The Final 24-Item Early Onset Scoliosis Questionnaires (EOSQ-24): Validity, Reliability and Responsiveness.
BACKGROUND
The goal of early-onset scoliosis (EOS) treatment is to improve health-related quality of life (HRQoL) for patients and to reduce the burden on their parents or caregivers. The purpose of this study is to develop and finalize the 24-item Early-Onset Scoliosis Questionnaire (EOSQ-24), and examine the validity, reliability, and responsiveness of the EOSQ-24 in measuring patients' HRQoL, the burden on their caregivers, and the burden on their caregiver's finances. The study also established aged-matched normative values for the EOSQ-24.


METHODS
The EOSQ-24 was administered to caregivers of male and female patients aged 0 to 18 years with EOS. Patients with EOS are diagnosed before 10 years of age. Criterion validity was investigated by measuring agreement between its scores and pulmonary function testing. Construct validity was established by comparing values across different etiology groups using the known-group method, and measuring internal consistency reliability. Content validity was confirmed by reviewing caregiver and health provider ratings for the relativity and clarity of the EOSQ-24 questions. Test-retest reliability was examined through intraclass correlation coefficients. Responsiveness of the EOSQ-24 before and after surgical interventions was also investigated. Age-matched, healthy patients, without spinal deformity, were enrolled to establish normative EOSQ-24 values.


RESULTS
The pulmonary function subdomain score in the EOSQ-24 was positively correlated with pulmonary function testing values, establishing criterion validity. The EOSQ-24 scores for neuromuscular patients were significantly decreased compared with idiopathic or congenital/structural patients, demonstrating known-group validity. Internal consistency reliability of patients' HRQoL was excellent (0.92), but Family Burden was questionable (0.64) indicating that Parental Burden and Financial Burden should be in separate domains. All 24 EOSQ items were rated as essential and clear, confirming content validity. All EOSQ-24 domains demonstrated good to excellent agreement (0.68 to 0.98) between test and retest scores. Neuromuscular patients improved their HRQoL postoperatively, whereas idiopathic patients worsened their HRQoL postoperatively, indicating the ability of the instrument to respond to different trajectories of HRQoL according to etiology in patients with EOS.


CONCLUSIONS
The developed and finalized EOSQ-24 is a valid, reliable, and responsive instrument that is able to serve as a patient-reported outcome measure evaluating health status for patients with EOS and burden of their caregivers. This instrument will be able to serve as an outcome measure for future research including clinical trials evaluating the effectiveness of various treatments. In addition, the EOSQ-24 allows assessment of patients' HRQoL, and the burden on their caregivers relative to age-matched healthy peers.


METHODS
Level II-diagnostic study with consecutive patients enrolled in national registries.
Department of Orthopaedic Surgery, Columbia University Medical Center.|Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY.
Hiroko
H
Matsumoto
Department of Orthopaedics and Rehabilitation, University of Florida, Gainesville, FL.
Brendan
B
Williams
Department of Orthopaedic Surgery, David Geffen School of Medicine at University of California at Los Angeles.
Howard Y
HY
Park
Department of Orthopaedic Surgery, Columbia University Medical Center.
Julie Y
JY
Yoshimachi
Department of Orthopaedic Surgery, Columbia University Medical Center.
Benjamin D
BD
Roye
Department of Orthopaedic Surgery, Columbia University Medical Center.
David P
DP
Roye
Department of Orthopaedics, University of California, San Diego, La Jolla, CA.
Behrooz A
BA
Akbarnia
Department of Orthopaedic Surgery, Children's Hospital, Harvard Medical School, Boston, MA.
John
J
Emans
Department of Orthopaedic Surgery, David Geffen School of Medicine at University of California at Los Angeles.|Children's Orthopaedic Center, Children's Hospital Los Angeles, Los Angeles.
David
D
Skaggs
Department of Orthopaedics and Pediatrics, Primary Children's Medical Center, The University of Utah School of Medicine, Salt Lake City, UT.
John T
JT
Smith
Department of Orthopaedic Surgery, Columbia University Medical Center.
Michael G
MG
Vitale
D000293:Adolescent; D017028:Caregivers / Q000523:psychology*; D002648:Child; D002675:Child, Preschool; D005260:Female; D006801:Humans; D007223:Infant; D007231:Infant, Newborn; D008297:Male; D017063:Outcome Assessment, Health Care; D010290:Parents / Q000523:psychology*; D011184:Postoperative Period; D011594:Psychometrics; D011788:Quality of Life; D012042:Registries; D015203:Reproducibility of Results; D012600:Scoliosis / Q000175:diagnosis* / Q000601:surgery; D011795:Surveys and Questionnaires