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The transition zone (TZ) at the base of the cilium is crucial for cilia function, and defects in several TZ proteins are associated with human congenital ciliopathies such as nephronophthisis (NPHP) and Meckel\u2013Gruber syndrome (MKS). In several species, MKS and NPHP proteins form separate complexes that cooperate with Cep290 to assemble the TZ, but flies seem to lack core components of the NPHP module. We show that MKS proteins in flies are spatially separated from Cep290 at the TZ, and that flies mutant for individual MKS genes fail to recruit other MKS proteins to the TZ, whereas Cep290 seems to be recruited normally. Although there are abnormalities in microtubule and membrane organisation in developing MKS mutant cilia, these defects are less apparent in adults, where sensory cilia and sperm flagella seem to function quite normally. Thus, localising MKS proteins to the cilium or flagellum is not essential for viability or fertility in flies.<\/jats:p>","DOI":"10.1242\/jcs.194621","type":"journal-article","created":{"date-parts":[[2016,8,30]],"date-time":"2016-08-30T22:14:17Z","timestamp":1472595257000},"page":"3732-3743","source":"Crossref","is-referenced-by-count":34,"title":["<i>Drosophila<\/i>\n                    sensory cilia lacking MKS proteins exhibit striking defects in development but only subtle defects in adults"],"prefix":"10.1242","volume":"129","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5296-1370","authenticated-orcid":false,"given":"Metta B.","family":"Pratt","sequence":"first","affiliation":[{"name":"University of Oxford 1 The Sir William Dunn School of Pathology , , South Parks Road, Oxford OX1 3RE , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua S.","family":"Titlow","sequence":"additional","affiliation":[{"name":"University of Oxford 2 Department of Biochemistry , , South Parks Road, Oxford OX1 3QU , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilan","family":"Davis","sequence":"additional","affiliation":[{"name":"University of Oxford 2 Department of Biochemistry , , South Parks Road, Oxford OX1 3QU , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amy R.","family":"Barker","sequence":"additional","affiliation":[{"name":"William Harvey Research Institute, Queen Mary University of London 3 Centre for Microvascular Research , , London EC1M 6BQ , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helen R.","family":"Dawe","sequence":"additional","affiliation":[{"name":"College of Life and Environmental Sciences, University of Exeter 4 Biosciences , , Exeter EX4 4QD , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4689-1297","authenticated-orcid":false,"given":"Jordan W.","family":"Raff","sequence":"additional","affiliation":[{"name":"University of Oxford 1 The Sir William Dunn School of Pathology , , South Parks Road, Oxford OX1 3RE , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5930-1989","authenticated-orcid":false,"given":"Helio","family":"Roque","sequence":"additional","affiliation":[{"name":"University of Oxford 1 The Sir William Dunn School of Pathology , , South Parks Road, Oxford OX1 3RE , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2024061316263266600_JCS194621C1","doi-asserted-by":"publisher","first-page":"2185","DOI":"10.1093\/hmg\/ddu738","article-title":"Nephrocystin proteins NPHP5 and Cep290 regulate BBSome integrity, ciliary trafficking and cargo delivery","volume":"24","author":"Barbelanne","year":"2015","journal-title":"Hum. 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