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巴西男性血液透析患者中对法布里病的靶向筛查凸显了家族筛查的重要性。

Targeted Screening of Fabry Disease in Male Hemodialysis Patients in Brazil Highlights Importance of Family Screening.

作者信息

Silva Cassiano Augusto Braga, Barreto Fellype Carvalho, Dos Reis Marlene Antonia, Moura Junior José Andrade, Cruz Constança Margarida Sampaio

出版信息

Nephron. 2016;134(4):221-230. doi: 10.1159/000448740. Epub 2016 Aug 31.

Abstract

INTRODUCTION

Fabry disease (FD) is a lysosomal storage disorder caused by enzyme α galactosidase A (α-Gal A) deficiency due to mutations in the galactosidase alpha (GLA) gene. It leads to damage several organs, such as the kidneys, due to progressive accumulation of glycosphingolipids.

OBJECTIVE

To estimate the prevalence of FD among male hemodialysis (HD) patients in a northern state of Brazil.

METHODS

Screening was performed using a dried blood spot on filter paper to identify patients with low α-Gal A enzyme activity (≤2.2 µmol/l/h). Those with low enzyme activity underwent genetic analysis of the GLA gene. Family screening was conducted in the index cases.

RESULTS

2,583 male HD patients (age: 52 (18-91 years)) were screened. The α-Gal A assay identified 72 males (2.78%) with low enzyme activity. Genotyping identified 3 patients with GLA mutations: W204X, A368T, both previously reported; and C52F, a novel missense mutation. Only the patient with W204X mutation had classic FD. The prevalence rate was 0.12%. Family screening of the index cases identified 23 family members with the same mutations.

CONCLUSIONS

The prevalence of FD amongst male HD patients found in the Northern of Brazil was low (0.12%). However, family screening of the 3 index cases identified family members at an early stage of the disease, which may benefit from earlier treatment.

摘要

引言

法布里病(FD)是一种溶酶体贮积症,由半乳糖苷酶α(GLA)基因突变导致α半乳糖苷酶A(α-Gal A)缺乏引起。由于糖鞘脂的进行性积累,它会导致多个器官受损,如肾脏。

目的

评估巴西北部某州男性血液透析(HD)患者中法布里病的患病率。

方法

使用滤纸上的干血斑进行筛查,以识别α-Gal A酶活性低(≤2.2 µmol/l/h)的患者。酶活性低的患者接受GLA基因的遗传分析。对索引病例进行家系筛查。

结果

共筛查了2583名男性HD患者(年龄:52岁(18 - 91岁))。α-Gal A检测发现72名男性(2.78%)酶活性低。基因分型鉴定出3例GLA突变患者:W204X、A368T,均为先前报道的突变;以及C52F,一种新的错义突变。只有携带W204X突变者患有典型的法布里病。患病率为0.12%。对索引病例的家系筛查发现23名家庭成员有相同突变。

结论

在巴西北部发现的男性HD患者中法布里病的患病率较低(0.12%)。然而,对3例索引病例的家系筛查在疾病早期识别出了家庭成员,他们可能从早期治疗中获益。

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