Tolosa-Hunt症候群が疑われたFungal Aneurysmの1例

A Fungal Aneurysm in a Patient with Presumed Tolosa-Hunt Syndrome

I．はじめに

　Tolosa-Hunt syndrome（以下，THS）は，一側性，反復性の眼筋麻痺と眼窩深部の疼痛を症状とし，その本態は海綿静脈洞から上眼窩裂にかけての非特異的炎症性肉芽腫によるものとされ，ステロイドが有効であると言われている^11）．

　今回われわれは，THS様症状を呈した患者にステロイドを投与したところ，一部症状の改善を認めたが，突然脳内出血を来した症例を経験した．手術並びに剖検の結果，THS様症状は海綿静脈洞部の真菌性膿瘍に起因し，脳内出血は真菌性脳動脈瘤の破裂によることが判明した．

Abstract

A case of fungal aneurysm associated with presumed Tolosa-Hunt syndrome is reported. A 57-year-old man was admitted to our hospital with complaints of left blephaloptosis, headache and weight loss. Neurological examination revealed left ophthalmople-gia without facial hypesthesia. Visual acuity was nor-mal. Laboratory studies showed raised ESR, 4+ glycos-uria, and a blood sugar of 351mg/di. Computerized tomography (CT) scan and left carotid angiography were considered normal. Left orbital venography showed no filling of the left cavernous sinus. Diabetic ophthalmoplegia was suspected by a neurologist.The patient was treated with insulin therapy, but visual acuity worsened, and hypesthesia was noted in the first and second divisions of the left trigeminal nerve. Subse-quent CT scan demonstrated a high density lesion, which was homogenously enhanced, in the left caver-nous portion and the superior orbital fissure. The pa-tient was presumed of Tolosa-Hunt syndrome, and pred-nine therapy (30mg/day) was started.

On the second day after the administration of pred-nine, hypesthesia of the first and second division of the left trigeminal nerve improved. After 9 days of pred-nine therapy, the patient suddenly complained of severe headache, and lapsed into a coma. Massive hemorrhage with subarachnoid hemorrhage was recognized on the CT scan, with a marked midline shift to the right.

The hematoma was immediately removed. A rup-tured cerebral aneurysm was found at the bottom of the hematoma. The aneurysm was located in the distal portion of the left middle cerebral artery. Aneurysm clipping with external decompression and bilateral ven-tricular drainage was performed. The patient died nine days after the operation. An autopsy disclosed an abs-cess extending from the left cavernous portion to the left superior orbital fissure.

Microscopic examination showed that the abscess contained necrotic debris with a mass of fungal hyphae, subsequently identified as Phyomycetes.

The risk of diagnostic steroid therapy for patients presumed of Tolosa-Hunt syndrome is discussed.