Assessing the impact of grief on quality of life, work productivity, and health outcomes for parents bereaved from SMA: A study protocol

Cost Effectiveness and Resource Allocation

Study Protocol

Abigail G. Riley¹  Tara A. Lavelle¹  Lisa Belter²  Colleen McCarthy O’Toole²  Christina M. Mulé³  Tamara Vesel⁴  David Fox⁵  Sheila Shapouri⁵  Stacey Kowal⁵  Debra Lerner⁶ 

[1] Center for the Evaluation of Value and Risk in Health, Institute for Clinical Research and Health Policy Studies, Tufts Medical Center, Boston, MA, USA;Cure SMA, Elk Grove Village, IL, USA;Department of Pediatrics, University of Rochester Medical Center, Rochester, NY, USA;Department of Pediatrics, Tufts Medical Center, Tufts University School of Medicine, Boston, MA, USA;Division of Palliative Care, Department of Medicine, Tufts Medical Center, Boston, MA, USA;Genentech, Inc, South San Francisco, CA, USA;Program on Health, Work and Productivity, Institute for Clinical Research and Health Policy Studies, Tufts Medical Center and Tufts University School of Medicine Department of Psychiatry, Boston, MA, USA;

关键词: Grief;    Health-related quality of life;    Work productivity;    Spinal muscular atrophy;    Mental health;   

DOI  :  10.1186/s12962-023-00465-5

 received in 2023-05-17, accepted in 2023-08-02,  发布年份 2023

来源: Springer

【 摘 要 】

BackgroundU.S. cost-effectiveness recommendations suggest that analyses should include all costs and effects relevant to the decision problem [1]. However, in many diseases, including spinal muscular atrophy (SMA), few studies have evaluated bereaved family outcomes after a child has died, neglecting potential impacts on their health-related quality of life (HRQoL), work productivity, and mental health. Additionally, grief-related outcomes are rarely included in economic evaluations. This manuscript outlines the protocol of a study that will estimate the HRQoL, work functioning, and mental health of bereaved parents of children with SMA type 1 to determine how outcomes vary based on parent’s sex and the time since a child’s death.MethodsThis study will involve two phases. In Phase 1, we will conduct a literature review to identify prior research that has measured how parental grief impacts HRQoL, work productivity, and mental health. We will also interview four bereaved parents of children with SMA type 1, stratified by parent sex and time since their child’s death, and analyze findings using a thematic analysis. In Phase 2, we will develop a survey draft based on Phase 1 findings. Parents bereaved from SMA type 1 will review our survey draft and we will revise the survey based on their feedback. We will send a cross-sectional survey to approximately 880 parents bereaved from SMA type 1. We will analyze findings from the survey to investigate whether the severity of grief symptoms is correlated with HRQoL, productivity, depression and anxiety symptom severity. We will also evaluate whether the mean scores of grief and each of the outcomes vary significantly when stratified by parent sex and the time since the child’s death.DiscussionOur results will provide preliminary information on how parental grief can impact HRQoL, productivity, and mental health outcomes over time. Increasing the availability of family outcomes data will potentially assist organizations performing health economic evaluations, such as the Institute of Clinical and Economic Review (ICER) in the U.S. This research will also help to inform the development of future economic guidelines on this topic.

【 授权许可】

CC BY   
© BioMed Central Ltd., part of Springer Nature 2023