title: Recurrent cerebral aneurysm formation and rupture within a short period due to invasive aspergillosis of the nasal sinus; pathological analysis of the catastrophic clinical course.
abstract: Destructive infiltration of invasive fungal sinusitis can easily occur into the central nervous system (CNS). Cerebral aneurysms associated with fungal infection are highly vulnerable to rupture, and can frequently and rapidly take a serious clinical course. We experienced a patient who twice developed cerebral aneurysm followed by rupture due to invasive fugal sinusitis. This 77-year-old man was admitted for progressive bilateral visual disturbance, which was initially treated as idiopathic hypertrophic pachymeningitis. The patient subsequently suffered subarachnoid hemorrhage (SAH) twice in only 12 days. Both SAH originated from different newly formed cerebral aneurysms. Trapping was performed for both ruptured aneurysms. Pathological examination of the resected aneurysms indicated the presence of fungi determined to be Aspergillus. This Aspergillus infection was also discovered inside the frontal sinus by endoscopic biopsy, so a regimen of antifungal agents was instituted. Prolonged antifungal therapy caused renal impairment, which ultimately led to the patient's death. Autopsy detected no mycotic infiltration of the major cerebral arteries, except for the 2 ruptured cerebral aneurysms. However, prolonged mycosis of the CNS, such as in the deep part in the falx cerebri and in the small veins proximal to the tentorium cerebelli, was observed, indicating that mycosis invading the cranium is refractory even to long-term administration of antifungal agents. The present case strongly suggests that urgent and proactive definitive diagnosis is essential to successfully treat invasive paranasal sinus aspergillosis. If infiltration of the CNS is suspected, early surgical resection and antifungal therapy must be initiated immediately.
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Yuki
authors.initials: Y
authors.lastname: Shinya
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Satoru
authors.initials: S
authors.lastname: Miyawaki
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Hirofumi
authors.initials: H
authors.lastname: Nakatomi
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Atsushi
authors.initials: A
authors.lastname: Okano
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Hideaki
authors.initials: H
authors.lastname: Imai
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Masahiro
authors.initials: M
authors.lastname: Shin
authors.affiliation: Department of Neurology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Kazuya
authors.initials: K
authors.lastname: Sato
authors.affiliation: Department of Neurology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Takeyuki
authors.initials: T
authors.lastname: Tsuchida
authors.affiliation: Department of Neurology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Toshihiro
authors.initials: T
authors.lastname: Hayashi
authors.affiliation: Department of Neurology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Yasuo
authors.initials: Y
authors.lastname: Terao
authors.affiliation: Department of Pathology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Satoe
authors.initials: S
authors.lastname: Numakura
authors.affiliation: Department of Pathology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Teppei
authors.initials: T
authors.lastname: Morikawa
authors.affiliation: Department of Pathology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Junji
authors.initials: J
authors.lastname: Shibahara
authors.affiliation: Department of Otolaryngology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Shu
authors.initials: S
authors.lastname: Kikuta
authors.affiliation: Department of Otolaryngology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Kenji
authors.initials: K
authors.lastname: Kondo
authors.affiliation: Department of Infectious Diseases, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Keita
authors.initials: K
authors.lastname: Tatsuno
authors.affiliation: Department of Radiology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Harushi
authors.initials: H
authors.lastname: Mori
authors.affiliation: Department of Radiology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Akira
authors.initials: A
authors.lastname: Kunimatsu
authors.affiliation: Department of Neurology, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Shoji
authors.initials: S
authors.lastname: Tsuji
authors.affiliation: Department of Neurosurgery, Faculty of Medicine, The University of Tokyo Tokyo, Japan.
authors.forename: Nobuhito
authors.initials: N
authors.lastname: Saito
mesh_terms: D000368:Aged; D017542:Aneurysm, Ruptured / Q000382:microbiology* / Q000473:pathology; D000935:Antifungal Agents / Q000627:therapeutic use; D001228:Aspergillosis / Q000150:complications* / Q000188:drug therapy / Q000473:pathology; D006801:Humans; D002532:Intracranial Aneurysm / Q000382:microbiology* / Q000473:pathology; D008297:Male; D012008:Recurrence; D012852:Sinusitis / Q000382:microbiology* / Q000473:pathology
chemical_list: D000935:Antifungal Agents
keywords: Aspergillus; Mycotic cerebral aneurysm; fungal sinusitis; subarachnoid hemorrhage