This gene encodes a zinc-sensitive transcriptional repressor that regulates cellular zinc homeostasis by binding to zinc transcriptional regulatory elements in gene promoters and controls ribosome biogenesis by modulating rRNA and ribosomal protein levels. The gene is extremely intolerant to loss-of-function variants (pLI ~1.0, LOEUF 1.9), suggesting that haploinsufficiency would be highly deleterious, but no established human disease phenotype has been reported to date. Given its critical role in zinc homeostasis and ribosome biogenesis, mutations would likely affect cellular metabolism and protein synthesis.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 1.90
Clinical SummaryZNF658
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
45 unique Pathogenic / Likely Pathogenic· 183 VUS of 247 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.90LOEUF
pLI 0.000
Z-score -1.37
OE 1.43 (0.961.90)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-2.86Z-score
OE missense 1.42 (1.321.52)
529 obs / 373.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.43 (0.961.90)
00.351.4
Missense OE1.42 (1.321.52)
00.61.4
Synonymous OE1.44
01.21.6
LoF obs/exp: 17 / 11.9Missense obs/exp: 529 / 373.4Syn Z: -3.94
DN
0.88top 5%
GOF
0.84top 5%
LOF
0.2873th %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

247 submitted variants in ClinVar

Classification Summary

Pathogenic41
Likely Pathogenic4
VUS183
Likely Benign15
Benign1
41
Pathogenic
4
Likely Pathogenic
183
VUS
15
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
41
0
41
Likely Pathogenic
0
0
4
0
4
VUS
0
182
1
0
183
Likely Benign
0
12
1
2
15
Benign
0
0
1
0
1
Total0194482244

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

View in ClinVar →

Protein Context — Lollipop Plot

ZNF658 · 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

No open access results found