BACKGROUND: In the United States, congenital cytomegalovirus disease (cCMVd) is the leading cause of non-genetic sensorineural hearing loss and causes other permanent disabilities. The impact of cCMVd on the patient, caregiver, and family is thought to be substantial but is poorly documented. Our objective was to develop a patient and caregiver survey to collect data on the economic and humanistic burden of cCMVd, in order to describe health-related quality of life (HRQOL), direct, and indirect non–health care costs among cCMVd patients and caregivers.
METHODS: Standard survey methodology was employed in study design and development of the protocol and questionnaire. A conceptual framework communicating the lifetime economic burden of cCMVd informed study design and questionnaire content. Input was obtained from cCMVd clinical experts, representative stakeholders, and by review of scientific literature.
RESULTS: Due to cCMVd’s multiple potential outcomes and impacts among patients, caregivers and families, it is not possible to characterize HRQOL and economic burden with a single instrument. Three validated instruments were identified to describe HRQOL and burden in caregivers (12-item Short Form Health Survey [SF-12], Work Productivity and Activity Impairment Questionnaire-General Health [WPAI-GH], Zarit Burden Interview), two validated instruments were identified for use with adult patients (SF-12 and WPAI-GH), and one for use with pediatric patients, (Pediatric Quality of Life Inventory). Additional questions were developed to collect data on caregiver economic impacts and mental health needs, as well as economic impacts on adults with cCMVd. Stakeholder feedback and cognitive debriefing of additional questions will be conducted prior to full survey study implementation.
CONCLUSIONS: Evidence on how cCMVd affects the physical, emotional, economic and social functioning of patients and caregivers is important for policy and health care resource allocation decisions. We anticipate this survey will characterize the economic and humanistic burden experience of those affected by cCMVd.
