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Record W4414821733 · doi:10.1093/jbmr/zjaf112

Bone microstructural and strength changes over 1 year in children with osteogenesis imperfecta are comparable to age- and sex-matched healthy controls

2025· article· en· W4414821733 on OpenAlexaff
Seyedmahdi Hosseinitabatabaei, Samantha McCluskey, Carolyn A. Denton, Elizabeth A. Zimmermann, Francis H. Glorieux, F. Charbonneau, Frank Rauch, Bettina M. Willie

Bibliographic record

VenueJournal of Bone and Mineral Research · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicConnective tissue disorders research
Canadian institutionsMcGill UniversityShriners Hospitals for Children - CanadaMcGill University Health Centre
Fundersnot available
KeywordsOsteogenesis imperfectaMetaphysisBone mineralCortical boneTibiaDiaphysisQuantitative computed tomographyBone densityOsteoporosis

Abstract

fetched live from OpenAlex

INTRODUCTION: Osteogenesis imperfecta (OI) is characterized by bone fragility with frequent fractures, especially in children. Some studies have used peripheral-QCT (pQCT) to examine bone density in children with OI and one cross-sectional study used HR-pQCT in 9 children with OI. MATERIALS AND METHODS: We compared bone density, microstructure, and strength changes over 1 yr at the metaphysis and diaphysis of the radius and tibia, as well as lumbar spine (LS) bone mass and area, in 20 children with OI under bisphosphonate treatment and 20 age- and sex-matched controls. For the double-stack metaphyseal scans, we developed an algorithm to correct stack misalignment. For the LS, we used DXA. RESULTS: At the tibial metaphysis, children with OI had lower baseline total and trabecular volumetric BMD (vBMD), trabecular and cortical microstructure, and strength. At the radial metaphysis, baseline trabecular microstructure and strength were lower in children with OI. At the tibial and radial diaphysis, children with OI had lower bone area and strength. At the tibial and radial metaphysis, 1-yr increases in most measurements were similar for both groups. Cortical vBMD and trabecular separation (radius) only increased in the OI group. Trabecular vBMD, volume fraction (tibia), number (tibia), and total vBMD (radius) only increased in the control group. At the tibia and radius diaphysis, cortical vBMD increased in the OI group. Tibial Ct.Po decreased in children with OI. At the LS, the OI group had lower bone mass, density, and area, but similar longitudinal changes. DISCUSSION: In summary, baseline trabecular bone density, trabecular and cortical microstructure, and strength were lower in metaphyseal regions of children with OI. At diaphysis, children with OI had lower bone area and strength. While longitudinal changes over 1 yr were generally comparable, certain outcomes demonstrated differences. These data are essential for powering future clinical trials.

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)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.637
Threshold uncertainty score0.371

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
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 head0.018
GPT teacher head0.338
Teacher spread0.320 · 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

Citations1
Published2025
Admission routes1
Has abstractyes

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