DYX1C1 is required for axonemal dynein assembly and ciliary motility.

doi:10.1038/ng.2707

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Article

DYX1C1 is required for axonemal dynein assembly and ciliary motility.

Nature genetics - 1 Sept 2013

Tarkar Aarti, Loges Niki T, Slagle Christopher E, Francis Richard, Dougherty Gerard W, Tamayo Joel V, Shook Brett, Cantino Marie, Schwartz Daniel, Jahnke Charlotte, Olbrich Heike, Werner Claudius, Raidt Johanna, Pennekamp Petra, Abouhamed Marouan, Hjeij Rim, Köhler Gabriele, Griese Matthias, Li You, Lemke Kristi, Klena Nikolas, Liu Xiaoqin, Gabriel George, Tobita Kimimasa, Jaspers Martine, Morgan Lucy C, Shapiro Adam J, Letteboer Stef J F, Mans Dorus A, Carson Johnny L, Leigh Margaret W, Wolf Whitney E, Chen Serafine, Lucas Jane S, Onoufriadis Alexandros, Plagnol Vincent, Schmidts Miriam, Boldt Karsten, Roepman Ronald, Zariwala Maimoona A, Lo Cecilia W, Mitchison Hannah M, Knowles Michael R, Burdine Rebecca D, Loturco Joseph J, Omran Heymut

Abstract excerpt

DYX1C1 has been associated with dyslexia and neuronal migration in the developing neocortex. Unexpectedly, we found that deleting exons 2-4 of Dyx1c1 in mice caused a phenotype resembling primary ciliary dyskinesia (PCD), a disorder characterized by chronic airway disease, laterality defects and male infertility. This phenotype was confirmed independently in mice with a Dyx1c1 c.T2A start-codon mutation recovered...

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Methodology Data sources Editorial policy Privacy About Contact Helena Bioinformatics

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Identifiers

PMID: 23872636
DOI: 10.1038/ng.2707
PMCID: PMC4000444

Genes

DNAAF2
DNAAF4

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PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 7 Sept 2026. Manifest publication-graph-cohort-2026-09-07.1.

Source receipt: pubmed, retrieved 7 Sept 2026.

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DYX1C1 is required for axonemal dynein assembly and ciliary motility.PMID 23872636Read ↗

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Mutations in GAS8, a Gene Encoding a Nexin-Dynein Regulatory Complex Subunit, Cause Primary Ciliary Dyskinesia with Axonemal Disorganization.Semantic proximity Read ↗

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