PUBLICATION
Shootin-1 Is Required for Nervous System Development in Zebrafish
- Authors
- Emerson, S.E., Stergas, H.R., Bupp-Chickering, S.O., Ebert, A.M.
- ID
- ZDB-PUB-200515-6
- Date
- 2020
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 249(10): 1285-1295 (Journal)
- Registered Authors
- Keywords
- PlexinA2, Retina, Semaphorin6A, cytoskeleton, microarray, migration
- MeSH Terms
-
- Animals
- Axons/physiology
- Body Patterning
- Cell Movement
- Cytoskeletal Proteins/genetics
- Cytoskeletal Proteins/physiology*
- Gene Expression Regulation, Developmental*
- Humans
- Motor Neurons/metabolism
- Nerve Tissue Proteins/genetics
- Nerve Tissue Proteins/physiology*
- Nervous System/embryology*
- Neurons/metabolism
- Peripheral Nervous System/physiology
- Phenotype
- Retinal Pigment Epithelium/physiology
- Semaphorins/genetics
- Semaphorins/physiology*
- Zebrafish
- Zebrafish Proteins/genetics
- Zebrafish Proteins/physiology*
- PubMed
- 32406957 Full text @ Dev. Dyn.
Citation
Emerson, S.E., Stergas, H.R., Bupp-Chickering, S.O., Ebert, A.M. (2020) Shootin-1 Is Required for Nervous System Development in Zebrafish. Developmental Dynamics : an official publication of the American Association of Anatomists. 249(10):1285-1295.
Abstract
Background Semaphorin6A (Sema6A) and its PlexinA2 (PlxnA2) receptor canonically function as repulsive axon guidance cues. To understand downstream signaling mechanisms, we performed a microarray screen and identified the "clutch molecule" shootin-1 (shtn-1) as a transcriptionally repressed target (Emerson et al., 2017). Shtn-1 is a key proponent of cell migration and neuronal polarization and must be regulated during nervous system development (Toriyama et al., 2006; Shimada et al., 2008; Kubo et al., 2015) The mechanisms of Shtn-1 regulation and the phenotypic consequences of loss of repression are poorly understood.
Results We demonstrate shtn-1 overexpression results in impaired migration of the optic vesicles, lack of retinal pigmented epithelium, and pathfinding errors of retinotectal projections. We also observed patterning defects in the peripheral nervous system. Importantly, these phenotypes were rescued by overexpressing PlxnA2.
Conclusions We demonstrate a functional role for repression of shtn-1 by PlxnA2 in development of the eyes and peripheral nervous system in zebrafish. These results demonstrate that careful regulation of shtn-1 is critical for development of the nervous system. This article is protected by copyright. All rights reserved.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping