Division of Neurology, Faculty of Medicine, University of British Columbia, 2215 Wesbrook Mall, Vancouver, BC, V6T 1Z3, Canada.
Department of Medical Genetics, University of British Columbia, Vancouver, Canada.
Hum Genet. 2017 Jun;136(6):705-714. doi: 10.1007/s00439-017-1784-9. Epub 2017 Mar 23.
Several single-gene disorders with clinical and radiological characteristics similar to those observed in multiple sclerosis (MS) patients have been described. To evaluate whether this phenotypic overlap can be ascribed to a common genetic etiology, 28 genes known to present pathogenic mutations for 24 of these disorders were sequenced in 270 MS patients. All identified variants were genotyped in 2131 MS cases and 830 healthy controls, and those exclusively observed in patients were assessed for segregation within families. This analysis identified 9 rare variants in 6 genes segregating with disease in 13 families. Four different mutations were identified in CYP27A1, including a reported pathogenic mutation for cerebrotendinous xanthomatosis (p.R405W), which was observed in six patients from a multi-incident family, three diagnosed with MS, two with an undefined neurological disease and one seemingly healthy. A LYST p.V1678A and a PDHA1 p.K387Q mutation were both observed in five MS patients from three separate multi-incident families. In addition, CLCN2 p.V174G, GALC p.D162E and POLG p.R361G were each identified in two MS patients from one family. This study suggests a shared genetic etiology between MS and the characterized single-gene disorders, and highlights cholesterol metabolism and the synthesis of oxysterols as important biological mechanisms for familial MS.
已经描述了几种具有与多发性硬化症(MS)患者相似的临床和影像学特征的单基因疾病。为了评估这种表型重叠是否可以归因于共同的遗传病因,我们对 270 名 MS 患者进行了 28 个已知存在致病性突变的基因测序,这些基因与 24 种此类疾病有关。在 2131 例 MS 病例和 830 例健康对照中对所有鉴定的变体进行了基因分型,并对仅在患者中观察到的变体进行了家系内分离评估。这项分析在 13 个家系中发现了 6 个基因中的 9 个罕见变体与疾病共分离。在 CYP27A1 中发现了 4 种不同的突变,包括报道的对脑腱黄瘤病的致病性突变(p.R405W),在一个多发病例家族的 6 名患者中观察到,其中 3 名被诊断为 MS,2 名患有未明确的神经疾病,1 名似乎健康。LYST p.V1678A 和 PDHA1 p.K387Q 突变均在三个独立的多发病例家族的 5 名 MS 患者中观察到。此外,CLCN2 p.V174G、GALC p.D162E 和 POLG p.R361G 分别在一个家族的 2 名 MS 患者中发现。这项研究表明 MS 与已描述的单基因疾病之间存在共同的遗传病因,并强调胆固醇代谢和氧化固醇的合成是家族性 MS 的重要生物学机制。