Do Patient- and Parent-reported Outcomes Measures for Children With Congenital Hand Differences Capture WHO-ICF Domains?

BACKGROUND: Patient- and parent-reported outcome measures (PROMs) are increasingly used to evaluate the effectiveness of surgery for congenital hand differences (CHDs). Knowledge of an existing outcome measure's ability to assess self-reported health, including psychosocial aspects, can inform the future development and application of PROMs for CHD. However, the extent to which measures used among children with CHD align with common, accepted metrics of self-reported disability remains unexplored.

QUESTIONS/PURPOSES: We reviewed studies that used PROMs to evaluate surgery for CHD to determine (1) the number of World Health Organization-International Classification of Functioning, Disability and Health (WHO-ICF) domains covered by existing PROMs; (2) the proportion of studies that used PROMs specifically validated among children with CHD; and (3) the proportion of PROMs that targets patients and/or parents.

METHODS: We performed a comprehensive review of the literature through a bibliographic search of MEDLINE®, PubMed, and EMBASE from January 1966 to December 2014 to identify articles related to patient outcomes and surgery for CHD. We evaluated the 42 studies that used PROMs to identify the number and type of WHO-ICF domains captured by existing PROMs for CHD and the proportion of studies that use PROMs validated for use among children with CHD. The most common instruments used to measure patient- and parent-reported outcomes after reconstruction for CHD included the Prosthetic Upper Extremity Functional Index (PUFI), Disabilities of the Arm, Shoulder, and Hand questionnaire, Childhood Experience Questionnaire, and Pediatric Quality of Life Inventory.

RESULTS: Current PROMs that have been used for CHD covered a mean of 1.3 WHO-ICF domains (SD ± 1.3). Only the Child Behavior Checklist and the Piers-Harris Children's Self-Concept Scale captured all ICF domains (body functions and structures, activity, participation, and environmental factors). The PUFI, the only PROM validated specifically for children with congenital longitudinal and transverse deficiency, was used in only four of 42 studies. Only 13 of the 42 studies assessed patient-reported outcomes, whereas five assessed both patient- and parent-reported outcomes.

CONCLUSIONS: The PROMs used to assess patients after CHD surgery do not evaluate all WHO-ICF domains (ie, body structure, body function, environmental factors, and activity and participation) and generally are not validated for children with CHD. Given the psychological and sociological aspects of CHD illness, a PROM that encompasses all components of the biopsychosocial model of illness and validated in children with CHD is desirable.

LEVEL OF EVIDENCE: Level III, therapeutic study.