The Impact on Families of Children With Congenital Upper Extremity Differences

Sarah Ballatori; Andrea Bauer; Angela Wang; Julie Samora; Suzanne Steinman; Claire Manske; Apurva Shah; Lindley B Wall; CoULD Study Group

doi:10.1016/j.jhsa.2024.02.011

The Impact on Families of Children With Congenital Upper Extremity Differences

J Hand Surg Am. 2024 Apr 16:S0363-5023(24)00095-9. doi: 10.1016/j.jhsa.2024.02.011. Online ahead of print.

Authors

Sarah Ballatori¹, Andrea Bauer², Angela Wang³, Julie Samora⁴, Suzanne Steinman⁵, Claire Manske⁶, Apurva Shah⁷, Lindley B Wall⁸; CoULD Study Group

Affiliations

¹ Department of Orthopaedic Surgery, Washington University School of Medicine, Saint Louis, MO.
² Department of Orthopaedic Surgery, Boston Children's Hospital, Boston, MA.
³ Department of Orthopeadic Surgery, University of Utah, Salt Lake City, UT.
⁴ Department of Orthopaedic Surgery, Nationwide Children's Hospital, Columbus, OH.
⁵ Department of Orthopeadic Surgery, Seattle Children's Hospital, Seattle, WA.
⁶ Department of Orthopaedic Surgery, Shriner's Hospital, Sacramento, CA.
⁷ Department of Orthopaedic Surgery, Children's Hospital of Philadelphia, Philadelphia, PA.
⁸ Department of Orthopaedic Surgery, Washington University School of Medicine, Saint Louis, MO. Electronic address: wallli@wustl.edu.

PMID: 38639681
DOI: 10.1016/j.jhsa.2024.02.011

Abstract

Purpose: To investigate the impact on caregivers of caring for a child with congenital upper extremity differences.

Methods: In this cross-sectional study, caregivers of patients enrolled in the multi-institutional Congenital Upper Limb Difference (CoULD) registry were contacted. Demographic information and the Impact on Family Scale (IOFS), a validated measure of perceived caregiver strain, were collected. Patient-reported outcome measures from the CoULD registry, the Pediatric Outcomes Data Collection Instrument (PODCI), and Patient-Reported Outcomes Measurement Information System (PROMIS) were also analyzed for correlation with IOFS.

Results: Two hundred ninety-nine caregivers participated. Factors with significantly stronger impact on family included public insurance; bilateral upper extremity involvement; household income of $20,000-40,000; additional musculoskeletal diagnosis; and a single adult caregiver household. There was a significantly increased subcategory of IOFS-Finance score for distant travel to see the surgeon. Additionally, all categories of the PODCI (upper extremity, mobility, sports, pain, happiness, and global) demonstrated a negative correlation with IOFS. PROMIS upper extremity and peer relations also demonstrated an inverse relationship with IOFS, whereas PROMIS pain interference had a positive correlation with IOFS. The overall IOFS for children with CoULDs was greater than previously reported for children with brachial plexus birth injury, and less than cerebral palsy and congenital heart disease.

Conclusions: Caregivers of children with congenital upper extremity differences report a significant impact on family life. Socioeconomic factors, such as economically disadvantaged or single-caregiver households, and clinical factors, such as bilateral upper extremity involvement, correlate with greater family impact. These findings represent opportunities to identify at-risk families and underscore the importance of caring for the whole family through a multidisciplinary approach.

Type of study/level of evidence: Prognostic II.

Keywords: Congenital; PROMIS; families; impact; patient-reported outcomes.