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基因检测确诊一名7个月大婴儿患X连锁低磷性佝偻病

Genetic Testing Confirmed the Early Diagnosis of X-Linked Hypophosphatemic Rickets in a 7-Month-Old Infant.

作者信息

Poon Kok Siong, Sng Andrew Anjian, Ho Cindy Weili, Koay Evelyn Siew-Chuan, Loke Kah Yin

机构信息

National University Health System, Singapore.

National University of Singapore, Singapore.

出版信息

J Investig Med High Impact Case Rep. 2015 Aug 3;3(3):2324709615598167. doi: 10.1177/2324709615598167. eCollection 2015 Jul-Sep.

Abstract

Loss-of-function mutations in the phosphate regulating gene with homologies to endopeptidases on the X-chromosome (PHEX) have been causally associated with X-linked hypophosphatemic rickets (XLHR). The early diagnosis of XLHR in infants is challenging when it is based solely on clinical features and biochemical findings. We report a 7-month-old boy with a family history of hypophosphatemic rickets., who demonstrated early clinical evidence of rickets, although serial biochemical findings could not definitively confirm rickets. A sequencing assay targeting the PHEX gene was first performed on the mother's DNA to screen for mutations in the 5'UTR, 22 coding exons, and the exon-intron junctions. Targeted mutation analysis and mRNA studies were subsequently performed on the boys' DNA to investigate the pathogenicity of the identified mutation. Genetic screening of the PHEX gene revealed a novel mutation, c.1080-2A>C, at the splice acceptor site in intron 9. The detection of an aberrant mRNA transcript with skipped (loss of) exon 10 establishes its pathogenicity and confirms the diagnosis of XLHR in this infant. Genetic testing of the PHEX gene resulted in early diagnosis of XLHR, thus enabling initiation of therapy and prevention of progressive rachitic changes in the infant.

摘要

与X染色体上内肽酶具有同源性的磷酸盐调节基因(PHEX)的功能丧失突变已被证实与X连锁低磷性佝偻病(XLHR)存在因果关系。仅基于临床特征和生化检查结果对婴儿期XLHR进行早期诊断具有挑战性。我们报告了一名7个月大的男婴,其家族中有低磷性佝偻病病史,该患儿虽有佝偻病的早期临床证据,但系列生化检查结果未能明确确诊佝偻病。首先对母亲的DNA进行靶向PHEX基因的测序检测,以筛查5'非翻译区、22个编码外显子及外显子 - 内含子连接处的突变。随后对患儿的DNA进行靶向突变分析和mRNA研究,以调查所鉴定突变的致病性。PHEX基因的基因筛查发现内含子9的剪接受体位点存在一个新的突变c.1080 - 2A>C。检测到外显子10跳跃(缺失)的异常mRNA转录本,证实了该突变的致病性,并确诊该婴儿患有XLHR。对PHEX基因进行基因检测实现了XLHR的早期诊断,从而能够在婴儿期开始治疗并预防进行性佝偻病改变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7884/4748509/57a64768eb36/10.1177_2324709615598167-fig1.jpg

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