- Division of Neurosurgery, Department of Surgery, Kaohsiung Veterans General Hospital, Kaohsiung, Taiwan, R.O.C
Correspondence Address:
Chi-Man Yip
Division of Neurosurgery, Department of Surgery, Kaohsiung Veterans General Hospital, Kaohsiung, Taiwan, R.O.C
DOI:10.4103/2152-7806.102349
Copyright: © 2012 Yip C. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.How to cite this article: Yip C, Hsu S, Liao W, Chen J, Liu S, Chen C. Orbital apex syndrome due to aspergillosis with subsequent fatal subarachnoid hemorrhage. Surg Neurol Int 13-Oct-2012;3:124
How to cite this URL: Yip C, Hsu S, Liao W, Chen J, Liu S, Chen C. Orbital apex syndrome due to aspergillosis with subsequent fatal subarachnoid hemorrhage. Surg Neurol Int 13-Oct-2012;3:124. Available from: http://sni.wpengine.com/surgicalint_articles/orbital-apex-syndrome-due-to-aspergillosis-with-subsequent-fatal-subarachnoid-hemorrhage/
Abstract
Background:Orbital apex syndrome has been described previously as a syndrome involving damage to the oculomotor nerve (III), trochlear nerve (IV), abducens nerve (VI), and ophthalmic branch of the trigeminal nerve (V1), in association with optic nerve dysfunction. It may be caused by inflammatory, infectious, neoplastic, iatrogenic, or vascular processes.
Case Description:A 73-year-old female having hypertension and rheumatoid arthritis stage 4 under long-term corticosteroid therapy presented to us with the right side orbital apex syndrome. Her magnetic resonance imaging (MRI) of orbit showed progression of a lesion at the right orbital apex and adjacent right superior orbital fissure with mild extension to the right posterior ethmoid sinus. She underwent endoscopic endonasal transethmoid approach with the removal of the lesion. The pathology showed a picture of fungal infection and the culture of the specimen proved Aspergillus fumigatus. Her postoperative course was smooth until 5 days after surgery, when she suffered a massive spontaneous subarachnoid hemorrhage resulting from a ruptured aneurysm, which was proven by computed tomography angiography (CTA) of brain. Unfortunately, she expired due to central failure.
Conclusion:In cases of immunocompromised patients having orbital apex syndrome, fungal infection should be kept in mind. One of the most lethal but rare sequels of CNS fungal infection is intracranial aneurysms. Early diagnosis and radical resection, combined with antifungal medications is the key to save this particular group of patients.
Keywords: Aspergillosis, fungal aneurysm, orbital apex syndrome
INTRODUCTION
Orbital aspergillosis is usually seen in immunocompromised individuals. The paranasal sinuses are the usual portal of entry for the pathogen, with orbital extension when host defenses are impaired leading to orbital apex syndrome which is a paralysis of all three nerves supplying the external ocular muscles and a sensory deficit in the distribution of the first division of the trigeminal nerve, combined with an optic nerve lesion.[
CASE REPORT
A 73-year-old woman with past medical history of hypertension and rheumatoid arthritis stage 4 under regular anti-hypertension medication and long-term corticosteroid therapy, presented to us in August 2010 with the chief complaint of decline of visual acuity of her right eye and right periorbital pain for 2 months. At the beginning of the clinical course, she had brain computed tomography (CT) scan and orbit magnetic resonance imaging (MRI) done in June 2010, which disclosed a small enhancing lesion, about 1.2 cm × 1.1 cm × 1 cm in size, near the right side orbital apex and adjacent right side superior orbital fissure with mild encasement of the right optic nerve, and this lesion showed mild extension to the adjacent right side posterior ethmoid sinus [Figure
Figure 1
Orbit MRI T1-weighted post-Gadolinium enhancement. (a, axial view) A small enhancing lesion about 1.2 cm × 1.1 cm × 1 cm noted near the right side orbital apex and adjacent right side superior orbital fissure with mild extension to the adjacent right side posterior ethmoid sinus region. (b, coronal view) This lesion had mild encasement of the right optic nerve
Figure 3
(a) Non-contrast brain CT demonstrated diffuse high-density acute subarachnoid hemorrhage in the basal cistern, pre-pontine cistern, ambient cistern, quadrigeminal cistern, cerebellomedullary cistern, and right sylvian fissure. (b) computed tomography angiography showed the presence of several bleb-like wide base aneurysms over right supraclinoid internal carotid artery. One aneurysm of about 4 mm showed extravasation of contrast medium. The dome of the ruptured aneurysm projected medially and superiorly
DISCUSSION
The orbital apex syndrome can be caused by tumors, trauma, aneurysm of internal carotid artery, inflammatory disorders, and infectious diseases, and aspergillosis is one of the causative pathologies.[
References
1. Ahsan H, Ajmal F, Saleem MF, Sonawala AB. Cerebral fungal infection with mycotic aneurysm of basilar artery and subarachnoid haemorrhage. Singapore Med J. 2009. 50: 22-5
2. Ahuja GK, Jain N, Vijayaraghavan M, Roy S. Cerebral mycotic aneurysm of fungal origin Case report. J Neurosurg. 1978. 49: 107-10
3. Fernandes YB, Ramina R, Borges G, Queiroz LS, Maldaun MVC, Maciel JA. Orbital Apex Syndrome due to Aspergillosis. Arq Neuropsiquiatr. 2001. 59: 806-8
4. Iihara K, Makita Y, Nabeshima S, Tei T, Keyaki A, Nioka H. Aspergillosis of the Central Nervous System causing Subarachnoid Hemorrhage from Mycotic Aneurysm of the Basilar Artery- Case Report. Neurol Med Chir (Tokyo). 1990. 30: 618-23
5. Ishikawa T, Kazumata K, Ni-iya Y, Kamiyama H, Andoh M. Subarachnoid hemorrhage as a result of fungal aneurysm at the posterior communicating artery associated with occlusion of the internal carotid artery Case Report. Surg Neurol. 2002. 58: 261-5
6. Komatsu Y, Narushima K, Kobayashi E, Tomono Y, Nose T. Aspergillus Mycotic Aneurysm – Case Report. Neuro Med Chir (Tokyo). 1991. 31: 346-50
7. Levin LA, Avery R, Shore JW, Woog JJ, Baker AS. The Spectrum of Orbital Aspergillosis: A Clinicopathological Review. Surv Ophthalmol. 1996. 41: 142-54
8. Masago A, Fukuoka H, Yoshida T, Majima K, Tada T, Nagai H. Intracranial Mycotic Aneurysm caused by Aspergillus – Case Report. Neurol Med Chir (Tokyo). 1992. 32: 904-7
9. O’Toole L’, Acheson JA, Kidd D. Orbital apex lesion due to Aspergillosis presenting in immunocompetent patients without apparent sinus disease. J Neurol. 2008. 255: 1798-801
10. Wilson WR, Hawrych A, Olan W. Rapid Development of Bilateral Internal Carotid Artery Aneurysm from Sphenoid Sinus Aspergillosis. Skull Base Surg. 1998. 8: 211-4
11. Yeh S, Foroozan R. Orbital apex syndrome. Curr Opin Ophthalmol. 2004. 15: 490-8