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FUS

FUS RNA Binding Protein

16p11.2Autosomal DominantOMIM 137070
Also known as: ALS6, ETM4, FUS1, HNRNPP2, POMP75, TLS, altFUS
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Gene Summary

RefSeq / NCBI

This gene encodes a multifunctional protein component of the heterogeneous nuclear ribonucleoprotein (hnRNP) complex. The hnRNP complex is involved in pre-mRNA splicing and the export of fully processed mRNA to the cytoplasm. This protein belongs to the FET family of RNA-binding proteins which have been implicated in cellular processes that include regulation of gene expression, maintenance of genomic integrity and mRNA/microRNA processing. Alternative splicing results in multiple transcript variants. Defects in this gene result in amyotrophic lateral sclerosis type 6. [provided by RefSeq, Sep 2009]

Clinical Phenotype

Familial ALS (FUS-ALS) and FTD-FUS. FUS-ALS typically has very early onset (teens to 30s) with rapid progression. Often presents with flail arm or flail leg syndrome. FUS mutations account for ~4% of fALS.

Molecular Mechanism

FUS (Fused in Sarcoma) is an RNA-binding protein involved in transcription, pre-mRNA splicing, and mRNA transport. ALS mutations impair nuclear import (disrupting PY-NLS interaction with Transportin-1), causing cytoplasmic mislocalization and stress granule incorporation, leading to toxic aggregates.

Clinical Hallmarks & Key Evidence

1

FUS-ALS uniquely features cytoplasmic FUS inclusions (not TDP-43), and pathology is absent from TDP-43 IHC — a key pathological distinction from the majority of ALS cases.

Kwiatkowski TJ Jr et al. Science. 2009;323(5918):1205-8.

2

Juvenile-onset ALS (before age 25) is disproportionately caused by FUS mutations — any young ALS patient should trigger urgent FUS genetic testing and family assessment.

Conte A et al. J Neurol Neurosurg Psychiatry. 2012;83(3):328-30.

3

Arginine methylation of FUS (by PRMT1) regulates its nuclear/cytoplasmic partitioning; arginine-to-cysteine mutations abolish methylation and maximally mislocalise FUS — correlating with the most aggressive phenotypes.

Dormann D et al. Nature. 2010;467(7311):729-33.

Associated Conditions

Neuromuscular Journal Club: Articles
Autosomal DominantAutosomal RecessiveMitochondrialGeneral

External Resources

WUSTL Neuromuscular
Washington University Disease Center
OMIM
Online Mendelian Inheritance in Man
ClinGen
Clinical Genome Resource
G2P
Gene-to-Phenotype (EBI)
GeneReviews
NCBI Expert-Authored Reviews
NCBI Gene
National Center for Biotechnology Information
PubMed
Biomedical Literature
UniProt
Universal Protein Resource
DECIPHER
DatabasE of genomiC varIation and Phenotype in Humans

Gene data compiled from the Washington University Neuromuscular Disease Center, NCBI Gene, and OMIM. For clinical use, always refer to primary sources.