Hospital incidence, management and direct cost of osteogenesis imperfecta in Spain: a retrospective database analysis

Abstract

Objective: The objective of this study was to analyse hospital incidence of osteogenesis imperfecta (OI) in Spanish hospitals and the associated medical costs from a healthcare system perspective. Methods: To this aim, a retrospective multicentre study was designed analysing admission records from patients admitted with OI in specialized care settings in Spain between 2000 and 2017. Direct medical costs were calculated based on the diagnosis-related group-based hospital payment systems, determined by the Spanish Ministry of Health. Results: Overall, 3,747 admissions were reviewed, corresponding to 998 patients, 48.20% of which were males and 51.80% females. Hospital incidence was 5.64 per 100,000 patients (95% CI, 4.80–6.60) over the study period, whereas incidence at birth was 10.14 per 100,000 (95% CI, 8.16–12.05). In-hospital mortality appeared primarily associated to neonatal conditions and acute respiratory failure. Mean length of hospital stay was 2.83 days, decreasing significantly during the study period (p < 0.0001). Readmission rate was significantly higher in younger patients (p = 0.0110). In most hospital admissions other disorders of bone and cartilage (osteoporosis and pathologic fractures) were registered, together with delayed growth and hypocalcaemia. The mean annual direct medical cost per hospital admission was €2,571, increasing significantly over the study period (p < 0.0001). Conclusions: Overall, this study provides data that should be taken into account for the development of improved and more efficient treatment protocols, and in reducing the burden of OI at the healthcare system level.