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Progressive Muscular Atrophy.

Teerin Liewluck1, David S Saperstein2

  • 1Department of Neurology, University of Colorado School of Medicine, Anschutz Medical Campus, 12631 East 17th Avenue, Mail Stop B-185, Aurora, CO 80045, USA; Department of Neurology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905, USA.

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|October 31, 2015
PubMed
Summary
This summary is machine-generated.

Progressive muscular atrophy (PMA) is a motor neuron disease often showing only lower motor neuron signs. However, many patients have hidden upper motor neuron involvement, impacting clinical trial eligibility.

Keywords:
Lower motor neuron syndromeLower motor neuron-onset ALSPMAProgressive muscular atrophy

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Area of Science:

  • Neurology
  • Neuroscience
  • Motor Neuron Diseases

Background:

  • Progressive muscular atrophy (PMA) is a rare motor neuron disease.
  • PMA typically presents with isolated lower motor neuron (LMN) features.
  • Upper motor neuron (UMN) signs can emerge later in some PMA patients.

Purpose of the Study:

  • To investigate the prevalence and implications of subclinical UMN involvement in PMA.
  • To assess the diagnostic and clinical trial eligibility criteria for PMA patients.
  • To evaluate the efficacy of intravenous immunoglobulin (IVIg) in PMA.

Main Methods:

  • Pathological, radiological, and neurophysiological assessments were used to identify subclinical UMN involvement.
  • Analysis of patient data against the revised El Escorial criteria for amyotrophic lateral sclerosis (ALS) clinical trials.
  • Review of outcomes for patients treated with IVIg.

Main Results:

  • A substantial number of PMA patients exhibit subclinical UMN involvement.
  • These patients do not meet the criteria for ALS clinical trials.
  • IVIg showed only marginal benefit in a small subgroup of LMN syndrome patients without conduction block.

Conclusions:

  • Subclinical UMN involvement is common in PMA and complicates clinical trial participation.
  • Current criteria may exclude a significant portion of PMA patients from relevant research.
  • Limited efficacy of IVIg suggests a need for alternative therapeutic strategies in PMA.