Patient-reported Outcomes in Arthrogryposis.
Arthrogryposis
/ epidemiology
Child
Female
Humans
Longitudinal Studies
Male
Patient Reported Outcome Measures
Physical Functional Performance
Prospective Studies
Psychosocial Functioning
Registries
/ statistics & numerical data
Surveys and Questionnaires
United States
/ epidemiology
Upper Extremity
/ physiopathology
Journal
Journal of pediatric orthopedics
ISSN: 1539-2570
Titre abrégé: J Pediatr Orthop
Pays: United States
ID NLM: 8109053
Informations de publication
Date de publication:
Aug 2020
Aug 2020
Historique:
pubmed:
11
2
2020
medline:
15
12
2020
entrez:
11
2
2020
Statut:
ppublish
Résumé
Little is known about patient-reported health status in children and adolescents with arthrogryposis. Utilizing the Patient-Reported Outcome Measurement Information System (PROMIS) and Pediatric Outcomes Data Collection Instrument (PODCI) questionnaires, we investigated functional and psychosocial measures in arthrogryposis. A total of 118 patients with arthrogryposis were identified from a prospective longitudinal cohort (the Congenital Upper Limb Difference Registry) from 2014 to 2018. Demographics and patient-reported outcome measures were evaluated, including the PROMIS [upper extremity (UE) function, pain, depression, anxiety, and peer relations] and PODCI questionnaires (UE function, pain, happiness, and global function). A total of 29 arthrogrypotic patients had complete PROMIS and PODCI data. This cohort was divided into distal arthrogryposis and amyoplasia groups, with 15 and 14 patients in each group, respectively. There were 8 males in the distal arthrogryposis group with a median age of 9 years and 7 males in the amyoplasia group with a median age of 8 years. For both cohorts, the median UE function PROMIS scores were significantly below population norms, 31 for distal arthrogryposis and 22 for amyoplasia. PODCI UE function was statistically lower for amyoplasia compared with the distal arthrogryposis cohort. PROMIS pain, depression, anxiety, and peer relations were in the normal range for both amyopasia and distal arthrogryposis. Median PODCI pain and happiness ranged from 85 to 88 for all patients with no statistical difference between groups. Arthrogryposis patients have lower UE function scores compared with population normals, but they have emotional states that are consistent with populations norms. Amyoplasia patients were functionally worse than distal arthrogryposis patients. Level II.
Sections du résumé
BACKGROUND
BACKGROUND
Little is known about patient-reported health status in children and adolescents with arthrogryposis. Utilizing the Patient-Reported Outcome Measurement Information System (PROMIS) and Pediatric Outcomes Data Collection Instrument (PODCI) questionnaires, we investigated functional and psychosocial measures in arthrogryposis.
METHODS
METHODS
A total of 118 patients with arthrogryposis were identified from a prospective longitudinal cohort (the Congenital Upper Limb Difference Registry) from 2014 to 2018. Demographics and patient-reported outcome measures were evaluated, including the PROMIS [upper extremity (UE) function, pain, depression, anxiety, and peer relations] and PODCI questionnaires (UE function, pain, happiness, and global function).
RESULTS
RESULTS
A total of 29 arthrogrypotic patients had complete PROMIS and PODCI data. This cohort was divided into distal arthrogryposis and amyoplasia groups, with 15 and 14 patients in each group, respectively. There were 8 males in the distal arthrogryposis group with a median age of 9 years and 7 males in the amyoplasia group with a median age of 8 years. For both cohorts, the median UE function PROMIS scores were significantly below population norms, 31 for distal arthrogryposis and 22 for amyoplasia. PODCI UE function was statistically lower for amyoplasia compared with the distal arthrogryposis cohort. PROMIS pain, depression, anxiety, and peer relations were in the normal range for both amyopasia and distal arthrogryposis. Median PODCI pain and happiness ranged from 85 to 88 for all patients with no statistical difference between groups.
CONCLUSIONS
CONCLUSIONS
Arthrogryposis patients have lower UE function scores compared with population normals, but they have emotional states that are consistent with populations norms. Amyoplasia patients were functionally worse than distal arthrogryposis patients.
LEVELS OF EVIDENCE
METHODS
Level II.
Identifiants
pubmed: 32040062
doi: 10.1097/BPO.0000000000001527
pii: 01241398-202008000-00025
doi:
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
357-360Références
Daltroy LH, Liang MH, Fossel AH, et al. The POSNA pediatric musculoskeletal functional health questionnaire: report on reliability, validity, and sensitivity to change. Pediatric Outcomes Instrument Development Group. Pediatric Orthopaedic Society of North America. J Pediatr Orthop. 1998;18:561–571.
Cella D, Riley W, Stone A, et al. The Patient-Reported Outcomes Measurement Information System (PROMIS) developed and tested its first wave of adult self-reported health outcome item banks: 2005-2008. J Clin Epidemiol. 2010;63:1179–1194.
Fries JF, Bruce B, Cella D. The promise of PROMIS: using item response theory to improve assessment of patient-reported outcomes. Clin Exp Rheumatol. 2005;23(5 suppl 39):S53–S57.
DeWalt DA, Gross HE, Gipson DS, et al. PROMIS® pediatric self-report scales distinguish subgroups of children within and across six common pediatric chronic health conditions. Qual Life Res. 2015;24:2195–2208.
Howell CR, Thompson LA, Gross HE, et al. Responsiveness to change in PROMIS® measures among children with asthma: a report from the PROMIS® pediatric asthma study. Value Health. 2016;19:192–201.
Ranganathan K, Shapiro D, Carlozzi NE, et al. The feasibility and validity of PROMIS: a novel measure of quality of life among children with cleft lip and palate. Plast Reconstr Surg. 2016;138:675e–681e.
Morgan EM, Mara CA, Huang B, et al. Establishing clinical meaning and defining important differences for Patient-Reported Outcomes Measurement Information System (PROMIS®) measures in juvenile idiopathic arthritis using standard setting with patients, parents, and providers. Qual Life Res. 2017;26:565–586.
Selewski DT, Troost JP, Cummings D, et al. Responsiveness of the PROMIS® measures to changes in disease status among pediatric nephrotic syndrome patients: a Midwest pediatric nephrology consortium study. Health Qual Life Outcomes. 2017;15:166.
Forrest CB, Zorc JJ, Moon J, et al. Evaluation of the PROMIS pediatric global health scale (PGH-7) in children with asthma. J Asthma. 2019;56:534–542.
Lai JS, Kupst MJ, Beaumont JL, et al. Using the Patient-Reported Outcomes Measurement Information System (PROMIS) to measure symptoms burden reported by patients with brain tumors. Pediatr Blood Cancer. 2019;66:e27526.
Gerull WD, Okoroafor UC, Guattery J, et al. Performance of pediatric PROMIS CATs in children with upper extremity fractures. Hand (N Y). 2018: 1558944718793195. [Epub ahead of print].
Bae DS, Canizares MF, Miller PE, et al. Functional impact of congenital hand differences: early results from the Congenital Upper Limb Differences (CoULD) Registry. J Hand Surg Am. 2018;43:321–330.
Waljee JF, Carlozzi N, Franzblau LE, et al. Applying the patient-reported outcomes measurement information system to assess upper extremity function among children with congenital hand differences. Plast Reconstr Surg. 2015;136:200e–207e.
Bamshad M, Van Heest AE, Pleasure D. Arthrogryposis: a review and update. J Bone Joint Surg Am. 2009;91(suppl 4):40–46.
Steen U, Christensen E, Samargian A. Adults living with amyoplasia: function, psychosocial aspoects, and the benefits of AMC support groups. J Pediatr Orthop. 2017;37:S31–S32.
Sodergard J, Hakamies-Blomqvist L, Sainio K, et al. Arthrogryposis Multiplex Congenita: Perinatal and electromyographic findings, disability, and psychosocial outcome. J Pediatr Orthop. 1997;6:167–171.
Amor CJ, Spaeth MC, Chafey DH, et al. Use of the pediatric outcomes data collection instrument to evaluate functional outcomes in arthrogryposis. J Pediatr Orthop. 2011;31:293–296.