BACKGROUND: The Pediatric Outcomes Data Collection Instrument (PODCI) is a subjective measurement tool designed to provide a standardized method of assessing outcomes in pediatric musculoskeletal conditions. It has earlier been shown to be useful in several pediatric musculoskeletal conditions, but there is currently no widely accepted subjective outcome measurement tool for children with arthrogryposis. METHODS: The PODCI was administered to parents of 74 children diagnosed with amyoplasia. The score distributions were compared with values published earlier for children without musculoskeletal disorders. For those patients with repeated PODCI administrations over time, the initial score was compared with the most recent score. Comparisons were made using the Student t test. RESULTS: PODCI scores in children with amyoplasia were significantly lower than those for typically developing children in all 6 domains. The scores were also more normally distributed than those for typically developing children in all 6 domains. Over an average follow-up period of approximately 3 years, children with amyoplasia had a statistically significant increase in scores for upper extremity function, sports participation, and global functioning. CONCLUSIONS: These results show that the PODCI is useful in evaluating functional outcomes of children with amyoplasia, and is sensitive to change in function over time. The PODCI shows promise as a tool to evaluate long-term outcomes of surgical management in amyoplasia. LEVEL OF EVIDENCE: Diagnostic Study, Level III.
BACKGROUND: The Pediatric Outcomes Data Collection Instrument (PODCI) is a subjective measurement tool designed to provide a standardized method of assessing outcomes in pediatric musculoskeletal conditions. It has earlier been shown to be useful in several pediatric musculoskeletal conditions, but there is currently no widely accepted subjective outcome measurement tool for children with arthrogryposis. METHODS: The PODCI was administered to parents of 74 children diagnosed with amyoplasia. The score distributions were compared with values published earlier for children without musculoskeletal disorders. For those patients with repeated PODCI administrations over time, the initial score was compared with the most recent score. Comparisons were made using the Student t test. RESULTS: PODCI scores in children with amyoplasia were significantly lower than those for typically developing children in all 6 domains. The scores were also more normally distributed than those for typically developing children in all 6 domains. Over an average follow-up period of approximately 3 years, children with amyoplasia had a statistically significant increase in scores for upper extremity function, sports participation, and global functioning. CONCLUSIONS: These results show that the PODCI is useful in evaluating functional outcomes of children with amyoplasia, and is sensitive to change in function over time. The PODCI shows promise as a tool to evaluate long-term outcomes of surgical management in amyoplasia. LEVEL OF EVIDENCE: Diagnostic Study, Level III.
Authors: Giovanni Trisolino; Stefano Stallone; Paola Zarantonello; Andrea Evangelista; Manila Boarini; Jacopo Faranda Cordella; Luca Lerma; Luisa Veronesi; Cosma Caterina Guerra; Luca Sangiorgi; Giovanni Luigi Di Gennaro; Renato Maria Toniolo Journal: Children (Basel) Date: 2022-06-08
Authors: Noémi Dahan-Oliel; Sarah Cachecho; Douglas Barnes; Tanya Bedard; Ann M Davison; Klaus Dieterich; Maureen Donohoe; Alicja Fąfara; Reggie Hamdy; Helgi T Hjartarson; Naimisha S Hoffman; Eva Kimber; Igor Komolkin; Ruth Lester; Eva Pontén; Harold J P van Bosse; Judith G Hall Journal: Am J Med Genet C Semin Med Genet Date: 2019-07-07 Impact factor: 3.908
Authors: Noémi Dahan-Oliel; Sarah Cachecho; Alicja Fąfara; Francis Lacombe; Ani Samargian; André Bussières Journal: Res Involv Engagem Date: 2022-02-18
Authors: Sarah Cachecho; Jill Boruff; Trudy Wong; Francis Lacombe; Noemi Dahan-Oliel Journal: Health Qual Life Outcomes Date: 2021-11-29 Impact factor: 3.186