CASE REPORT   

Chirurgia 2018 December;31(6):264-7

DOI: 10.23736/S0394-9508.18.04805-2

Copyright © 2018 EDIZIONI MINERVA MEDICA

language: English

Subarachnoid hemorrhage from rupture of a rapidly formed aneurysm following relapse of fungal orbital apex syndrome

Yusuke S. HORI ¹ ✉, Takashi SAITO ², Yuki EBISUDANI ¹, Haruto YAMADA ², Yusuke AKAGI ³, Mizuho AOI ¹, Yoko SHINNO ⁴, Hisashi NARAI ⁵, Hidenori MARUNAKA ³, Toru FUKUHARA ¹

¹ Department of Neurological Surgery, National Hospital Organization Okayama Medical Center, Okayama, Japan; ² Department of Infectious Diseases, National Hospital Organization Okayama Medical Center, Okayama, Japan; ³ Department of Otolaryngology, National Hospital Organization Okayama Medical Center, Okayama, Japan; ⁴ Department of Pathology, National Hospital Organization Okayama Medical Center, Okayama, Japan; ⁵ Department of Neurology, National Hospital Organization Okayama Medical Center, Okayama, Japan

Orbital apex syndrome-associated subarachnoid hemorrhage is a rare condition, and several reports have described fungal infection as a causative agent. No reports have described aneurysmal formation and subarachnoid hemorrhage secondary to relapse of this disease in its late clinical course. A 79-year-old man with a history of diabetes was diagnosed with orbital apex syndrome and eventually underwent surgical removal of a mass in the ethmoid sinus. Pathological examination findings were consistent with fungal infection, and serum Aspergillus antigen was positive (Cryptococcus and Candida antigens were negative). Four months postoperatively, the patient complained of left hemiparesis and was admitted to another hospital for treatment of a right parietal lobe infarction; no aneurysmal formation was noted. On admission day 20, his left hemiparesis and level of consciousness worsened, and he was transported to our hospital the next day. Subarachnoid hemorrhage from rupture of a rapidly formed right internal carotid artery aneurysm was diagnosed. Magnetic resonance imaging (MRI) showed a mass lesion in the right orbital apex, suggesting relapse of the disease. He was managed conservatively with antifungals. However, his level of consciousness deteriorated on day 14 at our institution. Diffusion-weighted MRI showed bilateral high-intensity areas and narrowing of the cerebral artery branches, suggesting vasospasm. His respiratory status and consciousness level worsened on day 17, and he died on day 18. This case suggests that careful long-term follow-up is needed to check for aneurysm formation in patients with previously diagnosed orbital apex syndrome with fungal infection, especially immunocompromised patients.

KEY WORDS: Aneurysm - Mycoses - Subarachnoid hemorrhage