Emery-Dreifuss muscular dystrophy 2, autosomal dominant

disease
On this page

Also known as autosomal dominant limb-girdle muscular dystrophy caused by mutation in LMNAbenign scapuloperoneal muscular dystrophy with cardiomyopathyEDMD2EMD2Hauptmann-Thannhauser muscular dystrophyLGMD1Blimb-girdle muscular dystrophy due to lamin A/C deficiencylimb-girdle muscular dystrophy type 1BLMNA autosomal dominant limb-girdle muscular dystrophymuscular dystrophy, limb-girdle type 1Bmuscular dystrophy, limb-girdle, type 1Bmuscular dystrophy, proximal, type 1Bproximal muscular dystrophy type 1B

Summary

Emery-Dreifuss muscular dystrophy 2, autosomal dominant (MONDO:0021569) is a disease caused by LMNA (GenCC Definitive), with 3 cohort genes and 1 clinical trial.

At a glance

  • Causal gene: LMNA (GenCC Definitive)
  • Cohort genes: 3
  • ClinVar variants: 280
  • Clinical trials: 1

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameEmery-Dreifuss muscular dystrophy 2, autosomal dominant
Mondo IDMONDO:0021569
MeSHC535898
OMIM159001, 181350
Orphanet264
DOIDDOID:0070247, DOID:0110301
NCITC126745
SNOMED CT240072005, 718178006
UMLSC0410190
MedGen98048
GARD0010230
Is cancer (heuristic)no

Also known as: autosomal dominant limb-girdle muscular dystrophy caused by mutation in LMNA · benign scapuloperoneal muscular dystrophy with cardiomyopathy · EDMD2 · EMD2 · Emery-Dreifuss muscular dystrophy 2, autosomal dominant · Hauptmann-Thannhauser muscular dystrophy · LGMD1B · limb-girdle muscular dystrophy due to lamin A/C deficiency · limb-girdle muscular dystrophy type 1B · LMNA autosomal dominant limb-girdle muscular dystrophy · muscular dystrophy, limb-girdle type 1B · muscular dystrophy, limb-girdle, type 1B · muscular dystrophy, proximal, type 1B · proximal muscular dystrophy type 1B

Data availability: 280 ClinVar variants · 3 GenCC gene-disease records · 5 cell lines.

Disease family

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary disease › autosomal genetic disease › autosomal dominant disease › muscular dystrophy, limb-girdle, autosomal dominantEmery-Dreifuss muscular dystrophy 2, autosomal dominant

Related subtypes (7): autosomal dominant limb-girdle muscular dystrophy type 1F, autosomal dominant limb-girdle muscular dystrophy type 1G, myofibrillar myopathy 3, autosomal dominant limb-girdle muscular dystrophy type 1H, autosomal dominant limb-girdle muscular dystrophy type 1E (DES), autosomal dominant limb-girdle muscular dystrophy type 1D (DNAJB6), muscular dystrophy, limb-girdle, autosomal dominant 4

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

280 retrieved; paginated sample, class counts are floors:

122 uncertain significance, 74 conflicting classifications of pathogenicity, 21 pathogenic, 19 pathogenic/likely pathogenic, 18 likely benign, 10 benign/likely benign, 8 likely pathogenic, 4 benign, 3 uncertain significance/uncertain risk allele, 1 not provided

ClinVarVariant (HGVS)GeneClassificationReview
14478NM_170707.4(LMNA):c.1357C>T (p.Arg453Trp)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
14481NM_170707.4(LMNA):c.1580G>C (p.Arg527Pro)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
14482NM_170707.4(LMNA):c.1589T>C (p.Leu530Pro)LMNAPathogenicno assertion criteria provided
14489NM_170707.4(LMNA):c.1444C>T (p.Arg482Trp)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
14491NM_170707.4(LMNA):c.960del (p.Arg321fs)LMNAPathogeniccriteria provided, single submitter
14495NM_170707.4(LMNA):c.1130G>A (p.Arg377His)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
14511NM_170707.4(LMNA):c.777T>A (p.Tyr259Ter)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
14514NM_170707.4(LMNA):c.1477C>T (p.Gln493Ter)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
14525NM_170707.4(LMNA):c.1072G>A (p.Glu358Lys)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
1457399NM_170707.4(LMNA):c.822del (p.Arg275fs)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1691500NM_170707.4(LMNA):c.1549_1550del (p.Gln517fs)LMNAPathogenicno assertion criteria provided
1695415NM_170707.4(LMNA):c.1381-2A>CLMNAPathogenicno assertion criteria provided
200941NM_170707.4(LMNA):c.768G>A (p.Val256=)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
216958NM_170707.4(LMNA):c.810+1G>CLMNAPathogeniccriteria provided, single submitter
246077NM_170707.4(LMNA):c.1540T>C (p.Trp514Arg)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
285468NM_170707.4(LMNA):c.83G>A (p.Arg28Gln)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
3595598NM_170707.4(LMNA):c.5del (p.Glu2fs)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
36473NM_170707.4(LMNA):c.1003C>T (p.Arg335Trp)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
48031NM_170707.4(LMNA):c.1129C>T (p.Arg377Cys)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
48070NM_170707.4(LMNA):c.607G>A (p.Glu203Lys)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
48074NM_170707.4(LMNA):c.673C>T (p.Arg225Ter)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
48077NM_170707.4(LMNA):c.745C>G (p.Arg249Gly)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
48096NM_170707.4(LMNA):c.961C>T (p.Arg321Ter)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
66762NM_170707.4(LMNA):c.1045C>T (p.Arg349Trp)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
66782NM_170707.4(LMNA):c.1157G>A (p.Arg386Lys)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
66791NM_170707.4(LMNA):c.116A>G (p.Asn39Ser)LMNAPathogeniccriteria provided, multiple submitters, no conflicts
66800NM_170707.4(LMNA):c.1294C>T (p.Gln432Ter)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
66808NM_170707.4(LMNA):c.1358G>C (p.Arg453Pro)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
66817NM_170707.4(LMNA):c.1380+1G>ALMNAPathogeniccriteria provided, multiple submitters, no conflicts
66849NM_170707.4(LMNA):c.1583C>A (p.Thr528Lys)LMNAPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 40 · Orphanet: 26 · 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
LMNADefinitiveSemidominantEmery-Dreifuss muscular dystrophy 2, autosomal dominant40

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
LMNAOrphanet:154Familial isolated dilated cardiomyopathy
LMNAOrphanet:157973Congenital muscular dystrophy due to LMNA mutation
LMNAOrphanet:1662Restrictive dermopathy
LMNAOrphanet:168796Heart-hand syndrome, Slovenian type
LMNAOrphanet:2229Dilated cardiomyopathy-hypergonadotropic hypogonadism syndrome
LMNAOrphanet:2348Familial partial lipodystrophy, Dunnigan type
LMNAOrphanet:280365Autosomal semi-dominant severe lipodystrophic laminopathy
LMNAOrphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
LMNAOrphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
LMNAOrphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
LMNAOrphanet:300751Familial dilated cardiomyopathy with conduction defect due to LMNA mutation
LMNAOrphanet:363618LMNA-related cardiocutaneous progeria syndrome
LMNAOrphanet:54260Left ventricular noncompaction
LMNAOrphanet:675396Epithelioid hemangioma
LMNAOrphanet:740Hutchinson-Gilford progeria syndrome
LMNAOrphanet:79084Familial partial lipodystrophy, Köbberling type
LMNAOrphanet:79474Atypical Werner syndrome
LMNAOrphanet:90153Mandibuloacral dysplasia with type A lipodystrophy
LMNAOrphanet:98853Autosomal dominant Emery-Dreifuss muscular dystrophy
LMNAOrphanet:98855Autosomal recessive Emery-Dreifuss muscular dystrophy
LMNAOrphanet:98856Charcot-Marie-Tooth disease type 2B1
SYNE2Orphanet:98853Autosomal dominant Emery-Dreifuss muscular dystrophy
TMEM43Orphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
TMEM43Orphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
TMEM43Orphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
TMEM43Orphanet:98853Autosomal dominant Emery-Dreifuss muscular dystrophy

Cohort genes → proteins

3 cohort genes, 3 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence3

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
LMNAHGNC:6636ENSG00000160789P02545Prelamin-A/Cgencc,clinvar
SYNE2HGNC:17084ENSG00000054654Q8WXH0Nesprin-2clinvar
TMEM43HGNC:28472ENSG00000170876Q9BTV4Transmembrane protein 43clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
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.
SYNE2Nesprin-2Multi-isomeric modular protein which forms a linking network between organelles and the actin cytoskeleton to maintain the subcellular spatial organization.
TMEM43Transmembrane protein 43May have an important role in maintaining nuclear envelope structure by organizing protein complexes at the inner nuclear membrane.

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)
LMNAOther/UnknownnoLamin_tail_dom, IF_conserved, Lamin_tail_dom_sf
SYNE2Other/UnknownnoActinin_actin-bd_CS, CH_dom, Spectrin_repeat
TMEM43Other/UnknownnoTMEM43_fam

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
mucosa of stomach1
nipple1
skin of abdomen1
ganglionic eminence1
skeletal muscle tissue of biceps brachii1
ventricular zone1
ascending aorta1
descending thoracic aorta1
thoracic aorta1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
LMNA295ubiquitousmarkernipple, mucosa of stomach, skin of abdomen
SYNE2284ubiquitousmarkerventricular zone, skeletal muscle tissue of biceps brachii, ganglionic eminence
TMEM43287ubiquitousmarkerdescending thoracic aorta, thoracic aorta, ascending aorta

Protein interactions among cohort

Intra-cohort edges: 3.

Hub genes (top 10 by interactor count)

SymbolInteractor count
LMNA7,173
SYNE22,276
TMEM431,864

Intra-cohort edges

ABSources
LMNASYNE2string_interaction
LMNATMEM43string_interaction
SYNE2TMEM43string_interaction

Structural data

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

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
LMNAP0254528
SYNE2Q8WXH03

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
TMEM43Q9BTV489.92

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 18. Enrichment computed across 3 evidence-associated genes (2 with Reactome annotation).

Pathways by enrichment

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

PathwayCohort genesFoldFDRSample cohort genes
Meiotic synapsis2141.0×9e-04LMNA, SYNE2
Breakdown of the nuclear lamina11903.3×0.005LMNA
Depolymerization of the Nuclear Lamina1380.7×0.011LMNA
Initiation of Nuclear Envelope (NE) Reformation1300.5×0.011LMNA
IRE1alpha activates chaperones1259.6×0.011LMNA
Deregulated CDK5 triggers multiple neurodegenerative pathways in Alzheimer’s disease models1259.6×0.011LMNA
Nuclear Envelope Breakdown1228.4×0.011LMNA
Unfolded Protein Response (UPR)1178.4×0.013LMNA
Meiosis1142.8×0.014SYNE2
Oncogenic MAPK signaling1124.1×0.014LMNA
XBP1(S) activates chaperone genes1107.7×0.015LMNA
Reproduction195.2×0.016SYNE2
Signaling by BRAF and RAF1 fusions185.2×0.016LMNA
Diseases of signal transduction by growth factor receptors and second messengers128.4×0.045LMNA
Cellular responses to stress118.4×0.062LMNA
Cell Cycle118.0×0.062SYNE2
Cellular responses to stimuli115.7×0.066LMNA
Disease16.5×0.147LMNA

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
nuclear migration2488.5×2e-04LMNA, SYNE2
positive regulation of cell communication by electrical coupling12808.7×0.004TMEM43
nuclear migration along microfilament12808.7×0.004SYNE2
nucleokinesis involved in cell motility in cerebral cortex radial glia guided migration11872.4×0.004SYNE2
DNA double-strand break attachment to nuclear envelope11872.4×0.004LMNA
establishment or maintenance of microtubule cytoskeleton polarity11404.3×0.005LMNA
nuclear pore localization11123.5×0.005LMNA
negative regulation of mesenchymal cell proliferation1936.2×0.005LMNA
nuclear membrane organization1802.5×0.005TMEM43
protein localization to nuclear envelope1702.2×0.005LMNA
regulation of protein localization to nucleus1702.2×0.005LMNA
metal ion transport1624.1×0.005TMEM43
negative regulation of cardiac muscle hypertrophy in response to stress1624.1×0.005LMNA
ventricular cardiac muscle cell development1510.7×0.005LMNA
nuclear envelope organization1330.4×0.008LMNA
centrosome localization1295.6×0.008SYNE2
regulation of telomere maintenance1280.9×0.008LMNA
negative regulation of release of cytochrome c from mitochondria1267.5×0.008LMNA
regulation of cilium assembly1200.6×0.010SYNE2
double-strand break repair via nonhomologous end joining1140.4×0.013LMNA
negative regulation of extrinsic apoptotic signaling pathway1140.4×0.013LMNA
protein localization to nucleus1117.0×0.015LMNA
cellular senescence198.5×0.017LMNA
sodium ion transport190.6×0.017TMEM43
heterochromatin formation185.1×0.018LMNA
potassium ion transport163.8×0.023TMEM43
memory161.1×0.023TMEM43
muscle organ development155.6×0.024LMNA
regulation of cell migration152.5×0.025LMNA
protein import into nucleus148.0×0.026LMNA

Therapeutics

Drug target analysis

Approved (phase 4): 1 · Phase ≥3: 1 · Phased (≥1): 1 · Undrugged: 2

Druggability breadth: 2 of 3 evidence-associated genes (67%) 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
LMNABEPRIDIL

Top cohort targets by molecule count

SymbolMoleculesMax phase
LMNA8234
SYNE200
TMEM4300

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
BEPRIDIL4LMNA
PHENYLBUTAZONE4LMNA
CEFOTAXIME SODIUM4LMNA
DIENESTROL4LMNA
IFOSFAMIDE4LMNA
PROGESTERONE4LMNA
CLOTRIMAZOLE4LMNA
DAPSONE4LMNA
AMINOCAPROIC ACID4LMNA
FLUCONAZOLE4LMNA
COLCHICINE4LMNA
NABUMETONE4LMNA
OXAPROZIN4LMNA
BUMETANIDE4LMNA
GLIPIZIDE4LMNA
BROMFENAC4LMNA
ROPIVACAINE4LMNA
TIZANIDINE4LMNA
METAXALONE4LMNA
CARBAMAZEPINE4LMNA
SALMETEROL XINAFOATE4LMNA
AMIODARONE HYDROCHLORIDE4LMNA
METHYL SALICYLATE4LMNA
DIBUCAINE4LMNA
PHENELZINE4LMNA
HYDROCORTISONE ACETATE4LMNA
BRETYLIUM TOSYLATE4LMNA
IMIPRAMINE4LMNA
FURAZOLIDONE4LMNA
DROPERIDOL4LMNA

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

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

SymbolAssaysType breakdown
LMNA12Binding:9, Functional:3
TMEM431Binding:1

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

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)
BEPRIDIL4LMNA
PHENYLBUTAZONE4LMNA
CEFOTAXIME SODIUM4LMNA
DIENESTROL4LMNA
IFOSFAMIDE4LMNA
PROGESTERONE4LMNA
CLOTRIMAZOLE4LMNA
DAPSONE4LMNA
AMINOCAPROIC ACID4LMNA
FLUCONAZOLE4LMNA
COLCHICINE4LMNA
NABUMETONE4LMNA
OXAPROZIN4LMNA
BUMETANIDE4LMNA
GLIPIZIDE4LMNA
BROMFENAC4LMNA
ROPIVACAINE4LMNA
TIZANIDINE4LMNA
METAXALONE4LMNA
CARBAMAZEPINE4LMNA
SALMETEROL XINAFOATE4LMNA
AMIODARONE HYDROCHLORIDE4LMNA
METHYL SALICYLATE4LMNA
DIBUCAINE4LMNA
PHENELZINE4LMNA
HYDROCORTISONE ACETATE4LMNA
BRETYLIUM TOSYLATE4LMNA
IMIPRAMINE4LMNA
FURAZOLIDONE4LMNA
DROPERIDOL4LMNA

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)1LMNA
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug0
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug2SYNE2, TMEM43

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
SYNE20LMNA
TMEM431

Clinical trials & evidence

Clinical trials

Clinical trials: 1.

Phase distribution (across all retrieved trials)

PhaseTrials
Not specified1

Top trials by phase / activity

NCTPhaseStatusTitle
NCT05989620Not specifiedRECRUITINGLong-Term Development of Muscular Dystrophy Outcome Assessments