Cost-effectiveness analysis of three algorithms for diagnosing primary ciliary dyskinesia: a simulation study.
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...
| Publicado en: | Orphanet Journal of Rare Diseases Vol. 14; no. 1 |
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| Autores principales: | , , , , , , , |
| Formato: | research Journal Article |
| Publicado: |
BioMed Central
6/13/2019
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| Acceso en línea: | Ver este registro en EBSCOhost |
| fields | @attributes: recordID: 1 pdfLink: plink: https://search.ebscohost.com/login.aspx?direct=true&db=ccm&AN=136999590&site=ehost-live header: @attributes: shortDbName: ccm uiTerm: 136999590 longDbName: CINAHL Complete uiTag: AN controlInfo: bkinfo: dissinfo: jinfo: jid: 17501172 38NP jtl: Orphanet Journal of Rare Diseases issn: 17501172 maglogo: N pubinfo: dt: 6/13/2019 vid: 14 iid: 1 pid: 24147 pub: BioMed Central artinfo: ui: 136999590 136999590 NLM31196140 136999590 10.1186/s13023-019-1116-3 NLM31196140 136999590 ppct: 1 formats: tig: atl: Cost-effectiveness analysis of three algorithms for diagnosing primary ciliary dyskinesia: a simulation study. aug: au: Kouis, Panayiotis Papatheodorou, Stefania I. Middleton, Nicos Giallouros, George Kyriacou, Kyriacos Cohen, Joshua T. Evans, John S. Yiallouros, Panayiotis K. affil: Respiratory Physiology Laboratory, Medical School, University of Cyprus, Nicosia, Cyprus sug: subj: Algorithms Ciliary Motility Disorders Diagnosis Ciliary Motility Disorders Physiopathology Cost Benefit Analysis Sensitivity and Specificity Female Ciliary Motility Disorders Metabolism Quality of Life Male Human Nitric Oxide Metabolism Microscopy, Electron Validation Studies Comparative Studies Evaluation Research Multicenter Studies Ferrans and Powers Quality of Life Index Female Male ab: 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. pubtype: Academic Journal doctype: research Journal Article ougenre: Article language: English refInfo: holdings: @attributes: islocal: N |
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