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Imputation of untreated LDL-C in treated subjects with homozygous familial hypercholesterolaemia: An international collaboration

2025· article· en· W4416425577 on OpenAlexafffund
G.B. John Mancini, Arnold Ryomoto, Isabelle L. Ruel, Iulia Iatan, Frederick J. Raal, Raul D. Santos, Ana Paula Marte Chacra, Robert A. Hegele, Brooke A. Kennedy, Liam R. Brunham, Daniel Gaudet, Miriam Larouche, Diane Brisson, Willemijn Schonck, Laurens F. Reeskamp, Jacques Genest

Bibliographic record

VenueAtherosclerosis · 2025
Typearticle
Languageen
FieldMedicine
TopicLipoproteins and Cardiovascular Health
Canadian institutionsUniversité du Québec à ChicoutimiWestern UniversityRobarts Clinical TrialsMcGill University Health CentreUniversity of British Columbia
FundersHLS TherapeuticsNovo NordiskDaiichi-SankyoAmryt PharmaDaiichi Sankyo EuropeSilence TherapeuticsUniversity of British ColumbiaPTC TherapeuticsSanofiKowa CompanyIonis PharmaceuticalsRegeneron PharmaceuticalsAmgenPfizerEsperion TherapeuticsUltragenyx PharmaceuticalAstraZenecaEli Lilly and Company
KeywordsImputation (statistics)Missing dataMedical recordMEDLINE

Abstract

fetched live from OpenAlex

BACKGROUND AND AIMS: Diagnosis of Homozygous Familial Hypercholesterolaemia (HoFH) relies on untreated low-density lipoprotein-cholesterol (LDL-C) which is often unknown. We determine whether untreated LDL-C can be imputed from treated LDL-C in HoFH. METHODS: Two groups with HoFH were identified: Group 1 (n = 193) from Canada, Brazil and South Africa; Group 2 (n = 206) from the HoFH International Clinical Collaboration. Pre- and post-treatment LDL-C and lipid lowering therapy (LLT) intensity from Group 1 were used to develop a regression model and applied to treated LDL-C in Group 2 to impute pre-treatment LDL-C. The same process was performed in reverse. A final imputation model was created from combining both groups. RESULTS: There was a curvilinear relationship between the expected and observed % lowering of LDL-C on LLT (r = 0.3923, p < 0.0001, Standard Error [SE] = 23 %). Using this relationship, LDL-C was imputed from treated values and showed significant correlation with pre-treatment LDL-C (r = 0.71, p < 0.001; mean values 13.4 ± 4.7 [Standard Deviation] and 13.6 ± 7.3 mmol/L, respectively, ns). Concordance between actual and imputed values ≥ 10 or <10 mmol/L was 80 %. Whereas 36 % of patients had treated LDL-C ≥ 10 mmol/L, 64 % had treated or imputed pre-treatment LDL-C ≥ 10 mmol/L. CONCLUSIONS: In HoFH, the response to LLT can be quantified and used to impute untreated LDL-C from treated LDL-C. Imputation may augment awareness of possible HoFH in treated subjects lacking records of untreated LDL-C.

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)0.084
metaresearch head score (Gemma)0.117
Version: metacan-v3-hybrid-931329e0061cValidation 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 score0.084
Threshold uncertainty score0.446

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0840.117
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0040.006
Bibliometrics0.0020.006
Science and technology studies0.0010.001
Scholarly communication0.0030.001
Open science0.0040.003
Research integrity0.0030.002
Insufficient payload (model declined to judge)0.0040.001

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 head0.011
GPT teacher head0.272
Teacher spread0.261 · 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 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".

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Citations0
Published2025
Admission routes2
Has abstractno

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