Clinical phenotype and outcome of hepatitis E virus - associated neuralgic amyotrophy

Jeroen van Eijk, Harry Dalton, Paolo Ripellino, Richard Madden, Catherine Jones, Miriam Fritz, Claudio Gobbi, Giorgia Melli, Emanuela Pasi, Jenny Herrod, Rebecca Lissmann, Hamad Ashraf, Mohamed Abdelrahim, Omar Masri, Montserrat Fraga, David Benninger, Thierry Kuntzer, Vincent Aubert, Roland Sahli, Darius MoradpourHelene Blasco-Perrin, Shahram Attarian, Rene Gérolami, Philippe Colson, Maria Giodani, Johannes Hartl, Sven Pischke, Nan Lin, Brendan McLean, Richard Bendall, Marcus Panning, Jean-Marie Peron, Nassim Kamar, Jacques Izopet, Bart Jacobs, Nens van Alfen, Baziel van Engelen

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Abstract

Objective: To determine the clinical phenotype and outcome in hepatitis E virus–associated neuralgic amyotrophy (HEV-NA).Methods: Cases of NA were identified in 11 centers from 7 European countries, with retrospective analysis of demographics, clinical/laboratory findings, and treatment and outcome. Cases of HEV-NA were compared with NA cases without evidence of HEV infection.Results: Fifty-seven cases of HEV-NA and 61 NA cases without HEV were studied. Fifty-six of 57 HEV-NA cases were anti-HEV IgM positive; 53/57 were IgG positive. In 38 cases, HEV RNA was recovered from the serum and in 1 from the CSF (all genotype 3). Fifty-one of 57 HEV-NA cases were anicteric; median alanine aminotransferase 259 IU/L (range 12–2,961 IU/L); in 6 cases, liver function tests were normal. HEV-NA cases were more likely to have bilateral involvement (80.0% vs 8.6%, p <0.001), damage outside the brachial plexus (58.5% vs 10.5%, p <0.01), including phrenic nerve and lumbosacral plexus injury (25.0% vs 3.5%, p = 0.01, and 26.4% vs 7.0%, p = 0.001), reduced reflexes (p = 0.03), sensory symptoms (p = 0.04) with more extensive damage to the brachial plexus. There was no difference in outcome between the 2 groups at 12 months.Conclusions: Patients with HEV-NA are usually anicteric and have a distinct clinical phenotype, with predominately bilateral asymmetrical involvement of, and more extensive damage to, the brachial plexus. Involvement outside the brachial plexus is more common in HEV-NA. The relationship between HEV and NA is likely to be causal, but is easily overlooked. Patients presenting with NA should be tested for HEV, irrespective of liver function test results. Prospective treatment/outcome studies of HEV-NA are warranted.
Original languageEnglish
Pages (from-to)909-917
JournalNeurology
Volume89
Issue number9
Early online date2 Aug 2017
DOIs
Publication statusPublished - 29 Aug 2017

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