The protein functions as a kelch domain-containing adaptor that regulates protein degradation through the ubiquitin-proteasome system. Mutations cause autosomal recessive intellectual disability with seizures and developmental delay. This gene shows low constraint against loss-of-function variants, consistent with a recessive inheritance pattern where both copies must be affected to cause disease.

LOEUF 1.86
Clinical SummaryKLHDC4
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
52 unique Pathogenic / Likely Pathogenic· 169 VUS of 259 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.86LOEUF
pLI 0.000
Z-score -2.23
OE 1.47 (1.131.86)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-2.62Z-score
OE missense 1.40 (1.301.51)
468 obs / 333.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.47 (1.131.86)
00.351.4
Missense OE1.40 (1.301.51)
00.61.4
Synonymous OE1.42
01.21.6
LoF obs/exp: 39 / 26.6Missense obs/exp: 468 / 333.5Syn Z: -4.09

ClinVar Variant Classifications

259 submitted variants in ClinVar

Classification Summary

Pathogenic43
Likely Pathogenic9
VUS169
Likely Benign14
43
Pathogenic
9
Likely Pathogenic
169
VUS
14
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
43
0
43
Likely Pathogenic
0
0
9
0
9
VUS
0
144
25
0
169
Likely Benign
0
10
3
1
14
Benign
0
0
0
0
0
Total0154801235

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

KLHDC4 · 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.

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Clinical Literature
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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC