CST3

Chr 20AD

cystatin C

Also known as: ADLDWA, ARMD11, HEL-S-2

Cystatin C functions as an extracellular inhibitor of cysteine proteases and is found in high concentrations throughout biological fluids and virtually all organs. Mutations cause autosomal dominant cerebral amyloid angiopathy and adult-onset leukodystrophy, with some variants also associated with age-related macular degeneration. The gene shows low constraint to loss-of-function variants (pLI 0.004, LOEUF 1.6), consistent with dominant conditions that may not require complete protein loss.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismADLOEUF 1.603 OMIM phenotypes
Clinical SummaryCST3
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Gene-Disease Validity (ClinGen)
leukodystrophy, adult-onset, autosomal dominant, without amyloid angiopathy · ADLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
💊
Clinical Trials
12 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.60LOEUF
pLI 0.004
Z-score 0.58
OE 0.73 (0.361.60)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.50Z-score
OE missense 1.17 (0.971.41)
81 obs / 69.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.73 (0.361.60)
00.351.4
Missense OE1.17 (0.971.41)
00.61.4
Synonymous OE1.12
01.21.6
LoF obs/exp: 4 / 5.5Missense obs/exp: 81 / 69.4Syn Z: -0.52
DN
0.76top 25%
GOF
0.5660th %ile
LOF
0.2678th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median

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.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CST3 · 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

Healthy Adults

Safety and Efficacy of Injectable Klotho Plasmid Gene Therapy in Humans

RECRUITING
NCT07216781Phase PHASE1MinicircleStarted 2025-10-06
Injectable Plasmid Klotho Gene Therapy
Chronic Kidney DiseasesMAFLD

Pathogenesis of Chronic Kidney Disease Associated With Metabolic Dysfunction- Associated Fatty Liver Disease (MAFLD) and Treatment Response of Oral Semaglutide.

NOT YET RECRUITING
NCT07391267Phase NAInstitute of Liver and Biliary Sciences, IndiaStarted 2026-02-01
Semaglutide Oral TabletPlaceboStandard medical treatment
Mitochondrial DiseasesPearson Syndrome

Evaluate the Safety and Therapeutic Effects of a Single Intravenous Infusion (IV) of Autologous CD34+ Cells Enriched With Allogenic Placenta-derived Mitochondria in Patients With a Diagnosis of Pearson Syndrome (PS)

RECRUITING
NCT06017869Phase PHASE2Minovia Therapeutics Ltd.Started 2023-07-31
MNV-201
Healthy Adults

Safety and Efficacy of Klotho and Follistatin Gene Therapy

RECRUITING
NCT07285629Phase EARLY_PHASE1MinicircleStarted 2025-12-16
Follistatin and klotho gene therapy
Focal Segmental Glomerulosclerosis

A Study to Find Out if BI 764198 Helps Adults and Adolescents With a Kidney Condition Called Focal Segmental Glomerulosclerosis (FSGS)

RECRUITING
NCT07220083Phase PHASE3Boehringer IngelheimStarted 2026-02-16
BI 764198Placebo
Age-related Muscle Decline

CALM-AF-AI: Counteracting Age-related Loss of Muscle With AAV-Follistatin Combined With Angiogenesis-Inducing VEGF Plasmid Gene Therapy

RECRUITING
NCT07443826Phase PHASE1, PHASE2Unlimited Biotechnology LLCStarted 2026-03-31
AAV9-Follistatin gene therapyVEGF Plasmid
Sphingosine Phosphate Lyase Insufficiency Syndrome (SPLIS)

Natural History of Sphingosine Phosphate Lyase Insufficiency Syndrome (SPLIS)

RECRUITING
NCT06669949University of California, San FranciscoStarted 2025-04-22
no intervention
Cognitive DisordersMuscular Disorders, Atrophic

Dietary Strategy to Tackle Cognitive and Locomotor Abilities in Early Elderly Subjects

RECRUITING
NCT06871384Phase NAUniversity Rovira i VirgiliStarted 2025-03-26
Nonalcoholic red wine group (Intervention group)Drinking water group (Control group)
Heart Failure

Acute Reno-Cardiac Action of Dapagliflozin In Advanced Heart Failure Patients on Heart Transplant Waiting List

RECRUITING
NCT06868797Phase NACentral Hospital, Nancy, FranceStarted 2025-08-29
Biological sample for the measurement of suPAR levels.
Chronic Kidney Disease 5D

Effect of Inulin on Gut Microbiota and Gut Barrier in Chronic Kidney Disease

RECRUITING
NCT05071131Phase NACharite University, Berlin, GermanyStarted 2022-02-01
InulinPlacebo
Kidney Disease, ChronicKidney Failure Chronic

Physical Exercise and Biomolecular Analysis to Reduce Uremic Toxins in Chronic Kidney Disease: An Exploratory Study

ENROLLING BY INVITATION
NCT06910475Phase NACatholic University of BrasíliaStarted 2025-04-01
Resistance trainingEndurance trainingConcurrent training
Hereditary Transthyretin Amyloidosis

Exploring Biomarkers in Hereditary Transthyretin Amyloidosis

RECRUITING
NCT05929209Phase NAFondazione Policlinico Universitario Agostino Gemelli IRCCSStarted 2023-05-01
Assessment of disease biomarkers
Clinical Literature
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