Abstract
The purpose of this study was to determine the accuracy of death certification for amyotrophic lateral sclerosis (ALS)/motor neuron disease (MND) as recorded by the Central Statistics Office in the Republic of Ireland, and to examine its utility in capture-recapture analysis. The database of the Irish Central Statistics Office (CSO) was searched for death certificates of individuals over 15 years of age for whom ALS/MND (ICD9) was listed as a primary, secondary or tertiary cause of death from 2002 to 2006. This dataset was compared with mortality data from the Irish Register for ALS/MND for the same period. Three hundred ninety-eight cases with a diagnosis of ALS were identified through the CSO. The cause of death as identified by the CSO was 318 out of 398 (80%) cases known to the Register during the study period. ALS/MND was listed as a cause of death by the CSO in 79 (20%) cases that were unknown to the ALS/MND Register. Of these, ALS/MND was listed as a secondary/tertiary cause of death in 76, and 75% of all such deaths occurred in rural areas. Capture-recapture analysis using the CSO and ALS Register as independent datasets generated a likely over-estimation of the crude incidence of ALS from 2.1/100,000 to 3.45/100,000 in Ireland. Population-based registers are most accurate when ascertaining from a wide network of inter-dependent sources. Death certification for ALS/MND lacks both sensitivity and specificity. Capture-recapture exercises using such data sources are likely to over-estimate the true incidence of disease.
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References
Phukan J, Hardiman O (2009) The management of amyotrophic lateral sclerosis. J Neurol 256(2):176–186 (Epub 2009 Feb 17)
Cronin S, Hardiman O, Traynor BJ (2007) Ethnic variation in the incidence of ALS: a systematic review. Neurology 68(13):1002–1007
Zaldivar T, Gutierrez J, Lara G, Carbonara M, Logroscino G, Hardiman O (2009) Reduced frequency of ALS in an ethnically mixed population: a population-based mortality study. Neurology 72(19):1640–1645
Sejvar JJ, Holman RC, Bresee JS, Kochanek KD, Schonberger LB (2005) Amyotrophic lateral sclerosis mortality in the United States, 1979–2001. Neuroepidemiology 25(3):144–152
Ragonese P, Filippini G, Salemi G, Beghi E, Citterio A, D’Alessandro R, Marini C, Monsurrò MR, Aiello I, Morgante L, Tempestini A, Fratti C, Ragno M, Pugliatti M, Epifanio A, Testa D, Savettieri G (2004) Accuracy of death certificates for amyotrophic lateral sclerosis varies significantly from north to south of Italy: implications for mortality studies. Neuroepidemiology 23(1–2):73–77
Traynor BJ, Codd MB, Corr B, Forde C, Frost E, Hardiman O (1999) Incidence and prevalence of ALS in Ireland, 1995–1997: a population-based study. Neurology 52(3):504–509
O’Toole O, Traynor BJ, Brennan P, Sheehan C, Frost E, Corr B et al (2008) Epidemiology and clinical features of amyotrophic lateral sclerosis in Ireland between 1995 and 2004. J Neurol Neurosurg Psychiatry 79:30–32
Beghi E, Logroscino G, Chio’ A, Hardiman O, Mitchell D, Swingler R et al (2006) The epidemiology of ALS and the role of population-based registries. Biochim Biophys Acta 1762:1150–1157
Logroscino G, Traynor BJ, Hardiman O, Chiò A, Mitchell D, Swingler RJ, Millul A, Benn E, Beghi E (2009) Incidence of amyotrophic lateral sclerosis in Europe. J Neurol Neurosurg Psychiatry (Epub ahead of print)
Hoffman PM, Brody JA (1971) The reliability of death certificate reporting fromamyotrophic lateral sclerosis. J Chron Dis 24:5–8
O’Malley F, Dean G, Elian M (1987) Multiple sclerosis and motor neurone disease: survival and how certified after death. J Epidemiol Community Health 41:14–17
Chio’ A, Magnani C, Oddenino E, Tolardo G, Schiffer D (1992) Accuracy of death certificate diagnosis of amyotrophic lateral sclerosis. J Epidemiol Community Health 46:517–518
Chancellor AM, Swingler RJ, Fraser J, Clarke JA, Warlow CP (1993) Utility of Scottish morbidity and mortality data for epidemiological studies of motor neurone. J Epidemiol Community Health 47:116–120
Forbes RB, Colville S, Swingler RJ (2004) Scottish ALS/MND Register: the epidemiology of amyotrophic lateral sclerosis (ALS/MND) in people aged 80 or over. Age Ageing 33(2):131–134
Kondo K, Tsubaki T (1977) Changing mortality patterns of motor neurone disease in Japan. J Neurol Sci 32:411–424
Forbes RB, Colville S, Parratt J, Swingler RJ (2007) The incidence of motor neuron disease in Scotland. J Neurol 254(7):866–869 (Epub 2007 Apr 10)
Hook EB, Regal RR (1995) Capture-recapture methods in epidemiology: methods and limitations. Epidemiol Rev 17:243–264
Tilling K (2001) Capture-recapture methods-useful or misleading? Int J Epidemiol 30:12–14
Acknowledgments
This study was sponsored in part by the Irish Motor Neuron Disease Research Foundation, and by the Royal College of Surgeons Summer Student Program. Prof. Hardiman is a HRB Clinician Scientist (HRB CSA 2007/3). She has received unrestricted research grant support from Bayer-Schering Pharma, Merck-Serono, and Sanofi-Aventis. She has served on advisory boards for Biogen Idec, Ono Pharmaceuticals, CytRx and Janssen-Cilag. She has received honoraria from Janssen-Cilag, Merck Serono, Ono Pharmaceuticals, CytRx and Biogen Idec. She is also funded by the Irish Health Research Board (CSA/2007/3).
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Yeo, L., Lynch, C. & Hardiman, O. Validating population-based registers for ALS: how accurate is death certification?. J Neurol 257, 1235–1239 (2010). https://doi.org/10.1007/s00415-010-5494-7
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DOI: https://doi.org/10.1007/s00415-010-5494-7