scholarly journals A model for estimating the health economic impact of earlier diagnosis of chronic thromboembolic pulmonary hypertension

2021 ◽  
pp. 00719-2020
Author(s):  
Gudula J.A.M. Boon ◽  
Wilbert B. van den Hout ◽  
Stefano Barco ◽  
Harm Jan Bogaard ◽  
Marion Delcroix ◽  
...  

BackgroundDiagnostic delay of chronic thromboembolic pulmonary hypertension (CTEPH) exceeds >1 year, contributing to higher mortality. Health-economic consequences of late CTEPH diagnosis are unknown. We aimed to develop a model for quantifying the impact of diagnosing CTEPH earlier on survival, quality-adjusted life years (QALYs) and healthcare costs.Material and methodsA Markov model was developed to estimate lifelong outcomes, depending on the degree of delay. Data on survival and quality of life were obtained from published literature. Hospital costs were assessed from patient records (n=498) at the Amsterdam UMC - VUmc, which is a Dutch CTEPH referral center. Medication costs were based on a mix of standard medication regimens.ResultsFor 63-year-old CTEPH patients with a 14 months diagnostic delay of CTEPH (median age and delay of patients in the European CTEPH Registry), lifelong healthcare costs were estimated at €117 100 for a mix of treatment options. In a hypothetical scenario of maximal reduction of current delay, improved survival was estimated at a gain of 3.01 life years and 2.04 QALYs. The associated cost increase was €44 654, of which 87% was due to prolonged medication use. This accounts for an incremental cost-utility ratio of €21 900/QALY.ConclusionOur constructed model based on the Dutch healthcare setting demonstrates a substantial health gain when CTEPH is diagnosed earlier. According to Dutch health-economic standards, additional costs remain below the deemed acceptable limit of €50 000/QALY for the particular disease burden. This model can be used for evaluating cost-effectiveness of diagnostic strategies aimed at reducing the diagnostic delay.

2020 ◽  
Vol 41 (Supplement_2) ◽  
Author(s):  
E Klok ◽  
G.J.A.M Boon ◽  
Y.M Ende-Verhaar ◽  
R Bavalia ◽  
M Delcroix ◽  
...  

Abstract Background The current diagnostic delay of chronic thromboembolic pulmonary hypertension (CTEPH) after acute pulmonary embolism (PE) is unacceptably long exceeding 1 year, causing loss of quality-adjusted life years and excess mortality. Validated screening strategies to diagnose CTEPH earlier are lacking. Importantly, performing echocardiography in all PE patients for this purpose has a low diagnostic yield, is associated with overdiagnosis and is not cost-effective. Moreover, expertise in performing high-quality PH-dedicated echocardiograms may not be available outside expert centers. Aim To validate a simple screening strategy aimed at identifying CTEPH early in the course after acute PE, avoiding echocardiography if possible (Figure 1). Methods In this prospective, international, multicenter management study, consecutive PE survivors were managed according to the predefined algorithm starting three months after acute PE. All were followed for a total period of two years. The study protocol was approved by all local IRBs and all patients provided informed consent. Results 424 patients were included across three European countries (Table 1). Following the algorithm, CTEPH was considered excluded in 343 (81%) patients based on clinical pre-test probability assessment by the “CTEPH prediction score”, evaluation of symptoms and application of the “CTEPH rule-out criteria” (Figure 1); only 19% was subjected to echocardiography. Only 1 of 343 patients managed without echocardiography was diagnosed with CTEPH, 10 months after initial PE, for a failure rate of 0.29% (95% CI 0–1.6%). Overall, 13 patients were diagnosed with CTEPH (incidence 3.1%), of whom 10 within 4 months after PE diagnosis. Conclusions The algorithm accurately ruled out CTEPH and avoided echocardiography in 81% of patients. The vast majority of CTEPH cases were identified early in the course of acute PE which is a considerable improvement compared to current clinical practice with an economic use of healthcare resources. Figure 1. Study flowchart Funding Acknowledgement Type of funding source: Foundation. Main funding source(s): This study was supported by unrestricted grants from Bayer/Merck Sharp & Dohme (MSD) and Actelion Pharmaceuticals Ltd. F.A. Klok and G.J.A.M. Boon were supported by the Dutch Heart Foundation (2017T064).


Thorax ◽  
2021 ◽  
pp. thoraxjnl-2020-216324
Author(s):  
Gudula J A M Boon ◽  
Yvonne M Ende-Verhaar ◽  
Roisin Bavalia ◽  
Lahassan H El Bouazzaoui ◽  
Marion Delcroix ◽  
...  

Background The current diagnostic delay of chronic thromboembolic pulmonary hypertension (CTEPH) after pulmonary embolism (PE) is unacceptably long, causing loss of quality-adjusted life years and excess mortality. Validated screening strategies for early CTEPH diagnosis are lacking. Echocardiographic screening among all PE survivors is associated with overdiagnosis and cost-ineffectiveness. We aimed to validate a simple screening strategy for excluding CTEPH early after acute PE, limiting the number of performed echocardiograms. Methods In this prospective, international, multicentre management study, consecutive patients were managed according to a screening algorithm starting 3 months after acute PE to determine whether echocardiographic evaluation of pulmonary hypertension (PH) was indicated. If the ‘CTEPH prediction score’ indicated high pretest probability or matching symptoms were present, the ‘CTEPH rule-out criteria’ were applied, consisting of ECG reading and N-terminalpro-brain natriuretic peptide. Only if these results could not rule out possible PH, the patients were referred for echocardiography. Results 424 patients were included. Based on the algorithm, CTEPH was considered absent in 343 (81%) patients, leaving 81 patients (19%) referred for echocardiography. During 2-year follow-up, one patient in whom echocardiography was deemed unnecessary by the algorithm was diagnosed with CTEPH, reflecting an algorithm failure rate of 0.29% (95% CI 0% to 1.6%). Overall CTEPH incidence was 3.1% (13/424), of whom 10 patients were diagnosed within 4 months after the PE presentation. Conclusions The InShape II algorithm accurately excluded CTEPH, without the need for echocardiography in the overall majority of patients. CTEPH was identified early after acute PE, resulting in a substantially shorter diagnostic delay than in current practice.


2021 ◽  
Vol 5 (1) ◽  
Author(s):  
Brooke Currie ◽  
Evan Davies ◽  
Amélie Beaudet ◽  
Larissa Stassek ◽  
Leah Kleinman

Abstract Background Chronic thromboembolic pulmonary hypertension (CTEPH) is a rare form of pulmonary hypertension caused by blood clots and scar tissue in the blood vessels of the lungs. Health-related quality of life is often significantly impaired in patients with CTEPH. However, a better understanding of how CTEPH symptoms affect patients’ lives is needed to optimally assess the impact of the disease and treatment. Objectives This qualitative study aimed to better understand the symptoms of CTEPH and how they affect patients’ lives, as well as to determine the appropriateness of the Pulmonary Arterial Hypertension – Symptoms and Impact (PAH-SYMPACT™) questionnaire for use in this patient population. Methods Adults diagnosed with CTEPH, recruited from two clinical sites in the US, participated in one-to-one qualitative telephone interviews. They described their experience of CTEPH symptoms and the impact these symptoms have on their lives. They also provided feedback on the comprehensibility and relevance of the PAH-SYMPACT™‘s instructions, items, and response options. Results Participants (N = 12) had a mean age of 62.5 years. Two thirds were female and most (83%) had undergone pulmonary endarterectomy and/or balloon pulmonary angioplasty. The most frequently endorsed symptoms were shortness of breath (endorsed by all 12 participants), fatigue (11 participants), and lightheadedness (10 participants). All participants identified shortness of breath as an “extremely important” symptom, and seven participants rated fatigue as “extremely important.” The most frequent impacts of CTEPH were on ability to walk quickly (endorsed by all 12 participants), ability to walk up inclines or stairs (11 participants), and ability to carry things (11 participants). The PAH-SYMPACT™ items were relevant to most participants and reflected their experience of CTEPH. All participants indicated that no important CTEPH symptoms were missing from the PAH-SYMPACT™. Overall, the instructions, items, and response options of the PAH-SYMPACT™ were clear and easy to understand. Conclusions The symptoms and impacts experienced by patients with CTEPH align with items included in the PAH-SYMPACT™. The PAH-SYMPACT™ appears to be fit for purpose for assessing disease status in patients with CTEPH.


2020 ◽  
Vol 3-4 (213-214) ◽  
pp. 8-16
Author(s):  
Andrey Avdeyev ◽  
◽  
Murat Mukarov ◽  
Sabira Zhaugasheva ◽  
Lubov Piven ◽  
...  

Riociguat is included in international guidelines and local clinical protocols for the treatment of pulmonary hypertension (PH) and chronic thromboembolic pulmonary hypertension (CTEPH), where it is the first choice drug for the treatment of patients with inoperable, persistent and recurrent forms of CTEPH. Therefore, it is necessary to include this drug in the GVoFMC (The Guaranteed Volume of Free Medical Care) reimbursement lists and the CSHI (Compulsory Social Health Insurance) system, which will alleviate access to medication for patients suffering from PH and CTEPH. Purpose of the study. Determination of the cost effectiveness parameters of using riociguat in comparison with bosentan and combined therapy of bosentan/sildenafil in medication therapy of CTEPH. Material and methods. A Markov model of CTEPH dynamics in patients with riociguat prescription was developed in comparison with application of bosentan and sildenafil. Modeling step was 16 weeks, modeling horizon – 10 years. Direct medical costs per patient were taken into account in the model, including the cost of drug therapy, costs of outpatient visits due to CTEPH, hospitalization costs due to clinical deterioration of CTEPH, emergency calls. Results and discussion. The results of “cost-effectiveness” Markov pharmacoeconomic modeling showed that riociguat application reveals a dominant position in comparison with the practice of using bosentan with addition of sildenafil for specific therapy at deterioration of patient's condition up to III-IV functional class (FC). Riociguat is characterized by the best values of “cost effectiveness” coefficients according to efficiency criterion of FC increase and the “value of 6-min walk distance test”. The results of analysis of the impact on budget showed that inclusion of riociguat in GVoFMC/CSHI will lead to budget savings of 154 million tenge in the first year, 294 million tenge in the second year and 415 million tenge in the third year compared to the existing clinical practice without riociguat use. Conclusions. On the basis of conducted complex pharmacoeconomic analysis, the application of riociguat in patients with inoperable or persistent/ recurrent form of CTEPH is a more preferable strategy in comparison with the strategy of application of bosentan with addition of sildenafil and will allow to reduce budget expenses within the framework of GVoFMC and CSHI system. Keywords: pulmonary hypertension, chronic thromboembolic pulmonary hypertension, riociguat.


2019 ◽  
Vol 40 (Supplement_1) ◽  
Author(s):  
P Martinez Santos ◽  
A Aurtenetxe Perez ◽  
M J Lopez Gude ◽  
J A Barbera ◽  
M Lopez Meseguer ◽  
...  

Abstract Background Chronic thromboembolic pulmonary hypertension (CTEPH) treatment has evolved over the last decade. Increasing evidence regarding new therapeutic developments has shown clinical benefit among these patients in different scenarios. However, there is scarce information about the long-term impact of these achievements in a real-life population on a national scale. We aimed to analyze the impact of current CTEPH therapies on survival in Spain. Methods We prospectively collected epidemiological, clinical and prognostic data from CTEPH patients consecutively included in the Spanish REHAP registry from January 1, 2007, to December 31, 2017. All-cause mortality data were gathered during this period. Results Eight hundred thirteen patients were included. The mean age was 61 (15) years and 58.1% were women. Out of the 813 patients, 537 (66%) were referred to an expert PH-center. Overall, 245 (30.1%) patients were selected for surgery and 52 (6.4%) for percutaneous treatment and 452 (60.5%) received medical treatment exclusively with specific PH drugs. Survival rates of patients who underwent an invasive procedure (pulmonary thrombendarterectomy or balloon pulmonary angioplasty) were remarkably high. Figure 1. Cumulative survival from date Conclusions Patients who underwent pulmonary thrombendarterectomy or balloon pulmonary angioplasty associated a better outcome. Acknowledgement/Funding We gratefully acknowledge all investigators of the REHAP Registry. We express our gratitude to Actelion, Ferrer, GlaxoSmithKline (GSK) and Merck Sharp


Author(s):  
Daniela Cavalet Blanco ◽  
Noris Coimbra Scaglia ◽  
Flavio Vinicius Costa Ferreira ◽  
Sabrina Rocha Machado ◽  
Luciana Zani Viegas da Silva

2017 ◽  
Vol 49 (5) ◽  
pp. 1601980 ◽  
Author(s):  
Raffaele Pesavento ◽  
Lucia Filippi ◽  
Antonio Palla ◽  
Adriana Visonà ◽  
Carlo Bova ◽  
...  

The impact of residual pulmonary obstruction on the outcome of patients with pulmonary embolism is uncertain.We recruited 647 consecutive symptomatic patients with a first episode of pulmonary embolism, with or without concomitant deep venous thrombosis. They received conventional anticoagulation, were assessed for residual pulmonary obstruction through perfusion lung scanning after 6 months and then were followed up for up to 3 years. Recurrent venous thromboembolism and chronic thromboembolic pulmonary hypertension were assessed according to widely accepted criteria.Residual pulmonary obstruction was detected in 324 patients (50.1%, 95% CI 46.2–54.0%). Patients with residual pulmonary obstruction were more likely to be older and to have an unprovoked episode. After a 3-year follow-up, recurrent venous thromboembolism and/or chronic thromboembolic pulmonary hypertension developed in 34 out of the 324 patients (10.5%) with residual pulmonary obstruction and in 15 out of the 323 patients (4.6%) without residual pulmonary obstruction, leading to an adjusted hazard ratio of 2.26 (95% CI 1.23–4.16).Residual pulmonary obstruction, as detected with perfusion lung scanning at 6 months after a first episode of pulmonary embolism, is an independent predictor of recurrent venous thromboembolism and/or chronic thromboembolic pulmonary hypertension.


2021 ◽  
Vol 28 (Supplement_1) ◽  
Author(s):  
C Schmidt ◽  
M Monteiro ◽  
A Reis ◽  
M Santos

Abstract Funding Acknowledgements Type of funding sources: Public grant(s) – National budget only. Main funding source(s): Fundação para a Ciência (FCT) Coordenação de Aperfeiçoamento de Pessoal de Nível (CAPES) Background  Limited data is available on physical activity (PA) levels in chronic thromboembolic pulmonary hypertension (CTEPH) patients, as well as on the impact of using different tools to assess PA such as questionnaires and accelerometers.  Purpose  We aimed to quantify PA levels of CTEPH patients and study its clinical correlates, as well as to compare PA levels measured by the International Physical Activity Questionnaire (IPAQ) with measures from accelerometers. Methods  This is a cross-sectional study (n = 50). Physical activity levels were measured using accelerometers and questionnaire (IPAQ). Clinical parameters evaluated were walked distance on the 6-minute-walking test (6MWT), pulmonary vascular resistance, N-terminal brain natriuretic peptide and quality of life (HRQoL) using the Cambridge Pulmonary Hypertension Outcome Review questionnaire. Results  Accelerometer-derived data showed that CTEPH patients spent 60% of the recorded time in sedentary behaviours and only 2% in moderate-to-vigorous PA (MVPA). MVPA was mildly correlated with 6MWT (r = 0.359; p= 0.023) and symptom domain of HRQoL (r=-0.371; p = 0.044) but not with NT-proBNP, pulmonary vascular resistance or functional domain of HRQoL. Time spent in sedentary behaviour was lower in self-reported measurement (279 ± 165min/day) compared to accelerometry (446 ± 117min/day, p < 0.001). Self-reported MVPA was significantly higher than the one registered by the accelerometer (411 ± 569 vs. 131 ± 108 min/week, p = 0.027). Bland-Altman analysis indicated poor agreement between the two methods.  Conclusions  Our results showed that CTEPH patients spend most of their days in sedentary behaviors and only a small amount of time in MVPA. MVPA was associated with symptoms domain of HRQoL and submaximal functional capacity. In addition, we showed a poor agreement between self-reported and accelerometer-derived PA in CTEPH patients, with the former overestimating the overall PA activity.


2020 ◽  
Vol 55 (6) ◽  
pp. 2000189 ◽  
Author(s):  
Fredrikus A. Klok ◽  
Francis Couturaud ◽  
Marion Delcroix ◽  
Marc Humbert

Chronic thromboembolic pulmonary hypertension (CTEPH) is the most severe long-term complication of acute pulmonary embolism (PE). Untreated CTEPH is fatal, but, if diagnosed in time, successful surgical (pulmonary endarterectomy), medical (pulmonary hypertension drugs) and/or interventional (balloon pulmonary angioplasty) therapies have been shown to improve clinical outcomes, especially in case of successful pulmonary endarterectomy. Early diagnosis has however been demonstrated to be challenging. Poor awareness of the disease by patients and physicians, high prevalence of the post-PE syndrome (i.e. persistent dyspnoea, functional limitations and/or decreased quality of life following an acute PE diagnosis), lack of clear guideline recommendations as well as inefficient application of diagnostic tests in clinical practice lead to a reported staggering diagnostic delay >1 year. Hence, there is a great need to improve current clinical practice and diagnose CTEPH earlier. In this review, we will focus on the clinical presentation of and risk factors for CTEPH, and provide best practices for PE follow-up programmes from expert centres, based on a clinical case.


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