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Title: Cost-effectiveness analysis of three algorithms for diagnosing primary ciliary dyskinesia: a simulation study
Authors: Kouis, Panayiotis 
Papatheodorou, Stefania 
Middleton, Nicos 
Giallouros, George 
Kyriacou, Kyriacos 
Cohen, Joshua T. 
Evans, John S. 
Yiallouros, Panayiotis K. 
Major Field of Science: Medical and Health Sciences
Field Category: Clinical Medicine
Keywords: Cost-effectiveness analysis;Decision analysis Kartagener syndrome;Diagnosis;High speed video microscopy;Nitric oxide;Primary ciliary dyskinesia;Transmission Electron microscopy
Issue Date: 13-Jun-2019
Source: Orphanet Journal of Rare Diseases, 2019, vol. 14, no. 1
Volume: 14
Issue: 1
Project: Better Experimental Screening and Treatment for Primary Ciliary Dyskinesia 
Journal: Orphanet Journal of Rare Diseases 
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.
ISSN: 1750-1172
DOI: 10.1186/s13023-019-1116-3
Rights: © The Author(s)
Type: Article
Affiliation : University of Cyprus 
Cyprus University of Technology 
Harvard University 
Cyprus Institute of Neurology and Genetics 
Cyprus School of Molecular Medicine 
Tufts Center for the Study of Drug Development 
Harvard T.H. Chan School of Public Health 
Shiakolas Educational Center of Clinical Medicine 
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