Vol 1, No 1 (2025): Current Issue (Volume 1, Issue 1), 2025

PublishedJune 30, 2025

Published June 30, 2025

View Vol. 1 No. 1 (2025): Current Issue (Volume 1, Issue 1), 2025

Original Article

Hyalinizing Trabecular Tumor: A Case Series with Literature Review

Abdulwahid M. Salih, Rebaz O. Mohammed, Hiwa O. Baba, Shko H. Hassan, Muhammed Bag A. Ali, Imad J. Habibullah, Imad S. Sedeeq, Dyari Q. Hamad, Karokh K. Mohammed, Marwan N. Hassan, Abdullah A. Qadir, Harun A. Ahmed, Meer M. Abdulkareem, Fahmi H. Kakamad, Hero KH. Mala (Author)

DOI: https://doi.org/10.70955/JCJ.2025.2

First Published : 2025-05-24

Page 1-6

250

Abstract

Introduction: Hyalinizing trabecular tumor (HTT) is a rare thyroid neoplasm originating from follicular cells and poses diagnostic challenges due to its cytologic and histologic overlap with other thyroid malignancies. This study aims to present the clinical features and management of HTT cases treated at a single center.

Methods: This was a single-center retrospective case series. The patients were included from January 2019 to November 2024. Data collection took place over one month, from November 15, 2024, to December 15, 2024. The study included patients with HTT whose diagnoses were confirmed histopathologically.

Results: The case series included 11 patients, predominantly female, 10 (90.9%), with a mean age of 50.7±19.01 years. The most common presenting symptom was anterior neck swelling, recorded in 5 (45.5%), while one case (9.1%) was discovered incidentally. Hyperthyroidism was present in 6 (54.5%). The tumors were distributed within the thyroid gland as follows: left lobe in 5 (45.5%) cases, right lobe in 4 (36.4%) cases, and isthmus in 2 (18.1%) cases. Total thyroidectomy was performed in 7 patients (63.6%), with tumor sizes ranging from 0.5 to 5.5 cm and a mean diameter of 2.6 ± 2.05 cm. All diagnoses were confirmed postoperatively through histopathological examination.

Conclusion: A rare benign tumor, HTT remains challenging to diagnose accurately. Both total thyroidectomy and lobectomy may result in good outcomes.

Introduction

Thyroid neoplasms include a wide range of lesions with varying behavior and prognosis [1]. They are generally classified as benign, low-grade malignant, or malignant. Benign tumors, such as adenomas, are common and usually do not cause symptoms. Low-grade malignant neoplasms, like follicular thyroid carcinoma, tend to be more aggressive but often have a good prognosis [2]. Hyalinizing trabecular tumor (HTT) of the thyroid was first identified in 1987. It accounts for approximately 1% of all thyroid tumors, occurs six times more often in men than women, and is most frequently diagnosed in individuals in their 50s [3]. HTT was originally classified as a variant of follicular adenoma but is frequently misdiagnosed due to overlapping morphological features with several thyroid neoplasms. These include papillary thyroid carcinoma (PTC) and medullary thyroid carcinoma. Diagnostic challenges also extend to rare tumors with trabecular architecture, such as fetal-type follicular adenoma, poorly differentiated carcinoma, intrathyroid parathyroid neoplasms, and metastatic lesions to the thyroid [4,5]. This poses a challenge in the clinical management of these lesions, as an accurate presurgical diagnosis of HTT is essential to prevent unnecessary overtreatment of this tumor. A literature review reveals that an accurate preoperative cytological diagnosis of HTT was made in only 8% of reported cases. More worrisome, however, is that the remaining 92% were misdiagnosed with false-positive results [6].

The classification of HTT as benign or malignant remains controversial. While it is generally regarded as benign, it is considered a borderline tumor with the potential for malignancy [7]. This diagnostic complexity has led to confusion in terminology, with HTT also being referred to by various other names, including hyalinizing trabecular adenoma, paraganglioma-like adenoma, hyalinizing trabecular neoplasm, and hyalinizing trabecular carcinoma [8]. This study aims to provide a comprehensive overview of HTT by retrospectively analyzing 11 cases, focusing on clinical presentations, diagnostic challenges, and treatment outcomes.

Methods

Study design

This study was a retrospective single-center case series. The patients were managed over five years, from January 2019 to November 2024. Data collection took place over one month, from November 15, 2024, to December 15, 2024. This study was approved by the Ethics Committee of Kscien organization (Approval No. 2025-33).

Participants

The study included all patients diagnosed with hyalinizing trabecular tumor. Diagnoses were confirmed through histopathological examination of resected thyroid tissue. Clinical and sociodemographic data were collected from patients, medical records, and healthcare providers.

Pre-intervention assessment

Assessments included vital signs monitoring, ultrasonography (U/S), thyroid function tests, serum calcium levels, vocal cord evaluation, viral screening (HBV, HCV, and HIV), and complete blood count.

Intervention

All patients underwent surgery under general anesthesia and were positioned supine with the neck extended and elevated using a roller placed beneath the shoulders. If visible, a 4 cm transverse collar incision was made in a natural skin crease of the lower neck for cosmetic purposes. Subplatysmal flaps were elevated superiorly and inferiorly to allow adequate thyroid gland exposure. A circular skin flap was then raised, with dissection carried laterally, medially, and toward the upper and lower cervical regions.

The midline between the strap muscles was divided, and the muscles were retracted laterally to expose the thyroid gland. Dissection commenced with ligation of the middle thyroid vein, followed by the superior and inferior pedicles. The superior and inferior thyroid vessels were ligated and divided close to the thyroid capsule to preserve the recurrent laryngeal nerves and avoid compromising the parathyroid gland vasculature. To minimize thermal injury, electrocautery use was limited; instead, multiple suture ligatures were applied to control oozing. Both sharp and blunt dissection techniques were employed to identify and preserve the recurrent laryngeal nerves, with all dissections maintained close to the thyroid capsule. The parathyroid glands were preserved in all cases, and surrounding adipose tissue was retained to maintain vascular integrity. In cases where devascularization was suspected, parathyroid autotransplantation into the sternocleidomastoid muscle was performed.

Of the 11 patients, 7 underwent total thyroidectomy. One of these patients underwent additional lateral and central neck dissection, with identification and preservation of the internal jugular vein, spinal accessory nerve, and phrenic nerve. The remaining 4 patients underwent lobectomy, performed using the same surgical technique but limited to the involved thyroid lobe.

After each procedure, hemostasis was confirmed. Closed suction drains (RediVac®) were placed in the patients. The strap muscles were reapproximated in the midline, and the skin was closed with absorbable subcuticular sutures and Steri-Strips. All patients tolerated the procedure without intraoperative complications.

Post-intervention Considerations

Postoperatively, all patients received intravenous paracetamol, and those who underwent total thyroidectomy were prescribed thyroid hormone replacement therapy (Levothyroxine) adjusted to their body weights. The diagnosis was confirmed through histopathological examination of the surgical specimens.

Results

Participants

The case series included 11 patients, of whom ten (90.9%) were female. Patient ages ranged from 32 to 85 years, with a mean age of 50.7±19.01 years. Seven patients (63.6%) had no significant past medical history, while three (27.3%) had hypertension, including one with concurrent type 2 diabetes and another with a history of renal stones. Surgical history was positive in six patients (55.5%). The most common presenting symptom was anterior neck swelling, observed in six patients (54.5%), followed by weight loss in three patients (27.3%). In one case (9.1%), the finding was incidental. Preoperative thyroid function assessment revealed hyperthyroidism in six patients (54.5%), and the remaining five (45.5%) were euthyroid (Table 1).

The left thyroid lobe was involved in five cases (45.5%), the right lobe in four cases (36.4%), and the isthmus in two cases (18.1%). The primary surgical approach was total thyroidectomy in seven cases (63.6%), including one patient who underwent concurrent neck dissection (Table 2). Tumor size ranged from 0.5 to 5.5 cm, with a mean size of 2.6 cm (Table 3). All the diagnoses were made post-operatively through histopathological examination (Figure 1).

Outcomes and follow-up

The follow-up period ranged from one month to five years, with a mean duration of 2.3±1.27 years. During this period, no cases of recurrence were reported, and all patients achieved complete recovery. No significant complications were observed during or after the surgical procedures.

**Table 1.** Clinical and sociodemographic data of the patients.
Case no.	Age	Sex	Medical history	Surgical history	Drug history	Chief complaint	Duration	Examination	Thyroid function test
1	39	F	Unremarkable	Unremarkable	Unremarkable	Incidental	1 week	Not palpable	Hyperthyroid
2	78	F	Hypertension	Cataract surgery & cholecystectomy	Statin, Anticoagulant & Anti-Hypertension	Shortness of breath	8 years	Hard	Hyperthyroid
3	74	M	Unremarkable	Kidney transplant	Corticosteroids	Weight-loss	5 months	Not palpable	Hyperthyroid
4	85	F	Hypertension & renal stone	Thyroid lobectomy & lithotripsy	Anti-Hypertension	Anterior neck swelling	6 years	Hard	Euthyroid
5	32	F	Unremarkable	Unremarkable	Unremarkable	Anterior neck swelling	3 months	Hard	Euthyroid
6	34	F	Hodgkin’s lymphoma	C-section & cervical cerclage	Antibiotics	Weight-loss	8 months	Not palpable	Euthyroid
7	38	F	Unremarkable	Cholecystectomy	Unremarkable	Anterior neck swelling	1.5 years	Hard	Hyperthyroid
8	42	F	Unremarkable	Unremarkable	Unremarkable	Anterior neck swelling	1 year	Hard	Euthyroid
9	51	F	Hypertension & Type 2 Diabetes Mellitus	Unremarkable	Anti-Hypertension & Anti-diabetics	Anterior neck swelling	2 months	Hard	Hyperthyroid
10	40	F	Unremarkable	Unremarkable	Unremarkable	Weight-loss	6 months	Not palpable	Euthyroid
11	45	F	Unremarkable	Unremarkable	Unremarkable	Anterior neck swelling	1 year	Hard	Hyperthyroid
M:Male, F:Female, S.Ca:Serum calcium, TG:Thyroglobulin, N/A:Not applicable

**Table 2.** Diagnostics and management.
Case no.	S. Ca (mg/dL)	US	US consistency	US echogenicity	Side	FNA Bethesda	Vocal cord assessment	Type of operation	Post Op complications	HPE	Tumor Size (cm)	Follow-up (years)
1	N/A	N/A	N/A	N/A	Left lobe	V	Normal	Lobectomy	None	HTT	1.4	5
2	9.6	TR3	Mixed	Heterogenous	Left lobe	IV	Normal	Total thyroidectomy	None	HTT	3.8	2
3	7.9	GD	No nodule	No nodule	Isthmus	NP	Normal	Total thyroidectomy	None	HTT	1	2
4	9.8	GD	No nodule	No nodule	Right lobe	NP	Normal	Total thyroidectomy	None	HTT	6	0.08
5	9.2	TR4	Solid	Hyperechoic	Left lobe	V	Normal	Total thyroidectomy	None	HTT	4.5	3
6	9.5	TR4	Solid	Hyperechoic	Left lobe	V	Normal	Total thyroidectomy	None	HTT	0.5	3
7	9.1	TR3	Solid	Heterogenous	Left lobe	NP	Normal	Lobectomy	None	HTT	1.4	1
8	9.5	TR4	Solid	Hypoechoic	Right lobe	IV	Normal	Lobectomy	None	HTT	3	2
9	9	TR3	N/A	N/A	Isthmus	NP	Normal	Total thyroidectomy	None	HTT	0.8	2
10	9.2	TR3	N/A	N/A	Right lobe	III	Normal	Total thyroidectomy	None	HTT	0.7	3
11	8.9	TR3	N/A	N/A	Right lobe	NP	Normal	Lobectomy	None	HTT	5.5	3
N/A: Not Applicable, S.Ca: Serum Calcium, US: Ultrasound, FNA: Fine Needle Aspiration, OP: Operation, HPE: Histopathology, TR: TI-RADS, GD: Graves' Disease, NP: Non-Productive, HTT: Hyalinizing trabecular tumor

**Table 3.** Patient demographics and clinical data.
Variables	Frequency
Sex
Male	1 (9.1%)
Female	10 (90.9)
Age groups (years)
30-39	4 (36.3%)
40-49	3 (27.3%)
50-59	1 (9.1%)
>60	3 (27.3%)
Mean ± SD	50.7 ± 19.0
Medical history
Unremarkable	7 (63.6%)
Hypertension	1 (9.1%)
Hypertension & renal stones	1 (9.1%)
Hodgkin’s lymphoma	1 (9.1%)
Hypertension & Type 2 Diabetes Mellitus	1 (9.1%)
Surgical history
Unremarkable	6 (54.5%)
Cholecystectomy	1 (9.1%)
C-section & cervical cerclage	1 (9.1%)
Cholecystectomy & cataract surgery	1 (9.1%)
Kidney transplant	1 (9.1%)
Thyroid lobectomy & lithotripsy	1 (9.1%)
Drug history
Negative	6 (54.5%)
Corticosteroids	1 (9.1%)
Antibiotics	1 (9.1%)
Antihypertensive	1 (9.1%)
Antihypertensive & Antidiabetic	1 (9.1%)
Chief complaint
Anterior neck swelling	6 (54.5%)
Shortness of breath	1 (9.1%)
Weight loss	3 (27.3%)
Incidental	1 (9.1%)
Thyroid examination
Not palpable	7 (63.6%)
Hard	4 (36.4%)
Thyroid function
Hyperthyroid	6 (54.5%)
Euthyroid	5 (45.5%)
Affected side
Right lobe	4 (36.3%)
Left lobe	5 (45.5%)
Isthmus	2 (18.2%)
Operation
Total thyroidectomy	7 (63.6%)
Lobectomy	4 (36.4%)
Tumor size (cm)
Mean ± SD	2.6 ± 1.95
Follow-up (years)
0.0 - 1.0	2 (18.18%)
1.1 - 2.0	4 (36.36%)
2.1 - 3.0	4 (36.36%)
3.1 - 4.0	0 (0.0%)
4.1 - 5.0	1 (9.09 %)
Mean ± SD	2.3 ± 1.27

Discussion

The diagnosis of HTT is challenging due to its resemblance to other thyroid neoplasms. While most cases are asymptomatic, Rossi et al. stated that symptom presentation may depend on tumor size and location [5]. Among the 11 cases, only one was asymptomatic, while the remaining ten exhibited clinical symptoms, including anterior neck swelling, weight loss, and shortness of breath. However, in the six reviewed cases, three were asymptomatic, two exhibited neck swelling, and one experienced both dyspnea and dysphagia (Table 4) [2,3,7-10].

**Table 4.** Summary of the cases of hyalinizing trabecular tumor in the literature.
Author/year	Age	Sex	Medical history	Drug history	Surgical history	Chief complaint	Examination	Duration (years)	Side	Size (cm)*	Distant metastasis	TI-RADS	FNA findings	Therapeutic approach	IHC findings	Diagnosis	Outcome	Follow-up (years)
Zhang et al./2025 [7]	31	F	Unremarkable	Unremarkable	Unremarkable	Incidental	Palpable mass	1.5	R	2.2	No	TR3	N/A	Lobectomy	TG +ve, CK19 +ve, TTF-1 +ve & Ki-67 5%	HTT	Resolved	0.6
Hayashi et al./2025 [2]	93	F	Diabetes mellitus, hyperlipidemia, hypertension & myocardial infarction	N/A	N/A	Loss of appetite, dyspnea & dysphagia	Enlarged, firm, non-tender, without palpable nodules	>12	B	>10	No	N/A	N/A	Conservative management & rehabilitation	N/A	HTT	Improved	N/A
Alsogair et al./2023 [3]	60	M	Unremarkable	Unremarkable	Hemorrhoidectomy	Mass on the neck	Palpable, firm & non-tender	N/A	R	3.8	No	N/A	60%-75% likelihood of malignancy	Total thyroidectomy followed by thyroxine	TG +ve & Ki-67 +ve	HTT	Resolved	0.06
Katano et al./ 2021 [10]	54	F	Panic disorder & chronic thyroiditis	Unremarkable	Unremarkable	Left cervical mass	Growing, painless & elastic	N/A	L	4.5	No	N/A	Chronic thyroiditis with possible malignancy	Lobectomy	Ki-67 +ve for cytoplasm & ColIV +ve	HTT	Resolved	1.5
Rhee et al./2018 [9]	63	F	Breast cancer	N/A	N/A	Incidental	N/A	N/A	L	0.6	No	N/A	Features of PTC	Lobectomy	Ki-67 +ve, CD56 +ve & Galectin-3 +ve	HTT	Resolved	N/A
Jones et al./2017 [8]	70	F	N/A	N/A	N/A	Incidental	N/A	N/A	R	1.94	N/A	N/A	60-75% likelihood of malignancy	Total thyroidectomy	TG +ve, vimentin +ve & CK19 +ve	HTT	Resolved	0.08
M: Male, F: Female, N/A: Not applicable, FNA: Fine needle aspiration, IHC: Immunohistochemistry, HTT: Hyalinizing trabecular tumor, L: Left, R: Right, B: Both

The diagnostic evaluation of most thyroid nodules typically begins with U/S, followed by fine needle aspiration (FNA). The U/S findings suggestive of HTT are well-defined, solitary, oval or round, solid hypoechoic nodules, usually without microcalcifications and displaying peri or intra-nodular vascularity. However, these features are not specific to HTT and may also occur in other thyroid lesions [7]. Recognizing the variability in U/S findings is crucial, as some studies reported an absence of malignant features. In contrast, Choi et al. found that 29% of HTT cases displayed malignant features on U/S [5,11]. In the present study, five cases were classified as mildly suspicious for malignancy, and three were considered moderately suspicious for malignancy according to the thyroid imaging reporting and data system (TI-RADS). Among the reviewed cases, malignancy was also suspected in four patients based on the U/S findings [2,3,7-10].

The primary diagnostic tool for thyroid nodules is FNA, which often leads to the misclassification of HTT as PTC or medullary thyroid carcinoma [9]. Ito et al. suggested that this diagnostic confusion arises from shared cytological features, including intranuclear cytoplasmic inclusions and nuclear grooves, which represent hallmark characteristics of PTC [4]. The cytological appearance of HTT on liquid-based preparations reveals cohesive aggregates or syncytial fragments of tumor cells surrounding hyaline material. Although tumor cells in HTT show enlarged nuclei with hyperchromasia and occasional intranuclear pseudo-inclusions similar to papillary carcinoma, HTT cells typically display dispersed fine chromatin rather than the pale and clear chromatin pattern observed in PTC [9].

Additionally, HTT cells demonstrate less frequent nuclear membrane irregularity and exhibit a more stratified trabecular arrangement compared to papillary carcinoma. These subtle distinctions prove crucial for accurate cytological interpretation, though they remain challenging to discern consistently in clinical practice [9]. Dell’Aquila et al. reported that up to 75% of HTTs are classified within Bethesda categories IV to VI [12]. Among the cases included, two were diagnosed as Bethesda category IV, while three were classified as category V, emphasizing their frequent misinterpretation by cytopathologists. Equivocal cytomorphologic diagnoses, such as atypia of undetermined significance or follicular lesion of undetermined significance, require repeat FNA, as the malignancy risk for nodules in these categories ranges from 1% to 15% [8].

On gross examination, HTT typically presents as a solid, well-circumscribed mass, or less commonly, as an encapsulated tumor, with colors ranging from yellow to tan, opposite to PTC, which is usually white and does not have a capsule. HTT generally lacks invasion into the capsule, vasculature, or thyroid parenchyma [5,7]. However, Gowrishankar reported a case in which invasion and malignant behavior were observed in HTT [13].

Immunohistochemistry can aid in diagnosing HTT, although some biomarkers used may lack significant specificity. HBME-1 and galectin-3 are well-established markers for malignant thyroid lesions, particularly PTC and its variants. However, their expression in HTT

remains a subject of debate. In their series, Dell’Aquila et al. found that the majority of HTT cases exhibited a distinct immune profile, with negative immunoreactivity observed in 16 out of 18 (89%) lesions. This finding further supports the classification of HTT as a benign tumor [12].

Recent genetic studies have demonstrated that GLIS rearrangements, particularly the PAX8-GLIS3 gene fusion, are critical for diagnosing HTT. Research indicates this fusion was present in 93% of HTT cases (13 out of 14), with the remaining 7% involving a PAX8-GLIS1 rearrangement. These findings highlight the diagnostic utility of detecting GLIS-related fusions to distinguish HTT from morphologically similar thyroid neoplasms [4].

In 2012, Smith et al. suggested that HTT could potentially acquire mutations leading to RET/PTC expression and undergo malignant transformation into PTC [14]. Given the uncertainty regarding the malignant potential of HTT, treatment approaches typically involve complete resection, near-total thyroidectomy, or lobectomy [8]. However, evidence suggests that up to three-quarters of patients may be subjected to overtreatment, opting for total or subtotal thyroidectomy rather than the less invasive lobectomy. In contrast, some experts argue for a more conservative management strategy, advocating for close monitoring or lobectomy as a first-line approach, rather than resorting to total thyroidectomy immediately [8]. Among the cases included in the current series, seven patients underwent total thyroidectomy, accounting for 63.6% of the surgeries performed. Utilizing total thyroidectomy as the surgical method mainly resulted from uncertainty in diagnosis, as imaging and other pre-operative examinations don’t usually provide a solid diagnosis.

Conclusion

In conclusion, HTT is a rare tumor that is challenging to diagnose accurately. Both total thyroidectomy and lobectomy may result in good outcomes.

Declarations

Conflicts of interest: The authors have no conflicts of interest to disclose.

Ethical approval: Ethical approval for this study was obtained from the Ethics Committee of the Kscien Organization (Approval No. 2025-33)

Consent for participation: Not applicable.

Consent for publication: Written informed consent for publication was obtained from all patients.

Funding: The present study received no financial support.

Acknowledgments: None to be declared.

Authors' contributions: AMS, ROM, FHK, and MMA: Major contributors to the conception of the study, as well as the literature search for related studies, and manuscript writing. HOB, SHH, MBA, IJH, ISS, and DQH: Literature review, design of the study, critical revision of the manuscript, and processing of the tables. KKM, MNH, AAQ, HAA, and HKM: Literature review and processing of the figure. All authors have read and approved the final version of the manuscript.

Use of AI: ChatGPT-3.5 was used to assist with language refinement and improve the overall clarity of the manuscript. All content was thoroughly reviewed and approved by the authors, who bear full responsibility for the final version.

Data availability statement: Not applicable.

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Thyroid Hemiagenesis: A Single-Center Case Series

Abdulwahid M. Salih, Hiwa O. Baba, Shaho F. Ahmed, Karzan M. Salih, Abdullah A. Qadir, Ayman M. Mustafa, Shko H. Hassan, Harun A. Ahmed, Aras J. Qaradakhy, Rebaz O. Mohammed, Aso N. Qadir, Ahmed H. Ahmed, Dindar H. Hama, Rawand R. Rafiq (Author)

DOI: https://doi.org/10.70955/JCJ.2025.3

First Published : 2025-06-10

Page 7-11

Abstract

Introduction: Thyroid hemiagenesis (TH) is a rare congenital anomaly characterized by the complete absence of one thyroid lobe, with or without absence of the isthmus. Its etiology remains unclear, and epidemiological data are limited. Although TH is often asymptomatic and discovered incidentally, it may pose clinical challenges when accompanied by thyroid dysfunction or structural abnormalities. This study reviews a single-center experience in diagnosing this condition and highlights its clinical significance..

Methods: This single-center case series was conducted from July 2021–July 2024, analyzing TH cases confirmed via ultrasonography. Eligible patients had complete medical records, including demographics, clinical presentation, radiological findings, and thyroid function status. Data were retrieved from electronic records and analyzed using SPSS 27.0, employing descriptive statistics to summarize means, ranges, frequencies, and percentages, ensuring a comprehensive assessment of TH’s clinical and epidemiological characteristics..

Results: This study analyzed 11 patients with TH (mean age: 28.12 ± 18.14 years; range: <1–55 years), seven of whom were females (63.6%). The diagnosis was incidental in six cases (54.5%), while three (27.3%) presented with neck swelling and two (18.2%) with neck pain. Thyroid function was euthyroid in seven (63.6%), hyperthyroid in two (18.2%), and hypothyroid in two (18.2%). Ultrasound examination confirmed left lobe and isthmus agenesis in eight cases (72.7%). Follow-up ranged from 4 to 48 months.

Conclusion: This study confirms the female predominance of TH, with left-lobe absence being the most common. Congenital anomalies suggest embryological links. While thyroid function is typically preserved, those with hypo- and hyperthyroidism highlight the need for individualized endocrine assessment and monitoring.

Introduction

Thyroid hemiagenesis (TH), first described in 1852, is characterized by the absence of one thyroid lobe, with or without the isthmus. It is typically detected incidentally during neck imaging, as most affected individuals remain asymptomatic and undiagnosed. However, epidemiological studies suggest a higher occurrence in regions endemic for hypothyroidism, potentially indicating an underlying environmental or genetic predisposition [1].

Epidemiological analyses reveal distinct anatomical patterns in TH. Approximately 80% of documented cases involve agenesis of the left thyroid lobe, establishing a left-to-right prevalence ratio of 4:1. When the left lobe is absent, the isthmus is missing in nearly half of cases [2]. In contrast, right lobe agenesis is more frequently associated with complete isthmus absence. Additionally, a well-documented female predominance exists, though the mechanisms contributing to this gender disparity remain unclear [3].

The pathogenesis of TH likely results from disruptions in key developmental processes, including defective migration, differentiation, or proliferation of thyroid precursor cells. Normal thyroid development begins in the fourth gestational week as an endodermal outpouching from the pharyngeal floor, which elongates into a bilobed structure and descends to its final position in the neck [3]. Any disturbance in this sequence can lead to congenital thyroid anomalies, with TH being one of the rarer manifestations. TH remains underreported due to its typically asymptomatic nature compared to more commonly recognized thyroid malformations, such as thyroglossal duct cysts or ectopic thyroid tissue. This underscores the need for systematic studies to assess its true prevalence and clinical significance [4].

The precise molecular mechanisms underlying TH remain to be understood completely, though evidence suggests a multifactorial etiology involving genetic and environmental influences. Genetic analyses of thyroid dysgenesis have identified key regulatory genes, FOXE1, PAX8, NKX2-1, NKX2-5, and TSHR, which play crucial roles in thyroid organogenesis [3]. While TH is primarily considered a sporadic anomaly, familial clustering, in some cases, suggests the possibility of heritable genetic influences. However, establishing definitive genotype-phenotype correlations remains challenging, as many cases occur in isolation without clear inheritance patterns. The interplay between genetic susceptibility and developmental signaling pathways continues to be an area of ongoing research [5].

Although numerous case reports exist, large-scale case series on TH remain scarce. The rarity of the condition, coupled with its typically benign and asymptomatic presentation, has contributed to a gap in comprehensive epidemiological and developmental studies. This study aims to review a single-center experience in diagnosing and managing TH cases. Additionally, all referenced sources have undergone verification [6].

Methods

Study design and Setting

This study was conducted as a single-center case series at the Thyroid Clinic of Smart Health Tower. The study period extended from July 2021 to July 2024, during which all eligible patients diagnosed with TH were identified and analyzed. The clinic serves as a specialized referral center for thyroid disorders, ensuring comprehensive diagnostic evaluation and follow-up of affected individuals.

Participant Selection and Eligibility Criteria

The study included all patients with a confirmed diagnosis of TH based on ultrasonographic imaging. Patients were eligible for inclusion if they had complete medical records detailing their demographic characteristics, clinical presentation, and radiological findings. Cases with incomplete data, particularly those lacking essential imaging reports or follow-up details, were excluded to ensure consistency and reliability in the analysis.

Data Collection and Variables Assessed

Patient data were systematically retrieved from the hospital’s electronic medical records, radiology reports, and clinicaldocumentation. The collected variables included demographic characteristics such as age, sex, residency, and clinical presentation, including symptoms at diagnosis and the presence of thyroid dysfunction or associated comorbidities. Medical and surgical history, including prior thyroid conditions and interventions, was also documented. Radiological findings focused on the laterality of TH, the size of the contralateral lobe, and any evidence of compensatory hypertrophy. Additionally, laboratory investigations, including thyroid function tests (thyroid stimulating hormone (TSH), triiodothyronine (T3), and thyroxine (T4) levels), were analyzed to assess thyroid function status. Follow-up data were reviewed to evaluate disease progression, changes in thyroid function, and any medical or surgical interventions undertaken.

Data Processing and Statistical Analysis

All collected data were documented and organized using Microsoft Excel 2021. Statistical analyses were conducted using the Statistical Package for the Social Sciences (SPSS) version 27.0. Descriptive statistical methods were employed to summarize the findings, with continuous variables presented as mean and range, while categorical variables were expressed as frequencies and percentages.

Results

This study included 11 patients diagnosed with TH, with a mean age of 28.12 ± 18.14 years (range: <1 to 55 years). The cohort comprised seven females (63.6%) and four males (36.4%). Clinically, six patients (54.5%) were diagnosed incidentally, three (27.3%) presented with neck swelling, and two (18.2%) reported neck pain. Regarding associated congenital conditions, eight patients (72.7%) had no additional anomalies. Among the remaining three, one had a thyroglossal duct cyst, one had a history of prolonged neonatal jaundice, and another presented with both prolonged neonatal jaundice and a periumbilical hernia.

Thyroid function assessment revealed that the majority (7 cases ,63.6%) of patients were euthyroid (0.35-4.5µIU/mL), while two patients exhibited hyperthyroidism (<0.35 µIU/mL), and two of them had hypothyroidism (>4.5 µIU/mL). Ultrasound findings demonstrated left lobe and isthmus agenesis in eight cases (72.7%), while two cases (18.2%) exhibited isolated left lobe agenesis, and one patient exhibited isolated right lobe agenesis (9.1%) (Figures 1 and 2). The largest documented normal lobe measured 100 × 43 × 35 mm, whereas the smallest measured 15 × 6.9 × 7.9 mm. The follow-up period ranged from 4 to 48 months (Tables 1-3).

Figure 1. Ultrasound findings of the thyroid gland demonstrate a right lobe of normal size (53 × 16 × 14 mm) with homogeneous parenchymal echotexture and no evidence of focal lesions. Notably, there is agenesis of the isthmus and absence of the left thyroid lobe (indicated by white arrow), consistent with thyroid hemiagenesis.

Figure 2. Ultrasound findings of the thyroid gland reveal a mildly enlarged left lobe measuring 57 × 20 × 19 mm, exhibiting heterogeneous parenchymal echogenicity. Multiple solid isoechoic nodules are present, the largest measuring 20 × 16 × 14 mm and classified as TIRADS 3. The right lobe and isthmus are absent (indicated by white arrow), consistent with right thyroid hemiagenesis.

**Table 1.** Demographic, clinical, and surgical characteristics of patients.
Cases	Age (Y)	Gender	History						Clinical Thyroid Examinations
Cases	Age (Y)	Gender	Presentation	Duration (W)	Other Congenital Conditions	PMH	PSH	Drug Hx	Clinical Thyroid Examinations
Case 1	20	M	Neck Swelling	4	None	Hyperthyroidism	Negative	Methimazole	G2
Case 2	21	F	Neck Pain	3	None	Negative	Lymph Node Biopsy	None	G2
Case 3	16	F	Incidental	8	Jaundice, Periumbilical Hernia	Iron Deficiency Anaemia	Hernia Surgery	None	G0
Case 4	<1	M	Incidental	N/A	Jaundice	Hypothyroidism	Negative	Thyroxine	G1
Case 5	31	M	Incidental	N/A	None	Negative	Negative	None	G0
Case 6	55	F	Neck Pain	8	None	Negative	Bilateral Total Knee Replacement	None	G0
Case 7	26	F	Neck Swelling	1	None	Negative	Tonsillectomy	None	G2
Case 8	6	F	Neck Swelling	1	Thyroglossal Duct Cyst	Negative	Tonsillectomy	None	G2
Case 9	55	F	Incidental	N/A	None	Negative	C-section	None	G0
Case 10	53	F	Incidental	1	None	Negative	C-section	None	G0
Case 11	27	M	Incidental	3	None	Negative	Negative	None	G0
M: Male, F: Female, PMH: Past Medical History, PSH: Past Surgical History, Drug Hx: Drug History, G: grades of thyroid enlargement, Y: Years, W: Weeks.

**Table 2.** Laboratory, imaging, and follow-up profiles of included cases.
Cases	Blood Investigations					Ultrasound Reports		Follow up (Months)
Cases	First TSH (µIU/mL)	First Free T4 (pmol/L)	First Total T4 (nmol/mL)	ATPO (IU/mL)	First TRAB (IU/mL)	Agenesis Side	Normal Lobe Size (mm)	Follow up (Months)
Case 1	0.005	55.53	N/A	N/A	12.55	Left Lobe, Isthmus	70 × 26 × 24	48
Case 2	2.45	19.9	N/A	9	N/A	Left Lobe, Isthmus	56 × 19 × 19	12
Case 3	1.8	N/A	165.6	N/A	N/A	Left Lobe, Isthmus	53 × 16 × 14	12
Case 4	100	2.34	N/A	N/A	N/A	Left Lobe	15 × 6.9 × 7.9	8
Case 5	2.61	18.00	N/A	68.3	N/A	Left Lobe, Isthmus	100 × 43 × 35	12
Case 6	1.83	15.86	N/A	109.2	N/A	Left Lobe, Isthmus	64 × 31 × 27	4
Case 7	1.27	16.3	N/A	80.9	N/A	Left Lobe, Isthmus	55 × 19 × 19	6
Case 8	1.33	21.71	N/A	N/A	N/A	Left Lobe, Isthmus	34 × 10 × 13	36
Case 9	2.27	17.02	N/A	11.45	N/A	Left Lobe	48 × 16 × 15	48
Case 10	5.66	9.75	N/A	17.6	N/A	Left Lobe, Isthmus	48 × 18 × 20	4
Case 11	0.005	41.6	9.75	N/A	0.8	Right Lobe	58 × 26 × 25	24
TSH: Thyroid-Stimulating Hormone, T4: Thyroxine, ATPO: Anti-Thyroid Peroxidase Antibodies, TRAB: Thyrotropin Receptor Antibodies, N/A: Not Applicable

**Table 3.** Summary of clinical and demographic findings.
Variables	Frequency (percentage)
Gender Male Female	4 (36.4%) 7 (63.6%)


Age, Years (Mean ± SD)	28.12 ± 18.14
Clinical presentations Incidental Neck Pain Neck Swelling	6 (54.5%) 2 (18.2%) 3 (27.3%)



Past medical history Iron Deficiency Anemia Negative	1 (9.1%) 10 (90.9%)


Clinical thyroid examination G0 G1 G2	6 (54.5%) 1 (9.1%) 4 (36.4%)



Thyroid function status Euthyroid Hyperthyroidism Hypothyroidism	7 (63.6%) 2 (18.2%) 2 (18.2%)



Agenesis Side Right Left	1 (9.1%) 10 (90.9%)

Discussion

The clinical presentation of TH is predominantly asymptomatic, with most cases identified incidentally during imaging studies performed for unrelated thyroid conditions or neck abnormalities. When symptomatic, manifestations typically arise from concurrent thyroid disorders rather than the hemiagenesis itself. These may include neck swelling due to compensatory hypertrophy of the remaining lobe, thyroid dysfunction, or palpable nodules. The prevalence of thyroid abnormalities in individuals with TH appears to increase with age, likely due to chronic overstimulation of the remaining lobe by TSH, a factor contributing to the ongoing debate regarding the benign nature of the condition [7].

In a study in which 40 patients newly diagnosed with TH, aged between 12 and 79, were enrolled, it was found that 90% of their cohort were clinically asymptomatic regarding hemiagenesis itself, with associated conditions including euthyroid nodular goiters, multinodular goiters, Graves’ disease, and Hashimoto’s thyroiditis [8]. Another study emphasized that even symptomatic cases typically arise from coexisting thyroid pathologies rather than the anatomical defect itself [3]. Rare presentations such as hypothyroidism with prolonged neonatal jaundice and umbilical hernia have been documented in pediatric cases [3]. In the present study, 54.5% of patients were diagnosed incidentally, a lower rate than previously reported in the literature. A notable proportion exhibited clinical symptoms, with neck swelling in 27.3% and neck pain in 18.2% of cases, suggesting potential variations in clinical presentation, particularly among younger populations. Furthermore, congenital anomalies, including thyroglossal duct cyst, prolonged neonatal jaundice, and periumbilical hernia, were observed in 27.3% of cases, findings not prominently reported in earlier studies. These variations highlight the need for further investigation into potential demographic and pathophysiological factors influencing the clinical spectrum of TH.

The diagnosis of TH is mainly based on imaging modalities, with ultrasonography as the first-line investigation and thyroid scintigraphy as a complementary confirmatory tool. Ultrasound imaging is particularly valuable as the initial screening method due to its wide availability, lack of radiation exposure, and sensitivity in detecting the absence of a thyroid lobe and any structural changes in the remaining thyroid tissue [3]. Thyroid scintigraphy using technetium or iodine provides functional anatomical assessment with the advantage of detecting ectopic thyroid tissue and diagnosing concurrent thyroid pathologies in the remaining lobe. Combining these two imaging modalities remains essential for accurate diagnosis and differentiation from other conditions that might mimic hemiagenesis [3]. A large cohort case-control study by Ruchala et al. emphasizes the need for both ultrasonography and scintigraphy to distinguish true hemiagenesis from pseudoagenesis, which can occur in cases of severe atrophy or destruction of thyroid tissue [8]. In a study focused on pediatric cases with suspected thyroid dysgenesis, researchers utilized both thyroid scanning and ultrasonography to establish definitive diagnoses, with hemiagenesis identified in one of their subjects [9]. Another case report of a rare male pediatric patient with TH demonstrated how ultrasonography revealed the absence of the left lobe while the right lobe showed minimal hyperplasia without nodules; scintigraphy confirmed these findings and ruled out ectopic thyroid tissue. This case emphasized that when only one thyroid lobe is detected initially, physicians should consider TH and employ both imaging modalities before invasive procedures [10].

A retrospective evaluation of imaging for congenital hypothyroidism revealed that compared to 99mTc-pertechnetate scanning, ultrasound examination demonstrated 100% specificity but only 44% sensitivity for detecting thyroid abnormalities. This finding highlights the value of scintigraphy as a complementary method to ultrasound examination, particularly when ectopic thyroid tissue is suspected. The limitations of relying solely on ultrasonography were further illustrated in cases where thyroid agenesis was diagnosed with ultrasonography, but follow-up scintigraphy revealed sublingual thyroid tissue in a significant proportion of patients [11]. In the current study, ultrasonography was the primary diagnostic tool, revealing left lobe and isthmus agenesis in eight (72.7%) cases, while two cases (18.2%) exhibited isolated left lobe agenesis with preservation of the isthmus, and one case showed isolated right lobe agenesis with preserved isthmus (9.1%). The ultrasound findings documented normal lobe dimensions ranging from the smallest at 15 × 6.9 × 7.9 mm to the largest at 100 × 43 × 35 mm, providing valuable reference values for assessing potential compensatory hypertrophy.

Typically, TH is associated with normal thyroid function, as the remaining lobe compensates for the absent tissue. Most patients remain euthyroid, though biochemical patterns may reveal elevated TSH levels despite normal peripheral hormone concentrations, suggesting mild subclinical hypothyroidism or compensatory stimulation of the intact lobe. Functional thyroid disorders such as hyperthyroidism or hypothyroidism may coexist, often linked to concurrent pathologies like autoimmune thyroiditis or nodular goiter [3,12]. Recent studies highlight these trends. Ruchała et al. noted that while TSH and free T3 levels were elevated in TH patients compared to controls, most maintained euthyroidism [8]. Maiorana et al. documented subclinical hypothyroidism in pediatric cases [13]. Genetic factors, including potential PAX8 or FOXE1 gene involvement, may influence thyroid development but do not directly correlate with hormonal status [14]. Management focuses on addressing associated thyroid disorders rather than the anatomical defect itself. For asymptomatic patients, periodic monitoring with ultrasonography and thyroid function tests suffices [3,12]. Surgical intervention is indicated for malignancies or symptomatic nodules in the remaining lobe, necessitating lifelong thyroxine supplementation post-resection [14]. The current study aligns with these findings since 63.6% of patients were euthyroid, and only two cases of hyperthyroidism and two of hypothyroidism were identified, each requiring specific targeted therapy. While compensatory hypertrophy was observed, no evidence of progressive dysfunction emerged during follow-up, reinforcing the conservative approach for uncomplicated TH [3].

The follow-up and outcome of TH primarily focus on monitoring for potential thyroid pathologies and ensuring optimal thyroid function. Since TH itself is generally asymptomatic, the clinical significance lies in its association with other thyroid disorders. Therefore, regular follow-ups with thyroid function tests and ultrasonography are crucial to detect emerging thyroid conditions early. Recent studies emphasize the importance of long-term monitoring. For instance, a study by Peteiro-Gonzalez et al. highlighted that patients with TH are more prone to autoimmune thyroid disease and nodular goiter due to sustained compensatory stimulation of the remaining lobe, necessitating regular surveillance to manage these conditions effectively [12]. Another study suggested that patients with TH might benefit from thyroxine therapy to normalize TSH levels and potentially prevent associated thyroid pathologies. However, further research is needed to confirm this approach. In cases where TH coexists with malignancies like medullary thyroid cancer, follow-up involves regular biochemical monitoring (calcitonin and carcinoembryonic antigen levels) and ultrasonography to detect recurrence early [15]. The current study's follow-up period ranged from 4 to 48 months, with no significant thyroid dysfunction or complications reported during this time. The study's findings align with previous literature in emphasizing the need for ongoing surveillance to manage potential thyroid-related issues in patients with TH. Despite the absence of severe complications during the study period, the importance of continued monitoring cannot be overstated, given the potential for future development of thyroid pathologies in these patients.

One of the primary limitations of this study is the unavailability of advanced diagnostic tools such as thyroid scintigraphy and molecular genetic testing. Scintigraphy, which is considered the complementary tool for confirming thyroid hemiagenesis, was not performed in any of the cases due to lack of access to nuclear medicine facilities. However, all cases were assessed using high-resolution ultrasonography performed by experienced clinicians in a high-volume, thyroid-specialized center, supporting the reliability of the diagnoses. Similarly, molecular or genetic analyses that could provide insights into potential hereditary or developmental mechanisms were not feasible, primarily due to financial constraints and limited infrastructure in the setting of a developing country.

Conclusion

This study confirms the female predominance of TH, with a higher prevalence of left-lobe absence and frequent symptomatic presentations. The association with congenital anomalies suggests embryological links requiring further exploration. While thyroid function is generally preserved, cases of hypo- and hyperthyroidism underscore the need for individualized endocrine evaluation.

Declarations

Conflicts of interest: The authors have no conflicts of interest to disclose.

Ethical approval: Ethical approval for this study was obtained from the Ethics Committee of the Kscien Organization (Approval No. 2025-37)

Consent for participation: Not applicable.

Consent for publication: Written informed consent for publication was obtained from the patients or, in the case of minors, from their parents.

Funding: The present study received no financial support.

Acknowledgements: None to be declared.

Authors' contributions: AMS, HOB, ShFA, and AMM: Major contributors to the conception of the study, as well as the literature search for related studies, and manuscript writing. KMS, AQQ, SHH, HAA, AJQ and ROM: Literature review, design of the study, critical revision of the manuscript, and processing of the tables. ANQ, AHA, DHH, and RRR: Literature review, processing of the figures, data analysis and interpretation.

Data availability statement: Not applicable.

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Abstract PDF

Systematic Review and Meta-analyses

Blunt Chest Trauma and Chylothorax: A Systematic Review

Hiwa O. Abdullah, Fahmi H. Kakamad, Harem K. Ahmed, Bnar J. Hama Amin, Hadi M. Abdullah, Shvan H. Mohammed, Berun A. Abdalla, Sasan M. Ahmed, Marwan N. Hassan, Sarhang S. Abdalla, Yousif M. Mahmood, Kayhan N. Najar, Suhaib H. Kakamad, Bander A. Abdalla (Author)

DOI: https://doi.org/10.70955/JCJ.2025.4

First Published : 2025-06-10

Page 27-45

Abstract

Introduction: Although traumatic chylothorax is predominantly associated with penetrating injuries, instances following blunt trauma, as a rare and challenging condition, are being increasingly documented. This study aims to systematically review the reported cases of blunt chest traumatic chylothorax (BCTC) and provide comprehensive insights into the condition.

Methods: Related studies published until December 11, 2024, were identified through Google Scholar. All studies documenting instances of BCTC, without restriction on cause or patient demographics, were included. Studies were excluded if they focused on chylothorax caused by penetrating injuries, their content was unretrievable, they were review articles, or they were published in blacklisted journals.

Results: Sixty-five eligible studies, encompassing 69 cases of BCTC, were included in the review. It predominantly affected males (73.91%), with patient ages ranging from 11 months to 84 years old. The most common clinical findings were dyspnea (47.83%) and abnormal auscultation or percussion (34.78%), with road traffic accidents as the primary cause (59.42%). Unilateral chylothorax was found in 72.46% of cases, bilateral chylothorax occurred in 27.54%, and pleural effusion was the most frequent radiological finding (55.07% in X-ray and 33.33% in computed tomography). Treatment typically included drainage (94.20%), parenteral nutrition (50.72%), and thoracic duct closure (39.13%). Most patients achieved complete recovery (89.85%), and six cases (8.70%) died.

Conclusion: The condition is rare and complex, underscored by the wide variability in patient demographics, clinical presentations, chylothorax onset, and management approaches. Given the challenges posed by limited evidence, the findings emphasize the need for early recognition and individualized management strategies.

Introduction

Chylothorax is a rare condition characterized by the accumulation of chyle in the pleural cavity caused by a disruption of the thoracic duct [1]. Chyle is an opalescent fluid that consists of triglycerides, chylomicrons, proteins, electrolytes, immunoglobulins, and fat-soluble vitamins, transported from the gastrointestinal system into the bloodstream by the thoracic duct. It makes up about 1-3% of total body weight in adults. Chylothorax was initially described by Bartolet in 1633 and later reported in the literature by Quinke in 1875 [1,2]. It is categorized into congenital, neoplastic, traumatic, and miscellaneous forms. The most common cause is malignancy, which leads to obstruction of the thoracic duct, while traumatic chylothorax is typically iatrogenic, resulting from surgical procedures or catheter placement. Penetrating trauma is the usual cause of traumatic chylothorax, while blunt trauma is considered an infrequent cause [1,2]. It may also develop due to chest compression or changes in intrathoracic pressure, such as during coughing or persistent vomiting [2].

The incidence of chylothorax is about 0.2% following blunt thoracic trauma and 0.9% after penetrating trauma. Bilateral chylothorax resulting from blunt trauma, mainly when no other injuries are evident, is an infrequent but severe complication [3]. Without prompt treatment, chylothorax can lead to serious complications, such as cardiopulmonary distress and significant nutritional deficiencies, with a high mortality rate of up to 15.5% [2-4]. Although traumatic chylothorax is predominantly associated with penetrating injuries, instances following blunt trauma have been increasingly documented, highlighting the need for awareness among healthcare providers regarding this potential complication [5,6]. This study aims to systematically review the reported cases of blunt chest traumatic chylothorax (BCTC) and provide comprehensive insights into the condition.

Methods

Literature search

The study followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Related studies published until December 11, 2024, were identified through Google Scholar using a search strategy that combined the following keywords with the “allintitle” and “including citation” features: (blunt trauma chylothorax), (blunt thoracic trauma chylothorax), (blunt thoracic injury chylothorax), (blunt chest injury chylothorax), (blunt torso trauma chylothorax), (blunt thoracic duct trauma), (blunt thoracic duct injury) and (traumatic chylothorax). The search was limited to English-language publications.

Eligibility criteria

All studies or reports documenting instances of BCTC, without restriction on cause or patient demographics, were eligible for inclusion. Studies were excluded if they focused on chylothorax caused by factors other than blunt chest trauma, if their content was unretrievable, if they were review articles, or if they were published in blacklisted journals. The legitimacy of the studies was verified by cross-referencing with widely recognized predatory journal checklists [7].

Study selection

First, an author conducted a literature search using the specified keywords and collected the relevant results. The titles and abstracts of the identified studies were then screened to exclude duplicates, non-English studies, and those unrelated to the study objective. Full-text screening was conducted for studies that passed the initial filtration, excluding those with unretrievable content or irrelevant study designs, such as reviews. This step was supervised by two authors, who independently reviewed each study. Finally, the remaining studies underwent legitimacy filtering.

Data extraction

The collected data encompassed various parameters, including the first author's name, year of publication, patient demographics, clinical manifestations, causes of chylothorax, chyle volume and content, the onset of chyle production, diagnostic methods, ICU admission status, treatment modalities, outcomes, and follow-up.

Statistical Analysis

The extracted data were organized in an Excel sheet (2019) and analyzed descriptively using the Statistical Package for the Social Sciences (SPSS, v. 27, IBM Co.). The results were presented in frequencies with percentages, means with standard deviation, and ranges.

Results

Study identification

A systematic search yielded 201 studies, all of which were case reports. After removing duplicates (16) and non-English articles (17), 168 titles and abstracts were screened. Fifty-five case reports were excluded due to irrelevance, and full-text evaluation of the remaining 113 case reports led to the exclusion of an additional 42. Furthermore, six articles were excluded for being published in warning-listed journals. Consequently, 65 eligible case reports, encompassing 69 cases of BCTC, were included in the review [1-6,8-66] (Tables 1 and 2). The identification process is outlined in a PRISMA flowchart (Figure 1).

**Table 1.** Demographic, Cause and Chyle Characteristics
First author, year [Reference]	Country	Age (year)	Gender	CFP	Cause	SOC	Amount of chyle (ml)/day*	COAP (day)	Biochemical content of chyle
Harvey, 2024 ^[5]	USA	60	F	Chest pain, multiple rib fractures	RTA	Left	<500	2	Triglycerides
Burduniuc, 2023 ^[2]	Czech Republic	70	F	Blunt injury	Fall on stairs	Right	>1000-2000	3	Protein, cholesterol, triglycerides
Dung, 2023 ^[14]	Vietnam	32	M	Thoracic spine injury, paraplegia	RTA	Right	>1000-2000	At once	Cholesterol, triglycerides
Kim, 2023 ^[4]	South Korea	45	M	Hemodynamically unstable, chest discomfort, multiple fractures, hemopneumothorax	Crushed by a 2-ton metal frame	Left	>1000-2000	1.66	Triglycerides
Boateng 2023 ^[33]	USA	75	F	Respiratory distress	Fall from bed	Right	<500	At once	Triglycerides
Ruest 2023 ^[34]	USA	15 months	M	Tenderness over right paraspinal thoracolumbar back, abnormal auscultation	Child abuse	Right	N/A	At once	N/A
Mohanakrishnan 2022 ^[35]	USA	70	F	Dyspnea, back pain, abnormal auscultation	Violent coughing episode	Right	>1000-2000	At once	Chylomicrons, triglycerides
Mazhar, 2021^[23]	UK	42	F	Dyspnea, abnormal auscultation	Fall from horse 1 week before presentation	Right	>1000-2000	7	Triglycerides
Waseem, 2021^[32]	Pakistan	50	M	Dyspnea	RTA 2 days before presentation	Bilateral	>1000-2000	2	Triglycerides, cholesterol, fat-rich fluid with few inflammatory cells
Din Dar 2021 ^[36]	India	50	M	Blunt injury	RTA	Right	>1000-2000	25	Triglyceride, chylomicrons
Bacon, 2020 ^[9]	USA	53	M	Hemopneumothorax	RTA	Left	<500	5	N/A
Champion, 2020 ^[12]	Canada	29	M	Dyspnea, flushing, diaphoresis, vomiting, abnormal auscultation	RTA	Bilateral	>1000-2000	At once	Cholesterol, triglycerides
Jindal 2019 ^[37]	India	35	M	Dyspnea, respiratory distress	RTA	Bilateral	>1000-2000	4	Triglyceride, WBC, albumin, glucose, protein, LDH
Ahmed, 2018 ^[1]	Iraq	42	M	Severe back pain	RTA	Right	500-1000	2	Triglycerides and lymphocyte
Brown, 2018 ^[10]	USA	53	M	Thoracoabdominal injuries, subcutaneous emphysema, unstable pelvis	RTA	Left	>1000-2000	N/A	Triglycerides and lymphocyte
Litzau, 2018 ^[22]	USA	66	F	Dyspnea, multiple fractures, abnormal auscultation	RTA 7 days before presentation	Right	>1000-2000	7	Triglycerides
Kozul, 2017 ^[19]	Australia	18	M	Multiple injuries	RTA	Bilateral	500-1000	0.46	N/A
Lee, 2017 ^[21]	South Korea	70	M	Hemothorax, flail chest	RTA	Right	>2000	5	Triglycerides, cholesterol
Mohamed, 2017 ^[3]	USA	51	M	Dyspnea, chest pain, abnormal auscultation	Fall on stairs	Bilateral	>1000-2000	5	Triglycerides, Leukocytes, RBCs, LDH, protein
Spasić, 2017 ^[6]	Serbia	55	F	Multiple injuries	RTA	Right	>2000	5	N/A
Sriprasit, 2017 ^[31]	Thailand	27	F	Hemothorax, neurogenic shock, multiple fractures	RTA	Left	<500	5	Triglycerides, protein, glucose, LDH
Hara 2017 ^[38]	Japan	17	F	Breathing difficulty, abnormal percussion	Recurrent chylothorax, physical punishment	Left	500-1000	At once	N/A
Jahn 2017 ^[39]	Germany	8	F	Respiratory distress, abnormal percussion, and auscultation	Pedestrian hit by a motor vehicle	Left	<500	5	Protein, albumin, LDH, triglycerides, cholesterol
Ghodrati 2016 ^[40]	Iran	12	F	Dyspnea, respiratory distress	Chest trauma during play at school	Bilateral	N/A	N/A	N/A
Lee 2016 ^[41]	South Korea	50	M	Paraplegia	Fall from height during construction work	Right	>1000-2000	3	Triglycerides
Sendama 2015 ^[42]	UK	84	F	Dyspnea, abnormal auscultation	Fall	Right	500-1000	4	Protein, LDH, cholesterol, triglycerides
Snow 2015 ^[43]	USA	22 months	M	Cough, dyspnea, respiratory distress, altered mental status, abnormal auscultation	Fall from a chair	Right	<500	2	Triglycerides
Adams 2013 ^[44]	USA	73	M	Paraplegia, rib fractures, hemothorax	RTA	Right	N/A	9	Triglycerides, lymphocytes, glucose, LDH, lipemia
Kumar 2013 ^[45]	India	32	M	Blunt injury, fracture of right femur	RTA	Right	>1000-2000	2	Triglycerides, chylomicrons
		23	M	Hemopneumothorax, multiple rib fractures, subcutaneous emphysema	RTA	Right	500-1000	1	Triglycerides
		40	M	Hemothorax	RTA	Right	500-1000	2	Triglycerides
Sharkey 2012 ^[46]	UK	50	M	Multiple fractures, hematoma, hemothorax, pneumothorax	RTA	Right	500-1000	N/A	Triglyceride, cholesterol
Sokouti, 2011^[30]	Iran	15	M	Respiratory distress, dyspnea, back pain, abnormal auscultation	Fall 11 years ago	Bilateral	>2000	40	Triglycerides, protein, cholesterol, fat
Kurklinsky 2011 ^[47]	USA	26	F	Pleuritic chest pain	Water-skiing fall	Bilateral	>1000-2000	2	Triglycerides
Apostolakis, 2009 ^[8]	USA	75	F	Dyspnea, back pain	RTA	Bilateral	500-1000	At once	Protein, LDH, glucose, amylase, triglycerides, cholesterol, albumin, globulin, K, Na, lymphocytes, erythrocytes
Apostolakis, 2009 ^[8]	USA	22	M	Back pain, hematoma of left thigh, chest pain	RTA	Bilateral	<500	0.25	Protein, LDH, glucose, amylase, triglycerides, cholesterol, albumin, globulin, K, Na, lymphocytes, erythrocytes
Huber, 2009 ^[16]	USA	47	M	Dyspnea, chest pain	Crushed by multiple metal gates	Bilateral	500-1000	3	Triglycerides
Schurz, 2009 ^[28]	Austria	39	M	Dyspnea, thoracodorsal pain	RTA	Left	>1000-2000	14	N/A
Serin-Ezer, 2009 ^[29]	Turkey	4	M	Dyspnea, somnolence, abnormal auscultation	Hit by a manufacturing pipe falling from a truck	Bilateral	<500	At once	Triglycerides, cholesterol, protein, LDH, glucose
Kamiyoshihara, 2008 ^[18]	Japan	51	M	Dyspnea, dullness in percussion	RTA 20 years before presentation	Bilateral	>1000-2000	At once (but the trauma dated back to 20 years prior)	Triglycerides
Pandey 2008 ^[48]	Australia	36	M	Chest pain, flail chest, hypotension	Fall from balcony	Right	>1000-2000	2	N/A
Lee, 2006 ^[20]	South Korea	11	M	Dyspnea, nausea, vomiting, abdominal discomfort, abnormal auscultation	RTA 3 days before presentation	Bilateral	N/A	3	Triglycerides, cholesterol, protein
Ozcelik, 2004 ^[26]	Turkey	15	F	Respiratory distress, subcutaneous emphysema, pneumothorax	Trapping under rubble during a 7.8 magnitude earthquake	Right	N/A	45	Cholesterol, triglycerides
Robbins 2004 ^[49]	USA	41	M	Chest injury, refractory hiccups, nausea	RTA	Bilateral	N/A	N/A	N/A
Buchan 2001 ^[50]	UK	18	M	Dyspnea	RTA	Right	>1000-2000	4	N/A
Chamberlain, 2000 ^[11]	UK	29	M	Pneumothorax, abdominal and paraspinal pain, loss of motor power, and sensation below T12/L1	RTA	Right	>1000-2000	0.88	N/A
Glyn-Jones 2000 ^[51]	UK	28	M	Dyspnea, polytrauma, tachypnea	RTA	Left	>2000	N/A	Triglyceride, cholesterol, WBC
Golden, 1999 ^[15]	USA	53	F	Chest pain, multiple fractures, hemopneumothorax, abnormal auscultation	RTA	Left	>2000	6	Triglycerides
McCormick, 1999 ^[24]	USA	46	M	Chest pain, dyspnea, abnormal auscultation, dullness to percussion	Hit-and-run motor vehicle accident	Bilateral	>1000-2000	14	Protein, cholesterol, triglycerides
Ikonomidis, 1997 ^[17]	Canada	17	M	Closed head injury, multiple fractures, respiratory distress, tracheal hematoma	RTA	Bilateral	<500	At once	Triglycerides
Ikonomidis, 1997 ^[17]	Canada	24	M	Closed head injury, rib fractures, hemothorax	Snowboarding accident	Left	<500	At once	Triglycerides
Guleserian, 1996 ^[52]	USA	11 months	M	Dyspnea, coughing, cold symptoms, grunting, abnormal percussion	Child abuse	Right	500-1000	N/A	Triglycerides, cholesterol, WBC
Milano, 1994 ^[25]	Italy	26	F	Dyspnea	Fall while skiing 4 months prior	Left	>1000-2000	50	Triglycerides
Fogli, 1993 ^[53]	Italy	31	M	Suspected traumatic hemothorax, dyspnea, cough	RTA	Right	500-1000	N/A	N/A
Grant, 1991 ^[54]	New Zealand	32	M	Dyspnea, chest and back pain	RTA	Right	>1000-2000	N/A	N/A
Dulchavsky, 1988 ^[13]	USA	48	M	Dyspnea, chest pain, abnormal auscultation	Fistfight	Right	>1000-2000	At once	Cholesterol, HDL, triglycerides, pre-beta lipoprotein, chylomicrons
Brook 1988 ^[55]	USA	27	M	Respiratory distress, abnormal percussion, tachycardia	RTA	Bilateral	>2000	3	Triglycerides
Pai, 1984 ^[27]	USA	19	M	Neck, back, and chest pain	RTA	Right	>1000-2000	N/A	N/A
Krishnan 1982 ^[56]	Malaysia	29	M	Dyspnea, abnormal percussion	RTA	Right	>2000	2	N/A
Azambuja 1981 ^[57]	Brazil	42	M	Paraplegia, hemopneumothorax	RTA	Right	N/A	3	N/A
Rea 1960 ^[58]	UK	28	M	Dyspnea, apex beat displaced to the left	Crush injury from falling planks	Right	500-1000	4	Lipid, protein, RBC, lymphocytes
Guest 1955 ^[59]	Canada	19	M	Dyspnea, dry cough, tachypnea, dull percussion	RTA	Right	>1000-2000	26	N/A
Elliot 1948 ^[60]	Canada	56	M	Fall injury, paraplegia, respiratory distress	Fall from a tree	Right	500-1000	3	Fat
Dorsey 1942 ^[61]	USA	60	M	Alcoholic stupor, dyspnea, chest pain, abnormal percussion	Fall down a flight of stairs	Right	>2000	0.5	Protein, albumin, globulin, fat
Cellan-Jones 1940 ^[62]	UK	32	M	Dyspnea, chest tightness	A stone hitting the chest and dorsal spine striking a block of coal	Right	>1000-2000	3	Fat
Brown 1937 ^[63]	USA	N/A	F	Respiratory distress, abdominal distention	RTA	Bilateral	>1000-2000	At once	N/A
Bauersfeld 1937 ^[64]	USA	22	M	Breathing difficulty, laceration of the scalp, pain in lower abdomen and lumbar region, cyanosis, cold extremities	RTA	Right	>2000	8	Fat globules
Lillie 1935 ^[65]	USA	45	M	Blunt injury	Fall from a scaffold 20 feet high	Right	>2000	N/A	N/A
Macnab 1932 ^[66]	Canada	46	M	Chest and back pain, dullness percussion, dyspnea, anorexia, weakness, intermittent fever, hypotension	Fall from a height of 11 feet	Right	500-1000	6	N/A
F: female, M: male, CFP: clinical findings & presentation, SOC: Side of chylothorax, hr: hour, N/A: non-available, COAP: Chyle onset after presentation, RTA: road traffic accident, RBC: red blood cell, LDH: lactate dehydrogenase, HDL: high density lipoprotein, WBC: white blood cell. * The amount of chyle has been grouped rather than the actual amount.

Figure 1. Study selection PRISMA flow chart.

**Table 2.** Imaging findings, treatment and outcomes.
First author, year ^[Reference]	Chest X-ray	CT	MRI	ICU admission	Treatment	Thoracic duct ligation approach	Mode of drainage	Duration of chest tube placement (day)	Follow-up (weeks)	Outcome
Harvey, 2024 ^[5]	Pleural effusion	Hemopneumothorax, pneumomediastinum, retrosternal hematoma, multiple rib fracture, lung contusions, and manubrium fractures	N/A	Yes	Drainage, medium chain fatty acid diet	N/A	Chest tube	7	8	Recovered
Burduniuc, 2023 ^[2]	Pleural effusion, multiple rip fracture	Pleural effusion, multiple rib fracture	Th12 vertebral fracture	Yes	Drainage, thoracic duct ligation	Thoracotomy	Chest tube	N/A	N/A	Recovered
Dung, 2023 ^[14]	Pleural effusion	T9 and T10 vertebral fracture	N/A	No	Drainage, octreotide, TPN, thoracic duct embolization	2.7 Fr microcatheter, fluoroscopic guidance	Chest tube	7	N/A	Recovered
Kim, 2023 ^[4]	Pleural effusion	Lipiodol leakage near T10–11 level	N/A	Yes	Drainage, TPN, intranodal lymphangiography, therapeutic lipiodol injection	N/A	Chest tube	39	N/A	Recovered
Boateng 2023 ^[33]	N/A	Pleural effusion, lung collapse	N/A	No	Drainage, medium-chain triglyceride	N/A	Chest tube	N/A	N/A	Died
Ruest 2023 ^[34]	Pleural effusion	T12 vertebral body fracture, rib fractures	N/A	Yes	Drainage	N/A	Chest tube	N/A	N/A	Recovered
Mohanakrishnan 2022 ^[35]	N/A	Pleural effusion, minimal ascites	N/A	No	Drainage, octreotide, low-fat diet, NPO, TPN, pleurodesis, thoracic duct embolization	Coiling and glue embolization	Chest tube	N/A	N/A	Recovered
Mazhar, 2021^[23]	Pleural effusion	Pleural effusion, T10 spinous process fracture	N/A	No	Drainage, octreotide, medium-chain triglyceride diet	N/A	Chest tube	3	N/A	Recovered
Waseem, 2021^[32]	Pleural effusion	Pleural effusion	N/A	No	Only drainage	N/A	Chest tube	5	N/A	Recovered
Din Dar 2021 ^[36]	N/A	Multiple rib fractures, hemothorax	N/A	No	Drainage, NPO, TPN, octreotide, thoracic duct embolization	Thoracotomy	Chest tube	25	48	Recovered
Bacon, 2020 ^[9]	Multiple rib fracture	Multiple rib fracture	N/A	Yes	Drainage, free-fat diet	N/A	Chest tube	N/A	12	Recovered
Champion, 2020 ^[12]	Pleural effusion	Pleural effusion	N/A	No	Drainage, octreotide, TPN, thoracic duct ligation	Thoracotomy	Chest tube	N/A	6	Recovered
Jindal 2019 ^[37]	Pleural effusion	Multiple rib fractures, lung contusions, fracture of L1 and L2 vertebrae	N/A	Yes	Drainage, thoracic duct ligation, TPN, octreotide, fat-free and medium chain triglyceride	Thoracotomy	Chest tube	8	8	Recovered
Ahmed, 2018 ^[1]	Opacification of hemithorax	D10 vertebral fracture, multiple rib fracture	N/A	Yes	Drainage, low-fat diet, albumin vial, octreotide	N/A	Chest tube
Brown, 2018 ^[10]	N/A	Left temporal epidural hematoma, pulmonary contusions, multiple skeletal fractures, pneumomediastinum compressing the right atrium	Not mentioned the findings	No	Drainage, NPO, TPN, octreotide, thoracic duct embolization, and ligation	Thoracotomy, decortication	Chest tube
Litzau, 2018 ^[22]	Pleural effusion	Pleural effusion	N/A	No	Drainage, low-fat diet	N/A	Chest tube
Kozul, 2017 ^[19]	N/A	Hemopneumothorax, mediastinal shift to the right, pleural effusion	N/A	No	Drainage, No fat/low-fat diet	N/A	Chest tube
Lee, 2017 ^[21]	Pleural effusion	Multiple rib fracture, hemopneumothorax (left), subcutaneous emphysema (left), and atelectasis (right).	N/A	Yes	Drainage, TPN, NPO, fat-free diet, medium-chain lipid diet, thoracic duct ligation, pleurectomy	Thoracotomy	Chest tube
Mohamed, 2017 ^[3]	Obliteration of left costophrenic angle (pleural effusion)	Bilateral effusion	N/A	No	Drainage, fat-free diet with medium-chain triglycerides, octreotide	N/A	Thoracentesis
Spasić, 2017 ^[6]	Lung contusion	Rib and thoracic vertebral fracture, hydropneumothorax, lung contusion, pneumomediastinum	N/A	No	Drainage, TPN, thoracic duct suturing	Thoracotomy	Chest tube
Sriprasit, 2017 ^[31]	N/A	N/A	N/A	Yes	Drainage, NPO, TPN	N/A	Chest tube
Hara 2017 ^[38]	Pleural effusion	N/A	N/A	No	Drainage, low-fat diet with medium-chain triglycerides, intranodal lymphangiography with lipiodol	N/A	Chest tube
Jahn 2017 ^[39]	Pulmonary opacification	Lung contusions	N/A	No	Drainage, fat-free diet	N/A	Chest tube
Ghodrati 2016 ^[40]	Pleural effusion	N/A	N/A	Yes	Drainage, thoracic duct embolization	Unknown	Chest tube
Lee 2016 ^[41]	N/A	Incomplete cord injury at the thoracic spinal vertebrae (T10 and T11)	N/A	No	Drainage, TPN, NPO, thoracic duct ligation	VATS	Chest tube
Sendama 2015 ^[42]	Pleural effusion	Multisegment fracture of L1 vertebra	N/A	No	Drainage, medium chain fatty acid diet, octreotide	N/A	Chest tube
Snow 2015 ^[43]	Opacification of right chest, mediastinal shift to left	N/A	N/A	Yes	Drainage, NPO, TPN, octreotide, low-fat diet	N/A	Chest tube
Adams 2013 ^[44]	Pleural effusion, atelectasis	Pleural effusion, atelectasis	N/A	Yes	Drainage, NPO, TPN, octreotide	N/A	Thoracentesis, chest tube
Kumar 2013 ^[45]	N/A	Pleural effusion, multiple rib fractures	N/A	No	Drainage, NPO, TPN, octreotide, chest physiotherapy	N/A	Chest tube
	N/A	Hemopneumothorax, multiple rib fractures	N/A	No	Drainage, NPO, TPN, octreotide, chest physiotherapy	N/A	Chest tube
	N/A	Bilateral hemothorax, lung contusion	N/A	Yes	Drainage, NPO, TPN, octreotide, exploratory laparotomy for biliary leak	N/A	Chest tube
Sharkey 2012 ^[46]	N/A	N/A	N/A	Yes	Drainage, NPO, TPN, octreotide, medium fatty acid diet	N/A	Chest tube
Sokouti, 2011^[30]	Large cystic mass in left posterior mediastinum	Large low-density cystic mass in the left posterior mediastinum, left pleural effusion	N/A	No	Drainage, thoracic duct ligation, TPN	Laparotomy, Thoracotomy	Chest tube
Kurklinsky 2011 ^[47]	N/A	Pleural effusion, dilated cisterna chyli, middle mediastinum fluid collection	N/A	No	Drainage, TPN, thoracic duct embolization	3 Fr microcatheter with ultrasound guidance	Thoracentesis
Apostolakis, 2009 ^[8]	Pleural effusion	Pleural effusion	N/A	No	Drainage, starvation diet, TPN	N/A	Chest tube
Apostolakis, 2009 ^[8]	Pleural effusion, rib fractures, ipsilateral sternoclavicular joint dislocation	Lung contusion	N/A	No	Drainage, starvation diet, TPN	N/A	Chest tube
Huber, 2009 ^[16]	Pleural effusion	Pleural effusion, right pneumothorax, multiple rib fracture, aortic pseudoaneurysm, retrocrural hemorrhage	N/A	Yes	Drainage, thoracic duct ligation, medium chain fatty acid diet, mechanical pleurodesis	Thoracotomy	Chest tube
Schurz, 2009 ^[28]	Multiple rib fracture, pleural effusion	Pleural effusion	Osseous lesions and pleural effusion	Yes	Drainage, TPN, fat-free diet, plain tea, apple puree	N/A	Pleural puncture, chest tube
Serin-Ezer, 2009 ^[29]	Multiple rib fracture, pleural effusion	Pleural effusion	N/A	No	Drainage, NPO, TPN	N/A	Chest tube
Kamiyoshihara, 2008 ^[18]	Pleural effusion	Pleural effusion	N/A	No	Drainage, low-fat diet, TPN, thoracic duct ligation, pleurodesis	Thoracotomy	Thoracentesis, Chest tube
Pandey 2008 ^[48]	N/A	Hemopneumothorax, pulmonary contusion, multiple rib fractures, pneumomediastinum	N/A	Yes	Drainage, octreotide, thoracic duct ligation	Laparoscopic ligation	Chest tube
Lee, 2006 ^[20]	Elevation of diaphragms, cardiomegaly	Pleural effusion, massive ascites around liver and spleen	N/A	No	Drainage, medium-chain lipid solution, NPO	N/A	Thoracentesis, chest tube
Ozcelik, 2004 ^[26]	Pneumothorax, consolidated right lung, pleural effusion	Right lung consolidation, pleural effusion	N/A	No	Drainage, thoracic duct mass ligation, TPN	Thoracotomy	Chest tube
Robbins 2004 ^[49]	N/A	Pleural effusion, focal fluid collection	N/A	No	EUS-guided aspiration, injection of sodium morrhuate	N/A	Aspiration
Buchan 2001 ^[50]	Pleural effusion	N/A	N/A	No	Drainage, low-fat diet, medium-chain triglycerides, thoracic duct ligation	Thoracotomy	Chest tube
Chamberlain, 2000 ^[11]	Pneumothorax, hemithorax opacification	Free abdominal gas	Fractures of T4 and T10 with spinal cord contusion and hematoma	No	Drainage, TPN, NPO, Supradiaphragmatic duct ligation	Thoracotomy	Chest tube
Glyn-Jones 2000 ^[51]	Mediastinal shift	Minor anterior wedge fractures at T5 and T10	Cord injury at T10	No	Drainage, thoracic duct ligation, pleurodesis, fat-free diet	Thoracotomy	Chest tube
Golden, 1999 ^[15]	N/A	N/A	N/A	Yes	Drainage, TPN, NPO, thoracic duct ligation	Thoracotomy	Chest tube
McCormick, 1999 ^[24]	Pleural effusion	Disruption of the thoracic duct at the T5 level	N/A	No	Only drainage	N/A	Chest tube
Ikonomidis, 1997 ^[17]	Pneumomediastinum, pulmonary contusions	N/A	N/A	No	Drainage, TPN, bowel rest	N/A	Chest tube
Ikonomidis, 1997 ^[17]	Left hemothorax	Left mediastinal hematoma, T3 vertebral fracture	N/A	No	Drainage, TPN, bowel rest	N/A	Chest tube
Guleserian, 1996 ^[52]	Right lung opacification and mediastinal shift to left	N/A	N/A	No	Drainage, nasogastric feeding with medium-chain triglycerides, low-fat diet	N/A	Chest tube
Milano, 1994 ^[25]	Pleural effusion	Dense lymphatic opacification at L1-L2, chyloma at D11, pleural leakage from left duct	N/A	No	Drainage, low-fat diet, medium-chain triglycerides, TPN, pleuroperitoneal shunt	N/A	Thoracentesis
Fogli, 1993 ^[53]	Pleural effusion, mediastinal shift	N/A	N/A	No	Drainage, TPN	N/A	Chest tube
Grant, 1991 ^[54]	Pleural effusion	N/A	N/A	No	Drainage, thoracic duct ligation, TPN, low-fat diet	Thoracotomy	Thoracocentesis, chest tube
Dulchavsky, 1988 ^[13]	Pleural effusion	N/A	N/A	No	Drainage, TPN, NPO, thoracic duct ligation	Thoracotomy	Chest tube	N/A	144	Recovered
Brook 1988 ^[55]	Pleural effusion	N/A	N/A	Yes	Drainage, NPO, TPN, low-fat/ high-protein diet	N/A	Chest tube	10	32	Recovered
Pai, 1984 ^[27]	Fracture dislocations of C6-C7 and T11-T12, right hemothorax	N/A	N/A	No	Drainage, fat-free diet, TPN, thoracic duct ligation, parietal pleurectomy	Thoracotomy	Chest tube	N/A	N/A	Recovered
Krishnan 1982 ^[56]	Pleural effusion, obliteration of left costophrenic angle, multiple rib fractures	N/A	N/A	No	Drainage, thoracic duct ligation	Thoracotomy	Chest tube	19	5	Recovered
Azambuja 1981 ^[57]	Hemopneumothorax	N/A	N/A	No	Drainage, thoracic duct ligation, pleural flap to address fistula, pleural abrasion	Thoracotomy	Chest tube	6	N/A	Recovered
Rea 1960 ^[58]	Opaque hemithorax	N/A	N/A	No	Drainage, thoracic duct ligation	Thoracotomy	Chest tube	N/A	N/A	Recovered
Guest 1955 ^[59]	N/A	N/A	N/A	No	Aspiration, high-protein, low-fat diet	N/A	Thoracentesis	N/A	4	Recovered
Elliot 1948 ^[60]	Pleural effusion	N/A	N/A	No	Aspiration, thoracic duct ligation	Thoracotomy	Aspiration	N/A	N/A	Recovered
Dorsey 1942 ^[61]	Rib fracture, pleural effusion	N/A	N/A	No	Drainage, low-fat diet, high-carb, high-protein diet, NPO	N/A	Thoracentesis	N/A	N/A	Died due to uncontrolled leakage
Cellan-Jones 1940 ^[62]	Pleural effusion	N/A	N/A	No	Aspiration, low-fat diet, intravenous glucose-saline	N/A	Aspiration	N/A	N/A	Died due to uncontrolled leakage
Brown 1937 ^[63]	Pleural effusion	N/A	N/A	No	Drainage, dietary management	N/A	Thoracentesis, paracentesis	N/A	N/A	Died
Bauersfeld 1937 ^[64]	Pleural effusion, mediastinal shift	N/A	N/A	No	Drainage, intravenous dextrose, high-calorie diet	N/A	Thoracentesis	16	N/A	Recovered
Lillie 1935 ^[65]	Pleural effusion, mediastinal displacement	N/A	N/A	No	Drainage, fat-free diet	N/A	Thoracentesis	N/A	N/A	Recovered
Macnab 1932 ^[66]	Displacement of the heart, pleural effusion	N/A	N/A	No	Drainage, carbohydrates, protein	N/A	Aspiration	48	2	Died due to extreme asthenia
CT: computed tomography, MRI: magnetic resonance imaging, ICU: intensive care unit, NPO: Nulla Per Os, TPN: total parenteral nutrition, N/A: non-available, EUS: endoscopic ultrasound

Presentation and etiology

The patients ranged in age from 11 months to 84 years, with a mean of 37.4 ± 19.9 years. Most cases were male (73.91%), while females accounted for 26.09%. The most common presenting symptom or clinical findings were dyspnea, observed in 47.83% of cases, followed by abnormal findings on auscultation or percussion (34.78%) and multiple fractures or injuries (27.54%). Other frequent symptoms included chest pain (21.74%) and pneumothorax, hemothorax, or hemopneumothorax (20.29%). Road traffic accidents (RTA) were the most prevalent cause of BCTC, accounting for 59.42% of cases, followed by falls (23.19%), trauma caused by heavy objects (8.70%), physical punishment or child abuse (4.34%), and fistfights (1.45%). Bilateral chylothorax was observed in 27.54% of cases, while 55.07% had right-sided involvement and 17.39% had left-sided involvement. The chyle leakage ranged widely, which was >1000–2000 mL/day in 40.58% of cases. Smaller volumes (<500 mL/day) were noted in 14.50% and 500 – 1000 mL/day in 20.28%. In 14.50% of cases, >2000 mL/day was drained. Chyle onset occurred within two days of presentation in 40.58% of cases and within three days to a week in 31.88%. Delayed onset (beyond one week) was reported in 13.04% of cases. The chyle predominantly contained only lipids (40.57%). Other compositions included lipid-protein mixtures (11.59%) and lipid-inflammatory cells (7.24%). Complex mixtures of lipids, proteins, sugars, inflammatory cells, and ions were seen in smaller proportions (5.80%) (Table 3).

**Table 3.** Summary of findings of the reviewed cases
Variables	Frequency / Percentage
Patient demography
Age range (mean ± SD), years	11 months – 84 (37.4 ± 19.9)
Gender Male Female	51 (73.91%) 18 (26.09%)


Common presentation and clinical findings* Dyspnea Abnormal auscultation or percussion Multiple fractures or injuries Chest pain Pneumothorax/ hemothorax/ hemopneumothorax Back pain Respiratory distress	33 (47.83%) 24 (34.78%) 19 (27.54%) 15 (21.74%) 14 (20.29%) 11 (15.94%) 11 (15.94%)







Cause of blunt trauma Road traffic accident Fall Hit or crushed by heavy objects Physical punishment & child abuse Fistfight Others	41 (59.42%) 16 (23.19%) 6 (8.70%) 3 (4.34%) 1 (1.45%) 2 (2.90%)






Side of chylothorax Right Left Bilateral	38 (55.07%) 12 (17.39%) 19 (27.54%)



Amount of chyle (ml/day) <500 500 - 1000 >1000-2000 >2000 N/A	10 (14.50%) 14 (20.28%) 28 (40.58%) 10 (14.50%) 7 (10.14%)





Chyle onset after presentation (day) At once – 2 days 3 days – one week > one week – one month > one month N/A	28 (40.58%) 22 (31.88%) 6 (8.70%) 3 (4.34%) 10 (14.50%)





Biochemical content of chyle Lipid Lipid + Protein Lipid + Inflammatory cells Lipid + Protein + Sugar + Inflammatory cells + Ions Lipid + Protein + Sugar Lipid + Inflammatory cells + Protein N/A	28 (40.57%) 8 (11.59%) 5 (7.24%) 4 (5.80%) 2 (2.90%) 2 (2.90%) 20 (29.00%)







Imaging findings
Chest X-rays* Pleural effusion Rib Fracture Lung/ mediastinal/ heart shift Opacification of lung Pneumothorax/ hemothorax Lung contusion Pneumomediastinum Vertebral fracture Lung consolidation Others N/A	38 (55.07%) 7 (10.14%) 7 (10.14%) 6 (8.70%) 5 (7.24%) 2 (2.90%) 1 (1.45%) 1 (1.45%) 1 (1.45%) 5 (7.24%) 15 (21.74%)











CT scan findings* Pleural effusion Rib fracture Vertebral fracture Pneumothorax/ hemothorax/ hemopneumothorax Lung contusion Pneumomediastinum Hematoma Thoracic duct leakage Others N/A	23 (33.33%) 14 (20.29%) 10 (14.50%) 9 (13.04%) 8 (11.59%) 4 (5.80%) 3 (4.34%) 2 (2.90%) 17 (24.64%) 25 (36.23%)










ICU admission Yes No	19 (27.54%) 50 (72.46%)


Common treatment approach* Drainage Parenteral nutrition Thoracic duct ligation/embolization/suturing Medium-chain fatty acid or low-fat diet Nulla per Os Free fat diet/starvation diet Octreotide Pleurectomy/Pleurodesis	65 (94.20%) 35 (50.72%) 27 (39.13%) 24 (34.78%) 19 (27.54%) 12 (17.39%) 17 (24.64%) 6 (8.70%)








Thoracic duct closure approach Thoracotomy Fr microcatheter with fluoroscopic/ ultrasound guidance VATS/ laparoscopy Coiling and glue embolization Unknown Not performed	22 (31.88%) 2 (2.90%) 2 (2.90%) 1 (1.45%) 1 (1.45%) 41 (59.42%)






Mode of drainage Chest tube Thoracentesis Aspiration Chest tube + Thoracentesis	53 (76.81%) 9 (13.04%) 4 (5.80%) 3 (4.34%)




Duration of chest tube placement ≤ One week > One week – two weeks > Two weeks – one month > One month N/A	12 (17.39%) 11 (15.94%) 13 (18.84%) 4 (5.80%) 29 (42.03%)





Outcome Recovered Partially recovered Died	62 (89.85%) 1 (1.45%) 6 (8.70%)



SD: standard deviation, CT: computed tomography, ICU: intensive care unit, VATS: video-assisted thoracoscopic surgery, N/A: non-available. *Each data in the variable might be found in more than one case

Imaging characteristics and management

Chest X-rays revealed pleural effusion in 55.07% of cases, rib fractures, and lung or mediastinal or heart shift, each in 10.14%, lung opacification in 8.70%, and pneumothorax or hemothorax in 7.24%. Computed tomography (CT) scans confirmed pleural effusion in 33.33% and rib fractures in 20.29%. The vertebral fracture was found in 14.50%, and pneumothorax, hemothorax, or hemopneumothorax in 13.04%. Drainage was performed in 94.20%, predominantly via chest tubes (76.81%). In 17.39% of patients, the chest tube was in place for one week or less, while 15.94% required chest tube placement for more than one week until two weeks. Another 18.84% needed chest tube placement for over two weeks to one month, and 5.80% had chest tube placement exceeding one month. Additional treatments included parenteral nutrition (50.72%), thoracic duct closure (39.13%), and dietary modifications such as a medium-chain fatty acid or low-fat diet (34.78%). Pharmacological treatments included octreotide in 24.64% of cases. Thoracic duct closure was performed through thoracotomy in 31.88%. Other less-used techniques included Fr microcatheter under radiological guidance in 2.90%, video-assisted thoracoscopy or laparoscopy in 2.90%, and coiling and glue embolization in 1.45%. In 59.42% of cases, thoracic duct closure was not performed. The majority of patients (89.85%) achieved complete recovery, with one case showing partial recovery (1.45%), and six cases died (8.70%) (Table 3).

Discussion

Chylothorax is a pathological condition; if left untreated, it can result in respiratory distress and various complications. The etiology is multifaceted, including traumatic causes, while non-traumatic factors may involve conditions that elevate lymphatic pressure or cause obstruction, such as lymphoma or heart failure [1,67]. Chylothorax was first documented in the medical literature during the 19th century but has since garnered increasing recognition with advancements in diagnostic and surgical techniques. Improved imaging modalities and surgical innovations have significantly enhanced the understanding of its pathophysiology, facilitating more effective identification and management of its underlying causes [1,2].

The demographic data in the present review revealed an age range of 11 months to 84 years, with a mean age of 37.4 ± 19.9 years. This aligns with the literature, as Elsaied et al. reported an approximate mean age of 42.67 years within an age range of 18 to 76 years [68]. Case reports have identified young adults as particularly susceptible to chylothorax following blunt chest trauma, who are commonly involved in motor vehicle collisions or sports injuries [12,31]. Conversely, another study found that individuals aged 50 years or older represented the most common age group among blunt chest trauma patients, comprising 28.9% of the sample [69]. This reflects the increased risk of falls and accidents among older populations [70]. The slightly lower mean age in the present study may be attributable to the inclusion of pediatric cases, broadening the demographic scope. A significant male predominance was observed in the current review, with 73.91% of cases involving males. This finding concurs with the literature, where male representation ranged from 72.3% in a literature review [71] to 85.4% in a cohort study on blunt chest trauma cases [69]. This gender disparity is often linked to higher exposure to high-risk activities and occupations among males [12,31].

The clinical presentation of chylothorax is variable, with dyspnea being the most common symptom, reported by approximately 66.7% of patients. Dyspnea arises from fluid accumulation in the pleural space, which restricts lung expansion and impairs gas exchange. Patients may also experience a dry cough, often exacerbated by pleural fluid [72]. Pleuritic chest pain is another potential symptom, likely caused by pleural irritation from chyle [3,5]. On physical examination, percussion of the thorax often reveals dullness over the affected area due to fluid accumulation, contrasting with the typical resonance of healthy lung tissue [3,12,22]. Auscultation typically shows diminished or absent breath sounds over regions where fluid has accumulated, reflecting impaired air movement [72]. In this review, consistent with the literature, dyspnea was the most common presenting symptom (47.83%). This was followed by abnormal findings on auscultation or percussion in 34.78% of cases. Other frequently reported symptoms included chest pain (21.74%) and complications such as pneumothorax, hemothorax, or hemopneumothorax (20.29%).

The thoracic duct, the primary conduit for lymphatic fluid, can be ruptured or injured by blunt trauma, leading to chyle leakage into the pleural space [71,73]. Damage to adjacent structures, such as vertebral fractures or mediastinal injuries, can also contribute to chylothorax. For example, thoracic spine injuries have been associated with chylothorax due to their anatomical proximity to the thoracic duct [4,14]. Chylothorax is predominantly unilateral. In a study of 74 cases, 78% involved one hemithorax, with the right side being affected in 67% and the left in 33%. Bilateral pleural effusion was observed in 22% of cases [74]. The volume of chyle leakage varies based on the severity of the injury and the extent of thoracic duct damage. Low-output chylothorax (<1000 mL/day) is typically managed conservatively, whereas high-output cases (>1–1.5 L/day) often require surgical or radiological intervention [4,37,75]. In extreme cases, chyle output exceeding 2000 mL/day has been reported [6,15,21,30]. Blunt chest trauma frequently results from RTA, underscoring the risks of high-speed collisions [5,22,32]. Falls are another common cause, accounting for approximately 45% of cases in a study of patients with multiple traumas [76]. In this review, RTA was found to be the leading cause of injury in 41 cases (59.42%), followed by falls in 16 cases (23.19%), trauma by heavy objects in 6 cases (8.70%), physical punishment or child abuse in three cases (4.34%) and fistfights in one case (1.45%). Bilateral chylothorax occurred in 27.54% of cases, higher than previously reported. Consistent with the literature, right-sided involvement (55.07%) was more common than left-sided involvement (17.39%). This finding contrasts with the observation of Kakamad et al., who reported no laterality difference, but is similar to the findings of Maldonado et al., who reported right-sided involvement in 67% of cases and left-sided involvement in 33% [71,74]. Chyle volume varied significantly, with 40.58% of cases producing >1000–2000 mL/day, while 10 cases (14.50%) exceeded 2000 mL/day.

The timing of chyle onset in this review varied, with symptoms developing within two days in 40.58% of cases and within three days to a week in 31.88%. These align with the finding that chylothorax most commonly manifests within 2 to 7 days following blunt chest trauma due to gradual pleural accumulation from duct leakage [71]. However, delayed onset beyond one month, as observed in 4.34% of reviewed cases, is rare but documented in the literature, with an extreme case reported up to 20 years post-trauma [18]. The biochemical composition of chyle among the reviewed cases primarily consisted of lipids (40.57%), with smaller proportions of lipid-protein mixtures (11.59%) and lipid-inflammatory cell mixtures (7.24%). Complex mixtures, including lipids, proteins, sugars, inflammatory cells, and ions, were identified in 5.80% of cases. These findings are consistent with the established biochemical profile of chyle, which is rich in triglycerides (≥110 mg/dL) and lymphocytes [71]. As reported in the literature, immunoglobulins and protein levels ranging from 2.2 to 6 g/dL underscore the nutritional and immunological impact of chyle loss [1,8,32].

In the present review, chest X-rays revealed pleural effusion in 55.07% of cases, consistent with its status as the most common radiographic finding in chylothorax, typically presenting as a homogeneous opacity [2,4,32,71]. Rib fractures and lung or mediastinal or heart shift each were observed in 10.14% of cases, with lung opacification in 8.70% and pneumothorax, or hemothorax in 7.24%, aligning with literature that highlights the utility of chest X-rays in detecting associated traumatic injuries, such as rib fractures and pulmonary contusions [2,6,77]. CT scans in the reviewed cases showed pleural effusion in 33.33% of cases and rib fractures in 20.29%. The detection of pneumothorax, hemothorax, or hemopneumothorax in 13.04% of cases further emphasizes the role of CT in visualizing coexisting traumatic injuries with greater detail than X-rays [4,77].

The initial approach to managing chylothorax primarily involves conservative measures, including nil per os (nothing by mouth), total parenteral nutrition, and adherence to a low-fat diet. Pharmacological interventions, such as octreotide, may decrease lymphatic flow and facilitate the closure of the leak [10,14,21,23]. In chylothorax management, chest tube placement is commonly maintained until chyle drainage significantly decreases or resolves. The duration varies from a few days to several weeks, influenced by the effectiveness of conservative approaches [78]. In this review, the chest tube was in place for one week or less in 17.39% of patients, while 15.94% required chest tube placement for more than one week until two weeks. Another 18.84% needed chest tube placement for over two weeks to one month, and 5.80% had chest tube placement exceeding one month.

In cases where conservative management proves ineffective, surgical intervention becomes imperative. Thoracic duct ligation remains the definitive surgical option and can be performed via open thoracotomy or minimally invasive approaches [79]. Based on the findings of this review, besides drainage, treatments for chylothorax included parenteral nutrition (50.72%), thoracic duct closure (39.13%), and dietary modifications, such as a medium-chain fatty acid or low-fat diet (34.78%). Octreotide was administered in 24.64% of cases. Thoracic duct closure was performed via thoracotomy in 31.88% of cases, Fr microcatheter with fluoroscopic/ ultrasound guidance in 2.90%, VATS or laparoscopy in 2.90%, and coiling and glue embolization in 1.45%. In 59.42% of cases, thoracic duct closure was not performed or was unnecessary.

The limitations of this study include the inherent nature of the reviewed studies, which were exclusively case reports due to the rarity of the condition. Consequently, drawing conclusions based on statistical analyses was not feasible. Additionally, the small sample size and the non-standardized data reporting across the included reports may have introduced potential bias into the findings of this review. While every effort was made to include all relevant studies identified through the search using predefined keywords, there remains the possibility that some studies were inadvertently overlooked.

Conclusion

BCTC is rare and complex, underscored by the wide variability in patient demographics, clinical presentations, chylothorax onset, and management approaches. Given the challenges posed by limited evidence, the findings emphasize the need for early recognition and individualized management strategies.

Declarations

Conflicts of interest: The authors have no conflicts of interest to disclose.

Ethical approval: Not applicable.

Consent for participation: Not applicable.

Consent for publication: Not applicable.

Funding: The present study received no financial support.

Acknowledgements: None to be declared.

Authors' contributions: FHK and HOA: major contributors to the conception of the study, as well as the literature search for related studies, and manuscript writing. HKA, BJHA, and HMA: Literature review, critical revision of the manuscript, and processing of the tables. SHM, BeAA, SMA, MNH, SSA, YMM, KAN, SHK and BaAA: Data extraction, data organization, and critical revision. All authors have read and approved the final version of the manuscript.

Use of AI: ChatGPT-3.5 was used to assist with language refinement and improve the overall clarity of the manuscript. All content was thoroughly reviewed and approved by the authors, who bear full responsibility for the final version.

Data availability statement: Not applicable.

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Abstract PDF

Current Perspectives on Cystic Echinococcosis: A Systematic Review

Hawkar A. Nasralla, Berun A. Abdalla, Hiwa O. Abdullah, Sasan M. Ahmed, Fahmi H. Kakamad, Shvan H. Mohammed, Rawezh Q. Salih, Dahat A. Hussein, Tomas M. Mikael, Marwan N. Hassan, Hunar A. Hassan, Suhaib H. Kakamad, Kayhan A. Najar, Karukh K. Mohammed, Diyar A. Omar, Shadi H. Sidiq, Fakher Abdullah, Yousif M. Mahmood (Author)

DOI: https://doi.org/10.70955/JCJ.2025.1

First Published : 2025-02-02

Page 12-26

843

Abstract

Introduction: Hydatidosis, a zoonotic disease caused by the larval stage of Echinococcus granulosus, is a significant public health concern with notable economic impact. It leads to morbidity and mortality worldwide, particularly in endemic regions. This study systematically reviews recent literature on cystic echinococcosis (CE) to provide updated insights into its prevalence, impact, and management.
Methods: A systematic review was conducted using PubMed to find original articles on hydatid cysts published between
September 1, 2019, and September 1, 2024. Data extracted included the first author's name, country, publication year, study type, number of cases, clinical presentation, diagnostic methods, cyst location and quantity, cyst status, treatment type and medications, follow-up details, recurrence, and mortality rates. Data were organized and qualitatively analyzed.
Results: A total of 398 articles were identified, of which 229 articles with 1,002 patients met the inclusion criteria. Spain reported the highest number of CE cases at 362 (36.13%). Asia accounted for 487 cases (48.60%), and Europe contributed 460 cases (45.91%). The liver was the most frequently affected organ, accounting for 731 cases (72.95%), followed by the lungs with 110 cases (10.98%), and the kidney with 43 cases (4.29%). The age distribution of the cases showed that 63 (6.29%) were aged between 3 and 18 years.
Conclusion: Hydatidosis remains a significant global public health concern, impacting developing and developed countries. The liver and lungs remain the primary sites of infection. Preventive strategies, including regular animal screening and enhanced public health education, are essential for controlling the spread of the disease.

Introduction

Cystic echinococcosis (CE), also known as hydatid disease (HD) or hydatidosis, is a well-known zoonotic disease caused by the larval stage of the tapeworm Echinococcus granulosus. Humans usually act as intermediate hosts, contracting the infection through direct contact with primary hosts like sheep, goats, cattle, dogs, and other canines or consuming food and water contaminated with the parasite's eggs [1, 2].

To date, HD is a serious public health problem that carries considerable economic implications. It leads to morbidity and mortality in various regions, notably in Mediterranean countries, the Middle East, New Zealand, Australia, India, and South America, mainly due to the close connections between sheep, dogs, and humans. It remains a neglected disease in many regions, necessitating concerted efforts for prevention and control, especially in rural areas where it is more prevalent [3, 4].

Hydatidosis can affect nearly any part of the body, but the liver is the organ most frequently impacted (75%), followed by the lungs (15%) and other organs like the brain (2%) and spine (1%) [3]. Hydatidosis is marked by a prolonged asymptomatic incubation period, often lasting several years. Clinical symptoms appear when the cysts grow large enough to compress nearby tissues. Additionally, cyst rupture into the peritoneal cavity can result in secondary cyst formation and the development of daughter cysts within them [3, 4]. This study systematically reviews recent literature on CE to provide updated insights into its prevalence, impact, and management.

Methods

Study design

This systematic review was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines.

Data sources and search strategy

A systematic review was performed using PubMed to identify original articles on hydatid cysts. The search strategy targeted recent, peer-reviewed clinical studies on echinococcosis in human populations, published from September 1, 2019, to September 1, 2024, and restricted to English-language research. The search was limited to the literature of the last five years to shed light on the current disease situation.

Eligibility criteria

This systematic review included only original studies and case reports. Exclusion criteria were as follows: 1) articles not in English, 2) abstract only, 3) studies on alveolar echinococcosis, 4) studies unrelated to humans, 5) inadequately peer-reviewed articles, and 6) any study types that did not meet the inclusion criteria. All references in this study were evaluated for eligibility [5].

Study selection and data extraction

The titles and abstracts of the selected studies were initially screened, followed by an in-depth full-text review to assess eligibility. Data extracted from each included study encompassed the first author’s name, country of origin, publication year, study type, number of cases, clinical presentation, diagnostic approaches, hydatid cyst location, cyst quantity, cyst status (intact or ruptured), treatment type, medications used, follow-up details, recurrence rate, and mortality rate.

Statistical analyses

Data were organized in an Excel spreadsheet (Microsoft Excel, 2021) and qualitatively analyzed using the Statistical Package for the Social Sciences (SPSS, version 27.0). Key findings were summarized as median, range, frequencies, and percentages.

Results

A total of 398 articles were identified through the search. After an initial review, 11 articles were excluded due to duplication and non-English language. The remaining 387 articles underwent title and abstract screening, during which 64 articles were excluded for not meeting the inclusion criteria. Consequently, 323 articles proceeded to full-text screening, and 93 were excluded due to unretrievable data, editorials, letters, or incomplete information. The remaining articles were then assessed for eligibility, resulting in 229 articles [1, 3, 4, 6-231] with 1,002 patients meeting the inclusion criteria and included in the study [Fig. 1].

Figure 1. The PRISMA diagram illustrating the study selection process

Among the included studies, 217 (94.76%) were case reports, seven (3.06%) were cohort studies, three (1.31%) were case series, one (0.44%) was a cross-sectional study, and one (0.44%) was a randomized controlled trial (Table 1). Among the countries, Spain recorded the highest number of cases with 362 (36.13%), followed by China with 270 cases (26.95%) and Turkey with 128 cases (12.77%), collectively accounting for approximately 75% of the total reported cases (Table 2). In terms of continental distribution, Asia recorded 487 cases (48.60%), while Europe was not far behind with 460 cases (45.91%) (Table 3).

**Table 1**. The details of the included studies
First Author, Year	Study design	Country	No. of cases	First Author, Year	Study design	Country	No. of cases	First Author, Year	Study design	Country	No. of cases
AlRashed, 2024 ^[¹^]	A	Saudi Arabia	1	Mutlu, 2022 ^[⁸²^]	A	Turkey	1	Gatt, 2020 ^[¹⁵⁷^]	A	Israel	1
Amer, 2024 ^[⁶^]	A	Iran	1	Ozgokce, 2022 ^[⁸³^]	A	Turkey	1	Giri, 2020 ^[¹⁵⁸^]	A	Bhutan	1
Babiker, 2024 ^[⁷^]	A	Qatar	1	Passarelli, 2022 ^[⁸⁴^]	A	US	1	Gopivallabha, 2020 ^[¹⁵⁹^]	A	India	1
Bazzi, 2024 ^[⁸^]	A	Lebanon	1	Pulavarty, 2022 ^[⁸⁵^]	A	India	1	Delgado, 2020 ^[¹⁶⁰^]	A	Spain	1
Brezeanu, 2024 ^[⁹^]	A	Romania	1	Rodríguez-Laiz, 2022 ^[⁸⁶^]	A	Spain	1	Handran, 2020 ^[¹⁶¹^]	A	US	1
Calu, 2024 ^[¹⁰^]	A	Romania	2	Sezer, 2022 ^[⁸⁷^]	A	Turkey	1	İriz, 2020 ^[¹⁶²^]	A	Turkey	1
Chen, 2024 ^[¹¹^]	A	Taiwan	1	Shahid, 2022 ^[⁸⁸^]	A	Pakistan	1	İyigün, 2020 ^[¹⁶³^]	A	Turkey	1
Darestani, 2024 ^[¹²^]	A	Iran	1	Sharma, 2022 ^[⁸⁹^]	A	India	1	Jarovsky, 2020 ^[¹⁶⁴^]	A	Brazil	1
Ghaedamini, 2024 ^[¹³^]	A	Iran	1	Sozutok, 2022 ^[⁹⁰^]	A	Turkey	1	Johny, 2020 ^[¹⁶⁵^]	A	India	1
Gulati, 2024 ^[¹⁴^]	A	US	1	Sun, 2022 ^[⁹¹^]	B	China	2	Kankilic, 2020 ^[¹⁶⁶^]	B	Turkey	6
Hasnaoui, 2024 ^[¹⁵^]	A	Tunisia	1	Ulusoy, 2022 ^[⁹²^]	A	Turkey	1	Kaskar, 2020 ^[¹⁶⁷^]	A	India	1
Haydar, 2024 ^[¹⁶^]	A	Iran	1	Uzunoğlu, 2022 ^[⁹³^]	A	Turkey	1	Kiran, 2020 ^[¹⁶⁸^]	A	India	1
Jalayeri, 2024 ^[¹⁷^]	A	Iran	1	Wang, 2022 ^[⁹⁴^]	A	China	1	Kırmacı, 2020 ^[¹⁶⁹^]	A	Turkey	1
Jellali, 2024 ^[¹⁸^]	A	Tunisia	1	Agarwal, 2021 ^[⁹⁵^]	A	India	1	Kumar, 2020 ^[¹⁷⁰^]	A	India	1
Karahan, 2024 ^[¹⁹^]	A	India	1	Aghajanzadeh, 2021 ^[⁴^]	A	Iran	1	Lahdhil, 2020 ^[¹⁷¹^]	A	Tunisia	1
Koren, 2024 ^[²⁰^]	A	Israel	1	Aili, 2021 ^[⁹⁶^]	A	China	1	Lapierre, 2020 ^[¹⁷²^]	A	Canada	1
Mahesan, 2024 ^[²¹^]	A	India	1	Akhan, 2021 ^[⁹⁷^]	A	Turkey	1	Llanos, 2020 ^[¹⁷³^]	A	US	1
Bouhout, 2024 ^[²²^]	A	Morocco	1	Basne, 2021 ^[⁹⁸^]	A	Nepal	1	Lodhia, 2020 ^[¹⁷⁴^]	A	Tanzania	2
Manuel, 2024 ^[²³^]	A	Angola	1	Biswas, 2021 ^[⁹⁹^]	A	India	3	Lyske, 2020 ^[¹⁷⁵^]	A	Canada	1
Mierzejewski, 2024 ^[²⁴^]	A	Poland	1	Boumarah, 2021 ^[¹⁰⁰^]	A	Saudi Arabia	1	Ma, 2020 ^[¹⁷⁶^]	E	China	195
Mutlu, 2024 ^[²⁵^]	A	Turkey	1	Çankaya, 2021 ^[¹⁰¹^]	A	Turkey	1	Mitrovic, 2020 ^[¹⁷⁷^]	A	Serbia	1
Remmerswaal, 2024 ^[²⁶^]	A	Netherlands	1	Cathomas, 2021 ^[¹⁰²^]	A	Switzerland	2	Mittal, 2020 ^[¹⁷⁸^]	A	India	1
Reyimu, 2024 ^[²⁷^]	A	China	1	Chatzifotiou, 2021 ^[¹⁰³^]	A	Germany	1	Moghtadaie, 2020 ^[¹⁷⁹^]	A	Iran	1
Sączek, 2024 ^[²⁸^]	A	Poland	1	Christodouli dis, 2021 ^[³^]	C	Greece	50	Nistor, 2020 ^[¹⁸⁰^]	A	Romania	1
Thakar, 2023 ^[²⁹^]	A	India	1	Ciftci, 2021 ^[¹⁰⁴^]	C	Turkey	34	Ogul, 2020 ^[¹⁸¹^]	A	Turkey	1
Voicu, 2024 ^[³⁰^]	A	Romania	1	Conlon, 2021 ^[¹⁰⁵^]	A	Ireland	1	Pappas, 2020 ^[¹⁸²^]	A	Greece	1
Aarif, 2023 ^[³¹^]	A	India	1	Elvan-Tuz, 2021 ^[¹⁰⁶^]	A	Turkey	2	Ramia, 2020 ^[¹⁸³^]	C	Spain	71
Alsulami, 2023 ^[³²^]	A	Saudi Arabia	1	Gautam, 2021 ^[¹⁰⁷^]	A	India	1	Samadian, 2020 ^[¹⁸⁴^]	A	Iran	1
Ammar, 2023 ^[³³^]	A	Tunisia	1	Ghabisha, 2021 ^[¹⁰⁸^]	A	Yemen	1	Sangal, 2020 ^[¹⁸⁵^]	A	India	1
Borni, 2023 ^[³⁴^]	A	Tunisia	1	Gonder, 2021 ^[¹⁰⁹^]	C	Turkey	9	Sauteur, 2020 ^[¹⁸⁶^]	A	Switzerland	1
Carrel, 2023 ^[³⁵^]	A	Uzbekistan	1	Govindaraj, 2021 ^[¹¹⁰^]	A	India	1	Savu, 2020 ^[¹⁸⁷^]	A	Romania	1
Casulli, 2023 ^[³⁶^]	A	Italy	1	Guha, 2021 ^[¹¹¹^]	A	India	1	Schleenvoigt, 2020 ^[¹⁸⁸^]	A	Germany	1
Caushi, 2023 ^[³⁷^]	A	Albania	1	Hãlmaciu, 2021 ^[¹¹²^]	A	Romania	1	Singh, 2020 ^[¹⁸⁹^]	A	India	1
Das, 2023 ^[³⁸^]	A	India	1	Harmouchi, 2021 ^[¹¹³^]	A	Morocco	1	Singh, 2020 ^[¹⁹⁰^]	A	India	1
Galvis, 2023 ^[³⁹^]	A	Colombia	1	Helvaci, 2021 ^[¹¹⁴^]	A	Turkey	1	Singla, 2020 ^[¹⁹¹^]	A	India	1
Göktürk, 2023 ^[⁴⁰^]	A	Turkey	1	Hermosa, 2021 ^[¹¹⁵^]	A	Spain	1	Sonsoz, 2020 ^[¹⁹²^]	A	Turkey	1
Hakimi, 2023 ^[⁴¹^]	A	Afghanistan	1	Iken, 2021 ^[¹¹⁶^]	A	Morocco	1	Tekin, 2020 ^[¹⁹³^]	A	Turkey	1
Hasnaoui, 2023 ^[⁴²^]	A	Tunisia	1	Jaén-Torrejimeno, 2021 ^[¹¹⁷^]	C	Spain	287	Tlili, 2020 ^[¹⁹⁴^]	A	Tunisia	1
Jia, 2023 ^[⁴³^]	A	China	1	Jindal, 2021 ^[¹¹⁸^]	A	India	1	Tonkaz, 2020 ^[¹⁹⁵^]	A	Turkey	1
Kardoun, 2023 ^[⁴⁴^]	A	Tunisia	1	Kafadar, 2021 ^[¹¹⁹^]	A	Turkey	1	Van De, 2020 ^[¹⁹⁶^]	A	Korea	2
Lao, 2023 ^[⁴⁵^]	A	China	1	Kankam, 2021 ^[¹²⁰^]	A	Iran	1	Vasilescu, 2020 ^[¹⁹⁷^]	A	Romania	1
Lees, 203 ^[⁴⁶^]	A	UK	1	Kechiche, 2021 ^[¹²¹^]	B	Tunisia	10	Verma, 2020 ^[¹⁹⁸^]	A	India	1
Li, 2023 ^[⁴⁷^]	A	China	1	Khasawneh, 2021 ^[¹²²^]	A	Jordan	2	Villalobos, 2020 ^[¹⁹⁹^]	A	US	1
Ma, 2023 ^[⁴⁸^]	A	China	1	Kumar, 2021 ^[¹²³^]	A	India	1	Xu, 2020 ^[²⁰⁰^]	A	China	1
Maggioni, 2023 ^[⁴⁹^]	A	Italy	1	Li, 2021 ^[¹²⁴^]	A	China	1	Yang, 2020 ^[²⁰¹^]	A	China	1
Mahmood, 2023 ^[⁵⁰^]	A	Pakistan	1	Maliqari, 2021 ^[¹²⁵^]	A	Albania	1	Yimamu, 2020 ^[²⁰²^]	A	China	1
Mayekar, 2023 ^[⁵¹^]	A	India	1	Moshref, 2021 ^[¹²⁶^]	A	Saudi Arabia	1	Abbas, 2019 ^[²⁰³^]	A	Morocco	1
Moraes, 2023 ^[⁵²^]	A	Brazil	1	Mozafar, 2021 ^[¹²⁷^]	A	Iran	1	Aydin, 2019 ^[²⁰⁴^]	A	Turkey	1
Moscatelli, 2023 ^[⁵³^]	A	Argentina	1	Rabhi, 2021 ^[¹²⁸^]	A	Tunisia	1	Banerjee, 2019 ^[²⁰⁵^]	A	India	1
Ntombela, 2023 ^[⁵⁴^]	A	South Africa	2	Rhissassi, 2021 ^[¹²⁹^]	A	Morocco	1	Beyhan, 2019 ^[²⁰⁶^]	A	Turkey	1
Peralta, 2023 ^[⁵⁵^]	A	Ecuador	1	Safari, 2021 ^[¹³⁰^]	A	Iran	1	Bracha, 2019 ^[²⁰⁷^]	A	Israel	2
Peulier‐Maitre, 2023 ^[⁵⁶^]	A	France	1	Shakerian, 2021 ^[¹³¹^]	A	Iran	1	Chaouch, 2019 ^[²⁰⁸^]	A	Tunisia	1
Ruíz-Pérez, 2023 ^[⁵⁷^]	A	Peru	1	Shariﬁ, 2021 ^[¹³²^]	A	Iran	1	Demir, 2019 ^[²⁰⁹^]	A	Turkey	1
Safarpour, 2023 ^[⁵⁸^]	A	Iran	2	Sharma, 2021 ^[¹³³^]	A	India	1	Derbel, 2019 ^[²¹⁰^]	A	Tunisia	1
Shah, 2023 ^[⁵⁹^]	A	India	1	Shuaibi, 2021 ^[¹³⁴^]	A	US	1	Gök, 2019 ^[²¹¹^]	A	Turkey	1
Türkoğlu, 2023 ^[⁶⁰^]	A	Turkey	1	Simsek, 2021 ^[¹³⁵^]	A	Turkey	3	Kandemirli, 2019 ^[²¹²^]	A	Turkey	1
Wang, 2023 ^[⁶¹^]	A	China	1	Şimşek, 2021 ^[¹³⁶^]	A	Turkey	1	Kang, 2019 ^[²¹³^]	A	Korea	1
Ahmady‑Nezhad, 2023 ^[⁶²^]	A	Iran	1	Singh, 2021 ^[¹³⁷^]	A	India	1	Kaya, 2019 ^[²¹⁴^]	A	Turkey	1
Assefa, 2022 ^[⁶³^]	A	Ethiopia	1	Ucar, 2021 ^[¹³⁸^]	A	Turkey	1	Khullar, 2019 ^[²¹⁵^]	A	India	1
Bicer, 2022 ^[⁶⁴^]	A	Turkey	1	van Zijl, 2021 ^[¹³⁹^]	A	South Africa	1	Kuzmanovska, 2019 ^[²¹⁶^]	A	Macedonia	2
Bishnoi, 2022 ^[⁶⁵^]	A	India	1	Velho, 2021 ^[¹⁴⁰^]	A	Portugal	1	MadissonBernardo, 2019 ^[²¹⁷^]	A	Brazil	1
Castro, 2022 ^[⁶⁶^]	A	Brazil	1	Wang, 2021 ^[¹⁴¹^]	A	China	1	Magistri, 2019 ^[²¹⁸^]	C	Italy	15
Dantis, 2022 ^[⁶⁷^]	A	India	1	Wu, 2021 ^[¹⁴²^]	A	China	1	Milosavljevic, 2019 ^[²¹⁹^]	A	Serbia	1
Dere, 2022 ^[⁶⁸^]	A	Turkey	1	Yasin, 2021 ^[¹⁴³^]	A	Malaysia	1	Ramteke, 2019 ^[²²⁰^]	A	India	2
Fourati, 2022 ^[⁶⁹^]	A	Tunisia	1	Zedelj, 2021 ^[¹⁴⁴^]	A	Croatia	1	Sharma, 2019 ^[²²¹^]	A	India	1
González Arboleda, 2022 ^[⁷⁰^]	A	Chile	1	Zhang, 2021 ^[¹⁴⁵^]	A	China	1	Singh, 2019 ^[²²²^]	A	India	1
Hammade, 2022 ^[⁷¹^]	A	Syria	1	Zouaghi, 2021 ^[¹⁴⁶^]	A	Tunisia	1	Syllaios, 2019 ^[²²³^]	A	Greece	1
Hanalioglu, 2022 ^[⁷²^]	A	Turkey	1	Aboksari, 2020 ^[¹⁴⁷^]	A	Iran	1	Taşlıçay, 2019 ^[²²⁴^]	A	Turkey	1
Çeviker,2022 ^[⁷³^]	A	Turkey	1	Acharya, 2020 ^[¹⁴⁸^]	A	Nepal	1	Tonkaz, 2019 ^[²²⁵^]	A	Turkey	1
Huertas, 2022 ^[⁷⁴^]	A	Spain	1	Akhan, 2020 ^[¹⁴⁹^]	D	Turkey	38	Trawinski, 2019 ^[²²⁶^]	A	Germany	1
Ijaz, 2022 ^[⁷⁵^]	A	Pakistan	1	Akhtar, 2020 ^[¹⁵⁰^]	A	India	1	Wa, 2019 ^[²²⁷^]	A	China	1
Karahan, 2022 ^[⁷⁶^]	A	Turkey	1	Arora, 2020 ^[¹⁵¹^]	A	India	1	Wang, 2019 ^[²²⁸^]	A	China	1
Karami, 2022 ^[⁷⁷^]	A	Iran	1	Assimakopoulos, 2020 ^[¹⁵²^]	A	Greece	1	Xu, 2019 ^[²²⁹^]	A	China	1
Kartavya, 2022 ^[⁷⁸^]	A	India	1	Bakshi, 2020 ^[¹⁵³^]	A	India	1	Yacine, 2019 ^[²³⁰^]	A	Tunisia	1
Kumar, 2022 ^[⁷⁹^]	A	India	1	Destek, 2020 ^[¹⁵⁴^]	A	Turkey	1	Zhuoli, 2019 ^[²³¹^]	A	China	1
Li, 2022 ^[⁸⁰^]	C	China	53	Dkhissi, 2020 ^[¹⁵⁵^]	A	Morocco	1
Li, 2022 ^[⁸¹^]	A	China	1	Ewnte, 2020 ^[¹⁵⁶^]	A	Ethiopia	1
A: case report, B: case series, C: cohort, D: randomized control trial, E: cross-sectional study, US:* United States, UK: United Kingdom*

**Table 2.** The distribution of reported cases across countries
Country	Number of Cases	Percentage (%)
Spain	362	36.13%
China	270	26.95%
Turkey	128	12.77%
Greece	53	5.29%
India	44	4.39%
Tunisia	24	2.40%
Iran	18	1.80%
Italy	17	1.70%
Romania	8	0.80%
Morocco	6	0.60%
United States	6	0.60%
Brazil	4	0.40%
Israel	4	0.40%
Saudi Arabia	4	0.40%
Germany	3	0.30%
Pakistan	3	0.30%
South Africa	3	0.30%
South Korea	3	0.30%
Switzerland	3	0.30%
Albania	2	0.20%
Canada	2	0.20%
Ethiopia	2	0.20%
Jordan	2	0.20%
Macedonia	2	0.20%
Nepal	2	0.20%
Poland	2	0.20%
Serbia	2	0.20%
Tanzania	2	0.20%
Afghanistan	1	0.10%
Angola	1	0.10%
Argentina	1	0.10%
Bhutan	1	0.10%
Chile	1	0.10%
Colombia	1	0.10%
Croatia	1	0.10%
Ecuador	1	0.10%
France	1	0.10%
Ireland	1	0.10%
Lebanon	1	0.10%
Malaysia	1	0.10%
Netherlands	1	0.10%
Peru	1	0.10%
Portugal	1	0.10%
Qatar	1	0.10%
Syria	1	0.10%
Taiwan	1	0.10%
United Kingdom	1	0.10%
Uzbekistan	1	0.10%
Yemen	1	0.10%

**Table 3.** Distribution of cases across continents
Continent	Number of cases	Percentage (%)
Asia	487	48.60%
Europe	460	45.91%
Africa	38	3.79%
South America	9	0.90%
North America	8	0.80%

The liver was the most frequently affected organ, accounting for 731 cases (72.95%), followed by the lungs with 110 cases (10.98%), the kidney with 43 cases (4.29%), the heart with 37 cases (3.69%), and muscle tissue with 24 cases (2.40%) (Table 4). The gender distribution among the cases was nearly equal, with 505 males (50.40%) and 496 females (49.50%). The age distribution revealed that 63 cases (6.29%) were between 3 and 18 years old, while the age of 740 patients (73.85%) was unspecified. Among the identified cases, the median age was 35 (QR:19-51) years. The most common clinical presentations included pain (18.86%), fever (6.59%), shortness of breath (4.09%), and cough (2.69%), with 51 cases (5.09%) being asymptomatic. Clinical presentation was not documented for 616 cases (61.48%). Regarding cyst characteristics, single cysts were identified in 466 cases (46.51%), and intact cysts were found in 100 patients (9.98%). Treatment predominantly involved surgical intervention, performed in 639 cases (63.77%), and Albendazole was the most commonly prescribed medication, given in 275 cases (27.44%). Follow-up data revealed that 347 patients were monitored for less than six months (34.63%). Recurrence was observed in 26 cases (2.59%), and mortality occurred in 17 patients (1.70%) (Table 5).

**Table 4.** Distribution of organs and body parts affected by hydatid cysts
Organ	Number of cases*	Percentage (%)
Liver	731	72.95%
Lung	110	10.98%
Kidney	43	4.29%
Heart	37	3.69%
Muscle	24	2.40%
Brain	21	2.10%
Abdominal cavity	16	1.60%
Bone	14	1.40%
Vertebral and spinal region	14	1.40%
Pelvic region	12	1.20%
Pancreas	9	0.90%
Spleen	8	0.80%
Mediastinum	5	0.50%
Breast	4	0.40%
Uterus	4	0.40%
Intra-orbital	3	0.30%
Thyroid gland	2	0.20%
Gallbladder	1	0.10%
Others	14	1.40%
* Some patients had hydatid cysts in more than one organ.

**Table 5.** Clinical and demographic characteristics of patients
Variables	Frequency (percentage)
Gender Male Female Non-Identified	505 (50.40%) 496 (49.50%) 1 (0.10%)



Age (year) 3 – 18 19 – 30 31 – 40 41 – 50 51 – 60 61 – 70 > 70 N/A Median (QR)	63 (6.29%) 52 (5.19%) 47 (4.69%) 34 (3.39) 18 (1.80%) 28 (2.79%) 20 (2.00%) 740 (73.85%) 35 (19–51)









Clinical presentations* Pain Fever Shortness of breath Cough Asymptomatic Others N/A	189 (18.86%) 66 (6.59%) 41 (4.09%) 27 (2.69%) 51 (5.09%) 42 (4.19%) 616 (61.48%)







Diagnostic findings
Hydatid serology Positive Negative N/A CT scan Ultrasound MRI X. ray Echocardiogram N/A	139 (13.87%) 31 (3.09%) 832 (83.03%) 806 (80.44%) 475 (47.40%) 168 (16.77%) 69 (6.88%) 23 (2.29%) 105 (10.48%)









Quantity of the cyst Single cyst Multiple cysts N/A	466 (46.51%) 203 (20.26%) 333 (33.23%)



Cyst status Patients with intact cysts Patients with ruptured cysts Patients with intact and ruptured cysts N/A	100 (9.98%) 97 (9.68%) 6 (0.60%) 799 (79.74%)




Type of treatment Surgical intervention Percutaneous treatment Medical treatment None N/A	639 (63.77%) 75 (7.49%) 31 (3.09%) 2 (0.20%) 255 (25.45%)





Medication Albendazole Albendazole & Praziquantel Mebendazole None N/A	275 (27.44%) 9 (0.90%) 1 (0.10%) 26 (2.59%) 691 (68.96%)





Follow up < 1 month – 6 months > 6 months – 1 year > 1 year – 2 years > 2years – 4 years > 4 years – 7 years > 7 years N/A	347 (34.63%) 25 (2.50%) 27 (2.69%) 13 (1.30%) 5 (0.50%) 2 (0.20%) 583 (58.18%)







Recurrence Yes No N/A	26 (2.59%) 566 (56.49%) 410 (40.92%)



Death Yes No N/A	17 (1.70%) 666 (66.47%) 319 (31.83%)



* Some patients had multiple clinical presentations, CT scan: computed tomography scan, MRI: magnetic resonance imaging, N/A: not available

Discussion

Hydatidosis is most common in countries with extensive livestock industries but has recently become a significant global health issue due to rising immigration and travel [117, 232]. The disease is prevalent in Peru, Chile, Argentina, Uruguay, southern Brazil, the Mediterranean basin, Central Asia, western China, and East Africa. It remains absent in Antarctica and has been successfully eradicated through comprehensive control measures in Iceland, New Zealand, Tasmania, the Falkland Islands, and Cyprus [233]. In the present study, Spain unexpectedly showed the highest percentage of CE cases, with 36.13%, followed by China (26.95%) and Turkey (12.77%), collectively accounting for nearly 75% of all reported cases. Spain is known to be an endemic country for CE, but this unusually high percentage may be attributed to the fact that Spain has advanced healthcare infrastructure and diagnostic capabilities, which could lead to more accurate identification and reporting of CE cases. In contrast, in many developing countries, where resources and diagnostic tools may be limited, CE cases might be underreported or misdiagnosed [234]. This diversity underscores the importance of diagnostic and reporting capabilities when comparing CE prevalence across countries with differing healthcare systems. Regarding continental distribution, Asia represented 48.60% of cases, with Europe closely following at 45.91%. This distribution demonstrates that CE is not limited to traditionally endemic regions in developing countries but is also present in developed countries.

Studies have identified the liver as the most common site for hydatid cysts, accounting for 75% of cases, followed by the lungs (15%) and the brain (2%) [3]. Furthermore, cysts can form in various other organs and structures, including the abdominal and pleural cavities, kidneys, spleen, bones, eyes, ovaries, testes, and pancreas [233]. The current study's findings confirm that the liver remains the most frequently affected organ (73%), with the lungs (11%) being the second most common site, consistent with the literature. However, the current study found that the kidneys, heart, and muscles were more frequently affected than the brain (2%–4% of cases). This contrasts with previous studies identifying the brain as the third most commonly affected organ [3, 235].

The European Centre for Disease Prevention and Control (ECDC) indicates that the occurrence of echinococcosis does not display a notable difference between genders, presenting a nearly balanced male-to-female ratio of 1.1:1 [236]. This finding aligns with the results of the present study, where the gender distribution among the cases was nearly equal, with males comprising 50.39% and females 49.50%, yielding a male-to-female ratio of approximately 1:0.98. However, Otero-Abad et al. reported that women are more susceptible to echinococcosis than men. This increased risk was linked to their more significant participation in household activities, such as food preparation and caring for pets, heightening their exposure to infected dogs, soil, and vegetables [237].

Moro et al. stated that only 10-20% of CE cases are identified in patients under 16 years old [238]. This is likely because CE is slow-growing and often asymptomatic, with most liver and lung cysts becoming symptomatic and diagnosed in adults. In contrast, in the current study, the highest prevalence of CE was observed in the pediatric age group of 3-18 years, with a rate of 24.05% among the 262 patients with available age data. This difference may be attributed to advancements in early CE diagnosis and the fact that most cases in this study were based on case reports. However, it is important to note that age data were unavailable for 73.85% of patients, which could potentially influence the observed distribution, as the missing data might disproportionately affect certain age groups and alter the findings. The next highest prevalence was in the 19-30 years (19.85%) and 31-40 years (17.94%) age groups, indicating that CE is also common among young and middle-aged adults. In contrast, a study in Western Romania found the highest prevalence in individuals aged 50-59 years (21.7%) [239], while the current study showed a much lower prevalence in the 51-60 years age group (1.80%). These differences may be attributed to regional variations in risk factors, such as livestock exposure, environmental conditions, and access to healthcare, all of which can influence the age distribution of CE.

The clinical presentation of CE is highly variable, primarily influenced by factors such as the cyst's location, size, and condition. As CE cysts tend to grow slowly over time, they often result in a prolonged asymptomatic phase. Symptoms usually appear when the cysts reach a size that causes pressure effects or functional impairment in the affected organ [238]. Several studies highlight the differences in asymptomatic rates across various organ involvements. Ciftci et al. reported that 52.9% of patients with renal hydatid cysts were asymptomatic, indicating that cysts in the kidneys may remain unnoticed for extended periods until they cause local complications or are incidentally discovered [104]. In contrast, Akhan et al. found a higher rate of asymptomatic cases, 73.68%, among patients with liver hydatid cysts, suggesting that hepatic involvement might often go undetected, potentially due to the liver's capacity to accommodate growth without immediate symptoms [149]. In the present study, clinical presentation for 386 patients was available, and a much lower asymptomatic rate of 13.21% was observed. This discrepancy could be due to differences in cyst locations. It is possible that a higher proportion of patients in this study presented with symptoms due to the cysts being located in more clinically sensitive areas. Additionally, the lower asymptomatic rate might reflect a population with more advanced or larger cysts at the time of diagnosis, thereby increasing the likelihood of symptomatic presentation.

Early detection of CE can greatly improve the success of its management and treatment [240]. The definitive diagnosis of CE typically relies on imaging techniques such as radiology, ultrasound, computed tomography (CT), and magnetic resonance imaging (MRI) [241]. Serological tests like enzyme-linked immunosorbent assay (ELISA) offer high specificity for detecting HD, but a positive result does not accurately reveal the cyst’s location. Conversely, imaging methods provide detailed visualization, identifying cysts at specific sites. This limitation emphasizes combining serological testing with imaging to diagnose and precisely locate the hydatid cyst [242]. In the present study, among the 170 patients with CE who had serological test results available, 18.23% tested negative.

Preventing CE relies on disrupting E. granulosus's life cycle. For instance, regular screening and treatment of infected dogs have effectively eradicated the disease in endemic regions. Additional control measures include restricting the feeding of home-slaughtered livestock to dogs and vaccinating intermediate hosts, such as sheep [243]. Community education plays a crucial role in preventing disease spread by raising awareness of the risks associated with infected animals and contaminated environments. Ongoing monitoring and collaboration among healthcare providers, veterinarians, and the community are essential for early detection and prompt intervention [244].

One limitation of this study is the predominance of case reports among the included studies. While these reports offer valuable insights into rare occurrences, they are inherently limited by small sample sizes, and potential selection bias. As a result, the findings may not be easily generalized to broader populations, and the ability to draw strong conclusions. Further research is needed to address the diagnostic challenges of CE in non-endemic regions, with a focus on improving early detection and treatment. Future studies should prioritize increasing healthcare professionals' knowledge, refining diagnostic processes, and evaluating the effectiveness of serological and imaging tools in regions with low disease prevalence.

Conclusion

Hydatidosis persists as a significant global public health concern, impacting both developing and developed countries. The liver and lungs remain the primary sites of infection. Preventive strategies, including regular animal screening and enhanced public health education, are essential for controlling the spread of the disease.

Declarations

Conflicts of interest: The author(s) have no conflicts of interest to disclose.

Ethical approval: Not applicable.

Consent for participation: Not applicable.

Consent for publication: Not applicable.

Funding: The present study received no financial support.

Acknowledgments: None to be declared.

Authors' contributions: HAN, BAA, FHK, and HOA: major contributors to the conception of the study, as well as the literature search for related studies, and manuscript writing. SMA, SHM, RQS FA, YMM and DAH: Literature review, design of the study, critical revision of the manuscript, and processing of the tables. TMM, MNH, HAH, SHK, KKM, DAO, SHS and KAN: Literature review, data analysis and interpretation. All authors have read and approved the final version of the manuscript.

Use of AI: AI was not used in the drafting of the manuscript, the production of graphical elements, or the collection and analysis of data.

Data availability statement: Not applicable.

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Thyroid Hemiagenesis: A Single-Center Case Series

DOI: https://doi.org/10.70955/JCJ.2025.3

First Published : 2025-06-10

Page 7-11

Abstract

Introduction

Methods

Study design and Setting

Participant Selection and Eligibility Criteria

Data Collection and Variables Assessed

Data Processing and Statistical Analysis

Results

**Table 1.** Demographic, clinical, and surgical characteristics of patients.
Cases	Age (Y)	Gender	History						Clinical Thyroid Examinations
Cases	Age (Y)	Gender	Presentation	Duration (W)	Other Congenital Conditions	PMH	PSH	Drug Hx	Clinical Thyroid Examinations
Case 1	20	M	Neck Swelling	4	None	Hyperthyroidism	Negative	Methimazole	G2
Case 2	21	F	Neck Pain	3	None	Negative	Lymph Node Biopsy	None	G2
Case 3	16	F	Incidental	8	Jaundice, Periumbilical Hernia	Iron Deficiency Anaemia	Hernia Surgery	None	G0
Case 4	<1	M	Incidental	N/A	Jaundice	Hypothyroidism	Negative	Thyroxine	G1
Case 5	31	M	Incidental	N/A	None	Negative	Negative	None	G0
Case 6	55	F	Neck Pain	8	None	Negative	Bilateral Total Knee Replacement	None	G0
Case 7	26	F	Neck Swelling	1	None	Negative	Tonsillectomy	None	G2
Case 8	6	F	Neck Swelling	1	Thyroglossal Duct Cyst	Negative	Tonsillectomy	None	G2
Case 9	55	F	Incidental	N/A	None	Negative	C-section	None	G0
Case 10	53	F	Incidental	1	None	Negative	C-section	None	G0
Case 11	27	M	Incidental	3	None	Negative	Negative	None	G0
M: Male, F: Female, PMH: Past Medical History, PSH: Past Surgical History, Drug Hx: Drug History, G: grades of thyroid enlargement, Y: Years, W: Weeks.

**Table 2.** Laboratory, imaging, and follow-up profiles of included cases.
Cases	Blood Investigations					Ultrasound Reports		Follow up (Months)
Cases	First TSH (µIU/mL)	First Free T4 (pmol/L)	First Total T4 (nmol/mL)	ATPO (IU/mL)	First TRAB (IU/mL)	Agenesis Side	Normal Lobe Size (mm)	Follow up (Months)
Case 1	0.005	55.53	N/A	N/A	12.55	Left Lobe, Isthmus	70 × 26 × 24	48
Case 2	2.45	19.9	N/A	9	N/A	Left Lobe, Isthmus	56 × 19 × 19	12
Case 3	1.8	N/A	165.6	N/A	N/A	Left Lobe, Isthmus	53 × 16 × 14	12
Case 4	100	2.34	N/A	N/A	N/A	Left Lobe	15 × 6.9 × 7.9	8
Case 5	2.61	18.00	N/A	68.3	N/A	Left Lobe, Isthmus	100 × 43 × 35	12
Case 6	1.83	15.86	N/A	109.2	N/A	Left Lobe, Isthmus	64 × 31 × 27	4
Case 7	1.27	16.3	N/A	80.9	N/A	Left Lobe, Isthmus	55 × 19 × 19	6
Case 8	1.33	21.71	N/A	N/A	N/A	Left Lobe, Isthmus	34 × 10 × 13	36
Case 9	2.27	17.02	N/A	11.45	N/A	Left Lobe	48 × 16 × 15	48
Case 10	5.66	9.75	N/A	17.6	N/A	Left Lobe, Isthmus	48 × 18 × 20	4
Case 11	0.005	41.6	9.75	N/A	0.8	Right Lobe	58 × 26 × 25	24
TSH: Thyroid-Stimulating Hormone, T4: Thyroxine, ATPO: Anti-Thyroid Peroxidase Antibodies, TRAB: Thyrotropin Receptor Antibodies, N/A: Not Applicable

**Table 3.** Summary of clinical and demographic findings.
Variables	Frequency (percentage)
Gender Male Female	4 (36.4%) 7 (63.6%)


Age, Years (Mean ± SD)	28.12 ± 18.14
Clinical presentations Incidental Neck Pain Neck Swelling	6 (54.5%) 2 (18.2%) 3 (27.3%)



Past medical history Iron Deficiency Anemia Negative	1 (9.1%) 10 (90.9%)


Clinical thyroid examination G0 G1 G2	6 (54.5%) 1 (9.1%) 4 (36.4%)



Thyroid function status Euthyroid Hyperthyroidism Hypothyroidism	7 (63.6%) 2 (18.2%) 2 (18.2%)



Agenesis Side Right Left	1 (9.1%) 10 (90.9%)

Discussion

Conclusion

Declarations

Conflicts of interest: The authors have no conflicts of interest to disclose.

Ethical approval: Ethical approval for this study was obtained from the Ethics Committee of the Kscien Organization (Approval No. 2025-37)

Consent for participation: Not applicable.

Consent for publication: Written informed consent for publication was obtained from the patients or, in the case of minors, from their parents.

Funding: The present study received no financial support.

Acknowledgements: None to be declared.

Data availability statement: Not applicable.

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Blunt Chest Trauma and Chylothorax: A Systematic Review

DOI: https://doi.org/10.70955/JCJ.2025.4

First Published : 2025-06-10

Page 27-45

Abstract

Introduction

Methods

Literature search

Eligibility criteria

Study selection

Data extraction

Statistical Analysis

Results

Study identification

**Table 1.** Demographic, Cause and Chyle Characteristics
First author, year [Reference]	Country	Age (year)	Gender	CFP	Cause	SOC	Amount of chyle (ml)/day*	COAP (day)	Biochemical content of chyle
Harvey, 2024 ^[5]	USA	60	F	Chest pain, multiple rib fractures	RTA	Left	<500	2	Triglycerides
Burduniuc, 2023 ^[2]	Czech Republic	70	F	Blunt injury	Fall on stairs	Right	>1000-2000	3	Protein, cholesterol, triglycerides
Dung, 2023 ^[14]	Vietnam	32	M	Thoracic spine injury, paraplegia	RTA	Right	>1000-2000	At once	Cholesterol, triglycerides
Kim, 2023 ^[4]	South Korea	45	M	Hemodynamically unstable, chest discomfort, multiple fractures, hemopneumothorax	Crushed by a 2-ton metal frame	Left	>1000-2000	1.66	Triglycerides
Boateng 2023 ^[33]	USA	75	F	Respiratory distress	Fall from bed	Right	<500	At once	Triglycerides
Ruest 2023 ^[34]	USA	15 months	M	Tenderness over right paraspinal thoracolumbar back, abnormal auscultation	Child abuse	Right	N/A	At once	N/A
Mohanakrishnan 2022 ^[35]	USA	70	F	Dyspnea, back pain, abnormal auscultation	Violent coughing episode	Right	>1000-2000	At once	Chylomicrons, triglycerides
Mazhar, 2021^[23]	UK	42	F	Dyspnea, abnormal auscultation	Fall from horse 1 week before presentation	Right	>1000-2000	7	Triglycerides
Waseem, 2021^[32]	Pakistan	50	M	Dyspnea	RTA 2 days before presentation	Bilateral	>1000-2000	2	Triglycerides, cholesterol, fat-rich fluid with few inflammatory cells
Din Dar 2021 ^[36]	India	50	M	Blunt injury	RTA	Right	>1000-2000	25	Triglyceride, chylomicrons
Bacon, 2020 ^[9]	USA	53	M	Hemopneumothorax	RTA	Left	<500	5	N/A
Champion, 2020 ^[12]	Canada	29	M	Dyspnea, flushing, diaphoresis, vomiting, abnormal auscultation	RTA	Bilateral	>1000-2000	At once	Cholesterol, triglycerides
Jindal 2019 ^[37]	India	35	M	Dyspnea, respiratory distress	RTA	Bilateral	>1000-2000	4	Triglyceride, WBC, albumin, glucose, protein, LDH
Ahmed, 2018 ^[1]	Iraq	42	M	Severe back pain	RTA	Right	500-1000	2	Triglycerides and lymphocyte
Brown, 2018 ^[10]	USA	53	M	Thoracoabdominal injuries, subcutaneous emphysema, unstable pelvis	RTA	Left	>1000-2000	N/A	Triglycerides and lymphocyte
Litzau, 2018 ^[22]	USA	66	F	Dyspnea, multiple fractures, abnormal auscultation	RTA 7 days before presentation	Right	>1000-2000	7	Triglycerides
Kozul, 2017 ^[19]	Australia	18	M	Multiple injuries	RTA	Bilateral	500-1000	0.46	N/A
Lee, 2017 ^[21]	South Korea	70	M	Hemothorax, flail chest	RTA	Right	>2000	5	Triglycerides, cholesterol
Mohamed, 2017 ^[3]	USA	51	M	Dyspnea, chest pain, abnormal auscultation	Fall on stairs	Bilateral	>1000-2000	5	Triglycerides, Leukocytes, RBCs, LDH, protein
Spasić, 2017 ^[6]	Serbia	55	F	Multiple injuries	RTA	Right	>2000	5	N/A
Sriprasit, 2017 ^[31]	Thailand	27	F	Hemothorax, neurogenic shock, multiple fractures	RTA	Left	<500	5	Triglycerides, protein, glucose, LDH
Hara 2017 ^[38]	Japan	17	F	Breathing difficulty, abnormal percussion	Recurrent chylothorax, physical punishment	Left	500-1000	At once	N/A
Jahn 2017 ^[39]	Germany	8	F	Respiratory distress, abnormal percussion, and auscultation	Pedestrian hit by a motor vehicle	Left	<500	5	Protein, albumin, LDH, triglycerides, cholesterol
Ghodrati 2016 ^[40]	Iran	12	F	Dyspnea, respiratory distress	Chest trauma during play at school	Bilateral	N/A	N/A	N/A
Lee 2016 ^[41]	South Korea	50	M	Paraplegia	Fall from height during construction work	Right	>1000-2000	3	Triglycerides
Sendama 2015 ^[42]	UK	84	F	Dyspnea, abnormal auscultation	Fall	Right	500-1000	4	Protein, LDH, cholesterol, triglycerides
Snow 2015 ^[43]	USA	22 months	M	Cough, dyspnea, respiratory distress, altered mental status, abnormal auscultation	Fall from a chair	Right	<500	2	Triglycerides
Adams 2013 ^[44]	USA	73	M	Paraplegia, rib fractures, hemothorax	RTA	Right	N/A	9	Triglycerides, lymphocytes, glucose, LDH, lipemia
Kumar 2013 ^[45]	India	32	M	Blunt injury, fracture of right femur	RTA	Right	>1000-2000	2	Triglycerides, chylomicrons
		23	M	Hemopneumothorax, multiple rib fractures, subcutaneous emphysema	RTA	Right	500-1000	1	Triglycerides
		40	M	Hemothorax	RTA	Right	500-1000	2	Triglycerides
Sharkey 2012 ^[46]	UK	50	M	Multiple fractures, hematoma, hemothorax, pneumothorax	RTA	Right	500-1000	N/A	Triglyceride, cholesterol
Sokouti, 2011^[30]	Iran	15	M	Respiratory distress, dyspnea, back pain, abnormal auscultation	Fall 11 years ago	Bilateral	>2000	40	Triglycerides, protein, cholesterol, fat
Kurklinsky 2011 ^[47]	USA	26	F	Pleuritic chest pain	Water-skiing fall	Bilateral	>1000-2000	2	Triglycerides
Apostolakis, 2009 ^[8]	USA	75	F	Dyspnea, back pain	RTA	Bilateral	500-1000	At once	Protein, LDH, glucose, amylase, triglycerides, cholesterol, albumin, globulin, K, Na, lymphocytes, erythrocytes
Apostolakis, 2009 ^[8]	USA	22	M	Back pain, hematoma of left thigh, chest pain	RTA	Bilateral	<500	0.25	Protein, LDH, glucose, amylase, triglycerides, cholesterol, albumin, globulin, K, Na, lymphocytes, erythrocytes
Huber, 2009 ^[16]	USA	47	M	Dyspnea, chest pain	Crushed by multiple metal gates	Bilateral	500-1000	3	Triglycerides
Schurz, 2009 ^[28]	Austria	39	M	Dyspnea, thoracodorsal pain	RTA	Left	>1000-2000	14	N/A
Serin-Ezer, 2009 ^[29]	Turkey	4	M	Dyspnea, somnolence, abnormal auscultation	Hit by a manufacturing pipe falling from a truck	Bilateral	<500	At once	Triglycerides, cholesterol, protein, LDH, glucose
Kamiyoshihara, 2008 ^[18]	Japan	51	M	Dyspnea, dullness in percussion	RTA 20 years before presentation	Bilateral	>1000-2000	At once (but the trauma dated back to 20 years prior)	Triglycerides
Pandey 2008 ^[48]	Australia	36	M	Chest pain, flail chest, hypotension	Fall from balcony	Right	>1000-2000	2	N/A
Lee, 2006 ^[20]	South Korea	11	M	Dyspnea, nausea, vomiting, abdominal discomfort, abnormal auscultation	RTA 3 days before presentation	Bilateral	N/A	3	Triglycerides, cholesterol, protein
Ozcelik, 2004 ^[26]	Turkey	15	F	Respiratory distress, subcutaneous emphysema, pneumothorax	Trapping under rubble during a 7.8 magnitude earthquake	Right	N/A	45	Cholesterol, triglycerides
Robbins 2004 ^[49]	USA	41	M	Chest injury, refractory hiccups, nausea	RTA	Bilateral	N/A	N/A	N/A
Buchan 2001 ^[50]	UK	18	M	Dyspnea	RTA	Right	>1000-2000	4	N/A
Chamberlain, 2000 ^[11]	UK	29	M	Pneumothorax, abdominal and paraspinal pain, loss of motor power, and sensation below T12/L1	RTA	Right	>1000-2000	0.88	N/A
Glyn-Jones 2000 ^[51]	UK	28	M	Dyspnea, polytrauma, tachypnea	RTA	Left	>2000	N/A	Triglyceride, cholesterol, WBC
Golden, 1999 ^[15]	USA	53	F	Chest pain, multiple fractures, hemopneumothorax, abnormal auscultation	RTA	Left	>2000	6	Triglycerides
McCormick, 1999 ^[24]	USA	46	M	Chest pain, dyspnea, abnormal auscultation, dullness to percussion	Hit-and-run motor vehicle accident	Bilateral	>1000-2000	14	Protein, cholesterol, triglycerides
Ikonomidis, 1997 ^[17]	Canada	17	M	Closed head injury, multiple fractures, respiratory distress, tracheal hematoma	RTA	Bilateral	<500	At once	Triglycerides
Ikonomidis, 1997 ^[17]	Canada	24	M	Closed head injury, rib fractures, hemothorax	Snowboarding accident	Left	<500	At once	Triglycerides
Guleserian, 1996 ^[52]	USA	11 months	M	Dyspnea, coughing, cold symptoms, grunting, abnormal percussion	Child abuse	Right	500-1000	N/A	Triglycerides, cholesterol, WBC
Milano, 1994 ^[25]	Italy	26	F	Dyspnea	Fall while skiing 4 months prior	Left	>1000-2000	50	Triglycerides
Fogli, 1993 ^[53]	Italy	31	M	Suspected traumatic hemothorax, dyspnea, cough	RTA	Right	500-1000	N/A	N/A
Grant, 1991 ^[54]	New Zealand	32	M	Dyspnea, chest and back pain	RTA	Right	>1000-2000	N/A	N/A
Dulchavsky, 1988 ^[13]	USA	48	M	Dyspnea, chest pain, abnormal auscultation	Fistfight	Right	>1000-2000	At once	Cholesterol, HDL, triglycerides, pre-beta lipoprotein, chylomicrons
Brook 1988 ^[55]	USA	27	M	Respiratory distress, abnormal percussion, tachycardia	RTA	Bilateral	>2000	3	Triglycerides
Pai, 1984 ^[27]	USA	19	M	Neck, back, and chest pain	RTA	Right	>1000-2000	N/A	N/A
Krishnan 1982 ^[56]	Malaysia	29	M	Dyspnea, abnormal percussion	RTA	Right	>2000	2	N/A
Azambuja 1981 ^[57]	Brazil	42	M	Paraplegia, hemopneumothorax	RTA	Right	N/A	3	N/A
Rea 1960 ^[58]	UK	28	M	Dyspnea, apex beat displaced to the left	Crush injury from falling planks	Right	500-1000	4	Lipid, protein, RBC, lymphocytes
Guest 1955 ^[59]	Canada	19	M	Dyspnea, dry cough, tachypnea, dull percussion	RTA	Right	>1000-2000	26	N/A
Elliot 1948 ^[60]	Canada	56	M	Fall injury, paraplegia, respiratory distress	Fall from a tree	Right	500-1000	3	Fat
Dorsey 1942 ^[61]	USA	60	M	Alcoholic stupor, dyspnea, chest pain, abnormal percussion	Fall down a flight of stairs	Right	>2000	0.5	Protein, albumin, globulin, fat
Cellan-Jones 1940 ^[62]	UK	32	M	Dyspnea, chest tightness	A stone hitting the chest and dorsal spine striking a block of coal	Right	>1000-2000	3	Fat
Brown 1937 ^[63]	USA	N/A	F	Respiratory distress, abdominal distention	RTA	Bilateral	>1000-2000	At once	N/A
Bauersfeld 1937 ^[64]	USA	22	M	Breathing difficulty, laceration of the scalp, pain in lower abdomen and lumbar region, cyanosis, cold extremities	RTA	Right	>2000	8	Fat globules
Lillie 1935 ^[65]	USA	45	M	Blunt injury	Fall from a scaffold 20 feet high	Right	>2000	N/A	N/A
Macnab 1932 ^[66]	Canada	46	M	Chest and back pain, dullness percussion, dyspnea, anorexia, weakness, intermittent fever, hypotension	Fall from a height of 11 feet	Right	500-1000	6	N/A
F: female, M: male, CFP: clinical findings & presentation, SOC: Side of chylothorax, hr: hour, N/A: non-available, COAP: Chyle onset after presentation, RTA: road traffic accident, RBC: red blood cell, LDH: lactate dehydrogenase, HDL: high density lipoprotein, WBC: white blood cell. * The amount of chyle has been grouped rather than the actual amount.

Figure 1. Study selection PRISMA flow chart.

**Table 2.** Imaging findings, treatment and outcomes.
First author, year ^[Reference]	Chest X-ray	CT	MRI	ICU admission	Treatment	Thoracic duct ligation approach	Mode of drainage	Duration of chest tube placement (day)	Follow-up (weeks)	Outcome
Harvey, 2024 ^[5]	Pleural effusion	Hemopneumothorax, pneumomediastinum, retrosternal hematoma, multiple rib fracture, lung contusions, and manubrium fractures	N/A	Yes	Drainage, medium chain fatty acid diet	N/A	Chest tube	7	8	Recovered
Burduniuc, 2023 ^[2]	Pleural effusion, multiple rip fracture	Pleural effusion, multiple rib fracture	Th12 vertebral fracture	Yes	Drainage, thoracic duct ligation	Thoracotomy	Chest tube	N/A	N/A	Recovered
Dung, 2023 ^[14]	Pleural effusion	T9 and T10 vertebral fracture	N/A	No	Drainage, octreotide, TPN, thoracic duct embolization	2.7 Fr microcatheter, fluoroscopic guidance	Chest tube	7	N/A	Recovered
Kim, 2023 ^[4]	Pleural effusion	Lipiodol leakage near T10–11 level	N/A	Yes	Drainage, TPN, intranodal lymphangiography, therapeutic lipiodol injection	N/A	Chest tube	39	N/A	Recovered
Boateng 2023 ^[33]	N/A	Pleural effusion, lung collapse	N/A	No	Drainage, medium-chain triglyceride	N/A	Chest tube	N/A	N/A	Died
Ruest 2023 ^[34]	Pleural effusion	T12 vertebral body fracture, rib fractures	N/A	Yes	Drainage	N/A	Chest tube	N/A	N/A	Recovered
Mohanakrishnan 2022 ^[35]	N/A	Pleural effusion, minimal ascites	N/A	No	Drainage, octreotide, low-fat diet, NPO, TPN, pleurodesis, thoracic duct embolization	Coiling and glue embolization	Chest tube	N/A	N/A	Recovered
Mazhar, 2021^[23]	Pleural effusion	Pleural effusion, T10 spinous process fracture	N/A	No	Drainage, octreotide, medium-chain triglyceride diet	N/A	Chest tube	3	N/A	Recovered
Waseem, 2021^[32]	Pleural effusion	Pleural effusion	N/A	No	Only drainage	N/A	Chest tube	5	N/A	Recovered
Din Dar 2021 ^[36]	N/A	Multiple rib fractures, hemothorax	N/A	No	Drainage, NPO, TPN, octreotide, thoracic duct embolization	Thoracotomy	Chest tube	25	48	Recovered
Bacon, 2020 ^[9]	Multiple rib fracture	Multiple rib fracture	N/A	Yes	Drainage, free-fat diet	N/A	Chest tube	N/A	12	Recovered
Champion, 2020 ^[12]	Pleural effusion	Pleural effusion	N/A	No	Drainage, octreotide, TPN, thoracic duct ligation	Thoracotomy	Chest tube	N/A	6	Recovered
Jindal 2019 ^[37]	Pleural effusion	Multiple rib fractures, lung contusions, fracture of L1 and L2 vertebrae	N/A	Yes	Drainage, thoracic duct ligation, TPN, octreotide, fat-free and medium chain triglyceride	Thoracotomy	Chest tube	8	8	Recovered
Ahmed, 2018 ^[1]	Opacification of hemithorax	D10 vertebral fracture, multiple rib fracture	N/A	Yes	Drainage, low-fat diet, albumin vial, octreotide	N/A	Chest tube
Brown, 2018 ^[10]	N/A	Left temporal epidural hematoma, pulmonary contusions, multiple skeletal fractures, pneumomediastinum compressing the right atrium	Not mentioned the findings	No	Drainage, NPO, TPN, octreotide, thoracic duct embolization, and ligation	Thoracotomy, decortication	Chest tube
Litzau, 2018 ^[22]	Pleural effusion	Pleural effusion	N/A	No	Drainage, low-fat diet	N/A	Chest tube
Kozul, 2017 ^[19]	N/A	Hemopneumothorax, mediastinal shift to the right, pleural effusion	N/A	No	Drainage, No fat/low-fat diet	N/A	Chest tube
Lee, 2017 ^[21]	Pleural effusion	Multiple rib fracture, hemopneumothorax (left), subcutaneous emphysema (left), and atelectasis (right).	N/A	Yes	Drainage, TPN, NPO, fat-free diet, medium-chain lipid diet, thoracic duct ligation, pleurectomy	Thoracotomy	Chest tube
Mohamed, 2017 ^[3]	Obliteration of left costophrenic angle (pleural effusion)	Bilateral effusion	N/A	No	Drainage, fat-free diet with medium-chain triglycerides, octreotide	N/A	Thoracentesis
Spasić, 2017 ^[6]	Lung contusion	Rib and thoracic vertebral fracture, hydropneumothorax, lung contusion, pneumomediastinum	N/A	No	Drainage, TPN, thoracic duct suturing	Thoracotomy	Chest tube
Sriprasit, 2017 ^[31]	N/A	N/A	N/A	Yes	Drainage, NPO, TPN	N/A	Chest tube
Hara 2017 ^[38]	Pleural effusion	N/A	N/A	No	Drainage, low-fat diet with medium-chain triglycerides, intranodal lymphangiography with lipiodol	N/A	Chest tube
Jahn 2017 ^[39]	Pulmonary opacification	Lung contusions	N/A	No	Drainage, fat-free diet	N/A	Chest tube
Ghodrati 2016 ^[40]	Pleural effusion	N/A	N/A	Yes	Drainage, thoracic duct embolization	Unknown	Chest tube
Lee 2016 ^[41]	N/A	Incomplete cord injury at the thoracic spinal vertebrae (T10 and T11)	N/A	No	Drainage, TPN, NPO, thoracic duct ligation	VATS	Chest tube
Sendama 2015 ^[42]	Pleural effusion	Multisegment fracture of L1 vertebra	N/A	No	Drainage, medium chain fatty acid diet, octreotide	N/A	Chest tube
Snow 2015 ^[43]	Opacification of right chest, mediastinal shift to left	N/A	N/A	Yes	Drainage, NPO, TPN, octreotide, low-fat diet	N/A	Chest tube
Adams 2013 ^[44]	Pleural effusion, atelectasis	Pleural effusion, atelectasis	N/A	Yes	Drainage, NPO, TPN, octreotide	N/A	Thoracentesis, chest tube
Kumar 2013 ^[45]	N/A	Pleural effusion, multiple rib fractures	N/A	No	Drainage, NPO, TPN, octreotide, chest physiotherapy	N/A	Chest tube
	N/A	Hemopneumothorax, multiple rib fractures	N/A	No	Drainage, NPO, TPN, octreotide, chest physiotherapy	N/A	Chest tube
	N/A	Bilateral hemothorax, lung contusion	N/A	Yes	Drainage, NPO, TPN, octreotide, exploratory laparotomy for biliary leak	N/A	Chest tube
Sharkey 2012 ^[46]	N/A	N/A	N/A	Yes	Drainage, NPO, TPN, octreotide, medium fatty acid diet	N/A	Chest tube
Sokouti, 2011^[30]	Large cystic mass in left posterior mediastinum	Large low-density cystic mass in the left posterior mediastinum, left pleural effusion	N/A	No	Drainage, thoracic duct ligation, TPN	Laparotomy, Thoracotomy	Chest tube
Kurklinsky 2011 ^[47]	N/A	Pleural effusion, dilated cisterna chyli, middle mediastinum fluid collection	N/A	No	Drainage, TPN, thoracic duct embolization	3 Fr microcatheter with ultrasound guidance	Thoracentesis
Apostolakis, 2009 ^[8]	Pleural effusion	Pleural effusion	N/A	No	Drainage, starvation diet, TPN	N/A	Chest tube
Apostolakis, 2009 ^[8]	Pleural effusion, rib fractures, ipsilateral sternoclavicular joint dislocation	Lung contusion	N/A	No	Drainage, starvation diet, TPN	N/A	Chest tube
Huber, 2009 ^[16]	Pleural effusion	Pleural effusion, right pneumothorax, multiple rib fracture, aortic pseudoaneurysm, retrocrural hemorrhage	N/A	Yes	Drainage, thoracic duct ligation, medium chain fatty acid diet, mechanical pleurodesis	Thoracotomy	Chest tube
Schurz, 2009 ^[28]	Multiple rib fracture, pleural effusion	Pleural effusion	Osseous lesions and pleural effusion	Yes	Drainage, TPN, fat-free diet, plain tea, apple puree	N/A	Pleural puncture, chest tube
Serin-Ezer, 2009 ^[29]	Multiple rib fracture, pleural effusion	Pleural effusion	N/A	No	Drainage, NPO, TPN	N/A	Chest tube
Kamiyoshihara, 2008 ^[18]	Pleural effusion	Pleural effusion	N/A	No	Drainage, low-fat diet, TPN, thoracic duct ligation, pleurodesis	Thoracotomy	Thoracentesis, Chest tube
Pandey 2008 ^[48]	N/A	Hemopneumothorax, pulmonary contusion, multiple rib fractures, pneumomediastinum	N/A	Yes	Drainage, octreotide, thoracic duct ligation	Laparoscopic ligation	Chest tube
Lee, 2006 ^[20]	Elevation of diaphragms, cardiomegaly	Pleural effusion, massive ascites around liver and spleen	N/A	No	Drainage, medium-chain lipid solution, NPO	N/A	Thoracentesis, chest tube
Ozcelik, 2004 ^[26]	Pneumothorax, consolidated right lung, pleural effusion	Right lung consolidation, pleural effusion	N/A	No	Drainage, thoracic duct mass ligation, TPN	Thoracotomy	Chest tube
Robbins 2004 ^[49]	N/A	Pleural effusion, focal fluid collection	N/A	No	EUS-guided aspiration, injection of sodium morrhuate	N/A	Aspiration
Buchan 2001 ^[50]	Pleural effusion	N/A	N/A	No	Drainage, low-fat diet, medium-chain triglycerides, thoracic duct ligation	Thoracotomy	Chest tube
Chamberlain, 2000 ^[11]	Pneumothorax, hemithorax opacification	Free abdominal gas	Fractures of T4 and T10 with spinal cord contusion and hematoma	No	Drainage, TPN, NPO, Supradiaphragmatic duct ligation	Thoracotomy	Chest tube
Glyn-Jones 2000 ^[51]	Mediastinal shift	Minor anterior wedge fractures at T5 and T10	Cord injury at T10	No	Drainage, thoracic duct ligation, pleurodesis, fat-free diet	Thoracotomy	Chest tube
Golden, 1999 ^[15]	N/A	N/A	N/A	Yes	Drainage, TPN, NPO, thoracic duct ligation	Thoracotomy	Chest tube
McCormick, 1999 ^[24]	Pleural effusion	Disruption of the thoracic duct at the T5 level	N/A	No	Only drainage	N/A	Chest tube
Ikonomidis, 1997 ^[17]	Pneumomediastinum, pulmonary contusions	N/A	N/A	No	Drainage, TPN, bowel rest	N/A	Chest tube
Ikonomidis, 1997 ^[17]	Left hemothorax	Left mediastinal hematoma, T3 vertebral fracture	N/A	No	Drainage, TPN, bowel rest	N/A	Chest tube
Guleserian, 1996 ^[52]	Right lung opacification and mediastinal shift to left	N/A	N/A	No	Drainage, nasogastric feeding with medium-chain triglycerides, low-fat diet	N/A	Chest tube
Milano, 1994 ^[25]	Pleural effusion	Dense lymphatic opacification at L1-L2, chyloma at D11, pleural leakage from left duct	N/A	No	Drainage, low-fat diet, medium-chain triglycerides, TPN, pleuroperitoneal shunt	N/A	Thoracentesis
Fogli, 1993 ^[53]	Pleural effusion, mediastinal shift	N/A	N/A	No	Drainage, TPN	N/A	Chest tube
Grant, 1991 ^[54]	Pleural effusion	N/A	N/A	No	Drainage, thoracic duct ligation, TPN, low-fat diet	Thoracotomy	Thoracocentesis, chest tube
Dulchavsky, 1988 ^[13]	Pleural effusion	N/A	N/A	No	Drainage, TPN, NPO, thoracic duct ligation	Thoracotomy	Chest tube	N/A	144	Recovered
Brook 1988 ^[55]	Pleural effusion	N/A	N/A	Yes	Drainage, NPO, TPN, low-fat/ high-protein diet	N/A	Chest tube	10	32	Recovered
Pai, 1984 ^[27]	Fracture dislocations of C6-C7 and T11-T12, right hemothorax	N/A	N/A	No	Drainage, fat-free diet, TPN, thoracic duct ligation, parietal pleurectomy	Thoracotomy	Chest tube	N/A	N/A	Recovered
Krishnan 1982 ^[56]	Pleural effusion, obliteration of left costophrenic angle, multiple rib fractures	N/A	N/A	No	Drainage, thoracic duct ligation	Thoracotomy	Chest tube	19	5	Recovered
Azambuja 1981 ^[57]	Hemopneumothorax	N/A	N/A	No	Drainage, thoracic duct ligation, pleural flap to address fistula, pleural abrasion	Thoracotomy	Chest tube	6	N/A	Recovered
Rea 1960 ^[58]	Opaque hemithorax	N/A	N/A	No	Drainage, thoracic duct ligation	Thoracotomy	Chest tube	N/A	N/A	Recovered
Guest 1955 ^[59]	N/A	N/A	N/A	No	Aspiration, high-protein, low-fat diet	N/A	Thoracentesis	N/A	4	Recovered
Elliot 1948 ^[60]	Pleural effusion	N/A	N/A	No	Aspiration, thoracic duct ligation	Thoracotomy	Aspiration	N/A	N/A	Recovered
Dorsey 1942 ^[61]	Rib fracture, pleural effusion	N/A	N/A	No	Drainage, low-fat diet, high-carb, high-protein diet, NPO	N/A	Thoracentesis	N/A	N/A	Died due to uncontrolled leakage
Cellan-Jones 1940 ^[62]	Pleural effusion	N/A	N/A	No	Aspiration, low-fat diet, intravenous glucose-saline	N/A	Aspiration	N/A	N/A	Died due to uncontrolled leakage
Brown 1937 ^[63]	Pleural effusion	N/A	N/A	No	Drainage, dietary management	N/A	Thoracentesis, paracentesis	N/A	N/A	Died
Bauersfeld 1937 ^[64]	Pleural effusion, mediastinal shift	N/A	N/A	No	Drainage, intravenous dextrose, high-calorie diet	N/A	Thoracentesis	16	N/A	Recovered
Lillie 1935 ^[65]	Pleural effusion, mediastinal displacement	N/A	N/A	No	Drainage, fat-free diet	N/A	Thoracentesis	N/A	N/A	Recovered
Macnab 1932 ^[66]	Displacement of the heart, pleural effusion	N/A	N/A	No	Drainage, carbohydrates, protein	N/A	Aspiration	48	2	Died due to extreme asthenia
CT: computed tomography, MRI: magnetic resonance imaging, ICU: intensive care unit, NPO: Nulla Per Os, TPN: total parenteral nutrition, N/A: non-available, EUS: endoscopic ultrasound

Presentation and etiology

**Table 3.** Summary of findings of the reviewed cases
Variables	Frequency / Percentage
Patient demography
Age range (mean ± SD), years	11 months – 84 (37.4 ± 19.9)
Gender Male Female	51 (73.91%) 18 (26.09%)


Common presentation and clinical findings* Dyspnea Abnormal auscultation or percussion Multiple fractures or injuries Chest pain Pneumothorax/ hemothorax/ hemopneumothorax Back pain Respiratory distress	33 (47.83%) 24 (34.78%) 19 (27.54%) 15 (21.74%) 14 (20.29%) 11 (15.94%) 11 (15.94%)







Cause of blunt trauma Road traffic accident Fall Hit or crushed by heavy objects Physical punishment & child abuse Fistfight Others	41 (59.42%) 16 (23.19%) 6 (8.70%) 3 (4.34%) 1 (1.45%) 2 (2.90%)






Side of chylothorax Right Left Bilateral	38 (55.07%) 12 (17.39%) 19 (27.54%)



Amount of chyle (ml/day) <500 500 - 1000 >1000-2000 >2000 N/A	10 (14.50%) 14 (20.28%) 28 (40.58%) 10 (14.50%) 7 (10.14%)





Chyle onset after presentation (day) At once – 2 days 3 days – one week > one week – one month > one month N/A	28 (40.58%) 22 (31.88%) 6 (8.70%) 3 (4.34%) 10 (14.50%)





Biochemical content of chyle Lipid Lipid + Protein Lipid + Inflammatory cells Lipid + Protein + Sugar + Inflammatory cells + Ions Lipid + Protein + Sugar Lipid + Inflammatory cells + Protein N/A	28 (40.57%) 8 (11.59%) 5 (7.24%) 4 (5.80%) 2 (2.90%) 2 (2.90%) 20 (29.00%)







Imaging findings
Chest X-rays* Pleural effusion Rib Fracture Lung/ mediastinal/ heart shift Opacification of lung Pneumothorax/ hemothorax Lung contusion Pneumomediastinum Vertebral fracture Lung consolidation Others N/A	38 (55.07%) 7 (10.14%) 7 (10.14%) 6 (8.70%) 5 (7.24%) 2 (2.90%) 1 (1.45%) 1 (1.45%) 1 (1.45%) 5 (7.24%) 15 (21.74%)











CT scan findings* Pleural effusion Rib fracture Vertebral fracture Pneumothorax/ hemothorax/ hemopneumothorax Lung contusion Pneumomediastinum Hematoma Thoracic duct leakage Others N/A	23 (33.33%) 14 (20.29%) 10 (14.50%) 9 (13.04%) 8 (11.59%) 4 (5.80%) 3 (4.34%) 2 (2.90%) 17 (24.64%) 25 (36.23%)










ICU admission Yes No	19 (27.54%) 50 (72.46%)


Common treatment approach* Drainage Parenteral nutrition Thoracic duct ligation/embolization/suturing Medium-chain fatty acid or low-fat diet Nulla per Os Free fat diet/starvation diet Octreotide Pleurectomy/Pleurodesis	65 (94.20%) 35 (50.72%) 27 (39.13%) 24 (34.78%) 19 (27.54%) 12 (17.39%) 17 (24.64%) 6 (8.70%)








Thoracic duct closure approach Thoracotomy Fr microcatheter with fluoroscopic/ ultrasound guidance VATS/ laparoscopy Coiling and glue embolization Unknown Not performed	22 (31.88%) 2 (2.90%) 2 (2.90%) 1 (1.45%) 1 (1.45%) 41 (59.42%)






Mode of drainage Chest tube Thoracentesis Aspiration Chest tube + Thoracentesis	53 (76.81%) 9 (13.04%) 4 (5.80%) 3 (4.34%)




Duration of chest tube placement ≤ One week > One week – two weeks > Two weeks – one month > One month N/A	12 (17.39%) 11 (15.94%) 13 (18.84%) 4 (5.80%) 29 (42.03%)





Outcome Recovered Partially recovered Died	62 (89.85%) 1 (1.45%) 6 (8.70%)



SD: standard deviation, CT: computed tomography, ICU: intensive care unit, VATS: video-assisted thoracoscopic surgery, N/A: non-available. *Each data in the variable might be found in more than one case

Imaging characteristics and management

Discussion

Conclusion

Declarations

Conflicts of interest: The authors have no conflicts of interest to disclose.

Ethical approval: Not applicable.

Consent for participation: Not applicable.

Consent for publication: Not applicable.

Funding: The present study received no financial support.

Acknowledgements: None to be declared.

Data availability statement: Not applicable.

More.. PDF

Abstract PDF

Hyalinizing Trabecular Tumor: A Case Series with Literature Review

DOI: https://doi.org/10.70955/JCJ.2025.2

First Published : 2025-05-24

Page 1-6

250

Abstract

Conclusion: A rare benign tumor, HTT remains challenging to diagnose accurately. Both total thyroidectomy and lobectomy may result in good outcomes.

Introduction

Methods

Study design

Participants

Pre-intervention assessment

Assessments included vital signs monitoring, ultrasonography (U/S), thyroid function tests, serum calcium levels, vocal cord evaluation, viral screening (HBV, HCV, and HIV), and complete blood count.

Intervention

Post-intervention Considerations

Results

Participants

Outcomes and follow-up

**Table 1.** Clinical and sociodemographic data of the patients.
Case no.	Age	Sex	Medical history	Surgical history	Drug history	Chief complaint	Duration	Examination	Thyroid function test
1	39	F	Unremarkable	Unremarkable	Unremarkable	Incidental	1 week	Not palpable	Hyperthyroid
2	78	F	Hypertension	Cataract surgery & cholecystectomy	Statin, Anticoagulant & Anti-Hypertension	Shortness of breath	8 years	Hard	Hyperthyroid
3	74	M	Unremarkable	Kidney transplant	Corticosteroids	Weight-loss	5 months	Not palpable	Hyperthyroid
4	85	F	Hypertension & renal stone	Thyroid lobectomy & lithotripsy	Anti-Hypertension	Anterior neck swelling	6 years	Hard	Euthyroid
5	32	F	Unremarkable	Unremarkable	Unremarkable	Anterior neck swelling	3 months	Hard	Euthyroid
6	34	F	Hodgkin’s lymphoma	C-section & cervical cerclage	Antibiotics	Weight-loss	8 months	Not palpable	Euthyroid
7	38	F	Unremarkable	Cholecystectomy	Unremarkable	Anterior neck swelling	1.5 years	Hard	Hyperthyroid
8	42	F	Unremarkable	Unremarkable	Unremarkable	Anterior neck swelling	1 year	Hard	Euthyroid
9	51	F	Hypertension & Type 2 Diabetes Mellitus	Unremarkable	Anti-Hypertension & Anti-diabetics	Anterior neck swelling	2 months	Hard	Hyperthyroid
10	40	F	Unremarkable	Unremarkable	Unremarkable	Weight-loss	6 months	Not palpable	Euthyroid
11	45	F	Unremarkable	Unremarkable	Unremarkable	Anterior neck swelling	1 year	Hard	Hyperthyroid
M:Male, F:Female, S.Ca:Serum calcium, TG:Thyroglobulin, N/A:Not applicable

**Table 2.** Diagnostics and management.
Case no.	S. Ca (mg/dL)	US	US consistency	US echogenicity	Side	FNA Bethesda	Vocal cord assessment	Type of operation	Post Op complications	HPE	Tumor Size (cm)	Follow-up (years)
1	N/A	N/A	N/A	N/A	Left lobe	V	Normal	Lobectomy	None	HTT	1.4	5
2	9.6	TR3	Mixed	Heterogenous	Left lobe	IV	Normal	Total thyroidectomy	None	HTT	3.8	2
3	7.9	GD	No nodule	No nodule	Isthmus	NP	Normal	Total thyroidectomy	None	HTT	1	2
4	9.8	GD	No nodule	No nodule	Right lobe	NP	Normal	Total thyroidectomy	None	HTT	6	0.08
5	9.2	TR4	Solid	Hyperechoic	Left lobe	V	Normal	Total thyroidectomy	None	HTT	4.5	3
6	9.5	TR4	Solid	Hyperechoic	Left lobe	V	Normal	Total thyroidectomy	None	HTT	0.5	3
7	9.1	TR3	Solid	Heterogenous	Left lobe	NP	Normal	Lobectomy	None	HTT	1.4	1
8	9.5	TR4	Solid	Hypoechoic	Right lobe	IV	Normal	Lobectomy	None	HTT	3	2
9	9	TR3	N/A	N/A	Isthmus	NP	Normal	Total thyroidectomy	None	HTT	0.8	2
10	9.2	TR3	N/A	N/A	Right lobe	III	Normal	Total thyroidectomy	None	HTT	0.7	3
11	8.9	TR3	N/A	N/A	Right lobe	NP	Normal	Lobectomy	None	HTT	5.5	3
N/A: Not Applicable, S.Ca: Serum Calcium, US: Ultrasound, FNA: Fine Needle Aspiration, OP: Operation, HPE: Histopathology, TR: TI-RADS, GD: Graves' Disease, NP: Non-Productive, HTT: Hyalinizing trabecular tumor

**Table 3.** Patient demographics and clinical data.
Variables	Frequency
Sex
Male	1 (9.1%)
Female	10 (90.9)
Age groups (years)
30-39	4 (36.3%)
40-49	3 (27.3%)
50-59	1 (9.1%)
>60	3 (27.3%)
Mean ± SD	50.7 ± 19.0
Medical history
Unremarkable	7 (63.6%)
Hypertension	1 (9.1%)
Hypertension & renal stones	1 (9.1%)
Hodgkin’s lymphoma	1 (9.1%)
Hypertension & Type 2 Diabetes Mellitus	1 (9.1%)
Surgical history
Unremarkable	6 (54.5%)
Cholecystectomy	1 (9.1%)
C-section & cervical cerclage	1 (9.1%)
Cholecystectomy & cataract surgery	1 (9.1%)
Kidney transplant	1 (9.1%)
Thyroid lobectomy & lithotripsy	1 (9.1%)
Drug history
Negative	6 (54.5%)
Corticosteroids	1 (9.1%)
Antibiotics	1 (9.1%)
Antihypertensive	1 (9.1%)
Antihypertensive & Antidiabetic	1 (9.1%)
Chief complaint
Anterior neck swelling	6 (54.5%)
Shortness of breath	1 (9.1%)
Weight loss	3 (27.3%)
Incidental	1 (9.1%)
Thyroid examination
Not palpable	7 (63.6%)
Hard	4 (36.4%)
Thyroid function
Hyperthyroid	6 (54.5%)
Euthyroid	5 (45.5%)
Affected side
Right lobe	4 (36.3%)
Left lobe	5 (45.5%)
Isthmus	2 (18.2%)
Operation
Total thyroidectomy	7 (63.6%)
Lobectomy	4 (36.4%)
Tumor size (cm)
Mean ± SD	2.6 ± 1.95
Follow-up (years)
0.0 - 1.0	2 (18.18%)
1.1 - 2.0	4 (36.36%)
2.1 - 3.0	4 (36.36%)
3.1 - 4.0	0 (0.0%)
4.1 - 5.0	1 (9.09 %)
Mean ± SD	2.3 ± 1.27

Discussion

**Table 4.** Summary of the cases of hyalinizing trabecular tumor in the literature.
Author/year	Age	Sex	Medical history	Drug history	Surgical history	Chief complaint	Examination	Duration (years)	Side	Size (cm)*	Distant metastasis	TI-RADS	FNA findings	Therapeutic approach	IHC findings	Diagnosis	Outcome	Follow-up (years)
Zhang et al./2025 [7]	31	F	Unremarkable	Unremarkable	Unremarkable	Incidental	Palpable mass	1.5	R	2.2	No	TR3	N/A	Lobectomy	TG +ve, CK19 +ve, TTF-1 +ve & Ki-67 5%	HTT	Resolved	0.6
Hayashi et al./2025 [2]	93	F	Diabetes mellitus, hyperlipidemia, hypertension & myocardial infarction	N/A	N/A	Loss of appetite, dyspnea & dysphagia	Enlarged, firm, non-tender, without palpable nodules	>12	B	>10	No	N/A	N/A	Conservative management & rehabilitation	N/A	HTT	Improved	N/A
Alsogair et al./2023 [3]	60	M	Unremarkable	Unremarkable	Hemorrhoidectomy	Mass on the neck	Palpable, firm & non-tender	N/A	R	3.8	No	N/A	60%-75% likelihood of malignancy	Total thyroidectomy followed by thyroxine	TG +ve & Ki-67 +ve	HTT	Resolved	0.06
Katano et al./ 2021 [10]	54	F	Panic disorder & chronic thyroiditis	Unremarkable	Unremarkable	Left cervical mass	Growing, painless & elastic	N/A	L	4.5	No	N/A	Chronic thyroiditis with possible malignancy	Lobectomy	Ki-67 +ve for cytoplasm & ColIV +ve	HTT	Resolved	1.5
Rhee et al./2018 [9]	63	F	Breast cancer	N/A	N/A	Incidental	N/A	N/A	L	0.6	No	N/A	Features of PTC	Lobectomy	Ki-67 +ve, CD56 +ve & Galectin-3 +ve	HTT	Resolved	N/A
Jones et al./2017 [8]	70	F	N/A	N/A	N/A	Incidental	N/A	N/A	R	1.94	N/A	N/A	60-75% likelihood of malignancy	Total thyroidectomy	TG +ve, vimentin +ve & CK19 +ve	HTT	Resolved	0.08
M: Male, F: Female, N/A: Not applicable, FNA: Fine needle aspiration, IHC: Immunohistochemistry, HTT: Hyalinizing trabecular tumor, L: Left, R: Right, B: Both

Conclusion

In conclusion, HTT is a rare tumor that is challenging to diagnose accurately. Both total thyroidectomy and lobectomy may result in good outcomes.

Declarations

Conflicts of interest: The authors have no conflicts of interest to disclose.

Ethical approval: Ethical approval for this study was obtained from the Ethics Committee of the Kscien Organization (Approval No. 2025-33)

Consent for participation: Not applicable.

Consent for publication: Written informed consent for publication was obtained from all patients.

Funding: The present study received no financial support.

Acknowledgments: None to be declared.

Data availability statement: Not applicable.

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Current Perspectives on Cystic Echinococcosis: A Systematic Review

DOI: https://doi.org/10.70955/JCJ.2025.1

First Published : 2025-02-02

Page 12-26

843

Abstract

Introduction

Methods

Study design

This systematic review was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines.

Data sources and search strategy

Eligibility criteria

Study selection and data extraction

Statistical analyses

Results

Figure 1. The PRISMA diagram illustrating the study selection process

**Table 1**. The details of the included studies
First Author, Year	Study design	Country	No. of cases	First Author, Year	Study design	Country	No. of cases	First Author, Year	Study design	Country	No. of cases
AlRashed, 2024 ^[¹^]	A	Saudi Arabia	1	Mutlu, 2022 ^[⁸²^]	A	Turkey	1	Gatt, 2020 ^[¹⁵⁷^]	A	Israel	1
Amer, 2024 ^[⁶^]	A	Iran	1	Ozgokce, 2022 ^[⁸³^]	A	Turkey	1	Giri, 2020 ^[¹⁵⁸^]	A	Bhutan	1
Babiker, 2024 ^[⁷^]	A	Qatar	1	Passarelli, 2022 ^[⁸⁴^]	A	US	1	Gopivallabha, 2020 ^[¹⁵⁹^]	A	India	1
Bazzi, 2024 ^[⁸^]	A	Lebanon	1	Pulavarty, 2022 ^[⁸⁵^]	A	India	1	Delgado, 2020 ^[¹⁶⁰^]	A	Spain	1
Brezeanu, 2024 ^[⁹^]	A	Romania	1	Rodríguez-Laiz, 2022 ^[⁸⁶^]	A	Spain	1	Handran, 2020 ^[¹⁶¹^]	A	US	1
Calu, 2024 ^[¹⁰^]	A	Romania	2	Sezer, 2022 ^[⁸⁷^]	A	Turkey	1	İriz, 2020 ^[¹⁶²^]	A	Turkey	1
Chen, 2024 ^[¹¹^]	A	Taiwan	1	Shahid, 2022 ^[⁸⁸^]	A	Pakistan	1	İyigün, 2020 ^[¹⁶³^]	A	Turkey	1
Darestani, 2024 ^[¹²^]	A	Iran	1	Sharma, 2022 ^[⁸⁹^]	A	India	1	Jarovsky, 2020 ^[¹⁶⁴^]	A	Brazil	1
Ghaedamini, 2024 ^[¹³^]	A	Iran	1	Sozutok, 2022 ^[⁹⁰^]	A	Turkey	1	Johny, 2020 ^[¹⁶⁵^]	A	India	1
Gulati, 2024 ^[¹⁴^]	A	US	1	Sun, 2022 ^[⁹¹^]	B	China	2	Kankilic, 2020 ^[¹⁶⁶^]	B	Turkey	6
Hasnaoui, 2024 ^[¹⁵^]	A	Tunisia	1	Ulusoy, 2022 ^[⁹²^]	A	Turkey	1	Kaskar, 2020 ^[¹⁶⁷^]	A	India	1
Haydar, 2024 ^[¹⁶^]	A	Iran	1	Uzunoğlu, 2022 ^[⁹³^]	A	Turkey	1	Kiran, 2020 ^[¹⁶⁸^]	A	India	1
Jalayeri, 2024 ^[¹⁷^]	A	Iran	1	Wang, 2022 ^[⁹⁴^]	A	China	1	Kırmacı, 2020 ^[¹⁶⁹^]	A	Turkey	1
Jellali, 2024 ^[¹⁸^]	A	Tunisia	1	Agarwal, 2021 ^[⁹⁵^]	A	India	1	Kumar, 2020 ^[¹⁷⁰^]	A	India	1
Karahan, 2024 ^[¹⁹^]	A	India	1	Aghajanzadeh, 2021 ^[⁴^]	A	Iran	1	Lahdhil, 2020 ^[¹⁷¹^]	A	Tunisia	1
Koren, 2024 ^[²⁰^]	A	Israel	1	Aili, 2021 ^[⁹⁶^]	A	China	1	Lapierre, 2020 ^[¹⁷²^]	A	Canada	1
Mahesan, 2024 ^[²¹^]	A	India	1	Akhan, 2021 ^[⁹⁷^]	A	Turkey	1	Llanos, 2020 ^[¹⁷³^]	A	US	1
Bouhout, 2024 ^[²²^]	A	Morocco	1	Basne, 2021 ^[⁹⁸^]	A	Nepal	1	Lodhia, 2020 ^[¹⁷⁴^]	A	Tanzania	2
Manuel, 2024 ^[²³^]	A	Angola	1	Biswas, 2021 ^[⁹⁹^]	A	India	3	Lyske, 2020 ^[¹⁷⁵^]	A	Canada	1
Mierzejewski, 2024 ^[²⁴^]	A	Poland	1	Boumarah, 2021 ^[¹⁰⁰^]	A	Saudi Arabia	1	Ma, 2020 ^[¹⁷⁶^]	E	China	195
Mutlu, 2024 ^[²⁵^]	A	Turkey	1	Çankaya, 2021 ^[¹⁰¹^]	A	Turkey	1	Mitrovic, 2020 ^[¹⁷⁷^]	A	Serbia	1
Remmerswaal, 2024 ^[²⁶^]	A	Netherlands	1	Cathomas, 2021 ^[¹⁰²^]	A	Switzerland	2	Mittal, 2020 ^[¹⁷⁸^]	A	India	1
Reyimu, 2024 ^[²⁷^]	A	China	1	Chatzifotiou, 2021 ^[¹⁰³^]	A	Germany	1	Moghtadaie, 2020 ^[¹⁷⁹^]	A	Iran	1
Sączek, 2024 ^[²⁸^]	A	Poland	1	Christodouli dis, 2021 ^[³^]	C	Greece	50	Nistor, 2020 ^[¹⁸⁰^]	A	Romania	1
Thakar, 2023 ^[²⁹^]	A	India	1	Ciftci, 2021 ^[¹⁰⁴^]	C	Turkey	34	Ogul, 2020 ^[¹⁸¹^]	A	Turkey	1
Voicu, 2024 ^[³⁰^]	A	Romania	1	Conlon, 2021 ^[¹⁰⁵^]	A	Ireland	1	Pappas, 2020 ^[¹⁸²^]	A	Greece	1
Aarif, 2023 ^[³¹^]	A	India	1	Elvan-Tuz, 2021 ^[¹⁰⁶^]	A	Turkey	2	Ramia, 2020 ^[¹⁸³^]	C	Spain	71
Alsulami, 2023 ^[³²^]	A	Saudi Arabia	1	Gautam, 2021 ^[¹⁰⁷^]	A	India	1	Samadian, 2020 ^[¹⁸⁴^]	A	Iran	1
Ammar, 2023 ^[³³^]	A	Tunisia	1	Ghabisha, 2021 ^[¹⁰⁸^]	A	Yemen	1	Sangal, 2020 ^[¹⁸⁵^]	A	India	1
Borni, 2023 ^[³⁴^]	A	Tunisia	1	Gonder, 2021 ^[¹⁰⁹^]	C	Turkey	9	Sauteur, 2020 ^[¹⁸⁶^]	A	Switzerland	1
Carrel, 2023 ^[³⁵^]	A	Uzbekistan	1	Govindaraj, 2021 ^[¹¹⁰^]	A	India	1	Savu, 2020 ^[¹⁸⁷^]	A	Romania	1
Casulli, 2023 ^[³⁶^]	A	Italy	1	Guha, 2021 ^[¹¹¹^]	A	India	1	Schleenvoigt, 2020 ^[¹⁸⁸^]	A	Germany	1
Caushi, 2023 ^[³⁷^]	A	Albania	1	Hãlmaciu, 2021 ^[¹¹²^]	A	Romania	1	Singh, 2020 ^[¹⁸⁹^]	A	India	1
Das, 2023 ^[³⁸^]	A	India	1	Harmouchi, 2021 ^[¹¹³^]	A	Morocco	1	Singh, 2020 ^[¹⁹⁰^]	A	India	1
Galvis, 2023 ^[³⁹^]	A	Colombia	1	Helvaci, 2021 ^[¹¹⁴^]	A	Turkey	1	Singla, 2020 ^[¹⁹¹^]	A	India	1
Göktürk, 2023 ^[⁴⁰^]	A	Turkey	1	Hermosa, 2021 ^[¹¹⁵^]	A	Spain	1	Sonsoz, 2020 ^[¹⁹²^]	A	Turkey	1
Hakimi, 2023 ^[⁴¹^]	A	Afghanistan	1	Iken, 2021 ^[¹¹⁶^]	A	Morocco	1	Tekin, 2020 ^[¹⁹³^]	A	Turkey	1
Hasnaoui, 2023 ^[⁴²^]	A	Tunisia	1	Jaén-Torrejimeno, 2021 ^[¹¹⁷^]	C	Spain	287	Tlili, 2020 ^[¹⁹⁴^]	A	Tunisia	1
Jia, 2023 ^[⁴³^]	A	China	1	Jindal, 2021 ^[¹¹⁸^]	A	India	1	Tonkaz, 2020 ^[¹⁹⁵^]	A	Turkey	1
Kardoun, 2023 ^[⁴⁴^]	A	Tunisia	1	Kafadar, 2021 ^[¹¹⁹^]	A	Turkey	1	Van De, 2020 ^[¹⁹⁶^]	A	Korea	2
Lao, 2023 ^[⁴⁵^]	A	China	1	Kankam, 2021 ^[¹²⁰^]	A	Iran	1	Vasilescu, 2020 ^[¹⁹⁷^]	A	Romania	1
Lees, 203 ^[⁴⁶^]	A	UK	1	Kechiche, 2021 ^[¹²¹^]	B	Tunisia	10	Verma, 2020 ^[¹⁹⁸^]	A	India	1
Li, 2023 ^[⁴⁷^]	A	China	1	Khasawneh, 2021 ^[¹²²^]	A	Jordan	2	Villalobos, 2020 ^[¹⁹⁹^]	A	US	1
Ma, 2023 ^[⁴⁸^]	A	China	1	Kumar, 2021 ^[¹²³^]	A	India	1	Xu, 2020 ^[²⁰⁰^]	A	China	1
Maggioni, 2023 ^[⁴⁹^]	A	Italy	1	Li, 2021 ^[¹²⁴^]	A	China	1	Yang, 2020 ^[²⁰¹^]	A	China	1
Mahmood, 2023 ^[⁵⁰^]	A	Pakistan	1	Maliqari, 2021 ^[¹²⁵^]	A	Albania	1	Yimamu, 2020 ^[²⁰²^]	A	China	1
Mayekar, 2023 ^[⁵¹^]	A	India	1	Moshref, 2021 ^[¹²⁶^]	A	Saudi Arabia	1	Abbas, 2019 ^[²⁰³^]	A	Morocco	1
Moraes, 2023 ^[⁵²^]	A	Brazil	1	Mozafar, 2021 ^[¹²⁷^]	A	Iran	1	Aydin, 2019 ^[²⁰⁴^]	A	Turkey	1
Moscatelli, 2023 ^[⁵³^]	A	Argentina	1	Rabhi, 2021 ^[¹²⁸^]	A	Tunisia	1	Banerjee, 2019 ^[²⁰⁵^]	A	India	1
Ntombela, 2023 ^[⁵⁴^]	A	South Africa	2	Rhissassi, 2021 ^[¹²⁹^]	A	Morocco	1	Beyhan, 2019 ^[²⁰⁶^]	A	Turkey	1
Peralta, 2023 ^[⁵⁵^]	A	Ecuador	1	Safari, 2021 ^[¹³⁰^]	A	Iran	1	Bracha, 2019 ^[²⁰⁷^]	A	Israel	2
Peulier‐Maitre, 2023 ^[⁵⁶^]	A	France	1	Shakerian, 2021 ^[¹³¹^]	A	Iran	1	Chaouch, 2019 ^[²⁰⁸^]	A	Tunisia	1
Ruíz-Pérez, 2023 ^[⁵⁷^]	A	Peru	1	Shariﬁ, 2021 ^[¹³²^]	A	Iran	1	Demir, 2019 ^[²⁰⁹^]	A	Turkey	1
Safarpour, 2023 ^[⁵⁸^]	A	Iran	2	Sharma, 2021 ^[¹³³^]	A	India	1	Derbel, 2019 ^[²¹⁰^]	A	Tunisia	1
Shah, 2023 ^[⁵⁹^]	A	India	1	Shuaibi, 2021 ^[¹³⁴^]	A	US	1	Gök, 2019 ^[²¹¹^]	A	Turkey	1
Türkoğlu, 2023 ^[⁶⁰^]	A	Turkey	1	Simsek, 2021 ^[¹³⁵^]	A	Turkey	3	Kandemirli, 2019 ^[²¹²^]	A	Turkey	1
Wang, 2023 ^[⁶¹^]	A	China	1	Şimşek, 2021 ^[¹³⁶^]	A	Turkey	1	Kang, 2019 ^[²¹³^]	A	Korea	1
Ahmady‑Nezhad, 2023 ^[⁶²^]	A	Iran	1	Singh, 2021 ^[¹³⁷^]	A	India	1	Kaya, 2019 ^[²¹⁴^]	A	Turkey	1
Assefa, 2022 ^[⁶³^]	A	Ethiopia	1	Ucar, 2021 ^[¹³⁸^]	A	Turkey	1	Khullar, 2019 ^[²¹⁵^]	A	India	1
Bicer, 2022 ^[⁶⁴^]	A	Turkey	1	van Zijl, 2021 ^[¹³⁹^]	A	South Africa	1	Kuzmanovska, 2019 ^[²¹⁶^]	A	Macedonia	2
Bishnoi, 2022 ^[⁶⁵^]	A	India	1	Velho, 2021 ^[¹⁴⁰^]	A	Portugal	1	MadissonBernardo, 2019 ^[²¹⁷^]	A	Brazil	1
Castro, 2022 ^[⁶⁶^]	A	Brazil	1	Wang, 2021 ^[¹⁴¹^]	A	China	1	Magistri, 2019 ^[²¹⁸^]	C	Italy	15
Dantis, 2022 ^[⁶⁷^]	A	India	1	Wu, 2021 ^[¹⁴²^]	A	China	1	Milosavljevic, 2019 ^[²¹⁹^]	A	Serbia	1
Dere, 2022 ^[⁶⁸^]	A	Turkey	1	Yasin, 2021 ^[¹⁴³^]	A	Malaysia	1	Ramteke, 2019 ^[²²⁰^]	A	India	2
Fourati, 2022 ^[⁶⁹^]	A	Tunisia	1	Zedelj, 2021 ^[¹⁴⁴^]	A	Croatia	1	Sharma, 2019 ^[²²¹^]	A	India	1
González Arboleda, 2022 ^[⁷⁰^]	A	Chile	1	Zhang, 2021 ^[¹⁴⁵^]	A	China	1	Singh, 2019 ^[²²²^]	A	India	1
Hammade, 2022 ^[⁷¹^]	A	Syria	1	Zouaghi, 2021 ^[¹⁴⁶^]	A	Tunisia	1	Syllaios, 2019 ^[²²³^]	A	Greece	1
Hanalioglu, 2022 ^[⁷²^]	A	Turkey	1	Aboksari, 2020 ^[¹⁴⁷^]	A	Iran	1	Taşlıçay, 2019 ^[²²⁴^]	A	Turkey	1
Çeviker,2022 ^[⁷³^]	A	Turkey	1	Acharya, 2020 ^[¹⁴⁸^]	A	Nepal	1	Tonkaz, 2019 ^[²²⁵^]	A	Turkey	1
Huertas, 2022 ^[⁷⁴^]	A	Spain	1	Akhan, 2020 ^[¹⁴⁹^]	D	Turkey	38	Trawinski, 2019 ^[²²⁶^]	A	Germany	1
Ijaz, 2022 ^[⁷⁵^]	A	Pakistan	1	Akhtar, 2020 ^[¹⁵⁰^]	A	India	1	Wa, 2019 ^[²²⁷^]	A	China	1
Karahan, 2022 ^[⁷⁶^]	A	Turkey	1	Arora, 2020 ^[¹⁵¹^]	A	India	1	Wang, 2019 ^[²²⁸^]	A	China	1
Karami, 2022 ^[⁷⁷^]	A	Iran	1	Assimakopoulos, 2020 ^[¹⁵²^]	A	Greece	1	Xu, 2019 ^[²²⁹^]	A	China	1
Kartavya, 2022 ^[⁷⁸^]	A	India	1	Bakshi, 2020 ^[¹⁵³^]	A	India	1	Yacine, 2019 ^[²³⁰^]	A	Tunisia	1
Kumar, 2022 ^[⁷⁹^]	A	India	1	Destek, 2020 ^[¹⁵⁴^]	A	Turkey	1	Zhuoli, 2019 ^[²³¹^]	A	China	1
Li, 2022 ^[⁸⁰^]	C	China	53	Dkhissi, 2020 ^[¹⁵⁵^]	A	Morocco	1
Li, 2022 ^[⁸¹^]	A	China	1	Ewnte, 2020 ^[¹⁵⁶^]	A	Ethiopia	1
A: case report, B: case series, C: cohort, D: randomized control trial, E: cross-sectional study, US:* United States, UK: United Kingdom*

**Table 2.** The distribution of reported cases across countries
Country	Number of Cases	Percentage (%)
Spain	362	36.13%
China	270	26.95%
Turkey	128	12.77%
Greece	53	5.29%
India	44	4.39%
Tunisia	24	2.40%
Iran	18	1.80%
Italy	17	1.70%
Romania	8	0.80%
Morocco	6	0.60%
United States	6	0.60%
Brazil	4	0.40%
Israel	4	0.40%
Saudi Arabia	4	0.40%
Germany	3	0.30%
Pakistan	3	0.30%
South Africa	3	0.30%
South Korea	3	0.30%
Switzerland	3	0.30%
Albania	2	0.20%
Canada	2	0.20%
Ethiopia	2	0.20%
Jordan	2	0.20%
Macedonia	2	0.20%
Nepal	2	0.20%
Poland	2	0.20%
Serbia	2	0.20%
Tanzania	2	0.20%
Afghanistan	1	0.10%
Angola	1	0.10%
Argentina	1	0.10%
Bhutan	1	0.10%
Chile	1	0.10%
Colombia	1	0.10%
Croatia	1	0.10%
Ecuador	1	0.10%
France	1	0.10%
Ireland	1	0.10%
Lebanon	1	0.10%
Malaysia	1	0.10%
Netherlands	1	0.10%
Peru	1	0.10%
Portugal	1	0.10%
Qatar	1	0.10%
Syria	1	0.10%
Taiwan	1	0.10%
United Kingdom	1	0.10%
Uzbekistan	1	0.10%
Yemen	1	0.10%

**Table 3.** Distribution of cases across continents
Continent	Number of cases	Percentage (%)
Asia	487	48.60%
Europe	460	45.91%
Africa	38	3.79%
South America	9	0.90%
North America	8	0.80%

**Table 4.** Distribution of organs and body parts affected by hydatid cysts
Organ	Number of cases*	Percentage (%)
Liver	731	72.95%
Lung	110	10.98%
Kidney	43	4.29%
Heart	37	3.69%
Muscle	24	2.40%
Brain	21	2.10%
Abdominal cavity	16	1.60%
Bone	14	1.40%
Vertebral and spinal region	14	1.40%
Pelvic region	12	1.20%
Pancreas	9	0.90%
Spleen	8	0.80%
Mediastinum	5	0.50%
Breast	4	0.40%
Uterus	4	0.40%
Intra-orbital	3	0.30%
Thyroid gland	2	0.20%
Gallbladder	1	0.10%
Others	14	1.40%
* Some patients had hydatid cysts in more than one organ.

**Table 5.** Clinical and demographic characteristics of patients
Variables	Frequency (percentage)
Gender Male Female Non-Identified	505 (50.40%) 496 (49.50%) 1 (0.10%)



Age (year) 3 – 18 19 – 30 31 – 40 41 – 50 51 – 60 61 – 70 > 70 N/A Median (QR)	63 (6.29%) 52 (5.19%) 47 (4.69%) 34 (3.39) 18 (1.80%) 28 (2.79%) 20 (2.00%) 740 (73.85%) 35 (19–51)









Clinical presentations* Pain Fever Shortness of breath Cough Asymptomatic Others N/A	189 (18.86%) 66 (6.59%) 41 (4.09%) 27 (2.69%) 51 (5.09%) 42 (4.19%) 616 (61.48%)







Diagnostic findings
Hydatid serology Positive Negative N/A CT scan Ultrasound MRI X. ray Echocardiogram N/A	139 (13.87%) 31 (3.09%) 832 (83.03%) 806 (80.44%) 475 (47.40%) 168 (16.77%) 69 (6.88%) 23 (2.29%) 105 (10.48%)









Quantity of the cyst Single cyst Multiple cysts N/A	466 (46.51%) 203 (20.26%) 333 (33.23%)



Cyst status Patients with intact cysts Patients with ruptured cysts Patients with intact and ruptured cysts N/A	100 (9.98%) 97 (9.68%) 6 (0.60%) 799 (79.74%)




Type of treatment Surgical intervention Percutaneous treatment Medical treatment None N/A	639 (63.77%) 75 (7.49%) 31 (3.09%) 2 (0.20%) 255 (25.45%)





Medication Albendazole Albendazole & Praziquantel Mebendazole None N/A	275 (27.44%) 9 (0.90%) 1 (0.10%) 26 (2.59%) 691 (68.96%)





Follow up < 1 month – 6 months > 6 months – 1 year > 1 year – 2 years > 2years – 4 years > 4 years – 7 years > 7 years N/A	347 (34.63%) 25 (2.50%) 27 (2.69%) 13 (1.30%) 5 (0.50%) 2 (0.20%) 583 (58.18%)







Recurrence Yes No N/A	26 (2.59%) 566 (56.49%) 410 (40.92%)



Death Yes No N/A	17 (1.70%) 666 (66.47%) 319 (31.83%)



* Some patients had multiple clinical presentations, CT scan: computed tomography scan, MRI: magnetic resonance imaging, N/A: not available

Discussion

Conclusion

Declarations

Conflicts of interest: The author(s) have no conflicts of interest to disclose.

Ethical approval: Not applicable.

Consent for participation: Not applicable.

Consent for publication: Not applicable.

Funding: The present study received no financial support.

Acknowledgments: None to be declared.

Use of AI: AI was not used in the drafting of the manuscript, the production of graphical elements, or the collection and analysis of data.

Data availability statement: Not applicable.

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Abstract PDF