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Journal of Medical Case Reports
Published in: Journal of Medical Case Reports 1/2018

Open Access 01-12-2018 | Case report

Hydatidosis of infratemporal fossa with proptosis – an unusual presentation: a case report and review of the literature

Authors: Sushma Thapa, Arnab Ghosh, Dilasma Ghartimagar, Supriya Shrestha, Subita Lalchan, O. P. Talwar

Published in: Journal of Medical Case Reports | Issue 1/2018

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Abstract

Background

Hydatid disease is one of the common zoonotic diseases caused by the larval stage of Echinococcus granulosus. It is endemic in sheep-raising and cattle-raising areas worldwide and humans are an accidental intermediate host following the ingestion of the larvae. Head and neck involvement of echinococcosis is a rare entity and involvement of the infratemporal region is extremely rare even in endemic areas. Only a few cases of hydatid cysts located in the infratemporal fossa have been reported in the literature. Moreover, extension of the hydatid cyst into the intraorbital region and infiltrating into the surrounding orbital bone is even rarer.

Case presentation

We present a case of a 65-year-old Gurung Nepalese woman with painless proptosis of her left eyeball of 2 months’ duration with recent progressive diminution of vision for 15 days. Radiological findings showed a cystic mass in the left infratemporal fossa extending into the left orbit and involving the surrounding orbital bone. Surgical removal was carried out. On histopathological evaluation, it was reported as hydatid cyst infiltrating into the bone. She was prescribed albendazole and discharged after surgery. However, she was lost to follow up and returned after 15 months with recurrence and proptosis of the same eye. Repeat excision of the lesion was carried out and postoperatively she was administered tablet albendazole. She was found to be disease free after 6 months of follow up.

Conclusions

Clinical and radiological findings are important but may not be sufficient in the preoperative diagnosis of hydatid disease especially if rare sites are involved. Proptosis may be seen in several conditions and orbital or infratemporal hydatidosis, although rare, should be considered a differential diagnosis.
Literature
1.
Ghartimagar D, Ghosh A, Shrestha MK, Talwar OP, Sathian B. A 14 years hospital based study on clinical and morphological Spectrum of Hydatid disease. J Nepal Med Assoc. 2013;52(190):205–12.
2.
3.
Mandell GL, Douglas RG, Bennett JE, Dolin R. Mandell, Douglas and Bennett’s principles and practice of infectious disease. 5th ed. Philadelphia: Churchill Livingstone; 2000. p. 2956–65.
4.
Beşkonakli S, Çayli Y, Yalçinlar E. Primary intracranial extradural hydatid cyst extending above and below the tentorium. Br J Neurosurg. 1996;10(3):315–6.CrossRef
5.
Abbasioun K, Amirjamshidi A, Sabouri Deylamie M. Hydatid disease of the central nervous system: imaging characteristics and general features. Iran J Radiol. 2003;1(3–4):125–31.
6.
Nouroallahian M, Bakhshaee M, Afzalzdeh MR, Memar B. A hydatid cyst in an unusual location – the infratemporal fossa. Laryngoscope. 2013;123(2):407–9.CrossRef
7.
Kak VK, Mathuriya SN. Hydatid disease of the nervous system. In: Chopra JS, Sawhney BI, editors. Neurology in tropics. London: Churchill Livingstone; 1999. p. 244–58.
8.
Gabriele F, Bortoletti G, Conchedda M, Palmas C, Ecca AR. Epidemiology of hydatid disease in the Mediterranean basin with special reference to Italy. Parassitologia. 1997;39(1):47–52.PubMed
9.
Anandpara KM, Aswani Y, Hira P, Sathe PA. Isolated primary orbital hydatid disease presenting as multiple cystic lesions: a rare cause of proptosis. Ann Parasitol. 2015;61(3):193–5.PubMed
10.
11.
Kahveci R, Sanli AM, Gurer B, Sekerci Z. Orbital hydatid cyst: case report. J Neurosurg Pediatr. 2012;9:42–4.CrossRef
12.
Metanat M, Sharifi-mood B, Sandoghi M, Alavi-Naini R. Osseous Hydatid disease. Iran J Parasitol. 2008;3:60–4.
13.
Ciurea AV, Giuseppe G, Machinis TG, Coman TC, Fountas KN. Orbital hydatid cyst in childhood: a report of two cases. South Med J. 2006;99(6):620–4.CrossRef
14.
Sahin B, Comoglu S, Polat B, Deger K. Hydatid cyst in unusual location: Pterygopalatine fossa – infratemporal fossa. Auris Nasus Larynx. 2016;43(4):464–7.CrossRef
15.
Baglam T, Karatas E, Durucu C, Sirikci A, Kara F, Kanlikama M. Primary hydatid cyst of the infratemporal fossa. J Craniofac Surg. 2009;20:1200–1.CrossRef
16.
Pasaoglu E, Damgaci L, Tokoglu F, Yildirim N, Alp AO, Yuksel E. CT findings of hydatid cyst with unusual location; infratemporal fossa. Eur Radiol. 1998;8:1570–2.CrossRef
17.
Sennaroglu L, Onerci M, Turan E, Sungur A. Infratemporal hydatid cyst – unusual location of echinococcosis. J Laryngeal Otol. 1994;108:601–3.
18.
Tierney LM Jr, Mc Phee SJ, Papaddakis MA. Current medical diagnosis and treatment. 38th ed. London: Appleton and Lange; 1999. p. 1396–8.
19.
Katilmis H, Ozturkcan S, Ozdemir I, Guvenc IA, Ozturan S. Primary hydatid cyst of the neck. Am J Otolaryngol. 2007;28:205–7.CrossRef
20.
Akal M, Kera M. Primary hydatid cyst of the posterior cervical triangle. J Laryngol Otol. 2002;116:153–5.CrossRef
21.
EI Kohen A, Benjelloun A, EI Quessar A, Derraz S, Lazrak A, Jazouli N, et al. Multiple hydatid cysts of the neck, the nasopharynx and the skull base revealing cervical vertebral hydatid disease. Int J Pediatr Otorhinolaryngol. 2003;67:655–62.CrossRef
22.
Ataoglu H, Uckan S, Gulsun OZ, Altinors N. Maxillofacial hydatid cyst. J Oral Maxillofac Surg. 2002;60:454–6.CrossRef
23.
Gangopadhyay K, Abuzeid MO, Kfoury H. Hydatid cyst of the pterygopalatine-infratemporal fossa. J Laryngol Otol. 1996;110:978–80.PubMed
24.
Thatte S, Thatte S. Ocular Hydatid cyst. Ann Clin Pathol. 2016;4(5):1081.
25.
Morales AG, Croxatto JO, Crovetto L, Ebner R. Hydatid cyst of the orbit. A review of 35 cases. Ophthalmology. 1988;95:1027–32.CrossRef
26.
Umesh K, Sulabha A, Sameer A, Neelakant M, Sangamesh NC, Ali R. Hydatid cyst of infratemporal region-a rare case report. Al Ameen J Med Sci. 2010;3(1):94–8.
27.
Taori K, Disawal A, Rathod J, Hatgaonkar A, Dhakate S, Bakare V, et al. Role of MRI imaging and MR spectroscopy in the diagnosis of ocular Hydatid cyst: case report. Open J Med Imaging. 2013;3:7–11.CrossRef
28.
Iynen I, Sogutb O, Guldurc ME, Kosed R, Kayab H, Bozkusa F. Primary hydatid cyst: an unusual cause of a mass in the supraclavicular region of the neck. J Clin Med Res. 2011;3(1):52–4.PubMedPubMedCentral
Metadata
Title
Hydatidosis of infratemporal fossa with proptosis – an unusual presentation: a case report and review of the literature
Authors
Sushma Thapa
Arnab Ghosh
Dilasma Ghartimagar
Supriya Shrestha
Subita Lalchan
O. P. Talwar
Publication date
01-12-2018
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2018
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/s13256-018-1812-y

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