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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.
SBF2
SET binding factor 2
Chromosome 11 Β· 11p15.4
NCBI Gene: 81846Ensembl: ENSG00000133812.18HGNC: HGNC:2135UniProt: A0A6Q8PH87
51PubMed Papers
21Diseases
0Drugs
88Pathogenic Variants
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
guanyl-nucleotide exchange factor activityprotein bindingmembranecytoplasmCharcot-Marie-Tooth disease type 4B2Charcot-Marie-Tooth disease type 4genetic disorderhypertension
✦AI Summary

SBF2 encodes a guanine nucleotide exchange factor (GEF) that activates RAB21 and possibly RAB28 by promoting GDP-to-GTP exchange, converting inactive Rab proteins to their active forms 12. During starvation-induced autophagy, SBF2 activates RAB21, which regulates the SNARE protein VAMP8 to facilitate autophagosome-lysosome fusion 2. SBF2 also acts as an adapter for the phosphatase MTMR2, enhancing its catalytic activity toward phosphatidylinositol lipids. Biallelic SBF2 mutations cause Charcot-Marie-Tooth disease type 4B2 (CMT4B2), an autosomal recessive sensorimotor neuropathy characterized by demyelination and excess myelin outfoldings 3. Disease-associated mutations lead to shortened or truncated proteins, suggesting loss-of-function mechanisms 4. CMT4B2 typically manifests in the first decade with predominantly motor symptoms and moderate disease progression, with neuropathy >90% penetrant by age 10 years 3. Glaucoma, previously considered a hallmark feature, is absent in ~40% of CMT4B2 cases but may develop with age, warranting lifelong intraocular pressure monitoring 3. SBF2 shows broad tissue expression with cytoplasmic localization 4.

Sources cited
1
SBF2 is a GEF that activates RAB21 and possibly RAB28 through GDP-to-GTP exchange
PMID: 20937701
2
SBF2 activates RAB21 during autophagy to regulate VAMP8-mediated autophagosome-lysosome fusion
PMID: 25648148
3
Biallelic SBF2 mutations cause CMT4B2 with demyelination, excess myelin outfoldings, >90% neuropathy penetrance by age 10, and variable glaucoma penetrance
PMID: 30028002
4
SBF2 mutations cause loss-of-function; SBF2 shows broad tissue expression and cytoplasmic localization
PMID: 16750429
⚠Limited data available β€” This gene has 4 indexed publications. Summary and analysis may be incomplete.
Disease Associationsβ“˜21
Charcot-Marie-Tooth disease type 4B2Open Targets
0.77Strong
Charcot-Marie-Tooth disease type 4Open Targets
0.56Moderate
genetic disorderOpen Targets
0.50Moderate
hypertensionOpen Targets
0.48Moderate
Abnormality of the skeletal systemOpen Targets
0.46Moderate
Charcot-Marie-Tooth diseaseOpen Targets
0.46Moderate
congestive heart failureOpen Targets
0.45Moderate
Varicose veinsOpen Targets
0.41Moderate
essential hypertensionOpen Targets
0.41Moderate
Tip-toe gaitOpen Targets
0.40Moderate
colorectal cancerOpen Targets
0.38Weak
type 2 diabetes mellitusOpen Targets
0.33Weak
Increased blood pressureOpen Targets
0.33Weak
cardiovascular diseaseOpen Targets
0.33Weak
major salivary gland cancerOpen Targets
0.31Weak
albuminuriaOpen Targets
0.30Weak
atrial fibrillationOpen Targets
0.29Weak
vascular diseaseOpen Targets
0.29Weak
pelvic organ prolapseOpen Targets
0.27Weak
diabetes mellitusOpen Targets
0.27Weak
Charcot-Marie-Tooth disease, demyelinating, type 4B2UniProt
Pathogenic Variants88
NM_030962.4(SBF2):c.1024C>T (p.Arg342Ter)Pathogenic
Charcot-Marie-Tooth disease type 4B2|Charcot-Marie-Tooth disease type 4
β˜…β˜…β˜†β˜†2026β†’ Residue 342
NM_030962.4(SBF2):c.3653-1G>ALikely pathogenic
Charcot-Marie-Tooth disease type 4|Charcot-Marie-Tooth disease type 4B2
β˜…β˜…β˜†β˜†2025
NM_030962.4(SBF2):c.229C>T (p.Arg77Ter)Pathogenic
Inborn genetic diseases|Charcot-Marie-Tooth disease type 4
β˜…β˜…β˜†β˜†2025β†’ Residue 77
NM_030962.4(SBF2):c.1066C>T (p.Arg356Ter)Pathogenic
Charcot-Marie-Tooth disease|Charcot-Marie-Tooth disease type 4|Charcot-Marie-Tooth disease type 4B2
β˜…β˜…β˜†β˜†2025β†’ Residue 356
NM_030962.4(SBF2):c.1601-2A>GPathogenic
not provided|Charcot-Marie-Tooth disease type 4B2
β˜…β˜…β˜†β˜†2024
NM_030962.4(SBF2):c.2055T>G (p.Tyr685Ter)Pathogenic
Charcot-Marie-Tooth disease type 4B2|Charcot-Marie-Tooth disease type 4
β˜…β˜…β˜†β˜†2024β†’ Residue 685
NM_030962.4(SBF2):c.5254_5255insTTAT (p.Lys1752fs)Pathogenic
not provided|Charcot-Marie-Tooth disease type 4|SBF2-related disorder
β˜…β˜…β˜†β˜†2023β†’ Residue 1752
NM_030962.4(SBF2):c.1459C>T (p.Arg487Ter)Pathogenic
Charcot-Marie-Tooth disease|Charcot-Marie-Tooth disease type 4|not provided|Charcot-Marie-Tooth disease type 4B2
β˜…β˜…β˜†β˜†2023β†’ Residue 487
NM_030962.4(SBF2):c.619+1G>APathogenic
not provided
β˜…β˜…β˜†β˜†2023
NM_030962.4(SBF2):c.1907del (p.Pro636fs)Pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2025β†’ Residue 636
NM_030962.4(SBF2):c.2581G>T (p.Glu861Ter)Pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2025β†’ Residue 861
NM_030962.4(SBF2):c.3800G>A (p.Trp1267Ter)Pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2025β†’ Residue 1267
NM_030962.4(SBF2):c.5232-1G>ALikely pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2025
NM_030962.4(SBF2):c.659_660del (p.Thr220fs)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 220
NM_030962.4(SBF2):c.115dup (p.Thr39fs)Pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2025β†’ Residue 39
NM_030962.4(SBF2):c.1792C>T (p.Gln598Ter)Pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2025β†’ Residue 598
NM_030962.4(SBF2):c.5037+1G>APathogenic
Charcot-Marie-Tooth disease type 4|Ovarian serous cystadenocarcinoma
β˜…β˜†β˜†β˜†2025
NM_030962.4(SBF2):c.4444-1G>CLikely pathogenic
Charcot-Marie-Tooth disease type 4B2
β˜…β˜†β˜†β˜†2024
NM_030962.4(SBF2):c.1297-2A>GLikely pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2024
NM_030962.4(SBF2):c.4009G>T (p.Glu1337Ter)Pathogenic
Charcot-Marie-Tooth disease type 4
β˜…β˜†β˜†β˜†2024β†’ Residue 1337
View on ClinVar β†—
Related Genes
ZNF24Shared pathway100%BCAS1Shared pathway100%CMTM8Shared pathway100%MAL2Shared pathway100%MARVELD1Shared pathway100%RAB21Protein interaction82%
Tissue Expression6 tissues
Ovary
100%
Heart
83%
Brain
76%
Lung
72%
Liver
51%
Bone Marrow
32%
Gene Interaction Network
Click a node to explore
SBF2ZNF24BCAS1CMTM8MAL2MARVELD1RAB21
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt Q86WG5
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.59Moderately Constrained
pLIβ“˜
0.00Tolerant
Observed/Expected LoF0.49 [0.41–0.59]
RankingsWhere SBF2 stands among ~20K protein-coding genes
  • #8,727of 20,598
    Most Researched51
  • #860of 5,498
    Most Pathogenic Variants88 Β· top quartile
  • #4,011of 17,882
    Most Constrained (LOEUF)0.59 Β· top quartile
Genes detectedSBF2
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
LncRNA SBF2-AS1: A Budding Star in Various Cancers.
PMID: 35440300
Curr Pharm Des Β· 2022
0.90
3
Emerging roles of a pivotal lncRNA SBF2-AS1 in cancers.
PMID: 34372871
Cancer Cell Int Β· 2021
0.80
4
Novel SBF2 mutations and clinical spectrum of Charcot-Marie-Tooth neuropathy type 4B2.
PMID: 30028002
Clin Genet Β· 2018
0.70
5
A Subpathway and Target Gene Cluster-Based Approach Uncovers lncRNAs Associated with Human Primordial Follicle Activation.
PMID: 37445702
Int J Mol Sci Β· 2023
0.60