Shootin-1 is required for nervous system development in zebrafish.

Sarah E Emerson, Helaina R Stergas, Simon O Bupp-Chickering, Alicia M Ebert

Developmental dynamics : an official publication of the American Association of Anatomists 2020 Oct

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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. Shtn-1 is a key proponent of cell migration and neuronal polarization and must be regulated during nervous system development. The mechanisms of Shtn-1 regulation and the phenotypic consequences of loss of repression are poorly understood. 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. 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. © 2020 Wiley Periodicals, Inc.

Citation

Sarah E Emerson, Helaina R Stergas, Simon O Bupp-Chickering, Alicia M Ebert. Shootin-1 is required for nervous system development in zebrafish. Developmental dynamics : an official publication of the American Association of Anatomists. 2020 Oct;249(10):1285-1295