HomeAboutRankingsData Sources
Β© 2026 GeneE
🧬
GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
β“˜GeneE is for informational purposes only. It is not a substitute for professional medical advice, diagnosis, or treatment.
EXOSC3
exosome component 3
Chromosome 9 Β· 9p13.2
NCBI Gene: 51010Ensembl: ENSG00000107371.15HGNC: HGNC:17944UniProt: Q9NQT5
128PubMed Papers
21Diseases
0Drugs
50Pathogenic Variants
FUNCTIONAL ROLE
Hub Gene
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
protein bindingcytoplasmic exosome (RNase complex)rRNA processingeuchromatinpontocerebellar hypoplasia type 1pontocerebellar hypoplasianeurodegenerative diseaseNon-syndromic pontocerebellar hypoplasia
✦AI Summary

EXOSC3 encodes a non-catalytic structural component of the RNA exosome complex, a highly conserved ribonuclease machinery essential for RNA processing and degradation 1. As part of the catalytically inactive exosomal core complex, EXOSC3 stabilizes the hexameric ring of RNase PH-domain subunits and facilitates RNA binding and presentation for ribonucleolysis 2. In the nucleus, the exosome processes stable RNAs (rRNA, snRNA, snoRNA) and eliminates defective transcripts, while cytoplasmic exosome activity degrades unstable mRNAs and enforces RNA surveillance 1. Recent evidence indicates EXOSC3 interacts with the RNA helicase DDX1 in neuronal cells, modulating R-loop accumulation and gene expression regulation 3. Recessive EXOSC3 mutations cause pontocerebellar hypoplasia type 1b (PCH1b), a severe neurodegenerative disease characterized by cerebellar atrophy, progressive microcephaly, spinal motor neuron degeneration, and profound developmental delay 21. PCH1b presents as various neuromuscular phenotypes including motor neuronopathy and non-5q spinal muscular atrophy 4. Disease-causing mutations reduce EXOSC3 affinity for G-rich RNA sequences, impairing RNA-binding capacity 5. Notably, EXOSC3 mutations also cause eculizumab-nonresponsive atypical hemolytic uremic syndrome, suggesting pleiotropic effects of defective RNA exosome function 6. The tissue-specific neurological manifestation remains incompletely understood.

Sources cited
1
EXOSC3 mutations cause pontocerebellar hypoplasia type 1 with spinal motor neuron degeneration, cerebellar atrophy, and developmental delay; first RNA exosome core component gene linked to human disease
PMID: 22544365
2
EXOSC3 mutations cause PCH1b, a neurodegenerative disease; overview of RNA exosome structure and function in disease
PMID: 31768969
3
EXOSC3 mutations cause PCH1b, an autosomal-recessive neurodegenerative disease; discusses RNA exosome role in processing and degradation
PMID: 29093021
4
PCH1B-causing EXOSC3 mutations exhibit lower binding affinity to G-rich RNA sequences; small molecule modeling of EXOSC3 dysfunction produces PCH1B-like phenotype
PMID: 30141626
5
EXOSC3 associates with RNA helicase DDX1 in neuronal cells; EXOSC3 and DDX1 interact to modulate R-loops and regulate gene expression during cellular homeostasis
PMID: 38219817
6
Biallelic pathogenic mutations in EXOSC3 cause eculizumab-nonresponsive atypical hemolytic uremic syndrome
PMID: 37369098
7
EXOSC3 variants associated with PCH1 presenting as motor neuronopathy, spinal muscular atrophy, or sensorimotor polyneuropathy; PCH1 involves RNA metabolism dysfunction
PMID: 40428407
Disease Associationsβ“˜21
pontocerebellar hypoplasia type 1Open Targets
0.76Strong
pontocerebellar hypoplasiaOpen Targets
0.60Moderate
neurodegenerative diseaseOpen Targets
0.53Moderate
Non-syndromic pontocerebellar hypoplasiaOpen Targets
0.52Moderate
genetic disorderOpen Targets
0.41Moderate
pontocerebellar hypoplasia type 1AOpen Targets
0.37Weak
microcephalyOpen Targets
0.34Weak
Abnormal cerebellum morphologyOpen Targets
0.34Weak
Abnormality of the nervous systemOpen Targets
0.34Weak
congenital myopathyOpen Targets
0.34Weak
fetal akinesia deformation sequenceOpen Targets
0.34Weak
fetal akinesia deformation sequence 1Open Targets
0.34Weak
Hypoplasia of the ponsOpen Targets
0.34Weak
HypotoniaOpen Targets
0.34Weak
LissencephalyOpen Targets
0.34Weak
Paucity of anterior horn motor neuronsOpen Targets
0.34Weak
Severe intrauterine growth retardationOpen Targets
0.34Weak
complex hereditary spastic paraplegiaOpen Targets
0.18Weak
hepatocellular carcinomaOpen Targets
0.07Suggestive
renal cysts and diabetes syndromeOpen Targets
0.05Suggestive
Pontocerebellar hypoplasia 1BUniProt
Pathogenic Variants50
NM_016042.4(EXOSC3):c.238G>T (p.Val80Phe)Pathogenic
Pontocerebellar hypoplasia type 1B|Pontoneocerebellar hypoplasia|not provided
β˜…β˜…β˜†β˜†2026β†’ Residue 80
NM_016042.4(EXOSC3):c.619_622dup (p.Arg208delinsAsnTer)Pathogenic
not provided|Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2026β†’ Residue 208
NM_016042.4(EXOSC3):c.395A>C (p.Asp132Ala)Pathogenic
Pontocerebellar hypoplasia type 1B|not provided|Inborn genetic diseases|Hypotonia|Abnormality of the nervous system|See cases|Congenital pontocerebellar hypoplasia type 1|EXOSC3-related disorder
β˜…β˜…β˜†β˜†2026β†’ Residue 132
NM_016042.4(EXOSC3):c.92G>C (p.Gly31Ala)Pathogenic
Pontocerebellar hypoplasia type 1B|7 conditions|not provided|Congenital myopathy
β˜…β˜…β˜†β˜†2026β†’ Residue 31
NM_016042.4(EXOSC3):c.226dup (p.Asp76fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2025β†’ Residue 76
NM_016042.4(EXOSC3):c.672_673del (p.Tyr225fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2025β†’ Residue 225
NM_016042.4(EXOSC3):c.428_431del (p.Tyr143fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2024β†’ Residue 143
NM_016042.4(EXOSC3):c.167_174del (p.Asn56fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2024β†’ Residue 56
NM_016042.4(EXOSC3):c.556C>T (p.Arg186Ter)Pathogenic
Pontoneocerebellar hypoplasia|Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2024β†’ Residue 186
NM_016042.4(EXOSC3):c.155del (p.Pro52fs)Pathogenic
not provided|Pontocerebellar hypoplasia type 1B|Pontoneocerebellar hypoplasia
β˜…β˜…β˜†β˜†2024β†’ Residue 52
NM_016042.4(EXOSC3):c.1A>G (p.Met1Val)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2024β†’ Residue 1
NM_016042.4(EXOSC3):c.294_303del (p.Val99fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2024β†’ Residue 99
NM_016042.4(EXOSC3):c.297_298insG (p.Tyr100fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2024β†’ Residue 100
NM_016042.4(EXOSC3):c.572G>A (p.Gly191Asp)Likely pathogenic
not specified|Pontocerebellar hypoplasia type 1B|not provided
β˜…β˜…β˜†β˜†2024β†’ Residue 191
NM_016042.4(EXOSC3):c.112del (p.Glu38fs)Pathogenic
Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2023β†’ Residue 38
NM_016042.4(EXOSC3):c.2T>C (p.Met1Thr)Pathogenic
not provided|Pontoneocerebellar hypoplasia|Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2023β†’ Residue 1
NM_016042.4(EXOSC3):c.312_313del (p.Gln105fs)Pathogenic
Pontoneocerebellar hypoplasia|Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2023β†’ Residue 105
NM_016042.4(EXOSC3):c.624_626+1delLikely pathogenic
Pontoneocerebellar hypoplasia|Pontocerebellar hypoplasia type 1B
β˜…β˜…β˜†β˜†2023
NM_016042.4(EXOSC3):c.660dup (p.Val221fs)Pathogenic
Pontoneocerebellar hypoplasia
β˜…β˜†β˜†β˜†2025β†’ Residue 221
NM_016042.4(EXOSC3):c.404G>A (p.Gly135Glu)Likely pathogenic
Pontoneocerebellar hypoplasia
β˜…β˜†β˜†β˜†2025β†’ Residue 135
View on ClinVar β†—
Related Genes
RBM7Protein interaction100%HBS1LProtein interaction100%MTREXProtein interaction100%EXOSC1Protein interaction100%HELZ2Protein interaction100%CDK5RAP1Protein interaction100%
Tissue Expression6 tissues
Brain
100%
Ovary
69%
Liver
59%
Lung
51%
Bone Marrow
44%
Heart
39%
Gene Interaction Network
Click a node to explore
EXOSC3RBM7HBS1LMTREXEXOSC1HELZ2CDK5RAP1
PROTEIN STRUCTURE
Preparing viewer…
PDB9G8M Β· 3.30 Γ… Β· EM
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.07LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.72 [0.50–1.07]
RankingsWhere EXOSC3 stands among ~20K protein-coding genes
  • #3,650of 20,598
    Most Researched128 Β· top quartile
  • #1,332of 5,498
    Most Pathogenic Variants50 Β· top quartile
  • #10,836of 17,882
    Most Constrained (LOEUF)1.07
Genes detectedEXOSC3
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
A 20-year Clinical and Genetic Neuromuscular Cohort Analysis in Lebanon: An International Effort.
PMID: 34602496
J Neuromuscul Dis Β· 2022
1.00
2
Atypical hemolytic uremic syndrome in the era of terminal complement inhibition: an observational cohort study.
PMID: 37369098
Blood Β· 2023
0.90
3
The RNA Exosome and Human Disease.
PMID: 31768969
Methods Mol Biol Β· 2020
0.80
4
Mutations in the RNA exosome component gene EXOSC3 cause pontocerebellar hypoplasia and spinal motor neuron degeneration.
PMID: 22544365
Nat Genet Β· 2012
0.70
5
A Chemical Biology Approach to Model Pontocerebellar Hypoplasia Type 1B (PCH1B).
PMID: 30141626
ACS Chem Biol Β· 2018
0.60