Late-onset Tay-Sachs disease

Pract Neurol. 2017 Oct;17(5):396-399. doi: 10.1136/practneurol-2017-001665. Epub 2017 Jul 24.

Abstract

We discuss the assessment and differential diagnoses of a young adult Hungarian man with a 1-year history of a progressive and symmetric amyotrophic lateral sclerosis-like syndrome, along with irregular action tremor and stimulus-sensitive myoclonus of the arms. MR scan of the brain showed isolated cerebellar atrophy and formal neuropsychometric testing identified significant subclinical deficits in attention, processing speed and memory. We suspected a form of GM2 gangliosidosis, and white cell enzyme analysis showed markedly reduced enzymatic activity of β-hexosaminidase A. Genetic testing subsequently revealed two heterozygous pathogenic mutations in the HEXA gene (c.1499delT p.(Leu500fs) and c.805G>A p.(Gly269Ser)), confirming the very rare diagnosis of adult-onset Tay-Sachs disease.

Keywords: CEREBELLAR DEGENERATION; Hexosaminidase A Deficiency; MOTOR CONTROL; NEUROGENETICS; NEUROPSYCHOLOGY.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Age of Onset
  • Cerebellum / diagnostic imaging
  • Cerebellum / pathology
  • Creatine Kinase / blood
  • Diagnosis, Differential
  • Electrocardiography
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Tay-Sachs Disease / blood
  • Tay-Sachs Disease / diagnosis*
  • Tay-Sachs Disease / physiopathology*
  • Tomography, X-Ray Computed

Substances

  • Creatine Kinase