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 |