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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.
BSN
bassoon presynaptic cytomatrix protein
Chromosome 3 Β· 3p21.31
NCBI Gene: 8927Ensembl: ENSG00000164061.5HGNC: HGNC:1117UniProt: Q9UPA5
54PubMed Papers
20Diseases
0Drugs
4Pathogenic Variants
FUNCTIONAL ROLE
Highly Constrained
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
enzyme inhibitor activitystructural constituent of presynaptic active zoneprotein localization to synapsemodulation of chemical synaptic transmissionepilepsyneurodegenerative diseaseParkinson diseasevascular parkinsonism
✦AI Summary

BSN encodes Bassoon, a 420-kDa scaffold protein that organizes the presynaptic active zone, the site of neurotransmitter release 1. Bassoon participates in forming Piccolo-Bassoon transport vesicles that deliver synaptic proteins to developing synapses and regulates spatial organization of synaptic vesicles and membrane fusion machinery 1. Beyond structural functions, BSN regulates presynaptic protein ubiquitination, autophagy, and modulates neurotransmission by inhibiting presynaptic DAO enzyme activity. BSN variants cause a distinct neurodevelopmental disorder characterized by epilepsy (45% of cases), developmental delay (38%), and behavioral abnormalities including ADHD (25%) and autism (17%), with phenotypic severity varying across the age spectrum 2. Notably, protein-truncating BSN variants are associated with severe adult-onset obesity and type 2 diabetes, representing effects larger than established obesity genes, suggesting degenerative synaptic processes contribute to metabolic dysfunction 3. Recent mechanistic studies demonstrate that BSN mutations impair astrocyte lipid homeostasis and Kir4.1 potassium channel function, leading to reduced neurosupportive capacity and neuronal hyperexcitability 4. A mouse model carrying BSN mutations exhibits impaired working memory and dopaminergic neuronal loss in the substantia nigra 5. These findings establish BSN as critical for synaptic organization and reveal how presynaptic dysfunction extends to metabolic and glial-neuronal interactions.

Sources cited
1
BSN encodes a 420-kDa presynaptic cytomatrix protein and is localized to human chromosome 3p21
PMID: 10329005
2
BSN variants cause neurodevelopmental disorder with epilepsy (45%), developmental delay (38%), ADHD (25%), and autism (17%)
PMID: 40393460
3
BSN protein-truncating variants are associated with severe adult-onset obesity and type 2 diabetes with larger effects than established obesity genes
PMID: 38575728
4
BSN mutations impair astrocyte lipid metabolism and Kir4.1 function, leading to neuronal hyperexcitability
PMID: 40962686
5
Mouse model with BSN p.P3882A mutation shows impaired working memory and dopaminergic neuronal loss in substantia nigra
PMID: 39130376
Disease Associationsβ“˜20
epilepsyOpen Targets
0.32Weak
neurodegenerative diseaseOpen Targets
0.21Weak
Parkinson diseaseOpen Targets
0.13Weak
parkinsonian disorderOpen Targets
0.12Weak
vascular parkinsonismOpen Targets
0.12Weak
SeizureOpen Targets
0.11Weak
Mobius syndromeOpen Targets
0.11Weak
Moebius syndromeOpen Targets
0.11Weak
deafnessOpen Targets
0.10Suggestive
inflammatory bowel diseaseOpen Targets
0.10Suggestive
hearing loss, autosomal recessiveOpen Targets
0.10Suggestive
autosomal dominant nonsyndromic hearing lossOpen Targets
0.09Suggestive
ulcerative colitisOpen Targets
0.08Suggestive
colorectal carcinomaOpen Targets
0.07Suggestive
carpal tunnel syndromeOpen Targets
0.07Suggestive
chronic obstructive pulmonary diseaseOpen Targets
0.07Suggestive
auditory neuropathy, autosomal dominant 3Open Targets
0.06Suggestive
autosomal recessive nonsyndromic hearing loss 1BOpen Targets
0.06Suggestive
autosomal dominant nonsyndromic hearing loss 65Open Targets
0.06Suggestive
benign adult familial myoclonic epilepsyOpen Targets
0.06Suggestive
Pathogenic Variants4
NM_003458.4(BSN):c.3335del (p.Ala1112fs)Pathogenic
BSN-associated seizure disorder|BSN-associated neurodevelopmental disorder
β˜…β˜†β˜†β˜†2025β†’ Residue 1112
NM_003458.4(BSN):c.5886del (p.Pro1963fs)Pathogenic
BSN-associated seizure disorder
β˜…β˜†β˜†β˜†2025β†’ Residue 1963
NM_003458.4(BSN):c.2623C>T (p.Gln875Ter)Pathogenic
BSN-associated epilepsy
β˜…β˜†β˜†β˜†2025β†’ Residue 875
NM_003458.4(BSN):c.10255C>T (p.Gln3419Ter)Likely pathogenic
Epilepsy
β˜…β˜†β˜†β˜†2023β†’ Residue 3419
View on ClinVar β†—
Related Genes
VAMP2Protein interaction98%SNAP25Protein interaction93%CTBP2Protein interaction93%SNPHProtein interaction83%STX1AProtein interaction82%SYBUProtein interaction79%
Tissue Expression6 tissues
Brain
100%
Liver
9%
Ovary
5%
Heart
4%
Bone Marrow
3%
Lung
1%
Gene Interaction Network
Click a node to explore
BSNVAMP2SNAP25CTBP2SNPHSTX1ASYBU
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q9UPA5
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.27Highly Constrained
pLIβ“˜
1.00Intolerant
Observed/Expected LoF0.22 [0.17–0.27]
RankingsWhere BSN stands among ~20K protein-coding genes
  • #8,284of 20,598
    Most Researched54
  • #3,785of 5,498
    Most Pathogenic Variants4
  • #917of 17,882
    Most Constrained (LOEUF)0.27 Β· top 10%
Genes detectedBSN
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Tissue morphology influences the temporal program of human brain organoid development.
PMID: 37802039
Cell Stem Cell Β· 2023
1.00
2
Variants in BSN, encoding the presynaptic protein Bassoon, result in a distinct neurodevelopmental disorder with a broad phenotypic range.
PMID: 40393460
Am J Hum Genet Β· 2025
0.90
3
Protein-truncating variants in BSN are associated with severe adult-onset obesity, type 2 diabetes and fatty liver disease.
PMID: 38575728
Nat Genet Β· 2024
0.80
4
What Makes a BSN a BSN?
PMID: 30261819
West J Nurs Res Β· 2019
0.70
5
Behavioral and histological analyses of the mouse
PMID: 39130376
Front Neurosci Β· 2024
0.60