{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,16]],"date-time":"2025-12-16T05:20:27Z","timestamp":1765862427042,"version":"3.48.0"},"reference-count":76,"publisher":"MDPI AG","issue":"24","license":[{"start":{"date-parts":[[2025,12,12]],"date-time":"2025-12-12T00:00:00Z","timestamp":1765497600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Interdisciplinary Center for Clinical Research (IZKF) of the University of W\u00fcrzburg, Germany","award":["N-320"],"award-info":[{"award-number":["N-320"]}]},{"name":"Interdisciplinary Center for Clinical Research (IZKF) of the University of W\u00fcrzburg, Germany","award":["Z-4\/149"],"award-info":[{"award-number":["Z-4\/149"]}]},{"name":"Interdisciplinary Center for Clinical Research (IZKF) of the University of W\u00fcrzburg, Germany","award":["Z-6"],"award-info":[{"award-number":["Z-6"]}]},{"name":"EU Horizon Europe Research and Innovation Programme","award":["101086453"],"award-info":[{"award-number":["101086453"]}]},{"name":"Verein zur Durchf\u00fchrung Neurowissenschaftlicher Tagungen e.V"},{"DOI":"10.13039\/501100008769","name":"University of W\u00fcrzburg","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100008769","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cells"],"abstract":"<jats:p>Lbx1 plays important roles in different processes, including the development of sensory pathways, neuronal cell fate regulation, and muscle cell precursor migration. Genetic variation in the LBX1 locus has been associated with several human disease conditions, such as idiopathic scoliosis, congenital limb malformation, and neuropsychiatric illness, including attention-deficit\/hyperactivity disorder (ADHD) and anxiety disorders. Zebrafish (Danio rerio) were used to investigate the behavioral consequences of the loss of function of the two orthologs to the human LBX1 gene, zebrafish lbx1a and lbx1b. We observed a consistent locomotor hyperactivity phenotype induced by a novel environment in lbx1a mutants. Repeated dark stimuli provoked similar responses in both mutant lines, including the novelty-induced hyperactivity. We performed RNAseq on total RNA isolated from the head region of mutant and wildtype larvae. Several differentially expressed genes were identified, giving more insights into Lbx1 target genes and pathways, which could be relevant regarding the evaluation of zebrafish lbx1a or lbx1b as a human disease model. Furthermore, the analysis was complemented with a comparison to the expression profile of human LBX1 overexpression in cell culture, revealing a convergence on just two commonly regulated genes, namely alpha-Internexin (INA) and Fibrillin-3 (FBN3). In conclusion, our findings might further elucidate the multitude of functions of Lbx1 and its involvement in various human disease conditions.<\/jats:p>","DOI":"10.3390\/cells14241980","type":"journal-article","created":{"date-parts":[[2025,12,12]],"date-time":"2025-12-12T16:19:02Z","timestamp":1765556342000},"page":"1980","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Hyperactivity and Differential Gene Expression in lbx1a(\u2212\/\u2212) Zebrafish Larvae"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7883-6479","authenticated-orcid":false,"given":"Carsten","family":"Drepper","sequence":"first","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laura","family":"Kettenstock","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Simon","family":"St\u00f6ckl","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anna","family":"Elsenbach","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carina","family":"Lechermeier","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wonhyeok","family":"Lee","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9425-3698","authenticated-orcid":false,"given":"Susanne","family":"Kneitz","sequence":"additional","affiliation":[{"name":"Biochemistry and Cell Biology, Biocenter, University of W\u00fcrzburg, 97074 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Klaus-Peter","family":"Lesch","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"},{"name":"Division of Molecular Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"},{"name":"Department of Psychiatry and Neuropsychology, School for Mental Health and Neuroscience (MHeNs), Maastricht University, 6229 ER Maastricht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marcel","family":"Romanos","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5166-2851","authenticated-orcid":false,"given":"Christina","family":"Lillesaar","sequence":"additional","affiliation":[{"name":"Child and Adolescent Psychiatry, Center of Mental Health, University Hospital W\u00fcrzburg, 97080 W\u00fcrzburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2025,12,12]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"345","DOI":"10.1016\/0925-4773(95)00450-5","article-title":"Mouse Lbx1 and human LBX1 define a novel mammalian homeobox gene family related to the Drosophila lady bird genes","volume":"53","author":"Jagla","year":"1995","journal-title":"Mech. 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