Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States

Background: Limited data exist on the use of drugs in patients with pulmonary arterial hypertension associated with congenital heart disease (PAH-CHD). Therefore, we evaluated their real-world patient journey, including symptomatology, diagnostic pathway, treatment patterns, and guideline adherence....

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Main Authors: Jordan D. Awerbach, MD, MPH, Carly J. Paoli, PharmD, MPH, Megan Scott, BSc, Gurinderpal Doad, PharmD, Julia Harley, BSc, Daniel Graham, MSc, Mark Small, BSc, Sumeet Panjabi, PhD, Leigh C. Reardon, MD, FACC
Format: Article
Language:English
Published: Elsevier 2025-08-01
Series:JHLT Open
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Online Access:http://www.sciencedirect.com/science/article/pii/S2950133425001211
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author Jordan D. Awerbach, MD, MPH
Carly J. Paoli, PharmD, MPH
Megan Scott, BSc
Gurinderpal Doad, PharmD
Julia Harley, BSc
Daniel Graham, MSc
Mark Small, BSc
Sumeet Panjabi, PhD
Leigh C. Reardon, MD, FACC
author_facet Jordan D. Awerbach, MD, MPH
Carly J. Paoli, PharmD, MPH
Megan Scott, BSc
Gurinderpal Doad, PharmD
Julia Harley, BSc
Daniel Graham, MSc
Mark Small, BSc
Sumeet Panjabi, PhD
Leigh C. Reardon, MD, FACC
author_sort Jordan D. Awerbach, MD, MPH
collection DOAJ
description Background: Limited data exist on the use of drugs in patients with pulmonary arterial hypertension associated with congenital heart disease (PAH-CHD). Therefore, we evaluated their real-world patient journey, including symptomatology, diagnostic pathway, treatment patterns, and guideline adherence. Methods: Data were drawn from the Adelphi Real World pulmonary hypertension congenital heart disease Disease Specific Programme (DSP), a cross-sectional survey of clinicians and patients with PAH-CHD or Fontan circulation associated with elevated pulmonary vascular resistance (PVR). Data were collected in the United States from November 2021 to May 2022. Analyses were descriptive. Results: Overall, 51 clinicians reported data for 191 patients with PAH-CHD or Fontan circulation associated with elevated PVR. Fifty-eight patients voluntarily provided data. Overall, 10.5% of patients had a gap of ≥1 year in their disease management. Mean (standard deviation, SD) time from pulmonary hypertension symptom onset to diagnosis/confirmation was 1.7 (2.2) years. Overall, 75.0% of patients underwent a right heart catheterization (RHC) at diagnosis/confirmation. Clinicians reported that 75.9% of patients were prescribed treatment for their PAH-CHD or elevated PVR. Pulmonary hypertension specific therapy was prescribed as combination therapy for 47.6% of patients prescribed pulmonary hypertension specific treatment. Conclusions: Patients experienced delays to diagnosis and gaps in congenital heart disease management. We observed low utilization of RHC and combination therapy. Key unmet needs in this population include more frequent testing to shorten time-to-diagnosis and proactive management with initial combination therapy.
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spelling doaj-art-3d864e41acf64a69befee513eeafe6372025-08-20T03:36:37ZengElsevierJHLT Open2950-13342025-08-01910032610.1016/j.jhlto.2025.100326Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United StatesJordan D. Awerbach, MD, MPH0Carly J. Paoli, PharmD, MPH1Megan Scott, BSc2Gurinderpal Doad, PharmD3Julia Harley, BSc4Daniel Graham, MSc5Mark Small, BSc6Sumeet Panjabi, PhD7Leigh C. Reardon, MD, FACC8Center for Heart Care, Phoenix Children’s, Phoenix, AZ; Departments of Internal Medicine and Child Health, University of Arizona College of Medicine-Phoenix, Phoenix, AZ; Corresponding author: Jordan D. Awerbach, MD, MPH, Phoenix Children's Hospital, Thomas Campus 1919 E. Thomas Rd., Phoenix, AZ 85016.Actelion Pharmaceuticals US Inc, San Francisco, CAAdelphi Real World, Bollington, United KingdomActelion Pharmaceuticals US Inc, San Francisco, CAAdelphi Real World, Bollington, United KingdomAdelphi Real World, Bollington, United KingdomAdelphi Real World, Bollington, United KingdomActelion Pharmaceuticals US Inc, San Francisco, CAAhmanson/UCLA Adult Congenital Heart Disease Center and UCLA Mattel Children’s Hospital, University of California Los Angeles, Los Angeles, CABackground: Limited data exist on the use of drugs in patients with pulmonary arterial hypertension associated with congenital heart disease (PAH-CHD). Therefore, we evaluated their real-world patient journey, including symptomatology, diagnostic pathway, treatment patterns, and guideline adherence. Methods: Data were drawn from the Adelphi Real World pulmonary hypertension congenital heart disease Disease Specific Programme (DSP), a cross-sectional survey of clinicians and patients with PAH-CHD or Fontan circulation associated with elevated pulmonary vascular resistance (PVR). Data were collected in the United States from November 2021 to May 2022. Analyses were descriptive. Results: Overall, 51 clinicians reported data for 191 patients with PAH-CHD or Fontan circulation associated with elevated PVR. Fifty-eight patients voluntarily provided data. Overall, 10.5% of patients had a gap of ≥1 year in their disease management. Mean (standard deviation, SD) time from pulmonary hypertension symptom onset to diagnosis/confirmation was 1.7 (2.2) years. Overall, 75.0% of patients underwent a right heart catheterization (RHC) at diagnosis/confirmation. Clinicians reported that 75.9% of patients were prescribed treatment for their PAH-CHD or elevated PVR. Pulmonary hypertension specific therapy was prescribed as combination therapy for 47.6% of patients prescribed pulmonary hypertension specific treatment. Conclusions: Patients experienced delays to diagnosis and gaps in congenital heart disease management. We observed low utilization of RHC and combination therapy. Key unmet needs in this population include more frequent testing to shorten time-to-diagnosis and proactive management with initial combination therapy.http://www.sciencedirect.com/science/article/pii/S2950133425001211Pulmonary arterial hypertensionCongenital heart diseasePulmonary vascular diseaseFontan circulationTreatment patternsReal-world evidence
spellingShingle Jordan D. Awerbach, MD, MPH
Carly J. Paoli, PharmD, MPH
Megan Scott, BSc
Gurinderpal Doad, PharmD
Julia Harley, BSc
Daniel Graham, MSc
Mark Small, BSc
Sumeet Panjabi, PhD
Leigh C. Reardon, MD, FACC
Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States
JHLT Open
Pulmonary arterial hypertension
Congenital heart disease
Pulmonary vascular disease
Fontan circulation
Treatment patterns
Real-world evidence
title Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States
title_full Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States
title_fullStr Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States
title_full_unstemmed Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States
title_short Real-world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease: Results from a real-world survey in the United States
title_sort real world experiences and treatment patterns among congenital heart disease patients with associated pulmonary vascular disease results from a real world survey in the united states
topic Pulmonary arterial hypertension
Congenital heart disease
Pulmonary vascular disease
Fontan circulation
Treatment patterns
Real-world evidence
url http://www.sciencedirect.com/science/article/pii/S2950133425001211
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