Yohei Mineharu1, Yasushi Takagi2, Akio Koizumi3, Takaaki Morimoto4, Takeshi Funaki1, Tomohito Hishikawa5, Yoshio Araki6, Hitoshi Hasegawa7, Jun C Takahashi8, Satoshi Kuroda9, Kiyohiro Houkin10, Susumu Miyamoto1. 1. 1Department of Neurosurgery, Kyoto University Graduate School of Medicine, Kyoto. 2. 2Department of Neurosurgery, Tokushima University Graduate School of Medicine, Tokushima. 3. 3Social Health Welfare Medicine Laboratory, Kyoto. 4. 4Department of Neurosurgery, Hyogo Prefectural Amagasaki General Medical Center, Amagasaki. 5. 5Okayama University Graduate School of Medicine, Okayama. 6. 6Department of Neurosurgery, Nagoya University Graduate School of Medicine, Nagoya. 7. 7Department of Neurosurgery, Brain Research Institute, Niigata University, Niigata. 8. 8Department of Neurosurgery, National Cerebral and Cardiovascular Center, Suita. 9. 9Department of Neurosurgery, Toyama University Graduate School of Medicine, Toyama; and. 10. 10Department of Neurological Cell Therapy, Hokkaido University Graduate School of Medicine, Sapporo, Japan.
Abstract
OBJECTIVE: Although many studies have analyzed risk factors for contralateral progression in unilateral moyamoya disease, they have not been fully elucidated. The aim of this study was to examine whether genetic factors as well as nongenetic factors are involved in the contralateral progression. METHODS: The authors performed a multicenter cohort study in which 93 cases with unilateral moyamoya disease were retrospectively reviewed. The demographic features, RNF213 R4810K mutation, lifestyle factors such as smoking and drinking, past medical history, and angiographic findings were analyzed. A Cox proportional hazards model was used to find risk factors for contralateral progression. RESULTS: Contralateral progression was observed in 24.7% of cases during a mean follow-up period of 72.2 months. Clinical characteristics were not significantly different between 67 patients with the R4810K mutation and those without it. Cox regression analysis showed that the R4810K mutation (hazard ratio [HR] 4.64, p = 0.044), childhood onset (HR 7.21, p < 0.001), male sex (HR 2.85, p = 0.023), and daily alcohol drinking (HR 4.25, p = 0.034) were independent risk factors for contralateral progression. CONCLUSIONS: These results indicate that both genetic and nongenetic factors are associated with contralateral progression of unilateral moyamoya disease. The findings would serve to help us better understand the pathophysiology of moyamoya disease and to manage patients more appropriately.
OBJECTIVE: Although many studies have analyzed risk factors for contralateral progression in unilateral moyamoya disease, they have not been fully elucidated. The aim of this study was to examine whether genetic factors as well as nongenetic factors are involved in the contralateral progression. METHODS: The authors performed a multicenter cohort study in which 93 cases with unilateral moyamoya disease were retrospectively reviewed. The demographic features, RNF213 R4810K mutation, lifestyle factors such as smoking and drinking, past medical history, and angiographic findings were analyzed. A Cox proportional hazards model was used to find risk factors for contralateral progression. RESULTS: Contralateral progression was observed in 24.7% of cases during a mean follow-up period of 72.2 months. Clinical characteristics were not significantly different between 67 patients with the R4810K mutation and those without it. Cox regression analysis showed that the R4810K mutation (hazard ratio [HR] 4.64, p = 0.044), childhood onset (HR 7.21, p < 0.001), male sex (HR 2.85, p = 0.023), and daily alcohol drinking (HR 4.25, p = 0.034) were independent risk factors for contralateral progression. CONCLUSIONS: These results indicate that both genetic and nongenetic factors are associated with contralateral progression of unilateral moyamoya disease. The findings would serve to help us better understand the pathophysiology of moyamoya disease and to manage patients more appropriately.
Authors: Bryan J Neth; Samantha N Balakrishnan; Ivan D Carabenciov; Joon H Uhm; David J Daniels; Sani H Kizilbash; Michael W Ruff Journal: J Neurooncol Date: 2022-01-25 Impact factor: 4.130