A brain-specific decrease of the tyrosine hydroxylase protein in sepiapterin reductase-null mice--as a mouse model for Parkinson’s disease
Abstract
Sepiapterin reductase (SPR) is an enzyme that acts in the third and final step of tetrahydrobiopterin (BH4) biosynthesis. The human Spr gene locates within the region of 2.5MB mapped to PARK3, an autosomal dominant form of familial Parkinson's diseases. In order to explore the role of SPR in the metabolism of BH4, we produced and analyzed Spr-deficient mice. Most of Spr-null mice survived beyond two weeks. Whereas the BH4 contents in the homozygous mutant mice were greatly decreased than those in wild-type and heterozygous mice, the substantial amounts of BH4 were remained even 17 days after delivery. Spr-null mice exhibited severe monoamine deficiencies and a tremor-like phenotype after weaning. The amount of TH protein in the brain of Spr-null mice was less than 10% of wild-type, while TH protein in the adrenal, phenylalanine hydroxylase protein in the liver, and nNOS in the brain were not altered. These data suggest an essential role of SPR in the biosynthesis of BH4, and that the SPR gene could be a candidate gene for PARK3.
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Reduction of TH immunoreactivity is observed in the midbrain and striatum of 6-pyruvoyl-tetrahydrobiopterin synthase-null and sepiapterin reductase-null mice, which are mouse models of tetrahydrobiopterin biosynthesis dysfunction.
“The reduction of TH immunoreactivity can be observed in the midbrain and striatum of 6-pyruvoyl-tetrahydrobiopterin synthase-null and sepiapterin reductase-null mice, which are mouse models of tetrahydrobiopterin biosynthesis dysfunction [75,76].”
From Degradation of Tyrosine Hydroxylase by the Ubiquitin-Proteasome System in the Pathogenesis of Parkinson’s Disease and Dopa-Responsive Dystonia (2020) · cites this paper as [76]
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Tetrahydrobioterin (BH4) pathway: from metabolism to neuropsychiatry
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- Authors
- Takazawa, C., Fujimoto, K., Homma, D., Sumi-Ichinose, C., Nomura, T., Ichinose, H., & Katoh, S.
- Affiliations
- Meikai University (JP)
- Tokyo Institute of Technology (JP)
- Fujita Health University (JP)
- Corresponding author
- Ichinose, Hiroshi
- Katoh, Setsuko
- ORCID
- Ichinose, Hiroshi
- Venue
- Biochemical and Biophysical Research Communications
- Publisher
- Elsevier BV
- Published
- 2008-03
- Volume
- 367
- Issue
- 4
- Pages
- 787-792
- ISSN
- 0006-291X (ISSN-L)
- OpenAlex
- W2082650861
- Cited by
- 37
- References
- 24
- Citations by year
- 2026: 1 · 2024: 1 · 2023: 1 · 2022: 3 · 2020: 5 · 2019: 1 · 2018: 1 · 2017: 2 · 2016: 2 · 2015: 4 · 2013: 5 · 2012: 1
- FWCI
- 2.12
- Type
- journal article
- Language
- en
- Open access
- closed
- Licence
- Locations
- Biochemical and Biophysical Research Communications (publishedVersion)
- PubMed (publishedVersion)
- Institutional Repositories DataBase (IRDB) (submittedVersion)
- Tokyo Tech Research Repository (Tokyo Institute of Technology) (submittedVersion)
- Funders
- Ministry of Health, Labour and Welfare (15791067, 17791329)
- Ministry of Education, Culture, Sports, Science and Technology (17024017)
- Topics
- Health Sciences › Medicine › Neurology › Parkinson's Disease Mechanisms and Treatments
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Clinical Biochemistry › Metabolism and Genetic Disorders
- Life Sciences › Neuroscience › Neurology › Neurological diseases and metabolism
- Keywords
- sepiapterin reductase, tyrosine hydroxylase, tetrahydrobiopterin biosynthesis, Parkinson's disease, phenylalanine hydroxylase, neuronal nitric oxide synthase, candidate genes
- MeSH terms
- Alcohol Oxidoreductases, Animals, Brain, Disease Models, Animal, Metabolic Clearance Rate, Mice, Inbred C57BL, Parkinson Disease, Tissue Distribution, Tyrosine 3-Monooxygenase, Mice, Knockout, Mice
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