CIC

Chr 19AD

capicua transcriptional repressor

Also known as: MRD45

The protein encoded by this gene is an ortholog of the Drosophila melanogaster capicua gene, and is a member of the high mobility group (HMG)-box superfamily of transcriptional repressors. This protein contains a conserved HMG domain that is involved in DNA binding and nuclear localization, and a conserved C-terminus. Studies suggest that the N-terminal region of this protein interacts with Atxn1 (GeneID:6310), to form a transcription repressor complex, and in vitro studies suggest that polyglutamine-expansion of ATXN1 may alter the repressor activity of this complex. Mutations in this gene have been associated with olidogdendrogliomas (PMID:21817013). In addition, translocation events resulting in gene fusions of this gene with both DUX4 (GeneID:100288687) and FOXO4 (GeneID:4303) have been associated with round cell sarcomas. There are multiple pseudogenes of this gene found on chromosomes 1, 4, 6, 7, 16, 20, and the Y chromosome. Alternative splicing results in multiple transcript variants encoding different isoforms. [provided by RefSeq, Feb 2015]

OMIMResearchGenerating clinical summary…
LOFmechanismADLOEUF 0.181 OMIM phenotype
Clinical SummaryCIC
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · 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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ClinVar Variants
82 unique Pathogenic / Likely Pathogenic· 513 VUS of 979 total submissions
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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?
0.18LOEUF
pLI 1.000
Z-score 6.02
OE 0.08 (0.040.18)
Highly constrained

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

Missense Constraint?
0.73Z-score
OE missense 0.93 (0.880.99)
904 obs / 967.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.08 (0.040.18)
00.351.4
Missense OE?0.93 (0.880.99)
00.61.4
Synonymous OE?1.28
01.21.6
LoF obs/exp: 4 / 49.9Missense obs/exp: 904 / 967.5Syn Z: -4.48
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongCIC-related neurodevelopmental disorderLOFAD

This gene — mechanism propensity

DN
0.2299th %ile
GOF
0.2597th %ile
LOF
0.84top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · 1 literature citation · 84% of P/LP variants are LoF · LOEUF 0.18 · ClinGen HI: Sufficient evidence for dosage pathogenicity

Literature Evidence

LOFHere we review four examples from our research group where studying the consequences of altered gene dosage in a mouse led to the discovery of previously unrecognized human syndromes: MECP2 duplication syndrome, SHANK3 duplication syndrome, CIC haploinsufficiency syndrome, and PUM1-related disorders1

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

References

  1. 1.PMID 30030131

ClinVar Variant Classifications

979 submitted variants in ClinVar

Classification Summary

Pathogenic31
Likely Pathogenic51
VUS513
Likely Benign280
Benign17
Conflicting28
31
Pathogenic
51
Likely Pathogenic
513
VUS
280
Likely Benign
17
Benign
28
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
29
1
1
0
31
Likely Pathogenic
40
10
0
1
51
VUS
19
472
11
11
513
Likely Benign
1
105
17
157
280
Benign
0
6
2
9
17
Conflicting
28
Total8959431178920

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

View in ClinVar →

9 pathogenic / likely-pathogenic (of 11) ClinVar copy-number / structural variants overlap CIC — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

CIC · 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

LiposarcomaRecurrent LiposarcomaMetastatic Liposarcoma

BTX-A51 in Patients With Liposarcoma or CIC-rearranged Sarcoma

ACTIVE NOT RECRUITING
NCT06414434Phase PHASE1Michael Wagner, MDStarted 2024-09-30
BTX-A51
ImmunologyAllergyGenetic Diseases

Impaired Type I IFN Immunity Due to Autoantibodies or a Genetic Defect: a Prospective National Cohort

RECRUITING
NCT06762002Institut National de la Santé Et de la Recherche Médicale, FranceStarted 2025-04-09
blood sample
Anatomic Stage I Breast Cancer AJCC v8Anatomic Stage II Breast Cancer AJCC v8Anatomic Stage III Breast Cancer AJCC v8

Additional Support Program Via Text Messaging and Telephone-Based Counseling for Breast Cancer Patients Receiving Hormonal Therapy

ACTIVE NOT RECRUITING
NCT04379570Phase PHASE3Alliance for Clinical Trials in OncologyStarted 2021-02-15
Educational InterventionText Message-based Navigation InterventionMotivational Interviewing
Previously Treated Non-Small Cell Lung Cancer

Lung-MAP: A Master Screening Protocol for Previously-Treated Non-Small Cell Lung Cancer

RECRUITING
NCT03851445Phase PHASE2, PHASE3SWOG Cancer Research NetworkStarted 2019-02-06
Screening Platform
Recurrent Lung Non-Small Cell CarcinomaStage IV Lung Cancer AJCC v8

Adding the Immunotherapy Drug Cemiplimab to Usual Treatment for People With Advanced Non-Small Cell Lung Cancer Who Had Previous Treatment With Platinum Chemotherapy and Immunotherapy (An Expanded Lung-MAP Treatment Trial)

RECRUITING
NCT06616584Phase PHASE2, PHASE3SWOG Cancer Research NetworkStarted 2025-05-22
Biospecimen CollectionCemiplimabComputed Tomography
Autism DisorderASD

GENES AND AUTISM - Induced Pluripotent Stem Cells

NOT YET RECRUITING
NCT07311213Phase NAInstitut National de la Santé Et de la Recherche Médicale, FranceStarted 2026-08-01
Blood Sample : isolation of peripheral blood mononuclear cells (PBMCs)
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
Rare Fetal Genetic DiseasesCongenital Malformation

Characterization and Contribution of Genome-wide DNA Methylation (DNA Methylation Episignatures) in Rare Diseases With Prenatal Onset

RECRUITING
NCT06475651Assistance Publique - Hôpitaux de ParisStarted 2026-02-26
Methylation analysis
High Myopia

Risk of Posterior Staphyloma in Highly Myopic Europeans : From Epidemiology to Anatomy.

RECRUITING
NCT06949579Phase NAAssistance Publique - Hôpitaux de ParisStarted 2026-02-02
blood sampling for DNA
Artemis (DCLRE1C ) Deficient Severe Combined Immunodeficiency

Safety and Efficacy Study of Transplantation of Autologous CD34+ Cells Transduced With the G2ARTE Lentiviral Vector Expressing the DCLRE1C cDNA in Artemis (DCLRE1C) Deficient Severe Combined Immunodeficiency Patients (ARTEGENE)

RECRUITING
NCT05071222Phase PHASE1, PHASE2Assistance Publique - Hôpitaux de ParisStarted 2023-07-19
ARTEGENE drug product
16P11.2 Deletion Syndrome16p11.2 Duplications1Q21.1 Deletion

Online Study of People Who Have Genetic Changes and Features of Autism: Simons Searchlight

RECRUITING
NCT01238250Simons SearchlightStarted 2010-10
Advanced Solid Neoplasms

Efficacy and Safety of Olaparib (MK-7339) in Participants With Previously Treated, Homologous Recombination Repair Mutation (HRRm) or Homologous Recombination Deficiency (HRD) Positive Advanced Cancer (MK-7339-002 / LYNK-002)

ACTIVE NOT RECRUITING
NCT03742895Phase PHASE2Merck Sharp & Dohme LLCStarted 2018-12-12
Olaparib