ArticleFASEB journal : official publication of the Federation of American Societies for Experimental Biology2016
Ciliary dyslexia candidate genes DYX1C1 and DCDC2 are regulated by Regulatory Factor X (RFX) transcription factors through X-box promoter motifs.
Article in FASEB journal : official publication of the Federation of American Societies for Experimental Biology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed, 34 citations in OpenAlex.
- RFX5 in cancer: context-dependent molecular functions and emerging translational relevance.Frontiers in immunology · 2026Review
- Spatiotemporal expression pattern of dyslexia susceptibility 1 candidate 1 (DYX1C1) during rat cerebral cortex development.Pediatric research · 2025Article
- Primary cilia promote the differentiation of human neurons through the WNT signaling pathway.BMC biology · 2024Article
- Genetic Modifications of Developmental Dyslexia and Its Representation Using In Vivo, In Vitro Model.Global medical genetics · 2024Review
- A novel homozygous splice variant inActa biochimica et biophysica Sinica · 2023Article
- Genetic and protein interaction studies between the ciliary dyslexia candidate genes DYX1C1 and DCDC2.BMC molecular and cell biology · 2023Article
- The Polygenic Nature and Complex Genetic Architecture of Specific Learning Disorder.Brain sciences · 2021Review
- RFX1: a promising therapeutic arsenal against cancer.Cancer cell international · 2021Review
- Dyslexia Candidate Gene and Ciliary Gene Expression Dynamics During Human Neuronal Differentiation.Molecular neurobiology · 2020Article
- Rare variants in dynein heavy chain genes in two individuals with situs inversus and developmental dyslexia: a case report.BMC medical genetics · 2020Article
- Neuroimaging genetics studies of specific reading disability and developmental language disorder: A review.Language and linguistics compass · 2019Article
- ALMS1 and Alström syndrome: a recessive form of metabolic, neurosensory and cardiac deficits.Journal of molecular medicine (Berlin, Germany) · 2019Review
- Promoter expression of HERV-K (HML-2) provirus-derived sequences is related to LTR sequence variation and polymorphic transcription factor binding sites.Retrovirology · 2018Article
- Characterization of the human RFX transcription factor family by regulatory and target gene analysis.BMC genomics · 2018Article
- An Expanded Role for the RFX Transcription Factor DAF-19, with Dual Functions in Ciliated and Nonciliated Neurons.Genetics · 2018Article
- RFX1 and RFX3 Transcription Factors Interact with the D Sequence of Adeno-Associated Virus Inverted Terminal Repeat and Regulate AAV Transduction.Scientific reports · 2018Article
- The role of READ1 and KIAA0319 genetic variations in developmental dyslexia: testing main and interactive effects.Journal of human genetics · 2017Article
- Genes, Gender, Environment, and Novel Functions of Estrogen Receptor Beta in the Susceptibility to Neurodevelopmental Disorders.Brain sciences · 2017Review
- Neurogenetics of developmental dyslexia: from genes to behavior through brain neuroimaging and cognitive and sensorial mechanisms.Translational psychiatry · 2017Review
- Study of Genetic Association WithClinical practice and epidemiology in mental health : CP & EMH · 2017Article
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Authors and funding
11 authors at 3 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
DYX1C1, DCDC2, and KIAA0319 are three of the most replicated dyslexia candidate genes (DCGs). 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-box motifs in the promoter regions of DYX1C1, DCDC2, and KIAA0319 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 RFX1, RFX2, and RFX3 and their significant effect on modifying the endogenous expression of DYX1C1 and DCDC2 in a human retinal pigmented epithelial cell line immortalized with hTERT (hTERT-RPE1). In addition, induction of ciliogenesis increases the expression of RFX TFs and DCGs. At the protein level, we show that endogenous DYX1C1 localizes to the base of the cilium, whereas DCDC2 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-box promoter motifs, in DCG promoter regions, which thus can be targeted for mutation screening in dyslexia and ciliopathies associated with these genes.-Tammimies, K., Bieder, A., Lauter, G., Sugiaman-Trapman, D., Torchet, R., Hokkanen, M.-E., Burghoorn, J., Castrén, E., Kere, J., Tapia-Páez, I., Swoboda, P. Ciliary dyslexia candidate genes DYX1C1 and DCDC2 are regulated by Regulatory Factor (RF) X transcription factors through X-box promoter motifs.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.