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August 18, 2020; 95 (7) Article

Temporal trends and autopsy findings of SUDEP based on medico-legal investigations in the United States

View ORCID ProfileEsma Cihan, View ORCID ProfileOrrin Devinsky, View ORCID ProfileDale C. Hesdorffer, Michael Brandsoy, Ling Li, David R. Fowler, Jason K. Graham, Michael W. Karlovich, Jaclyn E. Yang, Anne E. Keller, Elizabeth J. Donner, View ORCID ProfileDaniel Friedman
First published July 7, 2020, DOI: https://doi.org/10.1212/WNL.0000000000009996
Esma Cihan
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Orrin Devinsky
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Dale C. Hesdorffer
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Michael Brandsoy
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Ling Li
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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David R. Fowler
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Jason K. Graham
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Michael W. Karlovich
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Jaclyn E. Yang
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Anne E. Keller
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Elizabeth J. Donner
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Daniel Friedman
From the Department of Neurology (E.C., O.D., M.W.K., J.E.Y., D.F.), NYU School of Medicine; Department of Epidemiology (D.C.H.), Columbia University Medical Center, New York, NY; San Diego County Medical Examiner's Office (M.B.), CA; Maryland Office of the Chief Medical Examiner (L.L., D.R.F.), Baltimore; New York City Office of Chief Medical Examiner (J.K.G.), New York; and Division of Neurology, Department of Paediatrics (A.E.K., E.J.D.), the Hospital for Sick Children, Toronto, Canada.
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Temporal trends and autopsy findings of SUDEP based on medico-legal investigations in the United States
Esma Cihan, Orrin Devinsky, Dale C. Hesdorffer, Michael Brandsoy, Ling Li, David R. Fowler, Jason K. Graham, Michael W. Karlovich, Jaclyn E. Yang, Anne E. Keller, Elizabeth J. Donner, Daniel Friedman
Neurology Aug 2020, 95 (7) e867-e877; DOI: 10.1212/WNL.0000000000009996

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Abstract

Objective To determine time trends and distinguishing autopsy findings of sudden unexpected death in epilepsy (SUDEP) in the United States.

Methods We identified decedents where epilepsy/seizure was listed as cause/contributor to death or comorbid condition on the death certificate among all decedents who underwent medico-legal investigation at 3 medical examiner (ME) offices across the country: New York City (2009–2016), San Diego County (2008–2016), and Maryland (2000–2016). After reviewing all available reports, deaths classified as definite/probable/near SUDEP or SUDEP plus were included for analysis. Mann-Kendall trend test was used to analyze temporal trends in SUDEP rate for 2009–2016. Definite SUDEPs were compared to sex- and age ±2 years–matched non-SUDEP deaths with a history of epilepsy regarding autopsy findings, circumstances, and comorbidities.

Results A total of 1,086 SUDEP cases were identified. There was a decreasing trend in ME-investigated SUDEP incidence between 2009 and 2016 (z = −2.2, S = −42, p = 0.028) among 3 regions. There was a 28% reduction in ME-investigated SUDEP incidence from 2009 to 2012 to 2013–2016 (confidence interval, 17%–38%, p < 0.0001). We found no correlation between SUDEP rates and the month of year or day of week. There was no difference between SUDEP and non-SUDEP deaths regarding neurodevelopmental abnormalities, pulmonary congestion/edema, and myocardial fibrosis.

Conclusions There was a decreasing monotonic trend in ME-investigated SUDEP incidence over 8 years, with a 28% reduction in incidence from 2009–2012 to 2013–2016. Unlike SIDS and sudden cardiac death, we found no correlation between SUDEP and the season of year or day of week. No autopsy findings distinguished SUDEP from non-SUDEP deaths.

Glossary

CI=
confidence interval;
MD=
Maryland;
ME=
medical examiner;
MLI=
medico-legal investigation;
NYC=
New York City;
OCME=
Office of Chief Medical Examiner;
PWE=
people with epilepsy;
SDC=
San Diego County;
SUDEP=
sudden unexpected death in epilepsy

Footnotes

  • Go to Neurology.org/N for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Received June 10, 2019.
  • Accepted in final form January 28, 2020.
  • © 2020 American Academy of Neurology
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Letters: Rapid online correspondence

  • Author response: Temporal trends and autopsy findings of SUDEP based on medico-legal investigations in the US
    • Daniel Friedman, Neurologist, NYU Grossman School of Medicine
    • Esma Cihan, Fellow, NYU Grossman School of Medicine
    • Orrin Devinsky, Neurologist, NYU Grossman School of Medicine
    Submitted August 07, 2020
  • Reader response: Temporal trends and autopsy findings of SUDEP based on medico-legal investigations in the US
    • Andrew Bleasel, Neurologist, Westmead Hospital, University of Sydney (Sydney, Australia)
    Submitted August 06, 2020
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