Striatal cholinergic interneuron development in models of DYT1 dystonia
Dystonia is a neurodevelopmental disorder characterized by severe involuntary twisting movements, hypothesized to arise from a dysfunctional motor network involving the cortex, basal ganglia, and cerebellum. Within this network, striatal cholinergic interneurons have been identified as possible cont...
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| Format: | Article |
| Language: | English |
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Frontiers Media S.A.
2024-05-01
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| Series: | Dystonia |
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| Online Access: | https://www.frontierspartnerships.org/articles/10.3389/dyst.2024.12413/full |
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| author | Lauren N. Miterko-Myers |
| author_facet | Lauren N. Miterko-Myers |
| author_sort | Lauren N. Miterko-Myers |
| collection | DOAJ |
| description | Dystonia is a neurodevelopmental disorder characterized by severe involuntary twisting movements, hypothesized to arise from a dysfunctional motor network involving the cortex, basal ganglia, and cerebellum. Within this network, striatal cholinergic interneurons have been identified as possible contributors to dystonia pathophysiology. However, little is known about striatal cholinergic interneuron development in the mammalian brain, limiting our understanding of its role in dystonia and therapeutic potential. Here, I review striatal cholinergic interneuron development in the context of early-onset DYT1 (or “DYT-TOR1A”) dystonia. I discuss clinical and laboratory research findings that support cholinergic dysfunction in DYT1 dystonia and the implications of abnormal cholinergic cell development on disease penetrance and striatal connectivity. |
| format | Article |
| id | doaj-art-0037101c6b304c8cb6a6ddaa9ac30184 |
| institution | OA Journals |
| issn | 2813-2106 |
| language | English |
| publishDate | 2024-05-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Dystonia |
| spelling | doaj-art-0037101c6b304c8cb6a6ddaa9ac301842025-08-20T02:23:31ZengFrontiers Media S.A.Dystonia2813-21062024-05-01310.3389/dyst.2024.1241312413Striatal cholinergic interneuron development in models of DYT1 dystoniaLauren N. Miterko-MyersDystonia is a neurodevelopmental disorder characterized by severe involuntary twisting movements, hypothesized to arise from a dysfunctional motor network involving the cortex, basal ganglia, and cerebellum. Within this network, striatal cholinergic interneurons have been identified as possible contributors to dystonia pathophysiology. However, little is known about striatal cholinergic interneuron development in the mammalian brain, limiting our understanding of its role in dystonia and therapeutic potential. Here, I review striatal cholinergic interneuron development in the context of early-onset DYT1 (or “DYT-TOR1A”) dystonia. I discuss clinical and laboratory research findings that support cholinergic dysfunction in DYT1 dystonia and the implications of abnormal cholinergic cell development on disease penetrance and striatal connectivity.https://www.frontierspartnerships.org/articles/10.3389/dyst.2024.12413/fulldevelopmentDYT1striatumearly-onset dystoniacholinergic interneurons |
| spellingShingle | Lauren N. Miterko-Myers Striatal cholinergic interneuron development in models of DYT1 dystonia Dystonia development DYT1 striatum early-onset dystonia cholinergic interneurons |
| title | Striatal cholinergic interneuron development in models of DYT1 dystonia |
| title_full | Striatal cholinergic interneuron development in models of DYT1 dystonia |
| title_fullStr | Striatal cholinergic interneuron development in models of DYT1 dystonia |
| title_full_unstemmed | Striatal cholinergic interneuron development in models of DYT1 dystonia |
| title_short | Striatal cholinergic interneuron development in models of DYT1 dystonia |
| title_sort | striatal cholinergic interneuron development in models of dyt1 dystonia |
| topic | development DYT1 striatum early-onset dystonia cholinergic interneurons |
| url | https://www.frontierspartnerships.org/articles/10.3389/dyst.2024.12413/full |
| work_keys_str_mv | AT laurennmiterkomyers striatalcholinergicinterneurondevelopmentinmodelsofdyt1dystonia |