PCGF2

Chr 17AD

polycomb group ring finger 2

Also known as: MEL-18, RNF110, TPFS, ZNF144

The protein is a transcriptional repressor that functions as a component of Polycomb repressive complex 1 (PRC1), maintaining transcriptional silencing of developmental genes including Hox genes through chromatin modification. Mutations cause Turnpenny-Fry syndrome with autosomal dominant inheritance. The gene is highly constrained against loss-of-function variants (pLI 0.86, LOEUF 0.41), indicating intolerance to gene disruption.

OMIMResearchSummary from RefSeq, OMIM, UniProt
GOFmechanismADLOEUF 0.411 OMIM phenotype
Clinical SummaryPCGF2
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Gene-Disease Validity (ClinGen)
turnpenny-fry syndrome · ADStrong

Strong evidence — appropriate for clinical testing

Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.86) — some intolerance to loss-of-function variants.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.41LOEUF
pLI 0.864
Z-score 3.15
OE 0.13 (0.050.41)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.95Z-score
OE missense 0.81 (0.720.93)
169 obs / 207.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.13 (0.050.41)
00.351.4
Missense OE0.81 (0.720.93)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 2 / 15.3Missense obs/exp: 169 / 207.6Syn Z: -0.35
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongPCGF2-related craniofacial neurological cardiovascular and skeletal features (Turnpenny-Fry syndrome)GOFAD
DN
0.4487th %ile
GOF
0.4480th %ile
LOF
0.70top 10%

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

LOFprediction above median · LOEUF 0.41
DN1 literature citation

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.

Literature Evidence

DNMutant PCGF2 may have dominant-negative effects, sequestering PRC1 components into complexes that lack the ability to interact efficiently with histones.PMID:30343942

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PCGF2 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗