Current trends of high-risk gene Cul3 in neurodevelopmental disorders
Cul3 encodes Cullin-3, a core component of the ubiquitin E3 ligase that is involved in protein ubiquitination. Recent studies have identified Cul3 as a high-confidence risk gene in neurodevelopmental disorders (NDDs), especially autism spectrum disorder (ASD). Different strategies have been used to...
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| Format: | Article |
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Frontiers Media S.A.
2023-07-01
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| Series: | Frontiers in Psychiatry |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fpsyt.2023.1215110/full |
| _version_ | 1797770460999450624 |
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| author | Ping Lin Jie Yang Shumin Wu Tong Ye Wenting Zhuang Wei Wang Wei Wang Tao Tan |
| author_facet | Ping Lin Jie Yang Shumin Wu Tong Ye Wenting Zhuang Wei Wang Wei Wang Tao Tan |
| author_sort | Ping Lin |
| collection | DOAJ |
| description | Cul3 encodes Cullin-3, a core component of the ubiquitin E3 ligase that is involved in protein ubiquitination. Recent studies have identified Cul3 as a high-confidence risk gene in neurodevelopmental disorders (NDDs), especially autism spectrum disorder (ASD). Different strategies have been used to generate animal models with Cul3 deficiency in the central nervous system, including whole-brain knockout (KO), cell-type specific conditional KO (cKO), and brain region-specific knockdown. In this review, we revisited the basic properties of CUL3 and its function under physiological and pathological conditions. Recent clinical studies including case reports and large cohort sequencing studies related to CUl3 in NDDs have been summarized. Moreover, we characterized the behavioral, electrophysiological, and molecular changes in newly developed Cul3 deficiency models. This would guide further studies related to Cul3 in CNS and provide potential therapeutic targets for Cul3-deficiency-induced NDDs, including ASD. |
| first_indexed | 2024-03-12T21:23:37Z |
| format | Article |
| id | doaj.art-57d35a4046344e6ca3c107d3990aa14b |
| institution | Directory Open Access Journal |
| issn | 1664-0640 |
| language | English |
| last_indexed | 2024-03-12T21:23:37Z |
| publishDate | 2023-07-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Psychiatry |
| spelling | doaj.art-57d35a4046344e6ca3c107d3990aa14b2023-07-28T11:59:37ZengFrontiers Media S.A.Frontiers in Psychiatry1664-06402023-07-011410.3389/fpsyt.2023.12151101215110Current trends of high-risk gene Cul3 in neurodevelopmental disordersPing Lin0Jie Yang1Shumin Wu2Tong Ye3Wenting Zhuang4Wei Wang5Wei Wang6Tao Tan7Oujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaOujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaOujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaOujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaOujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaOujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaDepartment of Neuroscience, Baylor College of Medicine, Houston, TX, United StatesOujiang Laboratory (Zhejiang Lab for Regenerative Medicine, Vision and Brain Health), Key Laboratory of Alzheimer's Disease of Zhejiang Province, Institute of Aging, Wenzhou Medical University, Wenzhou, Zhejiang, ChinaCul3 encodes Cullin-3, a core component of the ubiquitin E3 ligase that is involved in protein ubiquitination. Recent studies have identified Cul3 as a high-confidence risk gene in neurodevelopmental disorders (NDDs), especially autism spectrum disorder (ASD). Different strategies have been used to generate animal models with Cul3 deficiency in the central nervous system, including whole-brain knockout (KO), cell-type specific conditional KO (cKO), and brain region-specific knockdown. In this review, we revisited the basic properties of CUL3 and its function under physiological and pathological conditions. Recent clinical studies including case reports and large cohort sequencing studies related to CUl3 in NDDs have been summarized. Moreover, we characterized the behavioral, electrophysiological, and molecular changes in newly developed Cul3 deficiency models. This would guide further studies related to Cul3 in CNS and provide potential therapeutic targets for Cul3-deficiency-induced NDDs, including ASD.https://www.frontiersin.org/articles/10.3389/fpsyt.2023.1215110/fullCul3Cullin3-RING E3 ubiquitin ligasesneurodevelopmental disorders (NDDs)autism spectrum disorder (ASD)knockout (KO) mice |
| spellingShingle | Ping Lin Jie Yang Shumin Wu Tong Ye Wenting Zhuang Wei Wang Wei Wang Tao Tan Current trends of high-risk gene Cul3 in neurodevelopmental disorders Frontiers in Psychiatry Cul3 Cullin3-RING E3 ubiquitin ligases neurodevelopmental disorders (NDDs) autism spectrum disorder (ASD) knockout (KO) mice |
| title | Current trends of high-risk gene Cul3 in neurodevelopmental disorders |
| title_full | Current trends of high-risk gene Cul3 in neurodevelopmental disorders |
| title_fullStr | Current trends of high-risk gene Cul3 in neurodevelopmental disorders |
| title_full_unstemmed | Current trends of high-risk gene Cul3 in neurodevelopmental disorders |
| title_short | Current trends of high-risk gene Cul3 in neurodevelopmental disorders |
| title_sort | current trends of high risk gene cul3 in neurodevelopmental disorders |
| topic | Cul3 Cullin3-RING E3 ubiquitin ligases neurodevelopmental disorders (NDDs) autism spectrum disorder (ASD) knockout (KO) mice |
| url | https://www.frontiersin.org/articles/10.3389/fpsyt.2023.1215110/full |
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