Familial adenomatous polyposis 1

disease
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Also known as adenoma, periampullary, somaticadenomatous polyposis coliAPC-related adenomatous polyposisFAP1

Summary

Familial adenomatous polyposis 1 (MONDO:0021056) is a disease caused by APC (GenCC Definitive), with 7 cohort genes and 12 clinical trials. Top therapeutic interventions include guselkumab, sulindac, and celecoxib.

At a glance

  • Causal gene: APC (GenCC Definitive)
  • Cohort genes: 7
  • ClinVar variants: 12,375
  • Clinical trials: 12

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namefamilial adenomatous polyposis 1
Mondo IDMONDO:0021056
OMIM175100
DOIDDOID:0080409
UMLSC2713442
MedGen398651
GARD0025282
Is cancer (heuristic)no

Also known as: adenoma, periampullary, somatic · adenomatous polyposis coli · APC-related adenomatous polyposis · familial adenomatous polyposis 1 · FAP1

Data availability: 12,375 ClinVar variants · 104 ClinGen variant curations · 4 GenCC gene-disease records.

Disease family

An umbrella term covering 1 Mondo subtype.

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary diseasehereditary neoplastic syndromeintestinal polyposis syndromeclassic or attenuated familial adenomatous polyposisfamilial adenomatous polyposis 1

Related subtypes (7): familial adenomatous polyposis 2, familial adenomatous polyposis 3, attenuated familial adenomatous polyposis, AXIN2-related attenuated familial adenomatous polyposis, Polymerase proofreading-related adenomatous polyposis, classic familial adenomatous polyposis, familial adenomatous polyposis 4

Subtypes (1): APC-related attenuated familial adenomatous polyposis

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:

558 uncertain significance, 16 pathogenic, 11 conflicting classifications of pathogenicity, 6 benign/likely benign, 4 likely pathogenic, 3 pathogenic/likely pathogenic, 2 likely benign

ClinVarVariant (HGVS)GeneClassificationReview
1002757NM_001127511.3(APC):c.-40G>AAPCPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1012188NM_000038.6(APC):c.1908_1909dup (p.Gly637fs)APCPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1012191NM_000038.6(APC):c.1490dup (p.Arg498fs)APCPathogeniccriteria provided, single submitter
1012192NM_000038.6(APC):c.1744G>T (p.Glu582Ter)APCPathogeniccriteria provided, single submitter
1012193NM_000038.6(APC):c.2510del (p.Ser836_Ser837insTer)APCPathogeniccriteria provided, single submitter
1048837NM_000038.6(APC):c.1742_1743del (p.Lys581fs)APCPathogenicno assertion criteria provided
1049077NM_000038.6(APC):c.2893_2896del (p.Asn965fs)APCPathogeniccriteria provided, multiple submitters, no conflicts
1049123NM_000038.6(APC):c.5782del (p.Gln1928fs)APCPathogeniccriteria provided, single submitter
1049151NM_000038.6(APC):c.442del (p.Asp149fs)APCPathogenicno assertion criteria provided
1049324NM_000038.6(APC):c.-2_135+1274delAPCPathogenicno assertion criteria provided
1049327NM_000038.6(APC):c.3411del (p.Asp1137fs)APCPathogenicno assertion criteria provided
1049668NM_000038.6(APC):c.4945dup (p.Ile1649fs)APCPathogenicno assertion criteria provided
1049772NM_000038.6(APC):c.1409-2delAPCPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
1050064NM_000038.6(APC):c.841dup (p.Thr281fs)APCPathogeniccriteria provided, single submitter
1050121NM_000038.6(APC):c.3260_3263dup (p.Lys1088fs)APCPathogenicno assertion criteria provided
1050316NM_000038.6(APC):c.2928_2929del (p.Gly977fs)APCPathogeniccriteria provided, multiple submitters, no conflicts
1050551NM_000038.6(APC):c.4606G>T (p.Glu1536Ter)APCPathogeniccriteria provided, single submitter
1050615NM_000038.6(APC):c.230T>A (p.Leu77Ter)APCPathogeniccriteria provided, single submitter
1050660NM_000038.6(APC):c.4161T>A (p.Cys1387Ter)APCPathogeniccriteria provided, single submitter
1012189NM_000038.6(APC):c.2743del (p.Cys914_Val915insTer)APCLikely pathogeniccriteria provided, single submitter
1012190NM_000038.6(APC):c.3991A>T (p.Arg1331Ter)APCLikely pathogeniccriteria provided, single submitter
1025291NM_000038.6(APC):c.933G>C (p.Lys311Asn)APCLikely pathogenicreviewed by expert panel
1050028NM_000038.6(APC):c.423-8A>GAPCLikely pathogenicreviewed by expert panel
1004817NM_000038.6(APC):c.2186T>C (p.Leu729Pro)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1017874NM_000038.6(APC):c.531T>C (p.Asn177=)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1018693NM_000038.6(APC):c.5600T>A (p.Phe1867Tyr)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1023798NM_000038.6(APC):c.4475C>A (p.Ala1492Asp)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1025685NM_000038.6(APC):c.6481A>C (p.Ser2161Arg)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1036972NM_000038.6(APC):c.827A>G (p.Asn276Ser)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications
1041349NM_000038.6(APC):c.4021A>G (p.Ser1341Gly)APCConflicting classifications of pathogenicitycriteria provided, conflicting classifications

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 15 · Orphanet: 10 · 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
APCDefinitiveAutosomal dominantfamilial adenomatous polyposis 115

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
APCOrphanet:220460Attenuated familial adenomatous polyposis
APCOrphanet:2615845q22 microdeletion syndrome
APCOrphanet:314022Gastric adenocarcinoma and proximal polyposis of the stomach
APCOrphanet:3258Cenani-Lenz syndrome
APCOrphanet:873Desmoid tumor
SRP19Orphanet:486Autosomal dominant severe congenital neutropenia
STK11Orphanet:2869Peutz-Jeghers syndrome
MUTYHOrphanet:247798MUTYH-related polyposis
MUTYHOrphanet:440437Familial colorectal cancer Type X
NTHL1Orphanet:454840NTHL1-related polyposis

Cohort genes → proteins

7 cohort genes, 7 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence7

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
APCHGNC:583ENSG00000134982P25054Adenomatous polyposis coli proteingencc,clinvar
SRP19HGNC:11300ENSG00000153037P09132Signal recognition particle 19 kDa proteinclinvar
STK11HGNC:11389ENSG00000118046Q15831Serine/threonine-protein kinase STK11clinvar
PCDHB16HGNC:14546ENSG00000272674Q9NRJ7Protocadherin beta-16clinvar
REEP5HGNC:30077ENSG00000129625Q00765Receptor expression-enhancing protein 5clinvar
MUTYHHGNC:7527ENSG00000132781Q9UIF7Adenine DNA glycosylaseclinvar
NTHL1HGNC:8028ENSG00000065057P78549Endonuclease III-like protein 1clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
APCAdenomatous polyposis coli proteinTumor suppressor.
SRP19Signal recognition particle 19 kDa proteinComponent of the signal recognition particle (SRP) complex, a ribonucleoprotein complex that mediates the cotranslational targeting of secretory and membrane proteins to the endoplasmic reticulum (ER).
STK11Serine/threonine-protein kinase STK11Tumor suppressor serine/threonine-protein kinase that controls the activity of AMP-activated protein kinase (AMPK) family members, thereby playing a role in various processes such as cell metabolism, cell polarity, apoptosis and DNA damage…
PCDHB16Protocadherin beta-16Potential calcium-dependent cell-adhesion protein.
REEP5Receptor expression-enhancing protein 5Plays an essential role in heart function and development by regulating the organization and function of the sarcoplasmic reticulum in cardiomyocytes.
MUTYHAdenine DNA glycosylaseInvolved in oxidative DNA damage repair.
NTHL1Endonuclease III-like protein 1Bifunctional DNA N-glycosylase with associated apurinic/apyrimidinic (AP) lyase function that catalyzes the first step in base excision repair (BER), the primary repair pathway for the repair of oxidative DNA damage.

Protein-family classification

Druggable: 2 · Difficult: 0 · Unknown: 5 · Druggable fraction: 0.29

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
Kinase14.0×0.456
Enzyme (other)11.7×0.456
Other/Unknown51.3×0.456

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
APCOther/UnknownnoArmadillo, APC_rpt, SAMP
SRP19Other/UnknownnoSignal_recog_particle_SRP19, SRP19-like_sf
STK11Kinaseyes2.7.11.1Prot_kinase_dom, Ser/Thr_kinase_AS, Kinase-like_dom_sf
PCDHB16Other/UnknownnoCadherin-like_dom, Cadherin_N, Cadherin-like_sf
REEP5Other/UnknownnoTB2_DP1_HVA22
MUTYHOther/UnknownnoNUDIX_hydrolase_dom, HhH_motif, HhH-GPD_domain
NTHL1Enzyme (other)yes4.2.99.18HhH_motif, HhH-GPD_domain, Endonuclease3_FeS-loop_motif

Expression context

Cohort genes with no expression data: 0.

7 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)7
unknown0

Top tissues across cohort

TissueCohort genes
medial globus pallidus1
substantia nigra pars compacta1
substantia nigra pars reticulata1
adenohypophysis1
body of pancreas1
islet of Langerhans1
hindlimb stylopod muscle1
left testis1
right testis1
caput epididymis1
cauda epididymis1
corpus epididymis1
endothelial cell1
lateral nuclear group of thalamus1
middle temporal gyrus1
cerebellar cortex1
cerebellar hemisphere1
right hemisphere of cerebellum1
apex of heart1
mucosa of transverse colon1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
APC297ubiquitousmarkersubstantia nigra pars compacta, substantia nigra pars reticulata, medial globus pallidus
SRP19169ubiquitousmarkeradenohypophysis, body of pancreas, islet of Langerhans
STK11238ubiquitousmarkerleft testis, right testis, hindlimb stylopod muscle
PCDHB16191broadmarkercorpus epididymis, caput epididymis, cauda epididymis
REEP5301ubiquitousmarkermiddle temporal gyrus, lateral nuclear group of thalamus, endothelial cell
MUTYH134ubiquitousmarkercerebellar hemisphere, cerebellar cortex, right hemisphere of cerebellum
NTHL1211ubiquitousmarkerright lobe of liver, apex of heart, mucosa of transverse colon

Protein interactions among cohort

Intra-cohort edges: 4.

Hub genes (top 10 by interactor count)

SymbolInteractor count
STK115,146
APC2,903
SRP192,099
NTHL11,994
MUTYH1,815
REEP51,764
PCDHB16489

Intra-cohort edges

ABSources
APCREEP5string_interaction
APCSRP19string_interaction
MUTYHNTHL1string_interaction
REEP5SRP19string_interaction

Structural data

PDB: 5 · AlphaFold-only: 2 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
APCP2505431
SRP19P091329
STK11Q158314
MUTYHQ9UIF73
NTHL1P785492

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
PCDHB16Q9NRJ782.84
REEP5Q0076578.88

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 55. Enrichment computed across 7 evidence-associated genes (5 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 5 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Displacement of DNA glycosylase by APEX12415.3×5e-04MUTYH, NTHL1
APC truncation mutants are not K63 polyubiquitinated12284.0×0.004APC
Defective MUTYH substrate binding12284.0×0.004MUTYH
Defective MUTYH substrate processing12284.0×0.004MUTYH
Defective NTHL1 substrate processing12284.0×0.004NTHL1
Defective NTHL1 substrate binding12284.0×0.004NTHL1
AMPK inhibits chREBP transcriptional activation activity1285.5×0.019STK11
Signaling by AXIN mutants1207.6×0.019APC
Signaling by CTNNB1 phospho-site mutants1207.6×0.019APC
Signaling by APC mutants1207.6×0.019APC
Signaling by AMER1 mutants1207.6×0.019APC
APC truncation mutants have impaired AXIN binding1163.1×0.019APC
AXIN missense mutants destabilize the destruction complex1163.1×0.019APC
Truncations of AMER1 destabilize the destruction complex1163.1×0.019APC
Signaling by GSK3beta mutants1152.3×0.019APC
CTNNB1 S33 mutants aren’t phosphorylated1152.3×0.019APC
CTNNB1 S37 mutants aren’t phosphorylated1152.3×0.019APC
CTNNB1 S45 mutants aren’t phosphorylated1152.3×0.019APC
CTNNB1 T41 mutants aren’t phosphorylated1152.3×0.019APC
FOXO-mediated transcription of cell death genes1142.8×0.019STK11
Beta-catenin phosphorylation cascade1134.3×0.019APC
Signaling by WNT in cancer1120.2×0.021APC
Apoptotic cleavage of cellular proteins195.2×0.024APC
Apoptotic execution phase195.2×0.024APC
Energy dependent regulation of mTOR by LKB1-AMPK178.8×0.028STK11
Disassembly of the destruction complex and recruitment of AXIN to the membrane171.4×0.029APC
FOXO-mediated transcription167.2×0.030STK11
Ovarian tumor domain proteases155.7×0.035APC
MTOR signalling153.1×0.035STK11
Deactivation of the beta-catenin transactivating complex146.6×0.039APC

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 7 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
positive regulation of vesicle transport along microtubule12407.4×0.018STK11
SRP-dependent cotranslational protein targeting to membrane, signal sequence recognition1601.9×0.018SRP19
depurination1601.9×0.018MUTYH
base-excision repair, AP site formation1481.5×0.018NTHL1
regulation of microtubule-based movement1401.2×0.018APC
negative regulation of epithelial cell proliferation involved in prostate gland development1401.2×0.018STK11
endoplasmic reticulum membrane organization1343.9×0.018REEP5
negative regulation of cell cycle G1/S phase transition1343.9×0.018APC
positive regulation of protein localization to centrosome1343.9×0.018APC
negative regulation of cyclin-dependent protein serine/threonine kinase activity1300.9×0.018APC
Golgi localization1300.9×0.018STK11
epithelial cell proliferation involved in prostate gland development1300.9×0.018STK11
regulation of microtubule-based process1267.5×0.018APC
depyrimidination1267.5×0.018NTHL1
cotranslational protein targeting to membrane1240.7×0.018SRP19
regulation of attachment of spindle microtubules to kinetochore1240.7×0.018APC
dendrite extension1240.7×0.018STK11
heart valve development1218.9×0.018APC
activation of protein kinase activity1218.9×0.018STK11
positive thymic T cell selection1200.6×0.018STK11
G1 to G0 transition1200.6×0.018STK11
cellular response to UV-B1200.6×0.018STK11
positive regulation of pseudopodium assembly1185.2×0.018APC
anoikis1185.2×0.018STK11
negative regulation of canonical Wnt signaling pathway233.7×0.018APC, STK11
vasculature development1160.5×0.020STK11
negative regulation of necroptotic process1141.6×0.021MUTYH
DNA damage response215.3×0.021APC, STK11
peptidyl-threonine phosphorylation1126.7×0.022STK11
regulation of Wnt signaling pathway1126.7×0.022STK11

Therapeutics

Drug target analysis

Approved (phase 4): 1 · Phase ≥3: 1 · Phased (≥1): 1 · Undrugged: 6

Druggability breadth: 6 of 7 evidence-associated genes (86%) 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
STK11FEDRATINIB

Top cohort targets by molecule count

SymbolMoleculesMax phase
STK11174
APC00
SRP1900
PCDHB1600
REEP500
MUTYH00
NTHL100

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
FEDRATINIB4STK11
PACRITINIB4STK11
NINTEDANIB4STK11
SUNITINIB4STK11
MIDOSTAURIN4STK11
DINACICLIB3STK11
DOVITINIB3STK11
LESTAURTINIB3STK11
RUBOXISTAURIN3STK11
AZD-14802STK11
SU-0148132STK11
R-4062STK11
TOZASERTIB2STK11
PF-005622711STK11
KW-24491STK11
PF-037583091STK11
XL-2281STK11

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 2.

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

SymbolAssaysType breakdown
STK11244Binding:244
APC24Binding:24
NTHL18Binding:7, Functional:1
SRP192Binding:2
REEP51Binding:1
MUTYH1Functional:1

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
STK112.7.11.1non-specific serine/threonine protein kinase
NTHL14.2.99.18DNA-(apurinic or apyrimidinic site) lyase

Cohort genes with high screening signal

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

SymbolChEMBL assays
STK11244

Pharmacogenomics

Cohort genes with a PharmGKB record: 7; with CPIC/DPWG dosing guidelines: 0.

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

Chemical tractability of cohort targets

17 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)
FEDRATINIB4STK11
PACRITINIB4STK11
NINTEDANIB4STK11
SUNITINIB4STK11
MIDOSTAURIN4STK11
DINACICLIB3STK11
DOVITINIB3STK11
LESTAURTINIB3STK11
RUBOXISTAURIN3STK11
AZD-14802STK11
SU-0148132STK11
R-4062STK11
TOZASERTIB2STK11
PF-005622711STK11
KW-24491STK11
PF-037583091STK11
XL-2281STK11

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)1STK11
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug1NTHL1
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug5APC, SRP19, PCDHB16, REEP5, MUTYH

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
APC24
SRP192
PCDHB160
REEP51
MUTYH1
NTHL18

Clinical trials & evidence

Clinical trials

Clinical trials: 12.

Phase distribution (across all retrieved trials)

PhaseTrials
Not specified4
PHASE23
PHASE13
PHASE41
PHASE31

Top trials by phase / activity

NCTPhaseStatusTitle
NCT00140894PHASE4TERMINATEDA Study of Rofecoxib in Familial Adenomatous Polyposis (FAP) (0966-205)(TERMINATED)
NCT00585312PHASE3TERMINATEDTrial In Pediatric Patients With Familial Adenomatous Polyposis (FAP)
NCT00319007PHASE2UNKNOWNInfluence of Sulindac and Probiotics on the Development of Pouch Adenomas in Patients With Familial Adenomatous Polyposis
NCT01187901PHASE2COMPLETEDA Clinical Trial of COX and EGFR Inhibition in Familial Polyposis Patients
NCT03095703PHASE2COMPLETEDSirolimus and Familial Adenomatous Polyposis (FAP)
NCT02113202PHASE1COMPLETEDMolecular Fluorescence Endoscopy in Patients With Familial Adenomatous Polyposis, Using Bevacizumab-IRDye800CW
NCT03649971PHASE1COMPLETEDA Study of Guselkumab in Participants With Familial Adenomatous Polyposis
NCT05014360PHASE1COMPLETEDA Study of JNJ-64251330 in Participants With Familial Adenomatous Polyposis
NCT06435533Not specifiedRECRUITINGCold Atmospheric Plasma for the Endoscopic Treatment of Duodenal Polyps in Patients With Familial Adenomatous Polyposis
NCT01604564Not specifiedCOMPLETEDRegistry With Information About Colitis Ulcerosa and Familial Adenomatous Polyposis Patients
NCT03027401Not specifiedWITHDRAWNClinical Sequencing of Cancer and Tissue Repository: OncoGenomics
NCT04948398Not specifiedNO_LONGER_AVAILABLEPost-Trial Access for Guselkumab in Participants With Familial Adenomatous Polyposis

Drugs tested across these trials (top 30)

MoleculeMax phaseTrials referencing
GUSELKUMAB42
SULINDAC42
CELECOXIB41
INULIN41