Literature DB >> 35792504

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

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.
© 2022 Wiley Periodicals LLC.

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


  1 in total

Review 1.  Comprehensive genetic testing approaches as the basis for personalized management of growth disturbances: current status and perspectives.

Authors:  Danielle Christine Maria van der Kaay; Anne Rochtus; Gerhard Binder; Ingo Kurth; Dirk Prawitt; Irène Netchine; Gudmundur Johannsson; Anita C S Hokken-Koelega; Miriam Elbracht; Thomas Eggermann
Journal:  Endocr Connect       Date:  2022-10-10       Impact factor: 3.221

  1 in total

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