Lennox-Gastaut syndrome

disease
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Also known as encephalopathy of childhoodepileptic encephalopathy Lennox-Gastaut typeLGS

Summary

Lennox-Gastaut syndrome (MONDO:0016532) is a disease with 15 cohort genes and 55 clinical trials. The dominant Reactome pathway is Interaction between L1 and Ankyrins (3 cohort genes). Top therapeutic interventions include cannabidiol, rufinamide, and fenfluramine.

At a glance

  • Prevalence: 1-5 / 10 000 (Europe) [Orphanet-validated]
  • Cohort genes: 15
  • ClinVar variants: 13
  • Phenotypes (HPO): 25
  • Clinical trials: 55

Clinical features

Epidemiology

Prevalence records

2 prevalence record(s), Orphanet:

TypeClassValueGeographyValidation
Annual incidence1-9 / 1 000 0000.1EuropeValidated
Point prevalence1-5 / 10 00015EuropeValidated

Signs & symptoms

Clinical features (HPO)

25 HPO clinical features (Orphanet curated; top 25 by frequency):

HPO IDTermFrequency
HP:0001249Intellectual disabilityVery frequent (80-99%)
HP:0001298EncephalopathyVery frequent (80-99%)
HP:0011195EEG with focal sharp slow wavesVery frequent (80-99%)
HP:0000708Atypical behaviorFrequent (30-79%)
HP:0000718Aggressive behaviorFrequent (30-79%)
HP:0000729Autistic behaviorFrequent (30-79%)
HP:0000737IrritabilityFrequent (30-79%)
HP:0000752HyperactivityFrequent (30-79%)
HP:0001268Mental deteriorationFrequent (30-79%)
HP:0001336MyoclonusFrequent (30-79%)
HP:0002069Bilateral tonic-clonic seizureFrequent (30-79%)
HP:0002353EEG abnormalityFrequent (30-79%)
HP:0002363Abnormal brainstem morphologyFrequent (30-79%)
HP:0002527FallsFrequent (30-79%)
HP:0007270Atypical absence seizureFrequent (30-79%)
HP:0010818Generalized tonic seizureFrequent (30-79%)
HP:0010819Atonic seizureFrequent (30-79%)
HP:0012075Personality disorderFrequent (30-79%)
HP:0012758Neurodevelopmental delayFrequent (30-79%)
HP:0000709PsychosisOccasional (5-29%)
HP:0000741ApathyOccasional (5-29%)
HP:0002123Generalized myoclonic seizureOccasional (5-29%)
HP:0002321VertigoOccasional (5-29%)
HP:0002376Developmental regressionOccasional (5-29%)
HP:0007359Focal-onset seizureOccasional (5-29%)

Identifiers

Disease identifiers

FieldValue
Canonical nameLennox-Gastaut syndrome
Mondo IDMONDO:0016532
MeSHD065768
OMIM606369
Orphanet2382
DOIDDOID:0050561
ICD-10-CMG40.81
ICD-11651135242
NCITC84816
SNOMED CT230418006
UMLSC0238111
MedGen116044
GARD0009912
MedDRA10048816
NORD1358
Is cancer (heuristic)no

Also known as: encephalopathy of childhood · epileptic encephalopathy Lennox-Gastaut type · LGS

Data availability: 13 ClinVar variants · 7 GenCC gene-disease records · 19 cell lines.

Disease family

An umbrella term covering 3 Mondo subtypes.

Classification path: disease › human disease › disease by body system or component › syndromic diseaseLennox-Gastaut syndrome

Related subtypes (1183): Neu-Laxova syndrome, inclusion body myopathy with Paget disease of bone and frontotemporal dementia, syndromic intellectual disability, abdominal obesity-metabolic syndrome, fibrogenesis imperfecta ossium, Fanconi renotubular syndrome, palindromic rheumatism, hepatorenal syndrome, Potter sequence, vertebral artery insufficiency, sick sinus syndrome, Tietze syndrome, toxic shock syndrome, capillary leak syndrome, dumping syndrome, FG syndrome, basilar artery insufficiency, long QT syndrome, Treacher-Collins syndrome, superior mesenteric artery syndrome, disappearing bone disease, Brown-Sequard syndrome, Froelich syndrome, diffuse infiltrative lymphocytosis syndrome, Capgras syndrome, compartment syndrome, central sleep apnea syndrome, irritable bowel syndrome, nephrotic syndrome, myalgic encephalomeyelitis/chronic fatigue syndrome, acute coronary syndrome, fibromyalgia, substance withdrawal syndrome, acute chest syndrome, Barre-Lieou syndrome, cauda equina syndrome, Kluver-Bucy syndrome, Miller Fisher syndrome, persian gulf syndrome, Reye syndrome, thoracic outlet syndrome, Waterhouse-Friderichsen syndrome, Wissler syndrome, acute respiratory distress syndrome, Achenbach syndrome, miliaria, anterior spinal artery syndrome, burning mouth syndrome, dry eye syndrome, empty sella syndrome, euthyroid sick syndrome, lateral medullary syndrome, subclavian steal syndrome, tarsal tunnel syndrome, tethered spinal cord syndrome, branchio-oto-renal syndrome, prune belly syndrome, Achard syndrome, alopecia-epilepsy-pyorrhea-intellectual disability syndrome, Finnish type amyloidosis, Angelman syndrome, aniridia-absent patella syndrome, ankyloblepharon filiforme adnatum-cleft palate syndrome, Townes-Brocks syndrome, obstructive sleep apnea syndrome, Lown-Ganong-Levine syndrome, Behcet disease, brachydactyly-arterial hypertension syndrome, brachydactyly type A2, fibular aplasia-ectrodactyly syndrome, brachydactyly-nystagmus-cerebellar ataxia syndrome, Sillence syndrome, Brachymorphism-onychodysplasia-dysphalangism syndrome, brachytelephalangy-dysmorphism-Kallmann syndrome, dilated cardiomyopathy 1A, cat-eye syndrome, cerebrocostomandibular syndrome, Alagille syndrome, autosomal dominant chondrodysplasia punctata, cleidocranial dysplasia 1, cleidorhizomelic syndrome, cochleosaccular degeneration-cataract syndrome, renal coloboma syndrome, Cri-du-chat syndrome, autosomal dominant deafness - onychodystrophy syndrome, cerebral arteriopathy with subcortical infarcts and leukoencephalopathy, Duane retraction syndrome, 3-M syndrome, dyschondrosteosis-nephritis syndrome, hereditary benign intraepithelial dyskeratosis, encephalopathy, recurrent, of childhood, Camurati-Engelmann disease, Felty syndrome, chromosome 16p12.1 deletion syndrome, 520kb, Frasier syndrome, Gamstorp-Wohlfart syndrome, Tourette syndrome, glaucoma-sleep apnea syndrome, renal cysts and diabetes syndrome, hypotrichosis-lymphedema-telangiectasia syndrome (grouping), GMS syndrome, gray platelet syndrome, hand-foot-genital syndrome, facial hemiatrophy, Bencze syndrome, alpha thalassemia-intellectual disability syndrome type 1, Gilbert syndrome, mullerian duct anomalies-limb anomalies syndrome, hypoparathyroidism-deafness-renal disease syndrome, chromosome 18p deletion syndrome, Pallister-Hall syndrome, ichthyosis-cheek-eyebrow syndrome, Jacobsen syndrome, palmoplantar keratoderma-hereditary motor and sensory neuropathy syndrome, palmoplantar keratoderma-esophageal carcinoma syndrome, Kleine-Levin syndrome, angioosteohypertrophic syndrome, congenital laryngeal web, Lenz-Majewski hyperostotic dwarfism, Noonan syndrome with multiple lentigines, lymphedema-cerebral arteriovenous anomaly syndrome, microcephaly with or without chorioretinopathy, lymphedema, or intellectual disability, yellow nail syndrome, lymphedema-distichiasis syndrome, Nager acrofacial dysostosis, jaw-winking syndrome, Marfan syndrome, Melkersson-Rosenthal syndrome, metaphyseal chondrodysplasia, Jansen type, Schmid metaphyseal chondrodysplasia, metaphyseal dysplasia-maxillary hypoplasia-brachydacty syndrome, microgastria-limb reduction defect syndrome, Mobius syndrome, muscular atrophy-ataxia-retinitis pigmentosa-diabetes mellitus syndrome, nail-patella syndrome, Schilbach-Rott syndrome, syndromic orbital border hypoplasia, Buschke-Ollendorff syndrome, nasopalpebral lipoma-coloboma syndrome, Perry syndrome, Poland syndrome, polydactyly-myopia syndrome, Greig cephalopolysyndactyly syndrome, Prader-Willi syndrome, Guttmacher syndrome, Currarino triad, Hutchinson-Gilford progeria syndrome, progeria-short stature-pigmented nevi syndrome, Liddle syndrome, exfoliation syndrome, ptosis-strabismus-ectopic pupils syndrome, radial hypoplasia-triphalangeal thumbs-hypospadias-maxillary diastema syndrome, radial ray hypoplasia-choanal atresia syndrome, Roussy-Levy syndrome, Silver-Russell syndrome, Ruvalcaba syndrome, oculodental syndrome, Rutherfurd type, aplasia of lacrimal and salivary glands, scalp defects-postaxial polydactyly syndrome, ulnar-mammary syndrome, septooptic dysplasia, Czeizel-Losonci syndrome, polycystic ovary syndrome, stiff-person syndrome, syndactyly-polydactyly-ear lobe syndrome, HELLP syndrome, double uterus-hemivagina-renal agenesis syndrome, VACTERL/vater association, posterior fusion of lumbosacral vertebrae-blepharoptosis syndrome, ptosis-vocal cord paralysis syndrome, Freeman-Sheldon syndrome, Williams syndrome, Denys-Drash syndrome, Wolf-Hirschhorn syndrome, ablepharon macrostomia syndrome, acrocallosal syndrome, PAGOD syndrome, alopecia - intellectual disability syndrome, mitochondrial DNA depletion syndrome 4a, Alstrom syndrome, aniridia-cerebellar ataxia-intellectual disability syndrome, aniridia-renal agenesis-psychomotor retardation syndrome, aplasia cutis congenita-intestinal lymphangiectasia syndrome, fetal akinesia deformation sequence, blepharophimosis-ptosis-esotropia-syndactyly-short stature syndrome, Bloom syndrome, Elsahy-Waters syndrome, campomelia, Cumming type, camptomelic syndrome, long-limb type, congenital cataract-ichthyosis syndrome, colobomatous optic disc-macular atrophy-chorioretinopathy syndrome, hepatic fibrosis-renal cysts-intellectual disability syndrome, Charcot-Marie-Tooth disease-hearing loss-intellectual disability syndrome, CHARGE syndrome, Aagenaes syndrome, infantile choroidocerebral calcification syndrome, Yunis-Varon syndrome, corneal dystrophy-perceptive deafness syndrome, cortical blindness-intellectual disability-polydactyly syndrome, Crigler-Najjar syndrome, cataract-nephropathy-encephalopathy syndrome, Fraser syndrome, cystic fibrosis-gastritis-megaloblastic anemia syndrome, cystinuria, DOORS syndrome, high myopia-sensorineural deafness syndrome, dermochondrocorneal dystrophy, nephrogenic diabetes insipidus-intracranial calcification syndrome, diverticulosis of bowel, hernia, and retinal detachment, Dyggve-Melchior-Clausen disease, cerebellar ataxia, intellectual disability, and dysequilibrium, ectrodactyly-polydactyly syndrome, Bonnemann-Meinecke-Reich syndrome, epidermolysis bullosa simplex 5B, with muscular dystrophy, Wolcott-Rallison syndrome, ermine phenotype, eyebrow duplication-syndactyly syndrome, Fanconi-like syndrome, gingival fibromatosis-facial dysmorphism syndrome, frontofacionasal dysplasia, Fryns syndrome, German syndrome, Bernard-Soulier syndrome, triple-A syndrome, Grubben-de Cock-Borghgraef syndrome, mullerian derivatives-lymphangiectasia-polydactyly syndrome, Hirschsprung disease-hearing loss-polydactyly syndrome, Hirschsprung disease-nail hypoplasia-dysmorphism syndrome, Holzgreve-Wagner-Rehder syndrome, multinucleated neurons-anhydramnios-renal dysplasia-cerebellar hypoplasia-hydranencephaly syndrome, growth delay-hydrocephaly-lung hypoplasia syndrome, Dubin-Johnson syndrome, ornithine translocase deficiency, acrofrontofacionasal dysostosis 2, hypertrichotic osteochondrodysplasia Cantu type, hypergonadotropic hypogonadism-cataract syndrome, primary hypergonadotropic hypogonadism-partial alopecia syndrome, hypoparathyroidism-retardation-dysmorphism syndrome, hypospadias-intellectual disability, Goldblatt type syndrome, Bamforth-Lazarus syndrome, ichthyosis-hepatosplenomegaly-cerebellar degeneration syndrome, ichthyosis-intellectual disability-dwarfism-renal impairment syndrome, Vici syndrome, channelopathy-associated congenital insensitivity to pain, autosomal recessive, Joubert syndrome with oculorenal defect, oculocerebrofacial syndrome, Kaufman type, Landau-Kleffner syndrome, laryngo-onycho-cutaneous syndrome, Laurence-Moon syndrome, Donohue syndrome, Miller-Dieker lissencephaly syndrome, Marinesco-Sjogren syndrome, Frank-Ter Haar syndrome, Mietens syndrome, mesomelic dwarfism-cleft palate-camptodactyly syndrome, metaphyseal chondrodysplasia-retinitis pigmentosa syndrome, metaphyseal dysostosis-intellectual disability-conductive deafness syndrome, microcephalic primordial dwarfism, Toriello type, Say-Barber-Miller syndrome, microcephaly and chorioretinopathy 1, Galloway-Mowat syndrome, microtia with meatal atresia and conductive deafness, mucopolysaccharidosis type 6, mulibrey nanism, lethal multiple pterygium syndrome, lethal congenital contracture syndrome 1, Schwartz-Jampel syndrome, Nathalie syndrome, nephronophthisis 1, nephropathy - deafness - hyperparathyroidism syndrome, nephrosis-deafness-urinary tract-digital malformations syndrome, Netherton syndrome, Norman-Roberts syndrome, cloacal exstrophy, ichthyosis-oral and digital anomalies syndrome, Primrose syndrome, familial osteodysplasia, Anderson type, multicentric osteolysis, nodulosis, and arthropathy, osteopenia-intellectual disability-sparse hair syndrome, osteoporosis-pseudoglioma syndrome, Shwachman-Diamond syndrome, Parana hard-skin syndrome, PEHO syndrome, Imerslund-Grasbeck syndrome, Peters plus syndrome, pili torti-developmental delay-neurological abnormalities syndrome, Rabson-Mendenhall syndrome, postaxial acrofacial dysostosis, Gitelman syndrome, Wiedemann-Rautenstrauch syndrome, Acrootoocular syndrome, holoprosencephaly-postaxial polydactyly syndrome, autosomal recessive multiple pterygium syndrome, Perlman syndrome, retinitis pigmentosa-intellectual disability-deafness-hypogenitalism syndrome, EEC syndrome, SHORT syndrome, Sjogren syndrome, spastic tetraplegia-retinitis pigmentosa-intellectual disability syndrome, corneal-cerebellar syndrome, spastic ataxia-corneal dystrophy syndrome, spondylocostal dysostosis-anal and genitourinary malformations syndrome, familial infantile bilateral striatal necrosis, subaortic stenosis-short stature syndrome, thrombocytopenia-absent radius syndrome, thyrocerebrorenal syndrome, Pendred syndrome, VACTERL with hydrocephalus, Weaver syndrome, Werner syndrome, Wernicke-Korsakoff syndrome, wooly hair-hypotrichosis-everted lower lip-outstanding ears syndrome, de Sanctis-Cacchione syndrome, corpus callosum agenesis-abnormal genitalia syndrome, Alport syndrome-intellectual disability-midface hypoplasia-elliptocytosis syndrome, X-linked lissencephaly with abnormal genitalia, X-linked myotubular myopathy-abnormal genitalia syndrome, Christianson syndrome, Armfield syndrome, Lesch-Nyhan syndrome, Atkin-Flaitz syndrome, alpha-thalassemia-myelodysplastic syndrome, deafness-intellectual disability, Martin-Probst type syndrome, fragile X-associated tremor/ataxia syndrome, fragile X syndrome, syndactyly-telecanthus-anogenital and renal malformations syndrome, X-linked dominant chondrodysplasia, Chassaing-Lacombe type, X-linked central congenital hypothyroidism with late-onset testicular enlargement, Meester-Loeys syndrome, Arts syndrome, X-linked mandibulofacial dysostosis, Abruzzo-Erickson syndrome, Aicardi syndrome, craniofrontonasal syndrome, deafness-hypogonadism syndrome, X-linked corneal dermoid, immune dysregulation-polyendocrinopathy-enteropathy-X-linked syndrome, hydrocephaly-cerebellar agenesis syndrome, keratosis follicularis-dwarfism-cerebral atrophy syndrome, laryngeal abductor paralysis-intellectual disability syndrome, oculocerebrorenal syndrome, Menkes disease, paraplegia-intellectual disability-hyperkeratosis syndrome, mucopolysaccharidosis type 2, orofaciodigital syndrome I, otopalatodigital syndrome type 1, Pallister-W syndrome, Rett syndrome, SCARF syndrome, Simpson-Golabi-Behmel syndrome, torticollis-keloids-cryptorchidism-renal dysplasia syndrome, trigonocephaly-short stature-developmental delay syndrome, ulnar hypoplasia-split foot syndrome, van den Bosch syndrome, Wildervanck syndrome, Kearns-Sayre syndrome, MELAS syndrome, MERRF syndrome, Pearson syndrome, proximal tubulopathy-diabetes mellitus-cerebellar ataxia syndrome, pancreatic hypoplasia-diabetes-congenital heart disease syndrome, chondrodysplasia-pseudohermaphroditism syndrome, Qazi Markouizos syndrome, familial developmental dysphasia, atrioventricular defect-blepharophimosis-radial and anal defect syndrome, CODAS syndrome, lethal hemolytic anemia-genital anomalies syndrome, HEC syndrome, anophthalmia plus syndrome, infundibulopelvic stenosis-multicystic kidney syndrome, Ayme-Gripp syndrome, dilated cardiomyopathy 1E, diaphragmatic defect-limb deficiency-skull defect syndrome, skeletal dysplasia-epilepsy-short stature syndrome, Potocki-Shaffer syndrome, amelia cleft lip palate hydrocephalus iris coloboma, human HOXA1 syndromes, dyssegmental dysplasia-glaucoma syndrome, lung agenesis-heart defect-thumb anomalies syndrome, tetrasomy 12p, chromosome 18q deletion syndrome, lymphedema-atrial septal defects-facial changes syndrome, infantile convulsions and choreoathetosis, RHYNS syndrome, Pierre Robin sequence with pectus excavatum and rib and scapular anomalies, colobomatous macrophthalmia-microcornea syndrome, Marshall-Smith syndrome, distal monosomy 13q, MPI-congenital disorder of glycosylation, camptodactyly, myopia, and fibrosis of the medial rectus muscle of eye, radioulnar synostosis-microcephaly-scoliosis syndrome, blepharophimosis - intellectual disability syndrome, SBBYS type, complex regional pain syndrome type 1, temtamy preaxial brachydactyly syndrome, Diamond-Blackfan anemia 2, genitopatellar syndrome, Phelan-McDermid syndrome, hypotonia-cystinuria syndrome, DNA ligase IV deficiency, Hurler syndrome, Hurler-Scheie syndrome, Scheie syndrome, Duane-radial ray syndrome, psoriatic arthritis, neonatal ichthyosis-sclerosing cholangitis syndrome, skin fragility-woolly hair-palmoplantar keratoderma syndrome, tubulointerstitial nephritis and uveitis syndrome, caudal duplication, sweet syndrome, ichthyosis prematurity syndrome, Meacham syndrome, BNAR syndrome, PCWH syndrome, foveal hypoplasia - optic nerve decussation defect - anterior segment dysgenesis syndrome, B-cell immunodeficiency, distal limb anomalies, and urogenital malformations, MEDNIK syndrome, Cerebrorenodigital syndrome, fetal valproate syndrome, Goldberg-Shprintzen syndrome, Al-Gazali syndrome, CEDNIK syndrome, osteosclerosis-ichthyosis-premature ovarian failure syndrome, cortical dysplasia-focal epilepsy syndrome, DK1-congenital disorder of glycosylation, Potocki-Lupski syndrome, Pitt-Hopkins syndrome, XFE progeroid syndrome, deafness-infertility syndrome, COG1-congenital disorder of glycosylation, autosomal dominant familial hematuria-retinal arteriolar tortuosity-contractures syndrome, microtia-eye coloboma-imperforation of the nasolacrimal duct syndrome, mitochondrial DNA depletion syndrome, encephalomyopathic form with methylmalonic aciduria, ANE syndrome, CLOVES syndrome, Hirschsprung disease-ganglioneuroblastoma syndrome, parkinsonism-dystonia, infantile, alpha 1-antitrypsin deficiency, COG5-congenital disorder of glycosylation, chromosome 13q14 deletion syndrome, deafness-lymphedema-leukemia syndrome, microcephaly-capillary malformation syndrome, EDICT syndrome, peripheral neuropathy-myopathy-hoarseness-hearing loss syndrome, hypomyelinating leukodystrophy 8 with or without oligodontia and-or hypogonadotropic hypogonadism, IMAGe syndrome, short stature-onychodysplasia-facial dysmorphism-hypotrichosis syndrome, microcephalic primordial dwarfism, Alazami type, intellectual disability-strabismus syndrome, estrogen resistance syndrome, Hartsfield-Bixler-Demyer syndrome, severe dermatitis-multiple allergies-metabolic wasting syndrome, alacrima, achalasia, and intellectual disability syndrome, familial episodic pain syndrome with predominantly lower limb involvement, congenital microcephaly - severe encephalopathy - progressive cerebral atrophy syndrome, diffuse cerebral and cerebellar atrophy - intractable seizures - progressive microcephaly syndrome, postaxial polydactyly-anterior pituitary anomalies-facial dysmorphism syndrome, tall stature-scoliosis-macrodactyly of the great toes syndrome, intellectual disability, autosomal dominant 29, intellectual disability, autosomal dominant 30, congenital sideroblastic anemia-B-cell immunodeficiency-periodic fever-developmental delay syndrome, retinitis pigmentosa-juvenile cataract-short stature-intellectual disability syndrome, polyendocrine-polyneuropathy syndrome, chronic atrial and intestinal dysrhythmia, motor developmental delay due to 14q32.2 paternally expressed gene defect, peeling skin-leukonuchia-acral punctate keratoses-cheilitis-knuckle pads syndrome, mandibulofacial dysostosis with alopecia, epilepsy with myoclonic atonic seizures, short stature, microcephaly, and endocrine dysfunction, progressive microcephaly-seizures-cortical blindness-developmental delay syndrome, PMP22-RAI1 contiguous gene duplication syndrome, acute infantile liver failure-cerebellar ataxia-peripheral sensory motor neuropathy syndrome, familial progressive retinal dystrophy-iris coloboma-congenital cataract syndrome, macrothrombocytopenia-lymphedema-developmental delay-facial dysmorphism-camptodactyly syndrome, Luscan-Lumish syndrome, even-plus syndrome, MIRAGE syndrome, growth retardation, intellectual developmental disorder, hypotonia, and hepatopathy, intellectual disability-epilepsy-extrapyramidal syndrome, 48,XXYY syndrome, FRAXF syndrome, complex hereditary spastic paraplegia, aniridia-ptosis-intellectual disability-familial obesity syndrome, aniridia - intellectual disability syndrome, ankyloblepharon filiforme-imperforate anus syndrome, pentasomy X, Bardet-Biedl syndrome, anophthalmia-megalocornea-cardiopathy-skeletal anomalies syndrome, Bartter syndrome, arachnodactyly-intellectual disability-dysmorphism syndrome, ataxia-photosensitivity-short stature syndrome, Brugada syndrome, Feingold syndrome, cardiomyopathy-cataract-hip spine disease syndrome, cataract - microcornea syndrome, autism-facial port-wine stain syndrome, cataract-intellectual disability-anal atresia-urinary defects syndrome, cataract-deafness-hypogonadism syndrome, syndromic craniosynostosis, drug rash with eosinophilia and systemic symptoms, multicentric reticulohistiocytosis, hereditary sensory and autonomic neuropathy with deafness and global delay, craniofacial microsomia, ring chromosome 10, Coffin-Siris syndrome, corpus callosum agenesis-double urinary collecting system syndrome, short rib-polydactyly syndrome, oromandibular-limb anomalies syndrome, hemophagocytic syndrome, cataract-glaucoma syndrome, diencephalic syndrome, hypereosinophilic syndrome, distal trisomy 14q, intellectual disability-cataracts-kyphosis syndrome, progressive familial intrahepatic cholestasis, thoraco-abdominal enteric duplication, oculomaxillofacial dysostosis, growth hormone insensitivity syndrome, syndromic dyslipidemia, macrothrombocytopenia and granulocyte inclusions with or without nephritis or sensorineural hearing loss, epiphyseal dysplasia-hearing loss-dysmorphism syndrome, eosinophilic granulomatosis with polyangiitis, axial mesodermal dysplasia spectrum, fetal hydantoin syndrome, vitamin K-antagonist embryofetopathy, fetal alcohol syndrome, methimazole embryofetopathy, Evans syndrome, Cornelia de Lange syndrome, cleft lip-retinopathy syndrome, cleft lip/palate-deafness-sacral lipoma syndrome, Crandall syndrome, syndromic microphthalmia, Cole-Carpenter syndrome, myotonic syndrome, Guillain-Barre syndrome, atypical hemolytic-uremic syndrome, Hennekam syndrome, Hernández-Aguirre Negrete syndrome, nodular neuronal heterotopia, Hirschsprung disease-type D brachydactyly syndrome, holoprosencephaly, hydrocephalus-obesity-hypogonadism syndrome, hydrocephalus-blue sclerae-nephropathy syndrome, xeroderma pigmentosum-Cockayne syndrome complex, Joubert syndrome with ocular defect, hypogonadism-mitral valve prolapse-intellectual disability syndrome, hypogonadotropic hypogonadism-retinitis pigmentosa syndrome, hypotrichosis-intellectual disability, Lopes type, congenital ichthyosis-microcephalus-tetraplegia syndrome, Hughes-Stovin syndrome, heart-hand syndrome, ptosis-upper ocular movement limitation-absence of lacrimal punctum syndrome, syndromic agammaglobulinemia, isotretinoin syndrome, microcornea-posterior megalolenticonus-persistent fetal vasculature-coloboma syndrome, Kabuki syndrome, Kenny-Caffey syndrome, muscular pseudohypertrophy-hypothyroidism syndrome, Kousseff syndrome, limb body wall complex, Lowe-Kohn-Cohen syndrome, macrocephaly-short stature-paraplegia syndrome, primary ciliary dyskinesia, familial intestinal malrotation-facial anomalies syndrome, primary hypertrophic osteoarthropathy, Melhem-Fahl syndrome, lower limb deficiency-hypospadias syndrome, 8p23.1 microdeletion syndrome, sickle cell-beta-thalassemia disease syndrome, sickle cell-hemoglobin c disease syndrome, sickle cell-hemoglobin d disease syndrome, sickle cell-hemoglobin E disease syndrome, hereditary persistence of fetal hemoglobin-sickle cell disease syndrome, microcephaly-brain defect-spasticity-hypernatremia syndrome, microcephaly-microcornea syndrome, Seemanova type, Meier-Gorlin syndrome, Mikati-Najjar-Sahli syndrome, shoulder and girdle defects-familial intellectual disability syndrome, myopathy-growth delay-intellectual disability-hypospadias syndrome, Fuchs heterochromic iridocyclitis, microcephalic osteodysplastic primordial dwarfism types I and III, osteochondrodysplatic nanism-deafness-retinitis pigmentosa syndrome, arthrogryposis-renal dysfunction-cholestasis syndrome, oculo-skeletal-renal syndrome, olivopontocerebellar atrophy-deafness syndrome, Opitz G/BBB syndrome, imperforate oropharynx-costo vetebral anomalies syndrome, Bruck syndrome, osteoporosis-macrocephaly-blindness-joint hyperlaxity syndrome, calciphylaxis, recessive intellectual disability-motor dysfunction-multiple joint contractures syndrome, X-linked ichthyosis syndrome, autoimmune polyendocrinopathy, renal caliceal diverticuli-deafness syndrome, tempi syndrome, syndromic oculocutaneous albinism, short stature-deafness-neutrophil dysfunction-dysmorphism syndrome, congenital varicella syndrome, polyneuropathy-intellectual disability-acromicria-premature menopause syndrome, celiac trunk compression syndrome, hypoplastic pancreas-intestinal atresia-hypoplastic gallbalder syndrome, fetal cytomegalovirus syndrome, Reunion island Larsen syndrome, 46,XX disorder of sex development-anorectal anomalies syndrome, mitochondrial neurogastrointestinal encephalomyopathy, Baraitser-Winter cerebrofrontofacial syndrome, mirror polydactyly-vertebral segmentation-limbs defects syndrome, intellectual disability-hypotonia-skin hyperpigmentation syndrome, congenital hereditary facial paralysis-variable hearing loss syndrome, intellectual disability-microcephaly-phalangeal-facial abnormalities syndrome, Mayer-Rokitansky-Kuster-Hauser syndrome, developmental and speech delay due to SOX5 deficiency, Spigelian hernia-cryptorchidism syndrome, autosomal recessive leukoencephalopathy-ischemic stroke-retinitis pigmentosa syndrome, severe neonatal hypotonia-seizures-encephalopathy syndrome due to 5q31.3 microdeletion, multiple sclerosis-ichthyosis-factor VIII deficiency syndrome, X-linked spasticity-intellectual disability-epilepsy syndrome, spina bifida-hypospadias syndrome, hantavirus pulmonary syndrome, white matter hypoplasia-corpus callosum agenesis-intellectual disability syndrome, deafness-genital anomalies-metacarpal and metatarsal synostosis syndrome, hearing loss-familial salivary gland insensitivity to aldosterone syndrome, multiple synostoses syndrome, central nervous system calcification-deafness-tubular acidosis-anemia syndrome, multiple paragangliomas associated with polycythemia, syngnathia multiple anomalies, Takayasu arteritis, severe early-onset obesity-insulin resistance syndrome due to SH2B1 deficiency, spondylocostal dysostosis-hypospadias-intellectual disability syndrome, hypotrichosis-deafness syndrome, thalidomide embryopathy, trisomy X, trisomy 13, trisomy 18, umbilical cord ulceration-intestinal atresia syndrome, microcephaly-brachydactyly-kyphoscoliosis syndrome, Waardenburg syndrome, Weill-Marchesani syndrome, infantile spasms, Wolfram syndrome, epidermal nevus syndrome, digital anomalies-intellectual disability-short stature syndrome, intellectual disability-obesity-brain malformations-facial dysmorphism syndrome, Erdheim-Chester disease, Stevens-Johnson syndrome, CADDS, finger hyperphalangy - toe anomalies - severe pectus excavatum syndrome, ataxia - telangiectasia variant, growth retardation-mild developmental delay-chronic hepatitis syndrome, primary microcephaly-mild intellectual disability-young-onset diabetes syndrome, ferro-cerebro-cutaneous syndrome, dystonia-aphonia syndrome, microcephaly-complex motor and sensory axonal neuropathy syndrome, X-linked microcephaly-growth retardation-prognathism-cryptorchidism syndrome, severe intellectual disability-hypotonia-strabismus-coarse face-planovalgus syndrome, intrauterine growth restriction-short stature-early adult-onset diabetes syndrome, pseudoxanthoma elasticum-like skin manifestations with retinitis pigmentosa, familial chylomicronemia syndrome, caudal regression-sirenomelia spectrum, visceral heterotaxy, Holmes-Adie syndrome, microcephalic primordial dwarfism due to RTTN deficiency, Joubert syndrome, congenital generalized hypercontractile muscle stiffness syndrome, Kallmann syndrome, Caroli syndrome, X-linked female restricted facial dysmorphism-short stature-choanal atresia-intellectual disability, microlissencephaly-micromelia syndrome, branchiootic syndrome, Plummer-Vinson syndrome, Cushing syndrome, McCune-Albright syndrome, Meckel syndrome, SUNCT syndrome, mucopolysaccharidosis type 3, mucopolysaccharidosis type 4, congenital myasthenic syndrome, Loeys-Dietz syndrome, Alport syndrome, schisis association, Tolosa-Hunt syndrome, iridocorneal endothelial syndrome, Noonan syndrome, short fifth metacarpals-insulin resistance syndrome, progressive supranuclear palsy, benign exophthalmos syndrome, Sandifer syndrome, global developmental delay-osteopenia-ectodermal defect syndrome, tubular renal disease-cardiomyopathy syndrome, angioosteohypotrophic syndrome, 6q terminal deletion syndrome, Axenfeld-Rieger syndrome, peroxisome biogenesis disorder, ectodermal dysplasia syndrome, Seckel syndrome, Sotos syndrome, Stickler syndrome, pelvis syndrome, Susac syndrome, ischio-vertebral syndrome, BRESEK syndrome, Turner syndrome, Usher syndrome, obesity-colitis-hypothyroidism-cardiac hypertrophy-developmental delay syndrome, CREST syndrome, Sheehan syndrome, polymyalgia rheumatica, autoinflammatory syndrome, neuroleptic malignant syndrome, pituitary stalk interruption syndrome, monosomy 13q34, ring chromosome 13, 48,XXXY syndrome, 49,XXXXY syndrome, hereditary continuous muscle fiber activity, Eisenmenger syndrome, Robinow syndrome, Ehlers-Danlos syndrome, hypoplastic right heart syndrome, shone complex, 48,XYYY syndrome, subcortical band heterotopia, complex regional pain syndrome type 2, faciodigitogenital syndrome, neoplastic syndrome, paraneoplastic syndrome, post-infectious syndrome, Achard-Thiers syndrome, acral dysostosis dyserythropoiesis syndrome, agnathia-microstomia-synotia, Aksu von Stockhausen syndrome, Aloi Tomasini Isaia syndrome, temporomandibular joint dysfunction syndrome, Apert-like polydactyly syndrome, arakawa syndrome 2, arena syndrome, Arnold stickler bourne syndrome, baetz-greenwalt syndrome, bagatelle Cassidy syndrome, baker Vinters syndrome, bobble-head doll syndrome, Boerhaave syndrome, Cantu Sanchez-Corona Fragoso syndrome, Cantu Sanchez-Corona Hernandez syndrome, carbon baby syndrome, Carnevale hernandez castillo syndrome, Cartwright Nelson Fryns syndrome, Charles bonnet syndrome, Parinaud syndrome, corticobasal degeneration disorder, hair defect with photosensitivity and intellectual disability syndrome, AIDS dysmorphic syndrome, congenital acardia, acute lymphoblastic leukemia congenital sporadic aniridia, aglossia and situs inversus, agyria pachygyria polymicrogyria, agyria-pachygyria type 1, Ahumada Del Castillo syndrome, alopecia congenita keratosis palmoplantaris, alpha-mannosidosis type 1, aluminosis, Mauriac syndrome, ankle defects short stature, ankyloblepharon filiforme imperforate anus, annular constricting bands, anophthalmia cleft palate micrognathia, anophthalmia esophageal atresia cryptorchidism, anotia facial palsy cardiac defect, aortic dissection lentiginosis, childhood aortic valve stenosis, arthrogryposis IUGR thoracic dystrophy, arthrogryposis multiplex congenita CNS calcification, arthrogryposis spinal muscular atrophy, asternia, atlanto-axial fusion, atrophoderma of Pierini and Pasini, Barnicoat Baraitser syndrome, Basedow’s coma, BD syndrome, Beardwell syndrome, bhaskar jagannathan syndrome, bidirectional tachycardia, bilirubin induced brain injury in the newborn, blepharo naso facial syndrome van Maldergem type, bone dysplasia corpus callosum agenesis, brachydactyly absence of distal phalanges, brachydactyly anonychia, brachydactyly small stature face anomalies, brachydactyly tibial hypoplasia, brittle bone syndrome lethal type, bronchiectasis oligospermia, Brunsting-Perry syndrome, bruyn scheltens syndrome, burn goodship syndrome, camptodactyly joint contractures and facial skeletal dysplasia, camptodactyly vertebral fusion, Cantu Sanchez-Corona Garcia-Cruz syndrome, cardiac hydatid cysts with intracavitary expansion, cardioencephalomyopathy, cardiofacial syndrome short limbs, cardiomelic syndrome stratton Koehler type, cardiomyopathy and deafness due to tRNA lysine gene mutation, cardiomyopathy diabetes deafness, cardiomyopathy hypogonadism collagenoma syndrome, cardiomyopathy hypogonadism metabolic anomalies, cardiomyopathy spherocytosis, carpo tarsal osteolysis recessive, autosomal dominant cataract, cataract skeletal anomalies, cennamo gangemi syndrome, cerebellar agenesis, cerebello-olivary atrophy, cerebral calcification cerebellar hypoplasia, cerebral calcifications opalescent teeth phosphaturia, oculo digital syndrome, chondrodysplasia, choreoacanthocytosis amyotrophic, chorioretinopathy dominant form microcephaly, Christian Demyer Franken syndrome, Christian Johnson angenieta syndrome, chromosome 3 duplication syndrome, chronic demyelinizing neuropathy with IgM monoclonal, ciliary dyskinesia-bronchiectasis, circumscribed cutaneous aplasia of the vertex, circumscribed disseminated keratosis Jadassohn lew type, cleft lip and palate malrotation cardiopathy, cleft lip and/or palate with mucous cysts of lower, cleft lip palate dysmorphism kumar type, cleft lip palate intellectual disability corneal opacity, cleft lip palate oligodontia syndactyly pili torti, cleft lip palate pituitary deficiency, cleft lip palate-tetraphocomelia, cleft lower lip cleft lateral canthi chorioretinal, cleft palate cardiac defect ectrodactyly, cleft palate colobomata radial synostosis deafness, cleft palate heart disease polydactyly absent tibia, cleft tongue, coarse face hypotonia constipation, Cohen Lockood Wyborney syndrome, type 2 collagenopathy, Collins-Sakati syndrome, coloboma porencephaly hydronephrosis, colobomata unilobar lung heart defect, colonic malakoplakia, Colver Steer Godman syndrome, Combarros Calleja Leno syndrome, complement receptor deficiency, congenital absence of the sternocleidomastoid muscle, congenital amputation, congenital aneurysms of the great vessels, congenital articular rigidity, congenital benign spinal muscular atrophy dominant, congenital cardiovascular shunt, congenital contractures, congenital craniosynostosis maternal hyperthyroiditis, congenital cystic eye, congenital heart disease ptosis hypodontia craniostosis, congenital heart disease radio ulnar synostosis intellectual disability, congenital mumps, congenital stenosis of cervical medullary canal, Dennis-Fairhurst-Moore syndrome, congenital unilateral pulmonary hypoplasia, congenital vagal hyperreflexivity, Cormier Rustin Munnich syndrome, corneal crystals myopathy neuropathy, corneal dystrophy ichthyosis microcephaly intellectual disability, corneal dystrophy pigmentary anomaly malabsorption, corpus callosum agenesis of blepharophimosis robin type, corpus callosum dysgenesis X-linked recessive, corpus callosum dysgenesis cleft spasm, corpus callosum dysgenesis hypopituitarism, cortada Koussef Matsumoto syndrome, Cortes Lacassie syndrome, craniofacial and skeletal defects, craniofacial dysostosis arthrogryposis progeroid appearance, craniofrontonasal syndrome Teebi type, craniostenosis with congenital heart disease intellectual disability, crawfurd syndrome, cutis gyratum acanthosis nigricans craniosynostosis, cutis laxa osteoporosis, Davenport-Donlan syndrome, Davis Lafer syndrome, de Hauwere Leroy adriaenssens syndrome, deafness conductive stapedial ear malformation facial palsy, deafness goiter stippled epiphyses, deafness hypospadias metacarpal and metatarsal syndrome, deafness mesenteric diverticula of small bowel neuropathy, deafness peripheral neuropathy arterial disease, deafness progressive cataract autosomal dominant, dermatocardioskeletal syndrome boronne type, dextrocardia with situs inversus, diabetes persistent mullerian ducts, diaphragmatic agenesis radial aplasia omphalocele, diaphragmatic hernia exomphalos corpus callosum agenesis, diaphragmatic hernia upper limb defects, die Smulders droog van dijk syndrome, diomedi bernardi placidi syndrome, diphallus rachischisis imperforate anus, distichiasis heart congenital anomalies, double discordia, double uterus-hemivagina-renal agenesis, Drachtman Weinblatt Sitarz syndrome, Duker-Weiss-Siber syndrome, duodenal atresia tetralogy of fallot, duplication of leg mirror foot, duplication of the thumb unilateral biphalangeal, dupont sellier chochillon syndrome, dwarfism bluish sclerae, dwarfism deafness retinitis pigmentosa, dwarfism lethal type advanced bone age, dwarfism thin bones multiple fractures, dysmorphism cleft palate loose skin, Eagle syndrome, ectrodactyly cardiopathy dysmorphism, Elliott ludman Teebi syndrome, enamel hypoplasia cataract hydrocephaly, encephalocele anencephaly, enchondromatosis dwarfism deafness, Engelhard Yatziv syndrome, enlarged vestibular aqueduct syndrome, epidermal nevus vitamin D resistant rickets, epimetaphyseal dysplasia cataract, epiphyseal dysplasia dysmorphism camptodactyly, esophageal atresia coloboma talipes, extrasystoles short stature hyperpigmentation microcephaly, facial clefting corpus callosum agenesis, facio digito genital syndrome recessive form, facio skeletal genital syndrome rippberger type, familial capillaro-venous leptomeningeal angiomatosis, Dursun syndrome, Faye-Petersen-Ward-Carey syndrome, feigenbaum Bergeron syndrome, Feingold trainer syndrome, fetal brain disruption sequence, fetal enterovirus syndrome, fetal parainfluenza virus type 3 syndrome, fetal phenothiazine syndrome, fibromatosis multiple non ossifying, fibula aplasia complex brachydactyly, fibular hypoplasia scapulo pelvic dysplasia absent, Fitz-Hugh-Curtis syndrome, focal alopecia congenital megalencephaly, focal or multifocal malformations in neuronal migration, foix chavany Marie syndrome, Fontaine farriaux blanckaert syndrome, Fraser Jequier Chen syndrome, Freiberg disease, Friedman Goodman syndrome, frontonasal malformation cloacal exstrophy, frontonasal dysplasia Klippel feil syndrome, frontonasal dysplasia phocomelic upper limbs, Fryns Fabry Remans syndrome, Fryns Smeets Thiry syndrome, Fuchs atrophia gyrata chorioideae et retinae, Fukuda-Miyanomae-Nakata syndrome, Fuqua Berkovitz syndrome, Garret-Tripp syndrome, gas bloat syndrome, Gaucher ichthyosis restrictive dermopathy, gershinibaruch Leibo syndrome, Ghose-Sachdev-Kumar syndrome, gigantism advanced bone age hoarse cry, glossopalatine ankylosis micrognathia ear anomalies, goldstein hutt syndrome, goniodysgenesis intellectual disability short stature, green sandford davison syndrome, grix Blankenship Peterson syndrome, Ho-Kaufman-McAlister syndrome, Jaffer-Beighton syndrome, Judge Misch wright syndrome, Kashani-Strom-Utley syndrome, Kasznica-Carlson-Coppedge syndrome, Katsantoni-Papadakou-Lagoyanni syndrome, Kocher-debre-Semelaigne syndrome, Koone-Rizzo-Elias syndrome, Kozlowski Brown Hardwick syndrome, Kozlowski Ouvrier syndrome, Kozlowski Rafinski Klicharska syndrome, Kozlowski Warren Fisher syndrome, Krauss Herman Holmes syndrome, Krieble Bixler syndrome, Kuster Majewski Hammerstein syndrome, Kuster syndrome, Laugier-Hunziker syndrome, Laurence-Prosser-Rocker syndrome, le Marec-Bracq-Picaud syndrome, levator syndrome, Marinesco-Sjogren-like syndrome, Milner-Khallouf-Gibson syndrome, radio-digito-facial dysplasia, Seckel like syndrome majoor-krakauer type, neonatal aspiration syndrome, muscular fibrosis multifocal obstructed vessels, short stature contractures hypotonia, Alice in Wonderland syndrome, megacystis-microcolon-intestinal hypoperistalsis syndrome, Basilicata-Akhtar syndrome, Liberfarb syndrome, craniofacial dysmorphism, skeletal anomalies, and impaired intellectual development syndrome, cardiac, facial, and digital anomalies with developmental delay, fibrosis, neurodegeneration, and cerebral angiomatosis, Duane anomaly-myopathy-scoliosis syndrome, congenital cataract-severe neonatal hepatopathy-global developmental delay syndrome, infantile hypotonia-oculomotor anomalies-hyperkinetic movements-developmental delay syndrome, acute radiation syndrome, monoclonal mast cell activation syndrome, oculocerebrodental syndrome, syndromic congenital sodium diarrhea, congenital brachyesophagus-intrathoracic stomach-vertebral anomalies syndrome, intellectual disability-cardiac anomalies-short stature-joint laxity syndrome, intrauterine growth restriction-congenital multiple café-au-lait macules-increased sister chromatid exchange syndrome, frontonasal dysplasia-bifid nose-upper limb anomalies syndrome, microcephaly-facial dysmorphism-ocular anomalies-multiple congenital anomalies syndrome, diaphragmatic hernia-short bowel-asplenia syndrome, warts-immunodeficiency-lymphedema-anogenital dysplasia syndrome, Cramp-fasciculation syndrome, choanal atresia-athelia-hypothyroidism-delayed puberty-short stature syndrome, blepharophimosis-intellectual disability syndrome/genitopatellar overlap syndrome, CCNK-related neurodevelopmental disorder-severe intellectual disability-facial dysmorphism syndrome, cerebellar hypoplasia-intellectual disability-congenital microcephaly-dystonia-anemia-growth retardation syndrome, KLHL7-related Bohring-Opitz-like syndrome, KLHL7-related cold-induced sweating-like syndrome, KAT6B-related multiple congenital anomalies syndrome, oculogastrointestinal-neurodevelopmental syndrome, spastic paraparesis-cataracts-speech delay syndrome, Ruzicka-Goerz-Anton syndrome, Sammartino-Decreccio syndrome, Samson-Gardner syndrome, Samson-Viljoen syndrome, Sanderson-Fraser syndrome, Sandhaus-Ben-Ami syndrome, prostatic malacoplakia associated with prostatic abscess, Saul-Wilkes-Stevenson syndrome, macrogyria, pseudobulbar palsy and intellectual disability, Schwartz-Cohen-addad-Lambert syndrome, Schlegelberger-Grote syndrome, Schrander-stumpel-Theunissen-Hulsmans syndrome, Saal-Bulas syndrome, Sackey-Sakati-Aur syndrome, Slti-Salem syndrome, Zerres Rietschel Majewski syndrome, Zazam Sheriff Phillips syndrome, Zadik-Barak-Levin syndrome, weinstein kliman scully syndrome, thickened earlobes with conductive deafness from incus-stapes abnormalities, ichthyosis linearis circumflexa, infantile striato thalamic degeneration, Landy-Donnai syndrome, merlob grunebaum reisner syndrome, Pavone Fiumara Rizzo syndrome, pfeiffer rockelein syndrome, Pfeiffer Tietze Welte syndrome, phosphoribosylpyrophosphate synthetase deficiency, piepkorn karp hickok syndrome, podder-tolmie syndrome, pointer syndrome, richieri-costa guion-almeida cohen syndrome, Rubinstein Taybi like syndrome, ruvalcaba churesigaew myhre syndrome, short limb dwarf lethal colavita kozlowski type, Mallory-Weiss syndrome, superior vena cava syndrome, piriformis syndrome, engraftment syndrome, Adams-Stokes syndrome, Leriche syndrome, multiple organ dysfunction syndrome, posterior leukoencephalopathy syndrome, cardio-renal syndrome, Rahman syndrome, X-linked keloid scarring-reduced joint mobility-increased optic cup-to-disc ratio syndrome, retinitis pigmentosa-hearing loss-premature aging-short stature-facial dysmorphism syndrome, congenital labioscrotal agenesis-cerebellar malformation-corneal dystrophy-facial dysmorphism syndrome, Lopes-Maciel-Rodan syndrome, Stankiewicz-Isidor syndrome, Skraban-Deardorff syndrome, joint laxity, short stature, and myopia, Sweeney-Cox syndrome, Alkuraya-Kucinskas syndrome, Jaberi-Elahi syndrome, neurodevelopmental disorder with regression, abnormal movements, loss of speech, and seizures, hearing impairment and infertile male syndrome, cardiocutaneous syndrome, neonatal diabetes, congenital sensorineural hearing loss and congenital cataracts, cardioectodermal syndrome, cannabinoid hyperemesis syndrome, retrograde cricopharyngeus dysfunction, Zinner syndrome, retinal dystrophy-ataxia-pituitary hormone abnormality-hypogonadism syndrome, IFAP syndrome, DICER1-related tumor predisposition, Roberts-SC phocomelia syndrome, carcinoid syndrome, Bonnevie-Ullrich syndrome, NKX2-1 related choreoathetosis and congenital hypothyroidism with or without pulmonary dysfunction, RNU4ATAC spectrum disorder, syndromic congenital heart disease, hand-foot syndrome, central hypoventilation syndrome, congenital, 1, with or without Hirschsprung disease, gastrointestinal defects and immunodeficiency syndrome 1, achalasia-alacrima syndrome, black locks with albinism and deafness syndrome, Birt-Hogg-Dube syndrome, trigeminal trophic syndrome, developmental and/or epileptic encephalopathy with spike-wave activation in sleep, syndromic microspherophakia, painful legs and moving toes syndrome, congenital aphakia-iris hypoplasia-microphthalmia-microcornea syndrome, hereditary persistence of fetal hemoglobin-intellectual disability syndrome, developmental delay-immunodeficiency-leukoencephalopathy-hypohomocysteinemia syndrome, primary hypomagnesemia-generalized seizures-intellectual disability-obesity syndrome, post-cardiac arrest syndrome, early-onset obesity-hyperphagia-severe developmental delay syndrome, hereditary alpha tryptasemia syndrome, KINSSHIP syndrome, developmental delay, hypotrophy, and dysmorphic features without moebius syndrome, breast implant illness, cataracts, hearing impairment, nephrotic syndrome, and enterocolitis, craniosynostosis-facial dysmorphism-chiari-1 malformation-developmental and language delay syndrome, MYT1L-related developmental delay-intellectual disability-obesity syndrome, Houge-Janssens syndrome, xerosis and growth failure with immune and pulmonary dysfunction syndrome, Fliedner-Zweier syndrome, Lui-Jee-Baron syndrome, Long-Olsen-Distelmaier syndrome, Tan-Almurshedi syndrome, diabetes, deafness, developmental delay, and short stature syndrome, Alfadhel syndrome, Hoxha-Aliu syndrome, cleft palate-congenital heart defect-intellectual disability syndrome, congenital insensitivity to pain syndrome, Marsili type, Yuksel-Vogel-Bauer syndrome, polydactyly-macrocephaly syndrome, pyoderma gangrenosum-acne-hidradenitis suppurativa-ankylosing spondylitis syndrome, psoriatic arthritis-pyoderma gangrenosum-acne-hidradenitis suppurativa syndrome, megalencephaly-polydactyly syndrome, Leigh syndrome, mitochondrial, auroneurodental syndrome, orofacial clefting-cardiac anomalies-facial dysmorphism syndrome, Grisel syndrome, arterial tortuosity-bone fragility syndrome, dialysis disequilibrium syndrome, brain abnormalities-severe developmental delay-facial dysmorphism-intellectual disability syndrome due to MEF2C mutation, Kariminejad neurodevelopmental syndrome, myelofibrosis, congenital, with anemia, neutropenia, developmental delay, and ocular abnormalities, brain malformation renal syndrome, myopathy with myalgia, increased serum creatine kinase, and with or without episodic rhabdomyolysis 2, Karayol-Borroto-Haghshenas neurodevelopmental syndrome, neurodegeneration, infantile-onset, with optic atrophy and brain abnormalities, Morimoto-Ryu-Malicdan neuromuscular syndrome, neurodevelopmental disorder with dysmorphic facies, absent speech and ambulation, and brain abnormalities, neurodevelopmental disorder with variable familial hypercholanemia, Pan-Chung-Bellen syndrome, telangiectasia, impaired intellectual development, microcephaly, metaphyseal dysplasia, eye abnormalities, and short stature, Muggenthaler-Chowdhury-Chioza syndrome, Tayoun-Maawali syndrome, ragopathy, cardiovascular-kidney-metabolic syndrome, craniofaciocardiohepatic syndrome, FICUS syndrome, Li-Takada-Miyake syndrome, Guillouet-Gordon syndrome, ICHAD syndrome, cataract, alopecia, oral mucosal disorder, and psoriasis-like syndrome, RAC2-related combined immunodeficiency-bronchiectasis-cancer-predisposing syndrome, oculovertebral syndrome, Alsahan-Harris syndrome, Ververi-Brady syndrome, Dursun-Ozgul neurodevelopmental syndrome, immune dysregulation, neurodevelopmental defects, and colitis, Harel-Tora neurodevelopmental syndrome, Valence-Farazi cerebellar ataxia syndrome, dyschromatosis, ichthyosis, deafness, and atopic disease, Ramond-Elliott neurodevelopmental syndrome, STAD syndrome, craniosynostosis-scoliosis syndrome, loin pain hematuria syndrome, IRF6-related condition, linkeropathy, NDUFB11-related disorders, CRYAB-related myofibrillar myopathy-cataract-cardiomyopathy spectrum disorder, TSEN2-related neurodevelopmental disorder with or without thrombotic microangiopathy, antiphospholipid syndrome

Subtypes (3): developmental and epileptic encephalopathy 94, developmental and epileptic encephalopathy, 31A, developmental and epileptic encephalopathy, 43

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

13 retrieved; paginated sample, class counts are floors:

5 uncertain significance, 3 pathogenic, 2 conflicting classifications of pathogenicity, 2 not provided, 1 pathogenic/likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
1705832NM_001271.4(CHD2):c.3199dup (p.Ile1067fs)CHD2Pathogeniccriteria provided, single submitter
1705841NM_004408.4(DNM1):c.590-2A>GDNM1Pathogeniccriteria provided, single submitter
205540NM_198904.4(GABRG2):c.406C>T (p.Arg136Ter)GABRG2Pathogeniccriteria provided, multiple submitters, no conflicts
205963NM_004519.4(KCNQ3):c.688C>T (p.Arg230Cys)KCNQ3Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
280448NM_001127222.2(CACNA1A):c.2127G>T (p.Leu709Phe)CACNA1AConflicting classifications of pathogenicitycriteria provided, conflicting classifications
206596NM_002693.3(POLG):c.1402A>G (p.Asn468Asp)POLGConflicting classifications of pathogenicitycriteria provided, conflicting classifications
560642NM_001482.3(GATM):c.367G>A (p.Ala123Thr)GATMUncertain significanceno assertion criteria provided
4070925NM_006597.6(HSPA8):c.1936_1938dup (p.Asp646_Ter647insAsp)HSPA8Uncertain significancecriteria provided, single submitter
1708030NM_001040142.2(SCN2A):c.269C>T (p.Thr90Met)SCN2AUncertain significancecriteria provided, multiple submitters, no conflicts
2444809NM_001040142.2(SCN2A):c.208C>G (p.Pro70Ala)SCN2AUncertain significancecriteria provided, single submitter
534635NM_014795.4(ZEB2):c.3267A>C (p.Glu1089Asp)ZEB2Uncertain significancecriteria provided, single submitter
2505076NM_000834.5(GRIN2B):c.2438T>C (p.Leu813Pro)GRIN2Bnot providedno classification provided
2505111NM_001040142.2(SCN2A):c.3671_3674dup (p.Ala1226fs)SCN2Anot providedno classification provided

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 70 · Orphanet: 53 · 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
CACNA1ASupportiveAutosomal dominantLennox-Gastaut syndrome23
CHD2SupportiveAutosomal dominantLennox-Gastaut syndrome4
CUX2SupportiveAutosomal dominantLennox-Gastaut syndrome4
DNM1SupportiveAutosomal dominantLennox-Gastaut syndrome10
GABRB3SupportiveAutosomal dominantLennox-Gastaut syndrome8
SCN1ASupportiveAutosomal dominantLennox-Gastaut syndrome20
MAPK10LimitedAutosomal dominantLennox-Gastaut syndrome

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
CACNA1AOrphanet:2131Alternating hemiplegia of childhood
CACNA1AOrphanet:2382Lennox-Gastaut syndrome
CACNA1AOrphanet:442835Non-specific early-onset epileptic encephalopathy
CACNA1AOrphanet:569Familial or sporadic hemiplegic migraine
CACNA1AOrphanet:71518Benign paroxysmal torticollis of infancy
CACNA1AOrphanet:97Familial paroxysmal ataxia
CACNA1AOrphanet:98758Spinocerebellar ataxia type 6
CHD2Orphanet:1942Epilepsy with myoclonic-atonic seizures
CHD2Orphanet:2382Lennox-Gastaut syndrome
DNM1Orphanet:2382Lennox-Gastaut syndrome
DNM1Orphanet:442835Non-specific early-onset epileptic encephalopathy
SCN1AOrphanet:1942Epilepsy with myoclonic-atonic seizures
SCN1AOrphanet:2382Lennox-Gastaut syndrome
SCN1AOrphanet:293181Epilepsy of infancy with migrating focal seizures
SCN1AOrphanet:33069Dravet syndrome
SCN1AOrphanet:36387Genetic epilepsy with febrile seizure plus
SCN1AOrphanet:442835Non-specific early-onset epileptic encephalopathy
SCN1AOrphanet:569Familial or sporadic hemiplegic migraine
CUX2Orphanet:2382Lennox-Gastaut syndrome
GABRB3Orphanet:2382Lennox-Gastaut syndrome
GABRB3Orphanet:64280Childhood absence epilepsy
MAPK10Orphanet:2382Lennox-Gastaut 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
ZEB2Orphanet:261537Mowat-Wilson syndrome due to monosomy 2q22
ZEB2Orphanet:261552Mowat-Wilson syndrome due to a ZEB2 point mutation
ZEB2Orphanet:626Large/giant congenital melanocytic nevus
GABRG2Orphanet:1945Self-limited epilepsy with centrotemporal spikes
GABRG2Orphanet:33069Dravet syndrome
GABRG2Orphanet:36387Genetic epilepsy with febrile seizure plus
GABRG2Orphanet:442835Non-specific early-onset epileptic encephalopathy
GABRG2Orphanet:64280Childhood absence epilepsy
GATMOrphanet:3337Primary Fanconi renotubular syndrome
GATMOrphanet:35704L-Arginine:glycine amidinotransferase deficiency
GRIN2BOrphanet:589547GRIN2B-related developmental delay, intellectual disability and autism spectrum disorder
GRIN2BOrphanet:697160Infantile epileptic spasms syndrome
KCNQ3Orphanet:1949Self-limited neonatal epilepsy
KCNQ3Orphanet:306Self-limited infantile epilepsy
KCNQ3Orphanet:307Juvenile myoclonic epilepsy
POLGOrphanet:254881Spinocerebellar ataxia with epilepsy
POLGOrphanet:254886Autosomal recessive progressive external ophthalmoplegia
POLGOrphanet:254892Autosomal dominant progressive external ophthalmoplegia
POLGOrphanet:298Mitochondrial neurogastrointestinal encephalomyopathy
POLGOrphanet:402082Progressive myoclonic epilepsy type 5

Cohort genes → proteins

15 cohort genes, 15 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence15

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
CACNA1AHGNC:1388ENSG00000141837O00555Voltage-dependent P/Q-type calcium channel subunit alpha-1Agencc,clinvar
CHD2HGNC:1917ENSG00000173575O14647ATP-dependent chromatin remodeler CHD2gencc,clinvar
DNM1HGNC:2972ENSG00000106976Q05193Dynamin-1gencc,clinvar
SCN1AHGNC:10585ENSG00000144285P35498Sodium channel protein type 1 subunit alphagencc
CUX2HGNC:19347ENSG00000111249O14529Homeobox protein cut-like 2gencc
GABRB3HGNC:4083ENSG00000166206P28472Gamma-aminobutyric acid receptor subunit beta-3gencc
MAPK10HGNC:6872ENSG00000109339P53779Mitogen-activated protein kinase 10gencc
SCN2AHGNC:10588ENSG00000136531Q99250Sodium channel protein type 2 subunit alphaclinvar
ZEB2HGNC:14881ENSG00000169554O60315Zinc finger E-box-binding homeobox 2clinvar
GABRG2HGNC:4087ENSG00000113327P18507Gamma-aminobutyric acid receptor subunit gamma-2clinvar
GATMHGNC:4175ENSG00000171766P50440Glycine amidinotransferase, mitochondrialclinvar
GRIN2BHGNC:4586ENSG00000273079Q13224Glutamate receptor ionotropic, NMDA 2Bclinvar
HSPA8HGNC:5241ENSG00000109971P11142Heat shock cognate 71 kDa proteinclinvar
KCNQ3HGNC:6297ENSG00000184156O43525Potassium voltage-gated channel subfamily KQT member 3clinvar
POLGHGNC:9179ENSG00000140521P54098DNA polymerase subunit gamma-1clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
CACNA1AVoltage-dependent P/Q-type calcium channel subunit alpha-1AVoltage-sensitive calcium channels (VSCC) mediate the entry of calcium ions into excitable cells and are also involved in a variety of calcium-dependent processes, including muscle contraction, hormone or neurotransmitter release, gene exp…
CHD2ATP-dependent chromatin remodeler CHD2ATP-dependent chromatin-remodeling factor that specifically binds to the promoter of target genes, leading to chromatin remodeling, possibly by promoting deposition of histone H3.3.
DNM1Dynamin-1Catalyzes the hydrolysis of GTP and utilizes this energy to mediate vesicle scission and participates in many forms of endocytosis, such as clathrin-mediated endocytosis or synaptic vesicle endocytosis as well as rapid endocytosis (RE).
SCN1ASodium channel protein type 1 subunit alphaPore-forming subunit of Nav1.1, a voltage-gated sodium (Nav) channel that directly mediates the depolarizing phase of action potentials in excitable membranes.
CUX2Homeobox protein cut-like 2Transcription factor involved in the control of neuronal proliferation and differentiation in the brain.
GABRB3Gamma-aminobutyric acid receptor subunit beta-3Beta subunit of the heteropentameric ligand-gated chloride channel gated by gamma-aminobutyric acid (GABA), a major inhibitory neurotransmitter in the brain.
MAPK10Mitogen-activated protein kinase 10Serine/threonine-protein kinase involved in various processes such as neuronal proliferation, differentiation, migration and programmed cell death.
SCN2ASodium channel protein type 2 subunit alphaMediates the voltage-dependent sodium ion permeability of excitable membranes.
ZEB2Zinc finger E-box-binding homeobox 2Transcriptional inhibitor that binds to DNA sequence 5’-CACCT-3’ in different promoters.
GABRG2Gamma-aminobutyric acid receptor subunit gamma-2Gamma subunit of the heteropentameric ligand-gated chloride channel gated by gamma-aminobutyric acid (GABA), a major inhibitory neurotransmitter in the brain.
GATMGlycine amidinotransferase, mitochondrialTransamidinase that catalyzes the transfer of the amidino group of L-arginine onto the amino moiety of acceptor metabolites such as glycine, beta-alanine, gamma-aminobutyric acid (GABA) and taurine yielding the corresponding guanidine deri…
GRIN2BGlutamate receptor ionotropic, NMDA 2BComponent of N-methyl-D-aspartate (NMDA) receptors (NMDARs) that function as heterotetrameric, ligand-gated cation channels with high calcium permeability and voltage-dependent block by Mg(2+).
HSPA8Heat shock cognate 71 kDa proteinMolecular chaperone implicated in a wide variety of cellular processes, including protection of the proteome from stress, folding and transport of newly synthesized polypeptides, chaperone-mediated autophagy, activation of proteolysis of m…
KCNQ3Potassium voltage-gated channel subfamily KQT member 3Pore-forming subunit of the voltage-gated potassium (Kv) M-channel which is responsible for the M-current, a key controller of neuronal excitability.
POLGDNA polymerase subunit gamma-1Catalytic subunit of DNA polymerase gamma solely responsible for replication of mitochondrial DNA (mtDNA).

Protein-family classification

Druggable: 7 · Difficult: 3 · Unknown: 5 · Druggable fraction: 0.47

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
Ion channel429.7×5e-05
Kinase11.9×0.709
Enzyme (other)21.6×0.709
Scaffold/PPI11.1×0.709
Transcription factor21.1×0.709
Other/Unknown50.6×0.978

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
CACNA1AIon channelyesVDCCAlpha1, CACNA1A, Ion_trans_dom
CHD2Other/UnknownnoSNF2_N, Chromo/chromo_shadow_dom, Helicase_C-like
DNM1Scaffold/PPIno3.6.5.5Dynamin_stalk, Dynamin_GTPase, PH_domain
SCN1AIon channelyesNa_channel_asu, Ion_trans_dom, Na_channel_a1su
CUX2Transcription factornoHD, CUT_dom, Homeodomain-like_sf
GABRB3Other/UnknownnoGABAAb_rcpt, GABAA/Glycine_rcpt, Neurotrans-gated_channel_TM
MAPK10Kinaseyes2.7.11.24Prot_kinase_dom, MAP_kinase_CS, Ser/Thr_kinase_AS
SCN2AIon channelyesIQ_motif_EF-hand-BS, Na_channel_asu, Ion_trans_dom
ZEB2Transcription factornoHD, Di19_Zn-bd, Homeodomain-like_sf
GABRG2Other/UnknownnoGABRG-1/4, GABBAg2_rcpt, GABAA/Glycine_rcpt
GATMEnzyme (other)yes2.1.4.1AmidinoTrfase
GRIN2BOther/UnknownnoIontro_rcpt_C, Iono_Glu_rcpt_met, ANF_lig-bd_rcpt
HSPA8Enzyme (other)yes7.4.2.3Hsp_70_fam, Heat_shock_70_CS, HSP70_peptide-bd_sf
KCNQ3Ion channelyesK_chnl_volt-dep_KCNQ, K_chnl_volt-dep_KCNQ3, Ion_trans_dom
POLGOther/UnknownnoDNA-dir_DNA_pol_A_palm_dom, DNA-dir_DNA_pol_A_mt, RNaseH-like_sf

Expression context

Cohort genes with no expression data: 0.

15 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)15
unknown0

Top tissues across cohort

TissueCohort genes
Brodmann (1909) area 237
middle temporal gyrus4
right hemisphere of cerebellum3
cortical plate3
cerebellar cortex2
cerebellar hemisphere2
sural nerve2
lateral nuclear group of thalamus2
buccal mucosa cell2
superior frontal gyrus2
calcaneal tendon1
colonic epithelium1
primary visual cortex1
adrenal tissue1
prefrontal cortex1
right frontal lobe1
cerebellar vermis1
monocyte1
adult organism1
body of pancreas1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
CACNA1A237broadmarkercerebellar hemisphere, right hemisphere of cerebellum, cerebellar cortex
CHD2268ubiquitousmarkercalcaneal tendon, sural nerve, colonic epithelium
DNM1257ubiquitousmarkerright hemisphere of cerebellum, cerebellar hemisphere, cerebellar cortex
SCN1A154tissue_specificmarkerBrodmann (1909) area 23, lateral nuclear group of thalamus, primary visual cortex
CUX2144broadmarkermiddle temporal gyrus, buccal mucosa cell, Brodmann (1909) area 23
GABRB3219broadmarkermiddle temporal gyrus, cortical plate, Brodmann (1909) area 23
MAPK10217ubiquitousmarkeradrenal tissue, prefrontal cortex, right frontal lobe
SCN2A187broadmarkermiddle temporal gyrus, Brodmann (1909) area 23, cerebellar vermis
ZEB2290ubiquitousmarkercortical plate, sural nerve, monocyte
GABRG2174tissue_specificmarkermiddle temporal gyrus, Brodmann (1909) area 23, superior frontal gyrus
GATM289ubiquitousmarkerbody of pancreas, adult organism, right lobe of liver
GRIN2B138broadmarkerbuccal mucosa cell, cortical plate, Brodmann (1909) area 23
HSPA8156ubiquitousmarkercorpus callosum, superior frontal gyrus, right hemisphere of cerebellum
KCNQ3180broadmarkerganglionic eminence, lateral nuclear group of thalamus, Brodmann (1909) area 23
POLG295ubiquitousmarkergranulocyte, small intestine Peyer’s patch, tibial nerve

Protein interactions among cohort

Intra-cohort edges: 7.

Hub genes (top 10 by interactor count)

SymbolInteractor count
HSPA812,858
DNM13,614
GRIN2B3,611
POLG3,400
ZEB23,193
SCN2A2,810
KCNQ32,665
GATM2,658
CHD22,392
GABRG22,392

Intra-cohort edges

ABSources
GABRB3GABRG2intact
GABRB3SCN1Astring_interaction
GABRG2SCN1Astring_interaction
GABRG2SCN2Astring_interaction
KCNQ3SCN1Astring_interaction
KCNQ3SCN2Astring_interaction
SCN1ASCN2Abiogrid_interaction, string_interaction

Structural data

PDB: 14 · AlphaFold-only: 1 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
GABRB3P2847295
GABRG2P1850775
MAPK10P5377965
HSPA8P1114238
GRIN2BQ1322436
POLGP5409836
DNM1Q0519322
KCNQ3O4352516
GATMP5044011
SCN2AQ992505
CACNA1AO005554
CUX2O145293
SCN1AP354981
ZEB2O603151

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
CHD2O1464760.81

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 98. Enrichment computed across 15 evidence-associated genes (14 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 14 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Interaction between L1 and Ankyrins378.9×6e-04SCN1A, SCN2A, KCNQ3
L1CAM interactions325.8×0.009SCN1A, SCN2A, KCNQ3
Respiratory syncytial virus genome transcription1815.7×0.020HSPA8
Phase 0 - rapid depolarisation249.4×0.020SCN1A, SCN2A
GABA receptor activation245.3×0.020GABRB3, GABRG2
Signaling by ERBB4238.8×0.020GABRB3, GABRG2
Axon guidance39.7×0.046SCN1A, SCN2A, KCNQ3
Nervous system development39.2×0.046SCN1A, SCN2A, KCNQ3
Toll Like Receptor 4 (TLR4) Cascade218.8×0.054DNM1, MAPK10
Activated NTRK2 signals through FYN1135.9×0.057GRIN2B
CHL1 interactions190.6×0.057HSPA8
Lipophagy190.6×0.057HSPA8
Activation of the AP-1 family of transcription factors181.6×0.057MAPK10
Formation of the posterior neural plate181.6×0.057ZEB2
Strand-asynchronous mitochondrial DNA replication181.6×0.057POLG
Formation of annular gap junctions174.2×0.057DNM1
Creatine metabolism174.2×0.057GATM
Regulation of CDH11 gene transcription174.2×0.057ZEB2
Formation of the anterior neural plate174.2×0.057ZEB2
Presynaptic depolarization and calcium channel opening168.0×0.057CACNA1A
Gap junction degradation168.0×0.057DNM1
Positive Regulation of CDH1 Gene Transcription168.0×0.057ZEB2
Cardiac conduction215.5×0.057SCN1A, SCN2A
Clathrin-mediated endocytosis212.2×0.057DNM1, HSPA8
Muscle contraction211.0×0.057SCN1A, SCN2A
Retrograde neurotrophin signalling158.3×0.064DNM1
Protein methylation148.0×0.073HSPA8
GABA synthesis, release, reuptake and degradation145.3×0.073HSPA8
MECP2 regulates neuronal receptors and channels142.9×0.073GRIN2B
Ras activation upon Ca2+ influx through NMDA receptor140.8×0.073GRIN2B

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 15 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
neuronal action potential396.3×6e-04SCN1A, SCN2A, KCNQ3
cellular response to histamine2374.5×1e-03GABRB3, GABRG2
excitatory chemical synaptic transmission2172.8×0.003GRIN2B, KCNQ3
nerve development2124.8×0.005SCN1A, KCNQ3
cardiac muscle cell action potential involved in contraction293.6×0.007SCN1A, SCN2A
inhibitory synapse assembly283.2×0.008GABRB3, GABRG2
reproductive behavior11123.5×0.008GABRB3
circadian sleep/wake cycle, REM sleep11123.5×0.008GABRB3
mammillary axonal complex development11123.5×0.008ZEB2
apoptosome assembly11123.5×0.008KCNQ3
clathrin coat assembly involved in endocytosis11123.5×0.008DNM1
regulation of action potential firing threshold11123.5×0.008KCNQ3
positive regulation of myofibroblast contraction11123.5×0.008ZEB2
regulation of protein import11123.5×0.008HSPA8
chaperone-mediated autophagy translocation complex disassembly11123.5×0.008HSPA8
regulation of blood-brain barrier permeability11123.5×0.008ZEB2
gamma-aminobutyric acid signaling pathway272.5×0.008GABRB3, GABRG2
positive regulation of excitatory postsynaptic potential270.2×0.008CUX2, GRIN2B
synaptic transmission, GABAergic266.1×0.008GABRB3, GABRG2
chemical synaptic transmission315.5×0.008CACNA1A, GRIN2B, KCNQ3
creatine biosynthetic process1561.7×0.009GATM
intrinsic apoptotic signaling pathway in response to osmotic stress1561.7×0.009SCN2A
muscle atrophy1561.7×0.009GATM
vesicle scission1561.7×0.009DNM1
positive regulation of lens fiber cell differentiation1561.7×0.009ZEB2
negative regulation of supramolecular fiber organization1561.7×0.009HSPA8
regulation of melanosome organization1561.7×0.009ZEB2
substantia propria of cornea development1561.7×0.009KCNQ3
regulation of myofibroblast cell apoptotic process1561.7×0.009ZEB2
sodium ion transport236.2×0.009SCN1A, SCN2A

Therapeutics

Drugs indicated or in trials for this disease

5 approved drugs — disease-direct ChEMBL indications, not inferred from the associated-gene cohort below.

DrugStatus
CannabidiolApproved (phase 4)
ClobazamApproved (phase 4)
LamotrigineApproved (phase 4)
RufinamideApproved (phase 4)
TopiramateApproved (phase 4)

3 drugs in clinical trials for this disease (phase 2–3, investigational): efficacy not established — a trial record, not an indication.

DrugHighest phase
FenfluraminePhase 3
PerampanelPhase 3
SoticlestatPhase 3

Drug target analysis

Approved (phase 4): 11 · Phase ≥3: 11 · Phased (≥1): 11 · Undrugged: 4

Druggability breadth: 11 of 15 evidence-associated genes (73%) 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
CACNA1ANIMODIPINE
DNM1CETRIMIDE
SCN1AMEXILETINE HYDROCHLORIDE
GABRB3LINDANE
MAPK10MOMELOTINIB
SCN2ABEPRIDIL
GABRG2ENZALUTAMIDE
GRIN2BHALOPERIDOL
HSPA8HEXACHLOROPHENE
KCNQ3FLUPIRTINE
POLGADEFOVIR DIPIVOXIL

Top cohort targets by molecule count

SymbolMoleculesMax phase
SCN2A994
SCN1A944
MAPK10584
GABRG2554
GRIN2B354
GABRB3324
DNM144
HSPA844
KCNQ334
CACNA1A24

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
NIMODIPINE4CACNA1A
TACRINE4CACNA1A, GRIN2B
CETRIMIDE4DNM1
MEXILETINE HYDROCHLORIDE4SCN1A
BEPRIDIL4SCN1A, SCN2A
DIBUCAINE4SCN1A, SCN2A
ARTICAINE4SCN1A, SCN2A
BUPIVACAINE4SCN1A, SCN2A
IMIPRAMINE4SCN1A, SCN2A
DROPERIDOL4SCN1A, SCN2A
DICYCLOMINE4SCN1A, SCN2A
TETRABENAZINE4SCN1A, SCN2A
PHENIRAMINE4SCN1A, SCN2A
PRILOCAINE4SCN1A, SCN2A
PROPOXYCAINE4SCN1A, SCN2A
PROPARACAINE4SCN1A, SCN2A
HEXYLCAINE4SCN1A, SCN2A
PRAMOXINE4SCN1A, SCN2A
BENOXINATE4SCN1A, SCN2A
QUINIDINE4SCN1A, SCN2A
FELODIPINE4SCN1A, SCN2A
PHENYTOIN4SCN1A, SCN2A
QUININE4SCN1A, SCN2A
NISOLDIPINE4SCN1A, SCN2A
NIFEDIPINE4SCN1A, SCN2A
PRAZOSIN4SCN1A, SCN2A
DILTIAZEM4SCN1A, SCN2A
PRENYLAMINE4SCN1A, SCN2A
COCAINE4SCN1A, SCN2A
TRIFLUOPERAZINE4SCN1A, SCN2A

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 4.

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

SymbolAssaysType breakdown
GABRG21,155Binding:940, Functional:201, ADMET:10, Toxicity:4
MAPK10900Binding:892, Functional:7, ADMET:1
GABRB3887Binding:722, Functional:156, ADMET:6, Toxicity:3
GRIN2B471Binding:429, Functional:36, ADMET:5, Toxicity:1
SCN2A203Binding:172, Functional:20, ADMET:10, Toxicity:1
SCN1A149Binding:115, Functional:18, ADMET:14, Toxicity:2
KCNQ397Binding:91, Functional:4, ADMET:1, Toxicity:1
HSPA844Binding:44
POLG33Binding:30, ADMET:2, Functional:1
CACNA1A19Binding:18, Functional:1
DNM111Binding:11

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
DNM13.6.5.5dynamin GTPase
MAPK102.7.11.24mitogen-activated protein kinase
GATM2.1.4.1glycine amidinotransferase
HSPA87.4.2.3mitochondrial protein-transporting ATPase

Cohort genes with high screening signal

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

SymbolChEMBL assays
SCN1A149
GABRB3887
MAPK10900
SCN2A203
GABRG21,155
GRIN2B471

Pharmacogenomics

Cohort genes with a PharmGKB record: 15; 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)
NIMODIPINE4CACNA1A
TACRINE4CACNA1A, GRIN2B
CETRIMIDE4DNM1
MEXILETINE HYDROCHLORIDE4SCN1A
BEPRIDIL4SCN1A, SCN2A
DIBUCAINE4SCN1A, SCN2A
ARTICAINE4SCN1A, SCN2A
BUPIVACAINE4SCN1A, SCN2A
IMIPRAMINE4SCN1A, SCN2A
DROPERIDOL4SCN1A, SCN2A
DICYCLOMINE4SCN1A, SCN2A
TETRABENAZINE4SCN1A, SCN2A
PHENIRAMINE4SCN1A, SCN2A
PRILOCAINE4SCN1A, SCN2A
PROPOXYCAINE4SCN1A, SCN2A
PROPARACAINE4SCN1A, SCN2A
HEXYLCAINE4SCN1A, SCN2A
PRAMOXINE4SCN1A, SCN2A
BENOXINATE4SCN1A, SCN2A
QUINIDINE4SCN1A, SCN2A
FELODIPINE4SCN1A, SCN2A
PHENYTOIN4SCN1A, SCN2A
QUININE4SCN1A, SCN2A
NISOLDIPINE4SCN1A, SCN2A
NIFEDIPINE4SCN1A, SCN2A
PRAZOSIN4SCN1A, SCN2A
DILTIAZEM4SCN1A, SCN2A
PRENYLAMINE4SCN1A, SCN2A
COCAINE4SCN1A, SCN2A
TRIFLUOPERAZINE4SCN1A, SCN2A

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)11CACNA1A, DNM1, SCN1A, GABRB3, MAPK10, SCN2A, GABRG2, GRIN2B, HSPA8, KCNQ3 (+1 more)
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug1GATM
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug3CHD2, CUX2, ZEB2

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
CHD20
CUX20
ZEB20
GATM0

Clinical trials & evidence

Clinical trials

Clinical trials: 55.

Phase distribution (across all retrieved trials)

PhaseTrials
Not specified19
PHASE318
PHASE26
PHASE45
PHASE1/PHASE23
PHASE13
EARLY_PHASE11

Top trials by phase / activity

NCTPhaseStatusTitle
NCT05044819PHASE4ACTIVE_NOT_RECRUITINGAssessment of Potential for Chronic Liver Injury in Participants Treated With Epidiolex (Cannabidiol) Oral Solution
NCT06924827PHASE4NOT_YET_RECRUITINGA Study to Investigate the Transition of Children From ‘Artisanal Cannabidiol (CBD) to Epidiolex
NCT01370486PHASE4WITHDRAWNMelatonin Versus Placebo in the Lennox-Gastaut Syndrome: Neurophysiological and Neuropsychological Effects
NCT02731300PHASE4COMPLETEDTranscranial Direct Current Stimulation, Treatment of Childhood Drug-Resistant Lennox-Gastaut Syndrome, A Pilot Study
NCT04133480PHASE4WITHDRAWNInvestigation of Cognitive Outcomes With Cannabidiol Oral Solution
NCT05066217PHASE3RECRUITINGAn Efficacy and Safety Study of Clemizole HCl in Patients With Lennox-Gastaut Syndrome
NCT05219617PHASE3RECRUITINGInvestigate Efficacy and Safety of Carisbamate as Adjunctive Treatment for Seizures Associated With LGS in Children and Adults
NCT00004776PHASE3COMPLETEDPhase III Randomized, Double-Blind, Placebo-Controlled Study of Oral Topiramate for Lennox-Gastaut Syndrome
NCT01146951PHASE3COMPLETEDA Placebo-Controlled, Double-Blind Comparative Study of E2080 in Lennox-Gastaut Syndrome Patients (Study E2080-J081-304)
NCT01151540PHASE3COMPLETEDA Long Term Extension Study of E2080 in Lennox-Gastaut Patients
NCT01160770PHASE3COMPLETEDSafety and Effectiveness of Open-Label Clobazam in Subjects With Lennox-Gastaut Syndrome
NCT01405053PHASE3COMPLETEDStudy of Rufinamide in Pediatric Subjects 1 to Less Than 4 Years of Age With Lennox-Gastaut Syndrome Inadequately Controlled With Other Anti-epileptic Drugs
NCT02224560PHASE3COMPLETEDEfficacy and Safety of GWP42003-P for Seizures Associated With Lennox-Gastaut Syndrome in Children and Adults
NCT02224573PHASE3COMPLETEDAn Open Label Extension Study of Cannabidiol (GWP42003-P) in Children and Adults With Dravet or Lennox-Gastaut Syndromes
NCT02224690PHASE3COMPLETEDA Study to Investigate the Efficacy and Safety of Cannabidiol (GWP42003-P; CBD) as Adjunctive Treatment for Seizures Associated With Lennox-Gastaut Syndrome in Children and Adults
NCT02318537PHASE3WITHDRAWNCannabidiol Oral Solution as an Adjunctive Therapy for Treatment of Participants With Inadequately Controlled Lennox-Gastaut Syndrome
NCT02834793PHASE3TERMINATEDStudy of Perampanel as Adjunctive Treatment for Inadequately Controlled Seizures Associated With Lennox-Gastaut Syndrome
NCT03355209PHASE3COMPLETEDA Study to Investigate the Efficacy and Safety of ZX008 (Fenfluramine Hydrochloride) as an Adjunctive Therapy in Children and Adults With Lennox-Gastaut Syndrome
NCT03936777PHASE3COMPLETEDA Study to Investigate the Long-Term Safety of ZX008 (Fenfluramine Hydrochloride) Oral Solution in Children and Adults With Epileptic Encephalopathy Including Dravet Syndrome and Lennox-Gastaut Syndrome
NCT04611438PHASE3UNKNOWNResearch on Cognitive Effect of Cannabidiol on Dravet Syndrome and Lennox-Gastaut SyndromeGastaut Syndrome
NCT04938427PHASE3COMPLETEDA Study of Soticlestat as an Add-on Therapy in Children, Teenagers, and Adults With Lennox-Gastaut Syndrome
NCT05163314PHASE3TERMINATEDA Study of Soticlestat as an Add-on Therapy in Children and Adults With Dravet Syndrome or Lennox-Gastaut Syndrome
NCT06422377PHASE3TERMINATEDA Study Evaluating Soticlestat in Participants With Dravet Syndrome or Lennox-Gastaut Syndrome Who Have Been Exposed to Fenfluramine
NCT05339126PHASE2ACTIVE_NOT_RECRUITINGRNS System LGS Feasibility Study
NCT06401538PHASE2RECRUITINGBMB-101 in Absence Epilepsy and DEE
NCT06738732PHASE1/PHASE2NOT_YET_RECRUITINGCBD Delivery with the A-Synaptic GT4 Transdermal Delivery System in with Dravet Syndrome And/or Lennox-Gastaut Syndrome
NCT02655198PHASE2UNKNOWNAdd-on Therapy With Low Dose Fenfluramine in Lennox Gastaut Epilepsy
NCT02815540PHASE1/PHASE2TERMINATEDThe Effects of Cannabidiol (CBD) on Electrical and Autonomic Cardiac Function in Children With Severe Epilepsy
NCT03635073PHASE2TERMINATEDA Study of Soticlestat in Adults and Children With Rare Epilepsies
NCT03650452PHASE2COMPLETEDA Phase 2, Multicenter, Randomized, Double-blind, Placebo-controlled Study to Evaluate the Efficacy, Safety, and Tolerability of TAK-935 (OV935) as an Adjunctive Therapy in Pediatric Participants With Developmental and/or Epileptic Encephalopathies
NCT05364021PHASE1/PHASE2COMPLETEDStudy to Investigate LP352 in Subjects With Developmental and Epileptic Encephalopathies
NCT05626634PHASE2COMPLETEDOpen-label, Long-term Safety Study of LP352 in Subjects With Developmental and Epileptic Encephalopathy
NCT03467113PHASE1COMPLETEDA Study to Assess the Safety and Tolerability of ZX008 in Children and Young Adults With Dravet Syndrome or Lennox Gastaut Syndrome Currently Taking Cannabidiol
NCT03731715PHASE1COMPLETEDCarisbamate in Adult & Pediatric Subjects With Lennox-Gastaut Syndrome
NCT04062981PHASE1COMPLETEDCarisbamate Safety Study in Adult and Pediatric Subjects With Lennox-Gastaut Syndrome
NCT02632149EARLY_PHASE1UNKNOWNTrial to Assess Vagus Nerve Stimulation Therapy in Children With Lennox-Gastaut Syndrome
NCT03778424Not specifiedAVAILABLEAn Extended Access Program (EAP) for Participants Who Have Completed Rufinamide Study E2080-G000-303
NCT05374824Not specifiedRECRUITINGComparative Effectiveness of Palliative Surgery Versus Additional Anti-Seizure Medications for Lennox-Gastaut Syndrome
NCT05485831Not specifiedRECRUITINGEpidyolex® in Lennox Gastaut, Dravet Syndrome and Tuberous Sclerosis Complex: an Observational Study in ITALY
NCT06149663Not specifiedAVAILABLEIntermediate-Size Expanded Access Protocol (EAP) for LP352

Drugs tested across these trials (top 30)

MoleculeMax phaseTrials referencing
CANNABIDIOL49
RUFINAMIDE45
FENFLURAMINE44
PERAMPANEL42
CLOBAZAM41
MELATONIN41
TOPIRAMATE41
SOTICLESTAT35
CARISBAMATE33
BEXICASERIN21
CLEMIZOLE21