The costs incurred by the families of children with cancer remain under researched. The objectives were to systematically review the literature and identify research and clinical implications. Thirteen studies were critically appraised using the Pediatric Quality Appraisal Questionnaire (PQAQ) [Ungar and Santos. Value Health 2003; 6:584-594]. Existing research indicates that families incur significant variable costs throughout cancer treatment. However, problems with the published studies related to various PQAQ domains (e.g., time horizon, sensitivity analysis) rendered the magnitude of families' childhood cancer costs somewhat uncertain. Strategies for enhancing the quality of childhood cancer cost of illness research are presented. Implications for clinical practice are discussed.
Families of children with cancer are confronted with a wide range of direct and time costs, the largest being travel and time allocated previously for unpaid activities.
Clinical interventions and research have mostly focused on the orthopedic, genetic, and pharmacological outcomes of individuals with osteogenesis imperfecta (OI), and although quality of life (QoL) has gained recognition as an important patient-outcome, it has received little attention in individuals with OI. This mixed-methods systematic review of the literature included five search engines and identified a total of 212 articles. Once study eligibility was reviewed, 10 studies met the inclusion criteria and were included in this mixed-methods review (9 quantitative and 1 qualitative). Among the 10 included QoL studies, six reported on children with OI, three on adults with OI, and one on the parents of children with OI. Physical QoL in children and adults with OI appears to be less than that of the general population, with individuals with more severe OI types reporting worse QoL. On the other hand, mental and psychosocial QoL is the same or better in individuals with OI than that of the general population. Pain, scoliosis activity limitations and participation restrictions due to decreased function are associated with lower levels of physical QoL. Researchers must agree on a definition of QoL as it relates to OI and use validated measures appropriate for evaluating QoL in OI. Pediatric studies should consider both the child and the parent's QOL perceptions as these may differ. QoL in the adult population should not be dismissed in order to offer proper client-centered interventions throughout the lifespan.
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