ERF

Chr 19AD

ETS2 repressor factor

Also known as: CHYTS, CRS4, PE-2, PE2

ERF encodes a transcriptional repressor that binds to the ETS2 promoter and regulates genes involved in cellular proliferation and trophoblast differentiation. Mutations cause Chitayat syndrome and craniosynostosis 4, both inherited in an autosomal dominant pattern. The gene is highly constrained against loss-of-function variants (pLI 0.99), indicating that haploinsufficiency is likely not tolerated in the general population.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismADLOEUF 0.262 OMIM phenotypes
Clinical SummaryERF
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Gene-Disease Validity (ClinGen)
craniosynostosis 4 · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.99). One damaged copy is likely sufficient to cause disease.
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Clinical Trials
8 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.26LOEUF
pLI 0.990
Z-score 3.73
OE 0.06 (0.020.26)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.25Z-score
OE missense 0.66 (0.600.74)
238 obs / 358.1 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.06 (0.020.26)
00.351.4
Missense OE0.66 (0.600.74)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 1 / 18.2Missense obs/exp: 238 / 358.1Syn Z: -1.84
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveERF-related complex craniosynostosisLOFAD
definitiveERF-related Chitayat syndrome: hyperphalangism, characteristic facies, hallux valgus and bronchomalaciaGOFAD
DN
0.3693th %ile
GOF
0.3590th %ile
LOF
0.82top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · 1 literature citation · LOEUF 0.26

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

LOFHeterozygous intragenic loss-of-function mutations of ERF, encoding an ETS transcription factor, were previously reported to cause a novel craniosynostosis syndrome, suggesting that ERF is haploinsufficient.PMID:33993607

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

ERF · 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

Melanoma

Clinical Study of Fianlimab in Combination With Cemiplimab Versus Pembrolizumab in Adolescent and Adult Patients With Previously Untreated Unresectable Locally Advanced or Metastatic Melanoma

ACTIVE NOT RECRUITING
NCT05352672Phase PHASE3Regeneron PharmaceuticalsStarted 2022-07-14
FianlimabCemiplimabPembrolizumab
Advanced (Stage IIIB/IV) Non-small Cell Lung Cancer (NSCLC) With MET Exon 14 (METex14) Skipping Alterations or MET AmplificationLung Adenocarcinoma Stage IIIB/IV

Tepotinib Phase II in NSCLC Harboring MET Alterations (VISION)

ACTIVE NOT RECRUITING
NCT02864992Phase PHASE2EMD Serono Research & Development Institute, Inc.Started 2016-09-13
Tepotinib
Basal Cell Carcinoma (BCC)First Line Treatment

Evaluation of Efficacy and Safety of Cemiplimab as First Line Treatment for Advanced Basal Cell Carcinoma (BCC) Patients

RECRUITING
NCT06981325Phase PHASE2Institut für Klinische Krebsforschung IKF GmbH at Krankenhaus NordwestStarted 2025-08-07
Cemiplimab
Melanoma

A Trial to See if the Combination of Fianlimab With Cemiplimab Works Better Than Pembrolizumab for Preventing or Delaying Melanoma From Coming Back After it Has Been Removed With Surgery

ACTIVE NOT RECRUITING
NCT05608291Phase PHASE3Regeneron PharmaceuticalsStarted 2023-01-16
FianlimabCemiplimabPembrolizumab
Melanoma

A Clinical Trial of Three Study Medicines (Encorafenib, Binimetinib, and Pembrolizumab) in Patients With Advanced or Metastatic Melanoma

ACTIVE NOT RECRUITING
NCT04657991Phase PHASE3PfizerStarted 2021-01-15
EncorafenibBinimetinibPembrolizumab
Malignant Melanoma Stage II

Adjuvant Nivolumab Treatment in Stage II (IIA, IIB, IIC) High-risk Melanoma

ACTIVE NOT RECRUITING
NCT04309409Phase PHASE3University Hospital, EssenStarted 2020-07-01
Nivolumab
Carcinoma, Non-Small-Cell LungNeoplasm Metastasis

A Study of First-Line Olomorasib (LY3537982) and Pembrolizumab With or Without Chemotherapy in Patients With Advanced KRAS G12C-Mutant Non-small Cell Lung Cancer

RECRUITING
NCT06119581Phase PHASE3Eli Lilly and CompanyStarted 2023-12-21
LY3537982PembrolizumabPlacebo
Healthy AdultsHigh AltitudeIsolation, Social

Human Milk Oligosaccharides (HMOs) and Gut Microbiota, Immune System in Antarctica

ENROLLING BY INVITATION
NCT06133530Phase NAIU University of Applied SciencesStarted 2023-09-24
Human milk oligosaccharidesMaltose
Clinical Literature
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