Neuralgic amyotrophy and hepatitis E virus infection

Eijk, J.J.J. van; Madden, R.G.; Eijk, Annemiek A. van der; Hunter, Jeremy; Reimerink, Johan; Bendall, Richard; Pas, Suzan D.; Ellis, Vic; Alfen, Nens van; Beynon, L. L.; Southwell, Lucy; McLean, Brendan; Jacobs, Bart C.; Engelen, B.G.M. van; Dalton, Harry R.

doi:10.1212/wnl.0000000000000112

Cited by 162 publications

(105 citation statements)

References 26 publications

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“…Although extrahepatic manifestations of HEV infection have scarcely been presented at academic meetings [https://www.med.kyushu-u.ac.jp/neuro/chihoukai/past/ 202program.pdf (in Japanese)] and there is no previous literature regarding such manifestations in Japan, several neurological manifestations have been described in patients with HEV infection in European countries, including Belgium, France, Germany, the Netherlands, Portugal and the United Kingdom (Loly et al 2009;Kamar et al 2011;Cheung et al 2012;Santos et al 2013;Scharn et al 2014;van den Berg et al 2014;van Eijk et al 2014;Woolson et al 2014), as well as Asian countries, including Bangladesh, China and India (Dixit et al 2006;Jha et al 2012;Geurtsvankessel et al 2013;Chen et al 2014;Wu et al 2015). A recent case series from Southwest England and Toulouse, France found a 5.6% prevalence (seven out of 126) of neurological complications in cases of locally acquired HEV infection (Kamar et al 2011).…”

Section: Discussionmentioning

confidence: 99%

Characteristics of 20 Patients with Autochthonous Acute Hepatitis E in Hokkaido, Japan: First Report of Bilateral Facial Palsy Following the Infection with Genotype 4 Hepatitis E Virus

Yazaki

Sugawara²,

Honda

et al. 2015

Tohoku J. Exp. Med.

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Autochthonous hepatitis E is increasingly being recognized in industrialized countries, including Japan. Although neurological abnormalities have been sporadically reported as an extrahepatic manifestation of hepatitis E virus (HEV) infection, it is rare and has not been reported in Japan. The present study aimed to characterize a total of 20 patients consecutively diagnosed with sporadic acute hepatitis E at a city hospital in Hokkaido, Japan, during 2001-2014, focusing on a patient complicated with neuropathy. Seventeen patients were infected with genotype 4 HEV, while the remaining three patients were with genotype 3 HEV. Although a 67-year-old male with severe hepatitis did not have predisposing factors associated with the development of neurological disorders, such as diabetes mellitus and the use of immunosuppressive agents, he developed bilateral peripheral facial palsy six days after admission. A neurological examination revealed the inability to smile, frown, close his eyes completely or puff out his cheeks. MRI brain scans were considered to be normal. Although it took 83 days after admission for the total bilirubin levels to normalize, his neurological symptoms resolved gradually within three weeks without any sequelae following conservative therapy. A full-length genomic analysis of the HEV strain (HE-JA30) isolated from the patient belonged to genotype 4 and was closest to that currently circulating in Hokkaido, Japan. This is the first report of HEV-associated neuropathy in Japan. While all of previous reports on HEV-related neuropathy involve genotype 3 HEV, the present report is unique in that genotype 4 HEV is responsible for the neuropathy.

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Section: Discussionmentioning

confidence: 99%

Characteristics of 20 Patients with Autochthonous Acute Hepatitis E in Hokkaido, Japan: First Report of Bilateral Facial Palsy Following the Infection with Genotype 4 Hepatitis E Virus

Yazaki

Sugawara²,

Honda

et al. 2015

Tohoku J. Exp. Med.

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“…As it is likely that neuralgic amyotrophy attacks are ultimately induced by an auto-immune response to the peripheral nervous system [1][2][3][4][5], efforts have been undertaken to attempt treatment in the acute phase with immunomodulants such as corticosteroids or iv gammaglobuline [6][7][8][9][10][11]. Unfortunately, a randomized controlled trial of corticosteroids failed because it proved impossible to recruit the needed number of participants within the three-year trial period.…”

Section: Introductionmentioning

confidence: 99%

Incidence of neuralgic amyotrophy (Parsonage–Turner syndrome) in a primary care setting: A prospective cohort study

Alfen

Groothuis

Nobacht³

et al. 2015

Neuromuscular Disorders

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ObjectiveNeuralgic amyotrophy is considered a rare peripheral nervous system disorder but in practice seems grossly under recognized, which negatively affects care for these patients. In this study we prospectively counted the one-year incidence rate of classic neuralgic amyotrophy in a primary care setting. MethodsIn a prospective cohort study during the year 2012 we registered all new cases of neck, shoulder or arm complaints from two large primary care centers serving a population of 14,118. Prior to study, general practitioners received a short training on how to diagnose classic neuralgic amyotrophy. Neuralgic amyotrophy was defined according to published criteria irrespective of family history. Only patients with a classic phenotype were counted as definite cases. After inclusion, patients with suspected neuralgic amyotrophy who had not yet seen a neurologist were offered neurologic evaluation for diagnostic confirmation. ResultsOf the 492 patients identified with new onset neck, shoulder or arm complaints, 34 were suspected of having neuralgic amyotrophy. After neurologic evaluation the diagnosis was confirmed in 14 patients. This amounts to a one-year incidence rate for classic neuralgic amyotrophy of 1 per 1000. Data Availability Statement: No centralized data collection was available for this study. Data were collected in an anonymized digital datafile and stored locally by the study group. The primary data file for this study is available as S1 Datafile.Funding: This study was sponsored by an unsolicited and unconditional gift from the Dutch Rare Diseases Fund (Stichting Zeldzame Ziekten Fonds; www.zzf.nl) and the ZABAWAS Foundation (www. zabawas.nl). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. ConclusionsOur findings suggest that neuralgic amyotrophy is 30-50 times more common than previously thought. Unawareness of the disorder and its clinical presentation seems the most likely explanation for this difference. An incidence rate of 1 per 1000 and the long-term sequelae many patients suffer warrant more vigilance in diagnosing the disorder, to pave the way for timely treatment and prevent complications.

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“…However, fulminant infection sporadically occurs, and in some patients, chronic infection may ensue, often with rapid fibrosis progression leading to cirrhosis; this most commonly occurs in immunocompromised individuals, such as solid organ recipients and patients receiving chemotherapy (6)(7)(8)(9)(10)(11). Additionally, hepatitis E may be associated with neurological manifestations, such as Guillain-Barré syndrome (12), neuralgic amyotrophy (13), and meningitis (14,15).…”

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confidence: 99%

Diagnostic Performance of Five Assays for Anti-Hepatitis E Virus IgG and IgM in a Large Cohort Study

et al. 2016

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Neuralgic amyotrophy and hepatitis E virus infection

Abstract: 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.

Cited by 162 publications

References 26 publications

Characteristics of 20 Patients with Autochthonous Acute Hepatitis E in Hokkaido, Japan: First Report of Bilateral Facial Palsy Following the Infection with Genotype 4 Hepatitis E Virus

Characteristics of 20 Patients with Autochthonous Acute Hepatitis E in Hokkaido, Japan: First Report of Bilateral Facial Palsy Following the Infection with Genotype 4 Hepatitis E Virus

Incidence of neuralgic amyotrophy (Parsonage–Turner syndrome) in a primary care setting: A prospective cohort study

Diagnostic Performance of Five Assays for Anti-Hepatitis E Virus IgG and IgM in a Large Cohort Study

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