Developmental and epileptic encephalopathy, 30

disease
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Also known as DEE30developmental and epileptic encephalopathy 30early infantile epileptic encephalopathy caused by mutation in SIK1EIEE30epileptic encephalopathy, early infantile, 30epileptic encephalopathy, early infantile, type 30SIK1 early infantile epileptic encephalopathy

Summary

Developmental and epileptic encephalopathy, 30 (MONDO:0014595) is a disease caused by SIK1 (GenCC Definitive), with 7 cohort genes.

At a glance

  • Causal gene: SIK1 (GenCC Definitive)
  • Cohort genes: 7
  • ClinVar variants: 1,034

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namedevelopmental and epileptic encephalopathy, 30
Mondo IDMONDO:0014595
OMIM616341
DOIDDOID:0080465
UMLSC4225360
MedGen898954
GARD0016093
Is cancer (heuristic)no

Also known as: DEE30 · developmental and epileptic encephalopathy 30 · early infantile epileptic encephalopathy caused by mutation in SIK1 · EIEE30 · epileptic encephalopathy, early infantile, 30 · epileptic encephalopathy, early infantile, type 30 · SIK1 early infantile epileptic encephalopathy

Data availability: 1,034 ClinVar variants · 2 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by body system or component › syndromic diseaseinfantile spasmsdevelopmental and epileptic encephalopathy, 30

Related subtypes (7): developmental and epileptic encephalopathy, 2, developmental and epileptic encephalopathy, 1, developmental and epileptic encephalopathy, 5, developmental and epileptic encephalopathy, 12, developmental and epileptic encephalopathy, 15, developmental and epileptic encephalopathy, 27, developmental and epileptic encephalopathy, 40

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

600 retrieved; paginated sample, class counts are floors:

281 likely benign, 269 uncertain significance, 22 benign, 21 conflicting classifications of pathogenicity, 4 pathogenic, 1 benign/likely benign, 1 likely pathogenic, 1 pathogenic/likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
206981NM_001040142.2(SCN2A):c.2995G>A (p.Glu999Lys)SCN2APathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
190105NM_173354.5(SIK1):c.859C>A (p.Pro287Thr)SIK1Pathogenicno assertion criteria provided
190106NM_173354.5(SIK1):c.1039G>T (p.Glu347Ter)SIK1Pathogenicno assertion criteria provided
190107NM_173354.5(SIK1):c.1840C>T (p.Gln614Ter)SIK1Pathogenicno assertion criteria provided
190109NM_173354.5(SIK1):c.1906G>A (p.Gly636Ser)SIK1Pathogenicno assertion criteria provided
1703110NM_173354.5(SIK1):c.1839C>A (p.Cys613Ter)SIK1Likely pathogeniccriteria provided, single submitter
2422361NC_000021.8:g.(?43160998)(47754702_?)delABCG1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1025710NM_173354.5(SIK1):c.1691T>C (p.Leu564Pro)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1030319NM_173354.5(SIK1):c.468G>A (p.Leu156=)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1036622NM_173354.5(SIK1):c.1783C>T (p.Arg595Trp)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1041574NM_173354.5(SIK1):c.938A>G (p.Gln313Arg)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1044080NM_173354.5(SIK1):c.1634C>T (p.Ser545Leu)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1063659NM_173354.5(SIK1):c.2201T>C (p.Ile734Thr)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1137642NM_173354.5(SIK1):c.1463-5C>ASIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1362958NM_173354.5(SIK1):c.835C>G (p.Arg279Gly)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1372665NM_173354.5(SIK1):c.1784G>A (p.Arg595Gln)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1393532NM_173354.5(SIK1):c.1235C>T (p.Ser412Leu)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1408200NM_173354.5(SIK1):c.1642G>A (p.Ala548Thr)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1462942NM_173354.5(SIK1):c.1279G>A (p.Gly427Arg)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1475064NM_173354.5(SIK1):c.691A>G (p.Thr231Ala)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1627773NM_173354.5(SIK1):c.1055A>G (p.Gln352Arg)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1763111NM_173354.5(SIK1):c.836G>A (p.Arg279Gln)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
190108NM_173354.5(SIK1):c.1897C>T (p.Gln633Ter)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1912697NM_173354.5(SIK1):c.1793G>C (p.Gly598Ala)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
2181702NM_173354.5(SIK1):c.692C>T (p.Thr231Met)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
2199574NM_173354.5(SIK1):c.2152G>A (p.Ala718Thr)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
2898107NM_173354.5(SIK1):c.1066C>T (p.Pro356Ser)SIK1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1409580NC_000021.8:g.(?43160998)(47865240_?)dupKRTAP12-2Uncertain significancecriteria provided, single submitter
1055976NC_000021.8:g.(?44838130)(45196150_?)dupRRP1BUncertain significancecriteria provided, single submitter
1000093NM_173354.5(SIK1):c.1988T>C (p.Leu663Ser)SIK1Uncertain significancecriteria provided, single submitter

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 7 · Orphanet: 10 · 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
SIK1DefinitiveAutosomal dominantdevelopmental and epileptic encephalopathy, 307

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
SIK1Orphanet:1934Early infantile developmental and epileptic encephalopathy
SIK1Orphanet:697160Infantile epileptic spasms syndrome
SCN2AOrphanet:140927Self-limited neonatal-infantile epilepsy
SCN2AOrphanet:1934Early infantile developmental and epileptic encephalopathy
SCN2AOrphanet:2131Alternating hemiplegia of childhood
SCN2AOrphanet:293181Epilepsy of infancy with migrating focal seizures
SCN2AOrphanet:306Self-limited infantile epilepsy
SCN2AOrphanet:33069Dravet syndrome
SCN2AOrphanet:36387Genetic epilepsy with febrile seizure plus
SCN2AOrphanet:697160Infantile epileptic spasms syndrome

Cohort genes → proteins

7 cohort genes, 7 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence7

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
SIK1HGNC:11142ENSG00000142178P57059Serine/threonine-protein kinase SIK1gencc,clinvar
SCN2AHGNC:10588ENSG00000136531Q99250Sodium channel protein type 2 subunit alphaclinvar
KRTAP12-2HGNC:20530ENSG00000221864P59991Keratin-associated protein 12-2clinvar
RRP1BHGNC:23818ENSG00000160208Q14684Ribosomal RNA processing protein 1 homolog Bclinvar
AGPAT3HGNC:326ENSG00000160216Q9NRZ71-acyl-sn-glycerol-3-phosphate acyltransferase gammaclinvar
ABCG1HGNC:73ENSG00000160179P45844ATP-binding cassette sub-family G member 1clinvar
PDXKHGNC:8819ENSG00000160209O00764Pyridoxal kinaseclinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
SIK1Serine/threonine-protein kinase SIK1Serine/threonine-protein kinase involved in various processes such as cell cycle regulation, gluconeogenesis and lipogenesis regulation, muscle growth and differentiation and tumor suppression.
SCN2ASodium channel protein type 2 subunit alphaMediates the voltage-dependent sodium ion permeability of excitable membranes.
KRTAP12-2Keratin-associated protein 12-2In the hair cortex, hair keratin intermediate filaments are embedded in an interfilamentous matrix, consisting of hair keratin-associated proteins (KRTAP), which are essential for the formation of a rigid and resistant hair shaft through t…
RRP1BRibosomal RNA processing protein 1 homolog BPositively regulates DNA damage-induced apoptosis by acting as a transcriptional coactivator of proapoptotic target genes of the transcriptional activator E2F1.
AGPAT31-acyl-sn-glycerol-3-phosphate acyltransferase gammaConverts 1-acyl-sn-glycerol-3-phosphate (lysophosphatidic acid or LPA) into 1,2-diacyl-sn-glycerol-3-phosphate (phosphatidic acid or PA) by incorporating an acyl moiety at the sn-2 position of the glycerol backbone.
ABCG1ATP-binding cassette sub-family G member 1Catalyzes the efflux of phospholipids such as sphingomyelin, cholesterol and its oxygenated derivatives like 7beta-hydroxycholesterol and this transport is coupled to hydrolysis of ATP.
PDXKPyridoxal kinaseCatalyzes the phosphorylation of the dietary vitamin B6 vitamers pyridoxal (PL), pyridoxine (PN) and pyridoxamine (PM) to form pyridoxal 5’-phosphate (PLP), pyridoxine 5’-phosphate (PNP) and pyridoxamine 5’-phosphate (PMP), respectively.

Protein-family classification

Druggable: 5 · Difficult: 0 · Unknown: 2 · Druggable fraction: 0.71

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
Kinase27.9×0.121
Ion channel115.9×0.144
Transporter111.1×0.144
Enzyme (other)11.7×0.571
Other/Unknown20.5×0.968

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
SIK1KinaseyesProt_kinase_dom, Ser/Thr_kinase_AS, Kinase-like_dom_sf
SCN2AIon channelyesIQ_motif_EF-hand-BS, Na_channel_asu, Ion_trans_dom
KRTAP12-2Other/UnknownnoKRTAP_PMG
RRP1BOther/UnknownnoRRP1
AGPAT3Enzyme (other)yes2.3.1.51Plipid/glycerol_acylTrfase, Acyltransf_C
ABCG1TransporteryesABC_transporter-like_ATP-bd, AAA+_ATPase, Pigment_permease/Abcg
PDXKKinaseyes2.7.1.35PyrdxlKinase, PM/HMP-P_kinase-1, Ribokinase-like

Expression context

Cohort genes with no expression data: 0.

6 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)1
broad (>20)6
unknown0

Top tissues across cohort

TissueCohort genes
Brodmann (1909) area 232
middle temporal gyrus2
mucosa of stomach1
skin of abdomen1
zone of skin1
cerebellar vermis1
colonic epithelium1
male germ line stem cell (sensu Vertebrata) in testis1
sural nerve1
germinal epithelium of ovary1
gingiva1
gingival epithelium1
lateral globus pallidus1
left adrenal gland1
right adrenal gland1
right adrenal gland cortex1
adult organism1
left testis1
right testis1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
SIK1138not_expressedmarkermucosa of stomach, skin of abdomen, zone of skin
SCN2A187broadmarkermiddle temporal gyrus, Brodmann (1909) area 23, cerebellar vermis
KRTAP12-216yesmale germ line stem cell (sensu Vertebrata) in testis, sural nerve, colonic epithelium
RRP1B286ubiquitousmarkergerminal epithelium of ovary, gingival epithelium, gingiva
AGPAT3274ubiquitousmarkerlateral globus pallidus, middle temporal gyrus, Brodmann (1909) area 23
ABCG1270ubiquitousmarkerright adrenal gland, left adrenal gland, right adrenal gland cortex
PDXK295ubiquitousmarkerleft testis, right testis, adult organism

Protein interactions among cohort

Intra-cohort edges: 1.

Hub genes (top 10 by interactor count)

SymbolInteractor count
RRP1B3,651
SCN2A2,810
ABCG12,178
AGPAT32,081
PDXK1,899
SIK11,840
KRTAP12-2773

Intra-cohort edges

ABSources
PDXKRRP1Bstring_interaction

Structural data

PDB: 4 · AlphaFold-only: 3 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
PDXKO0076410
SCN2AQ992505
ABCG1P458445
RRP1BQ146842

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
AGPAT3Q9NRZ794.63
KRTAP12-2P5999170.96
SIK1P5705961.31

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 28. Enrichment computed across 7 evidence-associated genes (6 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 6 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Vitamin B6 activation to pyridoxal phosphate1634.4×0.044PDXK
HDL remodeling1190.3×0.046ABCG1
ABC transporters in lipid homeostasis1100.2×0.046ABCG1
Plasma lipoprotein remodeling179.3×0.046ABCG1
Phosphorylated BMAL1:CLOCK (ARNTL:CLOCK) activates expression of core clock genes179.3×0.046SIK1
NR1H2 and NR1H3-mediated signaling165.6×0.046ABCG1
Interaction between L1 and Ankyrins161.4×0.046SCN2A
R-HSA-400253157.7×0.046SIK1
Phase 0 - rapid depolarisation157.7×0.046SCN2A
Sensory perception of taste156.0×0.046SCN2A
NR1H3 & NR1H2 regulate gene expression linked to cholesterol transport and efflux151.4×0.046ABCG1
Synthesis of PA148.8×0.046AGPAT3
Sensory perception of sweet, bitter, and umami (glutamate) taste146.4×0.046SCN2A
Plasma lipoprotein assembly, remodeling, and clearance138.1×0.052ABCG1
COPI-independent Golgi-to-ER retrograde traffic134.6×0.053AGPAT3
ABC-family protein mediated transport120.2×0.081ABCG1
L1CAM interactions120.0×0.081SCN2A
Cardiac conduction118.1×0.082SCN2A
Signaling by Nuclear Receptors117.0×0.082ABCG1
Sensory Perception115.9×0.082SCN2A
Developmental Biology24.8×0.082SCN2A, KRTAP12-2
Muscle contraction112.9×0.096SCN2A
Keratinization19.3×0.125KRTAP12-2
Axon guidance17.5×0.147SCN2A
Nervous system development17.2×0.148SCN2A
Transport of small molecules14.2×0.233ABCG1
Neutrophil degranulation13.9×0.242PDXK
Signal Transduction11.7×0.463ABCG1

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 6 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
pyridoxal 5’-phosphate salvage12808.7×0.011PDXK
pyridoxal metabolic process12808.7×0.011PDXK
intrinsic apoptotic signaling pathway in response to osmotic stress11404.3×0.011SCN2A
pyridoxamine metabolic process11404.3×0.011PDXK
glycoprotein transport1936.2×0.011ABCG1
cellular response to high density lipoprotein particle stimulus1936.2×0.011ABCG1
negative regulation of triglyceride biosynthetic process1702.2×0.011SIK1
obsolete negative regulation of CREB transcription factor activity1702.2×0.011SIK1
regulation of myotube differentiation1561.7×0.013SIK1
response to lipid1401.2×0.015ABCG1
positive regulation of anoikis1312.1×0.015SIK1
negative regulation of cholesterol storage1255.3×0.015ABCG1
negative regulation of macrophage derived foam cell differentiation1216.1×0.015ABCG1
CDP-diacylglycerol biosynthetic process1216.1×0.015AGPAT3
intracellular cholesterol transport1216.1×0.015ABCG1
anoikis1216.1×0.015SIK1
amyloid precursor protein catabolic process1200.6×0.015ABCG1
phospholipid efflux1187.2×0.015ABCG1
positive regulation of cholesterol biosynthetic process1187.2×0.015ABCG1
regulation of cholesterol metabolic process1187.2×0.015ABCG1
xenobiotic detoxification by transmembrane export across the plasma membrane1187.2×0.015ABCG1
negative regulation of GTPase activity1175.5×0.015RRP1B
low-density lipoprotein particle remodeling1175.5×0.015ABCG1
phospholipid homeostasis1165.2×0.015ABCG1
regulation of sodium ion transport1156.0×0.015SIK1
reverse cholesterol transport1156.0×0.015ABCG1
host-mediated activation of viral transcription1147.8×0.015RRP1B
positive regulation of amyloid-beta formation1147.8×0.015ABCG1
high-density lipoprotein particle remodeling1133.8×0.016ABCG1
negative regulation of gluconeogenesis1133.8×0.016SIK1

Therapeutics

Drug target analysis

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

Druggability breadth: 4 of 7 evidence-associated genes (57%) have a ChEMBL target (buckets above are over the deeply-mined display cohort).

Genes with an approved drug

The molecule shown is one approved compound that hits the gene — not necessarily a drug of choice or one indicated for this disease.

SymbolExample approved molecule
SIK1FEDRATINIB
SCN2ABEPRIDIL
PDXKSUNITINIB

Top cohort targets by molecule count

SymbolMoleculesMax phase
SCN2A994
SIK1194
PDXK64
KRTAP12-200
RRP1B00
AGPAT300
ABCG100

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
FEDRATINIB4SIK1
AXITINIB4SIK1
NERATINIB4SIK1
VANDETANIB4SIK1
BOSUTINIB4SIK1
PAZOPANIB4SIK1
NINTEDANIB4SIK1
SUNITINIB4PDXK, SIK1
DASATINIB4SIK1
MIDOSTAURIN4SIK1
BEPRIDIL4SCN2A
DIBUCAINE4SCN2A
ARTICAINE4SCN2A
BUPIVACAINE4SCN2A
IMIPRAMINE4SCN2A
DROPERIDOL4SCN2A
DICYCLOMINE4SCN2A
TETRABENAZINE4SCN2A
PHENIRAMINE4SCN2A
PRILOCAINE4SCN2A
PROPOXYCAINE4SCN2A
PROPARACAINE4SCN2A
HEXYLCAINE4SCN2A
PRAMOXINE4SCN2A
BENOXINATE4SCN2A
QUINIDINE4SCN2A
FELODIPINE4SCN2A
PHENYTOIN4SCN2A
QUININE4SCN2A
NISOLDIPINE4SCN2A

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 2.

Cohort genes with ChEMBL bioactivity (full, sorted by assay count)

SymbolAssaysType breakdown
SIK1210Binding:205, Toxicity:4, ADMET:1
SCN2A203Binding:172, Functional:20, ADMET:10, Toxicity:1
PDXK21Binding:20, ADMET:1
RRP1B1Binding:1

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
AGPAT32.3.1.511-acylglycerol-3-phosphate O-acyltransferase
PDXK2.7.1.35pyridoxal kinase

Cohort genes with high screening signal

≥100 ChEMBL assays — a studied-ness signal; see Therapeutics for approved-drug status.

SymbolChEMBL assays
SIK1210
SCN2A203

Pharmacogenomics

Cohort genes with a PharmGKB record: 7; with CPIC/DPWG dosing guidelines: 0.

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

Chemical tractability of cohort targets

30 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.

CompoundMax phaseCohort target (bioactivity)
FEDRATINIB4SIK1
AXITINIB4SIK1
NERATINIB4SIK1
VANDETANIB4SIK1
BOSUTINIB4SIK1
PAZOPANIB4SIK1
NINTEDANIB4SIK1
SUNITINIB4PDXK, SIK1
DASATINIB4SIK1
MIDOSTAURIN4SIK1
BEPRIDIL4SCN2A
DIBUCAINE4SCN2A
ARTICAINE4SCN2A
BUPIVACAINE4SCN2A
IMIPRAMINE4SCN2A
DROPERIDOL4SCN2A
DICYCLOMINE4SCN2A
TETRABENAZINE4SCN2A
PHENIRAMINE4SCN2A
PRILOCAINE4SCN2A
PROPOXYCAINE4SCN2A
PROPARACAINE4SCN2A
HEXYLCAINE4SCN2A
PRAMOXINE4SCN2A
BENOXINATE4SCN2A
QUINIDINE4SCN2A
FELODIPINE4SCN2A
PHENYTOIN4SCN2A
QUININE4SCN2A
NISOLDIPINE4SCN2A

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)3SIK1, SCN2A, PDXK
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug1ABCG1
DDruggable family + AlphaFold only, no drug1AGPAT3
EDifficult family or no structure, no drug2KRTAP12-2, RRP1B

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
RRP1B1PDXK
KRTAP12-20
AGPAT30
ABCG10

Clinical trials & evidence

Clinical trials

Clinical trials: 0.