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Zhongguo Dang Dai Er Ke Za Zhi. 2017 Nov;19(11):1159-1164. doi: 10.7499/j.issn.1008-8830.2017.11.007.
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A sporadic case of male-limited precocious puberty has the same constitutively activating point mutation in luteinizing hormone/choriogonadotropin receptor gene as familial cases.一例散发性男性性早熟病例与家族性病例在促黄体生成素/绒毛膜促性腺激素受体基因上具有相同的组成性激活点突变。
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本文引用的文献

1
[Analysis of a family affected with familial male-limited precocious puberty due to a Ala568Val mutation in LHCGR gene].[因LHCGR基因Ala568Val突变导致的家族性男性限性性早熟家系分析]
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2012 Dec;29(6):631-4. doi: 10.3760/cma.j.issn.1003-9406.2012.06.002.
2
[A report of familial male-limited precocious puberty caused by a germ-line heterozygous mutation (M398T) in luteinizing hormone receptor gene].[关于由促黄体生成素受体基因种系杂合突变(M398T)引起的家族性男性限性性早熟的报告]
Zhonghua Nei Ke Za Zhi. 2010 Dec;49(12):1024-7.
3
Novel C617Y mutation in the 7th transmembrane segment of luteinizing hormone/choriogonadotropin receptor in a Japanese boy with peripheral precocious puberty.日本一名外周性性早熟男孩的黄体生成素/绒毛膜促性腺激素受体第 7 跨膜区的新型 C617Y 突变。
Endocr J. 2010;57(12):1055-60. doi: 10.1507/endocrj.k10e-227. Epub 2010 Nov 2.
4
An update of the pathophysiology of human gonadotrophin subunit and receptor gene mutations and polymorphisms.人类促性腺激素亚基及受体基因突变与多态性病理生理学的最新进展
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Testicular seminoma in a patient with a constitutively activating mutation of the luteinizing hormone/chorionic gonadotropin receptor.一名患有促黄体生成素/绒毛膜促性腺激素受体组成性激活突变患者的睾丸精原细胞瘤。
Eur J Endocrinol. 1998 Jul;139(1):101-6. doi: 10.1530/eje.0.1390101.
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Cosegregation of missense mutations of the luteinizing hormone receptor gene with familial male-limited precocious puberty.
Hum Mol Genet. 1993 Nov;2(11):1779-83. doi: 10.1093/hmg/2.11.1779.
7
Genetic heterogeneity of constitutively activating mutations of the human luteinizing hormone receptor in familial male-limited precocious puberty.家族性男性性早熟中人类促黄体生成素受体组成性激活突变的遗传异质性。
Proc Natl Acad Sci U S A. 1995 Mar 14;92(6):1906-10. doi: 10.1073/pnas.92.6.1906.
8
A constitutively activating mutation of the luteinizing hormone receptor in familial male precocious puberty.
Nature. 1993 Oct 14;365(6447):652-4. doi: 10.1038/365652a0.
9
A sporadic case of male-limited precocious puberty has the same constitutively activating point mutation in luteinizing hormone/choriogonadotropin receptor gene as familial cases.一例散发性男性性早熟病例与家族性病例在促黄体生成素/绒毛膜促性腺激素受体基因上具有相同的组成性激活点突变。
J Clin Endocrinol Metab. 1994 Dec;79(6):1818-23. doi: 10.1210/jcem.79.6.7527413.
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[由于LHCGR基因Asp578His突变导致的家族性男性局限性性早熟:一名婴儿的临床特征及基因分析]

[Familial male-limited precocious puberty due to Asp578His mutations in the LHCGR gene: clinical characteristics and gene analysis in an infant].

作者信息

Wang Min, Li Min, Liu Yue-Sheng, Lei Si-Min, Xiao Yan-Feng

机构信息

Department of Pediatrics, The Second Affiliated Hospital of Xi'an Jiaotong University, Xi'an 710061, China.

出版信息

Zhongguo Dang Dai Er Ke Za Zhi. 2017 Nov;19(11):1159-1164. doi: 10.7499/j.issn.1008-8830.2017.11.007.

DOI:10.7499/j.issn.1008-8830.2017.11.007
PMID:29132462
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7389323/
Abstract

The aim of the study was to provide a descriptive analysis of familial male-limited precocious puberty (FMPP), which is a rare inherited disease caused by heterozygous constitutively activating mutations of the luteinizing hormone/choriogonadotropin receptor gene (LHCGR). The patient was a ten-month-old boy, presenting with penile enlargement, pubic hair formation, and spontaneous erections. Based on the clinical manifestations and laboratory data, including sexual characteristics, serum testosterone levels, GnRH stimulation test, and bone age, this boy was diagnosed with peripheral precocious puberty. Subsequently the precocious puberty-related genes were analyzed by direct DNA sequencing of amplified PCR products from the patient and his parents. Genetic analysis revealed a novel heterozygous missense mutation c.1732G>C (Asp578His) of the LHCGR gene exon11 in the patient, which had never been reported. His parents had no mutations. After combined treatment with aromatase inhibitor letrozole and anti-androgen spironolactone for six months, the patient's symptoms were controlled. The findings in this study expand the mutation spectrum of the LHCGR gene, and provide molecular evidence for the etiologic diagnosis as well as for the genetic counseling and prenatal diagnosis in the family.

摘要

本研究的目的是对家族性男性限性性早熟(FMPP)进行描述性分析,这是一种由促黄体生成素/绒毛膜促性腺激素受体基因(LHCGR)的杂合性组成型激活突变引起的罕见遗传病。该患者为一名10个月大的男孩,表现为阴茎增大、阴毛形成和自发性勃起。根据临床表现和实验室数据,包括性征、血清睾酮水平、GnRH刺激试验和骨龄,该男孩被诊断为外周性早熟。随后,通过对患者及其父母的扩增PCR产物进行直接DNA测序,分析了与性早熟相关的基因。基因分析发现患者LHCGR基因外显子11存在一个新的杂合错义突变c.1732G>C(Asp578His),此前未见报道。其父母无突变。在联合使用芳香化酶抑制剂来曲唑和抗雄激素螺内酯治疗6个月后,患者症状得到控制。本研究结果扩展了LHCGR基因的突变谱,为该家族的病因诊断、遗传咨询和产前诊断提供了分子证据。