{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T16:36:56Z","timestamp":1772210216855,"version":"3.50.1"},"reference-count":81,"publisher":"Public Library of Science (PLoS)","issue":"4","license":[{"start":{"date-parts":[[2021,4,1]],"date-time":"2021-04-01T00:00:00Z","timestamp":1617235200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:p>\n                    Multiple cilia-associated genes have been shown to affect hair cells in zebrafish (\n                    <jats:italic>Danio rerio)<\/jats:italic>\n                    , including the human deafness gene\n                    <jats:italic>dcdc2<\/jats:italic>\n                    , the radial spoke gene\n                    <jats:italic>rsph9<\/jats:italic>\n                    , and multiple intraflagellar transport (IFT) and transition zone genes. Recently a zebrafish\n                    <jats:italic>alms1<\/jats:italic>\n                    mutant was generated. The\n                    <jats:italic>ALMS1<\/jats:italic>\n                    gene is the gene mutated in the ciliopathy Alstr\u00f6m Syndrome a disease that causes hearing loss among other symptoms. The hearing loss seen in Alstr\u00f6m Syndrome may be due in part to hair cell defects as\n                    <jats:italic>Alms1<\/jats:italic>\n                    mutant mice show stereocilia polarity defects and a loss of hair cells. Hair cell loss is also seen in postmortem analysis of Alstr\u00f6m patients. The zebrafish\n                    <jats:italic>alms1<\/jats:italic>\n                    mutant has metabolic defects similar to those seen in Alstr\u00f6m syndrome and\n                    <jats:italic>Alms1<\/jats:italic>\n                    mutant mice. We wished to investigate if it also had hair cell defects. We, however, failed to find any hair cell related phenotypes in\n                    <jats:italic>alms1<\/jats:italic>\n                    mutant zebrafish. They had normal lateral line hair cell numbers as both larvae and adults and normal kinocilia formation. They also showed grossly normal swimming behavior, response to vibrational stimuli, and FM1-43 loading. Mutants also showed a normal degree of sensitivity to both short-term neomycin and long-term gentamicin treatment. These results indicate that cilia-associated genes differentially affect different hair cell types.\n                  <\/jats:p>","DOI":"10.1371\/journal.pone.0246844","type":"journal-article","created":{"date-parts":[[2021,4,1]],"date-time":"2021-04-01T14:25:37Z","timestamp":1617287137000},"page":"e0246844","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":4,"title":["alms1 mutant zebrafish do not show hair cell phenotypes seen in other cilia mutants"],"prefix":"10.1371","volume":"16","author":[{"given":"Lauren","family":"Parkinson","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2191-8550","authenticated-orcid":true,"given":"Tamara M.","family":"Stawicki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2021,4,1]]},"reference":[{"key":"pone.0246844.ref001","doi-asserted-by":"crossref","first-page":"1820","DOI":"10.2105\/AJPH.2016.303299","article-title":"Prevalence of Hearing Loss by Severity in the United States.","volume":"106","author":"AM Goman","year":"2016","journal-title":"Am J Public Health."},{"key":"pone.0246844.ref002","doi-asserted-by":"crossref","first-page":"938","DOI":"10.1001\/archinternmed.2009.66","article-title":"Disorders of balance and vestibular function in US adults: Data from the National Health and Nutrition Examination Survey, 2001\u20132004","volume":"169","author":"Y Agrawal","year":"2009","journal-title":"Arch Intern Med"},{"key":"pone.0246844.ref003","doi-asserted-by":"crossref","first-page":"2309","DOI":"10.3390\/jcm9072309","article-title":"Inner Ear Gene Therapies Take Off: Current Promises and Future Challenges.","volume":"9","author":"S Delmaghani","year":"2020","journal-title":"J Clin Med."},{"key":"pone.0246844.ref004","unstructured":"Van Camp G, Smith R. 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