Targeted ablation of the Pde6h gene in mice reveals cross-species differences in cone and rod phototransduction protein isoform inventory.

The Journal of Biological Chemistry
Christina BrennenstuhlRobert Lukowski

Abstract

Phosphodiesterase-6 (PDE6) is a multisubunit enzyme that plays a key role in the visual transduction cascade in rod and cone photoreceptors. Each type of photoreceptor utilizes discrete catalytic and inhibitory PDE6 subunits to fulfill its physiological tasks, i.e. the degradation of cyclic guanosine-3',5'-monophosphate at specifically tuned rates and kinetics. Recently, the human PDE6H gene was identified as a novel locus for autosomal recessive (incomplete) color blindness. However, the three different classes of cones were not affected to the same extent. Short wave cone function was more preserved than middle and long wave cone function indicating that some basic regulation of the PDE6 multisubunit enzyme was maintained albeit by a unknown mechanism. To study normal and disease-related functions of cone Pde6h in vivo, we generated Pde6h knock-out (Pde6h(-/-)) mice. Expression of PDE6H in murine eyes was restricted to both outer segments and synaptic terminals of short and long/middle cone photoreceptors, whereas Pde6h(-/-) retinae remained PDE6H-negative. Combined in vivo assessment of retinal morphology with histomorphological analyses revealed a normal overall integrity of the retinal organization and an unaltered distrib...Continue Reading

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Citations

Jan 18, 2017·The Journal of Gene Medicine·Stylianos MichalakisMartin Biel
Aug 2, 2017·Bioinformatics and Biology Insights·Apichat Suratanee, Kitiporn Plaimas
Jan 6, 2018·Ophthalmic Genetics·Nashila HirjiMichel Michaelides
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Jan 15, 2020·Computational and Structural Biotechnology Journal·Chia-Yin ChiangBen-Yang Liao
Aug 3, 2019·Progress in Retinal and Eye Research·Michael PowerFrançois Paquet-Durand
Apr 17, 2021·Proceedings of the National Academy of Sciences of the United States of America·Natalie AckerBruce E Vogel
Dec 4, 2021·Molecular Diagnosis & Therapy·Stylianos MichalakisClaudia Priglinger

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