Egyptian Journal of Ear, Nose, Throat and Allied Sciences (2016) 17, 103–105 H O S T E D BY Egyptian Society of Ear, Nose, Throat and Allied Sciences Egyptian Journal of Ear, Nose, Throat and Allied Sciences www.ejentas.com CASE REPORT Hydatid cyst of the neck. A case report and literature review Reem Khalifa, Firas Nasser, Ahmed Elsetouhy, Ismail Farag * Hamad Medical Corporation, Qatar Received 1 December 2015; accepted 28 January 2016 Available online 17 June 2016 KEYWORDS Hydatid disease; Neck; Surgery Abstract Echinococcosis or hydatid disease is a zoonotic infection caused by Echinococcus granulosus. It has been recognized by humans for centuries, as it was described by Hippocrates more than two thousand years as a ‘‘fluid-filled liver”, and the famous Arabian physician Al-Rahzes described it, but it took till the 17th century when Francesco Redi illustrated that the hydatid cysts of echinococcosis were of animal origin. Hydatid disease is endemic in the Middle East, India, Africa, South America, New Zealand, Australia, Turkey and Southern Europe keeping in mind that it can occur also in non-endemic countries because of the upsurge of emigration and trade. The primary hosts are dogs. Intermediate hosts are sheep, cattle, horses and occasionally man. Hydatid cyst develops most frequently in the liver (65%), the lungs (25%), and the remaining 10% occurs in muscle, spleen, bones, kidneys, brain, eye, heart, and pancreas. Multiorgan involvement is seen in 20–30% of the cases with involvement of the liver in all cases. Occurrence of hydatid cyst is extremely rare in the head and neck region even in geographical areas where echinococcal infestation is frequent. Only a few cases of hydatid cyst located in head and neck have been reported in the literature. We will present our experience in treating a case of hydatid cyst located in the neck area, which is considered one of the few cases published due to the relative rarity of the disease in the fore mentioned anatomical location. Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. All rights reserved. This is an open access article under the CC BY-NC-ND license (http:// creativecommons.org/licenses/by-nc-nd/4.0/). 1. Introduction Echinococcosis or hydatid disease is a zoonotic infection caused by Echinococcus granulosus.1 It has been recognized * Corresponding author at: Hamad Medical Corporation, P.O. BOX 3050, Qatar. Tel.: +974 55063872. E-mail addresses: [email protected] (R. Khalifa), fnasser@ hamad.qa (F. Nasser), [email protected] (A. Elsetouhy), ifarag@ hamad.qa (I. Farag). Peer review under responsibility of Egyptian Society of Ear, Nose, Throat and Allied Sciences. by humans for centuries, as it was described by Hippocrates more than two thousand years as a ‘‘fluid-filled liver”, and the famous Arabian physician Al-Rahzes described it, but it took till the 17th century when Francesco Redi illustrated that the hydatid cysts of echinococcosis were of animal origin.2 Hydatid disease is endemic in the Middle East, India, Africa, South America, New Zealand, Australia, Turkey and Southern Europe3,4 keeping in mind that it can occur also in nonendemic countries because of the upsurge of emigration and trade.5 The primary hosts are dogs. Intermediate hosts are sheep, cattle, horses and occasionally man.1 http://dx.doi.org/10.1016/j.ejenta.2016.01.002 2090-0740 Ó 2016 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). 104 R. Khalifa et al. Hydatid cyst develops most frequently in the liver (65%), the lungs (25%), and the remaining 10% occurs in muscle, spleen, bones, kidneys, brain, eye, heart, and pancreas.6,7 Multiorgan involvement is seen in 20–30% of the cases with involvement of the liver in all cases.8 Occurrence of hydatid cyst is extremely rare in the head and neck region even in geographical areas where echinococcal infestation is frequent. Only a few cases of hydatid cyst located in head and neck have been reported in the literature.9,10 We will present our experience in treating a case of hydatid cyst located in the neck area, which is considered one of the few cases published due to the relative rarity of the disease in the fore mentioned anatomical location. 2. Case report A 20 year old Nepalese male, who is not known to have any medical history, presented to the Oral and maxillofacial Surgery department in Hamad Medical Corporation, Doha, State of Qatar, with a chief complaint of a right neck swelling, noted about 6 month back. The general condition of the Figure 1 3D demonstration of the right upper neck mass. Figure 3 Coronal post contrast CT images of the neck showing the cystic lesion deep to the right angle of the mandible displacing the right submandibular gland superomedially. patient was good; he had no history of fever or weight loss. The patient lived almost his entire life in his home country Nepal, before recently moving to Doha. He reported that the swelling was gradually increasing in size but never caused pain or discharge. Clinical examination revealed a well-defined, non-tender right upper neck swelling of about 5 cm in diameter, systemic examination was unremarkable, Fig 1. Computed Tomography Scan showed a well-defined large cystic lesion involving the right upper neck area, of about 3.3–4.5 cm in size and the report favored an infected branchial cyst, laboratory investigations were within normal values, Figs. 2 and 3 shows axial and coronal views (respectively) of the cyst, excisional biopsy was planned and discussed with the patient. The surgical procedure was successfully executed and the postoperative course was uneventful. Other body parts were scanned, and a calcified hydatid cyst was found in the abdomen, for which he was referred to the infectious disease team for further management. 3. Discussion Figure 2 Axial Post contrast CT neck showing a well-defined cystic lesion medial to the right angle of mandible showing enhancing wall of uniform thickness. Hydatid cyst is endemic in the Middle East, India, Africa, South America, New Zealand, Australia.4 Though the State of Qatar is not among the endemic countries for the disease, nevertheless, this patient, in addition to some other cases in other anatomical locations, were reported, and the reason for that is the high emigration rate within the last 3 decades due to the fast expansion in the country. Al-Ani et al reported a Case of Echinococcosis in a pregnant lady with an unusual presentation. The disease occurred in the lungs and liver and was treated surgically following delivery. According to same study echinococcal disease is seen mainly in people coming from endemic areas and the disease is rare among Qataris.11 Occurrence of hydatid cyst is extremely rare in the head and neck region even in geographical areas where echinococcal infestation is frequent, and only few cases have been reported in the literature.6,7 Hydatid cysts are usually not considered in Hydatid cyst of the neck the differential diagnosis of head and neck cystic swellings, especially in non-endemic areas in the absence of hydatid disease elsewhere in the body. The rarity of the disease in this anatomical location presents a diagnostic difficulty for the physician if he or she is not familiar with the disease.9 If the cysts ruptured while in the body, whether during surgical extraction or by some kind of trauma to the body, the patient would most likely go into anaphylactic shock.12 This event didn’t occur in the patient included in the study. Standard treatment modality is surgical removal of the cysts combined with chemotherapy using albendazole and/or mebendazole before and after surgery, albendazole is preferred twice a day for 1–5 months.13 4. Conclusion When dealing with well-defined cystic lesions in the head and neck area hydatid cyst should be considered in the differential diagnosis even in non-endemic areas due to the high emigration rates. Care should be taken by the operating team during surgery as ruptured cyst may cause anaphylactic shock. This case report is granted for publication with reference: MRC/1367/2015. Conflict of interest None declared. References 1. Akal M, Kara M. Primary hydatid cyst of the posterior cervical triangle. J Laryngol Otol. 2002;116:153–155. 105 2. Jump up Connolly, Stephanie. Echinococcosis. 2006. Web. 5 February 2010. <http://www.stanford.edu/group/parasites/ParaSites2006/Echinococcus/index.html>. 3. Jastaniah S, Malatani TS, Abu-Eshy S, et al. Hydatid cyst disease (Echinococcus granulosus): experience at Asir Central Hospital. Saudi J Gastroenterol. 1997;3:140–143. 4. Abu-Eshy S, Elamin Ali M. Hydatid cyst associated with pregnancy: a case report and review of the literature. Ann Saudi Med. 1999;19:130–131. 5. Kehila M, Allegue M, Abdessalem M, Letaief R, Said R, Khalfallah A. Hydatic cyst of the psoas: one case. J Radiol. 1987;68: 265–268. 6. Aleksic-Shihabi A, Vidolin EP. Cystic echinococcosis of the heart and brain: a case report. Acta Med Okayama. 2008;6:341–344. 7. Katilmis H, Ozturkcan S, Ozdemir I, Adadan Guvenc I, Ozturan S. Primary hydatid cyst of the neck. Am J Otolaryngol. 2007;28:205–207. http://dx.doi.org/10.1016/j.amjoto.2006.08.007. 8. Aletras H, Symbas N. Hydatid disease of the lung. In: Shields TW, LoCicero J, Ponn RB, eds. General Thoracic Surgery. Philadelphia: Lippincott Williams and Wilkins; 2000:1113–1122. 9. Cangiotti L, Muiesan P, Begni A, et al. Unusual localizations of hydatid disease: a 18 year experience. G Chir. 1994;15:83–86. 10. Pandolfo I, Blandino G, Scribano E, Longo M, Certo A, Chirico G. CT findings in hepatic involvement by Echinococcus granulosus. J Comput Assist Tomogr. 1984;8(5):839–845. 11. Al Ani A, Elzouki A, Mazhar R, An imported case of Echinococcosis in a pregnant lady with unusual presentation. Department of Medicine, Hamad General Hospital, Hamad Medical Corporation, Doha, Qatar Case, Reports in Infectious Diseases, vol. 2013 (2013), Article ID 753848, p. 4. 12. Bitton M et al. Anaphylactic shock after traumatic rupture of a splenic echinococcal cyst. Harefuah. 1992;122(4):226–228. 13. Eckert J, Deplazes P. Biological, epidemiological, and clinical aspects of echinococcosis, a zoonosis of increasing concern. Clin Microbiol Rev. 2004;17(1):107–135. http://dx.doi.org/10.1128/ cmr.17.1.107-135.2004, PMC 321468. PMID 14726458.
