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Table 2 Cost and resource use inputs of the model

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

Input Unit Cost Resource Use Reference
Diagnosis
 Outpatient appointment at local tertiary hospital £330.14 [19] 1 appointment for each new patient NHS Reference Costs 2015–2016 [19] NHS UK Genetic Testing Network, 2016 [27]
 Genetic testing, WFS1 sequencing £550.00 [27]   NHS Reference Costs 2015–2016 [19] NHS UK Genetic Testing Network, 2016 [27]
Referral to specialised service (paediatric)
 Blood tests (serum osmolality, glucose, HbA1c) £1.18 [19]   NHS Reference Costs 2015–2016 [19]
 Urine osmolality test £1.18 [19]   NHS Reference Costs 2015–2016 [19]
 Genetic counselling £481.89 [19]   NHS Reference Costs 2015–2016 [19]
 MRI scan, brain £142.99 [19]   NHS Reference Costs 2015–2016 [19]
 Orthoptics assessment £66.05 [19]   NHS Reference Costs 2015–2016 [19]
 Retinal photography £174.16 [19]   NHS Reference Costs 2015–2016 [19]
 Ophthalmologist review £118.59 [19]   NHS Reference Costs 2015–2016 [19]
 Psychology assessment £249.26 [19] 1 appointment for each new patient NHS Reference Costs 2015–2016 [19]
 Audiological examination £151.69 [19]   NHS Reference Costs 2015–2016 [19]
 Auditory brain response (ABR) testing £29.19 [19]   NHS Reference Costs 2015–2016 [19]
 ENT review £122.78 [19]   NHS Reference Costs 2015–2016 [19]
 Consultant review £330.14 [19]   NHS Reference Costs 2015–2016 [19]
 Diabetic specialist review £391.00 [19]   NHS Reference Costs 2015–2016 [19]
 Urological examination £102.41 [19]   NHS Reference Costs 2015–2016 [19]
 Urology review £124.55 [19]   NHS Reference Costs 2015–2016 [19]
Specialised service (paediatric), annual review
 Blood tests (serum osmolality, glucose, HbA1c) £1.18 [19] 1 specialist paediatric review per patient per year [16] NHS Reference Costs 2015–2016 [19] NHS Wolfram Syndrome Service (Birmingham Children’s Hospital), 2016 [16]
 Urine osmolality test £1.18 [19] As above
 Genetic counselling £404.29 [19] As above
 MRI scan, brain £142.99 [19] As above
 Orthoptics assessment £65.21 [19] As above
 Retinal photography £174.16 [19] As above
 Ophthalmologist review £115.31 [19] As above
 Psychology assessment £195.59 [19] As above
 Audiological examination £151.69 [19] As above
 Auditory brain response (ABR) testing £29.19 [19] As above
 ENT review £102.65 [19] As above
 Consultant review £228.68 [19] As above
 Diabetic specialist review £252.57 [19] As above
 Urological examination £102.41 [19] As above
 Urology review £114.81 [19] As above
Specialised service (paediatric), additional costs
 Wellchild family liaison officer, per hour £59.00 [25] 33 h per clinic PSSRU, 2016 [25] NHS Wolfram Syndrome Service (Birmingham Children’s Hospital), 2016 [16] Expert opinion (Professor Timothy Barrett), 2016 NHS Reference Costs 2015–2016 [19]
 Nurse specialist, per hour £44.00 [25] 15 h per clinic [16] As above
 Research nurse, per hour £44.00 [25] As above
 Clinic co-ordinator, per hour £29.00 [25] 477 h per clinic As above
 MDT meeting, per patient £107.35 [19] 2 MDT meetings per clinic 6 patients discussed at MDT meetings [16] As above
 Consultant, per hour £104.00 [25] 6 h per clinic As above
 Number of paediatric specialist clinics, per year 4.00 [16] As above
Transition services
 Cost of transition services to adulthood, per year £979.00 [25] 1 per new patient seen at adult specialist centre PSSRU, 2016 [25]
Specialised service (adult), new patients
 Blood tests (serum osmolality, glucose, HbA1c) £1.18 [19] 1 per new patient seen at adult specialist centre NHS Reference Costs 2015–2016 [19]
 Urine osmolality test £1.18 [19] NHS Reference Costs 2015–2016 [19]
 Genetic counselling £481.89 [19] NHS Reference Costs 2015–2016 [19]
 Psychology assessment £249.26 [19] NHS Reference Costs 2015–2016 [19]
 Consultant review £195.87 [19] NHS Reference Costs 2015–2016 [19]
 Neurological review £216.58 [19] NHS Reference Costs 2015–2016 [19]
Specialised service (adult), follow-up review
 Blood tests (serum osmolality, glucose, HbA1c) £1.18 [19] 0.75 follow-up appointments per patient per year [17] NHS Reference Costs 2015–2016 [19] NHS Wolfram Syndrome Service (Queen Elizabeth Hospital), 2016 [17]
 Urine osmolality test £1.18 [19] As above
 Genetic counselling £404.29 [19] As above
 Psychology assessment £195.59 [19] As above
 Consultant review £150.38 [19] As above
 Neurological review £160.76 [19] As above
DM care
 Paediatric outpatient diabetes care, per year £2925.00 [20] N/A (costs given are per patient with DM per year) NHS National Tariff 2016–2017 [20] Cummins et al. 2010 [15] Patient experience (Wolfram Focus Group), 2016 [30] NHS Reference Costs 2015–2016 [19] Expert opinion (Professor Timothy Barrett), 2016 NHS Vale of York CCG, 2015 [21] NHS Southern Derbyshire CCG, 2016, UK [24]
 Insulin treatment, multiple daily injections, average annual cost of consumables £984.10 [15] As above
 Insulin treatment, continuous subcutaneous insulin infusions, average annual cost of consumables £2863.85 [15] As above
 Endocrinologist outpatient appointment for diabetes management, adult services £150.38 [19] 2 appointments per year [30] As above
 Diabetic specialist nurse appointment, adult services £70.59 [19] 2 appointments per year As above
 Diabetic specialist nurse telephone appointment, adult services £29.90 [19] 2 appointments per year As above
 Diabetic education service appointment £44.33 [19] 1 appointment per year As above
 Coeliac serology, IgA tTG test £23.75 [21] 1 per patient with symptoms suggestive of coeliac disease per year As above
 Coeliac serology, EMA test £11.62 [21] As above
 Duodenal biopsy £868.79 [19] 1 per patient with coeliac disease per year As above
 Dietician, community health service £81.32 [19] 3 visits for coeliac disease per year [24] As above
 Ketoacidosis, inpatient care £600.03 [19] 1 per patient with ketoacidosis per year As above
 Severe hypoglycaemic events, inpatient care £413.39 [19] 1 per patient with hypoglycaemic event per year As above
DM complications
 Myocardial infarction, year of event £6234.64 [26] N/A Roze et al. 2005 [26]
 Myocardial infarction, each subsequent year £1027.06 [26] Roze et al. 2005 [26]
 Angina, year of onset £3234.06 [26] Roze et al. 2005 [26]
 Angina, each subsequent year £1067.37 [26] Roze et al. 2005 [26]
 Congestive heart failure, year of onset £3606.53 [26] Roze et al. 2005 [26]
 Congestive heart failure, each subsequent year £1229.97 [26] Roze et al. 2005 [26]
 Stroke, fatal £4810.09 [26] Roze et al. 2005 [26]
 Stroke, year of event £3813.61 [26] Roze et al. 2005 [26]
 Stroke, each subsequent year £7225.56 [26] Roze et al. 2005 [26]
 Peripheral vascular disease, onset £3321.62 [26] Roze et al. 2005 [26]
 Haemodialysis £35,352.31 [26] Roze et al. 2005 [26]
 Peritoneal dialysis £26,543.77 [26] Roze et al. 2005 [26]
 Kidney transplant, first year £27,795.98 [26] Roze et al. 2005 [26]
 Kidney transplant, subsequent year £9151.83 [26] Roze et al. 2005 [26]
 Retinal photocoagulation £958.96 [26] Roze et al. 2005 [26]
 Cataract extraction £2208.39 [26] Roze et al. 2005 [26]
 Cataract annual follow-up £148.71 [26] Roze et al. 2005 [26]
 Neuropathy, onset £1352.27 [26] Roze et al. 2005 [26]
 Uninfected ulcer £1778.94 [26] Roze et al. 2005 [26]
 Infected ulcer £1823.42 [26] Roze et al. 2005 [26]
 Major hypoglycaemic event £307.15 [26] Roze et al. 2005 [26]
DI and other endocrine disorders care
 Blood tests (serum osmolality, glucose, HbA1c) £1.18 [19] 1 appointment for each new patient NHS Reference Costs 2015–2016 [19] BNF, March 2016 [14] BNF, May 2016 [14] PSSRU, 2016 [25]
 Desmopressin acetate, nasal spray, 10 micrograms/metered spray, 60 metered sprays £8.76 [14] 2 metered sprays per day [14] As above
 Testosterone enanthate, 250 mg/1 mL solution for injection ampoules, 3 ampoules £72.50 [14] 1 ampoule per month per male paediatric patient with hypogonadism As above
 Testosterone undecanoate, 250 mg/1 mL solution for injection, 1000 mg/4 mL, 1 vial £80.00 [14] 1 g per 12 weeks per male adult patient with hypogonadism As above
 GP nurse attendance for testosterone administration, per visit £9.30 [25] 1 per injection As above
 Estradiol valerate 1 mg, 28 tablets £3.53 [14] 1 per 3 months per female patient with hypogonadism As above
 Desogestrel 75 microgram, 84 tablets £5.21 [14] 1 per 3 months per female patient with hypogonadism As above
Visual impairment care
 Optometry assessment £95.84 [19] 2 per year per patient both before and after symptom onset [30] NHS Reference Costs 2015–2016 [19] Patient experience (Wolfram Focus Group), 2016 [30] Federation of (Ophthalmic and Dispensing) Opticians, 2016 [18] Warren 1995 [28]
 Retinal photography £174.16 [19] As above
 Ophthalmologist review at local centre £115.31 [19] As above
 Glasses, single vision lenses, ≤6 dioptres SPH £39.10 [18] 1 averaged cost per patient per year As above
 Glasses, single vision lenses, > 6–≤10 dioptres SPH £59.30 [18] As above
 Glasses, single vision lenses, > 10–≤14 dioptres SPH £86.90 [18] As above
 Glasses, single vision lenses, > 14 dioptres SPH £196.00 [18] As above
 Cataract surgery £875.11 [19] 1 per patient requiring cataract surgery As above
 Low vision services assessment £78.31 [19] 1 per patient requiring visual impairment care As above
 Occupational therapist visit £131.02 [19] 6 visits per year per patient requiring visual impairment care [28] As above
Hearing impairment care
 Audiological examination £151.69 [19] 1 per year per patient both before and after symptom onset [30] NHS Reference Costs 2015–2016 [19] Patient experience (Wolfram Focus Group), 2016 [30]
 Auditory brain response (ABR) testing £29.19 [19] As above
 ENT annual review £102.65 [19] As above
 Audiology hearing aid fitting £89.58 [19] 1 per patient requiring hearing aid As above
 Audiology hearing aid follow-up £96.90 [19] As above
 Audiology annual review £358.31 [19] 1 per patient with SNHL per year As above
 Cochlear implants, bilateral £31,481.77 [19] 1 per patient requiring cochlear implant As above
 ENT follow-up post-implant for external cochlear implant fitting £89.14 [19] As above
 Audiology follow-up post-implant, maintenance and programming £291.73 [19] As above
 ENT annual review for severe hearing loss £89.14 [19] As above
Renal/neurogenic bladder care
 Urodynamic studies, paediatrics £102.41 [19] 1 review per patient per year prior to symptom onset [30] 2 reviews per patient per year after symptom onset [29] NHS Reference Costs 2015–2016 [19] Patient experience (Wolfram Focus Group), 2016 [30] Wolfram Syndrome Guideline Development Group, 2014, Europe [29] NCGC Infection prevention and control, 2012 [12] NCGC Urinary incontinence in neurological disease, 2012 [12] NHS Drug Tariff, May 2016 [22]
 Urodynamic studies, adult services £134.75 [19] As above
 Urology review, paediatrics £114.81 [19] As above
 Urology review, adult services £99.79 [19] As above
 Urine dipstick £1.18 [19] As above
 Intermittent self-catheterisation, mean annual cost £2771.58 [12] Annual cost for patients managed by clean intermittent self-catheterisation [12] As above
 Indwelling catheter £5.22 [12] 7 indwelling catheter changes per patient per year with indwelling catheter [22] As above
 Catheter leg bag £2.39 [22] 60 drainage bag changes per patient per year with neurogenic bladder [22] As above
 Catheter drainage bag £1.10 [22]   As above
 Annual cost of symptomatic UTI £40.06 [12] Annual cost for patients with different forms of UTI and urethral complication As above
 Annual cost of first-line antibiotic resistant UTI £60.42 [12] As above
 Annual cost of multidrug resistant UTI £2232.50 [12] As above
 Annual cost of bacteraemia secondary to UTI £3535.95 [12] As above
 Annual cost of urethral complication £1402.07 [12] As above
 Mental health care initial assessment £257.59 [19] 1 appointment per patient after symptom onset [30] As above
 Community mental health care, adult £120.61 [19] 1 appointment per patient per year after symptom onset [30] As above
 Inpatient treatment of psychiatric disorders, per bed day £390.20 [19] 15 days review period per patient [30] As above
 Rehabilitation of psychiatric disorders £377.77 [19] 1 appointment per patient per year after symptom onset [30] As above
Neurological care
 Neurological review, paediatric £343.79 [19] 1 review per patients per year prior to symptom onset [29] 2 reviews per patient per year after symptom onset [4] NHS Reference Costs 2015–2016 [19] Wolfram Syndrome Guideline Development Group, 2014, Europe [29] Urano et al. 2016 [4] Expert opinion (Professor Timothy Barrett), 2016 NHS Bolton Foundation Trust, 2015 [23]
 Neurological review, adult services £160.76 [19] As above
 Specialist nurse, paediatric, community health services £137.36 [19] 2 appointments per patient per year after symptom onset As above
 Specialist nurse, telephone appointment, paediatric, community health services £43.23 [19] 2 appointments per patient per year after symptom onset As above
 Specialist nurse, adult services, community health services £77.24 [19] 1 appointment per patient per year after symptom onset As above
 Specialist nurse, adult services, telephone appointment, community health services £33.08 [19] As above
 Physiotherapy, first appointment £56.60 [19] As above
 Physiotherapy, follow-up £45.86 [19] As above
 Speech and language therapist, first appointment £87.04 [19] As above
 Speech and language therapist, follow-up £99.32 [19] As above
 Occupational therapist, first appointment £142.11 [19] As above
 Occupational therapist, follow-up £58.25 [19] As above
 Clinical physiology, first appointment £76.39 [19] As above
 Clinical physiology, follow-up £51.58 [19] As above
 Gastroenterologist, first appointment £164.54 [19] As above
 Gastroenterologist, follow-up £132.50 [19] As above
 Podiatry, community health services £39.88 [19] 1 appointment per patient per year after symptom onset and during late-stage symptoms As above
 Rehabilitation services £94.26 [19] 1 appointment per patient per year after symptom onset As above
 MRI scan, brain and spine £133.64 [19] 1 appointment per patient per year after symptom onset As above
 Nerve conduction studies, paediatric £281.58 [19] As above
 Nerve conduction studies, adult services £206.71 [19] As above
 Sleep studies £501.71 [19] As above
 Bronchoscopy, paediatric £1589.23 [19] As above
 Bronchoscopy, adult services £632.10 [19] As above
 Tracheostomy insertion £3489.92 [19] As above
 Orthopaedics, paediatrics, first appointment £135.97 [19] 1 appointment per patient per year after symptom onset As above
 Orthopaedics, adult services, first appointment £135.74 [19] 1 appointment per patient per year after symptom onset As above
 Orthopaedics, adult services, follow-up £109.51 [19] As above
 Walking aids £7.60 [23] 1 per patient requiring wheelchair or walking aids As above
 Wheelchair, assessment, paediatric £309.04 [19] As above
 Wheelchair, equipment, paediatric £485.57 [19] As above
 Wheelchair, maintenance, paediatric £66.47 [19] As above
 Wheelchair, review, paediatric £249.11 [19] As above
 Wheelchair, assessment £247.21 [19] As above
 Wheelchair, equipment £167.64 [19] As above
 Wheelchair, maintenance £60.03 [19] As above
 Wheelchair, review £151.66 [19] As above
Psychological/psychiatric care
 Community mental health care, paediatric £242.38 [19] 12 appointments per patient with symptoms per year [30] NHS Reference Costs 2015–2016 [19] Patient experience (Wolfram Focus Group), 2016 [30]
 Mental health care initial assessment £257.59 [19] 1 appointment per patient after symptom onset [30] As above
 Community mental health care, adult £120.61 [19] 1 appointment per patient per year after symptom onset [30] As above
 Inpatient treatment of psychiatric disorders, per bed day £390.20 [19] 15 days review period per patient [30] As above
 Rehabilitation of psychiatric disorders £377.77 [19] 1 appointment per patient per year after symptom onset [30] As above
  1. ABR auditory brain response, BNF British National Formulary, CCG Clinical Commissioning Group, DI diabetes insipidus, DM diabetes mellitus, EMA endomysial antibody, ENT ear, nose and throat, GP general practitioner, HbA1c haemoglobin A1c, IgA immunoglobulin A, MDT multi-disciplinary team, MRI magnetic resonance imaging, NCGC National Clinical Guideline Centre, NHS National Health Service, PSSRU Personal Social Services Research Unit, SALT Speech and Language Therapist, SPH sphere, tTG tissue transglutaminase, UTI urinary tract infection, WFS1 Wolfram syndrome type 1 (gene)