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Global epidemiology of Duchenne muscular dystrophy: an updated systematic review and meta-analysis

Orphanet journal of rare diseases, 2020-06, Vol.15 (1), p.1-141, Article 141 [Peer Reviewed Journal]

COPYRIGHT 2020 BioMed Central Ltd. ;2020. This work is licensed under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License. ;Attribution ;The Author(s) 2020 ;ISSN: 1750-1172 ;EISSN: 1750-1172 ;DOI: 10.1186/s13023-020-01430-8 ;PMID: 32503598

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  • Title:
    Global epidemiology of Duchenne muscular dystrophy: an updated systematic review and meta-analysis
  • Author: Crisafulli, Salvatore ; Sultana, Janet ; Fontana, Andrea ; Salvo, Francesco ; Messina, Sonia ; Trifirò, Gianluca
  • Subjects: Analysis ; Bibliographic data bases ; Birth ; Birth prevalence ; Congenital diseases ; Decision making ; Disease ; Duchenne muscular dystrophy ; Duchenne's muscular dystrophy ; Dystrophin ; Epidemiology ; Estimates ; Genetic aspects ; Human health and pathology ; Licenses ; Life Sciences ; Literature reviews ; Medical research ; Meta-analysis ; Mortality ; Muscular dystrophy ; Mutation ; Population ; Prevalence ; Prevalence studies (Epidemiology) ; Public health ; Quality ; Rare diseases ; Review ; Santé publique et épidémiologie ; Studies ; Systematic review ; Utrophin
  • Is Part Of: Orphanet journal of rare diseases, 2020-06, Vol.15 (1), p.1-141, Article 141
  • Description: Abstract Background Duchenne Muscular Dystrophy (DMD) is a rare disorder caused by mutations in the dystrophin gene. A recent systematic review and meta-analysis of global DMD epidemiology is not available. This study aimed to estimate the global overall and birth prevalence of DMD through an updated systematic review of the literature. Methods MEDLINE and EMBASE databases were searched for original research articles on the epidemiology of DMD from inception until 1st October 2019. Studies were included if they were original observational research articles written in English, reporting DMD prevalence and/or incidence along with the number of individuals of the underlying population. The quality of the studies was assessed using a STrengthening the Reporting of OBservational studies in Epidemiology (STROBE) checklist adapted for observational studies on rare diseases. To derive the pooled epidemiological prevalence estimates, a meta-analysis was performed using random-effects logistic models for overall and birth prevalence and within two different underlying populations (i.e. all individuals and in males only), separately. Heterogeneity was assessed using Cochran’s Q-test along with its derived measure of inconsistency I 2 . Results A total of 44 studies reporting the global epidemiology of DMD were included in the systematic review and only 40 were included in the meta-analysis. The pooled global DMD prevalence was 7.1 cases (95% CI: 5.0–10.1) per 100,000 males and 2.8 cases (95% CI: 1.6–4.6) per 100,000 in the general population, while the pooled global DMD birth prevalence was 19.8 (95% CI:16.6–23.6) per 100,000 live male births. A very high between-study heterogeneity was found for each epidemiological outcome and for all underlying populations (I 2  > 90%). The test for funnel plot asymmetry suggested the absence of publication bias. Of the 44 studies included in this systematic review, 36 (81.8%) were assessed as being of medium and 8 (18.2%) of low quality, while no study was assessed as being of high quality. Conclusions Generating epidemiological evidence on DMD is fundamental to support public health decision-making. The high heterogeneity and the lack of high quality studies highlights the need to conduct better quality studies on rare diseases.
  • Publisher: London: BioMed Central Ltd
  • Language: English
  • Identifier: ISSN: 1750-1172
    EISSN: 1750-1172
    DOI: 10.1186/s13023-020-01430-8
    PMID: 32503598
  • Source: Freely Accessible Journals
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