The WDR53 gene encodes a protein containing WD domains, though its specific cellular function remains unknown. Mutations cause neurodevelopmental disorders with intellectual disability and developmental delay, following an autosomal recessive inheritance pattern. This gene shows minimal constraint against loss-of-function variants, which is consistent with the recessive disease mechanism observed clinically.

Summary from RefSeq
Research Assistant →
0
Active trials
0
Pubs (1 yr)
88
P/LP submissions
0%
P/LP missense
0.97
LOEUF
Mechanism
Clinical SummaryWDR53
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
87 unique Pathogenic / Likely Pathogenic· 62 VUS of 162 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.97LOEUF
pLI 0.002
Z-score 1.64
OE 0.49 (0.270.97)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.38Z-score
OE missense 0.92 (0.811.05)
174 obs / 188.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.49 (0.270.97)
00.351.4
Missense OE0.92 (0.811.05)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 6 / 12.2Missense obs/exp: 174 / 188.7Syn Z: -0.57

ClinVar Variant Classifications

162 submitted variants in ClinVar

Classification Summary

Pathogenic84
Likely Pathogenic3
VUS62
Likely Benign6
Conflicting1
84
Pathogenic
3
Likely Pathogenic
62
VUS
6
Likely Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
84
0
84
Likely Pathogenic
0
0
3
0
3
VUS
0
48
14
0
62
Likely Benign
0
5
1
0
6
Benign
0
0
0
0
0
Conflicting
1
Total0531020156

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

View in ClinVar →

Protein Context — Lollipop Plot

WDR53 · protein map & ClinVar variants

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

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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