Generalized epilepsy with febrile seizures plus, type 9

disease
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Also known as GEFSP9generalised epilepsy with febrile seizures plus caused by mutation in STX1Bgeneralized epilepsy with febrile seizures plus caused by mutation in STX1BSTX1B generalised epilepsy with febrile seizures plusSTX1B generalized epilepsy with febrile seizures plus

Summary

Generalized epilepsy with febrile seizures plus, type 9 (MONDO:0014517) is a disease caused by STX1B (GenCC Strong), with 3 cohort genes.

At a glance

  • Causal gene: STX1B (GenCC Strong)
  • Cohort genes: 3
  • ClinVar variants: 370

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namegeneralized epilepsy with febrile seizures plus, type 9
Mondo IDMONDO:0014517
OMIM616172
DOIDDOID:0111301
UMLSC4015395
MedGen863832
GARD0018668
Is cancer (heuristic)no

Also known as: GEFSP9 · generalised epilepsy with febrile seizures plus caused by mutation in STX1B · generalised epilepsy with febrile seizures plus caused by mutation in STX1b · generalized epilepsy with febrile seizures plus caused by mutation in STX1B · generalized epilepsy with febrile seizures plus caused by mutation in STX1b · generalized epilepsy with febrile seizures plus, type 9 · STX1B generalised epilepsy with febrile seizures plus · STX1b generalised epilepsy with febrile seizures plus · STX1B generalized epilepsy with febrile seizures plus · STX1b generalized epilepsy with febrile seizures plus

Data availability: 370 ClinVar variants · 3 GenCC gene-disease records · 1 cell line.

Disease family

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary diseasehereditary neurological disease › hereditary generalized epilepsy › generalized epilepsy with febrile seizures plusgeneralized epilepsy with febrile seizures plus, type 9

Related subtypes (9): generalized epilepsy with febrile seizures plus, type 1, generalized epilepsy with febrile seizures plus, type 2, febrile seizures, familial, 8, generalized epilepsy with febrile seizures plus, type 4, generalized epilepsy with febrile seizures plus, type 6, generalized epilepsy with febrile seizures plus, type 8, generalized epilepsy with febrile seizures plus, type 7, generalized epilepsy with febrile seizures plus, type 10, generalized epilepsy with febrile seizures plus, type 12

Genetics & variants

GWAS landscape

No GWAS associations recorded — common-variant (GWAS) studies don’t cover this disease (typical for Mendelian / rare diseases). See the curated gene cohort and Mendelian overlap below.

Variant details and genetic-evidence tiers

ClinVar germline variants

370 retrieved; paginated sample, class counts are floors:

153 likely benign, 124 uncertain significance, 34 pathogenic, 27 likely pathogenic, 14 benign, 10 conflicting classifications of pathogenicity, 6 benign/likely benign, 2 pathogenic/likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
162396NM_052874.5(STX1B):c.[133_134insGGATGTGCATTG;135_136delinsGA]Pathogenicno assertion criteria provided
1458547NC_000016.9:g.(?30996980)(31021717_?)delHSD3B7Pathogeniccriteria provided, single submitter
475337NC_000016.10:g.(?30992801)(31068051_?)delLOC130058885Pathogeniccriteria provided, single submitter
2808202NM_052874.5(STX1B):c.340_354+123delLOC130058887Pathogeniccriteria provided, single submitter
3066028NC_000016.10:g.30996672_31010406delLOC130058887Pathogenicno assertion criteria provided
1066204NM_052874.5(STX1B):c.537+2T>CSTX1BPathogeniccriteria provided, single submitter
1070577NM_052874.5(STX1B):c.334del (p.Leu112fs)STX1BPathogeniccriteria provided, single submitter
1308652NM_052874.5(STX1B):c.160A>T (p.Lys54Ter)STX1BPathogeniccriteria provided, single submitter
1451994NM_052874.5(STX1B):c.205_205+1delSTX1BPathogeniccriteria provided, single submitter
1460098NM_052874.5(STX1B):c.214C>T (p.Gln72Ter)STX1BPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1481736NM_052874.5(STX1B):c.430T>C (p.Cys144Arg)STX1BPathogeniccriteria provided, single submitter
162395NM_052874.5(STX1B):c.166C>T (p.Gln56Ter)STX1BPathogenicno assertion criteria provided
162397NM_052874.5(STX1B):c.140C>A (p.Ser47Ter)STX1BPathogeniccriteria provided, single submitter
162398NM_052874.5(STX1B):c.647T>A (p.Val216Glu)STX1BPathogenicno assertion criteria provided
162399NM_052874.5(STX1B):c.676G>C (p.Gly226Arg)STX1BPathogenicno assertion criteria provided
2576582NM_052874.5(STX1B):c.354+1G>CSTX1BPathogeniccriteria provided, single submitter
2693031NM_052874.5(STX1B):c.786+1G>ASTX1BPathogeniccriteria provided, single submitter
2748620NM_052874.5(STX1B):c.815del (p.Val272fs)STX1BPathogeniccriteria provided, single submitter
2811860NM_052874.5(STX1B):c.786+1G>TSTX1BPathogeniccriteria provided, single submitter
2813188NM_052874.5(STX1B):c.39del (p.Asp13fs)STX1BPathogeniccriteria provided, single submitter
2849088NM_052874.5(STX1B):c.338_354+15delSTX1BPathogeniccriteria provided, single submitter
2864828NM_052874.5(STX1B):c.366del (p.Ser123fs)STX1BPathogeniccriteria provided, single submitter
3650187NM_052874.5(STX1B):c.565C>T (p.Gln189Ter)STX1BPathogeniccriteria provided, single submitter
3663941NM_052874.5(STX1B):c.2T>A (p.Met1Lys)STX1BPathogeniccriteria provided, single submitter
4726056NM_052874.5(STX1B):c.725dup (p.Tyr242Ter)STX1BPathogeniccriteria provided, single submitter
4732781NM_052874.5(STX1B):c.846dup (p.Gly283fs)STX1BPathogeniccriteria provided, single submitter
4735231NM_052874.5(STX1B):c.613G>T (p.Glu205Ter)STX1BPathogeniccriteria provided, single submitter
567167NM_052874.5(STX1B):c.35_36dup (p.Asp13fs)STX1BPathogeniccriteria provided, single submitter
590057NM_052874.5(STX1B):c.733C>T (p.Arg245Ter)STX1BPathogeniccriteria provided, multiple submitters, no conflicts
620559NM_052874.5(STX1B):c.394G>T (p.Glu132Ter)STX1BPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 4 · Orphanet: 2 · OMIM-shared: 0 · Dual-evidence (GWAS+Mendelian): 0

GenCC gene–disease validity (cohort genes)

the Disease column is the GenCC-asserted condition — a cohort gene’s strongest validity may be for a related predisposition syndrome.

GeneClassificationInheritanceDiseaseRecords
STX1BStrongAutosomal dominantgeneralized epilepsy with febrile seizures plus, type 94

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
STX1BOrphanet:36387Genetic epilepsy with febrile seizure plus
HSD3B7Orphanet:79301Congenital bile acid synthesis defect type 1

Cohort genes → proteins

3 cohort genes, 3 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence3

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
STX1BHGNC:18539ENSG00000099365P61266Syntaxin-1Bgencc,clinvar
HSD3B7HGNC:18324ENSG00000099377Q9H2F33 beta-hydroxysteroid dehydrogenase type 7clinvar
CFAP119HGNC:28078ENSG00000196118A1A4V9Cilia- and flagella-associated protein 119clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
STX1BSyntaxin-1BPotentially involved in docking of synaptic vesicles at presynaptic active zones.
HSD3B73 beta-hydroxysteroid dehydrogenase type 7The 3-beta-HSD enzymatic system plays a crucial role in the biosynthesis of all classes of hormonal steroids.

Protein-family classification

Druggable: 0 · Difficult: 0 · Unknown: 3 · Druggable fraction: 0.0

Family distribution

Cohort families vs a genome-wide background (hypergeometric, BH-FDR; fold = observed/expected). Counts kept; sorted by enrichment, so the catch-all Other/Unknown bucket no longer leads.

FamilyGenesFoldFDR
Other/Unknown31.8×0.174

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
STX1BOther/UnknownnoT_SNARE_dom, Syntaxin_N, Syntaxin/epimorphin_CS
HSD3B7Other/Unknownno3Beta_OHSteriod_DH/Estase, NAD(P)-bd_dom_sf, NAD(P)_dehydrat-like
CFAP119Other/UnknownnoCLAMP

Expression context

Cohort genes with no expression data: 0.

3 cohort genes are a single-cell marker in ≥1 SCXA experiment.

Breadth distribution (Bgee present_calls)

BucketGenes
narrow (1-5 tissues)0
moderate (6-20)0
broad (>20)3
unknown0

Top tissues across cohort

TissueCohort genes
cerebellar cortex1
cerebellar hemisphere1
right hemisphere of cerebellum1
left adrenal gland1
liver1
right lobe of liver1
bronchial epithelial cell1
bronchus1
right uterine tube1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
STX1B176ubiquitousmarkerright hemisphere of cerebellum, cerebellar hemisphere, cerebellar cortex
HSD3B7177ubiquitousmarkerright lobe of liver, liver, left adrenal gland
CFAP119184ubiquitousmarkerright uterine tube, bronchial epithelial cell, bronchus

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
HSD3B73,327
STX1B2,130
CFAP119884

Structural data

PDB: 0 · AlphaFold-only: 3 · No structure: 0

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
HSD3B7Q9H2F394.01
STX1BP6126684.17
CFAP119A1A4V974.17

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 16. Enrichment computed across 3 evidence-associated genes (2 with Reactome annotation).

Pathways by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 2 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Toxicity of botulinum toxin type C (botC)11903.3×0.007STX1B
Neurotoxicity of clostridium toxins1713.8×0.007STX1B
Synthesis of bile acids and bile salts via 24-hydroxycholesterol1439.2×0.007HSD3B7
Uptake and actions of bacterial toxins1407.9×0.007STX1B
LGI-ADAM interactions1407.9×0.007STX1B
Synthesis of bile acids and bile salts via 27-hydroxycholesterol1380.7×0.007HSD3B7
Bile acid and bile salt metabolism1248.3×0.009HSD3B7
Synthesis of bile acids and bile salts via 7alpha-hydroxycholesterol1228.4×0.009HSD3B7
Synthesis of bile acids and bile salts1203.9×0.009HSD3B7
Bacterial Infection Pathways1167.9×0.010STX1B
Metabolism of steroids168.8×0.021HSD3B7
Metabolism of lipids115.8×0.083HSD3B7
Infectious disease112.4×0.097STX1B
Developmental Biology17.2×0.153STX1B
Disease16.5×0.157STX1B
Metabolism15.8×0.165HSD3B7

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 3 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
negative regulation of synaptic vesicle recycling15617.3×0.001STX1B
positive regulation of spontaneous neurotransmitter secretion15617.3×0.001STX1B
negative regulation of macropinocytosis15617.3×0.001STX1B
calcium ion-regulated exocytosis of neurotransmitter11404.3×0.003STX1B
regulation of synaptic activity11404.3×0.003STX1B
B cell chemotaxis1936.2×0.004HSD3B7
spontaneous neurotransmitter secretion1936.2×0.004STX1B
positive regulation of neurotransmitter secretion1624.1×0.004STX1B
regulation of synaptic vesicle priming1561.7×0.004STX1B
synaptic vesicle fusion to presynaptic active zone membrane1561.7×0.004STX1B
obsolete synaptic vesicle docking1432.1×0.005STX1B
obsolete vesicle docking1255.3×0.007STX1B
obsolete vesicle docking involved in exocytosis1224.7×0.007STX1B
sperm flagellum assembly1224.7×0.007CFAP119
bile acid biosynthetic process1208.1×0.007HSD3B7
steroid biosynthetic process1200.6×0.007HSD3B7
positive regulation of excitatory postsynaptic potential1175.5×0.008STX1B
negative regulation of neuron projection development179.1×0.016STX1B
single fertilization161.1×0.020CFAP119
exocytosis150.6×0.023STX1B
regulation of gene expression127.8×0.039STX1B
gene expression126.6×0.039CFAP119
intracellular protein transport121.6×0.046STX1B

Therapeutics

Drug target analysis

Approved (phase 4): 0 · Phase ≥3: 0 · Phased (≥1): 0 · Undrugged: 3

Druggability breadth: 0 of 3 evidence-associated genes (0%) have a ChEMBL target (buckets above are over the deeply-mined display cohort).

Top cohort targets by molecule count

SymbolMoleculesMax phase
STX1B00
HSD3B700
CFAP11900

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

Pharmacogenomics

Cohort genes with a PharmGKB record: 3; with CPIC/DPWG dosing guidelines: 0.

No cohort gene has a CPIC/DPWG genotype-guided dosing guideline (PharmGKB).

Chemical tractability of cohort targets

0 approved/phased compounds have measured bioactivity against a cohort gene (and aren’t yet in disease-level trials). This is a research / tractability signal, NOT a therapeutic recommendation — a bioactivity row often reflects off-target or screening binding (e.g. promiscuous kinase inhibitors against a cohort kinase), implying no disease mechanism.

Druggability pyramid

Cohort genes binned by druggability tier (high → low):

TierDefinitionGenesSymbols
AApproved (phase 4 drug)0
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug0
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug3STX1B, HSD3B7, CFAP119

Undrugged target profiles

3 cohort genes are undrugged. Ranked by ‘starting-point quality’ (assay depth + drugged-partner adjacency).

SymbolChEMBL assaysDrugged partners (top 3)
STX1B0
HSD3B70
CFAP1190

Clinical trials & evidence

Clinical trials

Clinical trials: 0.