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胫骨发育不全、多指畸形和第一指三节指骨畸形患者中的ZRS 406A>G突变

ZRS 406A>G mutation in patients with tibial hypoplasia, polydactyly and triphalangeal first fingers.

作者信息

Norbnop Phatchara, Srichomthong Chalurmpon, Suphapeetiporn Kanya, Shotelersuk Vorasuk

机构信息

1] Doctor of Philosophy Program in Medical Sciences, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand [2] Center of Excellence for Medical Genetics, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand.

1] Center of Excellence for Medical Genetics, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand [2] Excellence Center for Medical Genetics, King Chulalongkorn Memorial Hospital, The Thai Red Cross Society, Bangkok, Thailand.

出版信息

J Hum Genet. 2014 Aug;59(8):467-70. doi: 10.1038/jhg.2014.50. Epub 2014 Jun 26.

Abstract

Werner mesomelic syndrome (WMS), an autosomal dominant disorder characterized by hypoplastic tibiae, triphalangeal thumbs and polydactyly, is caused by a specific point mutation at the position 404 in zone of polarizing activity regulatory sequence (ZRS). Here we identified two additional families with WMS. All three patients in three generations of Family 1 were found to harbor the same heterozygous 406A>G mutation in ZRS. The fourth patient from Family 2 was a sporadic case with the known 404 point mutation. The novel 406A>G mutation expands mutational spectrum in ZRS causing WMS, provides evidence for a functionally important nucleotide position 406 of ZRS in humans and has implications for genetic counseling.

摘要

维尔纳中肢发育不全综合征(WMS)是一种常染色体显性疾病,其特征为胫骨发育不全、拇指三节指骨和多指畸形,由极化活性调节序列(ZRS)第404位的特定点突变引起。在此,我们鉴定出另外两个患有WMS的家系。家系1三代中的所有三名患者均被发现ZRS中存在相同的杂合406A>G突变。家系2的第四名患者为散发病例,携带已知的404点突变。新发现的406A>G突变扩展了导致WMS的ZRS突变谱,为人类ZRS中功能重要的第406位核苷酸提供了证据,并对遗传咨询具有重要意义。

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