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Chow Chow 型成骨不全症 COL1A2 基因候选突变的鉴定。

Identification of a Candidate Mutation in the COL1A2 Gene of a Chow Chow With Osteogenesis Imperfecta.

机构信息

Department of Veterinary Pathobiology, College of Veterinary Medicine and Biomedical Sciences, Texas A&M University, College Station, TX.

Department of Population Health and Reproduction, School of Veterinary Medicine, University of California, Davis, CA.

出版信息

J Hered. 2018 Mar 16;109(3):308-314. doi: 10.1093/jhered/esx074.

Abstract

Osteogenesis imperfecta (OI) is a genetic disease that occurs in humans and animals. Individuals with OI exhibit signs of extreme bone fragility and osteopenia with frequent fractures and perinatal lethality in severe cases. In this study, we report the clinical diagnosis of OI in a dog and the use of targeted next-generation sequencing to identify a candidate autosomal dominant mutation in the COL1A2 gene. A 5-month-old male Chow Chow was examined with a fractured left humerus and resolving, bilateral femoral fractures. Radiographs revealed generalized osteopenia and bilateral humeral, radial, and femoral fractures. Targeted next-generation sequencing of genes associated with OI in humans (COL1A1, COL1A2, LEPRE1, SERPINH1, and CRTAP) revealed a G>A heterozygous mutation in the splice donor site of exon 18 of the COL1A2 gene (c.936 + 1G>A). The splice donor mutation was not detected among 91 control dogs representing 21 breeds. A comparative analysis of exon 18 and the exon-intron junction further showed that the mutated splice donor site is conserved among vertebrates. Altogether, these findings reveal a candidate autosomal splice donor site mutation causing OI in an individual Chow Chow.

摘要

成骨不全症(OI)是一种发生在人类和动物身上的遗传性疾病。OI 患者表现出极端的骨骼脆弱和骨质疏松症的迹象,严重情况下会频繁骨折和围产期致死。在这项研究中,我们报告了一只犬的 OI 临床诊断,并使用靶向下一代测序来鉴定 COL1A2 基因中的候选常染色体显性突变。一只 5 个月大的雄性 Chow Chow 因左肱骨骨折和双侧股骨骨折接受了检查。X 光片显示广泛的骨质疏松症和双侧肱骨、桡骨和股骨骨折。针对人类 OI 相关基因(COL1A1、COL1A2、LEPRE1、SERPINH1 和 CRTAP)的靶向下一代测序显示,COL1A2 基因第 18 外显子剪接受体位点存在 G>A 杂合突变(c.936 + 1G>A)。在代表 21 个品种的 91 只对照犬中未检测到剪接受突变。对第 18 外显子和外显子-内含子接头的比较分析进一步表明,突变的剪接受位点在脊椎动物中是保守的。总之,这些发现揭示了 Chow Chow 个体中导致 OI 的候选常染色体剪接受体突变。

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