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Long-term prophylaxis in hereditary angio-oedema: a systematic review.

Costantino G, Casazza G, Bossi I, Duca P, Cicardi M - BMJ Open (2012)

Bottom Line: Electronic databases were searched up to April 2011.Two reviewers selected the studies and extracted the study data, patient characteristics and outcomes of interest.The results were as follows: danazol (RR=0.023, 95% CI 0.003 to 0.162), methyltestosterone (RR=0.054, 95% CI 0.013 to 0.163), ε-aminocaproic acid (RR=0.095, 95% CI 0.025 to 0.356), tranexamic acid (RR=0.308, 95% CI 0.195 to 0.479) and C1-INH 0.491 (95% CI 0.395 to 0.607).

View Article: PubMed Central - PubMed

Affiliation: Unità Operativa di Medicina Interna II, Dipartimento di Scienze Cliniche "L. Sacco", Ospedale L. Sacco, Università degli Studi di Milano, Milan, Italy.

ABSTRACT

Objective: To systematically review the evidence regarding long-term prophylaxis in the prevention or reduction of attacks in hereditary angio-oedema (HAE).

Design: Systematic review and meta-analysis.

Data sources: Electronic databases were searched up to April 2011. Two reviewers selected the studies and extracted the study data, patient characteristics and outcomes of interest.

Eligibility criteria for selected studies: Controlled trials for HAE prophylaxis.

Results: 7 studies were included, for a total of 73 patients and 587 HAE attacks. Due to the paucity of studies, a meta-analysis was not possible. Since two studies did not report the number of HAE attacks, five studies (52 patients) were finally included in the summary analysis. Four classes of drugs with at least one controlled trial have been proposed for HAE prophylaxis. All those drugs, except heparin, were found to be more effective than placebo. In the absence of direct comparisons, the relative efficacies of these drugs were determined by calculating a RR of attacks (drug vs placebo). The results were as follows: danazol (RR=0.023, 95% CI 0.003 to 0.162), methyltestosterone (RR=0.054, 95% CI 0.013 to 0.163), ε-aminocaproic acid (RR=0.095, 95% CI 0.025 to 0.356), tranexamic acid (RR=0.308, 95% CI 0.195 to 0.479) and C1-INH 0.491 (95% CI 0.395 to 0.607).

Conclusions: Few trials have evaluated the benefits of HAE prophylaxis, and all drugs but heparin seem to be effective in this setting. Since there are no direct comparisons of HAE drugs, it was not possible to draw definitive conclusions on the most effective one. Thus, to accumulate evidence for HAE prophylaxis, further studies are needed that consider the dose-efficacy relationship and include a head-to-head comparison between drugs, with the active group, rather than placebo, as the control.

No MeSH data available.


Related in: MedlinePlus

Search history.
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Related In: Results  -  Collection

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fig1: Search history.

Mentions: From the 11 412 references identified by the search strategy, 11 344 were excluded after title/abstract review. Of the remaining 68 references, 61 were excluded after a full-text evaluation: 43 articles reported studies that were not trials, four studies were duplicate publications, three studies had no end point, one article was a trial with retrospective controls and the focus of 10 studies was on therapy only, not on prophylaxis. Thus, seven studies were eligible for descriptive analysis (figure 1). The total number of patients enrolled in the seven studies was 73 (range 4–22). The efficacy evaluation was based on HAE attack recurrences, with 587 recurrences registered by the studies. Two of the seven studies1718 did not report the number of attacks; thus five studies (52 patients) were considered for the analysis.19–23


Long-term prophylaxis in hereditary angio-oedema: a systematic review.

Costantino G, Casazza G, Bossi I, Duca P, Cicardi M - BMJ Open (2012)

Search history.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC3400069&req=5

fig1: Search history.
Mentions: From the 11 412 references identified by the search strategy, 11 344 were excluded after title/abstract review. Of the remaining 68 references, 61 were excluded after a full-text evaluation: 43 articles reported studies that were not trials, four studies were duplicate publications, three studies had no end point, one article was a trial with retrospective controls and the focus of 10 studies was on therapy only, not on prophylaxis. Thus, seven studies were eligible for descriptive analysis (figure 1). The total number of patients enrolled in the seven studies was 73 (range 4–22). The efficacy evaluation was based on HAE attack recurrences, with 587 recurrences registered by the studies. Two of the seven studies1718 did not report the number of attacks; thus five studies (52 patients) were considered for the analysis.19–23

Bottom Line: Electronic databases were searched up to April 2011.Two reviewers selected the studies and extracted the study data, patient characteristics and outcomes of interest.The results were as follows: danazol (RR=0.023, 95% CI 0.003 to 0.162), methyltestosterone (RR=0.054, 95% CI 0.013 to 0.163), ε-aminocaproic acid (RR=0.095, 95% CI 0.025 to 0.356), tranexamic acid (RR=0.308, 95% CI 0.195 to 0.479) and C1-INH 0.491 (95% CI 0.395 to 0.607).

View Article: PubMed Central - PubMed

Affiliation: Unità Operativa di Medicina Interna II, Dipartimento di Scienze Cliniche "L. Sacco", Ospedale L. Sacco, Università degli Studi di Milano, Milan, Italy.

ABSTRACT

Objective: To systematically review the evidence regarding long-term prophylaxis in the prevention or reduction of attacks in hereditary angio-oedema (HAE).

Design: Systematic review and meta-analysis.

Data sources: Electronic databases were searched up to April 2011. Two reviewers selected the studies and extracted the study data, patient characteristics and outcomes of interest.

Eligibility criteria for selected studies: Controlled trials for HAE prophylaxis.

Results: 7 studies were included, for a total of 73 patients and 587 HAE attacks. Due to the paucity of studies, a meta-analysis was not possible. Since two studies did not report the number of HAE attacks, five studies (52 patients) were finally included in the summary analysis. Four classes of drugs with at least one controlled trial have been proposed for HAE prophylaxis. All those drugs, except heparin, were found to be more effective than placebo. In the absence of direct comparisons, the relative efficacies of these drugs were determined by calculating a RR of attacks (drug vs placebo). The results were as follows: danazol (RR=0.023, 95% CI 0.003 to 0.162), methyltestosterone (RR=0.054, 95% CI 0.013 to 0.163), ε-aminocaproic acid (RR=0.095, 95% CI 0.025 to 0.356), tranexamic acid (RR=0.308, 95% CI 0.195 to 0.479) and C1-INH 0.491 (95% CI 0.395 to 0.607).

Conclusions: Few trials have evaluated the benefits of HAE prophylaxis, and all drugs but heparin seem to be effective in this setting. Since there are no direct comparisons of HAE drugs, it was not possible to draw definitive conclusions on the most effective one. Thus, to accumulate evidence for HAE prophylaxis, further studies are needed that consider the dose-efficacy relationship and include a head-to-head comparison between drugs, with the active group, rather than placebo, as the control.

No MeSH data available.


Related in: MedlinePlus