Factors Associated With Functional Mobility in 256 Children With Arthrogryposis Multiplex Congenita: A Multicentric Cross-Sectional Study
Bibliographic record
Abstract
OBJECTIVE: To describe functional mobility among children, adolescents, and young adults with arthrogryposis multiplex congenita (AMC) and identify factors associated with mobility outcomes. DESIGN: Multisite cross-sectional study. SETTING: Eight orthopedic hospitals for children. PARTICIPANTS: A total of 256 individuals (N=256) aged 5-21 years with a confirmed clinical diagnosis of AMC were recruited between October 2019 and December 2022. AMC subtypes included amyoplasia (n=122), distal arthrogryposis (n=62), and Central Nervous System (CNS)/syndromic AMC (n=39). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Functional mobility was assessed using the mobility domain of the Functional Independence Measure for Children (WeeFIM) and the Gillette Functional Assessment Questionnaire (FAQ). Multivariable models were used to examine associations with perinatal and current clinical and environmental factors. RESULTS: Children with CNS/syndromic and amyoplasia subtypes demonstrated significantly lower WeeFIM scores (coefficients [Exp(B)]=0.74 and 0.85, respectively; both P<.05) and reduced odds of higher FAQ levels (adjusted odds ratio [AOR]=0.16 and 0.18, respectively; both P<.001) compared with those with distal arthrogryposis. Greater joint involvement, particularly at the knees, was a strong negative factor. Each additional perinatal joint contracture was associated with a 3.1% reduction in WeeFIM scores (Exp(B)=0.97, P<.001) and an 8.2% decrease in the odds of higher FAQ levels (AOR=0.92, P<.05). Current knee involvement was associated with a 27.7% reduction in WeeFIM scores (Exp(B)=0.72, P<.001) and nearly 90% lower odds of higher FAQ levels (AOR=0.10, P<.001). Parental unemployment (AOR=0.44, P<.05) and higher musculoskeletal surgical burden (AOR=0.24, P<.001) were significantly associated with poorer mobility. CONCLUSIONS: This is the largest cohort to date examining functional mobility in AMC. Clinical and socioeconomic factors identified may guide tailored rehabilitation strategies to promote positive outcomes in children with AMC.
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How this classification was reachedexpand
Full frame machine prediction
Teacher imitationNot 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.
Distilled classifier scores by category (both heads)
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.001 | 0.001 |
| Meta-epidemiology (narrow) | 0.001 | 0.001 |
| Meta-epidemiology (broad) | 0.001 | 0.001 |
| Bibliometrics | 0.001 | 0.001 |
| Science and technology studies | 0.001 | 0.001 |
| Scholarly communication | 0.001 | 0.001 |
| Open science | 0.000 | 0.001 |
| Research integrity | 0.001 | 0.001 |
| Insufficient payload (model declined to judge) | 0.001 | 0.000 |
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.
score_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from itClassification
machine, unvalidatedMachine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.
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".