Abstract

Background -Research on the cost of care of patients with cystic fibrosis is scarce. The aim of this study was to estimate the costs using age-specific medical consumption from real patient data. Methods -The age-specific medical consumption ofpatients with cystic fibrosis in The Netherlands in 1991 was estimated from a survey of medical records and a patient questionnaire. A distinction was made between costs of hospital care, hospital and non-hospital medication, and home care. Costs per year were obtained by multiplying the yearly amount of care and the costs per unit. Results -On average the annual cost of a patient with cystic fibrosis in 1991 was £10 908 (hospital care 42%, medication 37%, home care 20%). The cost of care of cystic fibrosis in The Netherlands, with approximately 1000 patients, is estimated at £10-9 million per year, which is 0 07% of the total health care budget. The cost of care of a patient up to the age of 35 is estimated at £614 587. When year-to-year survival is taken into account and future costs are discounted to the year of birth with a yearly discount rate of 5%, the cost of care of a patient with cystic fibrosis is estimated at £164365 for 1991. This estimate will be used in a prospective evaluation of screening for cystic fibrosis carriers. Conclusions -The cost of care of patients with cystic fibrosis estimated by age-specific medical consumption of real patients is higher than that estimated by non-agespecific medical consumption and/or expert opinions.

T Th he e n no on nh ho os sp pi it ta al l c co os st ts s o of f c ca ar re e o of f p pa at ti ie en nt ts s w wi it th h C CF F i in n TThe average nonhospital costs of care amounted to £4,641 per child per year (range £712-13,269) and £10,242 per adult (range £1,653-26,571). Nonhospital medical care for children and adults accounted for, respectively, 8 and 5% of these costs, domestic help for 15 and 9%, diet for 10 and 7%, travelling because of CF for 4 and 8%, medication for 63 and 67%, and devices and special facilities at home, work or school for 1 and 4%.Nonhospital costs of care of cystic fibrosis are very high and amount to 50% of the total (medical and nonmedical) lifetime costs of cystic fibrosis.

Both single-entry two-step (SETS) couple screening and double-entry two-step (DETS) couple screening have been recommended as methods to screen for cystic fibrosis gene carriers. In this paper we compare the expected results from both types of screening. In general, DETS results in a higher detection rate of couples in which both partners are carriers, but also in a higher proportion of couples with only one identified carrier who have a significant remaining risk in their offspring.