Dysmyelination of the cerebral white matter with microdeletion at 6p25

Indian Pediatr. 2011 Sep;48(9):727-9. doi: 10.1007/s13312-011-0108-8.

Abstract

A 6 year old boy presented with mental retardation, hypotonia, abnormal facies, impaired hearing, protuberant eyes, visual impairment, short stature, Axenfeld-Rieger anomaly, a bicuspid aortic valve, and bilateral sensorineural deafness. CT scan of head suggested dysmyelination of the subcortical and periventricular white matter. FISH revealed a subtelomeric microdeletion encompassing both FOXC1 and FOXF2 loci within 6p25. Dysmyelination of the central nervous system has been infrequently described earlier in patients with 6p25 deletion.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / diagnosis
  • Abnormalities, Multiple / diagnostic imaging
  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / pathology
  • Brain / diagnostic imaging
  • Child
  • Chromosome Deletion*
  • Chromosomes, Human, Pair 6*
  • Demyelinating Diseases / diagnosis
  • Demyelinating Diseases / diagnostic imaging
  • Demyelinating Diseases / genetics*
  • Demyelinating Diseases / pathology
  • Eye Abnormalities / diagnosis
  • Eye Abnormalities / diagnostic imaging
  • Eye Abnormalities / genetics*
  • Eye Abnormalities / pathology
  • Forkhead Transcription Factors / genetics
  • Hearing Loss, Sensorineural / diagnosis
  • Hearing Loss, Sensorineural / diagnostic imaging
  • Hearing Loss, Sensorineural / genetics*
  • Hearing Loss, Sensorineural / pathology
  • Heart Septal Defects, Atrial / diagnosis
  • Heart Septal Defects, Atrial / diagnostic imaging
  • Heart Septal Defects, Atrial / genetics*
  • Heart Septal Defects, Atrial / pathology
  • Humans
  • Male
  • Tomography, X-Ray Computed

Substances

  • FOXC1 protein, human
  • FOXF2 protein, human
  • Forkhead Transcription Factors

Supplementary concepts

  • Axenfeld-Rieger anomaly with cardiac defects and sensorineural hearing loss