Amyloidosis, hereditary systemic 1

disease
On this page

Summary

Amyloidosis, hereditary systemic 1 (MONDO:0971004) is a disease caused by TTR (GenCC Definitive), with 9 cohort genes. The dominant Reactome pathway is Striated Muscle Contraction (4 cohort genes).

At a glance

  • Causal gene: TTR (GenCC Definitive)
  • Cohort genes: 9
  • ClinVar variants: 387

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameamyloidosis, hereditary systemic 1
Mondo IDMONDO:0971004
OMIM105210
UMLSC2751492
MedGen414031
GARD0027098
Is cancer (heuristic)no

Data availability: 387 ClinVar variants · 2 GenCC gene-disease records.

Disease family

An umbrella term covering 2 Mondo subtypes.

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary diseaseinborn errors of metabolismhereditary amyloidosisfamilial amyloid neuropathyamyloidosis, hereditary systemic 1

Related subtypes (3): amyloidosis, hereditary systemic 3, amyloidosis, hereditary systemic 5, amyloidosis, hereditary systemic 6

Subtypes (2): ATTRV122I amyloidosis, ATTRV30M amyloidosis

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

387 retrieved; paginated sample, class counts are floors:

111 uncertain significance, 90 likely benign, 53 pathogenic, 43 conflicting classifications of pathogenicity, 43 pathogenic/likely pathogenic, 32 likely pathogenic, 10 benign/likely benign, 5 benign

ClinVarVariant (HGVS)GeneClassificationReview
1012273NM_000371.4(TTR):c.[239C>T];[424G>A]Pathogenicno assertion criteria provided
1066429NM_000371.4(TTR):c.179C>A (p.Thr60Asn)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1067499NM_000371.4(TTR):c.217G>A (p.Gly73Arg)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1067500NM_000371.4(TTR):c.251T>C (p.Phe84Ser)TTRPathogeniccriteria provided, multiple submitters, no conflicts
1068392NM_000371.4(TTR):c.186G>C (p.Glu62Asp)TTRPathogeniccriteria provided, single submitter
1073324NM_000371.4(TTR):c.325G>A (p.Glu109Lys)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1073325NM_000371.4(TTR):c.381T>G (p.Ile127Met)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1075407NM_000371.4(TTR):c.94C>G (p.Leu32Val)TTRPathogeniccriteria provided, multiple submitters, no conflicts
1075408NM_000371.4(TTR):c.182G>T (p.Trp61Leu)TTRPathogeniccriteria provided, single submitter
1178327NM_000371.4(TTR):c.214T>C (p.Ser72Pro)TTRPathogeniccriteria provided, multiple submitters, no conflicts
1294424NM_000371.4(TTR):c.293A>T (p.Tyr98Phe)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1333466NM_000371.4(TTR):c.425T>C (p.Val142Ala)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13417NM_000371.4(TTR):c.148G>A (p.Val50Met)TTRPathogeniccriteria provided, multiple submitters, no conflicts
13418NM_000371.4(TTR):c.157T>A (p.Phe53Ile)TTRPathogenicno assertion criteria provided
13419NM_000371.4(TTR):c.401A>G (p.Tyr134Cys)TTRPathogeniccriteria provided, multiple submitters, no conflicts
13420NM_000371.4(TTR):c.233T>A (p.Leu78His)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13421NM_000371.4(TTR):c.238A>G (p.Thr80Ala)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13422NM_000371.4(TTR):c.290C>A (p.Ser97Tyr)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13423NM_000371.4(TTR):c.311T>G (p.Ile104Ser)TTRPathogeniccriteria provided, multiple submitters, no conflicts
13426NM_000371.4(TTR):c.424G>A (p.Val142Ile)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13428NM_000371.4(TTR):c.185A>G (p.Glu62Gly)TTRPathogenicno assertion criteria provided
13429NM_000371.4(TTR):c.210T>G (p.Ser70Arg)TTRPathogeniccriteria provided, multiple submitters, no conflicts
13430NM_000371.4(TTR):c.149T>C (p.Val50Ala)TTRPathogeniccriteria provided, multiple submitters, no conflicts
13432NM_000371.4(TTR):c.166G>C (p.Ala56Pro)TTRPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13435NM_000371.4(TTR):c.233T>G (p.Leu78Arg)TTRPathogenicno assertion criteria provided
13436NM_000371.4(TTR):c.199G>C (p.Gly67Arg)TTRPathogenicno assertion criteria provided
13437NM_000371.4(TTR):c.133G>A (p.Ala45Thr)TTRPathogenicno assertion criteria provided
13438NM_000371.4(TTR):c.224T>C (p.Leu75Pro)TTRPathogenicno assertion criteria provided
13439NM_000371.4(TTR):c.209G>T (p.Ser70Ile)TTRPathogenicno assertion criteria provided
13440NM_000371.4(TTR):c.148G>C (p.Val50Leu)TTRPathogeniccriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 8 · Orphanet: 30 · 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
TTRDefinitiveAutosomal dominantamyloidosis, hereditary systemic 18

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
TTROrphanet:597939Euthyroid dysprealbuminemic hyperthyroxinemia
TTROrphanet:85447ATTRV30M amyloidosis
TTROrphanet:85451ATTRV122I amyloidosis
TNNI3Orphanet:154Familial isolated dilated cardiomyopathy
TNNI3Orphanet:75249Familial isolated restrictive cardiomyopathy
TTNOrphanet:140922Titin-related limb-girdle muscular dystrophy R10
TTNOrphanet:154Familial isolated dilated cardiomyopathy
TTNOrphanet:169186Autosomal recessive centronuclear myopathy
TTNOrphanet:178464Hereditary myopathy with early respiratory failure
TTNOrphanet:289377Early-onset myopathy with fatal cardiomyopathy
TTNOrphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
TTNOrphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
TTNOrphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
TTNOrphanet:324604Classic multiminicore myopathy
TTNOrphanet:334Hereditary atrial fibrillation
TTNOrphanet:466921Childhood-onset progressive contractures-limb-girdle weakness-muscle dystrophy syndrome
TTNOrphanet:609Tibial muscular dystrophy
TTNOrphanet:707983Early-onset autosomal recessive TTN-related distal myopathy
DSC3Orphanet:217407Hereditary hypotrichosis with recurrent skin vesicles
DSG2Orphanet:154Familial isolated dilated cardiomyopathy
DSG2Orphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
DSG2Orphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
DSG2Orphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
MYBPC3Orphanet:154Familial isolated dilated cardiomyopathy
MYBPC3Orphanet:54260Left ventricular noncompaction
PKP2Orphanet:130Brugada syndrome
PKP2Orphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
PKP2Orphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
PKP2Orphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
PKP2Orphanet:54260Left ventricular noncompaction

Cohort genes → proteins

9 cohort genes, 9 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence9

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
TTRHGNC:12405ENSG00000118271P02766Transthyretingencc,clinvar
TNNI3HGNC:11947ENSG00000129991P19429Troponin I, cardiac muscleclinvar
TTNHGNC:12403ENSG00000155657Q8WZ42Titinclinvar
DSC1HGNC:3035ENSG00000134765Q08554Desmocollin-1clinvar
DSC3HGNC:3037ENSG00000134762Q14574Desmocollin-3clinvar
DSG2HGNC:3049ENSG00000046604Q14126Desmoglein-2clinvar
MYBPC3HGNC:7551ENSG00000134571Q14896Myosin-binding protein C, cardiac-typeclinvar
MYL3HGNC:7584ENSG00000160808P08590Myosin light chain 3clinvar
PKP2HGNC:9024ENSG00000057294Q99959Plakophilin-2clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
TTRTransthyretinThyroid hormone-binding protein.
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.
TTNTitinKey component in the assembly and functioning of vertebrate striated muscles.
DSC1Desmocollin-1A component of desmosome cell-cell junctions which are required for positive regulation of cellular adhesion.
DSC3Desmocollin-3A component of desmosome cell-cell junctions which are required for positive regulation of cellular adhesion.
DSG2Desmoglein-2A component of desmosome cell-cell junctions which are required for positive regulation of cellular adhesion.
MYBPC3Myosin-binding protein C, cardiac-typeThick filament-associated protein located in the crossbridge region of vertebrate striated muscle a bands.
MYL3Myosin light chain 3Regulatory light chain of myosin.
PKP2Plakophilin-2A component of desmosome cell-cell junctions which are required for positive regulation of cellular adhesion.

Protein-family classification

Druggable: 2 · Difficult: 0 · Unknown: 7 · Druggable fraction: 0.22

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/Immunoglobulin13.2×0.282
Kinase13.1×0.282
Other/Unknown71.4×0.282

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
TTROther/UnknownnoTransthyretin/HIU_hydrolase, Transthyretin/HIU_hydrolase_d, Thyroxine_BS
TNNI3Other/UnknownnoTroponin, Troponin-I_N, Troponin_sf
TTNKinaseyes2.7.11.1Prot_kinase_dom, Ig_sub2, Ig_sub
DSC1Other/UnknownnoCadherin-like_dom, Desmosomal_cadherin, Cadherin_pro_dom
DSC3Other/UnknownnoCadherin_Y-type_LIR, Cadherin-like_dom, Desmosomal_cadherin
DSG2Other/UnknownnoCadherin-like_dom, Desmosomal_cadherin, Cadherin-like_sf
MYBPC3Antibody/ImmunoglobulinyesIg_sub2, Ig_sub, FN3_dom
MYL3Other/UnknownnoEF_hand_dom, EF-hand-dom_pair, CALM/Myosin/TropC-like
PKP2Other/UnknownnoArmadillo, ARM-like, ARM-type_fold

Expression context

Cohort genes with no expression data: 0.

9 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)9
unknown0

Top tissues across cohort

TissueCohort genes
apex of heart4
left ventricle myocardium2
right atrium auricular region2
upper leg skin2
heart right ventricle2
choroid plexus epithelium1
right lobe of liver1
type B pancreatic cell1
biceps brachii1
gluteal muscle1
skeletal muscle tissue of biceps brachii1
skin of hip1
upper arm skin1
gingiva1
gingival epithelium1
colonic mucosa1
jejunal mucosa1
mucosa of sigmoid colon1
cardiac atrium1
hindlimb stylopod muscle1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
TTR185broadmarkerchoroid plexus epithelium, type B pancreatic cell, right lobe of liver
TNNI3169broadmarkerapex of heart, left ventricle myocardium, right atrium auricular region
TTN223broadmarkerbiceps brachii, gluteal muscle, skeletal muscle tissue of biceps brachii
DSC1102tissue_specificmarkerupper leg skin, skin of hip, upper arm skin
DSC3177broadmarkerupper leg skin, gingival epithelium, gingiva
DSG2238ubiquitousmarkermucosa of sigmoid colon, colonic mucosa, jejunal mucosa
MYBPC3149tissue_specificmarkerapex of heart, right atrium auricular region, cardiac atrium
MYL3198broadmarkerapex of heart, heart right ventricle, hindlimb stylopod muscle
PKP2237ubiquitousmarkerheart right ventricle, apex of heart, left ventricle myocardium

Protein interactions among cohort

Intra-cohort edges: 11.

Hub genes (top 10 by interactor count)

SymbolInteractor count
TTR4,528
TTN4,237
MYL32,255
DSG22,033
PKP21,861
TNNI31,836
MYBPC31,800
DSC31,474
DSC1831

Intra-cohort edges

ABSources
DSC1DSG2intact
DSC3DSG2intact, string_interaction
DSC3PKP2string_interaction
DSG2MYBPC3string_interaction
DSG2PKP2string_interaction
MYBPC3MYL3string_interaction
MYBPC3TNNI3string_interaction
MYBPC3TTNstring_interaction
MYL3TNNI3string_interaction
MYL3TTNstring_interaction
TNNI3TTNstring_interaction

Structural data

PDB: 8 · AlphaFold-only: 1 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
TTRP02766462
TTNQ8WZ4264
TNNI3P1942939
MYBPC3Q1489617
DSG2Q1412612
MYL3P085903
DSC1Q085541
PKP2Q999591

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
DSC3Q1457475.53

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 16. Enrichment computed across 9 evidence-associated genes (9 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 9 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Striated Muscle Contraction4137.2×2e-07TNNI3, TTN, MYBPC3, MYL3
Formation of the cornified envelope439.0×2e-05DSC1, DSC3, DSG2, PKP2
Keratinization424.8×6e-05DSC1, DSC3, DSG2, PKP2
Defective visual phototransduction due to STRA6 loss of function1423.0×0.009TTR
Muscle contraction217.1×0.018MYBPC3, MYL3
Apoptotic cleavage of cell adhesion proteins1115.3×0.023DSG2
The canonical retinoid cycle in rods (twilight vision)157.7×0.039TTR
Retinoid metabolism and transport127.6×0.071TTR
Ion homeostasis122.7×0.077TNNI3
Non-integrin membrane-ECM interactions117.1×0.079TTR
RHOG GTPase cycle116.5×0.079DSG2
Neutrophil degranulation25.1×0.079TTR, DSC1
RAC2 GTPase cycle114.1×0.084DSG2
RAC3 GTPase cycle113.2×0.084DSG2
Amyloid fiber formation111.4×0.090TTR
Platelet degranulation19.8×0.098TTN

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 9 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
ventricular cardiac muscle tissue morphogenesis4312.1×3e-08TNNI3, MYBPC3, MYL3, PKP2
cardiac muscle contraction4178.3×2e-07TNNI3, TTN, MYBPC3, MYL3
cell-cell adhesion445.1×3e-05DSC1, DSC3, DSG2, PKP2
bundle of His cell-Purkinje myocyte adhesion involved in cell communication2535.0×9e-05DSG2, PKP2
desmosome organization2468.1×9e-05DSG2, PKP2
regulation of striated muscle contraction2468.1×9e-05MYBPC3, MYL3
regulation of ventricular cardiac muscle cell action potential2312.1×2e-04DSG2, PKP2
muscle filament sliding2234.1×3e-04TNNI3, TTN
homophilic cell-cell adhesion346.8×3e-04DSC1, DSC3, DSG2
skeletal muscle contraction2113.5×0.001TNNI3, TTN
sarcomere organization285.1×0.002TTN, MYBPC3
regulation of heart rate by cardiac conduction283.2×0.002DSG2, PKP2
maintenance of protein localization at cell tip11872.4×0.003PKP2
muscle contraction246.2×0.004TTN, MYL3
regulation of systemic arterial blood pressure by ischemic conditions1936.2×0.005TNNI3
desmosome maintenance1936.2×0.005DSC1
Purkinje myocyte development1936.2×0.005DSG2
regulation of muscle filament sliding1936.2×0.005MYBPC3
regulation of cell-substrate adhesion1624.1×0.005PKP2
skeletal muscle myosin thick filament assembly1624.1×0.005TTN
sarcomerogenesis1624.1×0.005TTN
positive regulation of protein localization to cell-cell junction1624.1×0.005DSG2
cell adhesion312.5×0.005DSC3, DSG2, MYBPC3
negative regulation of glomerular filtration1468.1×0.007TTR
negative regulation of endothelial cell differentiation1374.5×0.008DSG2
maintenance of animal organ identity1374.5×0.008PKP2
regulation of substrate adhesion-dependent cell spreading1374.5×0.008PKP2
skeletal muscle thin filament assembly1312.1×0.008TTN
intermediate filament bundle assembly1312.1×0.008PKP2
desmosome assembly1267.5×0.009PKP2

Therapeutics

Drug target analysis

Approved (phase 4): 1 · Phase ≥3: 1 · Phased (≥1): 2 · Undrugged: 7

Druggability breadth: 5 of 9 evidence-associated genes (56%) 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
TTRTRICLABENDAZOLE

Top cohort targets by molecule count

SymbolMoleculesMax phase
TTR294
DSC112
TNNI300
TTN00
DSC300
DSG200
MYBPC300
MYL300
PKP200

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
TRICLABENDAZOLE4TTR
AMLEXANOX4TTR
TOLCAPONE4TTR
DICLOFENAC4TTR
LEVOTHYROXINE4TTR
TAFAMIDIS4TTR
BENZIODARONE4TTR
BITHIONOL4TTR
BENZBROMARONE4TTR
ACORAMIDIS4TTR
GEMFIBROZIL4TTR
MECLOFENAMIC ACID4TTR
DASATINIB4TTR
DEXTROTHYROXINE4TTR
TRICLOSAN4TTR
DIFLUNISAL4TTR
CAFFEIC ACID3TTR
RESVERATROL3TTR
EPIGALOCATECHIN GALLATE3TTR
DIACEREIN3TTR
TOLFENAMIC ACID2TTR
LUTEOLIN2TTR
FLUFENAMIC ACID2TTR
XANTHOHUMOL2TTR
GENISTEIN2TTR
NIFLUMIC ACID2TTR
DAIDZEIN2TTR
PTEROSTILBENE2TTR
ACECLOFENAC2TTR
MOLIBRESIB2DSC1

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 1.

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

SymbolAssaysType breakdown
TTR423Binding:391, Functional:32
DSC16Binding:6
TNNI32Binding:2
TTN1Binding:1

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
TTN2.7.11.1non-specific serine/threonine protein kinase

Cohort genes with high screening signal

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

SymbolChEMBL assays
TTR423

Pharmacogenomics

Cohort genes with a PharmGKB record: 9; 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)
TRICLABENDAZOLE4TTR
AMLEXANOX4TTR
TOLCAPONE4TTR
DICLOFENAC4TTR
LEVOTHYROXINE4TTR
TAFAMIDIS4TTR
BENZIODARONE4TTR
BITHIONOL4TTR
BENZBROMARONE4TTR
ACORAMIDIS4TTR
GEMFIBROZIL4TTR
MECLOFENAMIC ACID4TTR
DASATINIB4TTR
DEXTROTHYROXINE4TTR
TRICLOSAN4TTR
DIFLUNISAL4TTR
CAFFEIC ACID3TTR
RESVERATROL3TTR
EPIGALOCATECHIN GALLATE3TTR
DIACEREIN3TTR
TOLFENAMIC ACID2TTR
LUTEOLIN2TTR
FLUFENAMIC ACID2TTR
XANTHOHUMOL2TTR
GENISTEIN2TTR
NIFLUMIC ACID2TTR
DAIDZEIN2TTR
PTEROSTILBENE2TTR
ACECLOFENAC2TTR
MOLIBRESIB2DSC1

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)1TTR
BPhased (≥1) drug, not yet approved1DSC1
CDruggable family + PDB, no drug2TTN, MYBPC3
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug5TNNI3, DSC3, DSG2, MYL3, PKP2

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
TNNI32
TTN1
DSC30
DSG20
MYBPC30
MYL30
PKP20

Clinical trials & evidence

Clinical trials

Clinical trials: 0.