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Table 1 Key inputs in the model

From: The burden of congenital hyperinsulinism in the United Kingdom: a cost of illness study

Input

Value

Reference

Population inputs

 Prevalence of CHIb

0.003%

Estimated scope of the national CHI service (NHS England CHI Service Standard Contract, 2013) [4]

 Number of newly diagnosed patients with CHI per yeara,b

95

Expert opinion (GOSH, 2015; NORCHI, 2016) [19, 30]

 Maximum age of patients that have received a near-total pancreatectomyb

54

Harold N. Lovvorn III, 1999 [23]

Cost inputs per patient

 Daily cost of inpatient care at local hospital (excess)a

£486.62

NHS Reference Costs (2015–16) [20]

 Daily cost of inpatient care at specialist centre (excess)a

£486.62

NHS Reference Costs (2015–16) [20]

 Daily cost of neonatal care (excess)a

£274.11

NHS Reference Costs (2015–16) [20]

 Cost of inpatient care, per non-elective long stay at local hospitala

£2186.61

NHS Reference Costs (2015–16) [20]

 Annual cost of paediatric outpatient diabetes careb

£2925.00

NHS National Tariff (2014–15) [31]

 Total annual cost of IDDM for patients managed by multiple daily insulin injections (including severe and non-severe events)b

£2183.00

Evans, 2015 (2013)* [32]

 Cost of insertion of central venous catheter (Hickman line)a

£5925.98

NHS Reference Costs (2015–16) [20]

Clinical inputs

 Proportion of CHI patients responsive to first line therapy at local hospitala,b

75%

Expert opinion (NORCHI, 2016) [13]

 Proportion of IDDM patients managed by multiple daily injectionsb

90%

Expert opinion (GOSH and NORCHI, 2016) [13, 19]

 Incidence of IDDM at 11 years post near-total pancreatectomyb

96%

Arya, 2014 [15]

 Proportion of CHI patients with diffuse disease responsive to octreotideb

30%

Expert opinion (NORCHI, 2016) [13]

 Number of days until discharge from hospital following successful first-line therapya,b

14

Expert opinion (GOSH and NORCHI, 2016) [13, 19]

 Number of days to assess response to medical treatment in neonatal carea

10

Expert opinion (GOSH, 2016) [19]

 Number of days from octreotide non-response until surgerya

21

Expert opinion (NORCHI), 2016 [30]

  1. *Cost inflated to 2015/2016 cost year using the Personal Social Services Research Unit’s (PSSRU) hospital and community health services (HCHS) index [33]
  2. Key inputs shown include the ten greatest cost drivers of first year costsa and the ten greatest cost drivers of total annual costsb as identified by the DSA