Recurrent COLQ mutation in congenital myasthenic syndrome

Pediatr Neurol. 2012 Apr;46(4):253-6. doi: 10.1016/j.pediatrneurol.2012.02.003.

Abstract

Congenital myasthenic syndromes comprise clinically and genetically heterogeneous disorders resulting from presynaptic, synaptic, or postsynaptic defects. Mutations in the COLQ gene result in acetylcholinesterase deficiency and cause a rare, autosomal recessive synaptic form of congenital myasthenic syndrome, with variable age of onset and clinical severity. We present four unrelated patients with a homozygous W148X mutation in the COLQ gene. Signs began at birth in all, but subsequent severity ranged from independent ambulation to wheelchair use during childhood. Treatment was partly effective; one patient was asymptomatic with 3,4-diaminopyridine treatment. These cases illustrate the clinical features and treatment results associated with this particular genotype, which appears to be relatively frequent among Turkish patients with congenital myasthenic syndrome.

Publication types

  • Case Reports

MeSH terms

  • 4-Aminopyridine / analogs & derivatives
  • 4-Aminopyridine / therapeutic use
  • Acetylcholinesterase / genetics*
  • Adolescent
  • Amifampridine
  • Child
  • Collagen / genetics*
  • DNA Mutational Analysis
  • Female
  • Humans
  • Infant
  • Male
  • Muscle Proteins / genetics*
  • Mutation / genetics*
  • Myasthenic Syndromes, Congenital / drug therapy
  • Myasthenic Syndromes, Congenital / genetics*
  • Myasthenic Syndromes, Congenital / pathology
  • Potassium Channel Blockers / therapeutic use

Substances

  • Muscle Proteins
  • Potassium Channel Blockers
  • Collagen
  • 4-Aminopyridine
  • Acetylcholinesterase
  • COLQ protein, human
  • Amifampridine