C9ORF72

Chr 9AD

C9orf72-SMCR8 complex subunit

Also known as: ALSFTD, DENND9, DENNL72, FTDALS, FTDALS1

The protein encoded by this gene plays an important role in the regulation of endosomal trafficking, and has been shown to interact with Rab proteins that are involved in autophagy and endocytic transport. Expansion of a GGGGCC repeat from 2-22 copies to 700-1600 copies in the intronic sequence between alternate 5' exons in transcripts from this gene is associated with 9p-linked ALS (amyotrophic lateral sclerosis) and FTD (frontotemporal dementia) (PMID: 21944778, 21944779). Studies suggest that hexanucleotide expansions could result in the selective stabilization of repeat-containing pre-mRNA, and the accumulation of insoluble dipeptide repeat protein aggregates that could be pathogenic in FTD-ALS patients (PMID: 23393093). Alternative splicing results in multiple transcript variants encoding different isoforms. [provided by RefSeq, Jul 2016]

GeneReviewsOMIMResearchGenerating clinical summary…

Primary Disease Associations & Inheritance

Frontotemporal dementia and/or amyotrophic lateral sclerosis 1MIM #105550
AD

Clinical highlights

Gene-disease validity (ClinGen)
frontotemporal dementia and/or amyotrophic lateral sclerosis 1 · ADDefinitivesufficient evidence for diagnostic panels
12
Active trials
409
Pubs (1 yr)
P/LP submissions
P/LP missense
0.93
LOEUF
Mechanism
📖
GeneReview available — C9ORF72
Authoritative clinical overview · Recommended first read
Open GeneReview ↗
  • C9orf72 ASO programs (e.g. BIIB078, WVE-004)
    ASODiscontinued

    Lower the repeat-containing transcript.

    Delivery: Intrathecal

    Phase 1 programs discontinued after not meeting endpoints — shown for transparency

Therapeutic landscape as of 2026-07. Educational only. Investigational ≠ available; not medical advice or eligibility. Approved entries are precise; investigational program names/phases are conservative and move fast. Curated from FDA/EMA approvals and the clinical-trial literature; verify against current labeling + ClinicalTrials.gov.

ClinicalTrials.gov

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
0.93LOEUF
pLI 0.000
Z-score 1.82
OE 0.58 (0.380.93)
Tolerant

Typical tolerance to LoF variation

Missense Constraint?
0.24Z-score
OE missense 0.96 (0.861.06)
240 obs / 250.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.58 (0.380.93)
00.351.4
Missense OE?0.96 (0.861.06)
00.61.4
Synonymous OE?1.16
01.21.6
LoF obs/exp: 13 / 22.3Missense obs/exp: 240 / 250.9Syn Z: -1.20

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

C9ORF72 · 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

Amyotrophic Lateral Sclerosis

A Study Evaluating the Safety and Tolerability of QRL-201 in ALS

ACTIVE NOT RECRUITING
NCT05633459Phase PHASE1QurAlis CorporationStarted 2022-12-16
Multiple ascending doses of QRL-201Multiple ascending doses of PlaceboQRL-201
Neurodegenerative DiseaseBehavioral Variant Frontotemporal Dementia (bvFTD)Primary Progressive Aphasia(PPA)

Tracking and Predicting How Brain Damage Spreads in Neurodegenerative Diseases

ENROLLING BY INVITATION
NCT07567664Phase NAIRCCS San RaffaeleStarted 2017-06-01
3 Tesla MRI without contrast mediumBlood sample for genetic analysisCerebrospinal fluid sampling (CSF)
Corticobasal Syndrome(CBS)Primary Progressive Aphasia(PPA)Progressive Supranuclear Palsy(PSP)

Neuroinflammation in FTLD

ACTIVE NOT RECRUITING
NCT06870838Leiden University Medical CenterStarted 2023-07-25
7T MRI scanCSFBlood withdrawal
Neurodegenerative DiseasesDementia

Neurofilament Light Chain And Voice Acoustic Analyses In Dementia Diagnosis

RECRUITING
NCT06339190Monash UniversityStarted 2021-08-01
Venepuncture
Frontotemporal DementiaFTDFTD-GRN

A Study of PBFT02 in Participants With FTD and Mutations in the Granulin Precursor (GRN) or C9ORF72 Genes

ACTIVE NOT RECRUITING
NCT04747431Phase PHASE1, PHASE2Passage Bio, Inc.Started 2021-09-14
PBFT02
Amyotrophic Lateral SclerosisFrontotemporal DementiaALS-Frontotemporal Dementia

TRIAL READY (Clinical Trial Readiness)

ACTIVE NOT RECRUITING
NCT03912987University of MiamiStarted 2019-01-22
Frontotemporal DementiaFrontotemporal Lobar DegenerationFTD-GRN

Neurofilament Surveillance Project (NSP)

ACTIVE NOT RECRUITING
NCT04516499The Bluefield Project to Cure Frontotemporal DementiaStarted 2020-09-02
Cerebellar Ataxias

The Benefits of Long-read High-throughput Genomic Sequencing for the Causal Diagnosis of Cerebellar Ataxias

RECRUITING
NCT06467175Phase NACentre Hospitalier Universitaire DijonStarted 2024-12-11
blood sampling for high molecular weight DNA extraction
Amyotrophic Lateral Sclerosis

Needs of ALS Patients With C9orf72 Mutation and Their Caregivers

RECRUITING
NCT07302321Istituto Auxologico ItalianoStarted 2025-10-01
Survey for ALS patientsSurvey for Caregivers
Amyotrophic Lateral Sclerosis

Longitudinal Assessment of Autonomic and Sensory Nervous System in ALS

RECRUITING
NCT05747937Phase NAIstituti Clinici Scientifici Maugeri SpAStarted 2021-05-15
Skin biopsyCardiovascular Reflexes testingAdministration of clinical scales evaluating autonomic symptoms, pain small fiber neuropathy symptoms
Frontotemporal Dementia

GENetic Fronto Temporal Dementia Initiative in Lille

RECRUITING
NCT04639622Phase NAUniversity Hospital, LilleStarted 2019-04-23
Investigation procedures
Amyotrophic Lateral Sclerosis (ALS)

Testing Pulse Stimulation to Improve Motor Function in People With ALS: A Pilot Study

RECRUITING
NCT06681610Phase NAParc de Salut MarStarted 2024-10-24
Transcranial Pulse Stimulation