{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,8]],"date-time":"2026-09-08T22:33:26Z","timestamp":1788906806922,"version":"build-2803163510"},"reference-count":80,"publisher":"American Society for Clinical Investigation","issue":"7","license":[{"start":{"date-parts":[[2026,4,1]],"date-time":"2026-04-01T00:00:00Z","timestamp":1775001600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Radboudumc personal PhD fellowship","award":["n\/a"],"award-info":[{"award-number":["n\/a"]}]},{"name":"travelling fellowship by The Company of Biologists","award":["DMMTF1908278"],"award-info":[{"award-number":["DMMTF1908278"]}]},{"name":"Radboudumc junior researcher fellowship","award":["n\/a"],"award-info":[{"award-number":["n\/a"]}]},{"name":"Radboudumc junior researcher fellowship","award":["n\/a"],"award-info":[{"award-number":["n\/a"]}]},{"name":"Netherlands Organisation for Scientific Research (NWO) ZonMw Veni","award":["09150162010073"],"award-info":[{"award-number":["09150162010073"]}]},{"name":"Netherlands Organisation for Scientific Research (NWO) ZonMw Vidi","award":["91718310"],"award-info":[{"award-number":["91718310"]}]},{"name":"Netherlands Organisation for Scientific Research (NWO) ZonMw Vici","award":["09150181910022"],"award-info":[{"award-number":["09150181910022"]}]},{"name":"Max Planck Society","award":["n\/a"],"award-info":[{"award-number":["n\/a"]}]},{"name":"Instituto de Salud Carlos III (Madrid, Spain) through the Miguel Servet Program, co-funded by the European Union under the European Social Fund \u2018Investing in your future\u2019","award":["CP24\/00033"],"award-info":[{"award-number":["CP24\/00033"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,4,1]]},"abstract":"<jats:p>\n                    Sleep disturbances are among the most prevalent clinical features of FOXP1 syndrome, yet their nature and underlying mechanisms remain unclear. Here, we report that individuals with FOXP1 syndrome suffer from insomnia with sleep maintenance problems and early waking. Consistently, common variants in\n                    <jats:italic>FOXP<\/jats:italic>\n                    genes were associated with insomnia symptoms and short sleep. These sleep disturbances were recapitulated in\n                    <jats:italic>Drosophila<\/jats:italic>\n                    <jats:italic>FoxP<\/jats:italic>\n                    mutants, which exhibit severely fragmented and reduced sleep.\n                    <jats:italic>FoxP<\/jats:italic>\n                    loss also led to circadian arrhythmicity and impaired the plasticity of neuropeptide pigment dispersing factor\u2013secreting (PDF-secreting) neurons in a non-cell-autonomous manner.\n                    <jats:italic>FoxP<\/jats:italic>\n                    was required during development for adult sleep integrity, particularly in peptidergic neurons. Transcriptomic analyses revealed a dysregulation of genes involved in peptidergic signaling, including\n                    <jats:italic>hugin<\/jats:italic>\n                    .\n                    <jats:italic>FoxP<\/jats:italic>\n                    was expressed in hugin\n                    <jats:sup>+<\/jats:sup>\n                    neurons (afferent to PDF-secreting neurons) during development, and its knockdown in these cells was sufficient to induce sleep fragmentation. Our findings establish an evolutionarily conserved role for FOXP proteins in the peptidergic regulation of sleep.\n                  <\/jats:p>","DOI":"10.1172\/jci193475","type":"journal-article","created":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T18:00:57Z","timestamp":1774980057000},"source":"Crossref","is-referenced-by-count":3,"title":["Conserved sleep disturbances in FOXP1 syndrome originate from developmental dysregulation of peptidergic signaling"],"prefix":"10.1172","volume":"136","author":[{"given":"Mireia","family":"Coll-Tan\u00e9","sequence":"first","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilse","family":"Eidhof","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jie","family":"Han","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicholas","family":"Raun","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lara V.","family":"van Renssen","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3132-1996","authenticated-orcid":true,"given":"Simon E.","family":"Fisher","sequence":"additional","affiliation":[{"name":"Language and Genetics Department, Max Planck Institute for Psycholinguistics, Nijmegen, Netherlands."},{"name":"Donders Centre for Neuroscience, Donders Institute for Brain, Cognition, and Behaviour, Radboud University, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew S.","family":"Kayser","sequence":"additional","affiliation":[{"name":"Departments of Psychiatry and Neuroscience, Chronobiology and Sleep Institute, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tjitske","family":"Kleefstra","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Department of Clinical Genetics, Erasmus University Rotterdam, Rotterdam, Netherlands."},{"name":"Center of Excellence for Neuropsychiatry, Vincent van Gogh Institute for Psychiatry, Venray, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sigrid","family":"Pillen","sequence":"additional","affiliation":[{"name":"Kinderslaapexpert BV (Pediatric Sleep Expert Ltd.), Mook, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caitlin M.","family":"Hudac","sequence":"additional","affiliation":[{"name":"Department of Psychology and"},{"name":"Carolina Autism and Neurodevelopment Research Center, University of South Carolina, Columbia, South Carolina, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3788-3815","authenticated-orcid":true,"given":"Jordi","family":"Mayneris-Perxachs","sequence":"additional","affiliation":[{"name":"Nutrition, Eumetabolism and Health Group and Integrative Systems Medicine and Biology Group, Girona Biomedical Research Institute (IDIBGI-CERCA), Girona, Spain."},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red de Fisiopatolog\u00eda de la Obesidad y Nutrici\u00f3n (CIBEROBN), Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marieke","family":"Klein","sequence":"additional","affiliation":[{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."},{"name":"Department of Medical Neuroscience, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saskia","family":"Koene","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Castells-Nobau","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."},{"name":"Nutrition, Eumetabolism and Health Group and Integrative Systems Medicine and Biology Group, Girona Biomedical Research Institute (IDIBGI-CERCA), Girona, Spain."},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red de Fisiopatolog\u00eda de la Obesidad y Nutrici\u00f3n (CIBEROBN), Madrid, Spain."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annette","family":"Schenck","sequence":"additional","affiliation":[{"name":"Department of Human Genetics and"},{"name":"Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, Netherlands."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"232","published-online":{"date-parts":[[2026,4,1]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.1038\/s41582-024-01052-9"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1016\/j.smrv.2009.02.003"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1093\/sleep\/zsaa040"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.1038\/s41386-019-0409-5"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.1126\/sciadv.abe2626"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.1126\/sciadv.abe2597"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1002\/jnr.24619"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1002\/wdev.375"},{"key":"B9","doi-asserted-by":"publisher","DOI":"10.1186\/s11689-021-09358-1"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.14955"},{"key":"B11","doi-asserted-by":"crossref","first-page":"153","DOI":"10.1016\/bs.irrdd.2016.07.005","article-title":"Sleep in neurodevelopmental disorders","volume":"51","author":"Esbensen","year":"2016","journal-title":"Int Rev Res Dev Disabil"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1136\/jmg-2023-109537"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.1136\/jmg-2022-108734"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1038\/s41436-020-01016-6"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.24.2.809-822.2004"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddy035"},{"key":"B17","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2010.10.006"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2015.09.016"},{"key":"B19","doi-asserted-by":"publisher","DOI":"10.1016\/j.biopsych.2019.07.020"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1016\/S0002-7138(09)60942-0"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.1097\/00004703-198402000-00002"},{"key":"B22","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2869.1998.00107.x"},{"key":"B23"},{"key":"B24","doi-asserted-by":"publisher","DOI":"10.1038\/s41588-019-0481-0"},{"key":"B25","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2023.04.009"},{"key":"B26","doi-asserted-by":"publisher","DOI":"10.1093\/molbev\/msq182"},{"key":"B27","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0211652"},{"key":"B28","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M207174200"},{"key":"B29","doi-asserted-by":"publisher","DOI":"10.1242\/dev.118.2.401"},{"key":"B30","doi-asserted-by":"publisher","DOI":"10.1038\/nature05954"},{"key":"B31","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbr.2014.04.009"},{"key":"B32","doi-asserted-by":"publisher","DOI":"10.1126\/stke.2202004pl6"},{"key":"B33","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(00)81676-1"},{"key":"B34","doi-asserted-by":"publisher","DOI":"10.1038\/nature02935"},{"key":"B35","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.0060069"},{"key":"B36","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2020.06.009"},{"key":"B37","doi-asserted-by":"publisher","DOI":"10.1177\/0748730412440667"},{"key":"B38","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2019.08.079"},{"key":"B39","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1917573117"},{"key":"B40","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2013.04.002"},{"key":"B41","doi-asserted-by":"publisher","DOI":"10.1126\/science.1252114"},{"key":"B42","doi-asserted-by":"publisher","DOI":"10.1111\/j.1471-4159.2010.07130.x"},{"key":"B43","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1000967"},{"key":"B44","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.0040068"},{"key":"B45","doi-asserted-by":"publisher","DOI":"10.1098\/rsob.200295"},{"key":"B46","doi-asserted-by":"publisher","DOI":"10.1126\/science.aap8809"},{"key":"B47","doi-asserted-by":"publisher","DOI":"10.1038\/s41586-019-1506-7"},{"key":"B48","doi-asserted-by":"publisher","DOI":"10.1016\/j.smrv.2023.101814"},{"key":"B49","doi-asserted-by":"publisher","DOI":"10.1016\/j.sleh.2017.10.011"},{"key":"B50","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.2011.02204.x"},{"key":"B51","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2008.10.042"},{"key":"B52","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2008.08.033"},{"key":"B53","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2010.02.044"},{"key":"B54","doi-asserted-by":"publisher","DOI":"10.1038\/nature19097"},{"key":"B55","doi-asserted-by":"publisher","DOI":"10.1126\/sciadv.adm7039"},{"key":"B56","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2023.25152"},{"key":"B57","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.1001893"},{"key":"B58","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.0030305"},{"key":"B59","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.16799"},{"key":"B60","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2017.05.089"},{"key":"B61","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.2111183118"},{"key":"B62","doi-asserted-by":"publisher","DOI":"10.3390\/ijms23094599"},{"key":"B63","doi-asserted-by":"publisher","DOI":"10.1016\/j.npep.2011.01.004"},{"key":"B64","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2016.01.007"},{"key":"B65","doi-asserted-by":"publisher","DOI":"10.1002\/cne.25307"},{"key":"B66","doi-asserted-by":"publisher","DOI":"10.1002\/cne.25400"},{"key":"B67","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2004.08.005"},{"key":"B68","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2350-8-S1-S9"},{"key":"B69","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0098789"},{"key":"B70","doi-asserted-by":"publisher","DOI":"10.1126\/scitranslmed.aax2014"},{"key":"B71","doi-asserted-by":"publisher","DOI":"10.1016\/j.tins.2022.10.004"},{"key":"B72","doi-asserted-by":"publisher","DOI":"10.1111\/jne.13367"},{"key":"B73","doi-asserted-by":"publisher","DOI":"10.1038\/s41380-025-02898-1"},{"key":"B74","doi-asserted-by":"publisher","DOI":"10.1172\/JCI81389"},{"key":"B75","doi-asserted-by":"publisher","DOI":"10.1016\/S0196-9781(01)00647-7"},{"key":"B76","doi-asserted-by":"publisher","DOI":"10.1016\/S0031-6997(24)01352-8"},{"key":"B77","doi-asserted-by":"publisher","DOI":"10.1002\/neu.1012"},{"key":"B78","doi-asserted-by":"publisher","DOI":"10.1016\/j.biopha.2018.04.025"},{"key":"B79","doi-asserted-by":"publisher","DOI":"10.3390\/biomedicines10020343"},{"key":"B80","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroscience.2013.05.036"}],"container-title":["Journal of Clinical Investigation"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.jci.org\/articles\/view\/193475\/files\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,19]],"date-time":"2026-05-19T16:43:05Z","timestamp":1779208985000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.jci.org\/articles\/view\/193475"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,4,1]]},"references-count":80,"journal-issue":{"issue":"7","published-online":{"date-parts":[[2026,2,17]]},"published-print":{"date-parts":[[2026,4,1]]}},"alternative-id":["10.1172\/JCI193475"],"URL":"https:\/\/doi.org\/10.1172\/jci193475","relation":{},"ISSN":["1558-8238"],"issn-type":[{"value":"1558-8238","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,4,1]]},"article-number":"e193475"}}