KIAA0586

Chr 14AR

KIAA0586

Also known as: JBTS23, SRTD14, Talpid3

This gene encodes a centrosomal protein required for ciliogenesis and sonic hedgehog signaling, including centrosomal recruitment of RAB8A and targeting of centriole satellite proteins to centrosomes. Mutations cause Joubert syndrome 23 and short-rib thoracic dysplasia 14 with polydactyly through autosomal recessive inheritance. These are ciliopathies affecting the central nervous system, skeleton, and often multiple organ systems.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.722 OMIM phenotypes
Clinical SummaryKIAA0586
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.72LOEUF
pLI 0.000
Z-score 3.44
OE 0.54 (0.420.72)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.37Z-score
OE missense 0.96 (0.911.02)
735 obs / 763.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.54 (0.420.72)
00.351.4
Missense OE0.96 (0.911.02)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 36 / 66.2Missense obs/exp: 735 / 763.9Syn Z: -0.03
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveKIAA0586-related Joubert syndromeLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6647th %ile
GOF
0.6247th %ile
LOF
0.3941th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

KIAA0586 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC