CTLA4

Chr 2AD

cytotoxic T-lymphocyte associated protein 4

Also known as: ALPS5, CD, CD152, CELIAC3, CTLA-4, GRD4, GSE, IDDM12

The CTLA4 protein is an inhibitory receptor that acts as a major negative regulator of T-cell immune responses by outcompeting the stimulatory CD28 receptor for binding to CD80 and CD86 ligands. Mutations cause CTLA4 haploinsufficiency, which presents with immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX)-like syndrome featuring recurrent infections, autoimmune manifestations, and lymphoproliferation, typically with onset in infancy or early childhood. The gene is highly constrained against loss-of-function variants (pLI=0.94, LOEUF=0.33) and follows autosomal dominant inheritance with variable expressivity.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismADLOEUF 0.331 OMIM phenotype
Clinical SummaryCTLA4
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Gene-Disease Validity (ClinGen)
autoimmune lymphoproliferative syndrome due to CTLA4 haploinsufficiency · 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 0.94). 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

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.33LOEUF
pLI 0.941
Z-score 2.77
OE 0.00 (0.000.33)
Highly constrained

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

Missense Constraint
1.69Z-score
OE missense 0.58 (0.480.70)
74 obs / 127.6 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.33)
00.351.4
Missense OE0.58 (0.480.70)
00.61.4
Synonymous OE0.88
01.21.6
LoF obs/exp: 0 / 9.0Missense obs/exp: 74 / 127.6Syn Z: 0.65
DN
0.4884th %ile
GOF
0.6540th %ile
LOF
0.51top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function, gain-of-function and dominant-negative). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOF1 literature citation · LOEUF 0.33
GOFprediction above median
DN1 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

DNAs CTLA-4 forms homodimers and clusters with its ligands, these mutants may exert a dominant negative effect.PMID:25329329
LOFTaken together, mutations in CTLA4 resulting in CTLA-4 haploinsufficiency or impaired ligand binding result in disrupted T and B cell homeostasis and a complex immune dysregulation syndrome.PMID:25329329

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

CTLA4 · 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

Ovary Cancer

RC48 in Combination With AK104 and Bevacizumab in OCCC

RECRUITING
NCT06540729Phase PHASE2Sun Yat-Sen Memorial Hospital of Sun Yat-Sen UniversityStarted 2024-09-24
Disitamab vedotin in combination with AK104 and bevacizumab for the treatment of recurrent and persistent clear cell ovarian cancer
Cervical Cancer

Exploring the Efficacy, Safety and Cost-effectiveness Analysis of Cadonilimab in the Treatment of Cervical Cancer

RECRUITING
NCT06140589Fujian Cancer HospitalStarted 2024-09-07
Cadonilimab
Melanoma Stage IIIMelanoma Stage IVAdvanced Melanoma

Neoadjuvant and Adjuvant Anti-PD1 or Combinations for Locoregionally Advanced Melanoma

RECRUITING
NCT06295159Phase PHASE2H. Lee Moffitt Cancer Center and Research InstituteStarted 2024-05-17
NivolumabNivolumab + RelatlimabIpilimumab
Patients With Metastatic Tumours (Colorectal Cancer, Non-small Lung Cancer, Renal Cell Carcinoma or Sarcoma )

Atezolizumab With Stereotactic Ablative Radiotherapy in Patients With Metastatic Tumours

ACTIVE NOT RECRUITING
NCT02992912Phase PHASE2Gustave Roussy, Cancer Campus, Grand ParisStarted 2016-11-15
Anti-PD-L1 antibody atezolizumabSABR
Breast Cancer MetastaticBreast Carcinoma

Immun Checkpoint Washout in Patients With Invasive Ductal Breast Cancer

RECRUITING
NCT07003009Phase NAIstanbul Training and Research HospitalStarted 2024-01-01
Malign Lymph Node WashoutBenign Lymph Node Washout
Advanced Pancreatic Cancer

Recombinant Human IL-21-expressing Oncolytic Vaccinia Virus Injection (hV01) in Advanced Pancreatic Cancer

RECRUITING
NCT07006077Phase PHASE2Hangzhou Converd Co., Ltd.Started 2025-05-29
Recombinant human IL-21-expressing oncolytic vaccinia virus injection
Multiple Myeloma

A Clinical Study of TQB2029 for Injection in Subjects With Multiple Myeloma

RECRUITING
NCT06700395Phase PHASE1Chia Tai Tianqing Pharmaceutical Group Nanjing Shunxin Pharmaceutical Co., Ltd.Started 2024-11-28
TQB2029 injection
All Solid Tumors

Feasibility Study of MET-4: Evaluating Fecal Microbiome Effects in Immunotherapy Patients

ACTIVE NOT RECRUITING
NCT03686202Phase PHASE2, PHASE3University Health Network, TorontoStarted 2018-11-30
MET-4
Meningiomas

An Open-Label Phase II Study of Nivolumab or Nivolumab/Ipilimumab in Adult Participants With Progessive/ Recurrent Meningioma

ACTIVE NOT RECRUITING
NCT02648997Phase PHASE2Dana-Farber Cancer InstituteStarted 2016-03
Nivolumab - 240 mgIpilimumab - 1 mg/kgNivolumab - 480 mg
ALK Gene RearrangementEGFR Gene MutationLocally Advanced Lung Non-Small Cell Carcinoma

Ipilimumab and Nivolumab in Combination With Radiation Therapy in Treating Patients With Stage 2-3 Non-small Lung Cancer

ACTIVE NOT RECRUITING
NCT04013542Phase PHASE1M.D. Anderson Cancer CenterStarted 2019-09-13
IpilimumabNivolumabRadiation Therapy
Solid Cancers

Monitoring of Immunological Mechanisms and Biomarkers Underlying Efficacy and Toxicity of Cancer Immunotherapy

RECRUITING
NCT03514368Phase NAInstitut Claudius RegaudStarted 2018-05-28
Patients treated with immune checkpoint blockade
Clinical Stage III Cutaneous Melanoma AJCC v8Clinical Stage IV Cutaneous Melanoma AJCC v8Metastatic Melanoma

Dabrafenib and Trametinib Followed by Ipilimumab and Nivolumab or Ipilimumab and Nivolumab Followed by Dabrafenib and Trametinib in Treating Patients With Stage III-IV BRAFV600 Melanoma

ACTIVE NOT RECRUITING
NCT02224781Phase PHASE3National Cancer Institute (NCI)Started 2015-09-08
Biospecimen CollectionComputed TomographyDabrafenib Mesylate
Clinical Literature
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