| Summary: | Background: Infection of Lomentospora (Scedosporium) spp. is very rare but high mortality. Above all, a fungal aneurysm due to Lomentospora spp. is extremely rare. The prognosis of ruptured fungal aneurysm due to Lomentospora spp. is remarkably poor in spite of proper surgical treatment and chemotherapy. Case Description: A 70-year-old female who was having chemotherapy for acute myeloid leukemia suffered from a sudden onset of severe consciousness disturbance. A brain computed tomography (CT) scan revealed large hematoma at the right parietal lobe adjacent to subdural hematoma with a slight subarachnoid hemorrhage. Three dimensional-CT angiography showed an intracranial aneurysm at the right peripheral portion of the middle cerebral artery. Surgical clipping and resection of the aneurysm were successfully performed on the same day. Lomentospora prolificans was identified on the blood culture, and pathological examination of the aneurysmal wall recognized fungus body and hypha. Voriconazole (VRCZ) was administered immediately. However, she died of multiple organ failure 14 days after surgery. Conclusion: In all previously reported cases, fungal aneurysm after a Lomentospora infection led to miserable sequelae even if the aneurysm was properly treated, and VRCZ was administrated. The peripheral portion of the aneurysm due to lomentosporiosis has never been reported. Hematogenous transmission was suggested as a possible developing mechanism.
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