Amyotrophic lateral sclerosis-plus syndrome with TAR DNA-binding protein-43 pathology

Arch Neurol. 2009 Jan;66(1):121-4. doi: 10.1001/archneur.66.1.121.

Abstract

Background: Amyotrophic lateral sclerosis (ALS)-Plus syndromes meet clinical criteria for ALS but also include 1 or more additional features such as dementia, geographic clustering, extrapyramidal signs, objective sensory loss, autonomic dysfunction, cerebellar degeneration, or ocular motility disturbance.

Methods: We performed a whole-brain and spinal cord pathologic analysis in a patient with an ALS-Plus syndrome that included repetitive behaviors along with extrapyramidal and supranuclear ocular motility disturbances resembling the clinical phenotype of progressive supranuclear palsy.

Results: There was motoneuron cell loss and degeneration of the corticospinal tracts. Bunina bodies were present. TAR DNA-binding protein-43 pathology was diffuse. Significant tau pathology was absent.

Conclusions: TAR DNA-binding protein-43 disorders can produce a clinical spectrum of neurodegeneration that includes ALS, frontotemporal lobar degeneration, and ALS with frontotemporal lobar degeneration. The present case illustrates that isolated TAR DNA-binding protein-43 disorders can produce an ALS-Plus syndrome with extrapyramidal features and supranuclear gaze palsy resembling progressive supranuclear palsy.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Amyotrophic Lateral Sclerosis / complications*
  • Amyotrophic Lateral Sclerosis / genetics*
  • Amyotrophic Lateral Sclerosis / physiopathology
  • Brain / metabolism
  • Brain / pathology*
  • Brain / physiopathology
  • DNA Mutational Analysis
  • DNA-Binding Proteins / genetics*
  • Dementia / genetics*
  • Dementia / metabolism
  • Dementia / physiopathology
  • Disease Progression
  • Fatal Outcome
  • Genetic Markers / genetics
  • Genetic Predisposition to Disease / genetics
  • Genetic Testing
  • Genotype
  • Humans
  • Inclusion Bodies / pathology
  • Male
  • Motor Neurons / pathology
  • Mutation / genetics
  • Ocular Motility Disorders / genetics*
  • Ocular Motility Disorders / metabolism
  • Ocular Motility Disorders / physiopathology
  • Pyramidal Tracts / pathology

Substances

  • DNA-Binding Proteins
  • Genetic Markers