ITCH

Chr 20

itchy E3 ubiquitin protein ligase

Also known as: ADMFD, AIF4, AIP4, NAPP1

ITCH encodes a HECT domain E3 ubiquitin ligase that transfers ubiquitin to protein substrates for degradation and plays an essential role in regulating inflammatory signaling pathways and immune responses. Biallelic mutations cause autosomal recessive syndromic multisystem autoimmune disease with facial dysmorphism. The gene is highly constrained against loss-of-function variants (pLI 0.999, LOEUF 0.218), reflecting its critical role in immune system regulation.

GeneReviewsResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismLOEUF 0.22
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
30 unique Pathogenic / Likely Pathogenic· 163 VUS of 500 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 ↗
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

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 OE0.12 (0.070.22)
00.351.4
Missense OE0.54 (0.490.60)
00.61.4
Synonymous OE1.10
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

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

500 submitted variants in ClinVar

Classification Summary

Pathogenic20
Likely Pathogenic10
VUS163
Likely Benign251
Benign14
Conflicting6
20
Pathogenic
10
Likely Pathogenic
163
VUS
251
Likely Benign
14
Benign
6
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
11
0
9
0
20
Likely Pathogenic
5
1
4
0
10
VUS
2
146
11
4
163
Likely Benign
0
5
129
117
251
Benign
0
0
14
0
14
Conflicting
6
Total18152167121464

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

View in ClinVar →

Protein Context — Lollipop Plot

ITCH · 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

NaeviNeurodevelopmental DisorderCongenital Nevus

CongenItal Naevus Cohort for Longitudinal Evaluation

RECRUITING
NCT06828822Phase NANantes University HospitalStarted 2025-07-23
Neurodevelopmental assessmentMeeting with the parentsPatient quality of life assessment
Skin MicrobiomeGraft Versus Host DiseaseGraft Versus Host Disease in Skin

The Skin Microbiome in Graft Versus Host Disease

ACTIVE NOT RECRUITING
NCT04231500University Hospital, Basel, SwitzerlandStarted 2023-03-01
Skin swabsSkin punch biopsiesadditional blood sampling
Atopic DermatitisEczema

Defining the Skin and Blood Biomarkers of Pediatric Atopic Dermatitis

ACTIVE NOT RECRUITING
NCT01782703Northwestern UniversityStarted 2013-01
Breast Cancer

Long-Term Follow-Up in Women With Early Breast Cancer Three Years of More Post Primary Treatment

ACTIVE NOT RECRUITING
NCT05926024UNC Lineberger Comprehensive Cancer CenterStarted 2023-01-06
Exercise Recall Patient Reported OutcomesHealth Behavior Questionnaire (HBQ)30 Patient Reported OutcomesFunctional Assessment of Cancer Therapy-General (FACT-G) Patient Reported Outcomes
Genitourinary Syndrome of Menopause

Vaginal CO2 Laser Therapy for Genitourinary Syndrome in Breast Cancer Survivors

RECRUITING
NCT06007027Phase NAUniversity of AarhusStarted 2024-12-01
SmartXIDE2V2LR, Monalisa Touch, DEKA, Florence, Italy
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)
Gut MicrobiotaGastrointestinal SymptomsTemperament

Mother-Infant Cohort Study in Malaysia and China

RECRUITING
NCT04919265Universiti Putra MalaysiaStarted 2022-06-03
No intervention - mother-infant cohort study
Upper Respiratory Tract Infections

Study of the Preventive Effects and Mechanisms of Yeast β-Glucan on Upper Respiratory Tract Infections

NOT YET RECRUITING
NCT07085858Phase NALanZhou UniversityStarted 2025-07-25
yeast β-glucanplacebo
Sensitive Scalp

Pathophysiological Study of the Sensitive Scalp

RECRUITING
NCT07156422Phase NAUniversity Hospital, BrestStarted 2026-01-28
A scalp biopsy using a 4 mm punch in the retroauricular hair zonea general health questionnaireQuestionnaires concerning their sensitive scalp
Atopic Dermatitis

A Study to Assess the Efficacy and Safety of PG-011 Gel in Adolescents and Adults With Atopic Dermatitis

RECRUITING
NCT06587685Phase PHASE3Prime Gene Therapeutics Co., Ltd.Started 2024-03-15
PG-011GelVehicle
Itch

Identification of the Mechanism of Non-histaminergic Itch Inducing Epigenetic Changes

ACTIVE NOT RECRUITING
NCT06997926Phase NAAalborg UniversityStarted 2025-05-15
blood sampling
Drug Induced Liver InjuryPruritus

Drug-induced Liver Injury: Itching Study

RECRUITING
NCT06446609University of NottinghamStarted 2025-06-30
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
When an Itch is not just an Itch.
Smith RC et al.·Ann Allergy Asthma Immunol
2023
Itch Beyond the Skin:Mucosal Itch
Lesslar OJL et al.·Front Allergy
2021
When Itch Becomes Unbearable.
Yong JF et al.·Clin Exp Dermatol
2025
Mind the Itch.
Matuskey D·JAMA Neurol
2025
Top 5 full-text resultsSearch PubTator3 ↗
Key Publications
Landmark & review papers · by relevance
PubMed
Pathophysiology of Itch.
Lerner EA·Dermatol Clin
2018Review
Antihistamines and itch.
Thurmond RL et al.·Handb Exp Pharmacol
2015Review
Lichen Simplex Chronicus Itch: An Update.
Ju T et al.·Acta Derm Venereol
2022
Top 5 results · since 2015Search PubMed ↗