Top

Published in:

01-08-2021 | Amyotrophic Lateral Sclerosis | Original Communication

Time-trend evolution and determinants of sex ratio in Amyotrophic Lateral Sclerosis: a dose–response meta-analysis

Authors: Andrea Fontana, Benoit Marin, Jaime Luna, Ettore Beghi, Giancarlo Logroscino, Farid Boumédiene, Pierre-Marie Preux, Philippe Couratier, Massimilano Copetti

Published in: Journal of Neurology | Issue 8/2021

Abstract

Background

A noticeable change of the male-to-female sex ratio (SR) has been observed in Amyotrophic Lateral Sclerosis (ALS) leading to an apparent regression of SR with time (SR close to 1:1).

Objective

To provide a global SR estimate and investigate its relation with respect to population age.

Methods

A systematic review and meta-analysis was conducted including only population-based studies with a high-quality methodology in European ancestral origin population. Male-to-female SR was estimated by three different measures: SR number, SR crude incidence and SR standardized incidence. Standard and dose–response meta-analyses were performed to assess the pooled SR measures (irrespective of population age) and the evolution of the SR measures with respect to population age, respectively. Potential sources of heterogeneity were investigated via meta-regression.

Results

Overall, 3254 articles were retrieved in the literature search. Thirty-nine studies stratified by time periods were included. The overall pooled male-to-female ratio was 1.28 (95% CI 1.23–1.32) for SR number, 1.33 (95% CI 1.29–1.38) for SR crude incidence and 1.35 (95% CI 1.31–1.40) for SR standardized incidence. The SR number with respect to population age reveals a progressive reduction of SR at increasing age, while the SR crude incidence in relation to age displays a U-shaped curve.

Conclusions

The number and the incidence of ALS cases were consistently higher in males than females. Dose–response meta-analysis showed that SR measures change with respect to population age. Further original research is needed to clarify if our findings are reproducible in other populations.

Available only for authorised users

Beghi E, Logroscino G, Chiò A et al (2006) The epidemiology of ALS and the role of population-based registries. Biochim Biophys Acta 1762:1150–1157. https://doi.org/10.1016/j.bbadis.2006.09.008CrossRefPubMed

Logroscino G, Traynor BJ, Hardiman O et al (2008) Descriptive epidemiology of amyotrophic lateral sclerosis: new evidence and unsolved issues. J Neurol Neurosurg Psychiatry 79:6–11. https://doi.org/10.1136/jnnp.2006.104828CrossRefPubMed

Luna J, Logroscino G, Couratier P, Marin B (2017) Current issues in ALS epidemiology: variation of ALS occurrence between populations and physical activity as a risk factor. Rev Neurol (Paris) 173:244–253. https://doi.org/10.1016/j.neurol.2017.03.035CrossRef

Chiò A, Mora G, Moglia C et al (2017) Secular trends of amyotrophic lateral sclerosis: the piemonte and valle d’Aosta register. JAMA Neurol 74:1097–1104. https://doi.org/10.1001/jamaneurol.2017.1387CrossRefPubMedPubMedCentral

Marin B, Boumédiene F, Logroscino G et al (2017) Variation in worldwide incidence of amyotrophic lateral sclerosis: a meta-analysis. Int J Epidemiol 46:57–74. https://doi.org/10.1093/ije/dyw061CrossRefPubMed

Marin B, Logroscino G, Boumédiene F et al (2016) Clinical and demographic factors and outcome of amyotrophic lateral sclerosis in relation to population ancestral origin. Eur J Epidemiol 31:229–245. https://doi.org/10.1007/s10654-015-0090-xCrossRefPubMed

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

Stroup DF, Berlin JA, Morton SC et al (2000) Meta-analysis of observational studies in epidemiology: a proposal for reporting. Meta-analysis Of Observational Studies in Epidemiology (MOOSE) group. JAMA 283:2008–2012. https://doi.org/10.1001/jama.283.15.2008CrossRefPubMed

Fiest KM, Pringsheim T, Patten SB et al (2014) The role of systematic reviews and meta-analyses of incidence and prevalence studies in neuroepidemiology. Neuroepidemiology 42:16–24. https://doi.org/10.1159/000355533CrossRefPubMed

10.

Mitsumoto H, Chad DA, Pioro EP (1998) Amyotrophic lateral sclerosis. Oxford University Press, Oxford

11.

Szklo M (1998) Population-based cohort studies. Epidemiol Rev 20:81–90. https://doi.org/10.1093/oxfordjournals.epirev.a017974CrossRefPubMed

12.

(2000) Census, race and science. Nat Genet 24:97–98. https://doi.org/10.1038/72884

13.

United Nations Statistics Division Composition of macro geographical (continental) regions, geographical sub-regions, and selected economic and other groupings. https://unstats.un.org/unsd/methodology/m49/

14.

Sorenson EJ, Stalker AP, Kurland LT, Windebank AJ (2002) Amyotrophic lateral sclerosis in Olmsted County, Minnesota, 1925 to 1998. Neurology 59:280–282. https://doi.org/10.1212/wnl.59.2.280CrossRefPubMed

15.

United Nations Demographic yearbook. DYB annual issues. https://unstats.un.org/unsd/demographic-social/products/dyb/index.cshtml

16.

Greenland S (1987) Quantitative methods in the review of epidemiologic literature. Epidemiol Rev 9:1–30. https://doi.org/10.1093/oxfordjournals.epirev.a036298CrossRefPubMed

17.

van Houwelingen HC, Arends LR, Stijnen T (2002) Advanced methods in meta-analysis: multivariate approach and meta-regression. Stat Med 21:589–624. https://doi.org/10.1002/sim.1040CrossRefPubMed

18.

Higgins J, Thomas J (2011) Cochrane Handbook for Systematic Reviews of Interventions. The Cochrane Collaboration

19.

Manjaly ZR, Scott KM, Abhinav K et al (2010) The sex ratio in amyotrophic lateral sclerosis: a population based study. Amyotroph Lateral Scler Off Publ World Fed Neurol Res Group Mot Neuron Dis 11:439–442. https://doi.org/10.3109/17482961003610853CrossRef

20.

Trojsi F, D’Alvano G, Bonavita S, Tedeschi G (2020) Genetics and sex in the pathogenesis of amyotrophic lateral sclerosis (ALS): is there a link? Int J Mol Sci. https://doi.org/10.3390/ijms21103647CrossRefPubMedPubMedCentral

21.

Deeks JJ, Higgins JP, Douglas DG, on behalf of the Cochrane Statistical Methods Group (2019) Chapter 10: analysing data and undertaking meta-analyses. In: Cochrane Handbook for Systematic Reviews of Interventions, 2nd Edition. John Wiley & Sons, Chichester (UK)

22.

Radhakrishnan K, Ashok PP, Sridharan R, Mousa ME (1986) Descriptive epidemiology of motor neuron disease in Benghazi, Libya. Neuroepidemiology 5:47–54. https://doi.org/10.1159/000110812CrossRefPubMed

23.

Sajjadi M, Etemadifar M, Nemati A et al (2010) Epidemiology of amyotrophic lateral sclerosis in Isfahan. Iran Eur J Neurol 17:984–989. https://doi.org/10.1111/j.1468-1331.2010.02972.xCrossRefPubMed

24.

Ramanathan RS, Rana S (2018) Demographics and clinical characteristics of primary lateral sclerosis: case series and a review of literature. Neurodegener Dis Manag 8:17–23. https://doi.org/10.2217/nmt-2017-0051CrossRefPubMed

25.

Wijesekera LC, Mathers S, Talman P et al (2009) Natural history and clinical features of the flail arm and flail leg ALS variants. Neurology 72:1087–1094. https://doi.org/10.1212/01.wnl.0000345041.83406.a2CrossRefPubMedPubMedCentral

26.

Logroscino G, Marin B, Piccininni M et al (2018) Referral bias in ALS epidemiological studies. PLoS ONE 13:e0195821. https://doi.org/10.1371/journal.pone.0195821CrossRefPubMedPubMedCentral

27.

Finegan E, Chipika RH, Li Hi Shing S et al (2019) The clinical and radiological profile of primary lateral sclerosis: a population-based study. J Neurol 266:2718–2733. https://doi.org/10.1007/s00415-019-09473-zCrossRefPubMed

28.

McCombe PA, Henderson RD (2010) Effects of gender in amyotrophic lateral sclerosis. Gend Med 7:557–570. https://doi.org/10.1016/j.genm.2010.11.010CrossRefPubMed

29.

Alonso A, Logroscino G, Jick SS, Hernán MA (2010) Association of smoking with amyotrophic lateral sclerosis risk and survival in men and women: a prospective study. BMC Neurol 10:6. https://doi.org/10.1186/1471-2377-10-6CrossRefPubMedPubMedCentral

30.

Weisskopf MG, McCullough ML, Calle EE et al (2004) Prospective study of cigarette smoking and amyotrophic lateral sclerosis. Am J Epidemiol 160:26–33. https://doi.org/10.1093/aje/kwh179CrossRefPubMed

31.

Chiò A, Moglia C, Canosa A et al (2020) ALS phenotype is influenced by age, sex, and genetics: a population-based study. Neurology 94:e802–e810. https://doi.org/10.1212/WNL.0000000000008869CrossRefPubMed

Title: Time-trend evolution and determinants of sex ratio in Amyotrophic Lateral Sclerosis: a dose–response meta-analysis
Authors: Andrea Fontana
Benoit Marin
Jaime Luna
Ettore Beghi
Giancarlo Logroscino
Farid Boumédiene
Pierre-Marie Preux
Philippe Couratier
Massimilano Copetti
Publication date: 01-08-2021
Publisher: Springer Berlin Heidelberg
Keyword: Amyotrophic Lateral Sclerosis
Published in: Journal of Neurology / Issue 8/2021
Print ISSN: 0340-5354
Electronic ISSN: 1432-1459
DOI: https://doi.org/10.1007/s00415-021-10464-2

2024 ASCO Annual Meeting

Springer Medicine

Time-trend evolution and determinants of sex ratio in Amyotrophic Lateral Sclerosis: a dose–response meta-analysis

Abstract

Background

Objective

Methods

Results

Conclusions

2024 ASCO Annual Meeting

Springer Medicine

Abstract

Background

Objective

Methods

Results

Conclusions

Please log in to get access to this content

Other articles of this Issue 8/2021

Changes of clinical, neurophysiological and nerve ultrasound characteristics in CIDP over time: a 3-year follow-up

DRPLA: understanding the natural history and developing biomarkers to accelerate therapeutic trials in a globally rare repeat expansion disorder

Adjuvant chemotherapy after severe myelotoxicity during chemoradiation phase in malignant gliomas. Is it feasibile? Results from AINO study (Italian Association for Neuro-Oncology)

Quantitative analysis of dysautonomia in patients with autonomic dysreflexia

Critical illness-associated cerebral microbleeds for patients with severe COVID-19: etiologic hypotheses

Correction to: Heritable and non-heritable uncommon causes of stroke