{"publication_identifier":{"issn":["0950-1991"]},"type":"journal_article","extern":"1","volume":123,"date_updated":"2022-08-08T08:45:59Z","abstract":[{"text":"Mutations giving rise to anatomical defects in the inner ear have been isolated in a large scale screen for mutations causing visible abnormalities in the zebrafish embryo (Haffter, P., Granato, M., Brand, M. et al. (1996) Development 123, 1-36). 58 mutants have been classified as having a primary ear phenotype; these fall into several phenotypic classes, affecting presence or size of the otoliths, size and shape of the otic vesicle and formation of the semicircular canals, and define at least 20 complementation groups. Mutations in seven genes cause loss of one or both otoliths, but do not appear to affect development of other structures within the ear. Mutations in seven genes affect morphology and patterning of the inner ear epithelium, including formation of the semicircular canals and, in some, development of sensory patches (maculae and cristae). Within this class, dog-eared mutants show abnormal development of semicircular canals and lack cristae within the ear, while in van gogh, semicircular canals fail to form altogether, resulting in a tiny otic vesicle containing a single sensory patch. Both these mutants show defects in the expression of homeobox genes within the otic vesicle. In a further class of mutants, ear size is affected while patterning appears to be relatively normal; mutations in three genes cause expansion of the otic vesicle, while in little ears and microtic, the ear is abnormally small, but still contains all five sensory patches, as in the wild type. Many of the ear and otolith mutants show an expected behavioural phenotype: embryos fail to balance correctly, and may swim on their sides, upside down, or in circles. Several mutants with similar balance defects have also been isolated that have no obvious structural ear defect, but that may include mutants with vestibular dysfunction of the inner ear (Granato, M., van Eeden, F. J. M., Schach, U. et al. (1996) Development, 123, 399-413,). Mutations in 19 genes causing primary defects in other structures also show an ear defect. In particular, ear phenotypes are often found in conjunction with defects of neural crest derivatives (pigment cells and/or cartilaginous elements of the jaw). At least one mutant, dog-eared, shows defects in both the ear and another placodally derived sensory system, the lateral line, while hypersensitive mutants have additional trunk lateral line organs.","lang":"eng"}],"publication":"Development","publication_status":"published","doi":"10.1242/dev.123.1.241","author":[{"last_name":"Whitfield","first_name":"Tanya","full_name":"Whitfield, Tanya"},{"first_name":"Michael","full_name":"Granato, Michael","last_name":"Granato"},{"full_name":"Van Eeden, Fredericus","first_name":"Fredericus","last_name":"Van Eeden"},{"first_name":"Ursula","full_name":"Schach, Ursula","last_name":"Schach"},{"first_name":"Michael","full_name":"Brand, Michael","last_name":"Brand"},{"first_name":"Makoto","full_name":"Furutani Seiki, Makoto","last_name":"Furutani Seiki"},{"last_name":"Haffter","first_name":"Pascal","full_name":"Haffter, Pascal"},{"last_name":"Hammerschmidt","first_name":"Matthias","full_name":"Hammerschmidt, Matthias"},{"full_name":"Heisenberg, Carl-Philipp J","first_name":"Carl-Philipp J","orcid":"0000-0002-0912-4566","last_name":"Heisenberg","id":"39427864-F248-11E8-B48F-1D18A9856A87"},{"full_name":"Jiang, Yunjin","first_name":"Yunjin","last_name":"Jiang"},{"first_name":"Donald","full_name":"Kane, Donald","last_name":"Kane"},{"last_name":"Kelsh","first_name":"Robert","full_name":"Kelsh, Robert"},{"last_name":"Mullins","full_name":"Mullins, Mary","first_name":"Mary"},{"first_name":"Jörg","full_name":"Odenthal, Jörg","last_name":"Odenthal"},{"last_name":"Nüsslein Volhard","first_name":"Christiane","full_name":"Nüsslein Volhard, Christiane"}],"_id":"4142","date_created":"2018-12-11T12:07:11Z","month":"12","citation":{"mla":"Whitfield, Tanya, et al. “Mutations Affecting Development of the Zebrafish Inner Ear and Lateral Line.” <i>Development</i>, vol. 123, Company of Biologists, 1996, pp. 241–54, doi:<a href=\"https://doi.org/10.1242/dev.123.1.241\">10.1242/dev.123.1.241</a>.","ama":"Whitfield T, Granato M, Van Eeden F, et al. Mutations affecting development of the zebrafish inner ear and lateral line. <i>Development</i>. 1996;123:241-254. doi:<a href=\"https://doi.org/10.1242/dev.123.1.241\">10.1242/dev.123.1.241</a>","short":"T. Whitfield, M. Granato, F. Van Eeden, U. Schach, M. Brand, M. Furutani Seiki, P. Haffter, M. Hammerschmidt, C.-P.J. Heisenberg, Y. Jiang, D. Kane, R. Kelsh, M. Mullins, J. Odenthal, C. Nüsslein Volhard, Development 123 (1996) 241–254.","ieee":"T. Whitfield <i>et al.</i>, “Mutations affecting development of the zebrafish inner ear and lateral line,” <i>Development</i>, vol. 123. Company of Biologists, pp. 241–254, 1996.","ista":"Whitfield T, Granato M, Van Eeden F, Schach U, Brand M, Furutani Seiki M, Haffter P, Hammerschmidt M, Heisenberg C-PJ, Jiang Y, Kane D, Kelsh R, Mullins M, Odenthal J, Nüsslein Volhard C. 1996. Mutations affecting development of the zebrafish inner ear and lateral line. Development. 123, 241–254.","chicago":"Whitfield, Tanya, Michael Granato, Fredericus Van Eeden, Ursula Schach, Michael Brand, Makoto Furutani Seiki, Pascal Haffter, et al. “Mutations Affecting Development of the Zebrafish Inner Ear and Lateral Line.” <i>Development</i>. Company of Biologists, 1996. <a href=\"https://doi.org/10.1242/dev.123.1.241\">https://doi.org/10.1242/dev.123.1.241</a>.","apa":"Whitfield, T., Granato, M., Van Eeden, F., Schach, U., Brand, M., Furutani Seiki, M., … Nüsslein Volhard, C. (1996). Mutations affecting development of the zebrafish inner ear and lateral line. <i>Development</i>. Company of Biologists. <a href=\"https://doi.org/10.1242/dev.123.1.241\">https://doi.org/10.1242/dev.123.1.241</a>"},"article_processing_charge":"No","acknowledgement":"T. T. W. thanks all members of the Tübingen fish and fly groups for their hospitality and generosity during her visits to the laboratory. We thank Julian Lewis, in whose laboratory much of this work was carried out, for many helpful discussions and suggestions, Catherine Haddon for advice on wild-type ear development and techniques, and Stephen Massey for fish husbandry in Oxford. We are grateful to Julian Lewis, Catherine Haddon, Nick Monk and Patrick Blader for comments on the manuscript, and to Trevor Jowett, Tom Schilling,Eric Weinberg and Monte Westerfield for providing cDNAs. We also thank Jarema Malicki and Wolfgang Driever for making some of the Boston otolith mutants available before publication. T. T. W. thanks the EMBO (ASTF 7668; ASTF 7918), the Imperial Cancer Research Fund and the Wellcome Trust (03643/Z/92) for support.","user_id":"ea97e931-d5af-11eb-85d4-e6957dddbf17","external_id":{"pmid":["9007244"]},"page":"241 - 254","status":"public","language":[{"iso":"eng"}],"intvolume":"       123","publist_id":"1979","oa_version":"None","title":"Mutations affecting development of the zebrafish inner ear and lateral line","quality_controlled":"1","scopus_import":"1","pmid":1,"date_published":"1996-12-01T00:00:00Z","day":"01","article_type":"original","year":"1996","publisher":"Company of Biologists"}