DOI: http://dx.doi.org/10.18203/issn.2454-5929.ijohns20204647

Sino-orbito-cerebral aspergillosis in an immunocompetent patient: a rare case report

Jitender Phogat, Manisha Nada, Monika Dahiya, Sneha Solanki

Abstract


Invasive sino-orbito-cerebral aspergillosis is rarely seen in immunocompetent individuals; diagnosis and management of which is still a challenge. We report a case of invasive sino-orbito-cerebral aspergillosis in a 38 year immunocompetent male presenting with mild protrusion of right eye which was associated with no other complaint. His visual acuity was 6/6 in both eyes. There was mild proptosis of right eye of 2 mm on Hertel exophthalmometer with no restriction of the ocular movements. MRI brain, orbit and paranasal sinuses revealed soft tissue swelling in right ethmoid, frontal and sphenoidal air cells and orbit with bony destruction of lamina papyracea with intracranial extension of the mass with destruction of lamina cribrosa. The tissue biopsy was taken from the uncinate process and middle turbinate and sent for histopathological examination which revealed a fibrocollagenous soft tissue which was densely infiltrated by inflammatory cells with presence of large number of foreign body granulomas and filamentous aspergillus fungal hyphae. By radiological and histopathological findings, patient was diagnosed as a case of rhino-orbito-cerebral aspergillosis and given Tb Voriconazole 200 mg BD for 6 months after neurosurgery consultation. His proptosis was revealed at 3 months and repeat MRI showed slightly thickened mucosa of sinuses with no mass lesion seen in the orbit and brain with no evidence of any residual fungal granuloma. Orbital aspergillosis is quite challenging in terms of both diagnosis and treatment. Prolonged antifungal therapy is very effective in controlling infection, if patient is compliant.


Keywords


Aspergillosis, Fungal infections, Proptosis, Voriconazole

Full Text:

PDF

References


Fuqua TH, Sittitavornwong S, Knoll M, Said-Al-Naif N. Primary invasive oral aspergillosis: An update literature review. J Oral Maxillofac Surg. 2010;68:2557-63.

Sivak-Callcott JA, Livesley N, Nugent RA, Rasmussen SL, Saeed P, Rootman J. Localised invasive sino-orbital aspergillosis: characteristic features. Br J Ophthalmol. 2004;88(5):681-7.

Kontoyiannis DP, Bodey GP. Invasive aspergillosis in 2002: An update. Eur J Clin Microbiol Infect Dis. 2002;21:161-72.

Neeli AS, Kotrashetti SM, Vallavan P. Sino-orbital aspergillosis: a case report and brief review of literature. World J Dent. 2012;3(4):363-6.

Myoken Y, Sugata T, Kyo T, Fujihara M, Kohara T, Katsu M, Tamura M, Mikami Y. Invasive Aspergillus stomatitis in patients with acute leukemia: report of 12 cases. Clin Infect Dis. 2001; 33(12):1975-80.

Chambers MS, Lyzak WA, Martin JW, Lyzak JS, Toth BB. Oral complications associated with aspergillosis in patients with a hematologic malignancy. Presentation and treatment. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1995;79 (5):559-63.

Shamim MS, Siddiqui AA, Enam SA, Shah AA, Jooma R, Anwar S. Cranio-cerebral aspergillosis in immunocompetent hosts: Surgical prospectives. Neurol India. 2007;55:275-81.

Kameswaram M, Al-Wadei A, Khurana P, Okafor BC. Rhino-cerebral aspergillosis. J Laryngol Otol. 1992;106:981-5.

Green WR, Font RL, Zimmerman LE. Aspergillosis of orbit: Report of 10 cases and review of literature. Arch Ophthalmol. 1969;82:302-13.

Pushkar N, Meel R, Kashyap S, Bajaj M, Sen S. Invasive aspergillosis of orbit in immunocompetent patients: treatment and outcome. Ophthalmology. 2011;118:1886-91.

Fuqua TH Jr, Sittitavornwong S, Knoll M, Said-Al-Naief N. Primary invasive oral aspergillosis: An updated literature review. J Oral Maxillofac Surg. 2010;68:2557-63.

Lin SJ, Schranz J, Teutsch SM. Aspergillosis case-fatality rate: systematic review of the literature. Clin Infect Dis. 2001;32:358-66.

Udaipurwala IH. Allergic fungal sinusitis: A perplexing clinical entity. Pak J Otolaryngol. 2008; 24:25-7.

Levin LA, Avery R, Shore JW, Woog JJ, Baker AS. The spectrum of orbital aspergillosis: a clinicopathological review. Surv Ophthalmol. 1996; 41(2):142-54.

Khanna M, Mannan R, Kaur J. A unique cytological approach in diagnosing a case of invasive aspergillosis masquerading as retro-orbital neoplasm. J Clin Diagnos Res. 2010;(4):2311-15.

Shamim MS, Siddiqui AS, Enam SA, Shah AA, Jooma R. Craniocerebral aspergillosis in immunocompetent hosts: Surgical perspectives. Neurol India. 2007;55:274-81.

Austin P, Dekker A, Kennerdell JS. Orbital aspergillosis: Report of a case diagnosed by fine needle aspiration biopsy. Acta Cytol. 1983;27:166-9.

Spoor TC, Hartel WC, Harding S, Kocher G. Aspergillosis presenting as a corticosteroid responsive optic neuropathy. J Clin Neuroophthalmol. 1982;2:103-7.

Massry GG, Hornblass A, Harrison W. Itraconazole in the treatment of orbital aspergillosis. Ophthalmology. 1996;103:1467-70.

Adulkar N, Radhakrishan S, Kim U. Invasive sino-orbital aspergillosis in immunocompetent patients: A clinico-pathological study. Eye. 2019;33:988-94.

Sivak-Calcott JA, Livesley N, Nugent RA, Rootman J. Localised invasive sino-orbital aspergillosis: characteristic features. Br J Ophthalmol. 2004;88: 681-7.

Dhiwakar M, Thakar A, Bahadur S. Invasive sino-orbital aspergillosis: Surgical decision and dilemmas. J Laryngol Otol. 2003;117:280-5.

Slavin ML. Primary aspergillosis of the orbital apex. Arch Ophthalmol. 1991;109:1502-3.