Hemorrhagic Adult Unilateral Moyamoya Disease with Multiple Unruptured Intracranial Aneurysms: A Case Report

• Published in: Nō shinkei geka Vol. 44; no. 2; p. 121
• Main Authors: Ozaki, Saya, Inoue, Akihiro, Miyazaki, Hajime, Onoue, Shinji, Ichikawa, Haruhisa, Fukumoto, Shinya, Iwata, Shinji, Kohno, Kanehisa
• Format: Journal Article
• Language: Japanese
• Published: Japan 01.02.2016
• Subjects: Cerebral Angiography - methods; Cerebral Hemorrhage - diagnosis; Cerebral Hemorrhage - etiology; Cerebral Hemorrhage - surgery; Cerebral Revascularization - methods; Humans; Intracranial Aneurysm - diagnosis; Intracranial Aneurysm - etiology; Intracranial Aneurysm - surgery; Male; Middle Aged; Moyamoya Disease - complications; Moyamoya Disease - diagnosis; Moyamoya Disease - surgery
• ISSN: 0301-2603
• DOI: 10.11477/mf.1436203242

Adult unilateral moyamoya disease with intracranial aneurysm is frequently reported in the literature, but there is much variation in its treatment. In this case report, we describe the time course and treatment regimen of a patient with moyamoya disease and review the literature regarding moyamoya disease with intracranial aneurysm. A 64-year-old man had untreated intracranial aneurysm and unilateral moyamoya disease for 10 years. He presented with sudden-onset right hemiparesis and aphasia due to a subcortical hemorrhage. He was admitted to the local neurosurgical unit, and upon resolution of symptoms, he was admitted to our hospital. A cerebral angiogram revealed the champagne bottleneck sign of the left carotid artery and obliteration of the top of the left intracranial carotid artery with a moyamoya phenomenon. Two unruptured intracranial aneurysms were identified in the anterior communicating artery(Acom A) and the right intracranial carotid artery(C3). We performed superficial temporal artery-middle cerebral artery anastomosis followed by aneurysmal neck clipping of the Acom A aneurysm. Postoperative imaging showed no new ischemic damage and improved cerebral blood flow. Although the patient experienced temporal worsening of aphasia, his function recovered a few months later and he was able to resume his normal daily life activities. The combination of direct bypass surgery and aneurysmal neck clipping might be a therapeutic option for hemorrhagic unilateral moyamoya disease with unruptured intracranial aneurysm.