Division of Cardiology, Nephrology, Pulmonology, and Neurology, Department of Internal Medicine, Asahikawa Medical University, Japan.
Department of Neurology, Asahikawa City Hospital, Japan.
Intern Med. 2021 Jun 15;60(12):1819-1826. doi: 10.2169/internalmedicine.6420-20. Epub 2021 Jan 15.
Objective Fabry disease (FD) is a hereditary lysosomal storage disease that has been highlighted as a possible etiology of stroke at a young age and presents with other various neurological symptoms. Since FD is rare, limited information is currently available on the prevalence of neurological symptoms in Japanese patients with FD. Therefore, we examined the characteristics of neurological symptoms and brain magnetic resonance imaging (MRI) findings in adult Japanese patients with FD. Methods This was a retrospective, single-center study. We reviewed neurological symptoms and brain MRI findings in the medical records of 12 adult Japanese patients with FD diagnosed by a gene analysis of the α-galactosidase gene. Results Ten out of 12 patients with FD presented with the following neurological symptoms: acroparesthesia (n=6), headache (n=5) [migraine (n=4)], hypohidrosis (n=5), and cerebral infarction (n=3). Two and three of the patients with migraine were complicated by ischemic stroke and coronary spastic angina, respectively. Five and 10 patients presented with periventricular hyperintensity and deep white matter hyperintensity, respectively, on brain MRI. Two out of eight patients had cerebral microbleeds. Seven out of 11 patients had a dilated basilar artery diameter on magnetic resonance angiography. There were no patients with the pulvinar hyperintensity sign. Conclusion Patients with FD present with various neurological symptoms. Headache, particularly migraine, might be a major neurological symptom in patients with FD. Since migraine, ischemic stroke, and coronary spastic angina might occur together in FD, caution is needed when administering triptan to FD patients with migraine.
目的
法布里病(FD)是一种遗传性溶酶体贮积病,已被强调为年轻时发生中风的可能病因,并伴有其他各种神经症状。由于 FD 较为罕见,目前关于 FD 日本患者神经症状的患病率的信息有限。因此,我们研究了成年日本 FD 患者的神经症状和脑磁共振成像(MRI)表现的特征。
方法
这是一项回顾性、单中心研究。我们回顾了 12 例经α-半乳糖苷酶基因分析诊断为 FD 的成年日本患者的病历中的神经症状和脑 MRI 表现。
结果
12 例 FD 患者中有 10 例出现以下神经症状:肢端感觉异常(n=6)、头痛(n=5)[偏头痛(n=4)]、少汗(n=5)和脑梗死(n=3)。2 例偏头痛患者分别合并缺血性卒中和冠状动脉痉挛性心绞痛,3 例患者的偏头痛患者分别合并缺血性卒中和冠状动脉痉挛性心绞痛。5 例和 10 例患者的脑 MRI 分别显示脑室周围高信号和深部白质高信号。2 例患者有脑微出血。8 例患者中有 7 例磁共振血管造影显示基底动脉直径扩张。无患者出现丘脑出血高信号征。
结论
FD 患者表现出各种神经症状。头痛,尤其是偏头痛,可能是 FD 患者的主要神经症状。由于偏头痛、缺血性卒中和冠状动脉痉挛性心绞痛可能同时发生于 FD 中,因此在给偏头痛的 FD 患者使用曲坦类药物时需谨慎。