Haemophilia Utilization Group Study - Part Va (HUGS Va): Design, methods and baseline data

Haemophilia

Volumn 17, Issue 5, 2011, Pages 729-736

  Zhou, Z Y ^a   Wu, J ^a   Baker, J ^b   Curtis, R ^c   Forsberg, A ^d   Huszti, H ^e   Koerper, M ^f   Lou, M ^a   Miller, R ^g   Parish, K ^h   Riske, B ⁱ   Shapiro, A ^j   Ullman, M ^k   Johnson, K ^a  

^a UNIVERSITY OF SOUTHERN CALIFORNIA   (United States)

^b UNIVERSITY OF CALIFORNIA   (United States)

^c Factor VIII Computing   (United States)

^d UMASS MEMORIAL MEDICAL CENTER   (United States)

^e CHILDREN S HOSPITAL OF ORANGE COUNTY   (United States)

^f UNIVERSITY OF CALIFORNIA   (United States)

^g CHILDREN S HOSPITAL LOS ANGELES   (United States)

^h Indiana Hemophilia and Thrombosis Center ^*

ⁱ UNIVERSITY OF COLORADO   (United States)

^j INDIANA HEMOPHILIA AND THROMBOSIS CENTER   (United States)

^k UNIVERSITY OF TEXAS   (United States)

more..

Author keywords

Cost of illness; Haemophilia; Observational study; Outcome; Quality of life; Utilization

Indexed keywords

BLOOD CLOTTING FACTOR 8; BLOOD CLOTTING FACTOR 8 CONCENTRATE; BLOOD CLOTTING FACTOR 8 INHIBITOR;

ADOLESCENT; ADULT; AGE DISTRIBUTION; ARTHRALGIA; ARTICLE; CHILD; COMORBIDITY; DEMOGRAPHY; DISABILITY; DISEASE SEVERITY; EDUCATIONAL STATUS; EMPLOYMENT; FEMALE; FOLLOW UP; HEALTH CARE ACCESS; HEALTH CARE COST; HEALTH CARE UTILIZATION; HEALTH INSURANCE; HEALTH SURVEY; HEMOPHILIA A; HUMAN; INCOME; JOINT LIMITATION; MAJOR CLINICAL STUDY; MALE; MARRIAGE; MULTICENTER STUDY; OBSERVATIONAL STUDY; PRESCHOOL CHILD; PRIORITY JOURNAL; QUALITY OF LIFE; SCHOOL CHILD; SELF REPORT; STUDY DESIGN; TREATMENT OUTCOME;

ADOLESCENT; ADULT; CHILD; CHILD, PRESCHOOL; COHORT STUDIES; COST OF ILLNESS; FEMALE; HEALTH RESOURCES; HEALTH SERVICES ACCESSIBILITY; HEMOPHILIA A; HUMANS; MALE; MIDDLE AGED; OUTCOME ASSESSMENT (HEALTH CARE); QUALITY OF LIFE; UNITED STATES; YOUNG ADULT;

EID: 80052037388     PISSN: 13518216     EISSN: 13652516     Source Type: Journal    
DOI: 10.1111/j.1365-2516.2011.02595.x     Document Type: Article

References (24)

1. Royal S, Schramm W, Berntorp E et al. Quality-of-life differences between prophylactic and on-demand factor replacement therapy in European haemophilia patients. Haemophilia 2002; 8: 44-50.
2. World Federation of Hemophilia. Available at Accessed July 10, 2010.
3. The Centers for Disease Control and Prevention: Hemophilia Data and Statistics. Available at Accessed July 10, 2010.
4. Teitel JM, Barnard D, Israels S, Lillicrap D, Poon MC, Sek J. Home management of haemophilia. Haemophilia 2004; 10: 118-33.
5. Stobart K, Iorio A, Wu JK. Clotting factor concentrates given to prevent bleeding and bleeding-related complications in people with hemophilia A or B. Cochrane Database Syst Rev 2006; CD003429.
6. Szucs TD, Offner A, Kroner B et al. Resource utilisation in haemophiliacs treated in Europe: Results from the European Study on Socioeconomic Aspects of Haemophilia Care. Haemophilia 1998; 4: 498-501.
7. Aledort LM, Haschmeyer RH, Pettersson H. A longitudinal study of orthopaedic outcomes for severe factor-VIII-deficient haemophiliacs. The Orthopaedic Outcome Study Group. J Intern Med 1994; 236: 391-9.
8. Fischer K, van der Bom JG, Molho P et al. Prophylactic versus on-demand treatment strategies for severe haemophilia: a comparison of costs and long-term outcome. Haemophilia 2002; 8: 745-52.
9. Manco-Johnson MJ, Abshire TC, Shapiro AD et al. Prophylaxis versus episodic treatment to prevent joint disease in boys with severe hemophilia. N Engl J Med 2007; 357: 535-44.
10. Berntorp E, Boulyjenkov V, Brettler D et al. Modern treatment of haemophilia. Bull World Health Organ 1995; 73: 691-701.
11. Bohn RL, Avorn J, Glynn RJ, Choodnovskiy I, Haschemeyer R, Aledort LM. Prophylactic use of factor VIII: an economic evaluation. Thromb Haemost 1998; 79: 932-7.
12. Miners AH, Sabin CA, Tolley KH, Lee CA. Assessing the effectiveness and cost-effectiveness of prophylaxis against bleeding in patients with severe haemophilia and severe von Willebrand's disease. J Intern Med 1998; 244: 515-22.
13. Gringeri A, Mantovani LG, Scalone L, Mannucci PM. Cost of care and quality of life for patients with hemophilia complicated by inhibitors: the COCIS Study Group. Blood 2003; 102: 2358-63.
14. Auerswald G, von Depka Prondzinski M, Ehlken B et al. Treatment patterns and cost-of-illness of severe haemophilia in patients with inhibitors in Germany. Haemophilia 2004; 10: 499-508.
15. Globe DR, Curtis RG, Koerper MA. Utilization of care in haemophilia: a resource-based method for cost analysis from the Haemophilia Utilization Group Study (HUGS). Haemophilia 2004; 10(Suppl. 1): 63-70.
16. Globe DR, Cunningham WE, Andersen R et al. The Hemophilia Utilization Group Study (HUGS): determinants of costs of care in persons with haemophilia A. Haemophilia 2003; 9: 325-31.
17. Ware J Jr, Kosinski M, Keller SD. A 12-Item Short-Form Health Survey: construction of scales and preliminary tests of reliability and validity. Med Care 1996; 34: 220-33.
18. Varni JW, Seid M, Kurtin PS. PedsQL 4.0: reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Med Care 2001; 39: 800-12.
19. Varni JW, Seid M, Smith Knight T, Burwinkle T, Brown J, Szer IS. The PedsQL in pediatric rheumatology: reliability, validity, and responsiveness of the Pediatric Quality of Life Inventory Generic Core Scales and Rheumatology Module. Arthritis Rheum 2002; 46: 714-25.
20. Varni JW, Burwinkle TM, Seid M, Skarr D. The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr 2003; 3: 329-41.
21. Soucie JM, Cianfrini C, Janco RL et al. Joint range-of-motion limitations among young males with hemophilia: prevalence and risk factors. Blood 2004; 103: 2467-73.
22. Varni JW, Limbers CA, Burwinkle TM. Parent proxy-report of their children's health-related quality of life: an analysis of 13, 878 parents' reliability and validity across age subgroups using the PedsQL 4.0 Generic Core Scales. Health Qual Life Outcomes 2007; 5: 2.
23. Salomon JA, Tandon A, Murray CJ. Comparability of self rated health: cross sectional multi-country survey using anchoring vignettes. BMJ 2004; 328: 258.
24. Baker JR, Crudder SO, Riske B, Bias V, Forsberg A. A model for a regional system of care to promote the health and well-being of people with rare chronic genetic disorders. Am J Public Health 2005; 95: 1910-6.