Familial cramp due to potassium-aggravated myotonia

J Neurol Neurosurg Psychiatry. 1998 Oct;65(4):569-72. doi: 10.1136/jnnp.65.4.569.

Abstract

Clinical, electrophysiological, and molecular genetic features were investigated in two patients from a family a with dominantly inherited myotonic disease, characterised by painful cramps, stiffness without weakness, fluctuation of symptoms, and cold sensitivity. A reduction in amplitude of the compound muscle action potential was demonstrated on cooling and administration of potassium, although no clinical exacerbation was seen. A heterozygote mutation Val1589Met was identified in the alpha-subunit of the skeletal muscle sodium channel gene in both patients, consistent with the diagnosis of potassium-aggravated myotonia. The phenotype in this family is much milder than that previously described in another family with a mutation at this site.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • DNA Mutational Analysis
  • Electromyography / methods
  • Heterozygote
  • Humans
  • Muscle Cramp / genetics*
  • Muscle, Skeletal / innervation
  • Myotonia / genetics*
  • Neural Conduction / physiology
  • Pedigree
  • Phenotype
  • Point Mutation / genetics
  • Potassium Channels / genetics*

Substances

  • Potassium Channels