Cost-effectiveness analysis of three algorithms for diagnosing primary ciliary dyskinesia: a simulation study
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(2019) 14:142
RESEARCH
Open Access
Cost-effectiveness analysis of three algorithms for diagnosing primary ciliary dyskinesia: a simulation study Panayiotis Kouis1,2,9* , Stefania I. Papatheodorou2,3, Nicos Middleton4, George Giallouros1, Kyriacos Kyriacou5,6, Joshua T. Cohen7, John S. Evans8 and Panayiotis K. Yiallouros1
Abstract Background: Primary Ciliary Dyskinesia (PCD) diagnosis relies on a combination of tests which may include (a) nasal Nitric Oxide (nNO), (b) High Speed Video Microscopy (HSVM) and (c) Transmission Electron Microscopy (TEM). There is variability in the availability of these tests and lack of universal agreement whether diagnostic tests should be performed in sequence or in parallel. We assessed three combinations of tests for PCD diagnosis and estimated net sensitivity and specificity as well as cost-effectiveness (CE) and incremental cost-effectiveness (ICE) ratios. Methods and results: A hypothetical initial population of 1000 referrals (expected 320 PCD patients) was followed through a probabilistic decision analysis model which was created to assess the CE of three diagnostic algorithms (a) nNO + TEM in sequence, (b) nNO + HSVM in sequence and (c) nNO/HSVM in parallel followed, in cases with conflicting results, by confirmatory TEM (nNO/HSVM+TEM). Number of PCD patients identified, CE and ICE ratios were calculated using Monte Carlo simulations. Out of 320 expected PCD patients, 313 were identified by nNO/ HSVM+TEM, 274 with nNO + HSVM and 198 with nNO + TEM. The nNO/HSVM+TEM had the highest mean annual cost (€209 K) followed by nNO + TEM (€150 K) and nNO + HSVM (€136 K). The nNO + HSVM algorithm dominated the nNO + TEM algorithm (less costly and more effective). The ICE ratio for nNO/HSVM+TEM was €2.1 K per additional PCD patient identified. Conclusions: The diagnostic algorithm (nNO/HSVM+TEM) with parallel testing outperforms algorithms with tests in sequence. These findings, can inform the dialogue on the development of evidence-based guidelines for PCD diagnostic testing. Future research in understudied aspects of the disease, such as PCD-related quality of life and PCD-associated costs, is needed to help the better implementation of these guidelines across various healthcare systems. Keywords: Primary ciliary dyskinesia, Diagnosis, Cost-effectiveness analysis, Decision analysis Kartagener syndrome, Nitric oxide, High speed video microscopy, Transmission Electron microscopy
Introduction Primary Ciliary Dyskinesia (PCD) is a genetically heterogeneous disorder that affects one in approximately 15,000 live births [1]. PCD is characterized by chronic sinopulmonary symptoms and development of bronchiectasis, recurrent otitis, male infertility and situs inversus [2]. Defective * Correspondence: [email protected] 1 Respiratory Physiology Laboratory, Medical School, University of Cyprus, Nicosia, Cyprus 2 Cyprus International Institute for Environmental and Public Health, Cyprus University of Technology, Limassol, Cyprus Full list of author information is available at the end o
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