- Email: [email protected]
Hydatid cyst of thyroid gland, a rare case report with a literature review
Journal Pre-proof Hydatid cyst of thyroid gland, a rare case report with a literature review Abdwlwahid M. Salih, Zanyar Y. Abdulla, Dlawar A. Mohammed, Vanya I. Jwamer, Pshtiwan G. Ali, Ahmed G.Hamasaeed, Hawar H. Shkur, Jalal K. Omer, Rawezh Q. Salih, Shvan H. Mohammed, Aso S. Muhialdeen, Karzan Mohammed, M. Hassan, Fahmi H. Kakamad
PII:
S2210-2612(20)30093-6
DOI:
https://doi.org/10.1016/j.ijscr.2020.02.019
Reference:
IJSCR 4338
To appear in:
International Journal of Surgery Case Reports
Received Date:
29 November 2019
Revised Date:
30 January 2020
Accepted Date:
5 February 2020
Please cite this article as: Salih AM, Abdulla ZY, Mohammed DA, Jwamer VI, Ali PG, G.Hamasaeed A, Shkur HH, Omer JK, Salih RQ, Mohammed SH, Muhialdeen AS, Mohammed K, Hassan M, Kakamad FH, Hydatid cyst of thyroid gland, a rare case report with a literature review, International Journal of Surgery Case Reports (2020), doi: https://doi.org/10.1016/j.ijscr.2020.02.019
This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2020 Published by Elsevier.
Hydatid cyst of thyroid gland, a rare case report with a literature review
Abdwlwahid M.Salih1 , Zanyar Y.Abdulla2 , Dlawar A.Mohammed3 ,Vanya I.Jwamer4 , Pshtiwan G.Ali5 , Ahmed G.Hamasaeed5, Hawar H.Shkur6 , Jalal K.Omer7 , Rawezh Q.Salih8 , Shvan H.Mohammed8 , Aso S. Muhialdeen8, Karzan Mohammed8, M.Hassan9 , Fahmi H.Kakamad8,10
Corresponding author:
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Fahmi Hussein Kakamad
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1- Faculty of Medical Sciences/School of Medicine/Department of Surgery/ University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ 2- Sulaimaniy Polytechnical University Presidency/ Sulaimani/ Kurdistan/Iraq 3- shahid Dr. Kassem main health center.Directorate General of Health Germian ,Sulamani ,Kurdistan,Iraq 4- Faculty of Medical Sciences/School of Medicine / University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ 5- Faculty of Medical Sciences/School of Pharmacy/ University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ 6- Takya Main Health Center/Sub Directorate of health Chamchamal/ Kurdistan/IRAQ 7- BSc Biology/ Sulaimani/Kurdistan/IRAQ 8- Kscien Organization for Scientific Research/Hamdi Street/ Sulaimani/Kurdistan /IRAQ 9- Iraqi Board For Medical Specialties /Sulaimani Teaching Hospital/Sulaimani/Kurdistan/IRAQ 10- Faculty of Medical Sciences/School of Medicine/Department of Cardiothoracic and Vascular Surgery/ University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ
Doctor City, Building 11, Apartment 50 Sulaimani Iraq
Email: [email protected]
Jo
Tel: 009647717267454 Zip code: 00964
Highlights
Hydatid disease is an infestation by Echinococci species in the endemic areas. Liver and lungs are the main two affected organs. Hydatid disease of thyroid is an extreme rare variant of the condition
In this report, the details of thyroid hydatid disease occurring in a middle age female with brief literature review
Abstract Introduction: Although hydatid cysts can affect any organ in the body, reports regarding affection of thyroid gland are scanty in the literature. This report aims to present a case of thyroid hydatid disease with literature review.
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Case report: A 48 -year-old female presented with painless anterior neck mass of about 2 year duration. There was an ill-defined, central anterior neck mass, with a smooth surface and mobile with deglutition. Ultrasound of the thyroid gland revealed an enlarged left lobe of thyroid gland due to well defined thick wall cystic nodule. The patient underwent left thyroid lobectomy under general anesthesia, the pathology report revealed hydatid cyst of thyroid gland.
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Discussion: Liver and lungs are often the end destination for hydatid cysts, while other places like mediastinum, diaphragm, cardiac, smooth and skeletal muscles, abdominal and chest walls are rarely involved. In this case, the cyst affected even a rarer organ which was the left lobe of thyroid gland. Most of the time the disease is asymptomatic and is found accidentally, yet depending on the site and size of the cysts, symptoms can occur.
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Conclusion: Hydatid disease of thyroid gland is an extremely rare condition. The main presenting symptom is swelling. Operation under general anesthesia is the only modality of treatment.
ur na
Keywords: Echinococcus; multilocularis; granulosus; anaphylaxis,
Introduction
Jo
Hydatidosis as a zoonotic disease represents one of the oldest known diseases, dating back to Hippocrates era(1). It is caused by Echinococcus multilocularis and Echinococcus granulosus infestation (2) from the Taeniidae family in the Cestode class (3). Due to travelling, the disease is seen worldwide (1), however it is most endemic in areas with mild climates like Australia, Southeast Asia, New Zealand, Middle East, Mediterranean countries and South America (4). Up to two-third of the affected patients present with liver involvement, and up to 25% present with lung involvement, while other organs involvement like bone, kidney, muscle, pancreas, heart and brain constitute a small portion (5). However even in endemic areas head and neck hydatidosis is a rare finding (6). When mistook for
malignancy, needle aspiration can endanger the patient’s life (7). In line with SCARE guideline, we present a case of hydatid cyst in the thyroid gland, along with a brief literature review (8).
of
Patient Information: A 48 -year-old female patient presented with a painless anterior neck mass of about 2 year duration. The mass increased in size slowly and associated with mild shortness of breath. The patient was living in a village, working on a farm and she has a long history of animal contact such as sheep, goats and dogs. Past surgical history was positive for plastic operation for a burned face.
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Clinical Findings: On examination; there was an ill-defined, central anterior neck mass, with a smooth surface and mobile with deglutition. Investigations including complete blood counts, thyroid function tests, renal function tests, blood sugar and chest-X-ray were normal. Antithyroid peroxidase (anti-TPO) antibodies were negative. Ultrasound of the thyroid gland revealed an enlarged left lobe of thyroid gland ( 65x33x32 mm) due to a well-defined thick wall (reaching 4 mm) cystic nodule measuring about 40x32x32 mm, occupying the mid-low third of left lobe, with a mild peripheral vascularity and microcalcification.
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Therapeutic Intervention: The patient was prepared for general anesthesia (GA). Under GA, she underwent left thyroid lobectomy. The operation was completed within one hour. the pathology report revealed hydatid cyst of thyroid gland with pericystic adhesion (Figure 1).
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Follow-up and Outcomes: Postoperatively, the patient was sent for computed tomography scan (CT scan) of the head, neck, chest, abdomen and pelvis, the result was negative. The patient was remained in hospital for two days. The patient was put on anthelminthics for three months. Six months later, the patient was free from recurrence. Discussion
Jo
Hydatid disease is a parasitic infection seen all over the world, though mostly in regions like Eastern Europe, South Africa, Middle East, South America, Australia and Mediterranean region where cattle and sheep rearing is common (2). The current case has history of sheep and cattle contact. Liver and lungs are often the end destination for cysts, while other places like mediastinum, diaphragm, cardiac, smooth and skeletal muscles, abdominal and chest walls (9,10, 11, 12, 13, 14) are rarely involved. In this case, the cyst affected even a rarer organ which was the left lobe of thyroid gland. Just like this case, it is seen more frequently among middle-aged women(5). Most of the time, the disease is asymptomatic and is found accidentally, yet depending on the site and size of the cysts, symptoms can occur. Hewa et al received a 22-year-old with complete
paraplegia, later proven to be Hydatid disease of spine and recovered after operation (15). This patient presented with painless anterior neck mass of about 2 year duration.
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Echinococci species pass through three developmental stages in their life cycle, in the adult form they reside in their definitive host intestines and these are carnivores, most commonly dogs. Then their eggs will be excreted to the environment through feces and when ingested by intermediate hosts, which commonly include pig, camel, cattle, goats and sheep, they form metacestodes (10). Humans are not naturally included in their biological life cycle, instead may accidentally become an intermediate host when ingesting food contaminated by dog feces containing the echinococcosis eggs (16, 17). Once the eggs reach the gastrointestinal system, they will rupture and larvae emerge. These larvae penetrate the intestinal wall and reach the hepatic sinusoids through the portal system. Larvae that reach the lung are small enough to escape liver’s filtration system (3).
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Ultrasonography (USG), Computed Tomography scan (CT), Magnetic Resonance Imaging (MRI), Scintigraphy and fine-needle aspiration cytology (FNAC) are the standard methods for diagnosing Hydatid cyst (3). To this day worldwide, complete surgical excision with perioperative albendazole is most commonly practiced and regard as the standard. The results are outstanding with low recurrence rate.
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Two forms of thyroid gland hydatid cysts have been described (2,3). When the diagnosed hydatid disease is confined to the thyroid gland with no evidence of the disease elsewhere, this is referred to as primary form. The literature contains very few reports of the primary form of thyroid hydatid disease (table 1). Presence of other organ involvement like liver, lung and others labels the disease as secondary form. (3). This case is primary thyroid hydatid cyst due to not having the cyst before or elsewhere. When the size of the cyst increases, it may mimic thyroid carcinoma and adhere to surrounding structures like strap muscles, recurrent laryngeal nerve, trachea, esophagus and carotid sheath (2). In this case, the cyst was surrounded by normal thyroid tissue of the left lobe. Currently medical treatments are not effective, thus surgical excision still provides the best outcome. Calò et al resected hydatid cyst of the trapezius in an old age
male under local anesthesia [18]. Medical treatment with albendazole or mebendazole may still be tried in cases unfit for surgery, although adverse reactions and unpredictable results have been reported.
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When performing surgery, the aim is to remove the cyst completely while avoiding spillage of its content. Reported complication include organ damage, abscess and secondary cyst formation and most importantly anaphylaxis if the cyst ruptures. formation (19). The current case was free from complication. Conclusion
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Hydatid disease of thyroid gland is an extremely rare condition. The main presenting symptom is swelling. The diagnosis is suspected by ultrasound while it only can be confirmed after pathological examination of the specimen. Operation under general anesthesia is the only modality of treatment.
conflicts of interest
sources of funding No source to be stated.
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Ethical Approval
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here is no conflict to be declared.
Approval is not necessary for case report in our locality. Consent
Consent has been taken from the patient and the family of the patient.
Jo
Author contribution
Abdulwahid M. Salih: Surgeon performed the operation and follow up. Shvan H. Mohammed, Fahmi H. Kakamad, and Rawezh Q. Salih: Writing the manuscript and follow up. : literature review, final approval of the manuscript. Registration of Research Studies Not applicable
Guarantor Fahmi Hussein kakamad
Provenance and peer review
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Not commissioned, externally peer-reviewed
References
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1- Özdemir A, Korkmaz A, Darcin H, Aras N. Primary Hydatid Cyst of The Thyroid Gland and Other Organ Localizations of The Disease. Turkiye Klinikleri Journal of Case Reports. 1989;7(3):195-9. 2- Azendour I, Boulaich M, Ayoubi A, Oujilal A, Essakalli L, Kzadri M. Primary hydatid cyst of the thyroid gland. International Journal of Otolaryngology. 2011; 201(1):1-3 3- . Akbulut S, Demircan F, Sogutcu N. Hydatid cyst disease of the thyroid gland: report of two cases. International surgery. 2015 ;100(4):643-7. 4- Bartın MK, Yılmaz EM, Arslan H, Tekeli AE, Karataş S. A case of primary hydatid cyst in the thyroid gland. Turkish Journal of Surgery/Ulusal cerrahi dergisi. 2015;31(2):94. 5- Capoğlu I, Ünüvar N, Erdogan F, Yilmaz O, Caydere M. A hydatid cyst of the thyroid gland. Journal of international medical research. 2002 ;30(2):206-9. 6- Avcu S, Ünal Ö, Kotan Ç, Öztürk M, Özen Ö. Submandibular and thyroid gland involvement of hydatid cysts: a very rare association with percutaneous treatment. Diagnostic and Interventional Radiology. 2010;16(3):251. 7- Bastanhagh MH, Fatourechi V, Rajabian R. Hydatid cyst presenting as a thyroid nodule:Report of three cases. Acta Medica Iranica. 1995;33(1 & 2):31-4. 8- Agha RA, Borrelli MR, Farwana R, Koshy K, Fowler A, Orgill DP, For the SCARE Group. The SCARE 2018 Statement: Updating Consensus Surgical CAse REport (SCARE) Guidelines, International Journal of Surgery 2018;60:132-136
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9- Oraha AY, Faqe DA, Kadoura M, Kakamad FH, Yaldo FF, Aziz SQ. Cardiac Hydatid cysts; presentation and management. A case series. Annals of Medicine and Surgery. 2018;30(1):18-21. 10Salih AM, Kakamad FH, Salih RQ, Rahim HM, Habibullah IJ, Hassan HA et al. Hydatid cyst of the thigh: A case report with literature review. International journal of surgery case reports. 2018 ;51(1):8-10.
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11Salih AM, Kakamad FH, Hammood ZD, Yasin B, Ahmed DM. Abdominal wall Hydatid cyst: A review a literature with a case report. International journal of surgery case reports. 2017;37(1):154-6. 12Salih AM, Kakamad FH, Rauf GM. Isolated hydatid cyst of the diaphragm, a case report. International journal of surgery case reports. 2016;29(1):130-2. 13Baram A, Kakamad FH, Alwan AA. Primary posterior mediastinal hydatid cyst mimicking malignant mediastinal neurogenic tumor. International Journal of Case Report Images. 2014;5(1):54-7. 14Salih AM, Ahmed DM, Kakamad FH, Essa RA, Hunar AH, Ali HM. Primary chest wall Hydatid cyst: Review of literature with report of a new case. International journal of surgery case reports. 2017;41(1):404-6. 15Mustafa HM, Muhammad NH, Kakamad FH, Essa RA, Rahem HM. Hydatid disease causing paraplegia: a case report with literature review. Edorium Journal Infection Disease. 2017;3(1):17-20. 16Mahmood M, Yaldo FF, Kakamad FH, Abdulla K, Jaff A. Pediatric Cardiac hydatid cysts: A fatal manifestation of the disease. Journal of Cardiothoracic and Vascular Surgery. 2018;5(1):1-4. 17Salih AM, Kakamad FH, Mohammed SH, Baba HO, Mohammed KK, Ahmmad DR et al. Presentation and management of hydatid cyst of the thigh: A systematic review of literature. International Journal of Case Report Images. 2018;9(1):1-9. 18Calò PG, Tatti A, Tuveri M, Farris S, Nicolosi A. Hydatid cyst of trapezius muscle: an unusual localisation. Report of a case and review of the literature. Chir Ital. 2007;59(6):873-6. 19Sultana N, Shah W, Ullah S, Ali M, Basharat S. Primary Hydatid Cyst of the Thyroid Gland: A Case Report. Ann Clin Pathol. 2016;4(3):1070. 20Aköz G, Ekmekci S, Çakır E, Ünçel M, Özdemir T, Arıkan A, Şamlı A. Primary hydatid cyst of the thyroid gland; report of two cases. The Journal of Tepecik Education and Research Hospital. 2015;26(3):243-6. 21Erbil Y, Barbaros U, Baspinar I, Deveci U, Kapran Y, Bozbora A, Aral F, Özarmagan S. Hydatid cyst of the thyroid gland: two case reports. Infectious Diseases in Clinical Practice. 2005;13(6):318-20.
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22El Bousaadani A, El Mahdoufi R, Roubal M, Mahtar M. Thyroid hydatid cyst: An unusual site. European Annals of Otorhinolaryngology, Head and Neck Diseases .2016;133(1):75-6. 23Danilă R, Ionescu L, Livadariu R, Vulpoi C, Ciobanu D, Ungureanu MC. Primary hydatid cyst of the thyroid. Acta Endocrinologica (Buc). 2015;11(4):529. 24Jain SK, Jamdade PT, Muneshwar SS, Ambulgekar VK, Panzade SM. Hydatid cyst of thyroid: an unusual cause of stridor. Indian Journal of Otolaryngology and Head and Neck Surgery. 2005;57(1):80-1. 25Dey AK, Mandal PK, Dutta A, Pramanik S, Maji S, Chakraborti N, Dasgupta S. A case of hydatid cyst of thyroid gland. Asian Journal of Medical Sciences. 2014;5(2):143-5. 26Eshraghi M, Shahmoradi L, Ghoddoosi M, Sadati SJ. Diagnosis of Primary Hydatid Cyst of Thyroid Gland: A Case Report. Biomolecular concepts. 2019;10(1):106-10. 27Hoysal DR, Kulkarni S. Isolated primary hydatid disease of thyroid presenting as a solitary nodule: A rare. Indian Journal of Case Reports. 2019; 5(1): 79-81. 28Jiang T, Guo Q, Ran B, Zhang R, Aji T, Shao Y. Hydatid cyst of the thyroid gland with tracheal fistula: A case report and review of the literature. Experimental and Therapeutic Medicine. 2019;18(1):573-9. 29Oksuz S, Pektas E, Yavuz M, Aksungur N, Cayir Y, Akcay MN. An unusual cause of hoarseness: hydatid cyst of the thyroid. Trop Biomed. 2013;30(4):642-4. 30Rajabian R, Tavassoli M, Ggiassi T, Shslib I M. Hydatid cyst of the thyroid. Medical Journal of The Islamic Republic of Iran (MJIRI). 1991;5(3):165-6. 31Saha A, Paul UK, Kumar K. Diagnosis of primary hydatid cyst in thyroid by fine needle aspiration cytology. Journal of Cytology. 2007;24(3):137. 32Söğütlü G, Çıkım AS, Pişkin T, Dirican A, Mecit E, Kahraman L, Olmez A, Kırımlıoğlu V. Hydatid Cyst in Thyroid Gland: A Case Report. Inönü Üniversitesi Tip Fakültesi Dergisi.2007;14(3):185-7.
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33Amahzoune M, El Malki HO, Benkhraba K, Mohcine R, Ifrine L, Belkouchi A, Balafrej S. Recurred hydatid cyst of the thyroid: a case report. Ann Endocrinol (Paris). 2004 Oct;65(5):469-71. 34Lada P, Lermite E, Hennekinne-Mucci S, Etienne S, Pessaux P, Arnaud JP. Primary hydatid cyst of the thyroid, an unusual localisation of hidatidosis. Presse Med. 2005; 23:34(8):580.
Legends:
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Figure 1: intraoperative picture showing whitish cyst consistent with cyst’s germinal layers.
Table 1: brief review of the reported cases of thyroid hydatid disease. Presenting Symptoms
34 years
Female
growing tangible swelling in her neck (left side)
left lobectomy and isthmusectomy
2013
1
23 years
Male
hoarseness of voice and left lobe mass nodular goiter with moderate compression symptoms. swelling in front of her neck (left side) lump in her neck (Right), (Lift) and (Right) respectively neck lump Right side
subtotal thyroidectomy total thyroidectomy
Danilă et al/23
2015
1
26 years
Female
Hoysal et al/27
2019
1
14 years
Female
Bastanhagh et al/7
1995
3
16,24,60 Female
Dey et al/25
2014
1
30 years
Female
Jain et al/24 Azendour el/2
2005 2011
1 1
55 years 23 years
Female Female
Rajabian et al/30
1991
2
16,27
Jiang et al/28
2019
1
54 years
Söğütlü et al/32
2007
1
Yes¸im Erbil, MD/21
2005
2
ro
Oksuz et al/29
Female
lP
Male
18 years
Male
21,70 years
Male, Female
midline neck mass enlarging neck mass, Right side right thyroid nodule, right thyroid nodule gradual swelling on the left side of the lower part of the neck swelling of the thyroid gland left side swelling in the left lobe, right solitary thyroid nodule
2015
2
26,57 years
Female
neck swelling, hoarseness, and neck swelling
2007
1
30 years
Male
right sided thyroid swelling
Jo
Management
of
Sex
-p
2019
Saha et al/30
Age
re
Eshraghi et al/26
Number of cases 1
Akbulut et al/3
Year
ur na
Author/Reference
Ozdemir et al/1
1989
1
54 years
Female
nodular goiter
Avcu et al/6
2010
1
48 years
Male
right side neck lump
Sultana et al/19 Batrin et al/ 2
2016 2015
1 1
35 years 32 years
Female Female
Lada et al/34
2005
1
28-year
Male
swelling in neck palpable and growing mass in the neck right side left cervical tumefaction
left hemithyroidectomy Rt lobectomy, excision, excision Albendazole (conservative treatment) excision subtotal thyroidectomy excision, excision excision hemi-thyroidectomy with isthmusectomy left hemithyroidectomy, bilateral near-total thyroidectomy total thyroidectomy, total thyroidectomy patient refused surgery right total and left subtotal thyroidectomy Albendazole, and aspiration excision bilateral total thyroidectomy lobectomy with
2004
1
21-year
Male
cold thyroid nodule
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Amahzoune et al/33
isthmusectomy Resection
PII:
S2210-2612(20)30093-6
DOI:
https://doi.org/10.1016/j.ijscr.2020.02.019
Reference:
IJSCR 4338
To appear in:
International Journal of Surgery Case Reports
Received Date:
29 November 2019
Revised Date:
30 January 2020
Accepted Date:
5 February 2020
Please cite this article as: Salih AM, Abdulla ZY, Mohammed DA, Jwamer VI, Ali PG, G.Hamasaeed A, Shkur HH, Omer JK, Salih RQ, Mohammed SH, Muhialdeen AS, Mohammed K, Hassan M, Kakamad FH, Hydatid cyst of thyroid gland, a rare case report with a literature review, International Journal of Surgery Case Reports (2020), doi: https://doi.org/10.1016/j.ijscr.2020.02.019
This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2020 Published by Elsevier.
Hydatid cyst of thyroid gland, a rare case report with a literature review
Abdwlwahid M.Salih1 , Zanyar Y.Abdulla2 , Dlawar A.Mohammed3 ,Vanya I.Jwamer4 , Pshtiwan G.Ali5 , Ahmed G.Hamasaeed5, Hawar H.Shkur6 , Jalal K.Omer7 , Rawezh Q.Salih8 , Shvan H.Mohammed8 , Aso S. Muhialdeen8, Karzan Mohammed8, M.Hassan9 , Fahmi H.Kakamad8,10
Corresponding author:
ur na
Fahmi Hussein Kakamad
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1- Faculty of Medical Sciences/School of Medicine/Department of Surgery/ University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ 2- Sulaimaniy Polytechnical University Presidency/ Sulaimani/ Kurdistan/Iraq 3- shahid Dr. Kassem main health center.Directorate General of Health Germian ,Sulamani ,Kurdistan,Iraq 4- Faculty of Medical Sciences/School of Medicine / University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ 5- Faculty of Medical Sciences/School of Pharmacy/ University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ 6- Takya Main Health Center/Sub Directorate of health Chamchamal/ Kurdistan/IRAQ 7- BSc Biology/ Sulaimani/Kurdistan/IRAQ 8- Kscien Organization for Scientific Research/Hamdi Street/ Sulaimani/Kurdistan /IRAQ 9- Iraqi Board For Medical Specialties /Sulaimani Teaching Hospital/Sulaimani/Kurdistan/IRAQ 10- Faculty of Medical Sciences/School of Medicine/Department of Cardiothoracic and Vascular Surgery/ University of Sulaimani/ François Mitterrand Street/Sulaimani/Kurdistan/IRAQ
Doctor City, Building 11, Apartment 50 Sulaimani Iraq
Email: [email protected]
Jo
Tel: 009647717267454 Zip code: 00964
Highlights
Hydatid disease is an infestation by Echinococci species in the endemic areas. Liver and lungs are the main two affected organs. Hydatid disease of thyroid is an extreme rare variant of the condition
In this report, the details of thyroid hydatid disease occurring in a middle age female with brief literature review
Abstract Introduction: Although hydatid cysts can affect any organ in the body, reports regarding affection of thyroid gland are scanty in the literature. This report aims to present a case of thyroid hydatid disease with literature review.
ro
of
Case report: A 48 -year-old female presented with painless anterior neck mass of about 2 year duration. There was an ill-defined, central anterior neck mass, with a smooth surface and mobile with deglutition. Ultrasound of the thyroid gland revealed an enlarged left lobe of thyroid gland due to well defined thick wall cystic nodule. The patient underwent left thyroid lobectomy under general anesthesia, the pathology report revealed hydatid cyst of thyroid gland.
re
-p
Discussion: Liver and lungs are often the end destination for hydatid cysts, while other places like mediastinum, diaphragm, cardiac, smooth and skeletal muscles, abdominal and chest walls are rarely involved. In this case, the cyst affected even a rarer organ which was the left lobe of thyroid gland. Most of the time the disease is asymptomatic and is found accidentally, yet depending on the site and size of the cysts, symptoms can occur.
lP
Conclusion: Hydatid disease of thyroid gland is an extremely rare condition. The main presenting symptom is swelling. Operation under general anesthesia is the only modality of treatment.
ur na
Keywords: Echinococcus; multilocularis; granulosus; anaphylaxis,
Introduction
Jo
Hydatidosis as a zoonotic disease represents one of the oldest known diseases, dating back to Hippocrates era(1). It is caused by Echinococcus multilocularis and Echinococcus granulosus infestation (2) from the Taeniidae family in the Cestode class (3). Due to travelling, the disease is seen worldwide (1), however it is most endemic in areas with mild climates like Australia, Southeast Asia, New Zealand, Middle East, Mediterranean countries and South America (4). Up to two-third of the affected patients present with liver involvement, and up to 25% present with lung involvement, while other organs involvement like bone, kidney, muscle, pancreas, heart and brain constitute a small portion (5). However even in endemic areas head and neck hydatidosis is a rare finding (6). When mistook for
malignancy, needle aspiration can endanger the patient’s life (7). In line with SCARE guideline, we present a case of hydatid cyst in the thyroid gland, along with a brief literature review (8).
of
Patient Information: A 48 -year-old female patient presented with a painless anterior neck mass of about 2 year duration. The mass increased in size slowly and associated with mild shortness of breath. The patient was living in a village, working on a farm and she has a long history of animal contact such as sheep, goats and dogs. Past surgical history was positive for plastic operation for a burned face.
-p
ro
Clinical Findings: On examination; there was an ill-defined, central anterior neck mass, with a smooth surface and mobile with deglutition. Investigations including complete blood counts, thyroid function tests, renal function tests, blood sugar and chest-X-ray were normal. Antithyroid peroxidase (anti-TPO) antibodies were negative. Ultrasound of the thyroid gland revealed an enlarged left lobe of thyroid gland ( 65x33x32 mm) due to a well-defined thick wall (reaching 4 mm) cystic nodule measuring about 40x32x32 mm, occupying the mid-low third of left lobe, with a mild peripheral vascularity and microcalcification.
re
Therapeutic Intervention: The patient was prepared for general anesthesia (GA). Under GA, she underwent left thyroid lobectomy. The operation was completed within one hour. the pathology report revealed hydatid cyst of thyroid gland with pericystic adhesion (Figure 1).
ur na
lP
Follow-up and Outcomes: Postoperatively, the patient was sent for computed tomography scan (CT scan) of the head, neck, chest, abdomen and pelvis, the result was negative. The patient was remained in hospital for two days. The patient was put on anthelminthics for three months. Six months later, the patient was free from recurrence. Discussion
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Hydatid disease is a parasitic infection seen all over the world, though mostly in regions like Eastern Europe, South Africa, Middle East, South America, Australia and Mediterranean region where cattle and sheep rearing is common (2). The current case has history of sheep and cattle contact. Liver and lungs are often the end destination for cysts, while other places like mediastinum, diaphragm, cardiac, smooth and skeletal muscles, abdominal and chest walls (9,10, 11, 12, 13, 14) are rarely involved. In this case, the cyst affected even a rarer organ which was the left lobe of thyroid gland. Just like this case, it is seen more frequently among middle-aged women(5). Most of the time, the disease is asymptomatic and is found accidentally, yet depending on the site and size of the cysts, symptoms can occur. Hewa et al received a 22-year-old with complete
paraplegia, later proven to be Hydatid disease of spine and recovered after operation (15). This patient presented with painless anterior neck mass of about 2 year duration.
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Echinococci species pass through three developmental stages in their life cycle, in the adult form they reside in their definitive host intestines and these are carnivores, most commonly dogs. Then their eggs will be excreted to the environment through feces and when ingested by intermediate hosts, which commonly include pig, camel, cattle, goats and sheep, they form metacestodes (10). Humans are not naturally included in their biological life cycle, instead may accidentally become an intermediate host when ingesting food contaminated by dog feces containing the echinococcosis eggs (16, 17). Once the eggs reach the gastrointestinal system, they will rupture and larvae emerge. These larvae penetrate the intestinal wall and reach the hepatic sinusoids through the portal system. Larvae that reach the lung are small enough to escape liver’s filtration system (3).
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Ultrasonography (USG), Computed Tomography scan (CT), Magnetic Resonance Imaging (MRI), Scintigraphy and fine-needle aspiration cytology (FNAC) are the standard methods for diagnosing Hydatid cyst (3). To this day worldwide, complete surgical excision with perioperative albendazole is most commonly practiced and regard as the standard. The results are outstanding with low recurrence rate.
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Two forms of thyroid gland hydatid cysts have been described (2,3). When the diagnosed hydatid disease is confined to the thyroid gland with no evidence of the disease elsewhere, this is referred to as primary form. The literature contains very few reports of the primary form of thyroid hydatid disease (table 1). Presence of other organ involvement like liver, lung and others labels the disease as secondary form. (3). This case is primary thyroid hydatid cyst due to not having the cyst before or elsewhere. When the size of the cyst increases, it may mimic thyroid carcinoma and adhere to surrounding structures like strap muscles, recurrent laryngeal nerve, trachea, esophagus and carotid sheath (2). In this case, the cyst was surrounded by normal thyroid tissue of the left lobe. Currently medical treatments are not effective, thus surgical excision still provides the best outcome. Calò et al resected hydatid cyst of the trapezius in an old age
male under local anesthesia [18]. Medical treatment with albendazole or mebendazole may still be tried in cases unfit for surgery, although adverse reactions and unpredictable results have been reported.
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When performing surgery, the aim is to remove the cyst completely while avoiding spillage of its content. Reported complication include organ damage, abscess and secondary cyst formation and most importantly anaphylaxis if the cyst ruptures. formation (19). The current case was free from complication. Conclusion
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Hydatid disease of thyroid gland is an extremely rare condition. The main presenting symptom is swelling. The diagnosis is suspected by ultrasound while it only can be confirmed after pathological examination of the specimen. Operation under general anesthesia is the only modality of treatment.
conflicts of interest
sources of funding No source to be stated.
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Ethical Approval
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here is no conflict to be declared.
Approval is not necessary for case report in our locality. Consent
Consent has been taken from the patient and the family of the patient.
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Author contribution
Abdulwahid M. Salih: Surgeon performed the operation and follow up. Shvan H. Mohammed, Fahmi H. Kakamad, and Rawezh Q. Salih: Writing the manuscript and follow up. : literature review, final approval of the manuscript. Registration of Research Studies Not applicable
Guarantor Fahmi Hussein kakamad
Provenance and peer review
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Not commissioned, externally peer-reviewed
References
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11Salih AM, Kakamad FH, Hammood ZD, Yasin B, Ahmed DM. Abdominal wall Hydatid cyst: A review a literature with a case report. International journal of surgery case reports. 2017;37(1):154-6. 12Salih AM, Kakamad FH, Rauf GM. Isolated hydatid cyst of the diaphragm, a case report. International journal of surgery case reports. 2016;29(1):130-2. 13Baram A, Kakamad FH, Alwan AA. Primary posterior mediastinal hydatid cyst mimicking malignant mediastinal neurogenic tumor. International Journal of Case Report Images. 2014;5(1):54-7. 14Salih AM, Ahmed DM, Kakamad FH, Essa RA, Hunar AH, Ali HM. Primary chest wall Hydatid cyst: Review of literature with report of a new case. International journal of surgery case reports. 2017;41(1):404-6. 15Mustafa HM, Muhammad NH, Kakamad FH, Essa RA, Rahem HM. Hydatid disease causing paraplegia: a case report with literature review. Edorium Journal Infection Disease. 2017;3(1):17-20. 16Mahmood M, Yaldo FF, Kakamad FH, Abdulla K, Jaff A. Pediatric Cardiac hydatid cysts: A fatal manifestation of the disease. Journal of Cardiothoracic and Vascular Surgery. 2018;5(1):1-4. 17Salih AM, Kakamad FH, Mohammed SH, Baba HO, Mohammed KK, Ahmmad DR et al. Presentation and management of hydatid cyst of the thigh: A systematic review of literature. International Journal of Case Report Images. 2018;9(1):1-9. 18Calò PG, Tatti A, Tuveri M, Farris S, Nicolosi A. Hydatid cyst of trapezius muscle: an unusual localisation. Report of a case and review of the literature. Chir Ital. 2007;59(6):873-6. 19Sultana N, Shah W, Ullah S, Ali M, Basharat S. Primary Hydatid Cyst of the Thyroid Gland: A Case Report. Ann Clin Pathol. 2016;4(3):1070. 20Aköz G, Ekmekci S, Çakır E, Ünçel M, Özdemir T, Arıkan A, Şamlı A. Primary hydatid cyst of the thyroid gland; report of two cases. The Journal of Tepecik Education and Research Hospital. 2015;26(3):243-6. 21Erbil Y, Barbaros U, Baspinar I, Deveci U, Kapran Y, Bozbora A, Aral F, Özarmagan S. Hydatid cyst of the thyroid gland: two case reports. Infectious Diseases in Clinical Practice. 2005;13(6):318-20.
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22El Bousaadani A, El Mahdoufi R, Roubal M, Mahtar M. Thyroid hydatid cyst: An unusual site. European Annals of Otorhinolaryngology, Head and Neck Diseases .2016;133(1):75-6. 23Danilă R, Ionescu L, Livadariu R, Vulpoi C, Ciobanu D, Ungureanu MC. Primary hydatid cyst of the thyroid. Acta Endocrinologica (Buc). 2015;11(4):529. 24Jain SK, Jamdade PT, Muneshwar SS, Ambulgekar VK, Panzade SM. Hydatid cyst of thyroid: an unusual cause of stridor. Indian Journal of Otolaryngology and Head and Neck Surgery. 2005;57(1):80-1. 25Dey AK, Mandal PK, Dutta A, Pramanik S, Maji S, Chakraborti N, Dasgupta S. A case of hydatid cyst of thyroid gland. Asian Journal of Medical Sciences. 2014;5(2):143-5. 26Eshraghi M, Shahmoradi L, Ghoddoosi M, Sadati SJ. Diagnosis of Primary Hydatid Cyst of Thyroid Gland: A Case Report. Biomolecular concepts. 2019;10(1):106-10. 27Hoysal DR, Kulkarni S. Isolated primary hydatid disease of thyroid presenting as a solitary nodule: A rare. Indian Journal of Case Reports. 2019; 5(1): 79-81. 28Jiang T, Guo Q, Ran B, Zhang R, Aji T, Shao Y. Hydatid cyst of the thyroid gland with tracheal fistula: A case report and review of the literature. Experimental and Therapeutic Medicine. 2019;18(1):573-9. 29Oksuz S, Pektas E, Yavuz M, Aksungur N, Cayir Y, Akcay MN. An unusual cause of hoarseness: hydatid cyst of the thyroid. Trop Biomed. 2013;30(4):642-4. 30Rajabian R, Tavassoli M, Ggiassi T, Shslib I M. Hydatid cyst of the thyroid. Medical Journal of The Islamic Republic of Iran (MJIRI). 1991;5(3):165-6. 31Saha A, Paul UK, Kumar K. Diagnosis of primary hydatid cyst in thyroid by fine needle aspiration cytology. Journal of Cytology. 2007;24(3):137. 32Söğütlü G, Çıkım AS, Pişkin T, Dirican A, Mecit E, Kahraman L, Olmez A, Kırımlıoğlu V. Hydatid Cyst in Thyroid Gland: A Case Report. Inönü Üniversitesi Tip Fakültesi Dergisi.2007;14(3):185-7.
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33Amahzoune M, El Malki HO, Benkhraba K, Mohcine R, Ifrine L, Belkouchi A, Balafrej S. Recurred hydatid cyst of the thyroid: a case report. Ann Endocrinol (Paris). 2004 Oct;65(5):469-71. 34Lada P, Lermite E, Hennekinne-Mucci S, Etienne S, Pessaux P, Arnaud JP. Primary hydatid cyst of the thyroid, an unusual localisation of hidatidosis. Presse Med. 2005; 23:34(8):580.
Legends:
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Figure 1: intraoperative picture showing whitish cyst consistent with cyst’s germinal layers.
Table 1: brief review of the reported cases of thyroid hydatid disease. Presenting Symptoms
34 years
Female
growing tangible swelling in her neck (left side)
left lobectomy and isthmusectomy
2013
1
23 years
Male
hoarseness of voice and left lobe mass nodular goiter with moderate compression symptoms. swelling in front of her neck (left side) lump in her neck (Right), (Lift) and (Right) respectively neck lump Right side
subtotal thyroidectomy total thyroidectomy
Danilă et al/23
2015
1
26 years
Female
Hoysal et al/27
2019
1
14 years
Female
Bastanhagh et al/7
1995
3
16,24,60 Female
Dey et al/25
2014
1
30 years
Female
Jain et al/24 Azendour el/2
2005 2011
1 1
55 years 23 years
Female Female
Rajabian et al/30
1991
2
16,27
Jiang et al/28
2019
1
54 years
Söğütlü et al/32
2007
1
Yes¸im Erbil, MD/21
2005
2
ro
Oksuz et al/29
Female
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Male
18 years
Male
21,70 years
Male, Female
midline neck mass enlarging neck mass, Right side right thyroid nodule, right thyroid nodule gradual swelling on the left side of the lower part of the neck swelling of the thyroid gland left side swelling in the left lobe, right solitary thyroid nodule
2015
2
26,57 years
Female
neck swelling, hoarseness, and neck swelling
2007
1
30 years
Male
right sided thyroid swelling
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Management
of
Sex
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2019
Saha et al/30
Age
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Eshraghi et al/26
Number of cases 1
Akbulut et al/3
Year
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Author/Reference
Ozdemir et al/1
1989
1
54 years
Female
nodular goiter
Avcu et al/6
2010
1
48 years
Male
right side neck lump
Sultana et al/19 Batrin et al/ 2
2016 2015
1 1
35 years 32 years
Female Female
Lada et al/34
2005
1
28-year
Male
swelling in neck palpable and growing mass in the neck right side left cervical tumefaction
left hemithyroidectomy Rt lobectomy, excision, excision Albendazole (conservative treatment) excision subtotal thyroidectomy excision, excision excision hemi-thyroidectomy with isthmusectomy left hemithyroidectomy, bilateral near-total thyroidectomy total thyroidectomy, total thyroidectomy patient refused surgery right total and left subtotal thyroidectomy Albendazole, and aspiration excision bilateral total thyroidectomy lobectomy with
2004
1
21-year
Male
cold thyroid nodule
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Amahzoune et al/33
isthmusectomy Resection