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In autism and Fragile X Syndrome (FXS), disruptions in pup-dam USV communication hint at a possible connection between abnormal early developmental USV communication and the later emergence of communication and social deficits.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      Here, we gathered USVs from PND 10 FXS pups during a short period of separation from their mothers, encompassing animals of all possible genotypes and both sexes (i.e.,\n                      <jats:italic>Fmr1-\/y<\/jats:italic>\n                      vs.\n                      <jats:italic>Fmr1+\/y<\/jats:italic>\n                      males and\n                      <jats:italic>Fmr1+\/+<\/jats:italic>\n                      ,\n                      <jats:italic>+\/-<\/jats:italic>\n                      , and\n                      <jats:italic>-\/-<\/jats:italic>\n                      females). This allowed comparing the influence of sex and gene dosage on pups\u2019 communication capabilities. Leveraging DeepSqueak and analyzing vocal patterns, intricate vocal behaviors such as call structure, duration, frequency modulation, and temporal patterns were examined. Furthermore, homing behavior was assessed as a sensitive indicator of early cognitive development and social discrimination. This behavior relies on the use of olfactory and thermal cues to navigate and search for the maternal or nest odor in the surrounding space.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      The results show that FMRP-deficient pups of both sexes display an increased inclination to vocalize when separated from their mothers, and this behavior is accompanied by significant sex-specific changes in the main features of their USVs as well as in body weight. Analysis of the vocal repertoire and syntactic usage revealed that\n                      <jats:italic>Fmr1<\/jats:italic>\n                      gene silencing primarily alters the USVs\u2019 qualitative composition in males. Moreover, sex-specific effects of\n                      <jats:italic>Fmr1<\/jats:italic>\n                      silencing on locomotor activity and homing behavior were observed. FMRP deficiency in females increased activity, reduced nest-reaching time, and extended nest time. In males, it prolonged nest-reaching time and reduced nest time without affecting locomotion.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      These findings highlight the interplay between\n                      <jats:italic>Fmr1<\/jats:italic>\n                      gene dosage and sex in influencing communicative and cognitive skills during infancy.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13293-024-00594-3","type":"journal-article","created":{"date-parts":[[2024,2,21]],"date-time":"2024-02-21T07:02:33Z","timestamp":1708498953000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Sex-specific modulation of early life vocalization and cognition by Fmr1 gene dosage in a mouse model of Fragile X Syndrome"],"prefix":"10.1186","volume":"15","author":[{"given":"Gabriele","family":"Giua","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniela","family":"Iezzi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alba","family":"Caceres-Rodriguez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benjamin","family":"Strauss","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pascale","family":"Chavis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5579-6208","authenticated-orcid":false,"given":"Olivier J.","family":"Manzoni","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,2,21]]},"reference":[{"key":"594_CR1","doi-asserted-by":"publisher","first-page":"423","DOI":"10.1016\/B978-0-12-809600-0.00040-8","volume":"25","author":"S Granon","year":"2018","unstructured":"Granon S, Faure A, Chauveau F, Cressant A, Ey E. 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