Division of Nephrology, Department of Internal Medicine, Taichung Veterans General Hospital, Taichung, Taiwan.
Division of Nephrology, Taichung Veterans General Hospital Chiayi Branch.
Transplant Proc. 2023 May;55(4):788-791. doi: 10.1016/j.transproceed.2023.03.012. Epub 2023 May 23.
Fabry disease (FD) is an X-linked inborn error of lysosomal storage disorder, a deficiency in lysosomal hydrolase α-galactosidase A activity due to pathogenic variants in the GLA gene. Accumulation of globotriaosylceramide in multiple organs contributes to end-stage kidney disease, heart failure, and cerebrovascular accidents.
We began the FD screening program by involving male patients older than 20 years of age who were on chronic dialysis, had a post-kidney transplantation, and were part of the Pre-End Stage Renal Disease Program in our hospital. α-galactosidase A activity was detected through an initial dried blood spots screen assay, followed by levels of lyso-globotriaosylceramide and sequencing of the GLA gene when screening patients with suspected FD to confirm their diagnosis.
A total of 1812 patients had been FD screened, with the prevalence of FD being approximately 0.16 % (3/1812) up until June 2022. Interestingly, we confirmed a family cluster (2 sons and their mother) of having the c.936+919G>A mutation (designated GLA IVS4) with hypertrophic cardiomyopathy in Taiwan and another with the mutation c.644A>G (p.Asn215Ser), a more common later-onset variant reported in people of European or North American descent. Two patients were confirmed with cardiomyopathy through a cardiac biopsy, with their cardiac function later reversed after enzyme replacement therapy.
The FD screening test detects chronic kidney disease due to an unknown etiology and prevents other organ complications. Early detection of FD is crucial for reversing target organ damage with enzyme replacement therapy.
法布里病(FD)是一种 X 连锁的先天性溶酶体贮积病,由于 GLA 基因的致病性变异,溶酶体水解酶α-半乳糖苷酶 A 的活性缺失。糖鞘脂在多个器官中的积累导致终末期肾病、心力衰竭和脑血管意外。
我们通过参与我院慢性透析、肾移植后和终末期肾病前计划的 20 岁以上男性患者,开始了 FD 筛查计划。通过初始干血斑筛选检测α-半乳糖苷酶 A 活性,然后在筛选疑似 FD 患者以确认其诊断时检测溶酶体神经酰胺和 GLA 基因测序。
截至 2022 年 6 月,共对 1812 例患者进行了 FD 筛查,FD 的患病率约为 0.16%(3/1812)。有趣的是,我们在台湾确认了一个具有 c.936+919G>A 突变(命名为 GLA IVS4)的家族性聚集(2 个儿子及其母亲),伴有肥厚型心肌病,另一个具有突变 c.644A>G(p.Asn215Ser),这是一种在欧洲或北美裔人群中更为常见的迟发性变异。两名患者通过心脏活检确诊为心肌病,随后在酶替代治疗后心脏功能得到逆转。
FD 筛查检测到原因不明的慢性肾脏病,并预防其他器官并发症。早期发现 FD 对于用酶替代治疗逆转靶器官损伤至关重要。