DEVELOPMENT OF A COST OF CARE QUESTIONNAIRE FOR CHILDREN WITH ARTHROGRYPOSIS MULTIPLEX CONGENITA: CAREGIVER PERSPECTIVE

R.U. Elekanachi¹, S. Nematollahi^1,2, S. Cachecho², N. Dahan-Oliel^1,2, L. Snider¹

¹McGill University, School of Physical and Occupational Therapy, Montreal, Canada, ²Shriners Hospital for Children, Montreal, Canada

Background: Cost of care known as Cost of illness (COI) ranges from the incidence or prevalence of conditions to its effect on longevity, economic resources, morbidity with the decrease in health status, quality of life, and financial aspects including costs that result from disability, due to corresponding conditions and/or its comorbidities. Rare musculoskeletal (MSK) conditions (e.g., Arthrogryposis Multiplex Congenita (AMC)), are associated with an enormous health impact, affecting patients’ health, and causing direct and indirect costs for their families/caregivers. Families have reported that MSK conditions result in significant economic impact, but the exact cost is unknown despite it being an emerging area of study. A recent literature review on COI studies in childhood disability, reports that researchers fail to mention or provide the questionnaire used, and the validation process of these questionnaires is not mentioned. Although the identified questionnaires collected data that included demographics, income, and direct, and indirect costs, some studies lacked the psychosocial costs, and information on the questionnaire or adaptation choices posing challenges for further study replication in other conditions such as AMC.

Purpose: The aim of this study was to create a tool to measure the cost of caring for children with AMC from the caregivers’ perspectives. Specifically, Identify and develop items for a new measure on the cost of caring for a child with AMC; Estimate the extent to which the newly developed tool is acceptable and feasible by the target audience (caregivers of children with AMC); Contribute evidence towards the validation for use of the measure with the target audience.

Methods: This questionnaire was developed using an iterative design process that included a literature review, a review of existing measures, pilot testing, and translation with each phase contributing to its development. 5 Caregiver representatives and 10 health care other professionals were involved in the development, piloting, and validation of this tool. The questionnaire was created in English and translated into both French and Spanish.

Results: The questionnaire covers the following domain: Child and caregiver's demographics (e.g., age, gender, date of birth, ethnicity), Caregiver's socio-demographics (e.g., education, marital status, income), and Cost information (direct, indirect cost, and psychosocial costs). This tool was determined to be completed within 45mins of administration and is hosted on Qualtrics for electronics administration.

Conclusions: This tool was collaboratively developed with caregiver representatives, healthcare, and other professionals. Therefore, this tool will support economic evaluations of the impact of care on caregivers of children with disabilities. The development and validation process of this tool will also help other researchers to adapt this questionnaire for other studies, and develop similar questionnaires.

Implications: The estimation of the cost of caring for a child with AMC aids both researchers and policymakers to make informed decisions on its impact on the healthcare system and motivates change in access to care. Therefore, by measuring and comparing the cost of care to society, healthcare decision makers can benefit in setting up and prioritizing healthcare policies and interventions that are supposed to be implemented which in turn would benefit children with AMC and their families.

Funding acknowledgements: There are no funding acknowledgements to declare.

Keywords:
Cost of care
Childhood disabilities
Caregivers

Topics:
Paediatrics
Disability & rehabilitation
Research methodology, knowledge translation & implementation science

Did this work require ethics approval? No

Reason: This questionnaire was done by reviewing existing studies and questionnaires on the cost of care, cost of illness, and burden of care. Also in order to validate the questionnaire, caregiver representatives, health care, and other professionals were consulted to ensure the tool was rigorously developed. There was also low- to no- risk associated with the creation of the questionnaire.