Craniosynostosis 7

disease
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Also known as craniosynostosis 7, susceptibility toCRS7

Summary

Craniosynostosis 7 (MONDO:0044315) is a disease caused by SMAD6 (GenCC Strong), with 3 cohort genes.

At a glance

  • Causal gene: SMAD6 (GenCC Strong)
  • Cohort genes: 3
  • ClinVar variants: 28

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namecraniosynostosis 7
Mondo IDMONDO:0044315
OMIM617439
DOIDDOID:0060912
UMLSC4479496
MedGen1392447
Is cancer (heuristic)no

Also known as: craniosynostosis 7 · craniosynostosis 7, susceptibility to · CRS7

Data availability: 28 ClinVar variants · 4 GenCC gene-disease records.

Disease family

Classification path: disease susceptibility › inherited disease susceptibilitycraniosynostosis 7

Related subtypes (283): microvascular complications of diabetes, susceptibility, Mycobacterium tuberculosis, susceptibility, bacteremia, susceptibility, pregnancy loss, recurrent, susceptibility, autoimmune thyroid disease, susceptibility to, susceptibility to HIV infection, appendicitis, proneness to, atherosclerosis susceptibility, multiple sclerosis, susceptibility to, dyslexia, susceptibility to, 1, endometriosis, susceptibility to, 1, osteoarthritis susceptibility 2, essential hypertension, genetic, osteoarthritis susceptibility 1, otitis media, susceptibility to, pelvic organ prolapse, susceptibility to, pulmonary edema of mountaineers, susceptibility to, sarcoidosis, susceptibility to, 1, strabismus, susceptibility to, thyrotoxic periodic paralysis, susceptibility to, 1, predisposition to invasive fungal disease due to CARD9 deficiency, celiac disease, susceptibility to, 1, hemolytic uremic syndrome, atypical, susceptibility to, 1, hypervitaminosis a, susceptibility to, kuru, susceptibility to, leprosy, susceptibility to, 3, nonarteritic anterior ischemic optic neuropathy, susceptibility to, diabetes mellitus, insulin-dependent, X-linked, susceptibility to, coronary heart disease, susceptibility to, 3, dyslexia, susceptibility to, 9, systemic lupus erythematosus, susceptibility to, 15, Leber optic atrophy, susceptibility to, dyslexia, susceptibility to, 2, Helicobacter pylori infection, susceptibility to, epilepsy, idiopathic generalized, susceptibility to, 1, inherited susceptibility to asthma, renal dysplasia, cystic, susceptibility to, systemic lupus erythematosus, susceptibility to, 1, leishmaniasis, tegumentary, susceptibility to, xanthomatosis, susceptibility to, hyperlipidemia, combined, 1, urinary tract infections, recurrent, susceptibility to, hypertension, essential, susceptibility to, 1, hepatic fibrosis, severe, susceptibility to, due to Schistosoma mansoni infection, dyslexia, susceptibility to, 3, Ascaris lumbricoides infection, susceptibility to, hypertension, essential, susceptibility to, 2, epilepsy, idiopathic generalized, susceptibility to, 7, systemic lupus erythematosus, susceptibility to, 2, systemic lupus erythematosus, susceptibility to, 3, nephrolithiasis, uric acid, susceptibility to, atrioventricular septal defect, susceptibility to, 2, vitiligo-associated multiple autoimmune disease susceptibility 1, polysubstance abuse, susceptibility to, dyslexia, susceptibility to, 6, glaucoma, normal tension, susceptibility to, anorexia nervosa, susceptibility to, 1, stroke, susceptibility to, 1, dyslexia, susceptibility to, 5, epilepsy, idiopathic generalized, susceptibility to, 2, familial meningioma, celiac disease, susceptibility to, 5, systemic lupus erythematosus with hemolytic anemia, susceptibility to, 1, hypertension, essential, susceptibility to, 3, coronary heart disease, susceptibility to, 1, bulimia nervosa, susceptibility to, 1, leprosy, susceptibility to, 2, epilepsy, idiopathic generalized, susceptibility to, 9, Parkinson disease 11, autosomal dominant, susceptibility to, focal segmental glomerulosclerosis 3, susceptibility to, autoimmune disease, susceptibility to, 1, osteoarthritis susceptibility 3, systemic lupus erythematosus with nephritis, susceptibility to, 1, systemic lupus erythematosus with nephritis, susceptibility to, 2, systemic lupus erythematosus with nephritis, susceptibility to, 3, coronary heart disease, susceptibility to, 2, coronary heart disease, susceptibility to, 4, autoimmune disease, susceptibility to, 2, autoimmune disease, susceptibility to, 3, systemic lupus erythematosus, susceptibility to, 4, myocardial infarction, susceptibility to, major depressive disorder 1, legionnaire disease, susceptibility to, myocardial infarction, susceptibility to, 2, major depressive disorder 2, hypertension, essential, susceptibility to, 4, epilepsy, idiopathic generalized, susceptibility to, 3, myoclonic epilepsy, juvenile, susceptibility to, 3, orofacial cleft 6, susceptibility to, coronary heart disease, susceptibility to, 5, attention deficit-hyperactivity disorder, susceptibility to, 1, attention deficit-hyperactivity disorder, susceptibility to, 2, attention deficit-hyperactivity disorder, susceptibility to, 3, attention deficit-hyperactivity disorder, susceptibility to, 4, Alzheimer disease 9, susceptibility to respiratory infections associated with CD8alpha chain mutation, dyslexia, susceptibility to, 8, autoimmune disease, susceptibility to, 4, hepatitis C virus, susceptibility to, epilepsy, idiopathic generalized, susceptibility to, 4, celiac disease, susceptibility to, 4, celiac disease, susceptibility to, 2, celiac disease, susceptibility to, 3, leprosy, susceptibility to, 1, systemic lupus erythematosus, susceptibility to, 5, systemic lupus erythematosus, susceptibility to, 6, opioid dependence, susceptibility to, 1, systemic lupus erythematosus, susceptibility to, 7, systemic lupus erythematosus, susceptibility to, 8, hypertension, essential, susceptibility to, 5, hypertension, essential, susceptibility to, 6, Parkinson disease 13, autosomal dominant, susceptibility to, West Nile virus, susceptibility to, hepatitis B virus, susceptibility to, Buruli ulcer, susceptibility to, osteoarthritis susceptibility 4, systemic lupus erythematosus, susceptibility to, 9, coronary heart disease, susceptibility to, 7, hypertension, essential, susceptibility to, 7, leprosy, susceptibility to, 4, hypertension, essential, susceptibility to, 8, epilepsy, idiopathic generalized, susceptibility to, 13, coronary heart disease, susceptibility to, 8, myoclonic epilepsy, juvenile, susceptibility to, 4, susceptibility to visceral leishmaniasis, 2, susceptibility to visceral leishmaniasis, 3, celiac disease, susceptibility to, 6, epilepsy, idiopathic generalized, susceptibility to, 5, epilepsy, childhood absence, susceptibility to, 6, celiac disease, susceptibility to, 7, celiac disease, susceptibility to, 8, celiac disease, susceptibility to, 9, celiac disease, susceptibility to, 10, celiac disease, susceptibility to, 11, celiac disease, susceptibility to, 12, celiac disease, susceptibility to, 13, coronary heart disease, susceptibility to, 9, systemic lupus erythematosus, susceptibility to, 10, systemic lupus erythematosus, susceptibility to, 11, systemic lupus erythematosus, susceptibility to, 12, epilepsy, childhood absence, susceptibility to, 5, attention deficit-hyperactivity disorder, susceptibility to, 5, attention deficit-hyperactivity disorder, susceptibility to, 6, systemic lupus erythematosus, susceptibility to, 13, sarcoidosis, susceptibility to, 2, sarcoidosis, susceptibility to, 3, osteoarthritis susceptibility 5, osteoarthritis susceptibility 6, focal segmental glomerulosclerosis 4, susceptibility to, epilepsy, idiopathic generalized, susceptibility to, 8, herpes simplex encephalitis, susceptibility to, 2, attention deficit-hyperactivity disorder, susceptibility to, 7, epilepsy, idiopathic generalized, susceptibility to, 10, pelvic organ prolapse, susceptibility to, 2, systemic lupus erythematosus, susceptibility to, 14, leprosy, susceptibility to, 5, thyrotoxic periodic paralysis, susceptibility to, 2, hearing loss, cisplatin-induced, susceptibility to, susceptibility to mononeuropathy of the median nerve, mild, fatty liver disease, nonalcoholic, susceptibility to, 2, leprosy, susceptibility to, 6, autoimmune disease, susceptibility to, 6, Parkinson disease 5, autosomal dominant, susceptibility to, aspergillosis, susceptibility to, sick sinus syndrome 3, susceptibility to, Parkinson disease 18, autosomal dominant, susceptibility to, epilepsy, juvenile myoclonic, susceptibility to, 9, dengue virus, susceptibility to, coronary heart disease, susceptibility to, 6, podoconiosis, susceptibility to, peripartum cardiomyopathy, susceptibility to, influenza, severe, susceptibility to, thyrotoxic periodic paralysis, susceptibility to, 3, human herpesvirus 8, susceptibility to, epilepsy, idiopathic generalized, susceptibility to, 12, herpes simplex encephalitis, susceptibility to, 3, herpes simplex encephalitis, susceptibility to, 4, pulmonary hypertension, neonatal, susceptibility to, craniosynostosis 5, susceptibility to, melioidosis, susceptibility to, chronic mountain sickness, susceptibility to, herpes simplex encephalitis, susceptibility to, 7, epilepsy, idiopathic generalized, susceptibility to, 14, hereditary neoplastic syndrome, autosomal recessive Mendelian susceptibility to mycobacterial diseases due to partial IFNgammaR1 deficiency, autosomal recessive Mendelian susceptibility to mycobacterial diseases due to partial IFNgammaR2 deficiency, susceptibility to localized juvenile periodontitis, inherited susceptibility to mycobacterial diseases, neural tube defects, susceptibility to, multiple system atrophy 1, susceptibility to, nephrolithiasis susceptibility caused by SLC26A1, myoclonic epilepsy, juvenile, susceptibility to, 1, epilepsy, childhood absence, susceptibility to, 1, autism, susceptiblity to, susceptibility to visceral leishmaniasis, 1, malaria, susceptibility to, spondyloarthropathy, susceptibility to, herpes simplex encephalitis, susceptibility to, 1, epilepsy, idiopathic generalized, susceptibility to, 18, epilepsy, idiopathic generalized, susceptibility to, 15, hepatitis, fulminant viral, susceptibility to, epilepsy, idiopathic generalized, susceptibility to, 16, autosomal recessive mendelian susceptibility to mycobacterial diseases due to partial jak1 deficiency, aortic aneurysm, familial thoracic 11, susceptibility to, amyotrophic lateral sclerosis, susceptibility to, 24, amyotrophic lateral sclerosis, susceptibility to, 25, epilepsy, juvenile myoclonic, susceptibility to, 10, susceptibility to angioedema induced by ACE inhibitors, epidermodysplasia verruciformis, susceptibility to, exfoliation syndrome, susceptibility to, basal cell carcinoma, susceptibility to, graft-versus-host disease, susceptibility to, narcolepsy, susceptibility to, pulmonary disease, chronic obstructive, susceptibility to, restless legs syndrome, susceptibility to, psoriasis, susceptibility to, age related macular degeneration, susceptibility to, allergic rhinitis, susceptibility to, dermatitis, atopic, susceptibility to, Hirschsprung disease, susceptibility to, diabetes mellitus, ketosis-prone, schizophrenia, susceptibility to, radioulnar synostosis, nonsyndromic, susceptibility to, lumbar disk herniation, susceptibility to, lumbar disk degeneration, susceptibility to, psoriatic arthritis, susceptibility to, migraine with or without aura, susceptibility to, Alzheimer disease, susceptibility to, mitochondrial, Asperger syndrome, susceptibility to, tobacco addiction, susceptibility to, lipodystrophy, partial, acquired, susceptibility to, Graves disease, susceptibility to, 1, epilepsy, idiopathic generalized, susceptibility to, 17, breast-ovarian cancer, familial, susceptibility to, blepharospasm, benign essential, susceptibility to, hemorrhage, intracerebral, susceptibility to, IgA nephropathy, susceptibility to, hemolytic uremic syndrome, atypical, susceptibility to, 7, vitiligo-associated multiple autoimmune disease susceptibility 6, delayed sleep phase syndrome, susceptibility to, encephalitis, acute, infection-induced, susceptibility to, malignant hyperthermia, susceptibility to, multicentric Castleman disease, susceptibility to, pelvic organ prolapse, susceptibility to, 1, amyotrophic lateral sclerosis, susceptibility to, 13, Graves disease, susceptibility to, X-linked 2, autoimmune thyroid disease, susceptibility to, 5, atrioventricular septal defect, susceptibility to, 1, epilepsy, juvenile myoclonic, susceptibility to, 6, autoimmune disease, susceptibility to, 5, epilepsy, idiopathic generalized, susceptibility to, 6, seasonal affective disorder, susceptibility to, ventricular fibrillation during myocardial infarction, susceptibility to, UV-induced skin damage, susceptibility to, antisocial behavior, susceptibility to, aplastic anemia, susceptibility to, cardiomyopathy, familial hypertrophic, 4, susceptibility to, cirrhosis, noncryptogenic, susceptibility to, coronary artery disease, severe, susceptibility to, dengue fever, susceptibility to, dengue hemorrhagic fever, susceptibility to, dengue shock syndrome, susceptibility to, dyslexia, susceptibility to, 4, dyslexia, susceptibility to, 7, efavirenz central nervous system toxicity, susceptibility to, obesity, susceptibility to, bleeding disorder, platelet-type, 13, susceptibility to, Schistosoma mansoni infection, susceptibility/resistance to, Parkinson disease 24, autosomal dominant, susceptibility to, iron overload, susceptibility to, hydrocephalus, congenital, 5, susceptibility to, hearing loss, noise-induced, susceptibility to, hyperemesis gravidarum, susceptibility to, Parkinson disease 26, autosomal dominant, susceptibility to, epilepsy, idiopathic generalized, susceptibility to, 19, polycystic kidney disease 9, susceptibility to, rhabdomyolysis, susceptibility to, scoliosis, isolated, susceptibility to, dystonia 38, susceptibility to, gallbladder disease 4, epilepsy, juvenile absence, susceptibility to, GBA1-related Parkinson disease, susceptibility, SH2B3-related immune system disorder

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

28 retrieved; paginated sample, class counts are floors:

12 uncertain significance, 8 conflicting classifications of pathogenicity, 2 likely pathogenic, 2 pathogenic, 1 benign/likely benign, 1 pathogenic/likely pathogenic, 1 risk factor, 1 likely benign

ClinVarVariant (HGVS)GeneClassificationReview
1705020NM_005585.5(SMAD6):c.584T>G (p.Val195Gly)SMAD6Pathogenicno assertion criteria provided
3577612NM_005585.5(SMAD6):c.467_473dup (p.Ser158fs)SMAD6Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
37111NM_005585.5(SMAD6):c.1451G>T (p.Cys484Phe)SMAD6Pathogeniccriteria provided, single submitter
1174565NM_005585.5(SMAD6):c.243del (p.Arg82fs)SMAD6Likely pathogeniccriteria provided, single submitter
992966NM_005585.5(SMAD6):c.592del (p.Arg198fs)SMAD6Likely pathogenicno assertion criteria provided
427724NC_000020.11:g.7125642T>CBMP2risk factorno assertion criteria provided
1174567NM_005585.5(SMAD6):c.269dup (p.Arg91fs)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1315652NM_005585.5(SMAD6):c.1296dup (p.Gly433fs)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
3343869NM_005585.5(SMAD6):c.40T>C (p.Trp14Arg)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
405517NM_005585.5(SMAD6):c.973G>A (p.Ala325Thr)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
471762NM_005585.5(SMAD6):c.79_84del (p.Ser27_Gly28del)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
638816NM_005585.5(SMAD6):c.465_471del (p.Gly156fs)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
697752NM_005585.5(SMAD6):c.887C>T (p.Ser296Phe)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
871071NM_005585.5(SMAD6):c.1084C>T (p.Gln362Ter)SMAD6Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
4278319NM_080593.2(H2BC12):c.*10-670C>AH2BC12Uncertain significancecriteria provided, single submitter
1437413NM_005585.5(SMAD6):c.872T>C (p.Leu291Pro)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
1705021NM_005585.5(SMAD6):c.817G>A (p.Glu273Lys)SMAD6Uncertain significancecriteria provided, single submitter
1707547NM_005585.5(SMAD6):c.1412G>T (p.Gly471Val)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
2436118NM_005585.5(SMAD6):c.1300T>C (p.Tyr434His)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
3061860NM_005585.5(SMAD6):c.793C>G (p.His265Asp)SMAD6Uncertain significancecriteria provided, single submitter
3577611NM_005585.5(SMAD6):c.79_87del (p.Ser27_Gly29del)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
4279918NM_005585.5(SMAD6):c.1377C>A (p.Tyr459Ter)SMAD6Uncertain significancecriteria provided, single submitter
576250NM_005585.5(SMAD6):c.1377C>G (p.Tyr459Ter)SMAD6Uncertain significancecriteria provided, single submitter
579137NM_005585.5(SMAD6):c.776T>C (p.Val259Ala)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
581943NM_005585.5(SMAD6):c.262G>T (p.Gly88Cys)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
976003NM_005585.5(SMAD6):c.1419dup (p.Pro474fs)SMAD6Uncertain significancecriteria provided, multiple submitters, no conflicts
1625473NM_005585.5(SMAD6):c.952+13delSMAD6Benign/Likely benigncriteria provided, multiple submitters, no conflicts
471757NM_005585.5(SMAD6):c.336C>A (p.Gly112=)SMAD6Likely benigncriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 14 · Orphanet: 3 · 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
SMAD6StrongAutosomal dominantcraniosynostosis 714

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
SMAD6Orphanet:402075Familial bicuspid aortic valve
BMP2Orphanet:26129520p12.3 microdeletion syndrome
BMP2Orphanet:93396Brachydactyly type A2

Cohort genes → proteins

3 cohort genes, 3 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence3

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
SMAD6HGNC:6772ENSG00000137834O43541SMAD family member 6gencc,clinvar
BMP2HGNC:1069ENSG00000125845P12643Bone morphogenetic protein 2clinvar
H2BC12HGNC:13954ENSG00000197903O60814Histone H2B type 1-Kclinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
SMAD6SMAD family member 6Transforming growth factor-beta superfamily receptors signaling occurs through the Smad family of intracellular mediators.
BMP2Bone morphogenetic protein 2Growth factor of the TGF-beta superfamily that plays essential roles in many developmental processes, including cardiogenesis, neurogenesis, and osteogenesis.
H2BC12Histone H2B type 1-KCore component of nucleosome.

Protein-family classification

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

Family distribution

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

FamilyGenesFoldFDR
Other/Unknown31.8×0.174

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
SMAD6Other/UnknownnoSMAD_dom, MAD_homology1_Dwarfin-type, SMAD_FHA_dom_sf
BMP2Other/UnknownnoTGF-b_propeptide, TGF-b_C, TGF-beta-like
H2BC12Other/UnknownnoHistone_H2B, H2A/H2B/H3, Histone-fold

Expression context

Cohort genes with no expression data: 0.

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

Breadth distribution (Bgee present_calls)

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

Top tissues across cohort

TissueCohort genes
metanephric glomerulus1
renal glomerulus1
right lung1
cartilage tissue1
pancreatic ductal cell1
pigmented layer of retina1
blood1
bone marrow cell1
monocyte1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
SMAD6277ubiquitousmarkerright lung, renal glomerulus, metanephric glomerulus
BMP2238broadmarkercartilage tissue, pancreatic ductal cell, pigmented layer of retina
H2BC12134ubiquitousmarkerbone marrow cell, monocyte, blood

Protein interactions among cohort

Intra-cohort edges: 1.

Hub genes (top 10 by interactor count)

SymbolInteractor count
BMP23,131
H2BC123,113
SMAD62,006

Intra-cohort edges

ABSources
BMP2SMAD6string_interaction

Structural data

PDB: 2 · AlphaFold-only: 1 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
H2BC12O6081485
BMP2P1264321

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
SMAD6O4354172.34

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 70. Enrichment computed across 3 evidence-associated genes (3 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 3 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Signaling by BMP2237.9×0.002SMAD6, BMP2
Transcriptional regulation by RUNX22169.2×0.002SMAD6, BMP2
Signaling by TGFB family members276.9×0.005SMAD6, BMP2
RUNX2 regulates bone development1271.9×0.035SMAD6
Elastic fibre formation1112.0×0.035BMP2
Molecules associated with elastic fibres1102.9×0.035BMP2
Replacement of protamines by nucleosomes in the male pronucleus190.6×0.035H2BC12
Packaging Of Telomere Ends173.2×0.035H2BC12
Recognition and association of DNA glycosylase with site containing an affected purine168.0×0.035H2BC12
Cleavage of the damaged purine168.0×0.035H2BC12
E3 ubiquitin ligases ubiquitinate target proteins164.5×0.035H2BC12
Recognition and association of DNA glycosylase with site containing an affected pyrimidine161.4×0.035H2BC12
Cleavage of the damaged pyrimidine161.4×0.035H2BC12
RNA Polymerase I Promoter Opening161.4×0.035H2BC12
ChAHP complex assembly161.4×0.035H2BC12
Regulation of RUNX2 expression and activity160.4×0.035BMP2
DNA methylation159.5×0.035H2BC12
FXIIa activates plasma kallikrein-kinin system157.7×0.035H2BC12
SIRT1 negatively regulates rRNA expression156.8×0.035H2BC12
Activated PKN1 stimulates transcription of AR (androgen receptor) regulated genes KLK2 and KLK3156.0×0.035H2BC12
Nonhomologous End-Joining (NHEJ)156.0×0.035H2BC12
Inhibition of DNA recombination at telomere156.0×0.035H2BC12
Assembly of the ORC complex at the origin of replication155.2×0.035H2BC12
DNA Damage/Telomere Stress Induced Senescence154.4×0.035H2BC12
Chromatin modifications during the maternal to zygotic transition (MZT)154.4×0.035H2BC12
Regulation of endogenous retroelements by the Human Silencing Hub (HUSH) complex154.4×0.035H2BC12
Deposition of new CENPA-containing nucleosomes at the centromere152.9×0.035H2BC12
Condensation of Prophase Chromosomes152.1×0.035H2BC12
Defective pyroptosis152.1×0.035H2BC12
PRC2 methylates histones and DNA150.8×0.035H2BC12

GO biological processes by enrichment

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

GO termCohort genesFoldFDRSample cohort genes
positive regulation of miRNA transcription2193.7×0.004SMAD6, BMP2
BMP signaling pathway2133.8×0.004SMAD6, BMP2
negative regulation of transforming growth factor beta receptor signaling pathway2115.8×0.004SMAD6, BMP2
negative regulation of calcium-independent cell-cell adhesion15617.3×0.005BMP2
endodermal-mesodermal cell signaling12808.7×0.005BMP2
cardiac atrium formation12808.7×0.005BMP2
cardiocyte differentiation12808.7×0.005BMP2
mesenchymal cell proliferation involved in ureteric bud development12808.7×0.005BMP2
positive regulation of extracellular matrix constituent secretion11872.4×0.005BMP2
zygotic specification of dorsal/ventral axis11872.4×0.005SMAD6
embryonic heart tube anterior/posterior pattern specification11872.4×0.005BMP2
corticotropin hormone secreting cell differentiation11872.4×0.005BMP2
positive regulation of bone mineralization involved in bone maturation11872.4×0.005BMP2
cardiac jelly development11872.4×0.005BMP2
positive regulation of phosphatase activity11404.3×0.005BMP2
negative regulation of aldosterone biosynthetic process11404.3×0.005BMP2
response to laminar fluid shear stress11404.3×0.005SMAD6
atrioventricular canal morphogenesis11404.3×0.005BMP2
negative regulation of cortisol biosynthetic process11404.3×0.005BMP2
aortic valve development11123.5×0.005BMP2
negative regulation of steroid biosynthetic process11123.5×0.005BMP2
positive regulation of odontogenesis11123.5×0.005BMP2
telencephalon regionalization1936.2×0.006BMP2
thyroid-stimulating hormone-secreting cell differentiation1936.2×0.006BMP2
regulation of odontogenesis of dentin-containing tooth1802.5×0.006BMP2
mesenchyme development1802.5×0.006BMP2
positive regulation of odontoblast differentiation1802.5×0.006BMP2
negative regulation of cardiac muscle cell differentiation1802.5×0.006BMP2
heart induction1702.2×0.006BMP2
ameloblast differentiation1702.2×0.006BMP2

Therapeutics

Drug target analysis

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

Druggability breadth: 1 of 3 evidence-associated genes (33%) have a ChEMBL target (buckets above are over the deeply-mined display cohort).

Top cohort targets by molecule count

SymbolMoleculesMax phase
SMAD600
BMP200
H2BC1200

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

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

SymbolAssaysType breakdown
BMP222Binding:18, Functional:4

Pharmacogenomics

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

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

Chemical tractability of cohort targets

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

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)0
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug0
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug3SMAD6, BMP2, H2BC12

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
SMAD60
BMP222
H2BC120

Clinical trials & evidence

Clinical trials

Clinical trials: 0.