PUBLICATION
Capn5 Expression in the Healthy and Regenerating Zebrafish Retina
- Authors
- Coomer, C.E., Morris, A.C.
- ID
- ZDB-PUB-180722-10
- Date
- 2018
- Source
- Investigative ophthalmology & visual science 59: 3643-3654 (Journal)
- Registered Authors
- Morris, Ann C.
- Keywords
- none
- MeSH Terms
-
- Animals
- Calpain/biosynthesis
- Calpain/genetics*
- Disease Models, Animal
- Gene Expression Regulation*
- Immunohistochemistry
- In Situ Hybridization
- Photoreceptor Cells, Vertebrate/metabolism*
- Photoreceptor Cells, Vertebrate/pathology
- Polymerase Chain Reaction
- RNA/genetics*
- Regeneration*
- Retinal Degeneration/genetics*
- Retinal Degeneration/metabolism
- Retinal Degeneration/pathology
- Zebrafish
- PubMed
- 30029251 Full text @ Invest. Ophthalmol. Vis. Sci.
Citation
Coomer, C.E., Morris, A.C. (2018) Capn5 Expression in the Healthy and Regenerating Zebrafish Retina. Investigative ophthalmology & visual science. 59:3643-3654.
Abstract
Purpose Autosomal dominant neovascular inflammatory vitreoretinopathy (ADNIV) is a devastating inherited autoimmune disease of the eye that displays features commonly seen in other eye diseases, such as retinitis pigmentosa and diabetic retinopathy. ADNIV is caused by a gain-of-function mutation in Calpain-5 (CAPN5), a calcium-dependent cysteine protease. Very little is known about the normal function of CAPN5 in the adult retina, and there are conflicting results regarding its role during mammalian embryonic development. The zebrafish (Danio rerio) is an excellent animal model for studying vertebrate development and tissue regeneration, and represents a novel model to explore the function of Capn5 in the eye.
Methods We characterized the expression of Capn5 in the developing zebrafish central nervous system (CNS) and retina, in the adult zebrafish retina, and in response to photoreceptor degeneration and regeneration using whole-mount in situ hybridization, FISH, and immunohistochemistry.
Results In zebrafish, capn5 is strongly expressed in the developing embryonic brain, early optic vesicles, and in newly differentiated retinal photoreceptors. We found that expression of capn5 colocalized with cone-specific markers in the adult zebrafish retina. We observed an increase in expression of Capn5 in a zebrafish model of chronic rod photoreceptor degeneration and regeneration. Acute light damage to the zebrafish retina was accompanied by an increase in expression of Capn5 in the surviving cones and in a subset of Müller glia.
Conclusions These studies suggest that Capn5 may play a role in CNS development, photoreceptor maintenance, and photoreceptor regeneration.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping