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.
Authors
- Lauren C. Hyer (ORCID: https://orcid.org/0000-0003-3569-2513)
- Sarah Beth Nossov (ORCID: https://orcid.org/0000-0003-1191-074X)
- Joel A. Lerman
- Krister Freese
- Cary H. Mielke
- Noémi Dahan‐Oliel (ORCID: https://orcid.org/0000-0001-8567-7173)
- Thania Ordaz‐Robles
- Haluk Altiok
- F Rauch
- Ahlam Zidan
- Emmanouil Rampakakis
- Reggie Hamdy
- Sophia C. Pasia
- Philip F. Giampietro
Institutions
- 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)
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
- Field-Weighted Citation Impact
- 0.00