Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research

The vertebrate retina is made up of six specialized neuronal cell types and one glia that are generated from a common retinal progenitor. The development of these distinct cell types is programmed by transcription factors that regulate the expression of specific genes essential for cell fate specifi...

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Main Authors: Yiqiao Zheng, Shiming Chen
Format: Article
Language:English
Published: Frontiers Media S.A. 2024-02-01
Series:Frontiers in Cellular Neuroscience
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fncel.2024.1347436/full
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author Yiqiao Zheng
Yiqiao Zheng
Shiming Chen
Shiming Chen
Shiming Chen
author_facet Yiqiao Zheng
Yiqiao Zheng
Shiming Chen
Shiming Chen
Shiming Chen
author_sort Yiqiao Zheng
collection DOAJ
description The vertebrate retina is made up of six specialized neuronal cell types and one glia that are generated from a common retinal progenitor. The development of these distinct cell types is programmed by transcription factors that regulate the expression of specific genes essential for cell fate specification and differentiation. Because of the complex nature of transcriptional regulation, understanding transcription factor functions in development and disease is challenging. Research on the Cone-rod homeobox transcription factor CRX provides an excellent model to address these challenges. In this review, we reflect on 25 years of mammalian CRX research and discuss recent progress in elucidating the distinct pathogenic mechanisms of four CRX coding variant classes. We highlight how in vitro biochemical studies of CRX protein functions facilitate understanding CRX regulatory principles in animal models. We conclude with a brief discussion of the emerging systems biology approaches that could accelerate precision medicine for CRX-linked diseases and beyond.
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spelling doaj.art-e81898d3bbfc47ad88cbf3ec96275caf2024-02-13T04:30:07ZengFrontiers Media S.A.Frontiers in Cellular Neuroscience1662-51022024-02-011810.3389/fncel.2024.13474361347436Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX researchYiqiao Zheng0Yiqiao Zheng1Shiming Chen2Shiming Chen3Shiming Chen4Molecular Genetics and Genomics Graduate Program, Division of Biological and Biomedical Sciences, Saint Louis, MO, United StatesDepartment of Ophthalmology and Visual Sciences, Saint Louis, MO, United StatesMolecular Genetics and Genomics Graduate Program, Division of Biological and Biomedical Sciences, Saint Louis, MO, United StatesDepartment of Ophthalmology and Visual Sciences, Saint Louis, MO, United StatesDepartment of Developmental Biology, Washington University in St. Louis, Saint Louis, MO, United StatesThe vertebrate retina is made up of six specialized neuronal cell types and one glia that are generated from a common retinal progenitor. The development of these distinct cell types is programmed by transcription factors that regulate the expression of specific genes essential for cell fate specification and differentiation. Because of the complex nature of transcriptional regulation, understanding transcription factor functions in development and disease is challenging. Research on the Cone-rod homeobox transcription factor CRX provides an excellent model to address these challenges. In this review, we reflect on 25 years of mammalian CRX research and discuss recent progress in elucidating the distinct pathogenic mechanisms of four CRX coding variant classes. We highlight how in vitro biochemical studies of CRX protein functions facilitate understanding CRX regulatory principles in animal models. We conclude with a brief discussion of the emerging systems biology approaches that could accelerate precision medicine for CRX-linked diseases and beyond.https://www.frontiersin.org/articles/10.3389/fncel.2024.1347436/fullCRXhomeodomaingene regulationmolecular geneticsinherited retinopathydominant diseases
spellingShingle Yiqiao Zheng
Yiqiao Zheng
Shiming Chen
Shiming Chen
Shiming Chen
Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research
Frontiers in Cellular Neuroscience
CRX
homeodomain
gene regulation
molecular genetics
inherited retinopathy
dominant diseases
title Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research
title_full Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research
title_fullStr Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research
title_full_unstemmed Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research
title_short Transcriptional precision in photoreceptor development and diseases – Lessons from 25 years of CRX research
title_sort transcriptional precision in photoreceptor development and diseases lessons from 25 years of crx research
topic CRX
homeodomain
gene regulation
molecular genetics
inherited retinopathy
dominant diseases
url https://www.frontiersin.org/articles/10.3389/fncel.2024.1347436/full
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