Cytotoxic edema and diffusion restriction as an early pathoradiologic marker in canavan disease: case report and review of the literature

Orphanet J Rare Dis. 2016 Dec 7;11(1):169. doi: 10.1186/s13023-016-0549-1.

Abstract

Background: Canavan disease is a devastating autosomal recessive leukodystrophy leading to spongiform degeneration of the white matter. There is no cure or treatment for Canavan disease, and disease progression is poorly understood.

Results: We report a new presentation of a patient found to have Canavan disease; brain magnetic resonance imaging (MRI) revealed white matter cytotoxic edema, indicative of an acute active destructive process. We performed a comprehensive review of published cases of Canavan disease reporting brain MRI findings, and found that cytotoxic brain edema is frequently reported in early Canavan disease.

Conclusions: Our results and the literature review support the notion of an acute phase in Canavan disease progression. These findings suggest that there is a window available for therapeutic intervention and support the need for early identification of patients with Canavan disease.

Keywords: Canavan disease; Cytotoxic edema; Diffusion restriction; Leukodystrophy; MRI.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Biomarkers
  • Brain / diagnostic imaging*
  • Brain / pathology*
  • Canavan Disease / diagnostic imaging*
  • Canavan Disease / pathology*
  • Humans

Substances

  • Biomarkers