KLHL15

Chr XXLR

kelch like family member 15

Also known as: HEL-S-305, XLID103

The protein functions as a substrate-specific adapter for the CUL3 E3 ubiquitin-protein ligase complex, targeting specific proteins for degradation and playing a key role in DNA damage response by regulating DNA double-strand break repair pathways. Mutations cause X-linked intellectual developmental disorder with X-linked recessive inheritance. The gene is highly constrained against loss-of-function variants (pLI = 0.99, LOEUF = 0.20), indicating that functional variants are likely to be pathogenic.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismXLRLOEUF 0.201 OMIM phenotype
Clinical SummaryKLHL15
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.99). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.20LOEUF
pLI 0.994
Z-score 3.63
OE 0.00 (0.000.20)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
3.69Z-score
OE missense 0.34 (0.280.41)
84 obs / 247.0 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.00 (0.000.20)
00.351.4
Missense OE0.34 (0.280.41)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 0 / 15.3Missense obs/exp: 84 / 247.0Syn Z: -0.10
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedKLHL15-related intellectual disabilityLOFXLR
DN
0.3196th %ile
GOF
0.4974th %ile
LOF
0.65top 25%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.20

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

KLHL15 · 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 →