ZNF133

Chr 20

zinc finger protein 133

Also known as: ZNF150, pHZ-13, pHZ-66

The protein functions as a transcriptional repressor that binds to specific DNA sequences to negatively regulate RNA polymerase II transcription in the nucleus. Mutations in ZNF133 have been associated with neurodevelopmental disorders including developmental delay and intellectual disability. The gene shows moderate constraint against loss-of-function variants and inheritance patterns vary depending on the specific clinical presentation.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
23
P/LP submissions
0%
P/LP missense
0.62
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryZNF133
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
23 unique Pathogenic / Likely Pathogenic· 69 VUS of 102 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.62LOEUF
pLI 0.003
Z-score 3.01
OE 0.36 (0.210.62)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
2.01Z-score
OE missense 0.71 (0.640.79)
269 obs / 378.8 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.36 (0.210.62)
00.351.4
Missense OE0.71 (0.640.79)
00.61.4
Synonymous OE0.89
01.21.6
LoF obs/exp: 9 / 25.3Missense obs/exp: 269 / 378.8Syn Z: 1.03
DN
0.76top 25%
GOF
0.71top 25%
LOF
0.3066th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

ClinVar Variant Classifications

102 submitted variants in ClinVar

Classification Summary

Pathogenic23
VUS69
Likely Benign4
Benign2
23
Pathogenic
69
VUS
4
Likely Benign
2
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
23
0
23
Likely Pathogenic
0
0
0
0
0
VUS
0
66
3
0
69
Likely Benign
0
4
0
0
4
Benign
0
0
0
2
2
Total07026298

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

View in ClinVar →

Protein Context — Lollipop Plot

ZNF133 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →