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Age-specific ALS incidence: a dose–response meta-analysis

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Abstract

To evaluate the association between worldwide ALS incidence rates and age, using a dose–response meta-analysis. We reviewed Medline and Embase up to July 2016 and included all population-based studies of newly-diagnosed cases, using multiple sources for case ascertainment. A dose–response meta-analysis was performed. A meta-regression investigated potential sources of heterogeneity. Of 3254 articles identified in the literature, we included 41 incidence studies covering 42 geographical areas. Overall, the fit between observed and predicted age-specific rates was very good. The expected variation of ALS incidence with age was characterized, in each study, by a progressive increase in the incidence from the 40s leading to a peak in the 60s or 70s, followed by a sharp decrease. Cochran’s Q test suggested a significant heterogeneity between studies. Overall, estimated patterns of ALS age-specific incidence (at which the peak was reached) were similar among subcontinents of Europe and North America: peak of ALS incidence ranged in these areas between 6.98 and 8.17/100,000 PYFU, which referred to age in the range 71.6–77.4 years. The relationship between age and ALS incidence appeared different for Eastern Asia which was characterized by a peak of ALS incidence at 2.20/100,000 PYFU around 75 years of age. This study confirms the consistency of the age-specific ALS incidence pattern within different subcontinents. Age-specific incidence appears lower in Eastern Asia as compared to Europe and North America.

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Abbreviations

ALS:

Amyotrophic lateral sclerosis

PDC:

Parkinson dementia complex

PD:

Parkinson’s disease

MOOSE:

Meta-analysis of observational studies in epidemiology

PYFU:

Person years of follow-up

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Acknowledgements

We thank the following main authors or co-authors of population-based articles who answered to our solicitation and for the useful material for incidence standardization or phenotype characterization that they were able to provide: Kari Murros, Poul Joensen, Raeburn Forbes, Robert Swingler (the Scottish MND Register is funded by MND Scotland and supported by the Anne Rowling Regenerative Neurology Clinic), Ibrahim Imam, James Rooney, Albert Ludolph, Gabriele Nagel, Marwa Elamin, Orla Hardiman, Mark Heverin, Mark Huisman, Joachim Wolf, Adriano Chio, Federica Pisa, Jessica Mandrioli, Monica Bandettini, Stefano Zocollela, Maura Pugliatti, Leslie Parish, Paolo Ragonese, Valerie Mc Guire, Will Longstreth, Eric J. Sorenson, Farrah Mateen, James D. Bonaparte, Cristina Vazquez, Carlos Ketzoian, Kurupath Radhakrishnan, Chien-Hsu Lai, Chung Yan G Fong, Hitoshi Okumura, Tameko Kihira, Bruce Taylor and A Lannuzel. We thank Paul Mehta, Heather Jordan and Jhaqueline Valle for providing data to calculate US incidence. The data came from surveillance projects funded by the Agency for Toxic Substances and Disease Registry’s (ATSDR) National ALS Registry [www.cdc.gov/als] (Contract #200-2009-32577 and Contract #200-2010-F-36614). We also thank Walter Rocca and Brandon R. Grossardt for the detailed data on Olmsted county population with which they provided us, and Hidenao Sasaki, Robert Miller and Eric Denys as contact persons. We thank Vanna Pistotti for her advice during the literature search, as well as Mineko Terao, Lorenzo Moja and Claudio Pelucchi. We thank Limoges teaching hospital for its grant initiative for mobility. We thank Sylvie Gautier for English editing and Emilio G Gumiel for technical support.

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eFigure 1. Flow chart eligibility criteria of articles on ALS age-specific incidence (TIFF 29883 kb)

10654_2018_392_MOESM2_ESM.pdf

eFigure 2. ALS age-specific incidences reported in each original report (/100,000PYFU, point estimates) along with the estimation of the age-specific incidence curves estimated by the random effect dose–response meta-analysis. (2.a Northern Europe, 2.b Western Europe, 2.c Southern Europe, 2.d NorthernAmerica, 2.e East Asia, 2.f Other subcontinents) (PDF 994 kb)

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Marin, B., Fontana, A., Arcuti, S. et al. Age-specific ALS incidence: a dose–response meta-analysis. Eur J Epidemiol 33, 621–634 (2018). https://doi.org/10.1007/s10654-018-0392-x

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