Caregiver burden in haemophilia: results from a single UK centre


Haemophilia caregivers face limitations in their life leading to perceived burden. This single-centre study investigates the impact of burden on caregivers’ health-related quality of life (HRQoL). Methods: Questionnaires for caregivers comprised demographic data, HRQoL (EQ-5D, SF-36) and caregiver burden (IOF: Impact on Family Scale). Children were also asked about their HRQoL (EQ-5D, Haemo-QoL Short Form). Results: 20 consecutive parent/child dyads participated. 80% were mothers (mean age of 39.80±6.2 years) with 1-3 haemophilic children aged 8-17 years and 2.5±1.2 children <18 years living in the household. Mothers did most childcare (80%), 50% worked part-time, and 55% reported that haemophilia had an economic impact on their family. 80% of boys had haemophilia A; 60% were severely affected. Most received home treatment (85%) and prophylaxis (80%). Caregivers’ and boys’ HRQoL was similarly good (EQ-5Dparents M=0.90±0.1 vs. EQ-5Dchild M=0.81±0.3); by contrast, boys reported better values in the EQ-VAS (Mchild=90.25±10.0 vs. Mparents=82.16±14.8). Caregivers reported highest impairments in the dimensions ‘vitality’ (M=60.00±20.5) and ‘emotional role’ (M=70.37±42.6) of the SF-36. In the IOF, caregivers reported highest impairments in the dimension ‘negative impact’ (M=60.08±20.7). Caregivers reporting high burden had significantly worse HRQoL in the domains ‘bodily pain’ (p<.028) and ‘social functioning’ (p<0.024) of the SF-36. Caregivers who reported that haemophilia had an economic impact on their family and those with a chronic disease showed significant higher impairments in caregiver burden and their HRQoL. Conclusions: The perceived burden of haemophilia has a direct impact on caregivers’ HRQoL. Further studies with haemophilia-specific instruments are needed to verify these findings.

If the inline PDF is not rendering correctly, you can download the PDF file here.

  • 1. 2015 National Haemophilia Database Annual Returns for 2014. Available at last accessed 10th April 2016

  • 2. Valentino LA. Blood-induced joint disease: the pathophysiology of hemophilia arthropathy. J Thomb Haemost 2010; 8: 1895- 902.

  • 3. Biggs R, MacFarlane RG. Haemophilia and the related conditions: a survey of 187 cases. Br J Haematol 1958; 4: 1-27.

  • 4. Richards M, Williams M, Chalmers E, et al.; Paediatric Working Party of the United Kingdom Haemophilia Doctors’ Organisation. A United Kingdom Haemophilia Centre Doctors’ Organization guideline approved by the British Committee for Standards in Haematology: guideline on the use of prophylactic factor VIII concentrate in children and adults with severe haemophilia A. Br J Haematol. 2010; 149(4): 498-507.

  • 5. Vidler V. Teaching parents advanced clinical skills. Haemophilia. 1999; 5(5): 349-53.

  • 6. Beeton K, Neal D, Watson T, Lee CA. Parents of children with haemophilia - a transforming experience. Haemophilia. 2007; 13(5): 570-9.

  • 7. Myrin Westesson L, Sparud-Lundin C, Wallengren C, Baghaei F. A tortuous route to a capable fatherhood: the experience of being a father to a child with severe haemophilia. Haemophilia. 2015; 21(6): 799-805.

  • 8. Myrin-Westesson L, Baghaei F, Friberg F. The experience of being a female carrier of haemophilia and the mother of a haemophilic child. Haemophilia. 2013; 19(2): 219-24.

  • 9. Torres-Ortuno A, Cuesta-Barriuso R, Nieto-Munuera J. Parents of children with haemophilia at an early age: assessment of perceived stress and family functioning. Haemophilia 2014; 20: 756-62.

  • 10. Weidebusch S, Pollman H, Siegmund B, Muthny F. Quality of life, psychosocial strains and coping in parents of children with haemophilia. Haemophilia 2008; 14(5): 1014-22.

  • 11. Von Mackensen S, Gringeri A. Quality of life in haemophilia. In: Preedy VR, Watson RR (eds). Handbook of Disease Burdens and Quality of Life Measures. Heidelberg: Springer; 2009. Vol 3, Chapter 12, 1895-920.

  • 12. Von Mackensen S, Gringeri A. Quality of life in hemophilia. In: Lee CA, Berntop EE, Hoots WK (eds). Textbook of Hemophilia, 3rd edn. Oxford: Wiley Blackwell Publishing Ltd; 2014, 478-88.

  • 13. Department of Health. Equity and excellence: liberating the NHS. London: HMSO; 2010. Available from (accessed 15 December 2016).

  • 14. Szende A, Oppe M, Devlin N (eds). EQ-5D Value Sets: Inventory, Comparative Review and User Guide. Doordrecht: Springer; 2010.

  • 15. Ware J Jr, Kosinski M, Keller SD. SF-36 Physical and Mental Health Summary Scales: A User’s Manual. Boston, Massachussetts: The Health Institute, 1994.

  • 16. Ware JE Jr, Sherbourne CD. The MOS 36-item short-form health survey (SF-36). I. Conceptual framework and item selection. Med Care 1992; 30: 473-83.

  • 17. Ravens-Sieberer U, Wille N, Badia X, et al. Feasibility, reliability, and validity of the EQ-5D-Y: results from a multinational study. Qual Life Res 2010; 19(6): 875-86.

  • 18. Von Mackensen S, Bullinger M for The Haemo-Qol Group. Development and testing of an instrument to assess the quality of life of children with haemophilia in Europe. Haemophilia 2004, 10 (Suppl. 1): 17-25.

  • 19. Stein RE, Riessman CK The development of an impact-on-family scale: preliminary findings. Med Care. 1980; 18(4): 465-72.

  • 20. Ravens-Sieberer U, Morfeld M, Stein RE, et al. [The testing and validation of the German version of the impact on family scale in families with children with disabilities] [Article in German]. Psychother Psychosom Med Psychol 2001; 51(9-10): 384-93.

  • 21. Smith S, Cheater F, Bekker H. Parents’ experiences of living with a child with a long-term condition: a rapid structured review of the literature. Health Expect 2015; 18(4): 452-74.

  • 22. Murphy NA, Christian B, Caplin DA, Young P. The health of caregivers for children with disabilities: caregiver perspectives. Child Care Health Dev. 2007; 33(2): 180-7.

  • 23. Mullins LL, Wolfe-Christiansen C, Chaney JM, et al. The relationship between single-parent status and parenting capacities in mothers of youth with chronic health conditions: the mediating role of income. J Pediatr Psychol 2011; 36(3): 249-57.

  • 24. Shahly V, Chatterji S, Gruber MJ, et al. Cross-national differences in the prevalence and correlates of burden among older family caregivers in the World Health Organization World Mental Health (WMH) Surveys. Psychol Med 2013; 43(4): 865-79.

  • 25. DeKoven M, Karkare S, Kelley LA, et al. Understanding the experience of caring for children with haemophilia: crosssectional study of caregivers in the United States. Haemophilia 2014; 20(4): 541-9.

  • 26. Von Mackensen S, Wisniewski T, Urgo JC, Boggio L. Pilot test of the first hemophilia-specific burden scale for caregivers of children with hemophilia in the United States-the HEMOphilia associated CAregiver Burden scale (HEMOCAB™). Journal of Thrombosis and Haemostasis 2015; 13 (Suppl. 2): PO256.


Journal + Issues