ZFPM2

Chr 8AD

zinc finger protein, FOG family member 2

Also known as: DIH3, FOG2, PRDM19, SRXY9, ZC2HC11B, ZNF89B, hFOG-2

This gene encodes a zinc finger transcription factor that acts as an essential cofactor for GATA family proteins, regulating genes critical for heart development and gonadal differentiation. Mutations cause autosomal dominant conditions including tetralogy of Fallot, diaphragmatic hernia, and 46,XY sex reversal, affecting cardiac, pulmonary, and reproductive development. The gene is highly constrained against loss-of-function variants (pLI >0.99), indicating that most protein-disrupting variants are likely pathogenic.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

46XY sex reversal 9MIM #616067
AD
Diaphragmatic hernia 3MIM #610187
AD
Tetralogy of FallotMIM #187500
AD
UniProt46,XY sex reversal 9
UniProtConotruncal heart malformations
0
Active trials
18
Pubs (1 yr)
10
P/LP submissions
0%
P/LP missense
0.14
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryZFPM2
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
10 unique Pathogenic / Likely Pathogenic· 225 VUS of 300 total submissions
📖
GeneReview available — ZFPM2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.14LOEUF
pLI 1.000
Z-score 5.82
OE 0.05 (0.020.14)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
0.21Z-score
OE missense 0.98 (0.911.04)
581 obs / 595.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.05 (0.020.14)
00.351.4
Missense OE0.98 (0.911.04)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 2 / 43.3Missense obs/exp: 581 / 595.6Syn Z: 0.22
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedZFPM2-related malformation syndromeOTHERAD
DN
0.2698th %ile
GOF
0.1999th %ile
LOF
0.83top 5%

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

LOFprediction above median · 1 literature citation · 40% of P/LP variants are LoF · LOEUF 0.14

Literature Evidence

LOFThe same variant is seen in 2 additional family members, both of whom are asymptomatic, thus highlighting that ZFPM2 haploinsufficiency is associated with reduced penetrance.PMID:24769157

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

300 submitted variants in ClinVar

Classification Summary

Pathogenic2
Likely Pathogenic8
VUS225
Likely Benign46
Benign3
Conflicting2
2
Pathogenic
8
Likely Pathogenic
225
VUS
46
Likely Benign
3
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
2
0
2
Likely Pathogenic
4
0
4
0
8
VUS
1
215
9
0
225
Likely Benign
0
7
5
34
46
Benign
0
1
1
1
3
Conflicting
2
Total52232135286

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

View in ClinVar →

Protein Context — Lollipop Plot

ZFPM2 · 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
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