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Drosophila fatty acid transport protein regulates rhodopsin-1 metabolism and is required for photoreceptor neuron survival

Dourlen, Pierre
•
Bertin, Benjamin
•
Chatelain, Gilles
altro
Mollereau, Bertrand
2012
  • journal article

Periodico
PLOS GENETICS
Abstract
Tight regulation of the visual response is essential for photoreceptor function and survival. Visual response dysregulation often leads to photoreceptor cell degeneration, but the causes of such cell death are not well understood. In this study, we investigated a fatty acid transport protein (fatp) null mutation that caused adult-onset and progressive photoreceptor cell death. Consistent with fatp having a role in the retina, we showed that fatp is expressed in adult photoreceptors and accessory cells and that its re-expression in photoreceptors rescued photoreceptor viability in fatp mutants. The visual response in young fatp-mutant flies was abnormal with elevated electroretinogram amplitudes associated with high levels of Rhodopsin-1 (Rh1). Reducing Rh1 levels in rh1 mutants or depriving flies of vitamin A rescued photoreceptor cell death in fatp mutant flies. Our results indicate that fatp promotes photoreceptor survival by regulating Rh1 abundance.
DOI
10.1371/journal.pgen.1002833
WOS
WOS:000306840400045
Archivio
http://hdl.handle.net/11368/2930987
info:eu-repo/semantics/altIdentifier/scopus/2-s2.0-84864603834
http://www.plosgenetics.org/article/fetchObjectAttachment.action?uri=info%3Adoi%2F10.1371%2Fjournal.pgen.1002833&representation=PDF
Diritti
metadata only access
Soggetti
  • Animal

  • Cell Death

  • Electroretinography

  • Gene Expression

  • Mutation

  • Neuron

  • Photic Stimulation

  • Retina

  • Vitamin D

  • Drosophila melanogast...

  • Fatty Acid Transport ...

  • Photoreceptor Cells, ...

  • Retinal Degeneration

  • Rhodopsin

  • Ecology, Evolution, B...

  • Molecular Biology

  • Genetic

  • Genetics (clinical)

  • Cancer Research

Scopus© citazioni
34
Data di acquisizione
Jun 14, 2022
Vedi dettagli
Web of Science© citazioni
34
Data di acquisizione
Mar 20, 2024
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