Suppr超能文献

中国低磷性佝偻病/骨软化症患者中PHEX基因两个新突变的鉴定。

Identification of two novel mutations in the PHEX gene in Chinese patients with hypophosphatemic rickets/osteomalacia.

作者信息

Yue Hua, Yu Jin-bo, He Jin-wei, Zhang Zeng, Fu Wen-zhen, Zhang Hao, Wang Chun, Hu Wei-wei, Gu Jie-mei, Hu Yun-qiu, Li Miao, Liu Yu-juan, Zhang Zhen-Lin

机构信息

Department of Osteoporosis, Metabolic Bone Disease and Genetic Research Unit, Shanghai Jiao Tong University Affiliated Sixth People's Hospital, Shanghai, P. R. China.

Department of pediatrics, Shanghai Jiao Tong University Affiliated Sixth People's Hospital, Shanghai, P. R. China.

出版信息

PLoS One. 2014 May 16;9(5):e97830. doi: 10.1371/journal.pone.0097830. eCollection 2014.

Abstract

OBJECTIVE

X-linked dominant hypophosphatemia (XLH) is the most prevalent form of inherited rickets/osteomalacia in humans. The aim of this study was to identify PHEX gene mutations and describe the clinical features observed in 6 unrelated Chinese families and 3 sporadic patients with hypophosphatemic rickets/osteomalacia.

METHODS

For this study, 45 individuals from 9 unrelated families of Chinese Han ethnicity (including 16 patients and 29 normal phenotype subjects), and 250 healthy donors were recruited. All 22 exons and exon-intron boundaries of the PHEX gene were amplified by polymerase chain reaction (PCR) and directly sequenced.

RESULTS

The PHEX mutations were detected in 6 familial and 3 sporadic hypophosphatemic rickets/osteomalacia. Altogether, 2 novel mutations were detected: 1 missense mutation c.1183G>C in exon 11, resulting in p.Gly395Arg and 1 missense mutation c.1751A>C in exon 17, resulting in p.His584Pro. No mutations were found in the 250 healthy controls.

CONCLUSIONS

Our study increases knowledge of the PHEX gene mutation types and clinical phenotypes found in Chinese patients with XLH, which is important for understanding the genetic basis of XLH. The molecular diagnosis of a PHEX genetic mutation is of great importance for confirming the clinical diagnosis of XLH, conducting genetic counseling, and facilitating prenatal intervention, especially in the case of sporadic patients.

摘要

目的

X连锁显性低磷血症(XLH)是人类遗传性佝偻病/骨软化症最常见的形式。本研究旨在鉴定PHEX基因突变,并描述在6个无血缘关系的中国家庭和3例散发性低磷血症佝偻病/骨软化症患者中观察到的临床特征。

方法

本研究招募了9个无血缘关系的中国汉族家庭的45名个体(包括16例患者和29例表型正常的受试者)以及250名健康供体。通过聚合酶链反应(PCR)扩增PHEX基因的所有22个外显子和外显子-内含子边界,并直接进行测序。

结果

在6例家族性和3例散发性低磷血症佝偻病/骨软化症患者中检测到PHEX基因突变。共检测到2个新突变:外显子11中的1个错义突变c.1183G>C,导致p.Gly395Arg;外显子17中的1个错义突变c.1751A>C,导致p.His584Pro。在250名健康对照中未发现突变。

结论

我们的研究增加了对中国XLH患者中PHEX基因突变类型和临床表型的认识,这对于理解XLH的遗传基础很重要。PHEX基因突变的分子诊断对于确诊XLH的临床诊断、进行遗传咨询以及促进产前干预非常重要,尤其是对于散发性患者。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd86/4024000/ab4cef0d4abb/pone.0097830.g001.jpg

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验