StayCurrentMD · Early-Onset Pectus Excavatum Is More Likely to Be Part of a Genetic Variation
Article1 min read·Published May 2023Older

Early-Onset Pectus Excavatum Is More Likely to Be Part of a Genetic Variation

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Article · May 2023 · 1 min read

In brief

In brief

This study demonstrates that pectus excavatum presenting before age 11 has a 44% association with underlying genetic conditions, including syndromic disorders, chromosomal abnormalities, and connective tissue diseases. The findings support routine genetic counseling referral for early-onset cases to identify treatable conditions and inform family planning.

  • 44% of children with PE onset before age 11 had identifiable pathogenic genetic variations, suggesting strong genetic component in early cases
  • Early-onset PE was associated with syndromic disorders (Catel-Manzke, Noonan), chromosomal abnormalities, and connective tissue diseases
  • PE presenting before age 11 warrants genetic counseling referral, unlike adolescent-onset cases which are less likely genetic
  • Only one-fifth of PE cases present in first decade, making early-onset a distinct clinical entity requiring different evaluation approach
  • Molecular analysis identified diverse genetic etiologies including 16p13.11 and 22q11.21 microduplications and BICD2 pathogenic variants

Written by the GCMD Library team from the article.

Background Potential underlying genetic variations of pectus excavatum (PE) are quite rare. Only one-fifth of PE cases are identified in the first decade of life and thus are of congenital origin. The objective of this study is to test if early-onset PE is more likely to be part of genetic variations than PE that becomes apparent during puberty or adolescence. Materials and Methods Children younger than 11 years who presented with PE to the outpatient clinic of the Department of Pediatric Surgery at our center between 2014 and 2020 were screened by two clinical geneticists separately. Molecular analysis was performed based on the differential diagnosis. Data of all young PE patients who already had been referred for genetic counseling were analyzed retrospectively. Results Pathogenic genetic variations were found in 8 of the 18 participants (44%): 3 syndromic disorders (Catel–Manzke syndrome and two Noonan syndromes), 3 chromosomal disorders (16p13.11 microduplication syndrome, 22q11.21 microduplication syndrome, and genetic gain at 1q44), 1 connective tissue disease (Loeys–Dietz syndrome), and 1 neuromuscular disorder (pathogenic variation in BICD2 gene). Conclusion Early-onset PE is more likely to be part of genetic variations than PE that becomes apparent during puberty or adolescence. Referral for genetic counseling should therefore be considered. Trial Registration: NCT05443113

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