ITCH

Chr 20AR

itchy E3 ubiquitin protein ligase

Also known as: ADMFD, AIF4, AIP4, NAPP1

This gene encodes a member of the Nedd4 family of HECT domain E3 ubiquitin ligases. HECT domain E3 ubiquitin ligases transfer ubiquitin from E2 ubiquitin-conjugating enzymes to protein substrates, thus targeting specific proteins for lysosomal degradation. The encoded protein plays a role in multiple cellular processes including erythroid and lymphoid cell differentiation and the regulation of immune responses. Mutations in this gene are a cause of syndromic multisystem autoimmune disease. Alternatively spliced transcript variants encoding multiple isoforms have been observed for this gene. [provided by RefSeq, Mar 2012]

GeneReviewsOMIMResearchGenerating clinical summary…
LOFmechanismARLOEUF 0.221 OMIM phenotype
Clinical SummaryITCH
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Gene-Disease Validity (ClinGen)
syndromic multisystem autoimmune disease due to ITCH deficiency · ARDefinitive

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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ClinVar Variants
25 unique Pathogenic / Likely Pathogenic· 198 VUS of 557 total submissions
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Clinical Trials
12 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — ITCH
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint?
0.22LOEUF
pLI 1.000
Z-score 6.36
OE 0.12 (0.070.22)
Highly constrained

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

Missense Constraint?
3.58Z-score
OE missense 0.54 (0.490.60)
263 obs / 485.1 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios?
LoF OE?0.12 (0.070.22)
00.351.4
Missense OE?0.54 (0.490.60)
00.61.4
Synonymous OE?1.10
01.21.6
LoF obs/exp: 7 / 60.3Missense obs/exp: 263 / 485.1Syn Z: -1.03
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongITCH-related autoimmune disease, syndromic multisystemLOFAR

This gene — mechanism propensity

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.3694th %ile
GOF
0.4875th %ile
LOF
0.68top 10%

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

557 submitted variants in ClinVar

Classification Summary

Pathogenic17
Likely Pathogenic8
VUS198
Likely Benign274
Benign17
Conflicting7
17
Pathogenic
8
Likely Pathogenic
198
VUS
274
Likely Benign
17
Benign
7
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
13
0
4
0
17
Likely Pathogenic
6
1
1
0
8
VUS
2
178
14
4
198
Likely Benign
0
6
133
135
274
Benign
0
0
15
2
17
Conflicting
7
Total21185167141521

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

View in ClinVar →

16 pathogenic / likely-pathogenic (of 21) ClinVar copy-number / structural variants overlap ITCH — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

ITCH · 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

Cystinosis

European Cystinosis Cohort

RECRUITING
NCT05901077Institut National de la Santé Et de la Recherche Médicale, FranceStarted 2017-04-20
Recurrent Urinary Tract InfectionGenitourinary Syndrome of Menopause

RCT Comparing Intravaginal Laser Therapy to Sham in Post-menopausal Women with Recurrent Urinary Tract Infections

ACTIVE NOT RECRUITING
NCT06124820Phase NAKing's College Hospital NHS TrustStarted 2023-10-01
Deka SmartXide Touch C60 (MonaLisa Touch)Sham
Epidermolysis Bullosa Dystrophica, Recessive

Ex Vivo Gene Therapy Clinical Trial for RDEB Using Genetically Corrected Autologous Skin Equivalent Grafts

ACTIVE NOT RECRUITING
NCT04186650Phase PHASE1, PHASE2Institut National de la Santé Et de la Recherche Médicale, FranceStarted 2020-01-10
COL7A1-SIN retroviral vector engineered autologous tissue-engineered skin
IPEX

CD4^LVFOXP3 in Participants With IPEX

RECRUITING
NCT05241444Phase PHASE1Bacchetta, Rosa, MDStarted 2022-03-22
CD4^LVFOXP3
Lichen Simplex Chronicus

A Phase I Clinical Trial to Evaluate the Single Dose Ascending and Food Effects of PG-033 in Healthy Adults

RECRUITING
NCT07013097Phase PHASE1Prime Gene Therapeutics Co., Ltd.Started 2025-05-23
PG-033PG-033 placebo comparator
Vulvar AtrophyVaginal Atrophy

Clinical and Molecular Study to Evaluate the Effect of the Pixel CO2 Laser (FemiLiftTM) for the Treatment of Vulvo-Vaginal Atrophy

RECRUITING
NCT07024667Phase NAHillel Yaffe Medical CenterStarted 2022-09-02
Pixel CO2 Laser for the Treatment of Vulvo-Vaginal Atrophy (VVA)
Idiopathic Rhinitis

Posterior Nasal Nerve Combined with Anterior Ethmoid Neurotomy for Idiopathic Rhinitis

RECRUITING
NCT06870292Phase NAXu YuStarted 2025-03-01
Posterior nasal nerve combined with anterior ethmoid neurotomyDrug therapy group
Polycythemia Vera (PV)

Phase IIa Study on Flonoltinib Maleate Tablets in the Treatment of Patients With Polycythemia Vera

RECRUITING
NCT07232290Phase PHASE2Chengdu Zenitar Biomedical Technology Co., LtdStarted 2025-09-23
Flonoltinib 75mgFlonoltinib 100mgFlonoltinib 125mg
Primary MyelofibrosisPost-polycythemia Vera MyelofibrosisPost-essential Thrombocythemia Myelofibrosis

A Phase 3 Study of Pacritinib in Patients With Primary Myelofibrosis, Post Polycythemia Vera Myelofibrosis, or Post-Essential Thrombocythemia Myelofibrosis

ACTIVE NOT RECRUITING
NCT03165734Phase PHASE3Swedish Orphan BiovitrumStarted 2017-06-26
PacritinibPhysician's Choice medications
Atopic Dermatitis

Mind and Skin - the Neurocutaneous Axis in Atopic Eczema

RECRUITING
NCT05790330King's College LondonStarted 2023-03-10
MethotrexateDupilumab
Colo-rectal CancerPD-1 InhibitorAkkermansia Muciniphila

Akkermansia Probiotics Plus Anti-PD-1 Monoclonal Antibody in MSS/pMMR Advanced Colorectal Cancer

ACTIVE NOT RECRUITING
NCT06865521Phase NAWest China HospitalStarted 2025-02-21
Akkermansia Probiotics
Drug Induced Liver InjuryPruritus

Drug-induced Liver Injury: Itching Study

RECRUITING
NCT06446609University of NottinghamStarted 2025-06-30