OXPHOS and mtDNA alterations in a family with spastic paraparesis

Acta Neurol Scand. 2000 Apr;101(4):255-8. doi: 10.1034/j.1600-0404.2000.101004255.x.

Abstract

Objective: To study muscle biopsies in hereditary spastic paraparesis (HSP).

Methods: We analyzed oxidative phosphorylation activities and mtDNA in 3 individuals from an HSP family.

Results: We found histochemical evidence for mitochondrial proliferation and cytochrome c oxidase negative fibers. Biochemically, there was an important reduction of the activities of complexes I and IV in 3 patients. In addition, multiple mtDNA deletions (ranging 4.0-7.0 kb) were found in 2 cases by PCR but not by Southern blot.

Conclusion: We suggest the use of a muscle biopsy when examining HSP patients. HSP can represent a disorder of nuclear-mitochondrial intercommunication.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Biopsy
  • Blotting, Southern
  • DNA Mutational Analysis
  • DNA, Mitochondrial / genetics*
  • Female
  • Humans
  • Male
  • Middle Aged
  • Muscle, Skeletal / metabolism*
  • Oxidative Phosphorylation*
  • Paraparesis, Spastic / genetics*
  • Paraparesis, Spastic / metabolism*
  • Polymerase Chain Reaction
  • Predictive Value of Tests
  • Sequence Deletion / genetics*

Substances

  • DNA, Mitochondrial