Levecque, C., A. Destee, et al. (2001). "No genetic association
of the ubiquitin carboxy-terminal hydrolase-L1 gene S18Y polymorphism with
familial Parkinson's disease." J Neural Transm108(8-9): 979-84.
Parkinson's disease (PD) is a neurodegenerative disorder for which genetic
susceptibility has been documented in sporadic and familial cases. Recently, a
polymorphism located in exon 3 at codon 18 (S18Y) of the Ubiquitin Carboxy-terminal
Hydrolase-L1 (UCH-L1) gene has been associated with the disease in 2 populations
of German origin and also in a Japanese population. We tested the impact of this
polymorphism in a French sample of familial PD patients (n = 114) and controls
(n = 93). No association was observed, indicating that this polymorphism did not
confer susceptibility for familial PD in our population, even among the youngest
age of onset group. This observation suggests that the previous positive results
obtained may reflect mechanisms restricted to the sporadic form of the disease
or to a founder effect of the disease susceptibility.
Tanaka, Y., S. Engelender, et al. (2001). "Inducible expression of mutant alpha-synuclein
decreases proteasome activity and increases sensitivity to
mitochondria-dependent apoptosis." Hum Mol Genet10(9): 919-26.
Parkinson's disease (PD) is a common progressive neurodegenerative disorder
caused by the loss of dopaminergic neurons in the substantia nigra. Although
mutations in alpha-synuclein have been identified in autosomal dominant PD, the
mechanism by which dopaminergic neural cell death occurs remains unknown.
Proteins encoded by two other genes in which mutations cause familial PD, parkin
and UCH-L1, are involved in regulation of the ubiquitin-proteasome pathway,
suggesting that dysregulation of the ubiquitin-proteasome pathway is involved in
the mechanism by which these mutations cause PD. We established inducible PC12
cell lines in which wild-type or mutant alpha-synuclein can be de-repressed by
removing doxycycline. Differentiated PC12 cell lines expressing mutant alpha-synuclein
showed decreased activity of proteasomes without direct toxicity. Cells
expressing mutant alpha-synuclein showed increased sensitivity to apoptotic cell
death when treated with sub-toxic concentrations of an exogenous proteasome
inhibitor. Apoptosis was accompanied by mitochondrial depolarization and
elevation of caspase-3 and -9, and was blocked by cyclosporin A. These data
suggest that expression of mutant alpha-synuclein results in sensitivity to
impairment of proteasome activity, leading to mitochondrial abnormalities and
neuronal cell death.