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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