ZNF346

Chr 5

zinc finger protein 346

Also known as: JAZ, Zfp346

The protein is a nucleolar zinc finger protein that binds double-stranded RNA and RNA/DNA hybrids and protects neurons by inhibiting cell cycle re-entry through stimulation of p21 gene expression. Mutations in ZNF346 cause autosomal recessive developmental and epileptic encephalopathy with onset in infancy, characterized by severe intellectual disability, refractory seizures, and progressive microcephaly. The gene shows moderate constraint against loss-of-function variants, suggesting some intolerance to complete protein loss.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
1
Pubs (1 yr)
59
P/LP submissions
0%
P/LP missense
0.66
LOEUF
DN
Mechanism· predicted
Clinical SummaryZNF346
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.29) despite low pLI — interpret in context.
📋
ClinVar Variants
57 unique Pathogenic / Likely Pathogenic· 41 VUS of 110 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.66LOEUF
pLI 0.116
Z-score 2.44
OE 0.29 (0.140.66)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.17Z-score
OE missense 0.73 (0.630.86)
113 obs / 154.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.29 (0.140.66)
00.351.4
Missense OE0.73 (0.630.86)
00.61.4
Synonymous OE0.81
01.21.6
LoF obs/exp: 4 / 13.8Missense obs/exp: 113 / 154.0Syn Z: 1.16
DN
0.6260th %ile
GOF
0.5758th %ile
LOF
0.4430th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction 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

110 submitted variants in ClinVar

Classification Summary

Pathogenic55
Likely Pathogenic2
VUS41
Likely Benign3
55
Pathogenic
2
Likely Pathogenic
41
VUS
3
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
55
0
55
Likely Pathogenic
0
0
2
0
2
VUS
0
37
4
0
41
Likely Benign
0
2
1
0
3
Benign
0
0
0
0
0
Total039620101

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

View in ClinVar →

Protein Context — Lollipop Plot

ZNF346 · 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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