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Record W4410610542 · doi:10.1016/j.gim.2025.101465

Mainstreaming of clinical genetic testing: A conceptual framework

2025· article· en· W4410610542 on OpenAlexafffundabout
Michael P. Mackley, Julie Richer, Andrea Guerin, Oana Caluseriu, Linlea Armstrong, Katherine A Blood, François P. Bernier, Christie Boswell-Patterson, Marisa Chard, Gregory Costain, David A. Dyment, Alison Eaton, Hanna Faghfoury, Patrick Frosk, Meredith Gillespie, Elaine Goh, Robin Z. Hayeems, Bita Hashemi, A. Micheil Innes, Molly Jackson, Anne‐Marie Laberge, Jacqueline Limoges, Christian Marshall, Hugh J. McMillan, Tanya N. Nelson, Matthew Osmond, Jillian S. Parboosingh, Lynette S. Penney, Bradley Prince, Sarah L. Sawyer, Victoria Mok Siu, Mary Ann Thomas, Lesley Turner, Noémie Villeneuve‐Cloutier, Taila Hartley, Kym M Boycott

Bibliographic record

VenueGenetics in Medicine · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicBRCA gene mutations in cancer
Canadian institutionsBC Children's HospitalDalhousie UniversityCentre Hospitalier Universitaire Sainte-JustineWestern UniversityRoyal University HospitalAthabasca UniversityInstitute for Clinical Evaluative SciencesTrillium Health CentreUniversity of ManitobaUniversity of CalgaryMount Sinai HospitalMemorial University of NewfoundlandAlberta Children's HospitalHospital for Sick ChildrenUniversity of British ColumbiaChildren's Hospital of Eastern OntarioB.C. Women's Hospital & Health CentreUniversity of AlbertaQueen's UniversityToronto General HospitalSickKids Foundation
FundersCanadian Institutes of Health Research
KeywordsGenetic testingMainstreamingMedicineComputational biologyBiologyGeneticsPsychology

Abstract

fetched live from OpenAlex

PURPOSE: Demand for genetic testing is increasing across medicine, whereas the genetics workforce remains stable. In response, mainstreaming models are being introduced, in which nongeneticist clinicians are increasingly involved in the genetic testing pathway. Because a standardized approach would facilitate evaluation and optimal patient care, a unified framework is warranted. METHODS: Through a focus group with clinical genetics experts, a conceptual framework for the mainstreaming of clinical genetic testing is proposed. Through a consensus process, experts elucidated the steps in the diagnostic care pathway and defined a set of variables that influence which mainstreaming model is best suited to specific patient care scenarios. RESULTS: A total of 35 individuals representing 20 distinct clinical genetics services and all Canadian provinces participated in the development of the framework. The framework describes 4 generalizable mainstreaming models of care, each with varying levels of involvement of the clinical genetics service in the diagnostic care pathway. CONCLUSION: This framework will help guide clinical teams in the design and evaluation of mainstreaming efforts. It is critical that these programs are evaluated and shared in a standardized way so that we can implement strategies that allow optimal utilization of genetics resources and improve patient care.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.042
metaresearch head score (Gemma)0.027
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Theoretical or conceptual · Consensus signal: Theoretical or conceptual
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.105
Threshold uncertainty score0.349

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0420.027
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0010.002
Bibliometrics0.0090.005
Science and technology studies0.0110.049
Scholarly communication0.0140.013
Open science0.0060.009
Research integrity0.0050.005
Insufficient payload (model declined to judge)0.0030.000

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 head0.056
GPT teacher head0.402
Teacher spread0.346 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designTheoretical or conceptual
Domainnot available
GenreReview

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

Citations14
Published2025
Admission routes3
Has abstractyes

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