Medical Biology Department.
Neurology Department, Bursa Uludag University, Faculty of Medicine, Gorukle, Bursa.
Alzheimer Dis Assoc Disord. 2021;35(3):214-222. doi: 10.1097/WAD.0000000000000437.
Early-onset Alzheimer disease (EOAD) is an earlier Alzheimer disease form which is characterized by the mutations in the amyloid precursor protein, presenilin-1/2 (PSEN1/2), and triggering receptor expressed on myeloid cells 2 (TREM2). However, it is still necessary to report mutational screening in multiethnic groups to improve the genetic background of EOAD due to the variant classification challenge.
We performed targeted sequencing for the amyloid precursor protein, PSEN1, PSEN2, and TREM2 genes in 74 patients and 1 family diagnosed with EOAD.
Among the detected variants, 8 were coding and 6 were noncoding in 15 of 74 patients. In PSEN1, 2 pathogenic coding variants (T274K and L364P) detected in 2 patients were novel and 3 coding variants (G183V, E318G, and L219P) detected in 2 patients were previously reported. We found 4 patients with the compound heterozygosity for the PSEN2 A23= and N43= and a family with the coexistence of them, and 1 patient with TREM2 Y38C. The coding variation frequency was 12.1%. In silico analysis indicated pathogenic potentials and clinical interpretations of the detected variants.
Our study reveals the rare gene variants including novel ones from the Turkish EOAD cohort and provides to clinicians the list of detected variants in the screened genes, which may also be useful for accurate genetic counseling.
早发性阿尔茨海默病(EOAD)是一种更早的阿尔茨海默病形式,其特征是淀粉样前体蛋白、早老素-1/2(PSEN1/2)和髓样细胞表达的触发受体 2(TREM2)的突变。然而,由于变异分类的挑战,仍有必要在多民族群体中报告突变筛查,以改善 EOAD 的遗传背景。
我们对 74 名患者和 1 个被诊断为 EOAD 的家族进行了淀粉样前体蛋白、PSEN1、PSEN2 和 TREM2 基因的靶向测序。
在所检测的变异中,在 74 名患者中的 15 名中,有 8 个为编码变异,6 个为非编码变异。在 PSEN1 中,我们在 2 名患者中检测到 2 个新的致病性编码变异(T274K 和 L364P)和 2 名患者中以前报道的 3 个编码变异(G183V、E318G 和 L219P)。我们发现 4 名患者存在 PSEN2 A23=和 N43=的复合杂合性,1 个家系存在它们的共存,1 名患者存在 TREM2 Y38C。编码变异频率为 12.1%。计算机分析表明了所检测变异的致病性潜力和临床解释。
我们的研究揭示了来自土耳其 EOAD 队列的罕见基因变异,包括新的变异,并为临床医生提供了筛选基因中检测到的变异列表,这也可能有助于准确的遗传咨询。