Golbe, L. I., A. M. Lazzarini, et al. (2001). "The tau A0 allele
in Parkinson's disease." Mov Disord16(3): 442-7.
Parkinson's disease (PD) is primarily an alpha-synucleinopathy, rather than a
tauopathy, but there is evidence for an indirect association of tau with the
pathogenetic process in PD. We therefore assessed the frequency in PD of the tau
A0 allele, a dinucleotide repeat marker that has been associated with a sporadic
tauopathy, progressive supranuclear palsy (PSP). We found the A0 allele to
comprise 79.2% of 758 alleles from PD patients and 71.2% of 264 control alleles
(P = 0.008). We also performed a meta-analysis of three previous reports, two of
which failed to produce statistically significant results. Taken together, they
also support a PD/A0 allelic association, even after correction for misdiagnosis
of PSP as PD (P< 0.001). The A0/A0 genotype frequency in our patients (62.3%)
did not differ significantly from that in controls (53.0%, P = 0.062), but the
meta-analysis, even after correction for misdiagnosis, showed a significant
result, with P = 0.002. The frequency of A0 allele and the A0/A0 genotype were
compatible with Hardy-Weinberg equilibrium. The frequency of the A0 allele and
the A0/A0 genotype in our patients with familial PD was not significantly
greater than in those with sporadic PD. We conclude that the tau protein may
play a small role in the pathogenesis of PD and that biochemical
characterization of this role may suggest opportunities for PD prophylaxis.
Copyright 2001 Movement Disorder Society.
Horowitz, J. M., V. A. Vernace, et al. (2001). "Immunodetection of Parkin
protein in vertebrate and invertebrate brains: a comparative study using
specific antibodies." J Chem Neuroanat21(1): 75-93.
Parkin is an intracellular protein that plays a significant role in the
etiopathogenesis of autosomal recessive juvenile parkinsonism. Using immunoblot
methods, we found Parkin isoforms varying from 54 to 58 kDa in rat, mouse, bird,
frog and fruit-fly brains. Immunocytochemical studies carried out in rats, mice
and birds demonstrated multiple cell types bearing the phenotype for Parkin
throughout telencephalic, diencephalic, mesencephalic and metencephalic brain
structures. While in some instances Parkin-containing neurons tended to be
grouped into clusters, the majority of these labeled nerve cells were widely
scattered throughout the neuraxis. The topographical distribution and
organizational pattern of Parkin within major functional brain circuits was
comparable in both rats and mice. However, the subcellular localization of
Parkin was found to vary significantly as a function of antibody reactivity. A
consistent cytoplasmic labeling for Parkin was observed in rodent tissue
incubated with a polyclonal antibody raised against the human Parkin protein and
having an identical amino-acid sequence with that of the rat. In contrast,
rodent tissue alternately incubated with a polyclonal antibody raised against a
different region of the same human Parkin protein but having 10 mismatched
amino-acid sequence changes with those of the rat and mouse, resulted in nuclear
labeling for Parkin in rat but not mouse neurons. This difference in epitope
recognition, however, was reversed when mouse brain tissue was heated at 80
degrees C, apparently unmasking target epitopes against which the antisera were
directed. Collectively, these results show a high degree of conservation in the
cellular identity of Parkin in animals as different as drosophilids and mammals
and points to the possibility that the biochemical specificities of Parkin,
including analogous functional roles, may have been conserved during the course
of evolution.
Kang, U. J., W. Y. Lee, et al. (2001). "Gene therapy for Parkinson's disease:
determining the genes necessary for optimal dopamine replacement in rat models."
Hum Cell14(1): 39-48.
This article reviews the mechanism of dopamine delivery in the CNS in order to
determine the optimal set of genes for effective gene therapy in Parkinson's
disease (PD). Systematic neurobiological investigation of the biochemical steps
has revealed that tyrosine hydroxylase (TH), which has been used in earlier
studies, functions only when the essential cofactor, tetrahydrobiopterin (BH1)
is present. Transduction of the gene for GTP cyclohydrolase I, the first and
rate-limiting step in BH1 synthesis, along with the TH gene, generated cells
that are capable of producing L-DOPA spontaneously both in vitro and in vivo.
When the aromatic L-amino acid decarboxylase (AADC) gene was added as a third
gene, in an attempt to increase the conversion of L-DOPA to dopamine, feedback
inhibition by the end product, dopamine, on TH activity resulted. To circumvent
this problem, we employed a complementary strategy. Gene transfer of the
vesicular monoamine transporter was combined with AADC and produced genetically
modified cells that can convert L-DOPA to dopamine and store it for gradual
release. This approach provided a means to regulate final dopamine delivery by
controlling precursor doses and to achieve more sustained delivery of dopamine.
Our investigation into determining the genes necessary for optimal dopamine
delivery has been facilitated by in vivo biochemical assays using microdialysis.
This technique has provided us with a clear and quantitative tool to compare the
effects of various genes involved in dopamine synthesis and processing.
Sherer, T. B., R. Betarbet, et al. (2001). "Pathogenesis of Parkinson's
disease." Curr Opin Investig Drugs2(5): 657-62.
Parkinson's disease (PD) is a progressive neurodegenerative disorder
characterized by degeneration of the nigrostriatal dopaminergic pathway and the
appearance of cytoplasmic proteinaceous aggregates known as Lewy bodies. Studies
of familial PD have uncovered rare causative mutations in genes, including
alpha-synuclein. Mutations or oxidative modification of alpha-synuclein causes
it to aggregate; alpha-synuclein is a major component of the Lewy body in both
familial and sporadic PD. Biochemical analysis has implicated mitochondrial
dysfunction in PD. Epidemiological studies indicate a role of exposure to
pesticides, some of which are mitochondrial toxins. Mitochondrial dysfunction,
resulting from genetic defects, environmental toxins, or a combination of the
two, may cause alpha-synuclein aggregation and produce selective
neurodegeneration through mechanisms involving oxidative stress and
excitotoxicity. Efforts to better define PD pathogenesis should reveal novel
therapeutic targets.