Prenatal isolated clubfoot increases the risk for clinically significant exome sequencing results

Prenat Diagn. 2022 Dec;42(13):1622-1626. doi: 10.1002/pd.6259. Epub 2022 Nov 7.

Abstract

Objective: To examine the diagnostic yield of exome sequencing (ES) in singleton pregnancies with isolated fetal clubfoot.

Methods: Clinical data from singleton pregnancies with a sonographic diagnosis of isolated clubfoot and ES results between 2018 and 2021 were retrospectively obtained from a single referral medical center. The recorded data include maternal age, gestational age at sonographic diagnosis, the indication for genetic testing, ES results, and pregnancy outcomes.

Results: During the study period, 38 fetuses were prenatally diagnosed with isolated clubfoot by ultrasound and underwent ES after the copy number variant analysis was non-diagnostic. Through the trio-ES analysis, pathogenic or likely pathogenic variants were detected in 4 of 38 (10.5%) with the following genes: BRPF1, ANKRD17, FLNA, and KIF1A. All are de novo with three of autosomal dominant inheritance and one of X-linked recessive inheritance.

Conclusion: Sonographic diagnosis of clubfoot, even isolated, increases the risk for monogenic syndromes. Exome sequencing should be an option for genetic investigation for such pregnancies.

MeSH terms

  • Adaptor Proteins, Signal Transducing
  • Clubfoot* / diagnostic imaging
  • Clubfoot* / genetics
  • DNA-Binding Proteins
  • Exome Sequencing
  • Female
  • Fetus / diagnostic imaging
  • Humans
  • Kinesins
  • Pregnancy
  • Prenatal Diagnosis / methods
  • RNA-Binding Proteins
  • Retrospective Studies
  • Ultrasonography, Prenatal

Substances

  • BRPF1 protein, human
  • DNA-Binding Proteins
  • Adaptor Proteins, Signal Transducing
  • ANKRD17 protein, human
  • RNA-Binding Proteins
  • KIF1A protein, human
  • Kinesins