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