Whole-exome sequencing identifies a de novo PDE3A variant causing autosomal dominant hypertension with brachydactyly type E syndrome: a case report

BMC Med Genet. 2020 Jul 6;21(1):144. doi: 10.1186/s12881-020-01077-z.

Abstract

Background: Autosomal dominant hypertension with brachydactyly type E syndrome caused by pathogenic variants in the PDE3A gene was first reported in 2015. To date, there are only a few reports of this kind of syndrome. Other patients still lack a genetic diagnosis.

Case presentation: Whole-exome sequencing was performed in an 18-year-old female proband with a clinical diagnosis of hypertension with brachydactyly syndrome. Quantitative real-time PCR was used to identify pathogenic copy number variations (CNVs). After bioinformatics analysis and healthy control database filtering, we revealed a heterozygous missense PDE3A variant (c.1346G > A, p.Gly449Asp). The variant was absent in the ExAC database and located in a highly evolutionarily conserved cluster of reported PDE3A pathogenic variants. Importantly, this variant was predicted to affect protein function by both SIFT (score = 0) and PolyPhen-2 (score = 1). After Sanger sequencing, the variant was determined to be absent in the healthy parents of the proband as well as 800 ethnically and geographically matched healthy controls.

Conclusion: We present a report linking a de novo PDE3A variant to autosomal dominant hypertension with brachydactyly type E syndrome.

Keywords: Brachydactyly; Genetic diagnosis; Hypertension; Hypertension with brachydactyly type E syndrome; Whole-exome sequencing.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Brachydactyly / diagnostic imaging
  • Brachydactyly / genetics*
  • Cyclic Nucleotide Phosphodiesterases, Type 3 / genetics*
  • Exome Sequencing*
  • Exons / genetics
  • Female
  • Genes, Dominant*
  • Humans
  • Hypertension / congenital*
  • Hypertension / diagnostic imaging
  • Hypertension / genetics
  • Male
  • Mutation / genetics*
  • Pedigree
  • Syndrome

Substances

  • Cyclic Nucleotide Phosphodiesterases, Type 3
  • PDE3A protein, human

Supplementary concepts

  • Brachydactyly with hypertension