RESUMO
PURPOSE: This study describes the cost trajectory of the standard diagnostic care pathway for children with suspected genetic disorders in British Columbia, Canada. METHODS: Average annual per-patient costs were estimated using medical records review and a caregiver survey for a cohort of 498 children referred to BC Children's and Women's Hospitals (C&W) with unexplained intellectual disability (the TIDE-BC study) and families enrolled in the CAUSES study, which offered diagnostic genome-wide sequencing (GWS; exome and genome sequencing) to 500 families of children with suspected genetic disorders. RESULTS: Direct costs peaked in the first year of patients' diagnostic odyssey, with an average of C$2257 per patient (95% confidence interval [CI] C$2074, C$2441) for diagnostic testing and C$631 (95% CI C$543, C$727) for specialist consultations at C&W. In subsequent years, direct costs accrued at a constant rate, with an estimated annual per-patient cost of C$511 (95% CI C$473, C$551) for diagnostic testing and C$334 (95% CI C$295, C$369) for consultations at C&W. Travel costs and caregiver productivity loss associated with attending diagnosis-related physician appointments averaged C$1907/family/year. CONCLUSIONS: The continuing long-term accrual of costs by undiagnosed patients suggests that economic evaluations of diagnostic GWS services should use longer time horizons than have typically been used.
Assuntos
Doenças Genéticas Inatas/economia , Testes Genéticos/economia , Custos de Cuidados de Saúde/tendências , Adulto , Colúmbia Britânica/epidemiologia , Cuidadores/economia , Cuidadores/psicologia , Estudos de Coortes , Análise Custo-Benefício , Exoma/genética , Feminino , Custos de Cuidados de Saúde/ética , Humanos , Deficiência Intelectual/genética , Masculino , Análise de Sequência de DNA/economia , Sequenciamento do Exoma/economia , Sequenciamento do Exoma/métodosRESUMO
This study was conducted to investigate the trajectory of hospitalization costs, and to assess the determinants related to the membership of the identified trajectories, with the view of recommending future research directions. A retrospective study was performed in urban Yichang, China, where a total of 134 end-stage lung cancer patients were selected. The latent class analysis (LCA) model was used to investigate the heterogeneity in the trajectory of hospitalization cost amongst the different groups that were identified. A multi-nominal logit model was applied to explore the attributes of different classes. Three classes were defined as follows: Class 1 represented the trajectory with minimal cost, which had increased over the last two months. Classes 2 and 3 consisted of patients that incurred high costs, which had declined with the impending death of the patient. Patients in class 3 had a higher average cost than those in Class 2. The level of education, hospitalization, and place of death, were the attributes of membership to the different classes. LCA was useful in quantifying heterogeneity amongst the patients. The results showed the attributes were embedded in hospitalization cost trajectories. These findings are applicable to early identification and intervention in palliative care. Future studies should focus on the validation of the proposed model in clinical settings, as well as to identify the determinants of early discharge or aggressive care.