Suppr超能文献

家族性身材矮小的单基因病因。

Monogenic causes of familial short stature.

作者信息

Plachy Lukas, Dusatkova Petra, Amaratunga Shenali Anne, Neuman Vit, Sumnik Zdenek, Lebl Jan, Pruhova Stepanka

机构信息

Department of Pediatrics, 2nd Faculty of Medicine, Charles University in Prague and Motol University Hospital, Prague, Czechia.

出版信息

Front Endocrinol (Lausanne). 2024 Dec 19;15:1506323. doi: 10.3389/fendo.2024.1506323. eCollection 2024.

Abstract

Genetic factors play a crucial role in determining human height. Short stature commonly affects multiple family members and therefore, familial short stature (FSS) represents a significant proportion of growth disorders. Traditionally, FSS was considered a benign polygenic condition representing a subcategory of idiopathic short stature (ISS). However, advancements in genetic research have revealed that FSS can also be monogenic, inherited in an autosomal dominant manner and can result from different mechanisms including primary growth plate disorders, growth hormone deficiency/insensitivity or by the disruption of fundamental intracellular pathways. These discoveries have highlighted a broader phenotypic spectrum for monogenic forms of short stature, which may exhibit mild manifestations indistinguishable from ISS. Given the overlapping features and the difficulty in differentiating polygenic from monogenic FSS without genetic testing, some researchers redefine FSS as a descriptive term that encompasses any familial occurrence of short stature, regardless of the underlying cause. This shift emphasizes the complexity of diagnosing and managing short stature within families, reflecting the diverse genetic landscape that influences human growth.

摘要

遗传因素在决定人类身高方面起着至关重要的作用。身材矮小通常会影响多个家庭成员,因此,家族性身材矮小(FSS)在生长障碍中占相当大的比例。传统上,FSS被认为是一种良性多基因疾病,是特发性身材矮小(ISS)的一个子类别。然而,基因研究的进展表明,FSS也可能是单基因的,以常染色体显性方式遗传,并且可能由不同机制导致,包括原发性生长板疾病、生长激素缺乏/不敏感或基本细胞内途径的破坏。这些发现凸显了单基因形式身材矮小更广泛的表型谱,其可能表现出与ISS难以区分的轻微症状。鉴于其重叠特征以及在未经基因检测的情况下难以区分多基因FSS和单基因FSS,一些研究人员将FSS重新定义为一个描述性术语,涵盖任何家族性身材矮小情况,无论其潜在原因如何。这一转变强调了在家庭中诊断和管理身材矮小的复杂性,反映了影响人类生长的多样基因格局。

相似文献

1
Monogenic causes of familial short stature.家族性身材矮小的单基因病因。
Front Endocrinol (Lausanne). 2024 Dec 19;15:1506323. doi: 10.3389/fendo.2024.1506323. eCollection 2024.
3
Genetic Architecture Associated With Familial Short Stature.与家族性身材矮小相关的遗传结构。
J Clin Endocrinol Metab. 2020 Jun 1;105(6). doi: 10.1210/clinem/dgaa131.
5
Genetic short stature.遗传性身材矮小
Growth Horm IGF Res. 2018 Feb;38:29-33. doi: 10.1016/j.ghir.2017.12.003. Epub 2017 Dec 6.
6
gene variants in familial short stature: a single-center study.家族性矮小症中的基因变异:一项单中心研究。
J Pediatr Endocrinol Metab. 2021 Sep 27;35(2):185-190. doi: 10.1515/jpem-2021-0332. Print 2022 Feb 23.

本文引用的文献

8
Short stature related to Growth Hormone Insensitivity (GHI) in childhood.儿童时期生长激素不敏感(GHI)导致的身材矮小。
Front Endocrinol (Lausanne). 2023 Mar 15;14:1141039. doi: 10.3389/fendo.2023.1141039. eCollection 2023.
9
Clinical Indications for Growth Hormone Therapy.生长激素治疗的临床适应证。
Adv Pediatr. 2022 Aug;69(1):203-217. doi: 10.1016/j.yapd.2022.03.005. Epub 2022 Jun 17.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验