Congenital diaphragmatic eventration with pulmonary dysplasia in Frasier syndrome due to a WT1 mutation of c.1432+5(IVS9)G>A

Eur J Med Genet. 2022 Dec;65(12):104655. doi: 10.1016/j.ejmg.2022.104655. Epub 2022 Oct 28.

Abstract

WT1 disorder is caused by a heterozygous variant in the gene WT1 (Wilms' tumor suppressor gene 1), and is clinically diagnosed as Denys-Drash, Meacham, or Frasier syndrome, on a phenotypic continuum that presents as abnormalities of the urogenital system and gonads. Rarely, manifestations appear in the lung, especially in Frasier syndrome. Here we describe the first noted case of congenital diaphragmatic eventration with pulmonary dysplasia in a child with Frasier syndrome. A c.1432+5G > A mutation in intron 9 of WT1 was found. We also summarize pulmonary diseases associated with WT1 mutations in WT1 disorder.

Keywords: Congenital diaphragmatic eventration; Frasier syndrome; WT1 disorder.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Diaphragmatic Eventration*
  • Frasier Syndrome* / genetics
  • Humans
  • Lung
  • Mutation
  • WT1 Proteins / genetics

Substances

  • WT1 protein, human
  • WT1 Proteins