ZEB2

Chr 2AD

zinc finger E-box binding homeobox 2

Also known as: HSPC082, SIP-1, SIP1, SMADIP1, ZFHX1B

The encoded protein is a DNA-binding transcriptional repressor that interacts with activated SMADs and localizes to the nucleus. Loss-of-function mutations cause Mowat-Wilson syndrome through an autosomal dominant inheritance pattern. The gene is highly intolerant to loss-of-function variation, consistent with haploinsufficiency as the disease mechanism.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismADLOEUF 0.111 OMIM phenotype
Clinical SummaryZEB2
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Gene-Disease Validity (ClinGen)
Mowat-Wilson syndrome · 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 1.00). One damaged copy is likely sufficient to cause disease.
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Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.11LOEUF
pLI 1.000
Z-score 6.03
OE 0.02 (0.010.11)
Highly constrained

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

Missense Constraint
3.94Z-score
OE missense 0.57 (0.520.62)
378 obs / 663.9 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.02 (0.010.11)
00.351.4
Missense OE0.57 (0.520.62)
00.61.4
Synonymous OE1.02
01.21.6
LoF obs/exp: 1 / 44.3Missense obs/exp: 378 / 663.9Syn Z: -0.23
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveZEB2-related Mowat-Wilson syndromeLOFAD
DN
0.19100th %ile
GOF
0.1799th %ile
LOF
0.90top 5%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). 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 · 1 literature citation · LOEUF 0.11
GOF1 literature citation

Literature Evidence

GOFUsing a conditional gain-of-function mouse model, we recently demonstrated that ZEB2 is an oncogenic driver of immature T-cell acute lymphoblastic leukemia (T-ALL), a heterogenic subgroup of human leukemia characterized by a high incidence of remission failure or hematological relapse after conventiPMID:28069602
LOFHaploinsufficiency of the ZEB2 gene may predispose to MWS.PMID:31813148

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

ZEB2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold
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 ↗