Biallelic c.1263dupC in DOK7 results in fetal akinesia deformation sequence

Am J Med Genet A. 2020 Apr;182(4):804-807. doi: 10.1002/ajmg.a.61473. Epub 2019 Dec 27.

Abstract

Fetal akinesia deformation sequence (FADS) is a clinically and genetically heterogeneous condition. Pathogenic variants in DOK7 are known to cause myasthenic syndrome, congenital, 10 (MIM#254300) and, rarely (reported in a single family) lethal FADS. Herein, we describe a biallelic variant c.1263dupC in DOK7, known to cause congenital myasthenic syndrome 10, causing lethal FADS in a consanguineous family. The present report illustrates wide phenotypic variability caused by biallelic pathogenic variants in DOK7. We also describe the second family with FADS due to pathogenic variants in DOK7.

Keywords: DOK7; congenital myasthenic syndrome 10; fetal akinesia deformation sequence.

Publication types

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

MeSH terms

  • Abnormalities, Multiple / genetics
  • Abnormalities, Multiple / pathology*
  • Arthrogryposis / genetics
  • Arthrogryposis / pathology*
  • Developmental Disabilities / genetics
  • Developmental Disabilities / pathology*
  • Female
  • Fetus / metabolism
  • Fetus / pathology*
  • Gene Frequency
  • Gestational Age
  • Humans
  • Muscle Proteins / genetics*
  • Mutation*
  • Pedigree

Substances

  • DOK7 protein, human
  • Muscle Proteins

Supplementary concepts

  • Pena Shokeir syndrome, type 1