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极低出生体重儿矮小症患儿中遗传/表观遗传疾病的高频率。

High frequency of genetic/epigenetic disorders in short stature children born with very low birth weight.

机构信息

Unidade de Endocrinologia Genética (LIM25), Hospital das Clinicas da Faculdade de Medicina da Universidade de São Paulo (FMUSP), São Paulo, Consolacao, Brazil.

Unidade de Endocrinologia do Desenvolvimento, Laboratório de Hormônios e Genética Molecular LIM42, Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo (FMUSP), São Paulo, Consolacao, Brazil.

出版信息

Am J Med Genet A. 2022 Sep;188(9):2599-2604. doi: 10.1002/ajmg.a.62892. Epub 2022 Jul 6.

DOI:10.1002/ajmg.a.62892
PMID:35792504
Abstract

Most infants born with very low birth weight (VLBW, birth weight < 1500 g) show spontaneous catch-up growth in postnatal life. The reasons for the absence of catch-up growth are not entirely understood. We performed a comprehensive investigation of 52 children born with VLBW. Ten children had a history of an external cause that explained the VLBW and five refused genetic evaluation. Twenty-three cases were initially evaluated by a candidate gene approach. Patients with a negative result in the candidate gene approach (n = 14) or without clinical suspicion (n = 14) were assessed by chromosome microarray analysis (CMA) and/or whole-exome sequencing (WES). A genetic condition was identified in 19 of 37 (51.4%) patients without an external cause, nine by candidate gene approach, and 10 by a genomic approach (CMA/WES). Silver-Russell syndrome was the most frequent diagnosis (n = 5) and the remaining patients were diagnosed with other rare monogenic conditions. Almost all patients with a positive genetic diagnosis exhibited syndromic features (94.4%). However, microcephaly, neurodevelopmental disorders, major malformation, or facial dysmorphism were also frequently observed in children with an external cause. In conclusion, a significant proportion of children born with VLBW with persistent short stature have a genetic/epigenetic condition.

摘要

大多数极低出生体重儿(VLBW,出生体重<1500 克)在出生后会出现自发性追赶生长。导致追赶生长缺失的原因尚不完全清楚。我们对 52 名 VLBW 出生的儿童进行了全面调查。10 名儿童有导致 VLBW 的外部原因史,5 名儿童拒绝进行基因评估。23 例最初采用候选基因方法进行评估。候选基因方法阴性结果的患者(n=14)或无临床可疑的患者(n=14)进行染色体微阵列分析(CMA)和/或全外显子组测序(WES)。在无外部原因的 37 名患者中,有 19 名(51.4%)确定了遗传状况,其中 9 名通过候选基因方法,10 名通过基因组方法(CMA/WES)。银-罗素综合征是最常见的诊断(n=5),其余患者被诊断为其他罕见的单基因疾病。几乎所有阳性基因诊断的患者都表现出综合征特征(94.4%)。然而,有外部原因的儿童也经常出现小头畸形、神经发育障碍、重大畸形或面部畸形。总之,持续性身材矮小的 VLBW 出生儿中有相当比例存在遗传/表观遗传疾病。

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Imprinting disorders.印迹缺陷
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