The effect of beta-blocker therapy on clinical outcome in patients with Marfan's syndrome: A meta-analysis

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Abstract

Objective

To assess the effect of beta-blockade therapy on clinical outcome in patients with Marfan's syndrome.

Background

Despite the lack of definitive evidence to support its efficacy, beta-blocker therapy is widely used prophylactically in patients with Marfan's syndrome.

Methods

A meta-analysis was instituted, which included studies identified by a systematic review of MEDLINE of peer-reviewed publications and by abstracts from annual scientific meeting. Outcome measures of mortality and major morbidity were compared between patients treated and untreated with beta-blockade therapy. Data was combined according to both a fixed-effects and random-effects model. The endpoints included aortic dissection or rupture, cardiovascular surgery, or death.

Results

Six studies were included, 5 were non-randomized follow-up studies and 1 was a prospective randomized trial (802 patients). Ninety-six of 433 patients treated with beta-blocker therapy and 74 of 369 untreated patients reached designated endpoints. Utilizing a fixed-effects model, patients treated with beta-blocker therapy were more likely to reach an endpoint (odds ratio = 1.50 with 95% CI 1.05–2.16). However, by a random-effects model, the treatment effect failed to reach significance (1.54 with 95% CI 0.99–2.40).

Conclusions

On the basis of this meta-analysis, there is no evidence that beta-blockade therapy has clinical benefit in patients with Marfan's syndrome.

Section snippets

Background

The major causes of mortality and morbidity in Marfan's patients are the cardiovascular complications of aortic dissection and rupture [1]. In the late 1960s it was reported that blood pressure lowering medication improves survival in patients in the general population with acute dissection of aortic aneurysms [2], [3], [4], [5], [6]. This therapy then began to be utilized for the prophylactic treatment of patients with aortic root dilatation related to Marfan's syndrome. It has been reported

Search

We searched Medline database from 1965, with the latest electronic search on July 10, 2005 for publications of long-term follow-up of patients with Marfan's syndrome. Keywords included Marfan's syndrome and outcome or beta-blockade or beta-adrenergic blockade or cardiovascular surgery. Language of publication did not influence article selection. Titles and abstracts were screened to exclude ineligible studies. References from these studies and from related review articles or editorials were

Study selection and characteristics

Seventeen studies, which included relevant long-term follow-up data, were considered for inclusion in the study. Ten were excluded because of incomplete or redundant information. One study had no control group.

A total of 6 studies were thus included. All studies based the diagnosis on internationally established clinical criteria for Marfan's syndrome. The study by Salim et al. [10] used genetic information to confirm the diagnosis in one sub-group of patients. Clinical data from the six

Discussion

Clinical use of beta-blocker therapy and other anti-hypertensive agents for dissecting aortic aneurysms began in the late 1960s. This treatment was instituted after the observation that patients with aortic dissection or rupture had a high incidence of hypertension (75%) [4]. Studies with turkeys demonstrated that propanolol appeared to prevent aortic rupture at a dose which did not significantly alter mean arterial pressure but did decrease cardiac impulse [17], [18]. This finding suggested

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