A case of Canavan disease with microcephaly

Brain Dev. 2016 Sep;38(8):759-62. doi: 10.1016/j.braindev.2016.03.001. Epub 2016 Mar 15.

Abstract

Background: Canavan disease is an autosomal recessive disorder with spongy degeneration of white matter of the brain. It presents with developmental delay, visual problems and macrocephaly.

Patient description: We report a ten-month old boy with Canavan disease who presented with global developmental delay, seizures, abnormal eye movements and microcephaly.

Results: MRI brain revealed diffuse involvement of the supra tentorial white matter, globus pallidi, thalami, dentate nuclei and brainstem with sparing of the corpus callosum. The genetic testing revealed homozygous mutation of aspartoacylase gene [c.859 G>A (p.Ala287Thr)] in Exon 6.

Conclusion: Possibility of Canavan disease should be considered even in the presence of microcephaly.

Keywords: Canavan disease; Microcephaly; N-acetylaspartic acid (NAA).

Publication types

  • Case Reports

MeSH terms

  • Amidohydrolases / genetics
  • Brain / diagnostic imaging*
  • Canavan Disease / diagnosis*
  • Canavan Disease / genetics
  • Canavan Disease / physiopathology
  • Developmental Disabilities / diagnosis
  • Developmental Disabilities / genetics
  • Developmental Disabilities / physiopathology
  • Diagnosis, Differential
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Microcephaly / diagnosis*
  • Microcephaly / genetics
  • Microcephaly / physiopathology
  • Seizures / diagnosis
  • Seizures / genetics
  • Seizures / physiopathology

Substances

  • Amidohydrolases
  • aspartoacylase