FUS

Chr 16AD

FUS RNA binding protein

Also known as: ALS6, ETM4, FUS1, HNRNPP2, POMP75, TLS, altFUS

The protein is a component of the heterogeneous nuclear ribonucleoprotein complex that regulates pre-mRNA splicing and export of processed mRNA to the cytoplasm, and belongs to the FET family of RNA-binding proteins involved in gene expression regulation and genomic integrity maintenance. Mutations cause autosomal dominant amyotrophic lateral sclerosis type 6 with or without frontotemporal dementia, and hereditary essential tremor type 4. The pathogenic mechanism involves loss of function, with the gene showing extreme intolerance to loss-of-function variants.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismADLOEUF 0.242 OMIM phenotypes
Clinical SummaryFUS
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Gene-Disease Validity (ClinGen)
amyotrophic lateral sclerosis type 6 · 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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Clinical Trials
6 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
0.24LOEUF
pLI 0.999
Z-score 5.16
OE 0.10 (0.050.24)
Highly constrained

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

Missense Constraint
2.21Z-score
OE missense 0.66 (0.590.74)
217 obs / 329.7 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.10 (0.050.24)
00.351.4
Missense OE0.66 (0.590.74)
00.61.4
Synonymous OE1.25
01.21.6
LoF obs/exp: 4 / 38.6Missense obs/exp: 217 / 329.7Syn Z: -2.13
DN
0.4686th %ile
GOF
0.3392th %ile
LOF
0.79top 5%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to loss-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFprediction above median · LOEUF 0.24
GOF1 literature citation

Literature Evidence

GOFThe implications of this would be a progressive generalized disruption to transcription and RNA processing, and suggests a mechanism by which C-terminal FUS/TLS mutations may exert their effects by a toxic gain of function.PMID:20385912

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

FUS · 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

Amyotrophic Lateral Sclerosis

Development of Targeted RNA-Seq for Amyotrophic Lateral Sclerosis Diagnosis

RECRUITING
NCT06083584Centre Hospitalier Universitaire de NīmesStarted 2023-11-22
RNA sequencing
Amyotrophic Lateral Sclerosis (ALS)

Study on the Safety and Efficacy of RAG-21 in the Treatment of Amyotrophic Lateral Sclerosis Patients With FUS Gene Mutations

NOT YET RECRUITING
NCT07080801Phase EARLY_PHASE1Beijing Tiantan HospitalStarted 2025-08
RAG-21
ALS

Amyotrophic Lateral Sclerosis (ALS) Families Project

RECRUITING
NCT03865420Columbia UniversityStarted 2018-09-11
Brain NeoplasmsLiquid Biopsy

BRAINFUL (BRAIN Tumor Focused Ultrasound-enabled Liquid Biopsy) Trial

ACTIVE NOT RECRUITING
NCT04940507Phase NAUniversity Health Network, TorontoStarted 2021-06-10
Magnetic Resonance Guided Focused Ultrasound Tumor Ablation and Liquid Biopsy AcquisitionMagnetic Resonance Guided Focused Ultrasound Thalamotomy
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
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)
Clinical Literature
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