Neuralgic amyotrophy associated with hepatitis E virus

doi:10.1016/j.clineuro.2008.09.005

Clinical Neurology and Neurosurgery

Volume 111, Issue 2, February 2009, Pages 193-195

https://doi.org/10.1016/j.clineuro.2008.09.005 Get rights and content

Abstract

Neuralgic amyotrophy (NA) is characterised by neuropathic pain and patchy paresis of the upper or lower limbs, usually involving the upper and middle trunks of the brachial plexus. The aetiology of NA is varied, with precipitating factors that include trauma, surgery, pregnancy, inoculations and infections. Deranged liver enzymes have been noted in previous NA reports but no cause identified. We describe a case of bilateral NA in a 53-year-old man who presented with peripheral neuropathy and isolated derangement of liver enzymes. Serology was positive for hepatitis E infection and negative for other infections previously described to be associated with NA. The diagnosis was supported by electrophysiological findings. This case report suggests that hepatitis E is a potential cause of NA that had not previously been described in the literature, and further supports a proposed immune pathogenesis underlying the condition.

Introduction

Neuralgic amyotrophy (NA) is a form of peripheral neuropathy characterised by neuropathic pain and patchy paresis of the upper limbs, usually involving the upper and middle trunks of the brachial plexus. NA typically manifests initially with pain, with subsequent weakness and atrophy of affected muscles. The incidence of idiopathic NA is 2–3/100,000 per year [1]. First described in the nineteenth century, the publication of the first large case series of 136 patients in 1948 coined its eponymous name, Parsonage–Turner syndrome [2]. Precipitating causes of NA include trauma, surgery, inoculations and various infections, although the pathophysiology behind its development remains unclear.

Hepatitis E is an enterically transmitted virus prominent in the developing world, responsible for both sporadic and endemic hepatitis. It was first characterised in patients with serological markers distinct from hepatitis A or B virus, and became known as non-A, non-B hepatitis [3]. The course of illness is usually self-limiting with no reports of chronic hepatitis E infection. In addition, to our knowledge, there has not been any documented neurological complications or peripheral neuropathy arising subsequent to hepatitis E infection.

Section snippets

Case report

A 53-year-old ecologist presented to the Emergency Department with onset of bilateral shoulder pain radiating to the chest. He had returned from a trip to Equador 4 weeks prior to presentation. He had no past medical history, previous surgery to the shoulder or neck or history or trauma. He did not have any flu-like or coryzal symptoms. He reported minimal alcoholic intake. Systemic examination including neurological examination was normal. Investigations revealed no evidence of systemic

Discussion

This patient had NA because of his clinical presentation with a characteristic patchy peripheral neuropathy involving the brachial plexus that was confirmed with electrophysiological testing. The diagnosis of NA is based on clinical findings and is supported by nerve conduction studies and electromyography. However, factors such as the time of testing, the patchy distribution and varying degree of neural damage may influence the results and thus be inconclusive or yield false-negative results

Conflicts of interest

None to declare.

Acknowledgement

None.

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There are more references available in the full text version of this article.

Cited by (63)

Parsonage-Turner syndrome associated with hepatitis E infection in immunocompetent patients
2020, Virus Research
Introduction The hepatitis E virus (HEV) is the leading cause of acute hepatitis around the world. In recent years, knowledge has increased concerning extrahepatic manifestations caused by HEV, including neurological manifestations such as Parsonage-Turner syndrome (PTS). PTS is characterized by severe shoulder or arm pain and patchy paresis with muscle weakness. The aim of the present study was to assess the association between HEV and PTS. Materials and Methods We reported two cases of PTS associated with HEV, which were diagnosed in a short period of time in the same village. PTS was diagnosed by physical examination and electrophysiological studies, and serology testing for IgM, low-avidity IgG, and RNA of HEV established the diagnosis of acute HEV infection. Results A 44-year-old man who presented cervicobrachial pain accompanied by paresthesia, dyspnea, and isolated derangement of liver enzymes and 57-year-old women with cervical pain radiated to upper limbs, paresthesia, and liver cytolysis, although, this patient was initially diagnosed as having drug-induced hepatitis. Finally, the diagnosis was Parsonage- Turner syndrome associated with hepatitis e virus. In both patients, symptoms were bilateral and they required hospital admission. Both consumed vegetables are grown in a local patch and the phylogenetic analysis showed genotype 3f. Then, we reviewed the literature on PTS and HEV and we found 62 previously described cases that were more likely to be men (86.20 %) with more frequent bilateral symptoms (85.71 %). Genotype 3 is the most commonly associated. Three of those cases were diagnosed in Spain. Conclusions According to our findings, HEV should be considered in patients with neuralgic amyotrophy, including those with the absence of liver cytolysis.
Neuralgic Amyotrophy
2018, Challenging Neuropathic Pain Syndromes: Evaluation and Evidence-Based Treatment
Neuralgic amyotrophy, also known as Parsonage and Turner syndrome, is a painful nerve disorder that can present with a variety of clinical manifestations. The exact cause is unknown, but autoimmune, genetic, infectious, environmental, and biomechanical processes have been implicated. Neuralgic amyotrophy is a challenging neurologic condition to diagnose and treat. It usually affects portions of the brachial plexus, most commonly the suprascapular and long thoracic nerves, but it can also involve the cervical or lumbosacral plexus, particularly in hereditary neuralgic amyotrophy, where recurrence rates are higher. Treatment is usually conservative, with support for the early use of corticosteroids, and most cases lead to a favorable outcome.
Hepatitis E virus: Infection beyond the liver?
2017, Journal of Hepatology
Hepatitis E virus (HEV) infections are not limited to the liver but may also affect other organs. Several diseases, including Guillain-Barré syndrome, neuralgic amyotrophy, glomerulonephritis, cryoglobulinemia, pancreatitis, lymphoma, thrombopenia, meningitis, thyroiditis and myocarditis have been observed in the context of hepatitis E. To date, the definite pathophysiological links between HEV and extrahepatic manifestations are not yet established. However, it is suggested that HEV infection might be causative based on serological studies, case series, in vitro data and animal models. In particular, neuronal and renal diseases as well as pancreatitis seem to be caused by HEV, while a causative relationship between HEV and other diseases is more doubtful.
Either direct cytopathic tissue damage by extrahepatic replication, or immunological processes induced by an overwhelming host immune response, are possible origins of HEV-associated extrahepatic manifestations.
Hepatologists should be aware of the possibility that acute or chronically HEV-infected patients could develop extrahepatic manifestations. Neurologists, nephrologists, rheumatologists and other groups of physicians should consider HEV infection as a potential differential diagnosis when observing one of the diseases described in this review.
Ribavirin and steroids have been used in small groups of patients with extrahepatic manifestations of HEV, but the efficacy of these drugs still needs to be verified by large, multicenter studies.
This article comprehensively reviews the published literature regarding HEV and extrahepatic manifestations. We discuss the probability of specific extrahepatic diseases being caused by previous or ongoing HEV infection, and summarize the published knowledge about antiviral treatment in extrahepatic disorders.
Neuralgic Amyotrophy
2017, Challenging Neuropathic Pain Syndromes: Evaluation and Evidence-Based Treatment
Neuralgic amyotrophy, also known as Parsonage and Turner syndrome, is a painful nerve disorder that can present with a variety of clinical manifestations. The exact cause is unknown, but autoimmune, genetic, infectious, environmental, and biomechanical processes have been implicated. Neuralgic amyotrophy is a challenging neurologic condition to diagnose and treat. It usually affects portions of the brachial plexus, most commonly the suprascapular and long thoracic nerves, but it can also involve the cervical or lumbosacral plexus, particularly in hereditary neuralgic amyotrophy, where recurrence rates are higher. Treatment is usually conservative, with support for the early use of corticosteroids, and most cases lead to a favorable outcome.
Hepatitis E and neuralgic amyotrophy: Five cases and review of literature
2015, Journal of Clinical Virology
Citation Excerpt :
No symptomatic HEV cases were identified in close contacts. Twenty-one cases describing neuralgic amyotrophy associated with HEV were found in our literature search [11,12,16,18–29]. Including our patients, we count a total of 26 cases reports (Table 1).
Hepatitis E virus infection – mainly genotype 3 – is increasingly common in industrialized countries. Infection is usually asymptomatic, but cases of central or peripheral neurological symptoms with hepatitis E have been described. The most frequent is Guillain-Barre but somes cases of neuralgic amyotrophy have been described. In our center, since 2010, we have identified five cases of neuralgic amyotrophy associated with acute hepatitis E in immunocompetent patients. For all these patients, neuralgic amyotrophy was diagnosed with electromyogram and positive IgM for hepatitis E, and detectable HEV RNA in 4 of the cases. Including our patients, we count 26 cases in literature. The mean age of the patients was 44 years old, with a large predominance of males (88%). The disorder is bilateral and asymmetric in 69% of cases. Peripheral nerves other than the brachial plexus were affected in 6 patients (23%). In industrialized countries, any neuralgic amyotrophy, particularly if there is bilateral, asymmetric associated with involvement of nerves outside the brachial plexus, should lead physicians to consider a diagnosis of acute hepatitis E.
Significance of hepatitis E virus infection in HIV-infected patients: A challenging issue
2015, Asian Pacific Journal of Tropical Disease
Hepatitis E virus (HEV) is a small, single-stranded, non-enveloped RNA virus and belongs to the genus Hepevirus in the Hepeviridae family. Currently, the HEV infection is the most frequent cause of acute hepatitis in the world. In recent years, some studies have been demonstrated that immunosuppressed cases, such organ transplant recipients, cases with HIV infection and patients with hematological malignancies are at risk of HEV infection. But it is not clear whether HEV infection is a major concern in HIV infected patients or not? The answer has considerable significance, because HIV and HEV infection are now both highly endemic in many parts of the world. The purpose of this review is to provide data on the prevalence of HEV infection in HIV infected patients for determination of the significance of HEV/HIV co-infection.

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Case reportNeuralgic amyotrophy associated with hepatitis E virus

Abstract

Introduction

Section snippets

Case report

Discussion

Conflicts of interest

Acknowledgement

Lancet

Neurology

Biomed Pharmacother

The incidence and lifetime prevalence of neurological disorders in a prospective community-based study in the UK

Brain

Transfusion-associated hepatitis not due to viral hepatitis type A or B

N Engl J Med

The clinical spectrum of neuralgic amotrophy in 246 cases

Brain

Acute brachial plexus neuritis: an uncommon cause of shoulder pain

Am Fam Physician

Parsonage–Turner syndrome: MR imaging findings and clinical information of 27 patients

Radiology

Neuralgic amyotrophy

Orthop Rev

Case report
Neuralgic amyotrophy associated with hepatitis E virus