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The contribution of each gene in this syndrome has been studied using mutant mouse models, but no single mouse model recapitulates the whole spectrum of human 15q13.3 microdeletion syndrome. The behavior of\u00a0\n                    <jats:italic>Trpm1<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    \u00a0mice has not been investigated in relation to 15q13.3 microdeletion syndrome due to the visual impairment in these mice, which may confound the results of behavioral tests involving vision. We were able to perform a comprehensive behavioral test battery using\n                    <jats:italic>Trpm1<\/jats:italic>\n                    null mutant mice to investigate the role of\n                    <jats:italic>Trpm1<\/jats:italic>\n                    , which is thought to be expressed solely\u00a0in the retina, in the\u00a0central nervous system and to examine the relationship between TRPM1 and 15q13.3 microdeletion syndrome. Our data demonstrate that\u00a0\n                    <jats:italic>Trpm1<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice exhibit abnormal behaviors that may explain some phenotypes of 15q13.3 microdeletion syndrome, including reduced anxiety-like behavior, abnormal social interaction, attenuated fear memory, and the most prominent phenotype of\n                    <jats:italic>Trpm1<\/jats:italic>\n                    mutant mice,\u00a0hyperactivity. While the ON visual transduction pathway is impaired in\n                    <jats:italic>Trpm1<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice, we did not detect compensatory high sensitivities for other sensory modalities. The pathway for visual impairment is the same between\n                    <jats:italic>Trpm1<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice and\n                    <jats:italic>mGluR6<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice, but\u00a0hyperlocomotor activity has not been reported in\n                    <jats:italic>mGluR6<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice. These data suggest that the phenotype of\n                    <jats:italic>Trpm1<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice extends beyond that expected from visual impairment alone. Here, we provide the first evidence associating TRPM1 with impairment of cognitive function similar to that observed in phenotypes of 15q13.3 microdeletion syndrome.\n                  <\/jats:p>","DOI":"10.1186\/s13041-021-00749-y","type":"journal-article","created":{"date-parts":[[2021,3,30]],"date-time":"2021-03-30T05:02:46Z","timestamp":1617080566000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Mice with mutations in Trpm1, a gene in the locus of 15q13.3 microdeletion syndrome, display pronounced hyperactivity and decreased anxiety-like behavior"],"prefix":"10.1186","volume":"14","author":[{"given":"Tesshu","family":"Hori","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shohei","family":"Ikuta","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoko","family":"Hattori","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keizo","family":"Takao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tsuyoshi","family":"Miyakawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chieko","family":"Koike","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,3,30]]},"reference":[{"key":"749_CR1","doi-asserted-by":"publisher","first-page":"332","DOI":"10.1073\/pnas.0912730107","volume":"107","author":"C Koike","year":"2010","unstructured":"Koike C, Obara T, Uriu Y, Numata T, Sanuki R, Miyata K, et al. 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