An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo
Several inherited human syndromes that severely affect organogenesis and other developmental processes are caused by mutations in replication stress response (RSR) genes. Although the molecular machinery of RSR is conserved, disease-causing mutations in RSR-genes may have distinct tissue-specific ou...
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Frontiers Media S.A.
2021
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oai:doaj.org-article:6ceac0be911f4a9181217141128f9ca32021-11-04T06:47:05ZAn Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo2296-634X10.3389/fcell.2021.731308https://doaj.org/article/6ceac0be911f4a9181217141128f9ca32021-11-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fcell.2021.731308/fullhttps://doaj.org/toc/2296-634XSeveral inherited human syndromes that severely affect organogenesis and other developmental processes are caused by mutations in replication stress response (RSR) genes. Although the molecular machinery of RSR is conserved, disease-causing mutations in RSR-genes may have distinct tissue-specific outcomes, indicating that progenitor cells may differ in their responses to RSR inactivation. Therefore, understanding how different cell types respond to replication stress is crucial to uncover the mechanisms of RSR-related human syndromes. Here, we review the ocular manifestations in RSR-related human syndromes and summarize recent findings investigating the mechanisms of RSR during eye development in vivo. We highlight a remarkable heterogeneity of progenitor cells responses to RSR inactivation and discuss its implications for RSR-related human syndromes.Gabriel E. Matos-RodriguesRodrigo A. P. MartinsFrontiers Media S.A.articlegenome stabilitycell cycleDNA damagechekcpointATRorganogenesisBiology (General)QH301-705.5ENFrontiers in Cell and Developmental Biology, Vol 9 (2021) |
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genome stability cell cycle DNA damage chekcpoint ATR organogenesis Biology (General) QH301-705.5 |
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genome stability cell cycle DNA damage chekcpoint ATR organogenesis Biology (General) QH301-705.5 Gabriel E. Matos-Rodrigues Rodrigo A. P. Martins An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo |
description |
Several inherited human syndromes that severely affect organogenesis and other developmental processes are caused by mutations in replication stress response (RSR) genes. Although the molecular machinery of RSR is conserved, disease-causing mutations in RSR-genes may have distinct tissue-specific outcomes, indicating that progenitor cells may differ in their responses to RSR inactivation. Therefore, understanding how different cell types respond to replication stress is crucial to uncover the mechanisms of RSR-related human syndromes. Here, we review the ocular manifestations in RSR-related human syndromes and summarize recent findings investigating the mechanisms of RSR during eye development in vivo. We highlight a remarkable heterogeneity of progenitor cells responses to RSR inactivation and discuss its implications for RSR-related human syndromes. |
format |
article |
author |
Gabriel E. Matos-Rodrigues Rodrigo A. P. Martins |
author_facet |
Gabriel E. Matos-Rodrigues Rodrigo A. P. Martins |
author_sort |
Gabriel E. Matos-Rodrigues |
title |
An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo |
title_short |
An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo |
title_full |
An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo |
title_fullStr |
An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo |
title_full_unstemmed |
An Eye in the Replication Stress Response: Lessons From Tissue-Specific Studies in vivo |
title_sort |
eye in the replication stress response: lessons from tissue-specific studies in vivo |
publisher |
Frontiers Media S.A. |
publishDate |
2021 |
url |
https://doaj.org/article/6ceac0be911f4a9181217141128f9ca3 |
work_keys_str_mv |
AT gabrielematosrodrigues aneyeinthereplicationstressresponselessonsfromtissuespecificstudiesinvivo AT rodrigoapmartins aneyeinthereplicationstressresponselessonsfromtissuespecificstudiesinvivo AT gabrielematosrodrigues eyeinthereplicationstressresponselessonsfromtissuespecificstudiesinvivo AT rodrigoapmartins eyeinthereplicationstressresponselessonsfromtissuespecificstudiesinvivo |
_version_ |
1718445094332268544 |