Ye Min, Ma Ya, Qin Yi-Xuan, Cai Bo, Ma Li-Mei, Ma Zhen, Liu Yang, Jin Zi-Bing, Zhuang Wen-Juan
Third Clinical Medical College, Ningxia Medical University, Yinchuan, China.
Ningxia Eye Hospital, People's Hospital of Ningxia Hui Autonomous Region, Third Clinical Medical College of Ningxia Medical University, Yinchuan, China.
Mol Genet Genomics. 2023 May;298(3):669-682. doi: 10.1007/s00438-023-02003-7. Epub 2023 Mar 25.
High myopia (HM) is a leading cause of visual impairment in the world. To expand the genotypic and phenotypic spectra of HM in the Chinese population, we investigated genetic variations in a cohort of 113 families with nonsyndromic early-onset high myopia from northwestern China by whole-exome sequencing, with focus on 17 known genes. Sixteen potentially pathogenic variants predicted to affect protein function in eight of seventeen causative genes for HM in fifteen (13.3%) families were revealed, including seven novel variants, c.767 + 1G > A in ARR3, c.3214C > A/p.H1072N, and c.2195C > T/p.A732V in ZNF644, c.1270G > T/p.V424L in CPSF1, c.1918G > C/p.G640R and c.2786T > G/p.V929G in XYLT1, c.601G > C/p.E201Q in P4HA2; six rare variants, c.799G > A/p.E267K in NDUFAF7, c.1144C > T/p.R382W in TNFRSF21, c.1100C > T/p.P367L in ZNF644, c.3980C > T/p.S1327L in CPSF1, c.145G > A/p.E49K and c.325G > T/p.G109W in SLC39A5; and three known variants, c.2014A > G/p.S672G and c.3261A > C/p.E1087D in ZNF644, c.605C > T/p.P202L in TNFRSF21. Ten of them were co-segregated with HM. The mean (± SD) examination age of these 15 probands was 14.7 (± 11.61) years. The median spherical equivalent was - 9.50 D (IQ - 8.75 ~ - 12.00) for the right eye and - 11.25 D (IQ - 9.25 ~ - 14.13) for the left eye. The median axial length was 26.67 mm (IQ 25.83 ~ 27.13) for the right eye and 26.25 mm (IQ 25.97 ~ 27.32) for the left eye. These newly identified genetic variations not only broaden the genetic and clinical spectra, but also offer convincing evidence that the genes ARR3, NDUFAF7, TNFRSF21, and ZNF644 contribute to hereditable HM. This work improves further understanding of molecular mechanism of HM.
高度近视(HM)是全球视力损害的主要原因。为了扩大中国人群中HM的基因型和表型谱,我们通过全外显子测序研究了来自中国西北的113个非综合征性早发性高度近视家系的遗传变异,重点关注17个已知基因。在15个(13.3%)家系中,17个HM致病基因中的8个基因发现了16个预测会影响蛋白质功能的潜在致病变异,包括7个新变异,分别为ARR3基因中的c.767+1G>A、ZNF644基因中的c.3214C>A/p.H1072N和c.2195C>T/p.A732V、CPSF1基因中的c.1270G>T/p.V424L、XYLT1基因中的c.1918G>C/p.G640R和c.2786T>G/p.V929G、P4HA2基因中的c.601G>C/p.E201Q;6个罕见变异,分别为NDUFAF7基因中的c.799G>A/p.E267K、TNFRSF21基因中的c.1144C>T/p.R382W、ZNF644基因中的c.1100C>T/p.P367L、CPSF1基因中的c.3980C>T/p.S1327L、SLC39A5基因中的c.145G>A/p.E49K和c.325G>T/p.G109W;以及3个已知变异,分别为ZNF644基因中的c.2014A>G/p.S672G和c.3261A>C/p.E1087D、TNFRSF21基因中的c.605C>T/p.P202L。其中10个与HM共分离。这15名先证者的平均(±标准差)检查年龄为14.7(±11.61)岁。右眼等效球镜的中位数为-9.50D(四分位间距-8.75 ~ -12.00),左眼为-11.25D(四分位间距-9.25 ~ -14.13)。右眼眼轴长度的中位数为26.67mm(四分位间距25.83 ~ 27.13),左眼为26.25mm(四分位间距25.97 ~ 27.32)。这些新发现的遗传变异不仅拓宽了遗传和临床谱,也提供了令人信服的证据,证明ARR3、NDUFAF7、TNFRSF21和ZNF644基因与遗传性HM有关。这项工作增进了对HM分子机制的进一步理解。