USDA/ARS/Western Human Nutrition Research Center, Obesity and Metabolism Research Unit, Davis, California 95616, USA.
Graduate Group of Nutritional Biology, Department of Nutrition, University of California Davis, Davis, California 95616, USA.
Hum Mol Genet. 2023 Jun 5;32(12):2016-2031. doi: 10.1093/hmg/ddad033.
Zinc is an essential trace mineral. Dietary zinc deficiency results in stunted growth, skin lesions, hypogonadism and frequent infections in humans. Mice genetically lacking Slc30a7 suffer from mild zinc deficiency and are prone to development of prostate cancer and insulin resistance. Disease-causing variants or mutations in the human SLC30A7 (ZNT7) gene have not been previously reported. Here, we describe two-boy siblings from a French family with stunted growth, testicular hypoplasia and bone marrow failure. Exome sequencing revealed compound heterozygous variants in ZNT7 consisting of NM_133496.5:c.21dup; p.Asp8ArgfsTer3 and c.842 + 15 T > C inherited from their unaffected mother and father, respectively. The c.21dup variant led to a premature stop codon generated in exon 1 of the ZNT7 coding sequence. RNA-seq analysis demonstrated that the c.842 + 15 T > C variant resulted in a leaky mRNA splicing event generating a premature stop codon right after the splicing donor site of exon 8. Moreover, the expression of ZNT7 protein was remarkably reduced by 80-96% in the affected brothers compared to the control cells. These findings strongly suggest that biallelic variants in SLC30A7 should be considered as a cause of growth retardation, testicular hypoplasia and syndromic bone marrow failure.
锌是一种必需的微量元素。饮食中锌缺乏会导致人类生长迟缓、皮肤损伤、性腺功能减退和频繁感染。遗传缺乏 Slc30a7 的小鼠患有轻度锌缺乏症,并且容易发展为前列腺癌和胰岛素抵抗。人类 SLC30A7(ZNT7)基因中的致病变体或突变以前尚未报道过。在这里,我们描述了一对来自法国家庭的两兄弟,他们患有生长迟缓、睾丸发育不全和骨髓衰竭。外显子组测序显示 ZNT7 中的复合杂合变体由 NM_133496.5:c.21dup;p.Asp8ArgfsTer3 和 c.842 + 15T > C 组成,分别来自未受影响的母亲和父亲。c.21dup 变体导致 ZNT7 编码序列外显子 1 中产生一个过早的终止密码子。RNA-seq 分析表明,c.842 + 15T > C 变体导致一个渗漏的 mRNA 剪接事件,在 exon 8 的剪接受体位点之后产生一个过早的终止密码子。此外,与对照细胞相比,受影响的兄弟中 ZNT7 蛋白的表达显著降低了 80-96%。这些发现强烈表明 SLC30A7 的双等位基因变异应被视为生长迟缓、睾丸发育不全和综合征性骨髓衰竭的原因。