Motor-sensory neuropathy without minifascicles in a patient with 46XY gonadal dysgenesis

Brain Dev. 2008 Apr;30(4):291-4. doi: 10.1016/j.braindev.2007.07.010. Epub 2007 Sep 4.

Abstract

We report a 36-year-old patient with 46XY pure gonadal dysgenesis (GD), who manifested a syndrome of progressive motor-sensory neuropathy. Sural nerve biopsy showed severe axonal neuropathy. Since reported cases of chronic motor-sensory neuropathy and pure gonadal dysgenesis have been characterized by nerve biopsy evidence of minifascicle formation, we suggest that this clinical association may be a new type of hereditary motor-sensory neuropathy, not necessarily associated with minifascicle formation.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Axons / pathology*
  • Biopsy
  • Female
  • Gonadal Dysgenesis, 46,XY / complications*
  • Gonadal Dysgenesis, 46,XY / genetics
  • Hedgehog Proteins / genetics
  • Hereditary Sensory and Motor Neuropathy / complications*
  • Hereditary Sensory and Motor Neuropathy / genetics
  • Hereditary Sensory and Motor Neuropathy / pathology*
  • Humans
  • Sural Nerve / pathology

Substances

  • DHH protein, human
  • Hedgehog Proteins