Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models
BackgroundNeurexins, essential synaptic proteins, are linked to neurodevelopmental and neuropsychiatric disorders like autism spectrum disorder (ASD) and schizophrenia.ObjectiveThrough this systematic review, we aimed to shed light on the relationship between neurexin dysfunction and its implication...
| Main Authors: | , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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Frontiers Media S.A.
2024-02-01
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| Series: | Frontiers in Behavioral Neuroscience |
| Subjects: | |
| Online Access: | https://www.frontiersin.org/articles/10.3389/fnbeh.2024.1297374/full |
| _version_ | 1797323714063237120 |
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| author | Dan Shan Dan Shan Yuming Song Yanyi Zhang Cheong Wong Ho Wenxin Xia Zhi Li Fenfen Ge Qifeng Ou Zijie Dai Zhihao Dai |
| author_facet | Dan Shan Dan Shan Yuming Song Yanyi Zhang Cheong Wong Ho Wenxin Xia Zhi Li Fenfen Ge Qifeng Ou Zijie Dai Zhihao Dai |
| author_sort | Dan Shan |
| collection | DOAJ |
| description | BackgroundNeurexins, essential synaptic proteins, are linked to neurodevelopmental and neuropsychiatric disorders like autism spectrum disorder (ASD) and schizophrenia.ObjectiveThrough this systematic review, we aimed to shed light on the relationship between neurexin dysfunction and its implications in neurodevelopmental and neuropsychiatric manifestations. Both animal and human-induced pluripotent stem cell (hiPSC) models served as our primary investigative platforms.MethodsUtilizing the PRISMA 2020 guidelines, our search strategy involved scouring articles from the PubMed and Google Scholar databases covering a span of two decades (2003–2023). Of the initial collection, 27 rigorously evaluated studies formed the essence of our review.ResultsOur review suggested the significant ties between neurexin anomalies and neurodevelopmental and neuropsychiatric outcomes, most notably ASD. Rodent-based investigations delineated pronounced ASD-associated behaviors, and hiPSC models derived from ASD-diagnosed patients revealed the disruptions in calcium dynamics and synaptic activities. Additionally, our review underlined the integral role of specific neurexin variants, primarily NRXN1, in the pathology of schizophrenia. It was also evident from our observation that neurexin malfunctions were implicated in a broader array of these disorders, including ADHD, intellectual challenges, and seizure disorders.ConclusionThis review accentuates the cardinal role neurexins play in the pathological process of neurodevelopmental and neuropsychiatric disorders. The findings underscore a critical need for standardized methodologies in developing animal and hiPSC models for future studies, aiming to minimize heterogeneity. Moreover, we highlight the need to expand research into less studied neurexin variants (i.e., NRXN2 and NRXN3), broadening the scope of our understanding in this field. Our observation also projects hiPSC models as potent tools for bridging research gaps, promoting translational research, and fostering the development of patient-specific therapeutic interventions. |
| first_indexed | 2024-03-08T05:32:26Z |
| format | Article |
| id | doaj.art-cf34da3109254d9bbc753a60d34364c7 |
| institution | Directory Open Access Journal |
| issn | 1662-5153 |
| language | English |
| last_indexed | 2024-03-08T05:32:26Z |
| publishDate | 2024-02-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Behavioral Neuroscience |
| spelling | doaj.art-cf34da3109254d9bbc753a60d34364c72024-02-06T04:28:11ZengFrontiers Media S.A.Frontiers in Behavioral Neuroscience1662-51532024-02-011810.3389/fnbeh.2024.12973741297374Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal modelsDan Shan0Dan Shan1Yuming Song2Yanyi Zhang3Cheong Wong Ho4Wenxin Xia5Zhi Li6Fenfen Ge7Qifeng Ou8Zijie Dai9Zhihao Dai10Department of Biobehavioral Sciences, Columbia University, New York, NY, United StatesFaculty of Health and Medicine, Lancaster University, Lancaster, United KingdomSchool of Medical Imaging, Hebei Medical University, Shijiazhuang, ChinaSchool of Medicine, Shanghai Jiao Tong University, Shanghai, ChinaSchool of Medicine, University of Galway, Galway, IrelandSchool of Medicine, University of Galway, Galway, IrelandCollege of Health, Medicine and Wellbeing, University of Newcastle, Newcastle, NSW, AustraliaFaculty of Medicine, University of Iceland, Reykjavík, IcelandSchool of Medicine, University of Galway, Galway, IrelandDivision of Biosciences, Faculty of Life Sciences, University College London, London, United KingdomSchool of Medicine, Royal College of Surgeons in Ireland, Dublin, IrelandBackgroundNeurexins, essential synaptic proteins, are linked to neurodevelopmental and neuropsychiatric disorders like autism spectrum disorder (ASD) and schizophrenia.ObjectiveThrough this systematic review, we aimed to shed light on the relationship between neurexin dysfunction and its implications in neurodevelopmental and neuropsychiatric manifestations. Both animal and human-induced pluripotent stem cell (hiPSC) models served as our primary investigative platforms.MethodsUtilizing the PRISMA 2020 guidelines, our search strategy involved scouring articles from the PubMed and Google Scholar databases covering a span of two decades (2003–2023). Of the initial collection, 27 rigorously evaluated studies formed the essence of our review.ResultsOur review suggested the significant ties between neurexin anomalies and neurodevelopmental and neuropsychiatric outcomes, most notably ASD. Rodent-based investigations delineated pronounced ASD-associated behaviors, and hiPSC models derived from ASD-diagnosed patients revealed the disruptions in calcium dynamics and synaptic activities. Additionally, our review underlined the integral role of specific neurexin variants, primarily NRXN1, in the pathology of schizophrenia. It was also evident from our observation that neurexin malfunctions were implicated in a broader array of these disorders, including ADHD, intellectual challenges, and seizure disorders.ConclusionThis review accentuates the cardinal role neurexins play in the pathological process of neurodevelopmental and neuropsychiatric disorders. The findings underscore a critical need for standardized methodologies in developing animal and hiPSC models for future studies, aiming to minimize heterogeneity. Moreover, we highlight the need to expand research into less studied neurexin variants (i.e., NRXN2 and NRXN3), broadening the scope of our understanding in this field. Our observation also projects hiPSC models as potent tools for bridging research gaps, promoting translational research, and fostering the development of patient-specific therapeutic interventions.https://www.frontiersin.org/articles/10.3389/fnbeh.2024.1297374/fullanimal modelshuman induced pluripotent stem cellsdisease modelingneurexinsneuropsychiatric diseases |
| spellingShingle | Dan Shan Dan Shan Yuming Song Yanyi Zhang Cheong Wong Ho Wenxin Xia Zhi Li Fenfen Ge Qifeng Ou Zijie Dai Zhihao Dai Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models Frontiers in Behavioral Neuroscience animal models human induced pluripotent stem cells disease modeling neurexins neuropsychiatric diseases |
| title | Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models |
| title_full | Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models |
| title_fullStr | Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models |
| title_full_unstemmed | Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models |
| title_short | Neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders: a PRIMSA-based systematic review through iPSC and animal models |
| title_sort | neurexin dysfunction in neurodevelopmental and neuropsychiatric disorders a primsa based systematic review through ipsc and animal models |
| topic | animal models human induced pluripotent stem cells disease modeling neurexins neuropsychiatric diseases |
| url | https://www.frontiersin.org/articles/10.3389/fnbeh.2024.1297374/full |
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