Severe early onset retinitis pigmentosa in a Moroccan patient with Heimler syndrome due to novel homozygous mutation of PEX1 gene

Eur J Med Genet. 2016 Oct;59(10):507-11. doi: 10.1016/j.ejmg.2016.09.004. Epub 2016 Sep 12.

Abstract

Heimler syndrome (HS) is a rare recessive disorder characterized by sensorineural hearing loss (SNHL), amelogenesis imperfecta, nail abnormalities, and occasional or late-onset retinal pigmentation. It is the mildest form known to date of peroxisome biogenesis disorder caused by hypomorphic mutations of PEX1 and PEX6 genes. We report on a second Moroccan family with Heimler syndrome with early onset, severe visual impairment and important phenotypic overlap with Usher syndrome. The patient carried a novel homozygous missense variant c.3140T > C (p.Leu1047Pro) of PEX1 gene. As standard biochemical screening of blood for evidence of a peroxisomal disorder did not provide a diagnosis in the individuals with HS, patients with SNHL and retinal pigmentation should have mutation analysis of PEX1 and PEX6 genes.

Keywords: Heimler syndrome; Moroccan; PEX1; Retinitis pigmentosa; Variant.

MeSH terms

  • ATPases Associated with Diverse Cellular Activities
  • Adenosine Triphosphatases / genetics*
  • Amelogenesis Imperfecta / complications
  • Amelogenesis Imperfecta / genetics*
  • Child
  • Child, Preschool
  • DNA Mutational Analysis
  • Female
  • Hearing Loss, Sensorineural / complications
  • Hearing Loss, Sensorineural / genetics*
  • Hearing Loss, Sensorineural / physiopathology
  • Homozygote
  • Humans
  • Male
  • Membrane Proteins / genetics*
  • Mutation
  • Nails, Malformed / complications
  • Nails, Malformed / genetics*
  • Pedigree
  • Peroxisomal Disorders / genetics
  • Peroxisomal Disorders / physiopathology
  • Phenotype
  • Retinitis Pigmentosa / complications
  • Retinitis Pigmentosa / genetics*
  • Retinitis Pigmentosa / physiopathology

Substances

  • Membrane Proteins
  • Adenosine Triphosphatases
  • ATPases Associated with Diverse Cellular Activities
  • PEX1 protein, human
  • PEX6 protein, human

Supplementary concepts

  • Deafness enamel hypoplasia nail defects