Abstract
ObjectiveThe importance of environmental factors (especially leptospirosis) of moyamoya disease (MMD) has not been clarified. Here we investigated the epidemiological characteristics of MMD in Hubei province, China. MethodsWe conducted a population-based study to describe the epidemiologic characteristics of MMD in Hubei province between 2017 and 2019. The regional clusters of the hot spots (high incidence) and cold spots (low incidence) of MMD were identified using the spatial statistical method. To evaluate the role of leptospirosis in MMD, we performed an ecological comparison study to evaluate whether the socioeconomic and environmental variables of hot spots are more suitable for leptospirosis spread. ResultsThe average annual sex and age adjusted incidence of MMD was 3.07 per 100,000 person-years from 2017 to 2019. The middle-aged had apparently higher incidence of MMD than the children. There existed an obvious geographic distribution difference of MMD at county level. The hot spots were identified mainly in the low mountainous and hilly terrain, while cold spots were located in the Jianghan Plains. Compared to cold spots, the hot spots had larger cattle density (32.0 vs 3.1, p = 0.002), higher percentages of rice field (85.6% vs 47.0%, p = 0.007), and lower elevation (33.6 vs 157.4, p < 0.001) ConclusionsThere exists geographic distribution difference of MMD in Hubei province and was likely to be caused by leptospirosis in the early years. The MMD in China is dominant by middle-aged adults, which might be driven from leptospirosis outbreaks in the last century.
Highlights
Moyamoya disease (MMD) is a cerebrovascular disorder with unknown pathogenesis, characterized by chronic progressive occlusion of the terminal internal carotid artery and dilated collateral microvessels near the base skull, developing collateral circulation like "puff of smoke" on angiography[1,2]
We identified the obvious geographic distribution of MMD in the province, which initially strengthened the importance of environmental factors of this disease
In the 20th century when leptospirosis was epidemic in China, researchers reported that leptospiral cerebral vasculitis (LCV), one of the late complications of leptospirosis, which showed similar clinical, pathological as well as angiographic features with MMD7,8
Summary
Moyamoya disease (MMD) is a cerebrovascular disorder with unknown pathogenesis, characterized by chronic progressive occlusion of the terminal internal carotid artery and dilated collateral microvessels near the base skull, developing collateral circulation like "puff of smoke" on angiography[1,2]. MMD is not a common disease, it is the major reason for pediatric and young adult stroke in East Asian regions[3]. The etiology of MMD remain largely unknown. Genetical epidemiology has demonstrated that the ring finger protein 213 (RNF213) is a major susceptible gene for MMD among the East Asian populations[4,5]. The RNF213 mutation cannot explain the etiology of MMD alone because of its quite low penetrance in normal population[6]. Except for genetic factors, some environmental factors might trigger the start of MMD. The association between leptospirosis and MMD has not been clarified. The importance of environmental factors (especially leptospirosis) in moyamoya disease (MMD) has not been clarified. We investigated the epidemiological characteristics of MMD under perspective of ecology in Hubei province, China
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