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The effectiveness and cost-effectiveness of treatments for idiopathic pulmonary fibrosis: systematic review, network meta-analysis and health economic evaluation

The effectiveness and cost-effectiveness of treatments for idiopathic pulmonary fibrosis: systematic review, network meta-analysis and health economic evaluation
The effectiveness and cost-effectiveness of treatments for idiopathic pulmonary fibrosis: systematic review, network meta-analysis and health economic evaluation

BACKGROUND: Idiopathic pulmonary fibrosis (IPF) is a life-limiting lung disease with considerable impact on patients and carers as the disease progresses. Currently few treatments are available. We aimed to evaluate the clinical and cost-effectiveness of available treatments for IPF.

METHODS: Systematic reviews of clinical effectiveness, quality of life and cost effectiveness were undertaken. Eleven bibliographic databases were searched from inception to July 2013 and studies were assessed for eligibility against a set of pre-defined criteria. Two reviewers screened references, extracted data from included studies and appraised their quality. An advisory group was consulted about the choice of interventions. A narrative review was undertaken and where feasible fixed effect and random effects meta-analysis were undertaken including a network meta-analysis (NMA). A decision-analytic Markov model was developed to estimate cost-effectiveness of pharmacological treatments for IPF. Following best practice recommendations, the model perspective was of the national health service and personal social services, a discount rate of 3.5% for costs and health benefits was applied and outcomes were expressed as cost per quality adjusted life-year gained. Parameter values were obtained from the NMA and systematic reviews. Sensitivity analyses were undertaken.

RESULTS: Fourteen studies were included in the review of clinical effectiveness, of which one evaluated azathioprine, three N-acetylcysteine [NAC] (alone or in combination), four pirfenidone, one nintedanib, one sildenafil, one thalidomide, two pulmonary rehabilitation, and one a disease management programme. Study quality was generally good. Evidence suggests that some effective treatments are available. In NMA only nintedanib and pirfenidone show statistically significant improvements. The model results show increased survival for five pharmacological treatments (NAC triple therapy, inhaled NAC, nintedanib, pirfenidone, and sildenafil) compared with best supportive care, at increased cost. Only inhaled NAC was cost-effective at current willingness to pay thresholds but it may not be clinically effective.

CONCLUSIONS: Few interventions have any statistically significant effect and the cost-effectiveness of treatments is uncertain. A lack of studies on palliative care approaches was identified and there is a need for further research into pulmonary rehabilitation and thalidomide in particular. A well conducted RCT on inhaled NAC therapy should also be considered.

Cost-Benefit Analysis, Humans, Idiopathic Pulmonary Fibrosis/drug therapy, Models, Economic, Quality of Life, Treatment Outcome
2050-6511
1-13
Loveman, Emma
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Copley, Vicky R
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Colquitt, Jill L
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Scott, David A
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Clegg, Andy J
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Jones, Jeremy
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O'Reilly, Katherine M A
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Singh, Sally
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Bausewein, Claudia
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Wells, Athol
483d421a-1139-433d-8375-44a023d2043f
Loveman, Emma
06ff1bf1-0189-4330-b22d-f5a917e9871d
Copley, Vicky R
32f28acd-1199-4c6e-a08d-3d1a9c11778d
Colquitt, Jill L
b5872647-bc73-47d1-be44-8d1170eddbc6
Scott, David A
19b5fd34-9974-4ae4-8be0-27a693639e20
Clegg, Andy J
cf2169af-28bc-44d1-a273-9c0fe209f3ad
Jones, Jeremy
270b303b-6bad-4be7-8ea0-63d0e8015c91
O'Reilly, Katherine M A
05c7174f-1231-45d7-b303-a499ebe2e34d
Singh, Sally
6969feff-1101-412d-8869-742342132e81
Bausewein, Claudia
a1ee0e8d-033a-4e67-ae7e-4f8e07099f66
Wells, Athol
483d421a-1139-433d-8375-44a023d2043f

Loveman, Emma, Copley, Vicky R, Colquitt, Jill L, Scott, David A, Clegg, Andy J, Jones, Jeremy, O'Reilly, Katherine M A, Singh, Sally, Bausewein, Claudia and Wells, Athol (2014) The effectiveness and cost-effectiveness of treatments for idiopathic pulmonary fibrosis: systematic review, network meta-analysis and health economic evaluation. BMC Pharmacology & Toxicology, 15, 1-13, [63]. (doi:10.1186/2050-6511-15-63).

Record type: Review

Abstract

BACKGROUND: Idiopathic pulmonary fibrosis (IPF) is a life-limiting lung disease with considerable impact on patients and carers as the disease progresses. Currently few treatments are available. We aimed to evaluate the clinical and cost-effectiveness of available treatments for IPF.

METHODS: Systematic reviews of clinical effectiveness, quality of life and cost effectiveness were undertaken. Eleven bibliographic databases were searched from inception to July 2013 and studies were assessed for eligibility against a set of pre-defined criteria. Two reviewers screened references, extracted data from included studies and appraised their quality. An advisory group was consulted about the choice of interventions. A narrative review was undertaken and where feasible fixed effect and random effects meta-analysis were undertaken including a network meta-analysis (NMA). A decision-analytic Markov model was developed to estimate cost-effectiveness of pharmacological treatments for IPF. Following best practice recommendations, the model perspective was of the national health service and personal social services, a discount rate of 3.5% for costs and health benefits was applied and outcomes were expressed as cost per quality adjusted life-year gained. Parameter values were obtained from the NMA and systematic reviews. Sensitivity analyses were undertaken.

RESULTS: Fourteen studies were included in the review of clinical effectiveness, of which one evaluated azathioprine, three N-acetylcysteine [NAC] (alone or in combination), four pirfenidone, one nintedanib, one sildenafil, one thalidomide, two pulmonary rehabilitation, and one a disease management programme. Study quality was generally good. Evidence suggests that some effective treatments are available. In NMA only nintedanib and pirfenidone show statistically significant improvements. The model results show increased survival for five pharmacological treatments (NAC triple therapy, inhaled NAC, nintedanib, pirfenidone, and sildenafil) compared with best supportive care, at increased cost. Only inhaled NAC was cost-effective at current willingness to pay thresholds but it may not be clinically effective.

CONCLUSIONS: Few interventions have any statistically significant effect and the cost-effectiveness of treatments is uncertain. A lack of studies on palliative care approaches was identified and there is a need for further research into pulmonary rehabilitation and thalidomide in particular. A well conducted RCT on inhaled NAC therapy should also be considered.

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Published date: 19 November 2014
Keywords: Cost-Benefit Analysis, Humans, Idiopathic Pulmonary Fibrosis/drug therapy, Models, Economic, Quality of Life, Treatment Outcome

Identifiers

Local EPrints ID: 441420
URI: http://eprints.soton.ac.uk/id/eprint/441420
ISSN: 2050-6511
PURE UUID: 9dda6694-61c4-4cec-9b87-5628ae35653f
ORCID for David A Scott: ORCID iD orcid.org/0000-0001-6475-8046

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Date deposited: 11 Jun 2020 16:41
Last modified: 30 Jun 2020 00:47

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