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Recently, these DCGs were implicated in functions at the cilium. Here, we investigate the regulation of these DCGs by Regulatory Factor X transcription factors (RFX TFs), a gene family known for transcriptionally regulating ciliary genes. We identify conserved X\u2010box motifs in the promoter regions of\n                    <jats:italic>DYX1C1, DCDC2<\/jats:italic>\n                    , and\n                    <jats:italic>KIAA0319<\/jats:italic>\n                    and demonstrate their functionality, as well as the ability to recruit RFX TFs using reporter gene and electrophoretic mobility shift assays. Furthermore, we uncover a complex regulation pattern between\n                    <jats:italic>RFX1, RFX2<\/jats:italic>\n                    , and\n                    <jats:italic>RFX3<\/jats:italic>\n                    and their significant effect on modifying the endogenous expression of\n                    <jats:italic>DYX1C1<\/jats:italic>\n                    and\n                    <jats:italic>DCDC2<\/jats:italic>\n                    in a human retinal pigmented epithelial cell line immortalized with hTERT (hTERT\u2010RPE1). In addition, induction of ciliogenesis increases the expression of RFX TFs and DCGs. At the protein level, we show that endogenous\n                    <jats:italic>DYX1C1<\/jats:italic>\n                    localizes to the base of the cilium, whereas\n                    <jats:italic>DCDC2<\/jats:italic>\n                    localizes along the entire axoneme of the cilium, thereby validating earlier localization studies using overexpression models. Our results corroborate the emerging role of DCGs in ciliary function and characterize functional noncoding elements, X\u2010box promoter motifs, in DCG promoter regions, which thus can be targeted for mutation screening in dyslexia and ciliopathies associated with these genes.\u2014Tammimies, K., Bieder, A., Lauter, G., Sugiaman\u2010Trapman, D., Torchet, R., Hokkanen, M.\u2010E., Burghoorn, J., Castr\u00e9n, E., Kere, J., Tapia\u2010P\u00e1ez, I., Swoboda, P. Ciliary dyslexia candidate genes\n                    <jats:italic>DYX1C1<\/jats:italic>\n                    and\n                    <jats:italic>DCDC2<\/jats:italic>\n                    are regulated by Regulatory Factor (RF) X transcription factors through X\u2010box promoter motifs. FASEB J. 30, 3578\u20133587 (2016).\n                    <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"http:\/\/www.fasebj.org\">www.fasebj.org<\/jats:ext-link>\n                  <\/jats:p>","DOI":"10.1096\/fj.201500124rr","type":"journal-article","created":{"date-parts":[[2016,7,22]],"date-time":"2016-07-22T22:39:44Z","timestamp":1469227184000},"page":"3578-3587","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":32,"title":["Ciliary dyslexia candidate genes\n                    <i>DYX1C1<\/i>\n                    and\n                    <i>DCDC2<\/i>\n                    are regulated by Regulatory Factor X (RFX) transcription factors through X\u2010box promoter motifs"],"prefix":"10.1096","volume":"30","author":[{"given":"Kristiina","family":"Tammimies","sequence":"first","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"},{"name":"Center of Neurodevelopmental Disorders (KIND) Pediatric Neuropsychiatry Unit Department of Women's and Children's Health Karolinska Institutet  Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea","family":"Bieder","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gilbert","family":"Lauter","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Debora","family":"Sugiaman\u2010Trapman","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel","family":"Torchet","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie\u2010Estelle","family":"Hokkanen","sequence":"additional","affiliation":[{"name":"Neuroscience Center University of Helsinki  Helsinki Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan","family":"Burghoorn","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eero","family":"Castren","sequence":"additional","affiliation":[{"name":"Neuroscience Center University of Helsinki  Helsinki Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juha","family":"Kere","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"},{"name":"Molecular Neurology Research Program University of Helsinki  Helsinki Finland"},{"name":"Folkh\u00e4lsan Institute of Genetics  Helsinki Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabel","family":"Tapia\u2010P\u00e1ez","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Swoboda","sequence":"additional","affiliation":[{"name":"Department of Biosciences and Nutrition Karolinska Institutet  Huddinge Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,7,22]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.4065\/76.11.1081"},{"key":"e_1_2_8_3_1","first-page":"998","article-title":"Prevalence of reading disability in boys and girls","volume":"264","author":"Shaywitz S. 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