Abstract:
:Pulmonary arterial hypertension (PAH) associated with congenital heart disease (CHD) due to systemic to pulmonary shunting is associated with a high risk of morbidity and mortality. In this study we evaluated 4 years treatment effect of bosentan on exercise capacity and quality of life and survival rates in 64 adult patients with PAH associated with CHD, including patients with Down syndrome (DS). All patients were evaluated at baseline and during follow-up with laboratory tests, 6-minute walk test, quality of life questionnaires, and Doppler echocardiography. In total, 13 patients (20%) died during 4-years of follow-up; 4 patients with DS and 9 patients without DS. Mean follow-up of all patients treated with bosentan was 3.5 ± 1.2 year. We analyzed treatment efficacy separately within patients without DS (n=34) and patients with DS (n=30). Mean 6-minute walking distance (6 MWD) in patients without DS significantly increased at 6 months from 417 ± 108 to 458 ± 104 m (+41 m; p=0.002) and significant improvement continued to exist during at least 2.5 years of follow-up (p=0.003). Moreover, stroke volume increased significantly (p=0.02). In the patients with DS, 6-MWD, stroke volume and quality of life remained stable during treatment. In this study we demonstrate a prolonged beneficial effect of bosentan treatment on exercise capacity, stroke volume and quality of life in patients without DS. However the mortality rate of 20% of patients after 4 years of follow-up remains high.
journal_name
Int J Cardioljournal_title
International journal of cardiologyauthors
Vis JC,Duffels MG,Mulder P,de Bruin-Bon RH,Bouma BJ,Berger RM,Hoendermis ES,van Dijk AP,Mulder BJdoi
10.1016/j.ijcard.2011.06.064subject
Has Abstractpub_date
2013-03-20 00:00:00pages
64-9issue
1eissn
0167-5273issn
1874-1754pii
S0167-5273(11)00604-8journal_volume
164pub_type
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pub_type: 杂志文章,多中心研究,随机对照试验
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