Heterozygous carriers of a Parkin or PINK1 mutation share a common functional endophenotype

Neurology. 2009 Mar 24;72(12):1041-7. doi: 10.1212/01.wnl.0000338699.56379.11. Epub 2008 Nov 26.

Abstract

Objective: To use a combined neurogenetic-neuroimaging approach to examine the functional consequences of preclinical dopaminergic nigrostriatal dysfunction in the human motor system. Specifically, we examined how a single heterozygous mutation in different genes associated with recessively inherited Parkinson disease alters the cortical control of sequential finger movements.

Methods: Nonmanifesting individuals carrying a single heterozygous Parkin (n = 13) or PINK1 (n = 9) mutation and 23 healthy controls without these mutations were studied with functional MRI (fMRI). During fMRI, participants performed simple sequences of three thumb-to-finger opposition movements with their right dominant hand. Since heterozygous Parkin and PINK1 mutations cause a latent dopaminergic nigrostriatal dysfunction, we predicted a compensatory recruitment of those rostral premotor areas that are normally implicated in the control of complex motor sequences. We expected this overactivity to be independent of the underlying genotype.

Results: Task performance was comparable for all groups. The performance of a simple motor sequence task consistently activated the rostral supplementary motor area and right rostral dorsal premotor cortex in mutation carriers but not in controls. Task-related activation of these premotor areas was similar in carriers of a Parkin or PINK1 mutation.

Conclusion: Mutations in different genes linked to recessively inherited Parkinson disease are associated with an additional recruitment of rostral supplementary motor area and rostral dorsal premotor cortex during a simple motor sequence task. These premotor areas were recruited independently of the underlying genotype. The observed activation most likely reflects a "generic" compensatory mechanism to maintain motor function in the context of a mild dopaminergic deficit.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptation, Biological / genetics
  • Adult
  • Biomarkers
  • Brain Mapping
  • Female
  • Frontal Lobe / anatomy & histology
  • Frontal Lobe / physiopathology*
  • Genetic Carrier Screening / methods
  • Genetic Predisposition to Disease / genetics*
  • Heterozygote
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Motor Cortex / physiopathology
  • Movement / physiology
  • Mutation / genetics
  • Neuronal Plasticity / genetics*
  • Parkinsonian Disorders / diagnosis
  • Parkinsonian Disorders / genetics*
  • Parkinsonian Disorders / physiopathology*
  • Phenotype
  • Protein Kinases / genetics*
  • Ubiquitin-Protein Ligases / genetics*

Substances

  • Biomarkers
  • Ubiquitin-Protein Ligases
  • parkin protein
  • Protein Kinases
  • PTEN-induced putative kinase