Suppr超能文献

重新审视一个携带多种 GBA1 变异的家族中的 Gaucher 病诊断。

Revisiting the diagnosis of Gaucher disease in a family with multiple GBA1 variants.

机构信息

Medical Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland, USA.

出版信息

Am J Med Genet A. 2023 Oct;191(10):2647-2650. doi: 10.1002/ajmg.a.63345. Epub 2023 Jul 14.

Abstract

Our ability to identify different variants in GBA1, the gene mutated in the lysosomal storage disorder Gaucher disease (GD), has greatly improved. We describe a multigenerational family with type 1 GD initially evaluated over three decades ago. Re-evaluating both the genotype and phenotype, we determined that one family member with genotype N370S/T369M (p.N409S/p.T408M), was likely erroneously diagnosed with GD. This case substantiates that GBA1 variant T369M, while mildly reducing glucocerebrosidase activity, does not result in GD. The observation has clinical relevance as cases with this genotype will increasingly be ascertained through screening programs in newborns and in movement disorder clinics.

摘要

我们鉴定 GBA1 中不同变异的能力(GBA1 基因发生突变会导致溶酶体贮积症——戈谢病)已经大大提高。我们描述了一个多代家族,该家族患有戈谢病(GD),最初是在三十多年前进行评估的。重新评估基因型和表型后,我们确定一位基因型为 N370S/T369M(p.N409S/p.T408M)的家族成员可能被误诊为 GD。该病例证实,GBA1 变体 T369M 虽然会轻微降低葡萄糖脑苷脂酶的活性,但不会导致 GD。这种观察结果具有临床意义,因为通过新生儿筛查计划和运动障碍诊所,越来越多的此类基因型病例将会被发现。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验