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Health Policy (Amsterdam, Netherlands) Jul 2015Cost-of-illness studies, the systematic quantification of the economic burden of diseases on the individual and on society, help illustrate direct budgetary consequences... (Review)
Review
Cost-of-illness studies, the systematic quantification of the economic burden of diseases on the individual and on society, help illustrate direct budgetary consequences of diseases in the health system and indirect costs associated with patient or carer productivity losses. In the context of the BURQOL-RD project ("Social Economic Burden and Health-Related Quality of Life in patients with Rare Diseases in Europe") we studied the evidence on direct and indirect costs for 10 rare diseases (Cystic Fibrosis [CF], Duchenne Muscular Dystrophy [DMD], Fragile X Syndrome [FXS], Haemophilia, Juvenile Idiopathic Arthritis [JIA], Mucopolysaccharidosis [MPS], Scleroderma [SCL], Prader-Willi Syndrome [PWS], Histiocytosis [HIS] and Epidermolysis Bullosa [EB]). A systematic literature review of cost of illness studies was conducted using a keyword strategy in combination with the names of the 10 selected rare diseases. Available disease prevalence in Europe was found to range between 1 and 2 per 100,000 population (PWS, a sub-type of Histiocytosis, and EB) up to 42 per 100,000 population (Scleroderma). Overall, cost evidence on rare diseases appears to be very scarce (a total of 77 studies were identified across all diseases), with CF (n=29) and Haemophilia (n=22) being relatively well studied, compared to the other conditions, where very limited cost of illness information was available. In terms of data availability, total lifetime cost figures were found only across four diseases, and total annual costs (including indirect costs) across five diseases. Overall, data availability was found to correlate with the existence of a pharmaceutical treatment and indirect costs tended to account for a significant proportion of total costs. Although methodological variations prevent any detailed comparison between conditions and based on the evidence available, most of the rare diseases examined are associated with significant economic burden, both direct and indirect.
Topics: Cost of Illness; Europe; Health Care Costs; Humans; Orphan Drug Production; Prevalence; Quality of Life; Rare Diseases
PubMed: 25661982
DOI: 10.1016/j.healthpol.2014.12.016 -
Research in Developmental Disabilities Aug 2014Over the past three decades, the potential effects of lifestyle interventions targeting changes in body weight and composition (weight, body mass index, fat mass, waist... (Review)
Review
Over the past three decades, the potential effects of lifestyle interventions targeting changes in body weight and composition (weight, body mass index, fat mass, waist circumference) among adults with an intellectual disability (ID) have been examined in various systematic reviews. Nevertheless, since the middle of the 1980s, the potential effects of these interventions for youth with an ID remain an open question. The purpose of this article is to review the effects of lifestyle interventions targeting changes in body weight and composition among youth with an ID. This review will focus on changes in body weight and composition, healthy lifestyle, and secondary health conditions. A systematic review of English- and French-language studies, published between 1981 and 2013, was performed on Academic Search Complete, PsycARTICLES, Medline and Scopus. The nine studies included in this review focused mainly on: a sample with a wide age range (e.g., 7-22 years); males; overweight-obese youth having a mild-to-moderate ID with Down or Prader-Willi syndrome; physical activity interventions; cohort pre- and post-test designs with/without a control group; and changes in body weight and composition. Taken together, results from these studies suggest successful changes in weight, body mass index and fat mass. However, intervention effects on healthy lifestyle and secondary health conditions are scarce and inconclusive. Given the weaknesses of the reviewed studies, the present findings should be considered preliminary and indicative of the need for future research.
Topics: Adiposity; Body Composition; Body Weight; Health Promotion; Humans; Intellectual Disability; Life Style
PubMed: 24830882
DOI: 10.1016/j.ridd.2014.04.014