Cerebroretinal microangiopathy with calcifications and cysts 2

disease
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Also known as cerebroretinal microangiopathy with calcifications and cysts type 2Coats plus syndrome caused by mutation in STN1CRMCC2STN1 Coats plus syndrome

Summary

Cerebroretinal microangiopathy with calcifications and cysts 2 (MONDO:0015026) is a disease caused by STN1 (GenCC Strong), with 1 cohort gene.

At a glance

  • Causal gene: STN1 (GenCC Strong)
  • Cohort genes: 1
  • ClinVar variants: 12

Clinical features

No curated clinical features (Orphanet) for this disease.

Identifiers

Disease identifiers

FieldValue
Canonical namecerebroretinal microangiopathy with calcifications and cysts 2
Mondo IDMONDO:0015026
OMIM617341
UMLSC4479220
MedGen1390862
GARD0018442
Is cancer (heuristic)no

Also known as: cerebroretinal microangiopathy with calcifications and cysts 2 · cerebroretinal microangiopathy with calcifications and cysts type 2 · Coats plus syndrome caused by mutation in STN1 · CRMCC2 · STN1 Coats plus syndrome

Data availability: 12 ClinVar variants · 4 GenCC gene-disease records.

Disease family

Classification path: disease › human disease › disease by body system or component › nervous system disordercongenital nervous system disorder › congenital vitreoretinal dysplasia › Coats plus syndromecerebroretinal microangiopathy with calcifications and cysts 2

Related subtypes (2): cerebroretinal microangiopathy with calcifications and cysts 1, cerebroretinal microangiopathy with calcifications and cysts 3

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

12 retrieved; paginated sample, class counts are floors:

6 uncertain significance, 3 benign, 1 pathogenic, 1 conflicting classifications of pathogenicity, 1 benign/likely benign

ClinVarVariant (HGVS)GeneClassificationReview
375591NM_024928.5(STN1):c.404G>C (p.Arg135Thr)STN1Pathogenicno assertion criteria provided
728880NM_024928.5(STN1):c.793A>C (p.Ser265Arg)STN1Conflicting classifications of pathogenicitycriteria provided, conflicting classifications
1016833NM_024928.5(STN1):c.19C>T (p.Arg7Trp)STN1Uncertain significancecriteria provided, multiple submitters, no conflicts
1028354NM_024928.5(STN1):c.1100C>T (p.Ala367Val)STN1Uncertain significancecriteria provided, multiple submitters, no conflicts
1391941NM_024928.5(STN1):c.398G>A (p.Arg133Gln)STN1Uncertain significancecriteria provided, multiple submitters, no conflicts
2139047NM_024928.5(STN1):c.210T>G (p.Asp70Glu)STN1Uncertain significancecriteria provided, multiple submitters, no conflicts
3590585NM_024928.5(STN1):c.143T>C (p.Leu48Ser)STN1Uncertain significancecriteria provided, single submitter
375592NM_024928.5(STN1):c.469G>T (p.Asp157Tyr)STN1Uncertain significancecriteria provided, single submitter
1169155NM_024928.5(STN1):c.743C>G (p.Ser248Cys)STN1Benigncriteria provided, multiple submitters, no conflicts
1169156NM_024928.5(STN1):c.696G>C (p.Val232=)STN1Benigncriteria provided, multiple submitters, no conflicts
1169388NM_024928.5(STN1):c.451A>G (p.Thr151Ala)STN1Benigncriteria provided, multiple submitters, no conflicts
768387NM_024928.5(STN1):c.582-4G>ASTN1Benign/Likely benigncriteria provided, multiple submitters, no conflicts

Genes & proteins

Mendelian disease overlap and somatic drivers

GenCC: 5 · Orphanet: 2 · 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
STN1StrongAutosomal recessivecerebroretinal microangiopathy with calcifications and cysts 25

Orphanet rare-disease linkage (cohort genes)

GeneOrphanet IDRare disease
STN1Orphanet:2032Idiopathic pulmonary fibrosis
STN1Orphanet:313838Coats plus syndrome

Cohort genes → proteins

1 cohort genes, 1 distinct canonical proteins.

Evidence partition

SubsetGenes
multi_evidence1

Cohort genes (full)

SymbolHGNCEnsemblUniProtNameEvidence
STN1HGNC:26200ENSG00000107960Q9H668CST complex subunit STN1gencc,clinvar

Cohort function summary

Lead sentence per gene, UniProt-curated.

SymbolProtein nameFunction (lead sentence)
STN1CST complex subunit STN1Component 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.

Protein-family classification

Druggable: 0 · Difficult: 0 · Unknown: 1 · 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/Unknown11.8×0.558

Per-gene assignment

SymbolFamilyDruggable?ECInterPro (top 3)
STN1Other/UnknownnoNA-bd_OB_tRNA, NA-bd_OB-fold, Stn1

Expression context

Cohort genes with no expression data: 0.

1 cohort gene 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)1
unknown0

Top tissues across cohort

TissueCohort genes
esophagus mucosa1
lower esophagus mucosa1
oral cavity1

Per-gene tissue summary (top 30)

SymbolBgee breadthFANTOM5 breadthSCXATop tissues
STN1284ubiquitousmarkerlower esophagus mucosa, oral cavity, esophagus mucosa

Protein interactions among cohort

Intra-cohort edges: 0.

Hub genes (top 10 by interactor count)

SymbolInteractor count
STN11,863

Structural data

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

Cohort genes with PDB structures (top 30)

SymbolUniProtPDB entries
STN1Q9H6688

Function

Pathway analysis

Distinct Reactome pathways touched by cohort: 7. Enrichment computed across 1 evidence-associated genes (1 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 1 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

PathwayCohort genesFoldFDRSample cohort genes
Telomere C-strand synthesis initiation1815.7×0.004STN1
Telomere C-strand (Lagging Strand) Synthesis1761.3×0.004STN1
Extension of Telomeres1601.0×0.004STN1
Polymerase switching on the C-strand of the telomere1423.0×0.004STN1
Telomere Maintenance1368.4×0.004STN1
Chromosome Maintenance1211.5×0.006STN1
Cell Cycle136.0×0.028STN1

GO biological processes by enrichment

Over-representation of cohort genes vs the genome-wide background (hypergeometric test, Benjamini-Hochberg FDR; fold = observed/expected over 1 annotated cohort genes). Counts and members are kept as ground-truth; sorted by enrichment.

GO termCohort genesFoldFDRSample cohort genes
telomere maintenance via telomere lengthening11872.4×0.002STN1
telomere capping11296.3×0.002STN1
negative regulation of telomere maintenance via telomerase1732.7×0.002STN1
positive regulation of DNA replication1581.1×0.002STN1
telomere maintenance1267.5×0.004STN1

Therapeutics

Drug target analysis

Approved (phase 4): 0 · Phase ≥3: 0 · Phased (≥1): 0 · Undrugged: 1

Druggability breadth: 0 of 1 evidence-associated genes (0%) have a ChEMBL target (buckets above are over the deeply-mined display cohort).

Top cohort targets by molecule count

SymbolMoleculesMax phase
STN100

Bioactivity and enzyme data

Enzyme cohort genes (≥1 EC): 0.

Pharmacogenomics

Cohort genes with a PharmGKB record: 1; with CPIC/DPWG dosing guidelines: 0.

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

Chemical tractability of cohort targets

0 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.

Druggability pyramid

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

TierDefinitionGenesSymbols
AApproved (phase 4 drug)0
BPhased (≥1) drug, not yet approved0
CDruggable family + PDB, no drug0
DDruggable family + AlphaFold only, no drug0
EDifficult family or no structure, no drug1STN1

Undrugged target profiles

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

SymbolChEMBL assaysDrugged partners (top 3)
STN10

Clinical trials & evidence

Clinical trials

Clinical trials: 0.