Dopamine-agonist responsive Parkinsonism in a patient with the SANDO syndrome caused by POLG mutation

BMC Med Genet. 2013 Oct 7:14:105. doi: 10.1186/1471-2350-14-105.

Abstract

Background: Disorders of oxidative phosphorylation affects 1/5000 individuals and present heterogeneous involvement of tissues highly dependent upon ATP production.

Case presentation: Here we present the case of a 48-year-old woman carrying a homozygous mutation (p.A899T) in mitochondrial polymerase gamma (POLG) and manifesting with a complex neurological phenotype including Dopamine-agonist responsive Parkinsonism.

Conclusion: This case report is further evidence that mitochondrial dysfunction might play a role in Parkinson's Disease pathogenesis and helps in identification of apparent mutation-specific clinical characteristics. Mutations in POLG should be looked for in cases of Parkinsonism, especially when multisystem neurological involvement is found.

Publication types

  • Case Reports

MeSH terms

  • Benzothiazoles / therapeutic use
  • DNA Mutational Analysis
  • DNA Polymerase gamma
  • DNA-Directed DNA Polymerase / genetics*
  • DNA-Directed DNA Polymerase / metabolism
  • Dopamine Agonists / therapeutic use*
  • Duloxetine Hydrochloride
  • Dysarthria / complications
  • Dysarthria / genetics*
  • Dysarthria / pathology
  • Female
  • Hereditary Sensory and Motor Neuropathy / complications
  • Hereditary Sensory and Motor Neuropathy / genetics*
  • Hereditary Sensory and Motor Neuropathy / pathology
  • Humans
  • Middle Aged
  • Mitochondria / enzymology
  • Ophthalmoplegia / complications
  • Ophthalmoplegia / genetics*
  • Ophthalmoplegia / pathology
  • Parkinson Disease / complications
  • Parkinson Disease / drug therapy*
  • Pramipexole
  • Thiophenes / therapeutic use
  • Tomography, Emission-Computed, Single-Photon

Substances

  • Benzothiazoles
  • Dopamine Agonists
  • Thiophenes
  • Pramipexole
  • Duloxetine Hydrochloride
  • DNA Polymerase gamma
  • DNA-Directed DNA Polymerase
  • POLG protein, human

Supplementary concepts

  • Sensory ataxic neuropathy, dysarthria, and ophthalmoparesis