{"title":"Proline-Rich Transmembrane Protein 2 Is Variably Expressed Across Excitatory and Inhibitory Neurons in Mouse Motor Circuits","authors":"Daisuke Hatta, Kaori Watanabe, Akira Kinoshita, Koh-Ichiro Yoshiura, Naohiro Kurotaki, Keiro Shirotani, Nobuhisa Iwata","doi":"10.1002/cne.70176","DOIUrl":"10.1002/cne.70176","url":null,"abstract":"<p>Proline-rich transmembrane protein 2 (PRRT2) plays a pivotal role in the control of voluntary movements, as <i>PRRT2</i> mutations cause paroxysmal kinesigenic dyskinesia (PKD) in a loss-of-function manner. Although the cerebellum is considered a region responsible for PKD, we recently reported that Prrt2 also regulates dopaminergic activity in the striatum, suggesting that Prrt2 functions not only in the cerebellum but also in the basal ganglia motor circuits. However, the relationship between neuronal cell types expressing Prrt2 and motor functions remains poorly understood. In this study, we determined the neurochemical types of Prrt2-positive neurons using immunofluorescence staining of mouse midbrain primary neurons and brain sections. Prrt2 was expressed mainly in glutamatergic and GABAergic neurons, but not in dopaminergic or cholinergic neurons. We found that Prrt2 was expressed preferentially in Vglut1-positive, rather than Vglut2-positive, cortical projection neurons and cerebellar granule cells, and in GABAergic medium spiny neurons of the basal ganglia, where Prrt2 was localized in axonal tracts and at or near presynaptic terminals. Taken together, we conclude that Prrt2 is variably expressed across excitatory and inhibitory neurons in motor-related neural circuits, where it might play more diverse roles in the regulation of neuronal excitability and voluntary movement.</p>","PeriodicalId":15552,"journal":{"name":"Journal of Comparative Neurology","volume":"534 6","pages":""},"PeriodicalIF":2.1,"publicationDate":"2026-06-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cne.70176","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148163324","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Daniel Lozano, Adrián Chinarro, Nerea Moreno, Jesús M. López
{"title":"Brain Distribution of Orthopedia (Otp) Transcription Factor in Bony Fish: A Comparative Neuroanatomical Perspective","authors":"Daniel Lozano, Adrián Chinarro, Nerea Moreno, Jesús M. López","doi":"10.1002/cne.70174","DOIUrl":"https://doi.org/10.1002/cne.70174","url":null,"abstract":"<div>\u0000 \u0000 <p>The transcription factor Orthopedia (Otp) plays a key role in neuronal differentiation, regional specification, and long-term maintenance of neuronal identity across vertebrates. Although Otp expression is widely used as a landmark for hypothalamic regionalization, its distribution in bony fishes has remained poorly characterized. To assess the evolutionary conservation of Otp expression, we analyzed its immunohistochemical distribution in the central nervous system of representative species from all major clades of bony fishes: cladistians, chondrosteans, holosteans, teleosts, and lungfish. To refine anatomical localization and evaluate potential cellular coexpression, we additionally examined markers including calbindin, tyrosine hydroxylase (TH), and the transcription factors Islet-1, Pax7, and Satb1/2. Otp-immunoreactive neurons were consistently observed in the medial amygdala, preoptic area, paraventricular region, all basal hypothalamic domains except the mamillary region, as well as in a population in alar rhombomere 1, the dorsal interpeduncular nucleus, central gray, reticular formation, and the dorsal horn of the spinal cord. Notably, Otp-positive cells were detected in the posterior tubercle only in actinopterygian fishes, in contrast to lungfish. Overall, this Otp expression pattern is highly conserved in relation to that reported in cartilaginous fishes and tetrapods. This study closes a major phylogenetic gap and demonstrates that the brain-wide expression pattern of Otp is largely conserved across vertebrates, supporting its fundamental and ancient role in neuronal differentiation and cell-type specification in forebrain and hindbrain.</p>\u0000 </div>","PeriodicalId":15552,"journal":{"name":"Journal of Comparative Neurology","volume":"534 6","pages":""},"PeriodicalIF":2.1,"publicationDate":"2026-06-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148153601","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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