title: Care models in the management of haemophilia: a systematic review.
abstract: BACKGROUND
  Haemophilia care is commonly provided via multidisciplinary specialized management. To date, there has been no systematic assessment of the impact of haemophilia care delivery models on patient-important outcomes.
  
  
  OBJECTIVE
  To conduct a systematic review of published studies assessing the effects of the integrated care model for persons with haemophilia (PWH).
  
  
  METHODS
  We searched MEDLINE, EMBASE and CINAHL up to April 22, 2015, contacted experts in the field, and reviewed reference lists.
  
  
  METHODS
  Randomized and non-randomized studies of PWH or carriers, focusing mainly on the assessment of care models on delivery.
  
  
  METHODS
  Two investigators independently screened title, abstract, and full text of retrieved articles for inclusion. Risk of bias and overall quality of evidence was assessed using Cochrane's ACROBAT-NRSI tool and GRADE respectively. Relative risks, mean differences, proportions, and means and their variability were calculated as appropriate.
  
  
  RESULTS
  27 non-randomized studies were included: eight comparative and 19 non-comparative studies. We found low- to very low-quality evidence that in comparison to other models of care, integrated care may reduce mortality, hospitalizations and emergency room visits, may lead to fewer missed days of school and work, and may increase knowledge seeking.
  
  
  CONCLUSIONS
  Our comprehensive review found low- to very low-quality evidence from a limited number of non-randomized studies assessing the impact of haemophilia care models on some patient-important outcomes. While the available evidence suggests that adoption of the integrated care model may provide benefit to PWH, further high-quality research in the field is needed.
authors.affiliation: Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, ON, Canada.
authors.forename: C H T
authors.initials: CH
authors.lastname: Yeung
authors.affiliation: Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, ON, Canada.
authors.forename: N
authors.initials: N
authors.lastname: Santesso
authors.affiliation: Department of Medicine, McMaster University, Hamilton, ON, Canada.|Department of Pathology and Molecular Medicine, McMaster University, Hamilton, ON, Canada.
authors.forename: M
authors.initials: M
authors.lastname: Pai
authors.affiliation: Georgetown University, Washington, DC, USA.
authors.forename: C
authors.initials: C
authors.lastname: Kessler
authors.affiliation: Department of Medicine, University of North Carolina, Chapel Hill, NC, USA.
authors.forename: N S
authors.initials: NS
authors.lastname: Key
authors.affiliation: Department of Infection, Immunity and Cardiovascular Disease, University of Sheffield, Sheffield, UK.
authors.forename: M
authors.initials: M
authors.lastname: Makris
authors.affiliation: Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, ON, Canada.
authors.forename: T
authors.initials: T
authors.lastname: Navarro-Ruan
authors.affiliation: Centers for Disease Control and Prevention, Division of Blood Disorders, National Center for Birth Defects and Developmental Disabilities, Atlanta, GA, USA.
authors.forename: J M
authors.initials: JM
authors.lastname: Soucie
authors.affiliation: Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, ON, Canada.|Department of Medicine, McMaster University, Hamilton, ON, Canada.
authors.forename: H J
authors.initials: HJ
authors.lastname: Schünemann
authors.affiliation: Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, ON, Canada.|Department of Medicine, McMaster University, Hamilton, ON, Canada.
authors.forename: A
authors.initials: A
authors.lastname: Iorio
mesh_terms: D002986:Clinical Trials as Topic; D016208:Databases, Factual; D003695:Delivery of Health Care / Q000379:methods / Q000592:standards; D019468:Disease Management; D006467:Hemophilia A / Q000401:mortality / Q000473:pathology / Q000628:therapy*; D006801:Humans; D007902:Length of Stay; D016294:Models, Nursing
keywords: care model; delivery of health care; haemophilia; health care team; integrated care; review