Jeroen J J van Eijk1, Harry R Dalton2, Paolo Ripellino1, Richard G Madden1, Catherine Jones1, Miriam Fritz1, Claudio Gobbi1, Giorgia Melli1, Emanuela Pasi1, Jenny Herrod1, Rebecca F Lissmann1, Hamad H Ashraf1, Mohamed Abdelrahim1, Omar A B A L Masri1, Montserrat Fraga1, David Benninger1, Thierry Kuntzer1, Vincent Aubert1, Roland Sahli1, Darius Moradpour1, Hélène Blasco-Perrin1, Shahram Attarian1, Rene Gérolami1, Philippe Colson1, Maria T Giordani1, Johannes Hartl1, Sven Pischke1, Nan X Lin1, Brendan N Mclean1, Richard P Bendall1, Marcus Panning1, Jean-Marie Peron1, Nassim Kamar1, Jacques Izopet1, Bart C Jacobs1, Nens van Alfen1, Baziel G M van Engelen1. 1. From the Jeroen Bosch Hospital (J.J.J.v.E.), 's-Hertogenbosch, the Netherlands; Royal Cornwall Hospital (H.R.D., R.G.M., C.J., J. Herrod, R.F.L., H.H.A., M.A., O.A.B.A.L.M., B.N.M., R.P.B.); European Centre for the Environment and Human Health (H.R.D., R.P.B.), University of Exeter, Truro, UK; Neurocenter of Southern Switzerland (P.R., C.G., G.M.), Lugano; Department of Neurology and Neuroscience (M.F.) and Institute for Virology (M.P.), Medical Center-University of Freiburg and Faculty of Medicine, University of Freiburg, Germany; Microbiology Department (E.P.), EOLAB (SMIC), Bellinzona, Switzerland; University Hospital Lausanne (CHUV) (M.F., D.B., T.K., V.A., R.S., D.M.), Switzerland; Université Paul Sabatier (H.B.-P., J.-M.P., N.K., J.I.), Toulouse, France; Centre Hospitalo-Universitaire Timone (S.A., R.G., P.C.); IHU-Méditerranée Infection (P.C.), Aix-Marseille Université, AP-HM, France; San Bortolo Hospital (M.T.G.), Vicenza, Italy; University Medical Center Hamburg-Eppendorf (J. Hartl, S.P.), Germany; Northumbria University (N.X.L.), Newcastle upon Tyne, UK; CHU Rangueil (N.K.); CHU Purpan (H.B.-P., J.-M.P., N.K., J.I.), Toulouse, France; Erasmus MC (B.C.J.), University Medical Centre Rotterdam; and Department of Neurology (N.v.A., B.G.M.v.E.), Donders Center for Medical Neuroscience, Radboudumc Nijmegen, the Netherlands. 2. From the Jeroen Bosch Hospital (J.J.J.v.E.), 's-Hertogenbosch, the Netherlands; Royal Cornwall Hospital (H.R.D., R.G.M., C.J., J. Herrod, R.F.L., H.H.A., M.A., O.A.B.A.L.M., B.N.M., R.P.B.); European Centre for the Environment and Human Health (H.R.D., R.P.B.), University of Exeter, Truro, UK; Neurocenter of Southern Switzerland (P.R., C.G., G.M.), Lugano; Department of Neurology and Neuroscience (M.F.) and Institute for Virology (M.P.), Medical Center-University of Freiburg and Faculty of Medicine, University of Freiburg, Germany; Microbiology Department (E.P.), EOLAB (SMIC), Bellinzona, Switzerland; University Hospital Lausanne (CHUV) (M.F., D.B., T.K., V.A., R.S., D.M.), Switzerland; Université Paul Sabatier (H.B.-P., J.-M.P., N.K., J.I.), Toulouse, France; Centre Hospitalo-Universitaire Timone (S.A., R.G., P.C.); IHU-Méditerranée Infection (P.C.), Aix-Marseille Université, AP-HM, France; San Bortolo Hospital (M.T.G.), Vicenza, Italy; University Medical Center Hamburg-Eppendorf (J. Hartl, S.P.), Germany; Northumbria University (N.X.L.), Newcastle upon Tyne, UK; CHU Rangueil (N.K.); CHU Purpan (H.B.-P., J.-M.P., N.K., J.I.), Toulouse, France; Erasmus MC (B.C.J.), University Medical Centre Rotterdam; and Department of Neurology (N.v.A., B.G.M.v.E.), Donders Center for Medical Neuroscience, Radboudumc Nijmegen, the Netherlands. hardalton@gmail.com.
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.
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.
Authors: Thomas Horvatits; Julian Schulze Zur Wiesch; Marc Lütgehetmann; Ansgar W Lohse; Sven Pischke Journal: Viruses Date: 2019-07-05 Impact factor: 5.048
Authors: Johannes H Bannasch; Benjamin Berger; Claus-Peter Schwartkop; Marco Berning; Oliver Goetze; Marcus Panning; Miriam Fritz-Weltin; George Trendelenburg; Mathias Gelderblom; Marc Lütgehetmann; Fridrike Stute; Thomas Horvatits; Meike Dirks; Christoph Antoni; Patrick Behrendt; Sven Pischke Journal: Pathogens Date: 2021-05-30