Risk of Parkinson disease in carriers of parkin mutations: estimation using the kin-cohort method

Arch Neurol. 2008 Apr;65(4):467-74. doi: 10.1001/archneur.65.4.467.

Abstract

Objective: To estimate the risk of Parkinson disease (PD) in individuals with mutations in the Parkin gene.

Design: We assessed point mutations and exon deletions and duplications in the Parkin gene in 247 probands with PD (age at onset < or =50 years) and 104 control probands enrolled in the Genetic Epidemiology of Parkinson's Disease (GEPD) study. For each first-degree relative, a consensus diagnosis of PD was established. The probability that each relative carried a mutation was estimated from the proband's Parkin carrier status using Mendelian principles and from the relationship of the relative to the proband.

Setting: Tertiary care movement disorders center. Patients Cases, controls, and their first-degree relatives were enrolled in the GEPD study.

Main outcome measures: Estimated age-specific penetrance in first-degree relatives.

Results: Parkin mutations were identified in 25 probands with PD (10.1%), 18 (72.0%) of whom were heterozygotes. One Parkin homozygote was reported in 2 siblings with PD. The cumulative incidence of PD to age 65 years in carrier relatives (age-specific penetrance) was estimated to be 7.0% (95% confidence interval, 0.4%-71.9%), compared with 1.7% (95% confidence interval, 0.8%-3.4%) in noncarrier relatives of the cases (P = .59) and 1.1% (95% confidence interval, 0.3%-3.4%) in relatives of the controls (compared with noncarrier relatives, P = .52).

Conclusions: The cumulative risk of PD to age 65 years in a noncarrier relative of a case with an age at onset of 50 years or younger is not significantly greater than the general population risk among controls. Age-specific penetrance among Parkin carriers, in particular heterozygotes, deserves further study.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Age Factors
  • Aged
  • Alleles*
  • Case-Control Studies
  • Cohort Studies
  • DNA Mutational Analysis*
  • Female
  • Gene Frequency / genetics
  • Genetic Carrier Screening*
  • Genetic Predisposition to Disease / genetics*
  • Genetic Testing / statistics & numerical data
  • Genotype
  • Humans
  • Incidence
  • Leucine-Rich Repeat Serine-Threonine Protein Kinase-2
  • Male
  • Middle Aged
  • Parkinsonian Disorders / diagnosis
  • Parkinsonian Disorders / epidemiology
  • Parkinsonian Disorders / genetics*
  • Penetrance
  • Probability
  • Protein Serine-Threonine Kinases / genetics
  • Risk Assessment

Substances

  • LRRK2 protein, human
  • Leucine-Rich Repeat Serine-Threonine Protein Kinase-2
  • Protein Serine-Threonine Kinases