Evidence map›Paper›PMID 31196140›Full record

ArticleOrphanet journal of rare diseases2019

Cost-effectiveness analysis of three algorithms for diagnosing primary ciliary dyskinesia: a simulation study.

Panayiotis Kouis, Stefania I Papatheodorou, Nicos Middleton, George Giallouros, Kyriacos Kyriacou, Joshua T Cohen, John S Evans, Panayiotis K Yiallouros

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Orphanet journal of rare diseases, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.9field-weighted citation impact, top 24% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed, 6 citations in OpenAlex.

  1. Observational
  2. Article
  3. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

8 authors at 4 institutions in 2 countries.

Panayiotis KouisRespiratory Physiology Laboratory, Medical School, University of Cyprus, Nicosia, Cyprus. kouis.panayiotis@ucy.ac.cy.ORCID 0000-0003-0511-5352
Stefania I PapatheodorouCyprus International Institute for Environmental and Public Health, Cyprus University of Technology, Limassol, Cyprus.
Nicos MiddletonDepartment of Nursing, Cyprus University of Technology, Limassol, Cyprus.
George GiallourosRespiratory Physiology Laboratory, Medical School, University of Cyprus, Nicosia, Cyprus.
Kyriacos KyriacouDepartment of Electron Microscopy and Molecular Pathology, Cyprus Institute of Neurology and Genetics, Nicosia, Cyprus.
Joshua T CohenTufts Center for the Study of Drug Development, Boston, USA.
John S EvansDepartment of Environmental Health, Harvard T. H. Chan School of Public Health, Boston, USA.
Panayiotis K YiallourosRespiratory Physiology Laboratory, Medical School, University of Cyprus, Nicosia, Cyprus.
Cyprus University of Technology · CYHarvard University · USUniversity of Cyprus · CYCyprus Institute of Neurology and Genetics · CY

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundPrimary 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

resultsA 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.

conclusionsThe 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.

Indexed as

AlgorithmsCiliary Motility DisordersCost-Benefit AnalysisFemaleHumansMaleMicroscopy, Electron, TransmissionNitric OxideQuality of LifeSensitivity and SpecificityNitric OxideCost-effectiveness analysisDecision analysis Kartagener syndromeDiagnosisHigh speed video microscopyNitric oxidePrimary ciliary dyskinesiaTransmission Electron microscopy

Identifiers

PMID31196140
PMCPMC6567920
OpenAlexW2949337060

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.