Self-Reported Medication Adherence Measured with Morisky Scales in Rare Disease Patients: A Systematic Review and Meta-Analysis

Abstract

Background: The visibility of Rare Diseases is a new challenge for society. These diseases are numerous, heterogeneous in nature and distribution, characterized by a high mortality rate but low prevalence, and usually presenting a severe evolution. Adherence to medication studies in rare diseases are uncommon, due to treatment scarcity. Objectives: The main purpose of this study is to do a meta-analysis, evaluating the level of adherence to medication in the most prevalent rare diseases. Methods: This work is a systematic review, and meta-analysis was registered in the International Prospective Register of Systematic Reviews (PROSPERO) (Registration number: CRD42022372843) and conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. Adherence to treatment in this systematic review and meta-analysis was collected from all studies included, based on the crude numerators and denominators reported, using either the Morisky Medication Adherence Scale 4 or -8. Results: A total of 54 records were identified through database searches, or after screening relevant manuscripts’ references. Finally, 18 studies were included in this systematic review and meta-analysis. A total of 1559 participants (54.18% women) aged less than 84 years old were included. Twelve studies used the MMAS-8. In 8 of them, they established the level of adherence to treatment in three categories (low, medium, and high), with the mean prevalence in each of them being 41.4%, 30.4%, and 28.2%, respectively. Conclusions: The results observed in adherence to treatment in patients with rare diseases show great variability, due to the different aspects involved in the greater or lesser applicability of the medication.

Is meta-analysis the "top of the evidence pyramid" in cardiology?

“…При некоторых, достаточно редко встречающихся заболеваниях, даже проведение РКИ не всегда возможно. В этих случаях метаанализы, объединяющие результаты небольших исследований, являются практически единственным способом получить данные об истинном эффекте вмешательства [17], причем далеко не всегда эти исследования будут рандомизированными [18]. Правда, в этом случае обязательно возникнет вопрос об однородности методов проведения и оценки результата в этих небольших исследованиях.…”