A systematic review and meta-analysis on the epidemiology of Duchenne and Becker muscular dystrophy |
| |
| Affiliation: | 2. Laboratory of Neurogenetics and Neuroscience, Istituto G.Gaslini, Genoa, Italy;3. Centre of Myology and Neurodegenerative Disorders, Istituto G.Gaslini, Genoa, Italy;4. DINOGMI, University of Genoa, Genoa, Italy;5. Molecular Medicine Unit, Fondazione IRCCS Stella Maris, Pisa, Italy;1. Genetica y Biologia Molecular, Facultad de Farmacia y Bioquimica, Universidad de Buenos Aires, Argentina;2. Laboratorio de Genetica Molecular de Hemofilia, Instituto de Medicina Experimental IMEX, CONICET-Academia Nacional de Medicina, Buenos Aires, Argentina;3. Laboratorio de Genetica Molecular Diagnostica (GENOS S.A.), Buenos Aires, Argentina;1. Division of Child Neurology, Department of Neurology, National Hospital Organization Toneyama National Hospital, Toyonaka, Osaka, Japan;2. Department of Neurology, National Hospital Organization Higashisaitama Hospital, Hasuda, Saitama, Japan;3. National Center of Neurology and Psychiatry, Kodaira, Tokyo, Japan;4. Department of Neurology, National Hospital Organization Sendai-Nishitaga National Hospital, Sendai, Miyagi, Japan;5. Department of Neurology, National Hospital Organization National Akita Hospital, Yurihonjo, Akita, Japan;6. Department of Neurology, National Hospital Organization National Aomori Hospital, Aomori, Japan;7. Department of Neurology, National Hospital Organization National Suzuka Hospital, Suzuka, Mie, Japan;8. Department of Neurology, National Hospital Organization National Shimoshizu Hospital, Yotsukaido, Chiba, Japan;9. Department of Neurology, National Hospital Organization Toneyama National Hospital, Toyonaka, Osaka, Japan |
| |
| Abstract: | The muscular dystrophies are a broad group of hereditary muscle diseases with variable severity. Population-based prevalence estimates have been reported but pooled estimates are not available. We performed a systematic review of worldwide population-based studies reporting muscular dystrophies prevalence and/or incidence using MEDLINE and EMBASE databases. The search strategy included key terms related to muscular dystrophies, incidence, prevalence and epidemiology. Two reviewers independently reviewed all abstracts, full text articles and abstracted data using standardized forms. Pooling of prevalence estimates was performed using random effect models. 1104 abstracts and 167 full text articles were reviewed. Thirty-one studies met all eligibility criteria and were included in the final analysis. The studies differed widely in their approaches to case ascertainment, resulting in significant methodological heterogeneity and varied data quality. The pooled prevalence of DMD and BMD was 4.78 (95% CI 1.94–11.81) and 1.53 (95% CI 0.26–8.94) per 100,000 males respectively. The incidence of DMD ranged from 10.71 to 27.78 per 100,000. This is the first meta-analysis of worldwide prevalence estimates for muscular dystrophies. There is a need for more epidemiological studies addressing global estimates on incidence and prevalence of muscular dystrophies, utilizing standardized diagnostic criteria as well as multiple sources of case ascertainment. |
| |
| Keywords: | Incidence Prevalence Epidemiology Muscular dystrophy Population-based |
| 本文献已被 ScienceDirect 等数据库收录！ |
|
