PCSK9

Chr 1AD

proprotein convertase subtilisin/kexin type 9

Also known as: FH3, FHCL3, HCHOLA3, LDLCQ1, NARC-1, NARC1, PC9

The encoded protease regulates plasma cholesterol homeostasis by binding to low-density lipoprotein receptor family members and promoting their degradation, preventing cholesterol uptake from the bloodstream. Mutations cause autosomal dominant familial hypercholesterolemia, leading to elevated cholesterol levels and increased cardiovascular risk. This gene is not highly constrained against loss-of-function mutations and primarily affects lipid metabolism rather than neurological function.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
GOFmechanismADLOEUF 1.342 OMIM phenotypes
Clinical SummaryPCSK9
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Gene-Disease Validity (ClinGen)
hypercholesterolemia, autosomal dominant, 3 · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
12 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — PCSK9
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.34LOEUF
pLI 0.000
Z-score 0.16
OE 0.97 (0.711.34)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.27Z-score
OE missense 0.96 (0.891.04)
419 obs / 435.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.97 (0.711.34)
00.351.4
Missense OE0.96 (0.891.04)
00.61.4
Synonymous OE0.91
01.21.6
LoF obs/exp: 26 / 26.9Missense obs/exp: 419 / 435.0Syn Z: 1.01
DN
0.5870th %ile
GOF
0.5464th %ile
LOF
0.4234th %ile

The Badonyi & Marsh model scores dominant-negative highest, but genomic evidence most strongly supports gain-of-function as the primary mechanism.

GOF1 literature citation

Literature Evidence

GOFThe LDL-C and PCSK9 levels in patients with gain-of-function (GOF) variants of PCSK9 (n = 7) were mostly similar to those in patients with LDLR variants (n = 17) or variant-negative patients (n = 46).PMID:34652028

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

PCSK9 · 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

Familial Hypercholesterolemia

EPIRUS FH Reverse Cascade Screening

NOT YET RECRUITING
NCT05825612Hellenic Atherosclerosis SocietyStarted 2023-05
Coronary Artery DiseaseAtherosclerosis, Coronary

Latvian Early Atherosclerosis Registry

RECRUITING
NCT06393894Pauls Stradins Clinical University HospitalStarted 2019-04-01
Near infrared spectroscopyGenetic testing for LDLR, APOB, PCSK9 and LDLRAP1 mutations and niR-126, -145 and -155 expression.
Type 2 DiabetesInflammationInsulin Sensitivity/Resistance

Targeting Risk Factors for Diabetes in Subjects With Normal Blood Cholesterol Using Omega-3 Fatty Acids

RECRUITING
NCT04485871Phase NAInstitut de Recherches Cliniques de MontrealStarted 2019-12-19
Omega-3 fatty acids
LDL Hyper-responsiveness

A Study of Genetic Influence on LDL Hyper-responsiveness in Patients Following a Ketogenic Diet

RECRUITING
NCT07137286Mayo ClinicStarted 2025-09-11
Heterozygous Familial Hypercholesterolemia

Early-phase Study of ART002g1 Injection in HeFH: Safety, Tolerability and Preliminary Efficacy

NOT YET RECRUITING
NCT07353398Phase EARLY_PHASE1Shanghai General Hospital, Shanghai Jiao Tong University School of MedicineStarted 2026-03-11
ART002g1 Injection
Familial Hypercholesterolemia

Russian Familial Hypercholesterolemia Registry

RECRUITING
NCT02208869Russian Cardiology Research and Production CenterStarted 2014-01
Familial Hypercholesterolemia

Clinical Exploration Trial of YOLT-101 in the Treatment of Familial Hypercholesterolemia (FH)

RECRUITING
NCT06458010Phase EARLY_PHASE1RenJi HospitalStarted 2024-05-24
YOLT-101
Familial Hypercholesterolemia

EAS Familial Hypercholesterolaemia Studies Collaboration

RECRUITING
NCT04272697Imperial College LondonStarted 2015-03-22
Heterozygous Familial HypercholesterolemiaPremature Coronary Heart Disease

A Study of VERVE-102 in Patients With Familial Hypercholesterolemia or Premature Coronary Artery Disease

RECRUITING
NCT06164730Phase PHASE1Verve Therapeutics, Inc.Started 2024-04-30
VERVE-102
Ornithine Transcarbamylase DeficiencyOrnithine Transcarbamylase Deficiency DiseaseOrnithine Carbamoyltransferase Deficiency (Disorder)

An Open-label Study to Investigate ECUR-506 in Male Babies Less Than 9 Months of Age With Neonatal Onset OTC Deficiency

RECRUITING
NCT06255782Phase PHASE1, PHASE2iECURE, Inc.Started 2024-04-08
ECUR-506
Elevated LDL-C and High Cholesterol

A Study of STX-1150 in Participants With Elevated Low-Density Lipoprotein Cholesterol (LDL-C)

NOT YET RECRUITING
NCT07428473Phase PHASE1Monash UniversityStarted 2026-06-01
STX-1150
Type 1 Diabetes

Cholesterol Lowering and Residual Risk in Diabetes, Type 1

RECRUITING
NCT05641753Phase PHASE4NYU Langone HealthStarted 2022-12-06
Evolocumab CartridgeAtorvastatin Calcium TabletsEzetimibe Tablets
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
The Multifaceted Biology of PCSK9
Seidah NG et al.·Endocr Rev
2022
Safety of PCSK9 inhibitors.
Grześk G et al.·Biomed Pharmacother
2022
PCSK9 Inhibition
Ungvari Z·JACC Basic Transl Sci
2023
Phage display for targeting PCSK9
Ferri N·EBioMedicine
2021
Top 5 full-text resultsSearch PubTator3 ↗
Key Publications
Landmark & review papers · by relevance
PubMed
PCSK9 Inhibitors: Mechanism of Action, Efficacy, and Safety.
Roth EM et al.·Rev Cardiovasc Med
2018Review
Targeting PCSK9 to tackle cardiovascular disease.
Hummelgaard S et al.·Pharmacol Ther
2023Review
MK-0616: an oral PCSK9 inhibitor for hypercholesterolemia treatment.
Burnett JR et al.·Expert Opin Investig Drugs
2023Review
Top 5 results · since 2015Search PubMed ↗