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      Botulinum toxin as treatment for focal dystonia: a systematic review of the pharmaco-therapeutic and pharmaco-economic value

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          Abstract

          Focal dystonia is a common, invalidating neurologic condition characterized by involuntary, sustained muscle contractions causing twisting movements and abnormal postures in one body part. Currently, botulinum toxin is the treatment of first choice. We performed a systematic review towards the pharmaco-therapeutic and pharmaco-economic value of botulinum toxin as treatment for focal dystonia, which yielded the following results. Botulinum toxin is the most effective treatment for reducing dystonic symptoms measured with dystonia-specific and general questionnaires, and pain in patients with focal dystonia. Seventy-one percent of patients with cervical dystonia had a reduction in neck pain compared to 12 % in placebo groups. Adverse events occur in 58 % of patients during treatment with botulinum toxin compared to 46 % treated with placebo. Especially dry mouth, neck weakness, dysphagia, and voice changes are common. Adverse events are usually mild and self-limiting. Health-related quality of life, measured with the SF-36 is 20–50 points lower in patients with focal dystonia compared to controls and the effect of botulinum toxin on health-related quality of life is unclear. Botulinum toxin treatment is expensive because the drug itself is expensive. Yearly costs for treating a patient with focal dystonia with botulinum toxin range from EUR 347 to EUR 3,633 and the gain in QALYs with BTX treatment is small. Focal dystonia impairs the productivity and the ability to work. At start of botulinum toxin treatment only 47–50 % was working. Botulinum toxin partly improves this. Overall, we conclude that botulinum toxin is an expensive drug with good effects. From a societal perspective, the costs may well weigh up to the regained quality of life. However, the available literature concerning costs, health-related quality of life and labor participation is very limited. An extensive cost-effectiveness study should be performed incorporating all these aspects.

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          The online version of this article (doi:10.1007/s00415-012-6510-x) contains supplementary material, which is available to authorized users.

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          How common is the most common adult movement disorder? Update on the worldwide prevalence of essential tremor.

          Elan Louis, Joaquim Ferreira (2010)
          Essential tremor (ET) is among the more prevalent neurological disorders, yet prevalence estimates have varied enormously, making it difficult to establish prevalence with precision. We: (1) reviewed the worldwide prevalence of ET in population-based epidemiological studies, (2) derived as precisely as possible an estimate of disease prevalence, and (3) examined trends and important differences across studies. We identified 28 population-based prevalence studies (19 countries). In a meta-analysis, pooled prevalence (all ages) = 0.9%, with statistically significant heterogeneity across studies (I(2) = 99%, P or= 65 years) = 4.6%, and in additional descriptive analyses, median crude prevalence (age >or= 60-65) = 6.3%. In one study of those age >or= 95 years, crude prevalence = 21.7%. Several studies reported ethnic differences in prevalence, although more studies are needed. Greater than one-third of studies show a gender difference, with most demonstrating a higher prevalence among men. This possible gender preference is interesting given clinical, epidemiological, and pathological associations between ET and Parkinson's disease. Precise prevalence estimates such as those we provide are important because they form the numerical basis for planned public health initiatives, provide data on the background occurrence of disease for family studies, and offer clues about the existence of environmental or underlying biological factors of possible mechanistic importance. (c) 2010 Movement Disorder Society.
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            Prevalence and incidence of Parkinson's disease in Europe.

            Kai Bötzel, Sonja von Serényi, Mark R Wick … (2005)
            To provide an overview on the prevalence and incidence of Parkinson's disease (PD) in selected European countries. PD is a common disease of unknown etiology. Accurate information on the epidemiology of PD is critical to inform health policy. An aging population will lead to more patients with PD; thus, the high financial burden PD places on society will increase. A systematic literature search was performed to identify studies on the prevalence and incidence of PD in the following European countries: Austria, the Czech Republic, France, Germany, Italy, The Netherlands, Portugal, Spain, Sweden and United Kingdom. Only published studies were included. Abstracts, reviews, meta-analyses and letters to the editor were excluded. There were no language restrictions. Data were extracted using a standardized assessment form, and evidence tables were used to systematically report and compare the data. Of 39 identified studies, most (87%) reported estimates of PD prevalence rates, while only a few (13%) reported estimates of PD annual incidence rates. Crude prevalence rate estimates ranged from 65.6 per 100,000 to 12,500 per 100,000 and annual incidence estimates ranged from 5 per 100,000 to 346 per 100,000. No publications could be identified for Austria or the Czech Republic. The observed variations in prevalence and incidence rates may result from environmental or genetic factors, but might also be a consequence of differences in methodologies for case ascertainment, diagnostic criteria, or age distributions of the study populations. The comparability of existing studies is limited.
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              A prevalence study of primary dystonia in eight European countries.

              Laura Camfield (2000)
              There have been few epidemiological studies of dystonia. Most previous studies have provided estimates based on few cases. A European prevalence study was undertaken to provide more precise rates of dystonia by pooling data from eight European countries. Diagnosed cases were ascertained by adult neurologists with specialist movement disorder (and botulinum toxin) clinics. The crude annual period prevalence rate (1996-1997) for primary dystonia was 152 per million (95% confidence interval 142-162), with focal dystonia having the highest rate of 117 per million (108-126). Prevalence rates for cervical dystonia, blepharospasm and writer's cramp were as follows: 57 (95% confidence interval 51-63), 36 (31-41), and 14 (11-17). The age-adjusted relative rates were significantly higher in women than in men for segmental and focal dystonias with the exception of writer's cramp. Comparing rates between centres demonstrated significant variations for cervical dystonia, blepharospasm and writer's cramp, probably due to methodological differences. Our results provide the first data on the prevalence of primary dystonia and its subtypes across several European countries. Due to under-ascertainment of cases, our rates should be seen as conservative and an under-estimate of the true prevalence of dystonia.
              Article 0 comments Cited 57 times – based on 0 reviews     Review now
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                Author and article information

                Contributors
                : +31-50-3612400 , +31-50-3611707 , m.a.j.de.Koning-Tijssen@umcg.nl
                Journal
                Journal ID (nlm-ta): J Neurol
                Journal ID (iso-abbrev): J. Neurol
                Title: Journal of Neurology
                Publisher: Springer-Verlag (Berlin/Heidelberg )
                ISSN (Print): 0340-5354
                ISSN (Electronic): 1432-1459
                Publication date (Electronic): 3 May 2012
                Publication date PMC-release: 3 May 2012
                Publication date (Print): December 2012
                Volume: 259
                Issue: 12
                Pages: 2519-2526
                Affiliations
                [1 ]Department of Neurology, Academic Medical Centre, Amsterdam, The Netherlands
                [2 ]Clinical Research Unit, Academic Medical Centre, Amsterdam, The Netherlands
                [3 ]Movement Disorders, Department of Neurology AB 51, University Medical Centre Groningen (UMCG), PO Box 30.001, 9700 Groningen, The Netherlands
                Article
                Publisher ID: 6510
                DOI: 10.1007/s00415-012-6510-x
                PMC ID: 3506193
                PubMed ID: 22552527
                SO-VID: 220e4798-c6e9-4eca-96ec-dbebcf77dcfb
                Copyright © © The Author(s) 2012
                History
                Date received : 3 February 2012
                Date revision received : 30 March 2012
                Date accepted : 2 April 2012
                Categories
                Subject: Review
                Custom metadata
                issue-copyright-statement © Springer-Verlag Berlin Heidelberg 2012

                ScienceOpen disciplines: Neurology
                Keywords: botulinum toxin,focal dystonia,effectiveness
                Data availability:
                ScienceOpen disciplines: Neurology
                Keywords: botulinum toxin, focal dystonia, effectiveness

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