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| Verfasst von: | Breuer, Maximilian [VerfasserIn]  |
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| | Guglielmi, Luca [VerfasserIn] |
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| | Zielonka, Matthias [VerfasserIn] |
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| | Kölker, Stefan [VerfasserIn] |
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| | Okun, Jürgen G. [VerfasserIn] |
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| | Hoffmann, Georg Friedrich [VerfasserIn] |
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| | Carl, Matthias [VerfasserIn] |
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| | Sauer, Sven [VerfasserIn] |
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| | Opladen, Thomas [VerfasserIn] |
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| Titel: | QDPR homologues in Danio rerio regulate melanin synthesis, early gliogenesis, and glutamine homeostasis |
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| Verf.angabe: | Maximilian Breuer, Luca Guglielmi, Matthias Zielonka, Verena Hemberger, Stefan Kölker, Jürgen G. Okun, Georg F. Hoffmann, Matthias Carl, Sven W. Sauer, Thomas Opladen |
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| E-Jahr: | 2019 |
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| Jahr: | April 17, 2019 |
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| Umfang: | 19 S. |
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| Fussnoten: | Gesehen am 14.10.2019 |
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| Titel Quelle: | Enthalten in: PLOS ONE |
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| Ort Quelle: | San Francisco, California, US : PLOS, 2006 |
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| Jahr Quelle: | 2019 |
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| Band/Heft Quelle: | 14(2019,4) Artikel-Nummer e0215162, 19 Seiten |
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| ISSN Quelle: | 1932-6203 |
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| Abstract: | Dihydropteridine reductase (QDPR) catalyzes the recycling of tetrahydrobiopterin (BH4), a cofactor in dopamine, serotonin, and phenylalanine metabolism. QDPR-deficient patients develop neurological symptoms including hypokinesia, truncal hypotonia, intellectual disability and seizures. The underlying pathomechanisms are poorly understood. We established a zebrafish model for QDPR deficiency and analyzed the expression as well as function of all zebrafish QDPR homologues during embryonic development. The homologues qdpra is essential for pigmentation and phenylalanine metabolism. Qdprb1 is expressed in the proliferative zones of the optic tectum and eye. Knockdown of qdprb1 leads to up-regulation of pro-proliferative genes and increased number of phospho-histone3 positive mitotic cells. Expression of neuronal and astroglial marker genes is concomitantly decreased. Qdprb1 hypomorphic embryos develop microcephaly and reduced eye size indicating a role for qdprb1 in the transition from cell proliferation to differentiation. Glutamine accumulation biochemically accompanies the developmental changes. Our findings provide novel insights into the neuropathogenesis of QDPR deficiency. |
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| DOI: | doi:10.1371/journal.pone.0215162 |
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| URL: | Bitte beachten Sie: Dies ist ein Bibliographieeintrag. Ein Volltextzugriff für Mitglieder der Universität besteht hier nur, falls für die entsprechende Zeitschrift/den entsprechenden Sammelband ein Abonnement besteht oder es sich um einen OpenAccess-Titel handelt.
Volltext: https://doi.org/10.1371/journal.pone.0215162 |
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| | Verlag: https://journals.plos.org/plosone/article?id=10.1371/journal.pone.0215162 |
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| | DOI: https://doi.org/10.1371/journal.pone.0215162 |
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| Datenträger: | Online-Ressource |
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| Sprache: | eng |
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| Sach-SW: | Embryos |
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| | Eyes |
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| | Glutamine |
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| | Melanin |
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| | Phenylalanine |
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| | Pigments |
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| | Superior colliculus |
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| | Zebrafish |
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| K10plus-PPN: | 1678852651 |
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| Verknüpfungen: | → Zeitschrift |
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QDPR homologues in Danio rerio regulate melanin synthesis, early gliogenesis, and glutamine homeostasis / Breuer, Maximilian [VerfasserIn]; April 17, 2019 (Online-Ressource)
