Mowat-Wilson syndrome in a fetus with antenatal diagnosis of short corpus callosum: advocacy for standard autopsy

Eur J Med Genet. 2013 Jun;56(6):297-300. doi: 10.1016/j.ejmg.2013.03.003. Epub 2013 Mar 21.

Abstract

Mowat-Wilson syndrome (MWS) is a genetic disease caused by heterozygous mutations or deletions of the ZEB2 gene rarely diagnosed prenatally and with little fetal description reported. It is mainly characterized by moderate-to-severe intellectual disability, epilepsy, facial dysmorphism and various malformations including Hirschsprung disease and corpus callosum anomalies. Here we report a fetal case of MWS well described, suspected at standard autopsy. The association of a corpus callosum hypoplasia with a histological Hirschsprung disease and a typical facial gestalt allowed the guiding of genetic testing. Classical fetopathological examination still keeps indications in cases of syndromic association in the era of virtual autopsy.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / diagnosis
  • Abnormalities, Multiple / genetics
  • Adult
  • Agenesis of Corpus Callosum / diagnosis*
  • Agenesis of Corpus Callosum / genetics
  • Autopsy
  • Corpus Callosum / pathology
  • Facies
  • Female
  • Heterozygote
  • Hirschsprung Disease / diagnosis*
  • Hirschsprung Disease / genetics
  • Homeodomain Proteins / genetics
  • Humans
  • Infant, Newborn
  • Intellectual Disability / diagnosis*
  • Intellectual Disability / genetics
  • Magnetic Resonance Imaging
  • Male
  • Microcephaly / diagnosis*
  • Microcephaly / genetics
  • Mutation
  • Phenotype
  • Pregnancy
  • Prenatal Diagnosis
  • Repressor Proteins / genetics
  • Zinc Finger E-box Binding Homeobox 2

Substances

  • Homeodomain Proteins
  • Repressor Proteins
  • ZEB2 protein, human
  • Zinc Finger E-box Binding Homeobox 2

Supplementary concepts

  • Mowat-Wilson syndrome