Medical Genome Center, Research Institute, National Center for Geriatrics and Gerontology, Obu, Aichi, Japan.
RIKEN Center for Integrative Medical Sciences, Yokohama, Kanagawa, Japan.
Am J Med Genet B Neuropsychiatr Genet. 2022 Jul;189(5):139-150. doi: 10.1002/ajmg.b.32908. Epub 2022 Jun 28.
Dementia with Lewy bodies (DLB) is the second most common form of neurodegenerative dementia in elderly people, following Alzheimer's disease. Only three genes, SNCA (α-synuclein), APOE (apolipoprotein E), and GBA (glucosylceramidase), have been convincingly demonstrated to be associated with DLB. Here, we applied whole-genome sequencing to blood samples from 61 DLB patients and 45 cognitively normal controls. We used accumulation of candidate mutations to detect novel DLB-associated genes. Subsequent single nucleotide polymorphism (SNP) genotyping and association studies in a large number of samples from Japanese individuals revealed novel heterozygous variants in MFSD3 (rs143475431, c.888T>A:p.C296*; n = 5,421, p = 0.00063) and MRPL43 (chr10:102746730, c.241A>C:p.N81H; n = 4,782, p = 0.0029). We further found that the MFSD3 variant increased plasma levels of butyrylcholinesterase (n = 1,206, p = 0.029). We believe that our findings will contribute to the understanding of DLB and provide insight into its pathogenic mechanism for future studies.
路易体痴呆症 (DLB) 是仅次于阿尔茨海默病的老年人第二常见的神经退行性痴呆症。只有三个基因,即 SNCA(α-突触核蛋白)、APOE(载脂蛋白 E)和 GBA(葡萄糖脑苷脂酶),已被令人信服地证明与 DLB 相关。在这里,我们对 61 名 DLB 患者和 45 名认知正常对照者的血液样本进行了全基因组测序。我们使用候选突变的积累来检测新的与 DLB 相关的基因。随后在大量来自日本个体的样本中进行单核苷酸多态性 (SNP) 基因分型和关联研究,揭示了 MFSD3(rs143475431,c.888T>A:p.C296*;n=5,421,p=0.00063)和 MRPL43(chr10:102746730,c.241A>C:p.N81H;n=4,782,p=0.0029)中存在新的杂合变异。我们还发现 MFSD3 变异增加了血浆中丁酰胆碱酯酶的水平(n=1,206,p=0.029)。我们相信我们的发现将有助于理解 DLB,并为其发病机制提供见解,以用于未来的研究。