Article

Dynein-2 intermediate chains play crucial but distinct roles in primary cilia formation and function

2018-01-22

Abstract excerpt

The dynein-2 microtubule motor is the retrograde motor for intraflagellar transport. Mutations in dynein-2 components cause skeletal ciliopathies, notably Jeune syndrome. Dynein-2 comprises a heterodimer of two non-identical intermediate chains, WDR34 and WDR60. Here, we use knockout cell lines to demonstrate that each intermediate chain has a distinct role in cilia function. Both proteins are required to maintain...

Topics

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

Identifiers and source

Literature Corpus work: b14a7be1-4c36-5cb0-8844-1eb585ee98b3
DOI: 10.1101/251694

Open publication

Related research

Semantic proximity does not establish scientific evidence.

Click a neighbor to travelStep 1 · 12 closest

Dynein-2 intermediate chains play crucial but distinct roles in primary cilia formation and functionDOI 10.1101/251694Read

Disease-associated mutations in WDR34 lead to diverse impacts on the assembly and function of dynein-2Semantic proximity Read

Disease-associated mutations in WDR34 lead to diverse impacts on the assembly and function of dynein-2.Semantic proximity Read

Base editing derived models of human WDR34 and WDR60 disease alleles replicate retrograde IFT and hedgehog signaling defects and suggest disturbed Golgi protein transportSemantic proximity Read

Base editing derived models of human <i>WDR34</i> and <i>WDR60</i> disease alleles replicate retrograde IFT and hedgehog signaling defects and suggest disturbed Golgi protein transportSemantic proximity Read

Base editing-derived models of human WDR34 and WDR60 disease alleles replicate retrograde intraflagellar transport (IFT) and hedgehog signaling defects.Semantic proximity Read

Ciliary Dyneins and Dynein Related CiliopathiesSemantic proximity Read

Tctex1d2 associates with short-rib polydactyly syndrome proteins and is required for ciliogenesis.Semantic proximity Read

Distinct effects of hypomorphic IFT and dynein-2 skeletal ciliopathy disease alleles on chondrogenic differentiation, ECM composition and wnt signalling in ATDC5 derived cartilage-like organoidsSemantic proximity Read

Cytoplasmic dynein-2 at a glanceSemantic proximity Read

DYNC2LI1 mutations broaden the clinical spectrum of dynein-2 defects.Semantic proximity Read

Mutations in the gene encoding IFT dynein complex component WDR34 cause Jeune asphyxiating thoracic dystrophy.Semantic proximity Read

Consensus nomenclature for dyneins and associated assembly factors.Semantic proximity Read