{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"institution":[{"name":"bioRxiv"}],"indexed":{"date-parts":[[2026,1,15]],"date-time":"2026-01-15T20:59:13Z","timestamp":1768510753858,"version":"3.49.0"},"posted":{"date-parts":[[2024,4,27]]},"group-title":"Developmental Biology","reference-count":42,"publisher":"openRxiv","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2024,4,27]]},"abstract":"<jats:title>Summary<\/jats:title>\n                <jats:p>\n                  Neurofibromatosis Type 2 (NF-2) is a dominantly inherited genetic disorder that results from mutations in the tumor suppressor gene, neurofibromin 2 (NF2) gene. Here, we report the generation of a conditional zebrafish model of neurofibromatosis established by an inducible genetic knockout of\n                  <jats:italic>nf2a\/b<\/jats:italic>\n                  , the zebrafish homolog of human NF2. Analysis of\n                  <jats:italic>nf2a<\/jats:italic>\n                  and\n                  <jats:italic>nf2b<\/jats:italic>\n                  expression reveals ubiquitous expression of\n                  <jats:italic>nf2b<\/jats:italic>\n                  in the early embryo, with overlapping expression in the neural crest and its derivatives and in the cranial mesenchyme. In contrast,\n                  <jats:italic>nf2a<\/jats:italic>\n                  displays lower expression levels. Induction of\n                  <jats:italic>nf2a\/b<\/jats:italic>\n                  knockout at early stages increases the proliferation of larval Schwann cells and meningeal fibroblasts. Subsequently, in adult zebrafish,\n                  <jats:italic>nf2a\/b<\/jats:italic>\n                  knockout triggers the development of a spectrum of tumors, including vestibular schwannomas, spinal schwannomas, meningiomas, and retinal hamartomas, mirroring the tumor manifestations observed in patients with NF-2. Collectively, these findings highlight the generation of a novel zebrafish model that mimics the complexities of the human NF-2 disorder. Consequently, this model holds significant potential for facilitating therapeutic screening and elucidating key driver genes implicated in NF-2 onset.\n                <\/jats:p>","DOI":"10.1101\/2024.04.23.590787","type":"posted-content","created":{"date-parts":[[2024,4,28]],"date-time":"2024-04-28T04:10:12Z","timestamp":1714277412000},"source":"Crossref","is-referenced-by-count":1,"title":["Generation of a zebrafish neurofibromatosis model via inducible knockout of\n                  <i>nf2<\/i>"],"prefix":"10.64898","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0173-0465","authenticated-orcid":false,"given":"Ayyappa Raja","family":"Desingu Rajan","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuanyun","family":"Huang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3740-3378","authenticated-orcid":false,"given":"Jan","family":"Stundl","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Katelyn","family":"Chu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anushka","family":"Irodi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zihan","family":"Yang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2795-5678","authenticated-orcid":false,"given":"Brian E.","family":"Applegate","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4274-1862","authenticated-orcid":false,"given":"Marianne E.","family":"Bronner","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"54368","reference":[{"key":"2024050122050830000_2024.04.23.590787v1.1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.20883"},{"key":"2024050122050830000_2024.04.23.590787v1.2","doi-asserted-by":"crossref","unstructured":"Alasaadi, D. 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