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Correlations between patient-reported outcomes and self-reported characteristics in adults with hemophilia B and caregivers of children with hemophilia B: analysis of the B-HERO-S study

Authors Buckner TW, Sidonio Jr R, Witkop M, Guelcher C, Cutter S, Iyer NN, Cooper DL

Received 12 June 2019

Accepted for publication 19 August 2019

Published 18 September 2019 Volume 2019:10 Pages 299—314


Checked for plagiarism Yes

Review by Single-blind

Peer reviewer comments 2

Editor who approved publication: Professor Lynne Nemeth

Tyler W Buckner,1 Robert Sidonio Jr,2 Michelle Witkop,3 Christine Guelcher,4 Susan Cutter,5 Neeraj N Iyer,6 David L Cooper6

1Hemophilia and Thrombosis Center, University of Colorado School of Medicine, Aurora, CO, USA; 2Department of Pediatrics, Aflac Cancer and Blood Disorders Center, Emory University and Children’s Healthcare of Atlanta, Atlanta, GA, USA; 3National Hemophilia Foundation, New York, NY, USA; 4Hemostasis and Thrombosis Program, Center for Cancer and Blood Disorders, Children’s National Health System, Washington, DC, USA; 5Penn Comprehensive Hemophilia and Thrombosis Center, Hospital of the University of Pennsylvania, Philadelphia, PA, USA; 6Novo Nordisk Inc., Plainsboro, NJ, USA

Correspondence: Tyler W Buckner
Hemostasis and Thrombosis Center, University of Colorado School of Medicine, Anschutz Medical Campus, 13199 East Montview Blvd, Suite 100, Mailstop F416, Aurora, CO 80045, USA
Tel +1 303 724 0724
Fax +1 303 724 0947

Purpose: Pain, anxiety, depression, and other aspects of health-related quality of life (HRQoL) are important issues for people with hemophilia and caregivers of children with hemophilia. Patient-reported outcome (PRO) instruments may be used to assess aspects of HRQoL; however, the use of PROs in clinical management of patients with hemophilia is limited and inconsistent. The Bridging Hemophilia B Experiences, Results and Opportunities Into Solutions (B-HERO-S) study evaluated the impact of hemophilia B on HRQoL and other psychosocial aspects in affected adults and caregivers of children with hemophilia B. This post hoc analysis assessed correlations between PRO scores and psychosocial questions commonly asked in comprehensive care settings among B-HERO-S respondents.
Patients and methods: B-HERO-S consisted of two online surveys, one administered to adults with hemophilia B (n=299) and one administered to caregivers of children with hemophilia B (n=150). The adult survey included EQ-5D-5L with visual analog scale, BPI, HAL, and PHQ-9. The caregiver survey included PHQ-9 and GAD-7. Questions related to demographics, hemophilia treatment, and psychosocial questions asked in comprehensive care visits were also included in the surveys. A post hoc analysis was performed to assess correlations between responses to selected psychosocial questions with PRO scores.
Results: For adults with hemophilia B, greater pain severity and pain interference scores were associated with work-related problems, functional limitations, and relationship, psychological, and treatment issues. Significant correlations were also noted between some of these psychosocial outcomes and depressive symptoms. For caregivers, greater depression and anxiety were associated with employment issues, their child’s functional, relationship, and psychological issues, having had difficulty or concerns with treatment/factor availability or affordability, and having less frequent HTC visits.
Conclusion: High correlations were observed between PRO scores measuring pain, depression, and anxiety and questions commonly used in the comprehensive care setting to assess the psychosocial impact of hemophilia.

Keywords: health-related quality of life, depression, anxiety, employment, relationships

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