Medical Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland, USA.
Am J Med Genet A. 2023 Oct;191(10):2647-2650. doi: 10.1002/ajmg.a.63345. Epub 2023 Jul 14.
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.
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。