WT1

Chr 11ADSomatic

WT1 transcription factor

Also known as: AWT1, GUD, NPHS4, WAGR, WIT-2, WT-1, WT33

The WT1 protein is a transcription factor with four zinc-finger motifs that is essential for normal urogenital system development. Mutations cause Denys-Drash syndrome, Frasier syndrome, Meacham syndrome, and nephrotic syndrome type 4 through an autosomal dominant inheritance pattern, while somatic mutations can lead to Wilms tumor. The pathogenic mechanism involves loss of function of this critical developmental transcription factor.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismAD/SomaticLOEUF 0.259 OMIM phenotypes
Clinical SummaryWT1
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Gene-Disease Validity (ClinGen)
Wilms tumor 1 · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

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
12 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — WT1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.25LOEUF
pLI 0.996
Z-score 4.31
OE 0.08 (0.030.25)
Highly constrained

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

Missense Constraint
1.78Z-score
OE missense 0.70 (0.630.79)
201 obs / 285.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.08 (0.030.25)
00.351.4
Missense OE0.70 (0.630.79)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 2 / 25.5Missense obs/exp: 201 / 285.7Syn Z: -0.03
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveWT1-related Wilms tumourLOFAD
definitiveWT1-related Denys-Drash syndromeDNAD
DN
0.3892th %ile
GOF
0.2597th %ile
LOF
0.87top 5%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function, dominant-negative 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 0.25
DN1 literature citation
GOF1 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

DNHowever since the DDS phenotype is only elicited by mutations which lead to loss or alteration of ZF function (presumably DNA binding) while the N-terminal upstream portion of the gene remains intact, we suggest that a dominant negative mechanism is at work here.PMID:8388765
GOFDDS is inherited in an autosomal dominant fashion, implying a gain of function by mutant WT1 proteins.PMID:27596598
LOFHeterozygous mutations result in WT1 haploinsufficiency as they impair protein production.PMID:28081536

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

WT1 · 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

Hematopoietic and Lymphatic System NeoplasmRecurrent EpendymomaRecurrent Ewing Sarcoma

Selpercatinib for the Treatment of Advanced Solid Tumors, Lymphomas, or Histiocytic Disorders With Activating RET Gene Alterations, a Pediatric MATCH Treatment Trial

ACTIVE NOT RECRUITING
NCT04320888Phase PHASE2National Cancer Institute (NCI)Started 2021-05-03
Computed TomographyMagnetic Resonance ImagingPositron Emission Tomography
Glioblastoma

INO-5401 and INO-9012 Delivered by Electroporation (EP) in Combination With Cemiplimab (REGN2810) in Newly-Diagnosed Glioblastoma (GBM)

ACTIVE NOT RECRUITING
NCT03491683Phase PHASE1, PHASE2Inovio PharmaceuticalsStarted 2018-05-31
INO-5401INO-9012Cemiplimab
Acute LeukemiaAdenomatous PolyposisAdrenocortical Carcinoma

Familial Investigations of Childhood Cancer Predisposition

RECRUITING
NCT03050268St. Jude Children's Research HospitalStarted 2017-04-06
Pleuropulmonary BlastomaSertoli-Leydig Cell TumorDICER1 Syndrome

International PPB/DICER1 Registry

RECRUITING
NCT03382158Children's Hospitals and Clinics of MinnesotaStarted 2016-12-06
Ewing SarcomaDesmoplastic Small Round Cell TumorPediatric Cancer

Lurbinectedin in FET-Fused Tumors

RECRUITING
NCT05918640Phase PHASE1, PHASE2Children's Hospital of PhiladelphiaStarted 2023-07-27
Lurbinectedin
Malignant Solid NeoplasmRecurrent Adrenal Gland PheochromocytomaRecurrent Ectomesenchymoma

Tipifarnib for the Treatment of Advanced Solid Tumors, Lymphoma, or Histiocytic Disorders With HRAS Gene Alterations, a Pediatric MATCH Treatment Trial

ACTIVE NOT RECRUITING
NCT04284774Phase PHASE2National Cancer Institute (NCI)Started 2020-10-13
Tipifarnib
Diabetic Kidney Disease

Role of Finerenone in African American Veterans With Diabetic Kidney Disease

NOT YET RECRUITING
NCT07155694Phase PHASE4Washington D.C. Veterans Affairs Medical CenterStarted 2025-09
Finerenone 10 MGEmpagliflozin 10 MG
Liver Cell CarcinomaSolid TumorWilms Tumor

Interleukin-15 Armored Glypican 3-specific Chimeric Antigen Receptor Expressed in Autologous T Cells for Solid Tumors

ACTIVE NOT RECRUITING
NCT05103631Phase PHASE1Baylor College of MedicineStarted 2021-06-17
CATCH T cells
Rare DisordersUndiagnosed DisordersDisorders of Unknown Prevalence

Rare Disease Patient Registry & Natural History Study - Coordination of Rare Diseases at Sanford

RECRUITING
NCT01793168Sanford HealthStarted 2010-07
Acute Myeloid LeukemiaMyelodysplastic Syndrome

Administration of Donor Multi TAA-Specific T Cells for AML or MDS (ADSPAM)

ACTIVE NOT RECRUITING
NCT02494167Phase PHASE1Baylor College of MedicineStarted 2016-02
MultiTAA-specific T cells
Liver CancerRhabdomyosarcomaMalignant Rhabdoid Tumor

Interleukin-15 Armored Glypican 3-specific Chimeric Antigen Receptor Expressed in T Cells for Pediatric Solid Tumors

ACTIVE NOT RECRUITING
NCT04377932Phase PHASE1Baylor College of MedicineStarted 2021-08-08
AGAR T cells
Advanced Malignant Solid NeoplasmAnn Arbor Stage III Non-Hodgkin LymphomaAnn Arbor Stage IV Non-Hodgkin Lymphoma

Targeted Therapy Directed by Genetic Testing in Treating Pediatric Patients With Relapsed or Refractory Advanced Solid Tumors, Non-Hodgkin Lymphomas, or Histiocytic Disorders (The Pediatric MATCH Screening Trial)

ACTIVE NOT RECRUITING
NCT03155620Phase PHASE2National Cancer Institute (NCI)Started 2017-07-31
Biopsy ProcedureBiospecimen CollectionBone Marrow Aspiration and Biopsy
Clinical Literature
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