title: Patient/caregiver perceived benefits and barriers to elective orthopedic surgery (EOS) in patients with congenital hemophilia with inhibitors.
abstract: OBJECTIVE
  Congenital hemophilia patients with inhibitors are at greater risk for developing arthropathy and orthopedic complications compared to those without inhibitors. Elective orthopedic surgeries (EOS) may be an option for these patients and may provide long-term cost savings due to reduced bleed frequency. However, patient motivations and goals for undergoing or delaying such surgeries are not well understood. A US-based patient/caregiver survey was designed to describe inhibitor patient experiences and outcomes following EOS and to develop a comprehensive understanding of patient preferences for EOS, which are lacking in the literature.
  
  
  METHODS
  The paper-pencil questionnaire was mailed to 261 US inhibitor patients/caregivers and included history and timing of EOS, quality-of-life (QoL) and potential benefits of and barriers to receiving EOS. Univariate/bivariate descriptive analyses were performed to characterize those with/without a history of EOS.
  
  
  RESULTS
  For 103 subjects who responded, the mean age was 20.9 years. Approximately 25% (n = 26) of respondents underwent EOS, most commonly on the knee (21, 81%); 73.1% of surgery recipients reported the surgery improved or greatly improved their QoL based on single-item response. The highest ranked perceived benefits were less pain, fewer bleeds, and improved mobility. However, the leading concerns reported were lack of improved mobility (62.2%), fear of uncontrolled bleeding (61.3%), and surgical complications, such as blood clot (60.0%).
  
  
  CONCLUSIONS
  The study consisted of a small sample size, primarily due to the difficulty in trying to reach inhibitor patients or their caregivers, thereby restricting inferential and stratification analysis.
  
  
  CONCLUSIONS
  QoL improved for most inhibitor patients who reported having EOS. For those considering surgery, there is optimism about the potential benefits, but realistic concerns associated with bleed control and post-op complications.
authors.affiliation: Health Economics & Outcomes Research, IMS Consulting Group , Alexandria, VA 22314 , USA.
authors.forename: M
authors.initials: M
authors.lastname: Dekoven
authors.forename: T
authors.initials: T
authors.lastname: Wisniewski
authors.forename: A
authors.initials: A
authors.lastname: Petrilla
authors.forename: N
authors.initials: N
authors.lastname: Holot
authors.forename: W C
authors.initials: WC
authors.lastname: Lee
authors.forename: D
authors.initials: D
authors.lastname: Cooper
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D017028:Caregivers; D002648:Child; D002675:Child, Preschool; D003657:Decision Making; D017558:Elective Surgical Procedures; D007722:Health Knowledge, Attitudes, Practice; D006467:Hemophilia A / Q000150:complications*; D006801:Humans; D007223:Infant; D008297:Male; D008875:Middle Aged; D019637:Orthopedic Procedures; D057240:Patient Preference; D010361:Patients; D011788:Quality of Life; D011795:Surveys and Questionnaires; D055815:Young Adult