Hypertrophic cardiomyopathy 1

disease
On this page

Also known as cardiomyopathy, familial hypertrophic, 1cardiomyopathy, familial hypertrophic, Autosomal dominant, Digenic dominantcardiomyopathy, familial hypertrophic, type 1cardiomyopathy, hypertrophic, 1, Autosomal dominant, Digenic dominantcardiomyopathy, hypertrophic, 1, digenic, Autosomal dominant, Digenic dominantCMH1hypertrophic cardiomyopathy caused by mutation in MYH7hypertrophic cardiomyopathy type 1MYH7 hypertrophic cardiomyopathy

Summary

Hypertrophic cardiomyopathy 1 (MONDO:0008647) is a disease caused by MYH7 (GenCC Definitive), with 30 cohort genes. The dominant Reactome pathway is Striated Muscle Contraction (9 cohort genes).

At a glance

  • Causal gene: MYH7 (GenCC Definitive)
  • Cohort genes: 30
  • ClinVar variants: 1,477

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namehypertrophic cardiomyopathy 1
Mondo IDMONDO:0008647
OMIM192600
DOIDDOID:0110307
UMLSC3495498
MedGen501195
GARD0024636
Is cancer (heuristic)no

Also known as: cardiomyopathy, familial hypertrophic, 1 · cardiomyopathy, familial hypertrophic, Autosomal dominant, Digenic dominant · cardiomyopathy, familial hypertrophic, type 1 · cardiomyopathy, hypertrophic, 1, Autosomal dominant, Digenic dominant · cardiomyopathy, hypertrophic, 1, digenic, Autosomal dominant, Digenic dominant · CMH1 · hypertrophic cardiomyopathy 1 · hypertrophic cardiomyopathy caused by mutation in MYH7 · hypertrophic cardiomyopathy type 1 · MYH7 hypertrophic cardiomyopathy

Data availability: 1,477 ClinVar variants · 6 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by body system or component › musculoskeletal system disordermuscle tissue disordercardiomyopathyintrinsic cardiomyopathyhypertrophic cardiomyopathyfamilial hypertrophic cardiomyopathyhypertrophic cardiomyopathy 1

Related subtypes (39): hypertrophic cardiomyopathy 2, hypertrophic cardiomyopathy 3, hypertrophic cardiomyopathy 4, Beckwith-Wiedemann syndrome, myotonic dystrophy type 1, very long chain acyl-CoA dehydrogenase deficiency, multiple acyl-CoA dehydrogenase deficiency, 46,XY complete gonadal dysgenesis, hypertrophic cardiomyopathy 6, dilated cardiomyopathy 1C, hypertrophic cardiomyopathy 25, hypertrophic cardiomyopathy 8, hypertrophic cardiomyopathy 10, long chain 3-hydroxyacyl-CoA dehydrogenase deficiency, cardiomyopathy-hypotonia-lactic acidosis syndrome, hypertrophic cardiomyopathy 11, hypertrophic cardiomyopathy 12, hypertrophic cardiomyopathy 13, hypertrophic cardiomyopathy 14, hypertrophic cardiomyopathy 15, hypertrophic cardiomyopathy 7, hypertrophic cardiomyopathy 9, hypertrophic cardiomyopathy 16, hypertrophic cardiomyopathy 17, hypertrophic cardiomyopathy 18, hypertrophic cardiomyopathy 19, hypertrophic cardiomyopathy 20, hypertrophic cardiomyopathy 21, dilated cardiomyopathy 1KK, hypertrophic cardiomyopathy 26, Noonan syndrome and Noonan-related syndrome, long chain acyl-CoA dehydrogenase deficiency, cardiomyopathy, familial hypertrophic, 28, cardiomyopathy, familial hypertrophic 27, cardiomyopathy, familial hypertrophic, 23, with or without ventricular noncompaction, cardiomyopathy, familial restrictive, 5, cardiomyopathy, familial hypertrophic, 29, with polyglucosan bodies, cardiomyopathy, familial hypertrophic, 30, atrial, cardiomyopathy, familial hypertrophic, 31

Genetics & variants

GWAS landscape

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

Variant details and genetic-evidence tiers

ClinVar germline variants

600 retrieved; paginated sample, class counts are floors:

262 uncertain significance, 123 likely benign, 96 conflicting classifications of pathogenicity, 35 pathogenic, 28 likely pathogenic, 27 pathogenic/likely pathogenic, 19 benign/likely benign, 10 benign

ClinVarVariant (HGVS)GeneClassificationReview
14100NM_000257.4(MYH7):c.2333A>G (p.Asp778Gly)LOC126861898Pathogeniccriteria provided, single submitter
14120NM_000257.4(MYH7):c.2609G>A (p.Arg870His)LOC126861898Pathogenicreviewed by expert panel
164324NM_000257.4(MYH7):c.2572C>T (p.Arg858Cys)LOC126861898Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
164337NM_000257.4(MYH7):c.2302G>A (p.Gly768Arg)LOC126861898Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
181196NM_000257.4(MYH7):c.2605C>T (p.Arg869Cys)LOC126861898Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1801829NM_006767.4(LZTR1):c.736C>T (p.Gln246Ter)LZTR1Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
164284NM_000257.4(MYH7):c.4498C>T (p.Arg1500Trp)MHRTPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
181010NC_000011.10:g.47332705G>CMYBPC3Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
181076NM_000256.3(MYBPC3):c.1838dup (p.Asp613fs)MYBPC3Pathogeniccriteria provided, multiple submitters, no conflicts
217827NM_000256.3(MYBPC3):c.1302C>A (p.Tyr434Ter)MYBPC3Pathogeniccriteria provided, multiple submitters, no conflicts
217835NM_000256.3(MYBPC3):c.1359del (p.Val454fs)MYBPC3Pathogeniccriteria provided, multiple submitters, no conflicts
217836NM_000256.3(MYBPC3):c.3712_3713del (p.Leu1238fs)MYBPC3Pathogeniccriteria provided, multiple submitters, no conflicts
217832NM_002471.4(MYH6):c.3193dup (p.Gln1065fs)MYH6Pathogeniccriteria provided, single submitter
132925NM_000257.4(MYH7):c.1573G>A (p.Glu525Lys)MYH7Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
14087NM_000257.4(MYH7):c.1208G>A (p.Arg403Gln)MYH7Pathogenicreviewed by expert panel
14088NM_000257.4(MYH7):c.746G>A (p.Arg249Gln)MYH7Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
14089NM_000257.4(MYH7):c.1357C>T (p.Arg453Cys)MYH7Pathogenicreviewed by expert panel
14090NM_000257.4(MYH7):c.1750G>C (p.Gly584Arg)MYH7Pathogenicreviewed by expert panel
14091NM_000257.4(MYH7):c.1816G>A (p.Val606Met)MYH7Pathogeniccriteria provided, multiple submitters, no conflicts
14092NM_000257.4(MYH7):c.2770G>A (p.Glu924Lys)MYH7Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
14095NM_000257.4(MYH7):c.2167C>T (p.Arg723Cys)MYH7Pathogenicreviewed by expert panel
14096nsv513807MYH7Pathogenicno assertion criteria provided
14097NM_000257.4(MYH7):c.2722C>G (p.Leu908Val)MYH7Pathogenicreviewed by expert panel
14098NM_000257.4(MYH7):c.2221G>C (p.Gly741Arg)MYH7Pathogenicreviewed by expert panel
14099NM_000257.4(MYH7):c.767G>A (p.Gly256Glu)MYH7Pathogeniccriteria provided, multiple submitters, no conflicts
14101NM_000257.4(MYH7):c.1208G>T (p.Arg403Leu)MYH7Pathogeniccriteria provided, multiple submitters, no conflicts
14102NM_000257.4(MYH7):c.1207C>T (p.Arg403Trp)MYH7Pathogenicreviewed by expert panel
14103NM_000257.4(MYH7):c.1538T>G (p.Phe513Cys)MYH7Pathogenicno assertion criteria provided
14104NM_000257.4(MYH7):c.2155C>T (p.Arg719Trp)MYH7Pathogenicreviewed by expert panel
14105NM_000257.4(MYH7):c.2146G>A (p.Gly716Arg)MYH7Pathogenicreviewed by expert panel

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 24 · Orphanet: 87 · 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
MYH7DefinitiveAutosomal dominanthypertrophic cardiomyopathy 120
MYLK2LimitedAutosomal dominanthypertrophic cardiomyopathy 14

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
MYH7Orphanet:154Familial isolated dilated cardiomyopathy
MYH7Orphanet:1880Ebstein malformation of the tricuspid valve
MYH7Orphanet:324604Classic multiminicore myopathy
MYH7Orphanet:54260Left ventricular noncompaction
MYH7Orphanet:59135Laing distal myopathy
MYH7Orphanet:636965Autosomal dominant myosin storage myopathy
MYH7Orphanet:636970Autosomal recessive myosin storage myopathy
SGCDOrphanet:154Familial isolated dilated cardiomyopathy
SGCDOrphanet:219Delta-sarcoglycan-related limb-girdle muscular dystrophy R6
TCAPOrphanet:154Familial isolated dilated cardiomyopathy
TCAPOrphanet:34514Telethonin-related limb-girdle muscular dystrophy R7
TNNI3Orphanet:154Familial isolated dilated cardiomyopathy
TNNI3Orphanet:75249Familial isolated restrictive cardiomyopathy
TNNT2Orphanet:154Familial isolated dilated cardiomyopathy
TNNT2Orphanet:54260Left ventricular noncompaction
TNNT2Orphanet:75249Familial isolated restrictive cardiomyopathy
TPM1Orphanet:154Familial isolated dilated cardiomyopathy
TPM1Orphanet:54260Left ventricular noncompaction
CACNA1COrphanet:101016Romano-Ward syndrome
CACNA1COrphanet:130Brugada syndrome
CACNA1COrphanet:528084Non-specific syndromic intellectual disability
CACNA1COrphanet:595098Timothy syndrome type 1
CACNA1COrphanet:595105Timothy syndrome type 2
CACNA1COrphanet:595109Atypical Timothy syndrome
JPH2Orphanet:154Familial isolated dilated cardiomyopathy
CAV3Orphanet:101016Romano-Ward syndrome
CAV3Orphanet:206599Isolated asymptomatic elevation of creatine phosphokinase
CAV3Orphanet:488650Distal myopathy, Tateyama type
CAV3Orphanet:97238Rippling muscle disease
LDB3Orphanet:154Familial isolated dilated cardiomyopathy
LDB3Orphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
LDB3Orphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
LDB3Orphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
LDB3Orphanet:54260Left ventricular noncompaction
LDB3Orphanet:98912Late-onset distal myopathy, Markesbery-Griggs type
OBSCNOrphanet:99845Genetic recurrent myoglobinuria
ANKRD1Orphanet:154Familial isolated dilated cardiomyopathy
MYH7BOrphanet:54260Left ventricular noncompaction
ACTN2Orphanet:154Familial isolated dilated cardiomyopathy
ACTN2Orphanet:708129Autosomal recessive ACTN2-related distal myopathy
ACTN2Orphanet:708133Autosomal dominant ACTN2-related distal myopathy
MYPNOrphanet:154Familial isolated dilated cardiomyopathy
MYPNOrphanet:171439Childhood-onset nemaline myopathy
MYPNOrphanet:171881Cap myopathy
MYPNOrphanet:75249Familial isolated restrictive cardiomyopathy
CSRP3Orphanet:154Familial isolated dilated cardiomyopathy
NEXNOrphanet:154Familial isolated dilated cardiomyopathy
GLAOrphanet:324Fabry disease
LAMA4Orphanet:154Familial isolated dilated cardiomyopathy
LMNAOrphanet:154Familial isolated dilated cardiomyopathy

Cohort genes → proteins

30 cohort genes, 29 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence30

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
MYLK2HGNC:16243ENSG00000101306Q9H1R3Myosin light chain kinase 2, skeletal/cardiac musclegencc,clinvar
MYH7HGNC:7577ENSG00000092054P12883Myosin-7gencc,clinvar
SGCDHGNC:10807ENSG00000170624Q92629Delta-sarcoglycanclinvar
TCAPHGNC:11610ENSG00000173991O15273Telethoninclinvar
TNNI3HGNC:11947ENSG00000129991P19429Troponin I, cardiac muscleclinvar
TNNT2HGNC:11949ENSG00000118194P45379Troponin T, cardiac muscleclinvar
TPM1HGNC:12010ENSG00000140416P09493Tropomyosin alpha-1 chainclinvar
CACNA1CHGNC:1390ENSG00000151067Q13936Voltage-dependent L-type calcium channel subunit alpha-1Cclinvar
JPH2HGNC:14202ENSG00000149596Q9BR39Junctophilin-2clinvar
CAV3HGNC:1529ENSG00000182533P56539Caveolin-3clinvar
LDB3HGNC:15710ENSG00000122367O75112LIM domain-binding protein 3clinvar
OBSCNHGNC:15719ENSG00000154358Q5VST9Obscurinclinvar
ANKRD1HGNC:15819ENSG00000148677Q15327Ankyrin repeat domain-containing protein 1clinvar
MYH7BHGNC:15906ENSG00000078814A7E2Y1Myosin-7Bclinvar
ACTN2HGNC:164ENSG00000077522P35609Alpha-actinin-2clinvar
MYPNHGNC:23246ENSG00000138347Q86TC9Myopalladinclinvar
CSRP3HGNC:2472ENSG00000129170P50461Cysteine and glycine-rich protein 3clinvar
NEXNHGNC:29557ENSG00000162614Q0ZGT2Nexilinclinvar
GLAHGNC:4296ENSG00000102393P06280Alpha-galactosidase Aclinvar
MHRTHGNC:51291myosin heavy chain associated RNA transcriptclinvar
LAMA4HGNC:6484ENSG00000112769Q16363Laminin subunit alpha-4clinvar
LMNAHGNC:6636ENSG00000160789P02545Prelamin-A/Cclinvar
LZTR1HGNC:6742ENSG00000099949Q8N653Leucine-zipper-like transcriptional regulator 1clinvar
MYBPC3HGNC:7551ENSG00000134571Q14896Myosin-binding protein C, cardiac-typeclinvar
MYH6HGNC:7576ENSG00000197616P13533Myosin-6clinvar
MYL2HGNC:7583ENSG00000111245P10916Myosin regulatory light chain 2, ventricular/cardiac muscle isoformclinvar
MYL3HGNC:7584ENSG00000160808P08590Myosin light chain 3clinvar
OXTRHGNC:8529ENSG00000180914P30559Oxytocin receptorclinvar
PRKAG2HGNC:9386ENSG00000106617Q9UGJ05’-AMP-activated protein kinase subunit gamma-2clinvar
RAF1HGNC:9829ENSG00000132155P04049RAF proto-oncogene serine/threonine-protein kinaseclinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
MYLK2Myosin light chain kinase 2, skeletal/cardiac muscleImplicated in the level of global muscle contraction and cardiac function.
MYH7Myosin-7Myosins are actin-based motor molecules with ATPase activity essential for muscle contraction.
SGCDDelta-sarcoglycanComponent of the sarcoglycan complex, a subcomplex of the dystrophin-glycoprotein complex which forms a link between the F-actin cytoskeleton and the extracellular matrix.
TCAPTelethoninMuscle assembly regulating factor.
TNNI3Troponin I, cardiac muscleTroponin I is the inhibitory subunit of troponin, the thin filament regulatory complex which confers calcium-sensitivity to striated muscle actomyosin ATPase activity.
TNNT2Troponin T, cardiac muscleTroponin T is the tropomyosin-binding subunit of troponin, the thin filament regulatory complex which confers calcium-sensitivity to striated muscle actomyosin ATPase activity.
TPM1Tropomyosin alpha-1 chainBinds to actin filaments in muscle and non-muscle cells.
CACNA1CVoltage-dependent L-type calcium channel subunit alpha-1CPore-forming, alpha-1C subunit of the voltage-gated calcium channel that gives rise to L-type calcium currents.
JPH2Junctophilin-2Membrane-binding protein that provides a structural bridge between the plasma membrane and the sarcoplasmic reticulum and is required for normal excitation-contraction coupling in cardiomyocytes.
CAV3Caveolin-3May act as a scaffolding protein within caveolar membranes.
LDB3LIM domain-binding protein 3May function as an adapter in striated muscle to couple protein kinase C-mediated signaling via its LIM domains to the cytoskeleton.
OBSCNObscurinStructural component of striated muscles which plays a role in myofibrillogenesis.
ANKRD1Ankyrin repeat domain-containing protein 1May play an important role in endothelial cell activation.
MYH7BMyosin-7BInvolved in muscle contraction.
ACTN2Alpha-actinin-2F-actin cross-linking protein which is thought to anchor actin to a variety of intracellular structures.
MYPNMyopalladinComponent of the sarcomere that tethers together nebulin (skeletal muscle) and nebulette (cardiac muscle) to alpha-actinin, at the Z lines.
CSRP3Cysteine and glycine-rich protein 3Positive regulator of myogenesis.
NEXNNexilinInvolved in regulating cell migration through association with the actin cytoskeleton.
GLAAlpha-galactosidase ACatalyzes the hydrolysis of glycosphingolipids and participates in their degradation in the lysosome.
LAMA4Laminin subunit alpha-4Binding to cells via a high affinity receptor, laminin is thought to mediate the attachment, migration and organization of cells into tissues during embryonic development by interacting with other extracellular matrix components.
LMNAPrelamin-A/CLamins are intermediate filament proteins that assemble into a filamentous meshwork, and which constitute the major components of the nuclear lamina, a fibrous layer on the nucleoplasmic side of the inner nuclear membrane.
LZTR1Leucine-zipper-like transcriptional regulator 1Substrate-specific adapter of a BCR (BTB-CUL3-RBX1) E3 ubiquitin-protein ligase complex that mediates ubiquitination of Ras (K-Ras/KRAS, N-Ras/NRAS and H-Ras/HRAS).
MYBPC3Myosin-binding protein C, cardiac-typeThick filament-associated protein located in the crossbridge region of vertebrate striated muscle a bands.
MYH6Myosin-6Muscle contraction.
MYL2Myosin regulatory light chain 2, ventricular/cardiac muscle isoformContractile protein that plays a role in heart development and function.
MYL3Myosin light chain 3Regulatory light chain of myosin.
OXTROxytocin receptorReceptor for oxytocin.
PRKAG25’-AMP-activated protein kinase subunit gamma-2AMP/ATP-binding subunit of AMP-activated protein kinase (AMPK), an energy sensor protein kinase that plays a key role in regulating cellular energy metabolism.
RAF1RAF proto-oncogene serine/threonine-protein kinaseSerine/threonine-protein kinase that acts as a regulatory link between the membrane-associated Ras GTPases and the MAPK/ERK cascade, and this critical regulatory link functions as a switch determining cell fate decisions including prolifer…

Protein-family classification

Druggable: 9 · Difficult: 6 · Unknown: 15 · Druggable fraction: 0.3

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
Antibody/Immunoglobulin32.9×0.247
Kinase32.8×0.247
Scaffold/PPI42.3×0.247
Ion channel13.7×0.474
Other/Unknown150.9×0.927
GPCR10.8×0.927
Transcription factor20.6×0.927
Enzyme (other)10.4×0.927

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
MYLK2Kinaseyes2.7.11.18Prot_kinase_dom, Ser/Thr_kinase_AS, Kinase-like_dom_sf
MYH7Scaffold/PPInoIQ_motif_EF-hand-BS, Myosin_head_motor_dom-like, Myosin_tail
SGCDOther/UnknownnoSarcoglycan, Sarcoglycan_gamma/delta/zeta
TCAPOther/UnknownnoTelethonin, Titin-like_dom_sf
TNNI3Other/UnknownnoTroponin, Troponin-I_N, Troponin_sf
TNNT2Other/UnknownnoTroponin, TNNT, Troponin_sf
TPM1Other/UnknownnoTropomyosin
CACNA1CIon channelyesVDCCAlpha1, VDCC_L_a1su, VDCC_L_a1csu
JPH2Other/UnknownnoMORN, Junctophilin
CAV3Other/UnknownnoCaveolin, Caveolin_CS
LDB3Transcription factornoPDZ, Znf_LIM, Zasp-like_motif
OBSCNKinaseyesIQ_motif_EF-hand-BS, DH_dom, Prot_kinase_dom
ANKRD1Scaffold/PPInoAnkyrin_rpt, Ankyrin_rpt-contain_sf
MYH7BScaffold/PPInoMyosin_head_motor_dom-like, Myosin_tail, SH3_Myosin
ACTN2Other/UnknownnoActinin_actin-bd_CS, CH_dom, Spectrin_repeat
MYPNAntibody/ImmunoglobulinyesIg_sub2, Ig_sub, Ig-like_dom
CSRP3Transcription factornoZnf_LIM
NEXNAntibody/ImmunoglobulinyesIg_sub, Ig-like_dom, Ig_I-set
GLAEnzyme (other)yes3.2.1.22Glyco_hydro_27/36_CS, Glyco_hydro_27, Glyco_hydro_b
MHRTOther/Unknownno
LAMA4Other/UnknownnoEGF, Laminin_G, LE_dom
LMNAOther/UnknownnoLamin_tail_dom, IF_conserved, Lamin_tail_dom_sf
LZTR1Other/UnknownnoBTB/POZ_dom, Kelch_1, SKP1/BTB/POZ_sf
MYBPC3Antibody/ImmunoglobulinyesIg_sub2, Ig_sub, FN3_dom
MYH6Scaffold/PPInoMyosin_head_motor_dom-like, Myosin_tail, SH3_Myosin
MYL2Other/UnknownnoEF_hand_dom, EF-hand-dom_pair, EF_Hand_1_Ca_BS
MYL3Other/UnknownnoEF_hand_dom, EF-hand-dom_pair, CALM/Myosin/TropC-like
OXTRGPCRyesGPCR_Rhodpsn, Vasoprsn_rcpt, Oxytocn_rcpt
PRKAG2Other/UnknownnoCBS_dom, CBS_dom_sf, AMPK_gamma/SDS23_families
RAF1Kinaseyes2.7.10.2Prot_kinase_dom, Ser-Thr/Tyr_kinase_cat_dom, PKC_DAG/PE

Expression context

Cohort genes with no expression data: 1.

23 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)29
unknown1

Top tissues across cohort

TissueCohort genes
apex of heart12
hindlimb stylopod muscle10
skeletal muscle tissue of rectus abdominis7
left ventricle myocardium6
heart right ventricle5
right atrium auricular region5
cardiac atrium5
gastrocnemius4
skeletal muscle tissue of biceps brachii3
myocardium2
vastus lateralis2
cardiac muscle of right atrium2
muscle layer of sigmoid colon1
right coronary artery1
tibialis anterior1
triceps brachii1
heart left ventricle1
monocyte1
mononuclear cell1
pancreatic ductal cell1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
MYLK2148tissue_specificyeshindlimb stylopod muscle, skeletal muscle tissue of rectus abdominis, gastrocnemius
MYH7167tissue_specificmarkerapex of heart, hindlimb stylopod muscle, skeletal muscle tissue of biceps brachii
SGCD247broadmarkerleft ventricle myocardium, skeletal muscle tissue of rectus abdominis, heart right ventricle
TCAP213tissue_specificmarkerapex of heart, hindlimb stylopod muscle, skeletal muscle tissue of rectus abdominis
TNNI3169broadmarkerapex of heart, left ventricle myocardium, right atrium auricular region
TNNT2154broadmarkerapex of heart, right atrium auricular region, cardiac atrium
TPM1305ubiquitousmarkerleft ventricle myocardium, heart right ventricle, myocardium
CACNA1C134broadmarkerapex of heart, right coronary artery, muscle layer of sigmoid colon
JPH2173broadyesleft ventricle myocardium, skeletal muscle tissue of rectus abdominis, tibialis anterior
CAV3157tissue_specificyeshindlimb stylopod muscle, vastus lateralis, triceps brachii
LDB3247broadmarkerskeletal muscle tissue of biceps brachii, hindlimb stylopod muscle, apex of heart
OBSCN197ubiquitousmarkerhindlimb stylopod muscle, apex of heart, gastrocnemius
ANKRD1155ubiquitousmarkerapex of heart, right atrium auricular region, cardiac atrium
MYH7B195broadyesapex of heart, hindlimb stylopod muscle, heart left ventricle
ACTN2226broadmarkerskeletal muscle tissue of rectus abdominis, skeletal muscle tissue of biceps brachii, hindlimb stylopod muscle
MYPN116broadmarkerhindlimb stylopod muscle, gastrocnemius, vastus lateralis
CSRP3173tissue_specificmarkerskeletal muscle tissue of rectus abdominis, heart right ventricle, left ventricle myocardium
NEXN229ubiquitousmarkerleft ventricle myocardium, skeletal muscle tissue of rectus abdominis, myocardium
GLA263ubiquitousmarkerpancreatic ductal cell, monocyte, mononuclear cell
MHRT
LAMA4268ubiquitousmarkerlower esophagus muscularis layer, lower esophagus, nerve
LMNA295ubiquitousmarkernipple, mucosa of stomach, skin of abdomen
LZTR1134ubiquitousmarkersural nerve, pituitary gland, adenohypophysis
MYBPC3149tissue_specificmarkerapex of heart, right atrium auricular region, cardiac atrium
MYH6154tissue_specificyescardiac muscle of right atrium, cardiac atrium, vena cava
MYL2179tissue_specificmarkerheart right ventricle, diaphragm, apex of heart
MYL3198broadmarkerapex of heart, heart right ventricle, hindlimb stylopod muscle
OXTR204broadyesdecidua, bronchial epithelial cell, epithelium of mammary gland
PRKAG2258ubiquitousmarkerright atrium auricular region, cardiac atrium, cardiac muscle of right atrium
RAF1299ubiquitousmarkergastrocnemius, muscle of leg, ventricular zone

Protein interactions among cohort

Intra-cohort edges: 88.

Hub genes (top 10 by interactor count)

SymbolInteractor count
LMNA7,173
RAF16,574
TPM13,514
PRKAG23,212
CACNA1C3,145
MYH63,119
MYL23,119
ACTN22,781
MYH72,744
LAMA42,688

Intra-cohort edges

ABSources
ACTN2CACNA1Cbiogrid_interaction
ACTN2CSRP3intact, string_interaction
ACTN2LDB3biogrid_interaction, intact, string_interaction
ACTN2MYH6biogrid_interaction, string_interaction
ACTN2MYH7biogrid_interaction, string_interaction
ACTN2MYPNbiogrid_interaction, string_interaction
ACTN2NEXNstring_interaction
ACTN2TCAPstring_interaction
ACTN2TNNT2string_interaction
ACTN2TPM1biogrid_interaction, string_interaction
ANKRD1CSRP3string_interaction
ANKRD1MYPNbiogrid_interaction, string_interaction
ANKRD1TCAPstring_interaction
CACNA1CCAV3string_interaction
CACNA1CJPH2string_interaction
CAV3JPH2string_interaction
CSRP3LDB3string_interaction
CSRP3MYBPC3string_interaction
CSRP3MYH7string_interaction
CSRP3MYL2string_interaction
CSRP3MYL3string_interaction
CSRP3MYLK2string_interaction
CSRP3MYPNstring_interaction
CSRP3NEXNstring_interaction
CSRP3PRKAG2string_interaction
CSRP3TCAPintact, string_interaction
CSRP3TNNI3string_interaction
CSRP3TNNT2string_interaction
CSRP3TPM1string_interaction
JPH2TCAPstring_interaction
LDB3MYBPC3string_interaction
LDB3MYH7string_interaction
LDB3MYPNintact
LDB3NEXNstring_interaction
LDB3TCAPstring_interaction
LDB3TNNT2string_interaction
LMNASGCDstring_interaction
LZTR1MYH6intact
MYBPC3MYH6string_interaction
MYBPC3MYH7intact, string_interaction
MYBPC3MYL2string_interaction
MYBPC3MYL3string_interaction
MYBPC3MYLK2string_interaction
MYBPC3PRKAG2string_interaction
MYBPC3TCAPstring_interaction
MYBPC3TNNI3string_interaction
MYBPC3TNNT2string_interaction
MYBPC3TPM1string_interaction
MYH6MYL2string_interaction
MYH6MYL3string_interaction

Structural data

PDB: 20 · AlphaFold-only: 9 · No structure: 1

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
RAF1P0404976
MYH7P1288343
TNNI3P1942939
CACNA1CQ1393633
GLAP0628031
LMNAP0254528
TNNT2P4537925
OBSCNQ5VST925
MYBPC3Q1489617
ACTN2P3560916
TPM1P0949314
LZTR1Q8N6533
MYL2P109163
MYL3P085903
OXTRP305593
MYLK2Q9H1R32
TCAPO152732
JPH2Q9BR392
LDB3O751122
CSRP3P504612

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
CAV3P5653988.54
ANKRD1Q1532782.64
SGCDQ9262981.43
MYH6P1353374.91
MYH7BA7E2Y173.79
LAMA4Q1636373.75
NEXNQ0ZGT270.78
PRKAG2Q9UGJ067.24
MYPNQ86TC952.71

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 118. Enrichment computed across 30 evidence-associated genes (20 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 20 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Striated Muscle Contraction9138.9×3e-16TCAP, TNNI3, TNNT2, TPM1, ACTN2, MYBPC3, MYH6, MYL2 (+1 more)
Muscle contraction830.9×4e-09TCAP, CACNA1C, CAV3, ACTN2, MYBPC3, MYH6, MYL2, MYL3
Formation of the dystrophin-glycoprotein complex (DGC)230.9×0.074SGCD, LAMA4
Smooth Muscle Contraction226.6×0.074TPM1, CAV3
Breakdown of the nuclear lamina1190.3×0.079LMNA
Post NMDA receptor activation events220.4×0.079ACTN2, PRKAG2
Activation of NMDA receptors and postsynaptic events218.4×0.079ACTN2, PRKAG2
Integration of energy metabolism217.6×0.079CACNA1C, PRKAG2
Signaling by BRAF and RAF1 fusions217.0×0.079LMNA, RAF1
Non-integrin membrane-ECM interactions215.4×0.086SGCD, LAMA4
Vasopressin-like receptors195.2×0.103OXTR
Negative feedback regulation of MAPK pathway195.2×0.103RAF1
AMPK inhibits chREBP transcriptional activation activity171.4×0.103PRKAG2
SHOC2 M1731 mutant abolishes MRAS complex function171.4×0.103RAF1
Gain-of-function MRAS complexes activate RAF signaling171.4×0.103RAF1
IFNG signaling activates MAPKs171.4×0.103RAF1
Lipophagy163.4×0.108PRKAG2
Activation of PPARGC1A (PGC-1alpha) by phosphorylation157.1×0.108PRKAG2
Neurotransmitter receptors and postsynaptic signal transmission210.0×0.108ACTN2, PRKAG2
GP1b-IX-V activation signalling147.6×0.122RAF1
CREB1 phosphorylation through NMDA receptor-mediated activation of RAS signaling143.9×0.122ACTN2
Carnitine shuttle138.1×0.122PRKAG2
Depolymerization of the Nuclear Lamina138.1×0.122LMNA
Phase 2 - plateau phase138.1×0.122CACNA1C
Rap1 signalling135.7×0.122RAF1
Transmission across Chemical Synapses27.6×0.122ACTN2, PRKAG2
Metabolism of lipids34.7×0.122ANKRD1, GLA, PRKAG2
Initiation of Nuclear Envelope (NE) Reformation130.1×0.125LMNA
MET promotes cell motility130.1×0.125LAMA4
Ras activation upon Ca2+ influx through NMDA receptor128.6×0.125ACTN2

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 29 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
cardiac muscle contraction11152.2×5e-20MYLK2, MYH7, TCAP, TNNI3, TNNT2, TPM1, CSRP3, MYBPC3 (+3 more)
sarcomere organization11145.3×5e-20TCAP, TNNT2, TPM1, LDB3, OBSCN, ANKRD1, ACTN2, MYPN (+3 more)
ventricular cardiac muscle tissue morphogenesis8193.7×2e-15MYH7, TNNI3, TNNT2, TPM1, MYBPC3, MYH6, MYL2, MYL3
muscle filament sliding6217.9×8e-12MYH7, TCAP, TNNI3, TNNT2, TPM1, MYH6
regulation of the force of heart contraction5170.9×4e-09MYH7, CSRP3, MYH6, MYL2, MYL3
cardiac muscle cell development5107.6×4e-08SGCD, TCAP, CAV3, ACTN2, MYH6
detection of muscle stretch3249.0×6e-06TCAP, CAV3, CSRP3
regulation of striated muscle contraction3217.9×9e-06MYBPC3, MYL2, MYL3
regulation of heart contraction468.4×1e-05TNNT2, TPM1, CAV3, MYH6
cardiac muscle hypertrophy3174.3×2e-05TCAP, CAV3, CSRP3
cardiac muscle tissue morphogenesis3145.3×3e-05MYLK2, TCAP, ANKRD1
cardiac myofibril assembly3134.1×3e-05TCAP, CSRP3, MYL2
adult heart development3124.5×4e-05MYH7, TCAP, MYH6
cardiac muscle hypertrophy in response to stress3109.0×5e-05MYH7, TCAP, MYH6
regulation of muscle filament sliding2581.1×5e-05MYLK2, MYBPC3
striated muscle contraction387.2×9e-05MYLK2, MYH7, MYH6
response to muscle stretch379.2×1e-04TCAP, ANKRD1, RAF1
heart contraction379.2×1e-04SGCD, TNNI3, MYL2
muscle contraction428.7×2e-04MYH7, MYH6, MYL3, OXTR
skeletal muscle contraction352.8×3e-04MYH7, TCAP, TNNI3
regulation of heart rate348.4×4e-04MYH7, CAV3, MYH6
T-tubule organization2193.7×5e-04CAV3, CSRP3
phospholipase C/protein kinase C signal transduction2193.7×5e-04ANKRD1, CSRP3
regulation of muscle contraction2116.2×0.001TNNT2, TPM1
negative regulation of ATP-dependent activity2116.2×0.001TNNI3, TNNT2
regulation of cardiac muscle contraction by calcium ion signaling289.4×0.002TNNI3, JPH2
negative regulation of protein localization to cell surface289.4×0.002CAV3, ACTN2
calcium ion transport into cytosol283.0×0.003CACNA1C, JPH2
regulation of cardiac muscle cell contraction277.5×0.003MYH7B, MYBPC3
dendrite self-avoidance272.6×0.003MYPN, NEXN

Therapeutics

Drug target analysis

Approved (phase 4): 7 · Phase ≥3: 7 · Phased (≥1): 7 · Undrugged: 23

Druggability breadth: 16 of 30 evidence-associated genes (53%) 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
MYLK2FEDRATINIB
CACNA1CREMIFENTANIL
GLACLOTRIMAZOLE
LMNABEPRIDIL
OXTRDESMOPRESSIN
PRKAG2ADENOSINE PHOSPHATE
RAF1VEMURAFENIB

Top cohort targets by molecule count

SymbolMoleculesMax phase
LMNA8234
CACNA1C854
GLA624
RAF1314
MYLK2194
PRKAG2194
OXTR154
MYH700
SGCD00
TCAP00

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
FEDRATINIB4MYLK2
AXITINIB4MYLK2
SORAFENIB4MYLK2, RAF1
PAZOPANIB4MYLK2, RAF1
NINTEDANIB4MYLK2
SUNITINIB4CACNA1C, LMNA, MYLK2, PRKAG2
DASATINIB4CACNA1C, MYLK2, RAF1
ERLOTINIB4LMNA, MYLK2, RAF1
GEFITINIB4MYLK2
REMIFENTANIL4CACNA1C
BEPRIDIL4CACNA1C, LMNA
CLOTRIMAZOLE4CACNA1C, GLA, LMNA
PROPIVERINE4CACNA1C
DIBUCAINE4CACNA1C, LMNA
IMIPRAMINE4CACNA1C, LMNA
DULOXETINE4CACNA1C
QUINIDINE4CACNA1C
ESTRADIOL4CACNA1C, LMNA
TOLTERODINE4CACNA1C, LMNA
PIMOZIDE4CACNA1C, LMNA
NIMODIPINE4CACNA1C, LMNA
NICARDIPINE4CACNA1C, LMNA
AMLODIPINE4CACNA1C
VARDENAFIL4CACNA1C
CLEMASTINE4CACNA1C
ISRADIPINE4CACNA1C
TERFENADINE4CACNA1C, LMNA
NISOLDIPINE4CACNA1C, LMNA
SOLIFENACIN4CACNA1C
PINAVERIUM4CACNA1C

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 3.

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

SymbolAssaysType breakdown
RAF1839Binding:803, Functional:31, ADMET:5
CACNA1C575Binding:319, Functional:211, Toxicity:26, ADMET:19
PRKAG2266Binding:265, Functional:1
OXTR249Binding:149, Functional:99, Unclassified:1
MYLK2196Binding:196
GLA114Binding:104, Functional:10
LMNA12Binding:9, Functional:3
TPM13Binding:3
TNNI32Binding:2
TNNT22Binding:2

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
MYLK22.7.11.18myosin-light-chain kinase
GLA3.2.1.22alpha-galactosidase
RAF12.7.10.2non-specific protein-tyrosine kinase

Cohort genes with high screening signal

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

SymbolChEMBL assays
MYLK2196
CACNA1C575
GLA114
OXTR249
PRKAG2266
RAF1839

Pharmacogenomics

Cohort genes with a PharmGKB record: 29; 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)
FEDRATINIB4MYLK2
AXITINIB4MYLK2
SORAFENIB4MYLK2, RAF1
PAZOPANIB4MYLK2, RAF1
NINTEDANIB4MYLK2
SUNITINIB4CACNA1C, LMNA, MYLK2, PRKAG2
DASATINIB4CACNA1C, MYLK2, RAF1
ERLOTINIB4LMNA, MYLK2, RAF1
GEFITINIB4MYLK2
REMIFENTANIL4CACNA1C
BEPRIDIL4CACNA1C, LMNA
CLOTRIMAZOLE4CACNA1C, GLA, LMNA
PROPIVERINE4CACNA1C
DIBUCAINE4CACNA1C, LMNA
IMIPRAMINE4CACNA1C, LMNA
DULOXETINE4CACNA1C
QUINIDINE4CACNA1C
ESTRADIOL4CACNA1C, LMNA
TOLTERODINE4CACNA1C, LMNA
PIMOZIDE4CACNA1C, LMNA
NIMODIPINE4CACNA1C, LMNA
NICARDIPINE4CACNA1C, LMNA
AMLODIPINE4CACNA1C
VARDENAFIL4CACNA1C
CLEMASTINE4CACNA1C
ISRADIPINE4CACNA1C
TERFENADINE4CACNA1C, LMNA
NISOLDIPINE4CACNA1C, LMNA
SOLIFENACIN4CACNA1C
PINAVERIUM4CACNA1C

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)7MYLK2, CACNA1C, GLA, LMNA, OXTR, PRKAG2, RAF1
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug2OBSCN, MYBPC3
DDruggable family + AlphaFold only, no drug2MYPN, NEXN
EDifficult family or no structure, no drug19MYH7, SGCD, TCAP, TNNI3, TNNT2, TPM1, JPH2, CAV3, LDB3, ANKRD1 (+9 more)

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
MYH70
SGCD0
TCAP0
TNNI32
TNNT22
TPM13
JPH20
CAV30
LDB30
OBSCN0
ANKRD10
MYH7B0
ACTN20
MYPN0
CSRP30
NEXN0
MHRT0
LAMA40
LZTR10
MYBPC30
MYH60
MYL20
MYL30

Clinical trials & evidence

Clinical trials

Clinical trials: 0.