Milestone Attainment in Young Children With Arthrogryposis Multiplex Congenita: Developmental Profile and Associated Factors

Evidence on developmental milestones in children with arthrogryposis multiplex congenita (AMC) under the age of five is scarce. This multisite cross-sectional study described developmental status and examined factors associated with milestone attainment in 143 children aged 0-66 months from a pediatric AMC Registry. Development was assessed using the Ages and Stages Questionnaire, Third Edition (ASQ-3), across Communication, Gross Motor, Fine Motor, Problem-Solving, and Personal-Social domains. Independent variables included demographic, perinatal, clinical, and parental factors. Gross Motor function was the most limited domain, with more than 90% scoring close to/below the ASQ-3 domain-specific cutoff (≥ 2 SD below normative mean). Personal-Social scores were unexpectedly low, and Fine Motor limitations were also frequent, with 53.1% scoring close to/below the cutoff. Communication and Problem-Solving were relative strengths. Longer neonatal intensive-care unit stays were linked to poorer Communication and Fine Motor scores, and higher birth weight was associated with better Problem-Solving performance. Specific joint contractures, particularly those affecting the neck, shoulder, elbow, and knee, were associated with lower performance across domains, while higher parental education was related to better Personal-Social outcomes. Developmental attainment in AMC is shaped by interactions among musculoskeletal, perinatal, and family factors, underscoring the importance of early, coordinated orthopedic, developmental and family-centered interventions.

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Publication Details

Journal
American Journal of Medical Genetics Part C Seminars in Medical Genetics
Published
2026-03-16
DOI
https://doi.org/10.1002/ajmg.c.70006
Primary Topic
Neurogenetic and Muscular Disorders Research
Type
article
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article

Milestone Attainment in Young Children With Arthrogryposis Multiplex Congenita: Developmental Profile and Associated Factors

Lauren C. Hyer, Sarah Beth Nossov, Joel A. Lerman, Krister Freese et al.
American Journal of Medical Genetics Part C Seminars in Medical Genetics
Neurogenetic and Muscular Disorders Research
article

Milestone Attainment in Young Children With Arthrogryposis Multiplex Congenita: Developmental Profile and Associated Factors

Lauren C. Hyer, Sarah Beth Nossov, Joel A. Lerman, Krister Freese, Cary H. Mielke, Noémi Dahan‐Oliel, Thania Ordaz‐Robles, Haluk Altiok, F Rauch, Ahlam Zidan, Emmanouil Rampakakis, Reggie Hamdy, Sophia C. Pasia, Philip F. Giampietro
article en

Abstract

Evidence on developmental milestones in children with arthrogryposis multiplex congenita (AMC) under the age of five is scarce. This multisite cross-sectional study described developmental status and examined factors associated with milestone attainment in 143 children aged 0-66 months from a pediatric AMC Registry. Development was assessed using the Ages and Stages Questionnaire, Third Edition (ASQ-3), across Communication, Gross Motor, Fine Motor, Problem-Solving, and Personal-Social domains. Independent variables included demographic, perinatal, clinical, and parental factors. Gross Motor function was the most limited domain, with more than 90% scoring close to/below the ASQ-3 domain-specific cutoff (≥ 2 SD below normative mean). Personal-Social scores were unexpectedly low, and Fine Motor limitations were also frequent, with 53.1% scoring close to/below the cutoff. Communication and Problem-Solving were relative strengths. Longer neonatal intensive-care unit stays were linked to poorer Communication and Fine Motor scores, and higher birth weight was associated with better Problem-Solving performance. Specific joint contractures, particularly those affecting the neck, shoulder, elbow, and knee, were associated with lower performance across domains, while higher parental education was related to better Personal-Social outcomes. Developmental attainment in AMC is shaped by interactions among musculoskeletal, perinatal, and family factors, underscoring the importance of early, coordinated orthopedic, developmental and family-centered interventions.

American Journal of Medical Genetics Part C Seminars in Medical Genetics
Shriners Hospitals for Children - Portland (US), McGill University Health Centre (CA), Shriners Hospitals for Children - Canada (CA), University of Illinois Chicago (US), University of Chicago Medical Center (US), Shriners Hospitals for Children - Mexico (MX), Shriners Hospitals for Children - Greenville (US), Shriners Hospitals for Children - Shreveport (US), Shriners Hospitals for Children - Northern California (US), GDI Integrated Facility Services (Canada) (CA), Shriners Hospitals for Children - Philadelphia (US), Shriners Hospitals for Children - Chicago (US), McGill University (CA)
No poverty
Openalex Percentile: Top 21%
Neurogenetic and Muscular Disorders Research
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