Format

Send to

Choose Destination
Biol Open. 2017 Jun 15;6(6):881-890. doi: 10.1242/bio.024679.

Pias3 is necessary for dorso-ventral patterning and visual response of retinal cones but is not required for rod photoreceptor differentiation.

Author information

1
Neurobiology-Neurodegeneration and Repair Laboratory, National Eye Institute, National Institutes of Health, 6 Center Drive, Bethesda, MD 20892, USA.
2
Nuffield Laboratory of Ophthalmology, Nuffield Department of Clinical Neurosciences, University of Oxford, Oxford OX3 9DU, UK.
3
Genetic Engineering Core, National Eye Institute, National Institutes of Health, Bethesda, MD 20892, USA.
4
Neurobiology-Neurodegeneration and Repair Laboratory, National Eye Institute, National Institutes of Health, 6 Center Drive, Bethesda, MD 20892, USA swaroopa@nei.nih.gov jerome.roger@u-psud.fr.
5
Centre d'Etude et de Recherches Thérapeutiques en Ophtalmologie, Retina France, Orsay 91405, France.
6
Paris-Saclay Institute of Neuroscience, CNRS, Université Paris-Sud, Université Paris-Saclay, Orsay 91405, France.

Abstract

Protein inhibitor of activated Stat 3 (Pias3) is implicated in guiding specification of rod and cone photoreceptors through post-translational modification of key retinal transcription factors. To investigate its role during retinal development, we deleted exon 2-5 of the mouse Pias3 gene, which resulted in complete loss of the Pias3 protein. Pias3-/- mice did not show any overt phenotype, and retinal lamination appeared normal even at 18 months. We detected reduced photopic b-wave amplitude by electroretinography following green light stimulation of postnatal day (P)21 Pias3-/- retina, suggesting a compromised visual response of medium wavelength (M) cones. No change was evident in response of short wavelength (S) cones or rod photoreceptors until 7 months. Increased S-opsin expression in the M-cone dominant dorsal retina suggested altered distribution of cone photoreceptors. Transcriptome profiling of P21 and 18-month-old Pias3-/- retina revealed aberrant expression of a subset of photoreceptor genes. Our studies demonstrate functional redundancy in SUMOylation-associated transcriptional control mechanisms and identify a specific, though limited, role of Pias3 in modulating spatial patterning and optimal function of cone photoreceptor subtypes in the mouse retina.

KEYWORDS:

Cell type specification; Gene regulation; Mouse knockout; Retina development; SUMOylation; Vision

Supplemental Content

Full text links

Icon for HighWire Icon for PubMed Central
Loading ...
Support Center