Autosomal recessive truncating MAB21L1 mutation associated with a syndromic scrotal agenesis

Clin Genet. 2017 Feb;91(2):333-338. doi: 10.1111/cge.12794. Epub 2016 Jun 5.

Abstract

We report on a boy with a rare malformative association of scrotum agenesis, ophthalmological anomalies, cerebellar malformation, facial dysmorphism and global development delay. The reported patient was carrying a homozygous frameshift in MAB21L1 detected by whole-exome sequencing, considered as the most likely disease-causing variant. Mab21l1 knockout mice present a strikingly similar malformative association of ophthalmological malformations of the anterior chamber and preputial glands hypoplasia. We hypothesize that MAB21L1 haploinsufficiency cause a previously undescribed syndrome with scrotal agenesis, ophthalmological anomalies, facial dysmorphism and gross psychomotor delay as remarkable hallmarks. Four cases from the literature were reported with features suggestive of a similar and recognizable clinical entity. We hypothesize that MAB21L1 should be the culprit gene in these patients.

Keywords: MAB21L1; intellectual disability; scrotal agenesis; whole-exome sequencing.

Publication types

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

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / pathology
  • Animals
  • Child
  • Developmental Disabilities / genetics*
  • Developmental Disabilities / pathology
  • Exome / genetics
  • Frameshift Mutation / genetics
  • Homeodomain Proteins / genetics*
  • Homozygote
  • Humans
  • Intellectual Disability / genetics*
  • Intellectual Disability / pathology
  • Male
  • Mice
  • Mutation
  • Phenotype
  • Scrotum / pathology

Substances

  • Homeodomain Proteins
  • MAB21L1 protein, human