Diaphragmatic weakness with progressive sensory and motor polyneuropathy: case report of a neonatal IGHMBP2-related neuropathy

J Child Neurol. 2013 Jun;28(6):787-90. doi: 10.1177/0883073812450209. Epub 2012 Jul 12.

Abstract

The authors present a child affected with diaphragmatic paralysis in the early neonatal period. Although no electroneuromyographic abnormalities were reported, the patient developed dramatic motor and respiratory impairment with impossibility to wean from mechanical ventilation. Repeated electroneuromyographic study at age 4 months revealed severe neurogenic changes and sensory nerve abnormalities with more preserved nerve conduction velocities. Genetic studies identified 2 mutations in the gene IGHMBP2. These results support the consideration of this entity as a form of sensory-motor rapidly progressive polyneuropathy rather than a primary anterior horn disease (IGHMBP2-related neuropathy). A review of the series of mutated patients in the French National Database gives new insights of the incidence of this disease in France.

Keywords: diaphragmatic weakness; infantile neuropathy; palsy; peripheral nervous system diseases; spinal muscular atrophy with respiratory distress type I.

Publication types

  • Case Reports

MeSH terms

  • Cross-Sectional Studies
  • DNA Mutational Analysis*
  • DNA-Binding Proteins / genetics*
  • Diagnosis, Differential
  • Electromyography
  • Fatal Outcome
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Muscular Atrophy, Spinal / diagnosis
  • Muscular Atrophy, Spinal / genetics*
  • Muscular Atrophy, Spinal / therapy
  • Neurologic Examination
  • Phenotype
  • Respiratory Distress Syndrome, Newborn / diagnosis
  • Respiratory Distress Syndrome, Newborn / genetics*
  • Respiratory Distress Syndrome, Newborn / therapy
  • Respiratory Paralysis / diagnosis
  • Respiratory Paralysis / epidemiology
  • Respiratory Paralysis / genetics*
  • Respiratory Paralysis / therapy
  • Transcription Factors / genetics*
  • Ventilator Weaning

Substances

  • DNA-Binding Proteins
  • IGHMBP2 protein, human
  • Transcription Factors

Supplementary concepts

  • Spinal muscular atrophy with respiratory distress 1