Interstitial lung disease 2

disease
On this page

Also known as fibrocystic pulmonary dysplasiafibrosing alveolitis, cryptogenicidiopathic pulmonary fibrosis, familialinterstitial pneumonitis, usualpulmonary fibrosis, idiopathic

Summary

Interstitial lung disease 2 (MONDO:0800497) is a disease caused by SFTPA2 (GenCC Strong), with 26 cohort genes and 2 clinical trials. The dominant Reactome pathway is Defective CSF2RB causes SMDP5 (4 cohort genes).

At a glance

  • Causal gene: SFTPA2 (GenCC Strong)
  • Cohort genes: 26
  • ClinVar variants: 194
  • Clinical trials: 2

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameinterstitial lung disease 2
Mondo IDMONDO:0800497
OMIM178500
DOIDDOID:0060971
UMLSC5561926
MedGen1794136
GARD0028066
Is cancer (heuristic)no

Also known as: fibrocystic pulmonary dysplasia · fibrosing alveolitis, cryptogenic · idiopathic pulmonary fibrosis, familial · interstitial pneumonitis, usual · pulmonary fibrosis, idiopathic

Data availability: 194 ClinVar variants · 2 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by etiologic mechanism › disease of genetic or genomic mechanism › hereditary disease › inherited interstitial lung disease › interstitial lung disease 2

Related subtypes (12): pulmonary fibrosis and/or bone marrow failure, telomere-related, hypersensitivity pneumonitis, familial, alveolar capillary dysplasia with misalignment of pulmonary veins, Niemann-Pick disease type B, interstitial lung disease due to ABCA3 deficiency, lung fibrosis-immunodeficiency-46,XX gonadal dysgenesis syndrome, Hermansky-Pudlak syndrome with pulmonary fibrosis, familial hypocalciuric hypercalcemia, SFTPC-related interstitial lung disease, interstitial lung disease 1, Rajab interstitial lung disease with brain calcifications, Lane Hamilton syndrome

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

194 retrieved; paginated sample, class counts are floors:

82 uncertain significance, 39 conflicting classifications of pathogenicity, 16 pathogenic, 15 benign, 11 benign/likely benign, 10 pathogenic/likely pathogenic, 9 likely benign, 6 likely pathogenic, 2 uncertain significance; association, 1 pathogenic/likely risk allele, 1 benign; risk factor, 1 not provided, 1 likely risk allele

ClinVarVariant (HGVS)GeneClassificationReview
203381NM_001089.3(ABCA3):c.875A>T (p.Glu292Val)ABCA3Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
831918NC_000005.10:g.(?218349)(1297373_?)delAHRRPathogeniccriteria provided, single submitter
12738NM_198253.3(TERT):c.219+1G>ALOC110806263Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
36945NM_198253.3(TERT):c.164T>A (p.Leu55Gln)LOC110806263Pathogenicno assertion criteria provided
7327NR_001566.3(TERC):n.98G>ALOC110806306Pathogenicno assertion criteria provided
831255NC_000005.10:g.(?218346)(1295046_?)delNKD2Pathogeniccriteria provided, single submitter
217284NM_001283009.2(RTEL1):c.2413+1G>CRTEL1Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
217517NM_001283009.2(RTEL1):c.958+2dupRTEL1Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
42018NM_001283009.1(RTEL1):c.3791G>A (p.Arg1264His)RTEL1Pathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
42020NM_001283009.2(RTEL1):c.2920C>T (p.Arg974Ter)RTEL1Pathogeniccriteria provided, multiple submitters, no conflicts
217283NM_001283009.2(RTEL1):c.2219_2227del (p.His740_Ile742del)RTEL1-TNFRSF6BPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
217518NM_001283009.2(RTEL1):c.1482-1G>ARTEL1-TNFRSF6BPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
217519NM_001283009.2(RTEL1):c.1546G>C (p.Val516Leu)RTEL1-TNFRSF6BPathogeniccriteria provided, single submitter
559538NM_001283009.2(RTEL1):c.3559C>T (p.Gln1187Ter)RTEL1-TNFRSF6BPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
13199NM_001098668.4(SFTPA2):c.692G>T (p.Gly231Val)SFTPA2Pathogenic/Likely risk alleleno assertion criteria provided
13200NM_001098668.4(SFTPA2):c.593T>C (p.Phe198Ser)SFTPA2Pathogenicno assertion criteria provided
1322020NM_001098668.4(SFTPA2):c.512A>T (p.Asn171Ile)SFTPA2Pathogenicno assertion criteria provided
1325405NM_001098668.4(SFTPA2):c.697T>A (p.Trp233Arg)SFTPA2Pathogenicno assertion criteria provided
1074267NC_000005.9:g.(?1253843)(1297488_?)delTERTPathogeniccriteria provided, single submitter
12736NM_198253.3(TERT):c.2594G>A (p.Arg865His)TERTPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
12737NM_198253.3(TERT):c.2240del (p.Val747fs)TERTPathogeniccriteria provided, single submitter
29899NM_198253.3(TERT):c.1892G>A (p.Arg631Gln)TERTPathogeniccriteria provided, multiple submitters, no conflicts
36944NM_198253.3(TERT):c.2583-2A>CTERTPathogenicno assertion criteria provided
39123NM_198253.3(TERT):c.*6_*182delTERTPathogenicno assertion criteria provided
410651NM_198253.3(TERT):c.336dup (p.Glu113fs)TERTPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
584005NC_000005.10:g.(?1280152)(1280344_?)delTERTPathogeniccriteria provided, single submitter
843002NM_198253.3(TERT):c.1434G>A (p.Trp478Ter)TERTPathogeniccriteria provided, single submitter
915844NM_001089.3(ABCA3):c.2883C>T (p.Gly961=)ABCA3Likely pathogeniccriteria provided, multiple submitters, no conflicts
915845NM_001089.3(ABCA3):c.4885dup (p.Ala1629fs)ABCA3Likely pathogeniccriteria provided, single submitter
559539NM_173842.3(IL1RN):c.63A>G (p.Ser21=)IL1RNLikely pathogeniccriteria provided, single submitter

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 7 · Orphanet: 52 · 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
SFTPA2StrongAutosomal dominantinterstitial lung disease 25
MUC5BLimitedAutosomal recessiveinterstitial lung disease2

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
SFTPA2Orphanet:2032Idiopathic pulmonary fibrosis
MUC5BOrphanet:171700Diffuse panbronchiolitis
MUC5BOrphanet:2032Idiopathic pulmonary fibrosis
MUC5BOrphanet:686465Fibrotic hypersensitivity pneumonitis
SFTPA1Orphanet:2032Idiopathic pulmonary fibrosis
SFTPCOrphanet:2032Idiopathic pulmonary fibrosis
SFTPCOrphanet:217566Chronic respiratory distress with surfactant metabolism deficiency
SFTPCOrphanet:440392Interstitial lung disease due to SP-C deficiency
SFTPCOrphanet:685082Pediatric acute respiratory distress syndrome
TERTOrphanet:146Differentiated thyroid carcinoma
TERTOrphanet:1501Adrenocortical carcinoma
TERTOrphanet:1775Dyskeratosis congenita
TERTOrphanet:2032Idiopathic pulmonary fibrosis
TERTOrphanet:2495Meningioma
TERTOrphanet:3322Hoyeraal-Hreidarsson syndrome
TERTOrphanet:457246Clear cell sarcoma of kidney
TERTOrphanet:618Familial melanoma
TERTOrphanet:88Idiopathic aplastic anemia
NKX2-1Orphanet:1429Benign hereditary chorea
NKX2-1Orphanet:146Differentiated thyroid carcinoma
NKX2-1Orphanet:209905Brain-lung-thyroid syndrome
NKX2-1Orphanet:95713Athyreosis
HPS4Orphanet:231500Hermansky-Pudlak syndrome due to BLOC-3 deficiency
RTEL1Orphanet:1775Dyskeratosis congenita
RTEL1Orphanet:2032Idiopathic pulmonary fibrosis
RTEL1Orphanet:3322Hoyeraal-Hreidarsson syndrome
DTNBP1Orphanet:231531Hermansky-Pudlak syndrome due to BLOC-1 deficiency
FAM13AOrphanet:2032Idiopathic pulmonary fibrosis
CTC1Orphanet:1775Dyskeratosis congenita
CTC1Orphanet:313838Coats plus syndrome
DSPOrphanet:154Familial isolated dilated cardiomyopathy
DSPOrphanet:158687Lethal acantholytic erosive disorder
DSPOrphanet:2032Idiopathic pulmonary fibrosis
DSPOrphanet:293165Skin fragility-woolly hair-palmoplantar keratoderma syndrome
DSPOrphanet:293888Inherited isolated arrhythmogenic cardiomyopathy, dominant-left variant
DSPOrphanet:293899Inherited isolated arrhythmogenic ventricular dysplasia, biventricular variant
DSPOrphanet:293910Inherited isolated arrhythmogenic cardiomyopathy, dominant-right variant
DSPOrphanet:369992Severe dermatitis-multiple allergies-metabolic wasting syndrome
DSPOrphanet:476096Erythrokeratodermia-cardiomyopathy syndrome
DSPOrphanet:50942Striate palmoplantar keratoderma
DSPOrphanet:65282Carvajal syndrome
ABCA3Orphanet:2032Idiopathic pulmonary fibrosis
ABCA3Orphanet:217563Neonatal acute respiratory distress syndrome
ABCA3Orphanet:440402Interstitial lung disease due to ABCA3 deficiency
ABCA3Orphanet:685082Pediatric acute respiratory distress syndrome
IL1RNOrphanet:210115Sterile multifocal osteomyelitis with periostitis and pustulosis
LAMA3Orphanet:2407Laryngo-onycho-cutaneous syndrome
LAMA3Orphanet:79402Intermediate generalized junctional epidermolysis bullosa
LAMA3Orphanet:79404Severe generalized junctional epidermolysis bullosa
LAMA4Orphanet:154Familial isolated dilated cardiomyopathy

Cohort genes → proteins

26 cohort genes, 24 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence26

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
SFTPA2HGNC:10799ENSG00000185303Q8IWL1Pulmonary surfactant-associated protein A2gencc,clinvar
MUC5BHGNC:7516ENSG00000117983Q9HC84Mucin-5Bgencc,clinvar
SFTPA1HGNC:10798ENSG00000122852Q8IWL2Pulmonary surfactant-associated protein A1clinvar
SFTPCHGNC:10802ENSG00000168484P11686Surfactant protein Cclinvar
TERTHGNC:11730ENSG00000164362O14746Telomerase reverse transcriptaseclinvar
NKX2-1HGNC:11825ENSG00000136352P43699Homeobox protein Nkx-2.1clinvar
HPS4HGNC:15844ENSG00000100099Q9NQG7BLOC-3 complex member HPS4clinvar
RTEL1HGNC:15888ENSG00000258366Q9NZ71Regulator of telomere elongation helicase 1clinvar
TOLLIPHGNC:16476ENSG00000078902Q9H0E2Toll-interacting proteinclinvar
NKD2HGNC:17046ENSG00000145506Q969F2Protein naked cuticle homolog 2clinvar
DTNBP1HGNC:17328ENSG00000047579Q96EV8Dysbindinclinvar
SFTA3HGNC:18387ENSG00000229415P0C7M3Surfactant-associated protein 3clinvar
RTKN2HGNC:19364ENSG00000182010Q8IZC4Rhotekin-2clinvar
FAM13AHGNC:19367ENSG00000138640O94988Protein FAM13Aclinvar
EXOC3-AS1HGNC:25175ENSG00000221990Q8N2X6Uncharacterized protein EXOC3-AS1clinvar
CTC1HGNC:26169ENSG00000178971Q2NKJ3CST complex subunit CTC1clinvar
DSPHGNC:3052ENSG00000096696P15924Desmoplakinclinvar
ABCA3HGNC:33ENSG00000167972Q99758Phospholipid-transporting ATPase ABCA3clinvar
AHRRHGNC:346ENSG00000063438A9YTQ3Aryl hydrocarbon receptor repressorclinvar
RTEL1-TNFRSF6BHGNC:44095ENSG00000026036RTEL1-TNFRSF6B readthrough (NMD candidate)clinvar
MUC5B-AS1HGNC:53936ENSG00000255177MUC5B antisense RNA 1clinvar
IL1RNHGNC:6000ENSG00000136689P18510Interleukin-1 receptor antagonist proteinclinvar
LAMA3HGNC:6483ENSG00000053747Q16787Laminin subunit alpha-3clinvar
LAMA4HGNC:6484ENSG00000112769Q16363Laminin subunit alpha-4clinvar
MMP1HGNC:7155ENSG00000196611P03956Interstitial collagenaseclinvar
MMP19HGNC:7165ENSG00000123342Q99542Matrix metalloproteinase-19clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
SFTPA2Pulmonary surfactant-associated protein A2In presence of calcium ions, it binds to surfactant phospholipids and contributes to lower the surface tension at the air-liquid interface in the alveoli of the mammalian lung and is essential for normal respiration.
MUC5BMucin-5BGel-forming mucin that is thought to contribute to the lubricating and viscoelastic properties of whole saliva and cervical mucus.
SFTPA1Pulmonary surfactant-associated protein A1In presence of calcium ions, it binds to surfactant phospholipids and contributes to lower the surface tension at the air-liquid interface in the alveoli of the mammalian lung and is essential for normal respiration.
SFTPCSurfactant protein CPulmonary surfactant associated proteins promote alveolar stability by lowering the surface tension at the air-liquid interface in the peripheral air spaces.
TERTTelomerase reverse transcriptaseTelomerase is a ribonucleoprotein enzyme essential for the replication of chromosome termini in most eukaryotes.
NKX2-1Homeobox protein Nkx-2.1Transcription factor that binds and activates the promoter of thyroid specific genes such as thyroglobulin, thyroperoxidase, and thyrotropin receptor.
HPS4BLOC-3 complex member HPS4Component of the BLOC-3 complex, a complex that acts as a guanine exchange factor (GEF) for RAB32 and RAB38, promotes the exchange of GDP to GTP, converting them from an inactive GDP-bound form into an active GTP-bound form.
RTEL1Regulator of telomere elongation helicase 1A probable ATP-dependent DNA helicase implicated in telomere-length regulation, DNA repair and the maintenance of genomic stability.
TOLLIPToll-interacting proteinComponent of the signaling pathway of IL-1 and Toll-like receptors.
NKD2Protein naked cuticle homolog 2Cell autonomous antagonist of the canonical Wnt signaling pathway.
DTNBP1DysbindinComponent of the BLOC-1 complex, a complex that is required for normal biogenesis of lysosome-related organelles (LRO), such as platelet dense granules and melanosomes.
SFTA3Surfactant-associated protein 3Putative surfactant protein.
RTKN2Rhotekin-2May play an important role in lymphopoiesis.
CTC1CST complex subunit CTC1Component of the CST complex proposed to act as a specialized replication factor promoting DNA replication under conditions of replication stress or natural replication barriers such as the telomere duplex.
DSPDesmoplakinA component of desmosome cell-cell junctions which are required for positive regulation of cellular adhesion.
ABCA3Phospholipid-transporting ATPase ABCA3Catalyzes the ATP-dependent transport of phospholipids such as phosphatidylcholine and phosphoglycerol from the cytoplasm into the lumen side of lamellar bodies, in turn participates in the lamellar bodies biogenesis and homeostasis of pul…
AHRRAryl hydrocarbon receptor repressorMediates dioxin toxicity and is involved in regulation of cell growth and differentiation.
IL1RNInterleukin-1 receptor antagonist proteinAnti-inflammatory antagonist of interleukin-1 family of proinflammatory cytokines such as interleukin-1beta/IL1B and interleukin-1alpha/IL1A.
LAMA3Laminin subunit alpha-3Binding 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.
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.
MMP1Interstitial collagenaseCleaves collagens of types I, II, and III at one site in the helical domain.
MMP19Matrix metalloproteinase-19Endopeptidase that degrades various components of the extracellular matrix, such as aggrecan and cartilage oligomeric matrix protein (comp), during development, haemostasis and pathological conditions (arthritic disease).

Protein-family classification

Druggable: 3 · Difficult: 4 · Unknown: 19 · Druggable fraction: 0.12

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/Unknown191.3×0.276
Protease22.8×0.395
Transporter13.0×0.476
Scaffold/PPI21.3×0.561
Transcription factor20.6×0.840

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
SFTPA2Other/UnknownnoC-type_lectin-like, C-type_lectin-like/link_sf, CTDL_fold
MUC5BOther/UnknownnoVWF_dom, VWF_type-D, TIL_dom
SFTPA1Other/UnknownnoC-type_lectin-like, C-type_lectin-like/link_sf, CTDL_fold
SFTPCOther/UnknownnoSP-C, BRICHOS_dom, Surfactant_protein_propep
TERTOther/UnknownnoRT_dom, Telomerase_RT, Telomerase_RBD
NKX2-1Transcription factornoHD, Homeodomain-like_sf, Homeobox_CS
HPS4Other/UnknownnoHPS4, CCZ1/INTU/HSP4_longin_1, CCZ1/INTU/HSP4_longin_3
RTEL1Other/UnknownnoHelicase-like_DEXD_c2, ATP-dep_Helicase_C, RAD3-like_helicase_DEAD
TOLLIPOther/UnknownnoC2_dom, CUE, UBA-like_sf
NKD2Other/UnknownnoEF_hand_dom, EF-hand-dom_pair, Nkd-like
DTNBP1Other/UnknownnoDysbindin
SFTA3Other/Unknownno
RTKN2Scaffold/PPInoPH_domain, HR1_rho-bd, PH-like_dom_sf
FAM13AOther/UnknownnoRhoGAP_dom, Rho_GTPase_activation_prot, FAM13
EXOC3-AS1Other/Unknownno
CTC1Other/UnknownnoCTC1, CTC1-like
DSPScaffold/PPInoPlectin_repeat, SH3_domain, Spectrin/alpha-actinin
ABCA3TransporteryesABC_transporter-like_ATP-bd, AAA+_ATPase, ABC2_TM
AHRRTranscription factornoPAS, bHLH_dom, PAS_fold
RTEL1-TNFRSF6BOther/Unknownno
MUC5B-AS1Other/Unknownno
IL1RNOther/UnknownnoIL-1_fam, IL-1RA/IL-36, IL1/FGF
LAMA3Other/UnknownnoLaminin_IV, EGF, Laminin_G
LAMA4Other/UnknownnoEGF, Laminin_G, LE_dom
MMP1Proteaseyes3.4.24.7Hemopexin-like_dom, Pept_M10_metallopeptidase, Peptidoglycan-bd-like
MMP19ProteaseyesHemopexin-like_dom, Pept_M10_metallopeptidase, Peptidoglycan-bd-like

Expression context

Cohort genes with no expression data: 0.

20 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)26
unknown0

Top tissues across cohort

TissueCohort genes
right lung5
cerebellar hemisphere4
right hemisphere of cerebellum4
lower lobe of lung3
gall bladder3
upper lobe of left lung3
adult organism2
lung2
stromal cell of endometrium2
left lobe of thyroid gland2
right lobe of thyroid gland2
thyroid gland2
primordial germ cell in gonad2
buccal mucosa cell2
visceral pleura1
mucosa of transverse colon1
trachea1
olfactory bulb1
type B pancreatic cell1
cerebellar cortex1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
SFTPA2159tissue_specificmarkerlower lobe of lung, adult organism, visceral pleura
MUC5B171tissue_specificmarkertrachea, gall bladder, mucosa of transverse colon
SFTPA1111tissue_specificmarkerright lung, upper lobe of left lung, lung
SFTPC208tissue_specificmarkerlower lobe of lung, right lung, adult organism
TERT105broadyesstromal cell of endometrium, type B pancreatic cell, olfactory bulb
NKX2-1101broadmarkerright lobe of thyroid gland, left lobe of thyroid gland, thyroid gland
HPS4261ubiquitousmarkercerebellar hemisphere, right hemisphere of cerebellum, cerebellar cortex
RTEL1134ubiquitousyessural nerve, right hemisphere of cerebellum, cerebellar hemisphere
TOLLIP263ubiquitousmarkerright frontal lobe, anterior cingulate cortex, cingulate cortex
NKD2130broadyesupper lobe of left lung, right lung, lung
DTNBP1249ubiquitousmarkertendon of biceps brachii, nucleus accumbens, putamen
SFTA3107tissue_specificmarkerright lobe of thyroid gland, thyroid gland, left lobe of thyroid gland
RTKN2152ubiquitousmarkerright lung, ventricular zone, primordial germ cell in gonad
FAM13A293ubiquitousmarkersecondary oocyte, oocyte, jejunal mucosa
EXOC3-AS1188ubiquitousyesprimordial germ cell in gonad, adenohypophysis, buccal mucosa cell
CTC1198ubiquitousmarkergranulocyte, right hemisphere of cerebellum, cerebellar hemisphere
DSP253ubiquitousmarkerskin of hip, upper leg skin, hair follicle
ABCA3222ubiquitousmarkerlower lobe of lung, upper lobe of lung, upper lobe of left lung
AHRR131ubiquitousyesleft testis, stromal cell of endometrium, right testis
RTEL1-TNFRSF6B135markerright hemisphere of cerebellum, cerebellar hemisphere, cerebellum
MUC5B-AS185yescolonic epithelium, gall bladder, tonsil
IL1RN220broadmarkerlower esophagus mucosa, buccal mucosa cell, palpebral conjunctiva
LAMA3239broadmarkerright lung, skin of leg, skin of abdomen
LAMA4268ubiquitousmarkerlower esophagus muscularis layer, lower esophagus, nerve
MMP1172broadmarkerepithelial cell of pancreas, pancreatic ductal cell, islet of Langerhans
MMP19222ubiquitousmarkerleft uterine tube, gall bladder, omental fat pad

Protein interactions among cohort

Intra-cohort edges: 11.

Hub genes (top 10 by interactor count)

SymbolInteractor count
TERT5,717
MMP12,933
DSP2,897
LAMA42,688
MUC5B2,659
IL1RN2,550
TOLLIP2,543
NKX2-12,403
RTEL12,324
LAMA32,195

Intra-cohort edges

ABSources
ABCA3NKX2-1string_interaction
ABCA3RTEL1string_interaction
ABCA3SFTPCstring_interaction
CTC1RTEL1string_interaction
CTC1TERTstring_interaction
DTNBP1HPS4string_interaction
FAM13ARTEL1biogrid_interaction
FAM13ARTEL1-TNFRSF6Bintact
MUC5BSFTPCstring_interaction
NKX2-1SFTPCstring_interaction
RTEL1TERTstring_interaction

Structural data

PDB: 13 · AlphaFold-only: 11 · No structure: 2

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
TERTO1474623
MMP1P0395615
CTC1Q2NKJ37
IL1RNP185105
DSPP159244
SFTPCP116863
RTEL1Q9NZ713
MUC5BQ9HC842
NKX2-1P436992
TOLLIPQ9H0E22
ABCA3Q997582
HPS4Q9NQG71
AHRRA9YTQ31

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
SFTPA2Q8IWL184.09
SFTPA1Q8IWL283.15
MMP19Q9954279.70
DTNBP1Q96EV875.92
LAMA4Q1636373.75
RTKN2Q8IZC470.33
SFTA3P0C7M361.17
FAM13AO9498861.00
NKD2Q969F256.15
EXOC3-AS1Q8N2X650.37
LAMA3Q16787

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 99. Enrichment computed across 26 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
Defective CSF2RB causes SMDP54326.3×1e-08SFTPA2, SFTPA1, SFTPC, SFTA3
Defective CSF2RA causes SMDP44326.3×1e-08SFTPA2, SFTPA1, SFTPC, SFTA3
Surfactant metabolism592.1×5e-08SFTPA2, SFTPA1, SFTPC, SFTA3, ABCA3
Diseases associated with surfactant metabolism3428.2×5e-07SFTPA2, SFTPA1, ABCA3
Diseases of metabolism416.1×0.002SFTPA2, MUC5B, SFTPA1, ABCA3
Extracellular matrix organization412.6×0.004LAMA3, LAMA4, MMP1, MMP19
Signal regulatory protein family interactions267.2×0.005SFTPA2, SFTPA1
Cell-Cell communication320.6×0.005SFTPA2, SFTPA1, LAMA3
Extension of Telomeres260.1×0.005TERT, RTEL1
MET promotes cell motility260.1×0.005LAMA3, LAMA4
Degradation of the extracellular matrix317.7×0.005LAMA3, MMP1, MMP19
Regulation of TLR by endogenous ligand249.6×0.005SFTPA2, SFTPA1
Attachment of bacteria to epithelial cells249.6×0.005LAMA3, LAMA4
Telomere Extension By Telomerase245.7×0.006TERT, RTEL1
Laminin interactions238.1×0.008LAMA3, LAMA4
MET activates PTK2 signaling238.1×0.008LAMA3, LAMA4
Telomere Maintenance236.8×0.008TERT, RTEL1
Defective ABCA3 causes SMDP31571.0×0.008ABCA3
Defective SFTPA2 causes IPF1571.0×0.008SFTPA2
Defective pro-SFTPC causes SMDP2 and RDS1571.0×0.008SFTPC
Signaling by MET231.7×0.008LAMA3, LAMA4
Formation of the dystrophin-glycoprotein complex (DGC)230.9×0.008LAMA3, LAMA4
Developmental Lineage of Pancreatic Ductal Cells222.8×0.014LAMA3, LAMA4
Chromosome Maintenance221.1×0.016TERT, RTEL1
Collagen degradation217.6×0.021MMP1, MMP19
Toll Like Receptor 2 (TLR2) Cascade217.3×0.021SFTPA2, SFTPA1
Toll Like Receptor TLR1:TLR2 Cascade216.8×0.022SFTPA2, SFTPA1
Non-integrin membrane-ECM interactions215.4×0.025LAMA3, LAMA4
ECM proteoglycans215.0×0.025LAMA3, LAMA4

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 23 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
respiratory gaseous exchange by respiratory system381.4×0.001SFTPA2, SFTPA1, SFTPC
telomere maintenance334.9×0.009TERT, RTEL1, CTC1
replicative senescence286.2×0.017TERT, CTC1
DNA strand displacement1732.7×0.021RTEL1
RNA-templated transcription1732.7×0.021TERT
DNA strand elongation1732.7×0.021TERT
positive regulation of protein homooligomerization1732.7×0.021ABCA3
positive regulation of eye pigmentation1732.7×0.021HPS4
siRNA transcription1732.7×0.021TERT
positive regulation of transdifferentiation1732.7×0.021TERT
negative regulation of telomere maintenance in response to DNA damage1732.7×0.021RTEL1
positive regulation of telomeric loop disassembly1732.7×0.021RTEL1
platelet dense granule organization258.6×0.021HPS4, DTNBP1
response to hormone237.6×0.021NKX2-1, MMP19
collagen catabolic process234.1×0.022MMP1, MMP19
RNA-templated DNA biosynthetic process1366.4×0.023TERT
developmental induction1366.4×0.023NKX2-1
positive regulation of hair cycle1366.4×0.023TERT
Golgi vesicle fusion to target membrane1366.4×0.023NKD2
positive regulation of glutamate neurotransmitter secretion in response to membrane depolarization1366.4×0.023DTNBP1
telomeric loop disassembly1366.4×0.023RTEL1
regulation of phosphatidylcholine metabolic process1366.4×0.023ABCA3
extracellular matrix disassembly231.9×0.023MMP1, MMP19
regulation of embryonic development228.7×0.023LAMA3, LAMA4
response to glucocorticoid228.2×0.023ABCA3, IL1RN
regulation of cell adhesion226.6×0.023LAMA3, LAMA4
cerebral cortex GABAergic interneuron differentiation1244.2×0.029NKX2-1
xenobiotic export from cell1244.2×0.029ABCA3
negative regulation of single-species biofilm formation in or on host organism1244.2×0.029MUC5B
mitotic telomere maintenance via semi-conservative replication1244.2×0.029RTEL1

Therapeutics

Drug target analysis

Approved (phase 4): 2 · Phase ≥3: 2 · Phased (≥1): 2 · Undrugged: 24

Druggability breadth: 7 of 26 evidence-associated genes (27%) 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
TERTBERBERINE
MMP1TILUDRONATE DISODIUM

Top cohort targets by molecule count

SymbolMoleculesMax phase
MMP1214
TERT104
SFTPA200
MUC5B00
SFTPA100
SFTPC00
NKX2-100
HPS400
RTEL100
TOLLIP00

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
BERBERINE4TERT
DOXORUBICIN4TERT
TILUDRONATE DISODIUM4MMP1
PRAZOSIN4MMP1
SULFASALAZINE4MMP1
IRINOTECAN4MMP1
NIMESULIDE4MMP1
DOXAZOSIN4MMP1
RESVERATROL3TERT
EPIGALOCATECHIN GALLATE3MMP1, TERT
PERIFOSINE3TERT
CAFFEIC ACID3MMP1
MARIMASTAT3MMP1
QUERCETIN3MMP1
PRINOMASTAT3MMP1
ISOMETAMIDIUM2TERT
HOMIDIUM BROMIDE2TERT
ALLICIN2TERT
OLEIC ACID2TERT
ETHACRIDINE2TERT
CIPEMASTAT2MMP1
ILOMASTAT2MMP1
APRATASTAT2MMP1
SOLIMASTAT2MMP1
TANOMASTAT2MMP1
BATIMASTAT2MMP1
(+)-SECOISOLARICIRESINOL2MMP1
CTS-10272MMP1
REBIMASTAT2MMP1
AMINOQUINURIDE2MMP1

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 1.

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

SymbolAssaysType breakdown
MMP1588Binding:574, ADMET:10, Functional:3, Toxicity:1
TERT391Binding:389, Functional:2
IL1RN26Binding:26
MMP194ADMET:2, Binding:1, Toxicity:1
DSP2Binding:2

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
MMP13.4.24.7interstitial collagenase

Cohort genes with high screening signal

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

SymbolChEMBL assays
TERT391
MMP1588

Pharmacogenomics

Cohort genes with a PharmGKB record: 23; 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)
BERBERINE4TERT
DOXORUBICIN4TERT
TILUDRONATE DISODIUM4MMP1
PRAZOSIN4MMP1
SULFASALAZINE4MMP1
IRINOTECAN4MMP1
NIMESULIDE4MMP1
DOXAZOSIN4MMP1
RESVERATROL3TERT
EPIGALOCATECHIN GALLATE3MMP1, TERT
PERIFOSINE3TERT
CAFFEIC ACID3MMP1
MARIMASTAT3MMP1
QUERCETIN3MMP1
PRINOMASTAT3MMP1
ISOMETAMIDIUM2TERT
HOMIDIUM BROMIDE2TERT
ALLICIN2TERT
OLEIC ACID2TERT
ETHACRIDINE2TERT
CIPEMASTAT2MMP1
ILOMASTAT2MMP1
APRATASTAT2MMP1
SOLIMASTAT2MMP1
TANOMASTAT2MMP1
BATIMASTAT2MMP1
(+)-SECOISOLARICIRESINOL2MMP1
CTS-10272MMP1
REBIMASTAT2MMP1
AMINOQUINURIDE2MMP1

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)2TERT, MMP1
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug1ABCA3
DDruggable family + AlphaFold only, no drug1MMP19
EDifficult family or no structure, no drug22SFTPA2, MUC5B, SFTPA1, SFTPC, NKX2-1, HPS4, RTEL1, TOLLIP, NKD2, DTNBP1 (+12 more)

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
SFTPA20
MUC5B0
SFTPA10
SFTPC0
NKX2-10
HPS40
RTEL10
TOLLIP0
NKD20
DTNBP10
SFTA30
RTKN20
FAM13A0
EXOC3-AS10
CTC10
DSP2
ABCA30
AHRR0
RTEL1-TNFRSF6B0
MUC5B-AS10
IL1RN26
LAMA30
LAMA40
MMP194

Clinical trials & evidence

Clinical trials

Clinical trials: 2.

Phase distribution (across all retrieved trials)

PhaseTrials
Not specified2

Top trials by phase / activity

NCTPhaseStatusTitle
NCT06644144Not specifiedRECRUITINGP4O2 ILD Extension
NCT06372353Not specifiedCOMPLETEDThe Effect Of Baduanjin Exercises In Patients With Idiopathic Pulmonary Fibrosis