Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish
The zebrafish is a powerful model to investigate the developmental roles of electrical synapses because many signaling pathways that regulate the development of the nervous system are highly conserved from fish to humans. Here, we provide evidence linking the mammalian connexin-36 (Cx36) ortholog gj...
| Main Authors: | , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Frontiers Media S.A.
2023-03-01
|
| Series: | Frontiers in Cell and Developmental Biology |
| Subjects: | |
| Online Access: | https://www.frontiersin.org/articles/10.3389/fcell.2023.1150273/full |
| _version_ | 1811161904092020736 |
|---|---|
| author | Cherie A. Brown-Panton Cherie A. Brown-Panton Shiva Sabour Georg S. O. Zoidl Georg S. O. Zoidl Christiane Zoidl Christiane Zoidl Nima Tabatabaei Nima Tabatabaei Georg R. Zoidl Georg R. Zoidl Georg R. Zoidl |
| author_facet | Cherie A. Brown-Panton Cherie A. Brown-Panton Shiva Sabour Georg S. O. Zoidl Georg S. O. Zoidl Christiane Zoidl Christiane Zoidl Nima Tabatabaei Nima Tabatabaei Georg R. Zoidl Georg R. Zoidl Georg R. Zoidl |
| author_sort | Cherie A. Brown-Panton |
| collection | DOAJ |
| description | The zebrafish is a powerful model to investigate the developmental roles of electrical synapses because many signaling pathways that regulate the development of the nervous system are highly conserved from fish to humans. Here, we provide evidence linking the mammalian connexin-36 (Cx36) ortholog gjd2b/Cx35.1, a major component of electrical synapses in the zebrafish, with a refractive error in the context of morphological, molecular, and behavioral changes of zebrafish larvae. Two abnormalities were identified. The optical coherence tomography analysis of the adult retina confirmed changes to the refractive properties caused by eye axial length reduction, leading to hyperopic shifts. The gjd2b/Cx35.1 depletion was also correlated with morphological changes to the head and body ratios in larvae. The differential expression of Wnt/ß-catenin signaling genes, connexins, and dopamine receptors suggested a contribution to the observed phenotypic differences. The alteration of visual-motor behavioral responses to abrupt light transitions was aggravated in larvae, providing evidence that cone photoreceptor cell activity was enhanced when gjd2b/Cx35.1 was depleted. The visual disturbances were reversed under low light conditions in gjd2b−/−/Cx35.1−/− larvae. Since qRT-PCR data demonstrated that two rhodopsin genes were downregulated, we speculated that rod photoreceptor cells in gjd2b/Cx35.1−/− larvae were less sensitive to bright light transitions, thus providing additional evidence that a cone-mediated process caused the VMR light-ON hyperactivity after losing Cx35.1 expression. Together, this study provides evidence for the role of gjd2b/Cx35.1 in the development of the visual system and visually guided behaviors. |
| first_indexed | 2024-04-10T06:21:32Z |
| format | Article |
| id | doaj.art-4e88344ea87b483b979612b7cbbd219c |
| institution | Directory Open Access Journal |
| issn | 2296-634X |
| language | English |
| last_indexed | 2024-04-10T06:21:32Z |
| publishDate | 2023-03-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Cell and Developmental Biology |
| spelling | doaj.art-4e88344ea87b483b979612b7cbbd219c2023-03-02T04:47:34ZengFrontiers Media S.A.Frontiers in Cell and Developmental Biology2296-634X2023-03-011110.3389/fcell.2023.11502731150273Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafishCherie A. Brown-Panton0Cherie A. Brown-Panton1Shiva Sabour2Georg S. O. Zoidl3Georg S. O. Zoidl4Christiane Zoidl5Christiane Zoidl6Nima Tabatabaei7Nima Tabatabaei8Georg R. Zoidl9Georg R. Zoidl10Georg R. Zoidl11Department of Biology, York University, Toronto, ON, CanadaCenter for Vision Research, York University, Toronto, ON, CanadaDepartment of Mechanical Engineering, York University, Toronto, ON, CanadaDepartment of Biology, York University, Toronto, ON, CanadaCenter for Vision Research, York University, Toronto, ON, CanadaDepartment of Biology, York University, Toronto, ON, CanadaCenter for Vision Research, York University, Toronto, ON, CanadaDepartment of Mechanical Engineering, York University, Toronto, ON, CanadaCenter for Vision Research, York University, Toronto, ON, CanadaDepartment of Biology, York University, Toronto, ON, CanadaDepartment of Psychology, York University, Toronto, ON, CanadaCenter for Vision Research, York University, Toronto, ON, CanadaThe zebrafish is a powerful model to investigate the developmental roles of electrical synapses because many signaling pathways that regulate the development of the nervous system are highly conserved from fish to humans. Here, we provide evidence linking the mammalian connexin-36 (Cx36) ortholog gjd2b/Cx35.1, a major component of electrical synapses in the zebrafish, with a refractive error in the context of morphological, molecular, and behavioral changes of zebrafish larvae. Two abnormalities were identified. The optical coherence tomography analysis of the adult retina confirmed changes to the refractive properties caused by eye axial length reduction, leading to hyperopic shifts. The gjd2b/Cx35.1 depletion was also correlated with morphological changes to the head and body ratios in larvae. The differential expression of Wnt/ß-catenin signaling genes, connexins, and dopamine receptors suggested a contribution to the observed phenotypic differences. The alteration of visual-motor behavioral responses to abrupt light transitions was aggravated in larvae, providing evidence that cone photoreceptor cell activity was enhanced when gjd2b/Cx35.1 was depleted. The visual disturbances were reversed under low light conditions in gjd2b−/−/Cx35.1−/− larvae. Since qRT-PCR data demonstrated that two rhodopsin genes were downregulated, we speculated that rod photoreceptor cells in gjd2b/Cx35.1−/− larvae were less sensitive to bright light transitions, thus providing additional evidence that a cone-mediated process caused the VMR light-ON hyperactivity after losing Cx35.1 expression. Together, this study provides evidence for the role of gjd2b/Cx35.1 in the development of the visual system and visually guided behaviors.https://www.frontiersin.org/articles/10.3389/fcell.2023.1150273/fullelectrical synapseszebrafishconnexinCx36eyerefractive error |
| spellingShingle | Cherie A. Brown-Panton Cherie A. Brown-Panton Shiva Sabour Georg S. O. Zoidl Georg S. O. Zoidl Christiane Zoidl Christiane Zoidl Nima Tabatabaei Nima Tabatabaei Georg R. Zoidl Georg R. Zoidl Georg R. Zoidl Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish Frontiers in Cell and Developmental Biology electrical synapses zebrafish connexin Cx36 eye refractive error |
| title | Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish |
| title_full | Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish |
| title_fullStr | Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish |
| title_full_unstemmed | Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish |
| title_short | Gap junction Delta-2b (gjd2b/Cx35.1) depletion causes hyperopia and visual-motor deficiencies in the zebrafish |
| title_sort | gap junction delta 2b gjd2b cx35 1 depletion causes hyperopia and visual motor deficiencies in the zebrafish |
| topic | electrical synapses zebrafish connexin Cx36 eye refractive error |
| url | https://www.frontiersin.org/articles/10.3389/fcell.2023.1150273/full |
| work_keys_str_mv | AT cherieabrownpanton gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT cherieabrownpanton gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT shivasabour gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT georgsozoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT georgsozoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT christianezoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT christianezoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT nimatabatabaei gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT nimatabatabaei gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT georgrzoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT georgrzoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish AT georgrzoidl gapjunctiondelta2bgjd2bcx351depletioncauseshyperopiaandvisualmotordeficienciesinthezebrafish |