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Record W4414162073 · doi:10.1177/13674935251379181

A qualitative exploration of child, caregiver, and clinician perspectives on mental health in children with osteogenesis imperfecta

2025· article· en· W4414162073 on OpenAlexaff
Justin Qian, Andrew D. Wiese, Clarissa Gonzalez, Whitney S. Shepherd, Hannah Cho, Gianna M. Colombo, Julia Morales, Selena Guo, Kristin M. Kostick, Dianne Nguyen, Sophie C. Schneider, Kara Ayers, Marie‐Eve Robinson, Chaya N. Murali, Brendan Lee, V. Reid Sutton, Eric A. Storch

Bibliographic record

VenueJournal of Child Health Care · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicConnective tissue disorders research
Canadian institutionsUniversity of Ottawa
FundersEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentNational Institute of Arthritis and Musculoskeletal and Skin Diseases
KeywordsPsychosocialOsteogenesis imperfectaMental healthQualitative researchSocial isolationMoodMultidisciplinary approachSocial support

Abstract

fetched live from OpenAlex

Osteogenesis imperfecta (OI) is a heritable connective disorder with clinical manifestations including bone fragility and short stature. Previous research identified psychosocial themes related to mental health among children with OI and their caregivers, such as independence, social isolation, depression, and anxiety. Several studies have also examined clinicians of individuals with OI for their perspectives on mental health among their patients. However, no known studies have compared psychosocial themes among all three participant groups concurrently. This qualitative study examined mood and anxiety, along with risk and protective factors, from perspectives of youths, parents, and clinicians. Semi-structured interviews were conducted, which were coded independently by two researchers. Codes were analyzed for extraction of themes. Relevant themes include depressed and anxious mood experienced in individuals with OI, as well as risk factors and protective factors for these symptoms. Participants reported functional limitations and social isolation as key risk factors for symptoms of anxious or depressed mood. In contrast, they discussed positive attitudes, social support, multidisciplinary medical groups, and mental health access as key protective factors. Implications for clinical care and future research are discussed. Convergence and divergence of themes across study samples are discussed in detail.

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: Qualitative · Consensus signal: Qualitative
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.140
Threshold uncertainty score0.476

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 head0.016
GPT teacher head0.397
Teacher spread0.381 · 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 designQualitative
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

Citations0
Published2025
Admission routes1
Has abstractyes

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