Duchenne muscular dystrophy in a 4-year-old girl due to heterozygous frame shift deletion of the dystrophin gene and skewed X-inactivation

Klin Padiatr. 2012 Jul;224(4):256-8. doi: 10.1055/s-0032-1304626. Epub 2012 Apr 27.

Abstract

X-linked recessive diseases affect males, whereas female carriers are generally asymptomatic.We report on a 4-year-old girl who presented with a classical phenotype of Duchenne Muscular Dystrophy (DMD), a severe X-linked recessive type of muscular dystrophy affecting boys in early childhood.A thorough diagnostic work-up revealed that this resulted from a heterozygous out-of frame deletion in the DMD-gene in combination with an X-inactivation ratio of <10:90 in blood leukocytes and muscle.The case exemplifies that a skewed X-inactivation pattern has to be taken into account as mechanism causing clinical symptoms in female carriers of X-linked recessive disorders.

Publication types

  • Case Reports

MeSH terms

  • Biopsy
  • Child, Preschool
  • Creatine Kinase / blood
  • Dystrophin / genetics*
  • Exons / genetics
  • Female
  • Frameshift Mutation / genetics*
  • Genes, Recessive / genetics
  • Genetic Carrier Screening*
  • Humans
  • Leukocytes / metabolism
  • Muscle, Skeletal / metabolism
  • Muscle, Skeletal / pathology
  • Polymerase Chain Reaction
  • Sarcoglycanopathies / diagnosis
  • Sarcoglycanopathies / genetics*
  • X Chromosome Inactivation / genetics*

Substances

  • Dystrophin
  • Creatine Kinase

Supplementary concepts

  • Limb-girdle muscular dystrophy, type 2C