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February 11, 2014; 82 (6) Article

Neuralgic amyotrophy and hepatitis E virus infection

Jeroen J.J. van Eijk, Richie G. Madden, Annemiek A. van der Eijk, Jeremy G. Hunter, Johan H.J. Reimerink, Richard P. Bendall, Suzan D. Pas, Vic Ellis, Nens van Alfen, Laura Beynon, Lucy Southwell, Brendan McLean, Bart C. Jacobs, Baziel G.M. van Engelen, Harry R. Dalton
First published January 8, 2014, DOI: https://doi.org/10.1212/WNL.0000000000000112
Jeroen J.J. van Eijk
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Richie G. Madden
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Annemiek A. van der Eijk
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Jeremy G. Hunter
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Johan H.J. Reimerink
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Richard P. Bendall
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Suzan D. Pas
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Vic Ellis
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Nens van Alfen
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Laura Beynon
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Lucy Southwell
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Brendan McLean
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Bart C. Jacobs
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Baziel G.M. van Engelen
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Harry R. Dalton
From the Department of Neurology (J.J.J.v.E., N.v.A., B.G.M.v.E.), Radboud University Nijmegen Medical Centre; Department of Neurology (J.J.J.v.E.), Jeroen Bosch Hospital, Nijmegen, the Netherlands; Cornwall Gastrointestinal Unit (R.G.M., J.G.H., L.B., L.S., H.R.D.), Clinical Microbiology (R.P.B., V.E.), and Department of Neurology (B.M.), Royal Cornwall Hospital Trust; European Centre for the Environment and Human Health (R.G.M., J.G.H., R.P.B., L.B., H.R.D.), University of Exeter Medical School, Truro, UK; Centre for Infectious Disease Control (J.H.J.R.), Division Virology, National Institute for Public Health and the Environment, Bilthoven; Departments of Viroscience (A.A.v.d.E., S.D.P.), Immunology (B.C.J.), and Neurology (B.C.J.), Erasmus Medical Centre, University Medical Center Rotterdam, the Netherlands.
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Full PDF
Citation
Neuralgic amyotrophy and hepatitis E virus infection
Jeroen J.J. van Eijk, Richie G. Madden, Annemiek A. van der Eijk, Jeremy G. Hunter, Johan H.J. Reimerink, Richard P. Bendall, Suzan D. Pas, Vic Ellis, Nens van Alfen, Laura Beynon, Lucy Southwell, Brendan McLean, Bart C. Jacobs, Baziel G.M. van Engelen, Harry R. Dalton
Neurology Feb 2014, 82 (6) 498-503; DOI: 10.1212/WNL.0000000000000112

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Abstract

Objective: To determine whether there is an association between an acute preceding hepatitis E virus (HEV) infection and neuralgic amyotrophy (NA), and if so, whether patients with HEV-related NA differ from patients without an associated HEV infection.

Methods: HEV testing was conducted in a retrospective cohort of 28 Cornish patients with NA (2011–2013) and a prospective cohort of 38 consecutive Dutch patients with NA (2004–2007). Acute-phase serum samples were analyzed for the presence of anti-HEV immunoglobulin (Ig) M and IgG and HEV RNA (quantitative real-time PCR).

Results: Five cases (10.6%) of acute hepatitis E infection were identified in a total group of 47 patients with NA of whom serum samples were available. In 4 patients, HEV RNA was detected in serum samples taken at presentation. All patients with HEV-associated NA had clinical and electrophysiologic evidence of bilateral brachial plexus involvement. Anti-HEV IgM positivity was not related to age, sex, disease severity, disease course, or outcome.

Conclusions: Acute hepatitis E is found in 10% of patients with NA from the United Kingdom and the Netherlands. Further research is required to investigate the role of HEV in NA in other geographical locations and to determine pathophysiologic mechanisms.

GLOSSARY

GBS=
Guillain-Barré syndrome;
HEV=
hepatitis E virus;
Ig=
immunoglobulin;
NA=
neuralgic amyotrophy

Footnotes

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

  • See page 491

  • Supplemental data at www.neurology.org

  • Received August 23, 2013.
  • Accepted in final form November 22, 2013.
  • © 2014 American Academy of Neurology
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