| Literature DB >> 35792504 |
Bruna Lucheze Freire1,2, Thais Kataoka Homma1,2, Antônio Marcondes Lerario3, Go Hun Seo4, Heonjong Han4, Mariana Ferreira de Assis Funari1,2, Nathalia Lisboa Gomes5, Carla Rosemberg6, Ana Cristina Victorino Krepischi6, Gabriela de Andrade Vasques1,2, Alexsandra Christianne Malaquias1,7, Alexander Augusto de Lima Jorge1,2.
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.Entities:
Keywords: genetics; next-generation sequencing; short stature; very low birth weight
Mesh:
Year: 2022 PMID: 35792504 DOI: 10.1002/ajmg.a.62892
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.578