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生长激素不敏感症(拉龙综合征):一个新家族的报告及巴西患者综述

Growth Hormone insensitivity (Laron syndrome): Report of a new family and review of Brazilian patients.

作者信息

Villela Thais R, Freire Bruna L, Braga Nathalia T P, Arantes Rodrigo R, Funari Mariana F A, Alexander Jorge A L, Silva Ivani N

机构信息

Universidade Federal de Minas Gerais, Faculdade de Medicina, Hospital das Clínicas, Divisão de Endocrinologia Infantil e do Adolescente, Belo Horizonte, MG, Brazil.

Universidade de São Paulo, Hospital das Clínicas da Faculdade de Medicina, Unidade de Endocrinologia Genética, São Paulo, SP, Brazil.

出版信息

Genet Mol Biol. 2020 Jan 20;42(4):e20180197. doi: 10.1590/1678-4685-GMB-2018-0197. eCollection 2020.

Abstract

Laron's syndrome (LS) is a rare genetic disorder characterized by insensitivity to growth hormone (GH). Up to the present time, over 70 mutations of GH receptor (GHR) gene have been identified leading to GH/insulin-like growth factor type 1 (IGF1) signaling pathway defect. The number of LS patients worldwide is unknown, as many are probably undiagnosed. We report two sibs from a consanguineous family from Minas Gerais, southeastern Brazil. The parents have three children. The older, a 4-years-old girl was 80.2 cm tall (-5.7 SDS height/age), and the youngest sister, aged 3 years, was 73.2 cm tall (-5.82 SDS height/age). Their clinical and biochemical features are typical of LS patients, such as high serum level of GH and low IGF1 concentrations. A homozygous c.1A>T nucleotide substitution in GHR exon 2 in the probands' samples was identified. Their parents and healthy sister are heterozygous for the same variant that abolishes the translation initiation codon of GHR. This mutation has not been reported in Brazilian patients and was previously associated with an LS phenotype in a single 29-year-old Spanish man. In addition to this case report, we summarize the main characteristics and molecular data of the 21 LS Brazilian patients who have been published to date.

摘要

拉龙综合征(LS)是一种罕见的遗传性疾病,其特征是对生长激素(GH)不敏感。截至目前,已鉴定出超过70种生长激素受体(GHR)基因突变,导致GH/胰岛素样生长因子1型(IGF1)信号通路缺陷。全球拉龙综合征患者的数量尚不清楚,因为许多患者可能未被诊断出来。我们报告了来自巴西东南部米纳斯吉拉斯州一个近亲家庭的两名同胞。父母育有三个孩子。年龄较大的是一名4岁女孩,身高80.2厘米(身高/年龄标准差为-5.7),最小的妹妹3岁,身高73.2厘米(身高/年龄标准差为-5.82)。她们的临床和生化特征是拉龙综合征患者的典型特征,如血清GH水平高而IGF1浓度低。在先证者样本中,在GHR外显子2中鉴定出纯合的c.1A>T核苷酸替代。她们的父母和健康的妹妹对于同一变异是杂合的,该变异消除了GHR的翻译起始密码子。这种突变在巴西患者中尚未见报道,之前仅在一名29岁的西班牙男性中与拉龙综合征表型相关联。除了本病例报告外,我们还总结了迄今已发表的21例巴西拉龙综合征患者的主要特征和分子数据。

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Epilogue: The future of Laron syndrome - The need for changes.后记:拉龙综合征的未来——变革的必要性。
Growth Horm IGF Res. 2016 Jun;28:79-80. doi: 10.1016/j.ghir.2015.07.007. Epub 2015 Jul 30.

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