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Table 3 Values of the most influential COI model inputs

From: A cost of illness study evaluating the burden of Wolfram syndrome in the United Kingdom

Input Value Reference
Population inputs
 Number of specialist paediatric reviews per patient per year 1 NHS Wolfram Syndrome Service (Birmingham Children’s Hospital), 2016 [16]
DM: management
 Paediatric outpatient diabetes care, average annual cost £2925.00 NHS National Tariff 2016-2017 [20]
 Proportion of patients managed by continuous subcutaneous insulin infusions 0.19 Diabetes UK, 2012 [7]
 Insulin treatment, continuous subcutaneous insulin infusions, average annual cost of consumables £2863.85 Cummins et al. 2010 [15]
 Insulin treatment, multiple daily injections, average annual cost of consumables £984.10 Cummins et al. 2010 [15]
DM: complications
 Incidence of end-stage renal disease 0.31 Pratoomsoot et la. 2009 [10]
 Haemodialysis, average annual cost £35,352.31 Roze et al. 2005 [26]
 Kidney transplant, average cost per first year £27,795.98 Roze et al. 2005 [26]
 Peritoneal dialysis, average annual cost £26,543.77 Roze et al. 2005 [26]
 Incidence of myocardial infarction 0.32 Pratoomsoot et la. 2009 [10]
 Myocardial infarction, average cost in year of event £6234.64 Roze et al. 2005 [26]
Hearing impairment
 Proportion of patients with sensorineural hearing loss 0.66 Barrett et al. 1995 [2]
 Proportion of patients with sensorineural hearing loss requiring cochlear implants 0.17 Karzon et al. 2013 [11]
 Cochlear implants, bilateral, average cost (management) £31,481.77 NHS Reference Costs 2015–2016 [19]
Visual impairment
 Number of ophthalmology reviews at local centre after symptom onset, per year 2.00 Patient experience (Wolfram Focus Group), 2016 [30]
 Number of occupational therapist visits for low vision services 6.00 Warren 1995 [28]
 Cost of occupational therapist visit £131.02 NHS Reference Costs 2015–2016 [19]
 Retinal photography, average cost £174.16 NHS Reference Costs 2015–2016 [19]
Renal/neurogenic bladder
 Proportion of patients with neurogenic bladder 0.55 Barrett et al. 1995 [2]
 Intermittent self-catheterisation, mean annual cost £2771.58 NCGC Infection prevention and control, 2012 [12]
  1. DM diabetes mellitus, NCGC National Clinical Guideline Centre, NHS National Health Service