From the Department of Cerebrovascular Medicine, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan (K.M.).
Department of Neurology (K.M., M.S., H.M.), Osaka University Graduate School of Medicine, Japan.
Stroke. 2019 Apr;50(4):1010-1012. doi: 10.1161/STROKEAHA.118.024426.
Background and Purpose- The effect of enzyme replacement therapy (ERT) on cerebrovascular complications remains largely unexplored. We aimed to investigate the relationship between basilar artery (BA) diameter and long-term ERT in patients with Fabry disease. Methods- We obtained baseline magnetic resonance imaging data from 30 patients (40.5±16.3 years, male: 14) in the single institution; among them, 21 patients prospectively had follow-up magnetic resonance imaging assessments. The short axis of BA diameter was measured at the midpons level on axial T2-weighted images. Brain magnetic resonance imaging measurements included markers of cerebral small vessel disease (lacunas, white matter hyperintensities, and cerebral microbleeds). We assessed variables associated with baseline BA diameter and annual BA diameter change using linear regression analyses. Results- Hypertension, left ventricular hypertrophy, estimated glomerular filtration rate, and white matter hyperintensities correlated with the initial BA diameter. After a mean interval of 7.2±4.6 years, the annual BA diameter change correlated positively with severe white matter hyperintensities and inversely with the duration of ERT (β=-0.48, P=0.033). After stratifying patients by sex, a significant correlation between the duration of ERT and BA diameter was found only in men (β=-0.72, P=0.019). Conclusions- Our results show a possible relationship between ERT and changes in BA diameter. Future studies to elucidate the clinical impact of BA changes as a potential surrogate marker are needed.
背景与目的- 酶替代疗法(ERT)对脑血管并发症的影响在很大程度上仍未得到探索。我们旨在研究基底动脉(BA)直径与法布里病患者长期 ERT 之间的关系。
方法- 我们从单一机构中获得了 30 名患者(40.5±16.3 岁,男性:14)的基线磁共振成像数据;其中,21 名患者前瞻性地进行了磁共振成像随访评估。在轴向 T2 加权图像上于桥脑水平测量 BA 短轴直径。脑磁共振成像测量包括脑小血管疾病的标志物(腔隙、脑白质高信号和脑微出血)。我们使用线性回归分析评估与基线 BA 直径和年度 BA 直径变化相关的变量。
结果- 高血压、左心室肥厚、估计肾小球滤过率和脑白质高信号与初始 BA 直径相关。在平均 7.2±4.6 年的随访后,BA 直径的年变化与严重的脑白质高信号呈正相关,与 ERT 持续时间呈负相关(β=-0.48,P=0.033)。在按性别分层患者后,仅在男性中发现 ERT 持续时间与 BA 直径之间存在显著相关性(β=-0.72,P=0.019)。
结论- 我们的结果表明 ERT 与 BA 直径变化之间可能存在关系。需要进一步的研究来阐明 BA 变化作为潜在替代标志物的临床影响。