July 2019
Volume 60, Issue 9
Open Access
ARVO Annual Meeting Abstract  |   July 2019
RNA binding proteins in eye development: rbm24a regulates sox2 and leads to microphthalmia and visual processing defects in zebrafish
Author Affiliations & Notes
  • Lindy Brastrom
    Department of Biology, University of Iowa, Iowa City, Iowa, United States
  • Soma Dash
    Department of Biological Sciences, University of Delaware, Newark, Delaware, United States
  • C. Anthony Scott
    Department of Biology, University of Iowa, Iowa City, Iowa, United States
  • Deborah V. Dawson
    Department of Pediatric Dentistry, University of Iowa, Iowa City, Iowa, United States
  • Salil Anil Lachke
    Department of Biological Sciences, University of Delaware, Newark, Delaware, United States
  • Diane Slusarski
    Department of Biology, University of Iowa, Iowa City, Iowa, United States
  • Footnotes
    Commercial Relationships   Lindy Brastrom, None; Soma Dash, None; C. Scott, None; Deborah Dawson, None; Salil Lachke, None; Diane Slusarski, None
  • Footnotes
    Support  None
Investigative Ophthalmology & Visual Science July 2019, Vol.60, 4952. doi:
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      Lindy Brastrom, Soma Dash, C. Anthony Scott, Deborah V. Dawson, Salil Anil Lachke, Diane Slusarski; RNA binding proteins in eye development: rbm24a regulates sox2 and leads to microphthalmia and visual processing defects in zebrafish. Invest. Ophthalmol. Vis. Sci. 2019;60(9):4952.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose : Developmental eye disorders such as microphthalmia affect roughly 1 in every 7,000 live births. Nearly 20% of these cases are due to a mutation in the proliferative gene, sox2. While transcriptional control of sox2 is well understood, the mechanisms for its post-transcriptional control have not been addressed. Our preliminary data has found the RNA binding protein, rbm24a, regulates sox2. The purpose of our study is to determine the cellular mechanism underlying the microphthalmia phenotype and identify additional RNA targets of rbm24a.

Methods : We use gene knockdown and CRISPR genome editing to reduce rbm24a in zebrafish. We characterize developmental defects, evaluate predicted targets by qPCR, and perform behavioral studies to assess visual acuity.

Results : Knockdown and somatic CRISPR mutation of rbm24a leads to microphthalmia. We find that knockdown of rbm24a leads to a 70% decrease in sox2 expression. Using the vision startle response with software developed in our lab, we show rbm24a morphants are able to detect stark changes in light. However, rbm24a deficient embryos are unable to follow a sinusoidal pattern, suggesting a subtle defect in vision or a defect in visual processing.

Conclusions : Our study highlights a novel mechanism for rbm24a in the post-transcriptional regulation of sox2. Additionally, this work demonstrates the usefulness of the zebrafish as a model for visual function studies.

This abstract was presented at the 2019 ARVO Annual Meeting, held in Vancouver, Canada, April 28 - May 2, 2019.

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