Increasing and persistent DWI changes in a patient with hereditary diffuse leukoencephalopathy with spheroids

J Neurol Sci. 2013 Dec 15;335(1-2):213-5. doi: 10.1016/j.jns.2013.08.027. Epub 2013 Aug 30.

Abstract

We report a case with genetically confirmed hereditary diffuse leukoencephalopathy with spheroids with distinctive MRI features. A 52-year-old woman with a family history of juvenile dementia presented with an 18-month history of progressive cognitive decline. Longitudinal magnetic resonance imaging studies of the brain revealed increasing and persistent white matter hyperintensities on diffusion-weighted images. Linear high intensity signal along axonal fibers arisen from the cerebral cortex was also shown. Finding of subcortical calcifications was noted on brain CT scan. Sequence analysis of CSF1R showed a novel missense mutation c.2467C>T (p.Ala823Val). Persistent and increasing diffusion on magnetic resonance image, presumably reflecting intramyelinic oedema in regions of neurodegeneration, is a distinctive feature observed in this case. The presence of this unique finding can be a diagnostic clue in the early stage of the disease.

Keywords: Colony stimulating factor 1 receptor (CSF1R); Diagnosis; Diffusion-weighted imaging (DWI); Hereditary Diffuse Leukoencephalopathy with Spheroids (HDLS); Magnetic resonance imaging (MRI); dJuvenile dementia.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Diffusion Magnetic Resonance Imaging*
  • Female
  • Humans
  • Leukoencephalopathies / diagnosis*
  • Leukoencephalopathies / genetics
  • Middle Aged
  • Mutation / genetics
  • Receptor, Macrophage Colony-Stimulating Factor / genetics
  • Spheroids, Cellular / pathology*

Substances

  • Receptor, Macrophage Colony-Stimulating Factor