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Article

The hydrolethalus syndrome protein HYLS-1 links core centriole structure to cilia formation.

Genes & development - 1 Sept 2009

Dammermann Alexander, Pemble Hayley, Mitchell Brian J, McLeod Ian, Yates John R, Kintner Chris, Desai Arshad B, Oegema Karen

Abstract excerpt

Centrioles are subcellular organelles composed of a ninefold symmetric microtubule array that perform two important functions: (1) They build centrosomes that organize the microtubule cytoskeleton, and (2) they template cilia, microtubule-based projections with sensory and motile functions. We id...

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Methodology Data sources Editorial policy Privacy About Contact Helena Bioinformatics

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Source

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Identifiers

PMID: 19656802
DOI: 10.1101/gad.1810409
PMCID: PMC2751977

Genes

HYLS1

Provenance

PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 2 Sept 2026. Prepared record publication-19656802-2026-09-02.2. Manifest publication-graph-cohort-2026-09-02.2.

Source receipt: PubMed via Helena Literature Corpus, retrieved 2 Sept 2026.

Related research

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The hydrolethalus syndrome protein HYLS-1 links core centriole structure to cilia formation.PMID 19656802Read ↗

Centriole structural integrity defects are a crucial feature of hydrolethalus syndrome.Semantic proximity Read ↗

Centriole structural integrity defects are a crucial feature of Hydrolethalus SyndromeSemantic proximity Read ↗

Mutations in KIAA0586 Cause Lethal Ciliopathies Ranging from a Hydrolethalus Phenotype to Short-Rib Polydactyly Syndrome.Semantic proximity Read ↗

CCDC15 localizes to the centriole inner scaffold and regulates centriole integrity and ciliogenesisSemantic proximity Read ↗

<i>C. elegans</i> SAS-1 ensures centriole integrity and ciliary function, and operates with SSNA-1Semantic proximity Read ↗

Ahi1, whose human ortholog is mutated in Joubert syndrome, is required for Rab8a localization, ciliogenesis and vesicle trafficking.Semantic proximity Read ↗

Expanding the Phenotypic Spectrum of Pathogenic KIAA0586 Variants: From Joubert Syndrome to Hydrolethalus Syndrome.Semantic proximity Read ↗

A novel HYLS1 homozygous mutation in living siblings with Joubert syndrome.Semantic proximity Read ↗

SAS-4 is a C. elegans centriolar protein that controls centrosome size.Semantic proximity Read ↗

Sub-centrosomal mapping identifies augmin-γTuRC as part of a centriole-stabilizing scaffoldSemantic proximity Read ↗

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