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Welcome!

Community-curated standards & an infrastructure for predicted effector gene lists

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Why does this matter?

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Two approaches…

…One objective

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International team…

contributing to this effort

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  • Raise awareness & present the landscape
  • Establish a community standard for reporting PEG lists
  • Ensure that PEG lists with their supporting evidence & related metadata are FAIR and relevant to the user community
  • Define strategies for evaluation & collation
  • Identify challenges in sharing, maintaining, updating PEG lists 
  • Identify strategies for incentivizing implementation/use of the proposed standards

Our Objectives

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  • An engaged community of stewards (you all)
  • A white paper/manuscript
  • A resource for the deposition & access for these lists
  • A resource for the presentation, curation & interpretation of these lists
  • A roadmap make these results useful- to enable discovery

Our Outputs

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Strawman

PEGASUS

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What does this mean?

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Date (2024-25)

Publication & phenotype(s)

Size / ancestry of meta-analysis

Evidence sources combined in the gene-ranking step

Metric / rule published (files in supp. data unless noted)

10 Feb 2025

Schipper M et al. “Prioritizing effector genes at trait-associated loci using multimodal evidence” — Multi-trait (UK Biobank + GWAS Catalog)

14,000 GWAS loci, pan-trait

Fine-mapping (SuSiE), coding variant annotation, convergence layer (co-expression, Reactome/GO pathways, PPI)

FLAMES score (0-1 probability per gene/locus; TSV on Zenodo) PubMed

3 Jan 2025

Dang X et al. “Gene-level analysis reveals the genetic aetiology and therapeutic targets of schizophrenia” (Nat Hum Behav)

144 K cases / 1.3 M ctrls, multi-ancestry

MAGMA gene-level p, PoPS, TWAS (brain GTEx & PsychENCODE), chromatin-interaction links

Integrated gene score (normalized −log10 p across all sources) — ranked list of 298 genes (Supplementary Table 3) PubMed

21 Nov 2024

Zheng S L et al. “Genome-wide association analysis provides insights into the molecular etiology of dilated cardiomyopathy” (Nat Genet)

14 256 cases / 1.1 M ctrls (European + multi-ancestry)

PoPS, distance-to-lead SNP, OpenTargets V2G, multi-tissue TWAS, rare-variant colocalisation

Omnibus prioritisation score (weighted sum; sheet in Supplementary Data 8) Nature

04 Mar 2025

Henry A et al. “GWAS meta-analysis of heart-failure and its subtypes” (Nat Genet)

90 K cases / 1.3 M ctrls, multi-ancestry

PoPS, OpenTargets V2G fine-map score, multi-tissue S-MulTiXcan TWAS

Two-step rank: top gene per locus if best in ≥1 metric; 356 ranked genes (Extended Data Table 5) Nature

16 Apr 2025

Lange L M et al. “Prioritizing Parkinson’s disease risk genes in GWAS loci” (npj Parkinson’s Dis.)

49 K cases / 2.4 M ctrls; Eur. + E.Asian

PoPS, nearest-gene distance, exonic credible-set variants

PD-PoPS rank: PoPS-top gene intersected with distance / coding evidence; 46 high-confidence genes (Table 2) Nature

24 Apr 2025

Li H et al. “Multi-ancestry seqGWAS of C-reactive protein (CRP)” (Nat Commun)

513 273 WGS samples (TOPMed + UKB + AoU)

Cross-ancestry FINEMAP, PoPS, gene-based SKAT-O tests

PoPS-enriched list: 151 genes ranked by PoPS then SKAT-O significance (Supp. Data 9) Nature

Our community is still churning out large-scale GWASs with gene prioritization

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▲ Rank

Publication (Year)

Gene-prioritisation metric released

What makes the metric “curated”

Scope / data breadth

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Schipper et al. “Prioritizing effector genes at trait-associated loci using multimodal evidence” Nat Genet 2025

FLAMES score (machine-learning probability, 0-1)

Trained on an expert-vetted and data-driven truth set of 406 causal genes; integrates fine-mapping SNP→gene links plus convergence evidence (co-expression, pathways, PPI) for each locus PubMedmedRxiv

Pan-trait benchmark across UK Biobank & GWAS Catalog loci

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Open Targets Platform v25.03 (2025 release notes)

Locus-to-Gene (L2G) score v2

Gradient-boosting model trained on a hand-curated gold standard; >40 functional-genomics features (QTLs, chromatin, distance) exposed through an open API Open Targets

6.5 M variants, 2.6 M credible sets, >28 K traits

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Effector Index cited in Turner et al. Cell HGG Advances 2025

Ei probability (0-1 per gene)

Initial model trained on 12 common diseases with manual curation; emphasises interpretable distance, eQTL and chromatin features; score files downloadable from GitHub PMC

Genome-wide for all RefSeq genes at each GWAS locus

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Schipper et al. “CALDERA” pre-print 2024

CALDERA score (LASSO-logistic probability)

Uses only 12 biologically interpretable features; truth set constructed with fine-mapped coding signals and corrected for proximity bias medRxiv

4 764 gene–trait pairs across 19 UKB traits; externally validated

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Lange et al. “Prioritising Parkinson’s disease risk genes…” npj Parkinson’s Dis. 2025

PoPS rank / score (similarity-based)

>57 000 curated gene features (single-cell RNA-seq, pathways, PPI) merged into a polygenic priority score; benchmarked against PD literature genes Nature

Multi-ancestry PD GWAS; method generalisable genome-wide

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Law et al. “Systematic prioritisation… colorectal cancer risk” Nat Genet 2024

ABC-MPRA linked-gene score

Curated colon-specific enhancer maps, Micro-C contacts & MPRA validation to assign variant→gene links; publishes per-locus gene lists with confidence tiers Nature

170 CRC risk loci, 208 effector genes

New approaches- curated & computational

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New approaches- curated & computational

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Iterative, more comprehensive

Not available or hosted on lab websites!

Lui et al, 2025

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Final consideration- technology

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Final consideration- technology

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