Early-onset non-syndromic cataract

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Summary

Early-onset non-syndromic cataract (MONDO:0011060) is a disease (an umbrella term covering 29 Mondo subtypes) with 9 cohort genes.

At a glance

  • Umbrella term: 29 Mondo subtypes
  • Cohort genes: 9
  • ClinVar variants: 7

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical nameearly-onset non-syndromic cataract
Mondo IDMONDO:0011060
OMIM601371
Orphanet91492
ICD-111080602978
UMLSC1832423
MedGen371326
GARD0016801
Is cancer (heuristic)no

Data availability: 7 ClinVar variants · 3 GenCC gene-disease records.

Disease family

An umbrella term covering 29 Mondo subtypes.

Classification path: disease › human disease › disease by body system or component › disorder of orbital regioneye disorderlens disordercataractearly-onset non-syndromic cataract

Related subtypes (28): immature cataract, diabetic cataract, mature cataract, tetanic cataract, myotonic cataract, senile cataract, diabetes mellitus type 2 associated cataract, cataract 4 multiple types, cataract 29, cataract 1 multiple types, cataract 3 multiple types, cataract 9 multiple types, cataract 28, cataract 18, cataract 12 multiple types, cataract 34 multiple types, cataract 36, bhaskar jagannathan syndrome, autosomal dominant cataract, craniostenosis cataract, Kozlowski Rafinski Klicharska syndrome, cataract 49, cataract 48, hypermature cataract, nuclear cataract, cortical cataract, cataract 2, multiple types, cataract 50 with or without glaucoma

Subtypes (29): cataract 32 multiple types, cataract 8 multiple types, cataract 42, cataract 20 multiple types, cataract 6 multiple types, cataract 13 with adult I phenotype, cataract 5 multiple types, cataract 46 juvenile-onset, cataract 40, cataract 10 multiple types, cataract 14 multiple types, pulverulent cataract, cataract 31 multiple types, cataract 26 multiple types, cataract 22 multiple types, cataract 21 multiple types, cataract 23, cataract 11 multiple types, cataract 33, cataract 17 multiple types, cataract 38, cataract 39 multiple types, cataract 15 multiple types, cataract 19 multiple types, cataract 43, cataract 44, cataract 45, early-onset partial cataract, total early-onset cataract

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

7 retrieved; paginated sample, class counts are floors:

3 pathogenic, 2 uncertain significance, 1 pathogenic/likely pathogenic, 1 likely pathogenic

ClinVarVariant (HGVS)GeneClassificationReview
4796486NM_001887.4(CRYBB1):c.576-637_587delCRYBA4Pathogeniccriteria provided, single submitter
4796487NM_001886.3(CRYBA4):c.220T>C (p.Tyr74His)CRYBA4Pathogeniccriteria provided, single submitter
2574672NM_001353214.3(DYM):c.916C>T (p.Gln306Ter)DYMPathogenic/Likely pathogeniccriteria provided, multiple submitters, no conflicts
215406NM_006005.3(WFS1):c.1243_1245del (p.Val415del)WFS1Pathogeniccriteria provided, multiple submitters, no conflicts
4796489NM_001395002.1(MAP4K4):c.298C>T (p.Gln100Ter)MAP4K4Likely pathogeniccriteria provided, single submitter
4796490NM_020989.4(CRYGC):c.419G>C (p.Arg140Pro)CRYGCUncertain significancecriteria provided, single submitter
1305889NM_021954.4(GJA3):c.161G>A (p.Cys54Tyr)GJA3Uncertain significancecriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 28 · Orphanet: 24 · 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
BFSP2DefinitiveAutosomal dominantcataract 12 multiple types9
CRYBB3DefinitiveAutosomal recessivecataract 22 multiple types9
EPHA2DefinitiveAutosomal dominantcataract 6 multiple types10

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
BFSP2Orphanet:441452Early-onset lamellar cataract
BFSP2Orphanet:98984Pulverulent cataract
BFSP2Orphanet:98985Early-onset sutural cataract
CRYBB3Orphanet:98988Early-onset anterior polar cataract
CRYBB3Orphanet:98991Early-onset nuclear cataract
EPHA2Orphanet:441447Early-onset posterior subcapsular cataract
EPHA2Orphanet:98991Early-onset nuclear cataract
EPHA2Orphanet:98993Early-onset posterior polar cataract
EPHA2Orphanet:98994Total early-onset cataract
WFS1Orphanet:3463Wolfram syndrome
WFS1Orphanet:411590Wolfram-like syndrome
WFS1Orphanet:90635Rare autosomal dominant non-syndromic sensorineural deafness type DFNA
WFS1Orphanet:98991Early-onset nuclear cataract
DYMOrphanet:178355Smith-McCort dysplasia
DYMOrphanet:239Dyggve-Melchior-Clausen disease
CRYBA4Orphanet:1377Cataract-microcornea syndrome
CRYBA4Orphanet:441452Early-onset lamellar cataract
CRYGCOrphanet:1377Cataract-microcornea syndrome
CRYGCOrphanet:441452Early-onset lamellar cataract
CRYGCOrphanet:98984Pulverulent cataract
CRYGCOrphanet:98991Early-onset nuclear cataract
GJA3Orphanet:98984Pulverulent cataract
GJA3Orphanet:98991Early-onset nuclear cataract
GJA3Orphanet:98993Early-onset posterior polar cataract

Cohort genes → proteins

9 cohort genes, 9 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence9

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
BFSP2HGNC:1041ENSG00000170819Q13515Phakiningencc
CRYBB3HGNC:2400ENSG00000100053P26998Beta-crystallin B3gencc
EPHA2HGNC:3386ENSG00000142627P29317Ephrin type-A receptor 2gencc
WFS1HGNC:12762ENSG00000109501O76024Wolframinclinvar
DYMHGNC:21317ENSG00000141627Q7RTS9Dymeclinclinvar
CRYBA4HGNC:2396ENSG00000196431P53673Beta-crystallin A4clinvar
CRYGCHGNC:2410ENSG00000163254P07315Gamma-crystallin Cclinvar
GJA3HGNC:4277ENSG00000121743Q9Y6H8Gap junction alpha-3 proteinclinvar
MAP4K4HGNC:6866ENSG00000071054O95819Mitogen-activated protein kinase kinase kinase kinase 4clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
BFSP2PhakininRequired for the correct formation of lens intermediate filaments as part of a complex composed of BFSP1, BFSP2 and CRYAA.
CRYBB3Beta-crystallin B3Crystallins are the dominant structural components of the vertebrate eye lens.
EPHA2Ephrin type-A receptor 2Receptor tyrosine kinase which binds promiscuously membrane-bound ephrin-A family ligands residing on adjacent cells, leading to contact-dependent bidirectional signaling into neighboring cells.
WFS1WolframinParticipates in the regulation of cellular Ca(2+) homeostasis, at least partly, by modulating the filling state of the endoplasmic reticulum Ca(2+) store.
DYMDymeclinNecessary for correct organization of Golgi apparatus.
CRYBA4Beta-crystallin A4Crystallins are the dominant structural components of the vertebrate eye lens.
CRYGCGamma-crystallin CCrystallins are the dominant structural components of the vertebrate eye lens.
GJA3Gap junction alpha-3 proteinStructural component of lens fiber gap junctions.
MAP4K4Mitogen-activated protein kinase kinase kinase kinase 4Serine/threonine kinase that plays a role in the response to environmental stress and cytokines such as TNF.

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
Kinase26.2×0.079
Other/Unknown71.4×0.161

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
BFSP2Other/UnknownnoKeratin_I, IF_rod_dom
CRYBB3Other/UnknownnoBeta/gamma_crystallin, G_crystallin-like, Beta/Gamma-Crystallin
EPHA2Kinaseyes2.7.10.1Prot_kinase_dom, EPH_LBD, Ser-Thr/Tyr_kinase_cat_dom
WFS1Other/UnknownnoTPR-like_helical_dom_sf, Wolframin, Wolframin_fam
DYMOther/UnknownnoDymeclin
CRYBA4Other/UnknownnoBeta/gamma_crystallin, G_crystallin-like, Beta/Gamma-Crystallin
CRYGCOther/UnknownnoBeta/gamma_crystallin, G_crystallin-like, Beta/Gamma-Crystallin
GJA3Other/UnknownnoConnexin, Connexin46, Connexin_N
MAP4K4KinaseyesProt_kinase_dom, CNH_dom, Ser/Thr_kinase_AS

Expression context

Cohort genes with no expression data: 0.

5 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)1
broad (>20)8
unknown0

Top tissues across cohort

TissueCohort genes
male germ line stem cell (sensu Vertebrata) in testis4
primordial germ cell in gonad3
ganglionic eminence2
lens of camera-type eye1
buccal mucosa cell1
mucosa of transverse colon1
esophagus mucosa1
lower esophagus mucosa1
pharyngeal mucosa1
body of uterus1
left ovary1
right ovary1
bone marrow cell1
embryo1
frontal pole1
testis1
heart right ventricle1
left ventricle myocardium1
myocardium1
C1 segment of cervical spinal cord1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
BFSP2111tissue_specificyeslens of camera-type eye, male germ line stem cell (sensu Vertebrata) in testis, primordial germ cell in gonad
CRYBB3145tissue_specificyesmale germ line stem cell (sensu Vertebrata) in testis, buccal mucosa cell, mucosa of transverse colon
EPHA2224ubiquitousmarkerlower esophagus mucosa, esophagus mucosa, pharyngeal mucosa
WFS1280ubiquitousmarkerright ovary, left ovary, body of uterus
DYM254ubiquitousmarkerbone marrow cell, embryo, ganglionic eminence
CRYBA472tissue_specificmarkermale germ line stem cell (sensu Vertebrata) in testis, primordial germ cell in gonad, frontal pole
CRYGC15tissue_specificyesmale germ line stem cell (sensu Vertebrata) in testis, primordial germ cell in gonad, testis
GJA375broadyesleft ventricle myocardium, heart right ventricle, myocardium
MAP4K4295ubiquitousmarkerC1 segment of cervical spinal cord, cortical plate, ganglionic eminence

Protein interactions among cohort

Intra-cohort edges: 7.

Hub genes (top 10 by interactor count)

SymbolInteractor count
EPHA24,794
WFS13,409
CRYGC2,111
MAP4K41,975
CRYBB31,718
DYM1,298
BFSP21,203
CRYBA4601
GJA3449

Intra-cohort edges

ABSources
BFSP2CRYBA4string_interaction
BFSP2CRYBB3string_interaction
BFSP2CRYGCstring_interaction
BFSP2GJA3string_interaction
CRYBA4GJA3string_interaction
CRYBB3GJA3string_interaction
CRYGCGJA3string_interaction

Structural data

PDB: 5 · AlphaFold-only: 4 · No structure: 0

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
EPHA2P29317103
MAP4K4O9581918
CRYBB3P269981
CRYBA4P536731
CRYGCP073151

AlphaFold-only cohort genes (top 30 by pLDDT)

SymbolUniProtpLDDT
DYMQ7RTS987.91
BFSP2Q1351578.15
WFS1O7602473.85
GJA3Q9Y6H869.53

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 24. Enrichment computed across 9 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
Attenuation phase181.6×0.046CRYBA4
HSF1 activation176.1×0.046CRYBA4
EPHA-mediated growth cone collapse176.1×0.046EPHA2
HSF1-dependent transactivation163.4×0.046CRYBA4
Gap junction assembly158.6×0.046GJA3
RHOV GTPase cycle157.1×0.046EPHA2
RHOU GTPase cycle155.7×0.046EPHA2
RND1 GTPase cycle153.1×0.046EPHA2
RND3 GTPase cycle151.9×0.046EPHA2
RND2 GTPase cycle151.9×0.046EPHA2
EPH-ephrin mediated repulsion of cells143.9×0.046EPHA2
XBP1(S) activates chaperone genes143.1×0.046WFS1
EPH-Ephrin signaling133.1×0.054EPHA2
RHOG GTPase cycle129.7×0.054EPHA2
Regulation of HSF1-mediated heat shock response127.9×0.054CRYBA4
Cellular Senescence127.5×0.054MAP4K4
RAC2 GTPase cycle125.4×0.055EPHA2
RAC3 GTPase cycle123.8×0.055EPHA2
Post-translational protein phosphorylation120.0×0.062WFS1
Oxidative Stress Induced Senescence118.1×0.065MAP4K4
Regulation of Insulin-like Growth Factor (IGF) transport and uptake by Insulin-like Growth Factor Binding Proteins (IGFBPs)117.3×0.065WFS1
RAC1 GTPase cycle112.2×0.086EPHA2
Cellular responses to stress17.4×0.134MAP4K4
Cellular responses to stimuli16.3×0.149MAP4K4

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
visual perception653.0×3e-08BFSP2, WFS1, CRYBA4, CRYBB3, CRYGC, GJA3
lens development in camera-type eye3124.8×7e-05CRYBA4, CRYBB3, CRYGC
notochord cell development11872.4×0.017EPHA2
axial mesoderm formation1936.2×0.019EPHA2
negative regulation of ATF6-mediated unfolded protein response1936.2×0.019WFS1
notochord formation1624.1×0.019EPHA2
positive regulation of ARF protein signal transduction1624.1×0.019MAP4K4
negative regulation of lymphangiogenesis1624.1×0.019EPHA2
positive regulation of growth1468.1×0.020WFS1
cAMP metabolic process1468.1×0.020EPHA2
pericyte cell differentiation1374.5×0.020EPHA2
regulation of blood vessel endothelial cell migration1312.1×0.020EPHA2
gap junction-mediated intercellular transport1312.1×0.020GJA3
blood vessel endothelial cell proliferation involved in sprouting angiogenesis1267.5×0.020EPHA2
negative regulation of response to endoplasmic reticulum stress1267.5×0.020WFS1
negative regulation of chemokine production1234.1×0.020EPHA2
lens fiber cell development1234.1×0.020BFSP2
lens fiber cell morphogenesis1234.1×0.020EPHA2
negative regulation of type B pancreatic cell apoptotic process1234.1×0.020WFS1
regulation of lamellipodium assembly1208.1×0.020EPHA2
olfactory behavior1208.1×0.020WFS1
positive regulation of focal adhesion disassembly1208.1×0.020MAP4K4
positive regulation of bicellular tight junction assembly1187.2×0.022EPHA2
ER overload response1170.2×0.022WFS1
mammary gland epithelial cell proliferation1170.2×0.022EPHA2
branching involved in mammary gland duct morphogenesis1156.0×0.022EPHA2
response to growth factor1156.0×0.022EPHA2
negative regulation of cell adhesion mediated by integrin1144.0×0.022EPHA2
positive regulation of keratinocyte migration1144.0×0.022MAP4K4
nervous system process1133.8×0.023WFS1

Therapeutics

Drug target analysis

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

Druggability breadth: 4 of 9 evidence-associated genes (44%) 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
EPHA2PONATINIB
MAP4K4PONATINIB

Top cohort targets by molecule count

SymbolMoleculesMax phase
MAP4K4624
EPHA2504
BFSP200
CRYBB300
WFS100
DYM00
CRYBA400
CRYGC00
GJA300

Drugs targeting cohort genes (top 30)

MoleculeMax phaseTargets in cohort
PONATINIB4EPHA2, MAP4K4
FEDRATINIB4EPHA2
TIVOZANIB4EPHA2
SORAFENIB4EPHA2, MAP4K4
DASATINIB ANHYDROUS4EPHA2
REGORAFENIB4EPHA2
CABOZANTINIB4EPHA2
VANDETANIB4EPHA2, MAP4K4
NILOTINIB4EPHA2
BOSUTINIB4EPHA2, MAP4K4
TOVORAFENIB4EPHA2
NINTEDANIB4EPHA2, MAP4K4
DASATINIB4EPHA2, MAP4K4
CRIZOTINIB4EPHA2
AXITINIB4MAP4K4
NERATINIB4MAP4K4
PALBOCICLIB4MAP4K4
ENTRECTINIB4MAP4K4
PACRITINIB4MAP4K4
ABEMACICLIB4MAP4K4
GILTERITINIB4MAP4K4
PAZOPANIB4MAP4K4
SUNITINIB4MAP4K4
QUIZARTINIB4MAP4K4
MIDOSTAURIN4MAP4K4
GEFITINIB4MAP4K4
SARACATINIB3EPHA2, MAP4K4
LINIFANIB3EPHA2
TESEVATINIB3EPHA2, MAP4K4
ALVOCIDIB3EPHA2

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 1.

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

SymbolAssaysType breakdown
EPHA2567Binding:565, Functional:1, ADMET:1
MAP4K4407Binding:400, ADMET:5, Functional:1, Toxicity:1
CRYGC9Binding:9
WFS11Binding:1

Cohort enzymes (BRENDA EC)

SymbolEC numbersNames
EPHA22.7.10.1receptor protein-tyrosine kinase

Cohort genes with high screening signal

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

SymbolChEMBL assays
EPHA2567
MAP4K4407

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)
PONATINIB4EPHA2, MAP4K4
FEDRATINIB4EPHA2
TIVOZANIB4EPHA2
SORAFENIB4EPHA2, MAP4K4
DASATINIB ANHYDROUS4EPHA2
REGORAFENIB4EPHA2
CABOZANTINIB4EPHA2
VANDETANIB4EPHA2, MAP4K4
NILOTINIB4EPHA2
BOSUTINIB4EPHA2, MAP4K4
TOVORAFENIB4EPHA2
NINTEDANIB4EPHA2, MAP4K4
DASATINIB4EPHA2, MAP4K4
CRIZOTINIB4EPHA2
AXITINIB4MAP4K4
NERATINIB4MAP4K4
PALBOCICLIB4MAP4K4
ENTRECTINIB4MAP4K4
PACRITINIB4MAP4K4
ABEMACICLIB4MAP4K4
GILTERITINIB4MAP4K4
PAZOPANIB4MAP4K4
SUNITINIB4MAP4K4
QUIZARTINIB4MAP4K4
MIDOSTAURIN4MAP4K4
GEFITINIB4MAP4K4
SARACATINIB3EPHA2, MAP4K4
LINIFANIB3EPHA2
TESEVATINIB3EPHA2, MAP4K4
ALVOCIDIB3EPHA2

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)2EPHA2, MAP4K4
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug0
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug7BFSP2, CRYBB3, WFS1, DYM, CRYBA4, CRYGC, GJA3

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
BFSP20
CRYBB30
WFS11
DYM0
CRYBA40
CRYGC9
GJA30

Clinical trials & evidence

Clinical trials

Clinical trials: 0.