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Record W3038754476 · doi:10.1371/journal.pmed.1003152

Development of a polygenic risk score to improve screening for fracture risk: A genetic risk prediction study

2020· article· en· W3038754476 on OpenAlexafffund
Vincenzo Forgetta, Julyan Keller‐Baruch, Marie Forest, Audrey Durand, Sahir Bhatnagar, John P. Kemp, Maria Nethander, Daniel S. Evans, John Morris, Douglas P. Kiel, Fernando Rivadeneira, Helena Johansson, Nicholas C. Harvey, Dan Mellström, Magnus K. Karlsson, Cyrus Cooper, David M. Evans, Robert Clarke, John А. Kanis, Eric Orwoll, Eugène McCloskey, Claes Ohlsson, Joëlle Pineau, William D. Leslie, Celia M.T. Greenwood, J. Brent Richards

Bibliographic record

VenuePLoS Medicine · 2020
Typearticle
Languageen
FieldMedicine
TopicBone health and osteoporosis research
Canadian institutionsUniversity of ManitobaMcGill UniversityJewish General Hospital
FundersNational Institute on AgingNational Health and Medical Research CouncilFonds de Recherche du Québec - SantéNational Institutes of HealthServierNorthwest Regional Development AgencyEuropean CommissionNational Institute for Health and Care ResearchKing's College LondonRadius HealthBritish Heart FoundationNational Center for Advancing Translational SciencesWellcome TrustCanadian Institutes of Health ResearchDanoneGlaxoSmithKlineNational Institute of Arthritis and Musculoskeletal and Skin DiseasesMedical Research CouncilBiogenAmgenPfizerEli Lilly and Company
KeywordsFRAXMedicineGuidelineFramingham Risk ScoreRisk assessmentPopulationPolygenic risk scoreOsteoporosisBiobankInternal medicinePhysical therapyBone mineralBioinformaticsEnvironmental healthOsteoporotic fractureGeneticsDiseaseSingle-nucleotide polymorphismPathologyBiologyGenotype

Abstract

fetched live from OpenAlex

BACKGROUND: Since screening programs identify only a small proportion of the population as eligible for an intervention, genomic prediction of heritable risk factors could decrease the number needing to be screened by removing individuals at low genetic risk. We therefore tested whether a polygenic risk score for heel quantitative ultrasound speed of sound (SOS)-a heritable risk factor for osteoporotic fracture-can identify low-risk individuals who can safely be excluded from a fracture risk screening program. METHODS AND FINDINGS: A polygenic risk score for SOS was trained and selected in 2 separate subsets of UK Biobank (comprising 341,449 and 5,335 individuals). The top-performing prediction model was termed "gSOS", and its utility in fracture risk screening was tested in 5 validation cohorts using the National Osteoporosis Guideline Group clinical guidelines (N = 10,522 eligible participants). All individuals were genome-wide genotyped and had measured fracture risk factors. Across the 5 cohorts, the average age ranged from 57 to 75 years, and 54% of studied individuals were women. The main outcomes were the sensitivity and specificity to correctly identify individuals requiring treatment with and without genetic prescreening. The reference standard was a bone mineral density (BMD)-based Fracture Risk Assessment Tool (FRAX) score. The secondary outcomes were the proportions of the screened population requiring clinical-risk-factor-based FRAX (CRF-FRAX) screening and BMD-based FRAX (BMD-FRAX) screening. gSOS was strongly correlated with measured SOS (r2 = 23.2%, 95% CI 22.7% to 23.7%). Without genetic prescreening, guideline recommendations achieved a sensitivity and specificity for correct treatment assignment of 99.6% and 97.1%, respectively, in the validation cohorts. However, 81% of the population required CRF-FRAX tests, and 37% required BMD-FRAX tests to achieve this accuracy. Using gSOS in prescreening and limiting further assessment to those with a low gSOS resulted in small changes to the sensitivity and specificity (93.4% and 98.5%, respectively), but the proportions of individuals requiring CRF-FRAX tests and BMD-FRAX tests were reduced by 37% and 41%, respectively. Study limitations include a reliance on cohorts of predominantly European ethnicity and use of a proxy of fracture risk. CONCLUSIONS: Our results suggest that the use of a polygenic risk score in fracture risk screening could decrease the number of individuals requiring screening tests, including BMD measurement, while maintaining a high sensitivity and specificity to identify individuals who should be recommended an intervention.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.004
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.139
Threshold uncertainty score0.792

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.004
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.063
GPT teacher head0.339
Teacher spread0.276 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations73
Published2020
Admission routes2
Has abstractyes

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