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However, it remains elusive whether <jats:italic>RTL1<\/jats:italic> is also involved in their neurological symptoms, such as behavioral and developmental delay\/intellectual disability, feeding difficulties, motor delay, and delayed speech. Here, we demonstrate that the mouse RTL1 protein is widely expressed in the central nervous system (CNS), including the limbic system. Importantly, two disease model mice with over\u2010 and under\u2010expression of <jats:italic>Rtl1<\/jats:italic> exhibited reduced locomotor activity, increased anxiety, and impaired amygdala\u2010dependent cued fear, demonstrating that <jats:italic>Rtl1<\/jats:italic> also plays an important role in the CNS. These results indicate that the KOS14 and TS14 are neuromuscular as well as neuropsychiatric diseases caused by irregular CNS <jats:italic>RTL1<\/jats:italic> expression, presumably leading to impaired innervation of motor neurons to skeletal muscles as well as malfunction of the hippocampus\u2010amygdala complex. It is of considerable interest that eutherian\u2010specific <jats:italic>RTL1<\/jats:italic> is expressed in mammalian\u2010 and eutherian\u2010specific brain structures, that is, the corticospinal tract and corpus callosum, respectively, suggesting that <jats:italic>RTL1<\/jats:italic> might have contributed to the acquisition of both these structures themselves and fine motor skill in eutherian brain evolution.<\/jats:p>","DOI":"10.1111\/gtc.12830","type":"journal-article","created":{"date-parts":[[2021,1,23]],"date-time":"2021-01-23T20:20:00Z","timestamp":1611433200000},"page":"165-179","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":40,"title":["The role of eutherian\u2010specific <i>RTL1<\/i> in the nervous system and its implications for the Kagami\u2010Ogata and Temple syndromes"],"prefix":"10.1111","volume":"26","author":[{"given":"Moe","family":"Kitazawa","sequence":"first","affiliation":[{"name":"Department of 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