Article

Physical and functional interaction of Lrrc56 and Odad3 controls deployment of axonemal dyneins in vertebrate multiciliated cells

2025-07-05

Abstract excerpt

Primary ciliary dyskinesia is a genetically heterogeneous motile ciliopathy characterized chronic respiratory disease, laterality defects, hydrocephalus, and infertility, caused by impaired function of motile cilia. LRRC56 has recently emerged as a novel PCD candidate gene, but its role in vertebrate cilia remains poorly understood. Here, we used Xenopus laevis multiciliated cells, targeted knockdown, and in vivo...

Topics

Open a Topic to create a Post that cites this publication.

Identifiers and source

Literature Corpus work: 4fe23b21-acc5-50b7-98f1-1f8b931cd6f3
DOI: 10.1101/2025.07.02.662827

Open publication

Related research

Semantic proximity does not establish scientific evidence.

Click a neighbor to travelStep 1 · 12 closest

Physical and functional interaction of Lrrc56 and Odad3 controls deployment of axonemal dyneins in vertebrate multiciliated cellsDOI 10.1101/2025.07.02.662827Read

LRRC56 deletion causes primary ciliary dyskinesia in mice characterized by dynein arms defectsSemantic proximity Read

Biallelic Mutations in <i>LRRC56</i> encoding a protein associated with intraflagellar transport, cause mucociliary clearance and laterality defectsSemantic proximity Read

Loss-of-Function GAS8 Mutations Cause Primary Ciliary Dyskinesia and Disrupt the Nexin-Dynein Regulatory Complex.Semantic proximity Read

Pathogenic variants in CLXN encoding the outer dynein arm docking-associated calcium-binding protein calaxin cause primary ciliary dyskinesia.Semantic proximity Read

Mutations in ZMYND10, a gene essential for proper axonemal assembly of inner and outer dynein arms in humans and flies, cause primary ciliary dyskinesia.Semantic proximity Read

ZMYND10 is mutated in primary ciliary dyskinesia and interacts with LRRC6.Semantic proximity Read

Functional partitioning of a liquid-like organelle during assembly of axonemal dyneinsSemantic proximity Read

Loss-of-function mutations in LRRC6, a gene essential for proper axonemal assembly of inner and outer dynein arms, cause primary ciliary dyskinesia.Semantic proximity Read

CCDC151 mutations cause primary ciliary dyskinesia by disruption of the outer dynein arm docking complex formation.Semantic proximity Read

DRC2/CCDC65 is a central hub for assembly of the nexin-dynein regulatory complex and other regulators of ciliary and flagellar motility.Semantic proximity Read

LRRC6 Mutation Causes Primary Ciliary Dyskinesia with Dynein Arm DefectsSemantic proximity Read

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