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

Eijk, J.J.J. van; Dalton, Harry R.; Ripellino, Paolo; Madden, Richard G.; Jones, Catherine; Fritz, Miriam; Gobbi, Claudio; Melli, Giorgia; Pasi, Emanuela; Herrod, Jenny; Lissmann, Rebecca; Ashraf, Hamad H.; Abdelrahim, Mohamed; Masri, Omar; Fraga, Montserrat; Benninger, David; Küntzer, Thierry; Aubert, Vincent; Sahli, Roland; Moradpour, Darius; Blasco-Perrin, Hélène; Attarian, Shahram; Gerolami, René; Colson, Philippe; Giordani, Maria Teresa; Hartl, Johannes; Pischke, Sven; Lin, Nan; McLean, Brendan; Bendall, Richard; Panning, Marcus; Péron, Jean‐Marie; Kamar, Nassim; Izopet, Jacques; Jacobs, Bart C.; Alfen, Nens van; Engelen, B.G.M. van

doi:10.1212/wnl.0000000000004297

Cited by 87 publications

(91 citation statements)

References 23 publications

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“…A recent multi-center study demonstrated that HEV-associated NA patients tend to have more extensive and bilateral involvement of nerves, including nerves outside the brachial plexus. 6 The phenotype of our patients is in line with this conclusion, as all had three or more nerves affected, symptoms were bilateral for two, including involvement of the trunk and lower limb.…”

Section: Discussionsupporting

confidence: 86%

“…In one patient, each of the three bouts was heralded by the intensification and spreading of the pain; in the other two, nerve lesions appeared in two phases following the resolution of pain. Multiphasic presentation was not described in the multi-center study on HEVassociated NA of van Eijk et al, 6 but it is unclear whether this information was collected. However, in one case report of HEV-associated NA, symptoms were progressive over 3 weeks, 5 and in another phrenic nerve palsy developed several months after onset.…”

Section: Discussionmentioning

confidence: 99%

“…Since its first report in 2009, 3 an increasing number of cases have been published and a distinct clinical phenotype of HEV-associated NA has been described. [4][5][6][7] With the aim of further characterizing HEV-associated NA, we describe here 3 patients with an unusual multiphasic clinical presentation.…”

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Multiphasic presentation of neuralgic amyotrophy associated with hepatitis E virus infection

2019

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Background: The aim of this study was to further characterize the clinical phenotype of hepatitis E virus (HEV)-associated neuralgic amyotrophy (NA).Methods: Three patients with HEV-associated NA underwent clinical, electrodiagnostic, and ultrasound assessment.Results: In all patients, symptoms developed in several phases within a time span of 4-6 weeks, with three or more nerves involved. Symptoms were bilateral in two. In two patients, nerves of the trunk and the lower limb were affected as well. In one patient, three bouts occurred, each heralded by an increase in pain. In the other two, pain subsided quickly and nerve damage developed in two phases. Segmental enlargement with or without hourglass-like constrictions of the nerves was demonstrated by ultrasound in all. Conclusions:The multiphasic presentation, together with the extensive multi-nerve involvement, may reflect a severe and protracted inflammation of the nerves in HEVassociated NA. K E Y W O R D S clinical phenotype, hepatitis E virus infection, hourglass-like constriction, multiphasic presentation, neuralgic amyotrophy, Parsonage-Turner syndrome 1 | INTRODUCTION Neuralgic amyotrophy (NA) is an acute painful dysimmune neuropathy, typically affecting nerves originating from the brachial plexus. 1,2Lately, a specific association between hepatitis E virus (HEV) infection and NA has emerged. Since its first report in 2009, 3 an increasing number of cases have been published and a distinct clinical phenotype of HEV-associated NA has been described. [4][5][6][7] With the aim of further characterizing HEV-associated NA, we describe here 3 patients with an unusual multiphasic clinical presentation. | METHODSApproval was obtained from the Institutional Ethics Committee.The following criteria were used for the diagnosis of NA, (1) acute pain in the shoulder or arm, associated with weakness or sensory loss in the distribution of one or more peripheral nerves; (2) electrodiagnostic (EDX) proof of axon loss; (3) absence of other conditions explaining the symptoms. The diagnosis was further supported by imaging findings. Patients underwent clinical, EDX, and high-resolution ultrasound examination of affected nerves. The diagnosis of HEV infection associated with the acute stage of NA was based on elevated transaminase levels and HEV IgM antibody positivity.3 | RESULTS | Patient 1The 30-year-old man experienced an acute onset of intense pain in the left forearm, followed by loss of sensation on the lateral aspect of the left forearm (indicative of left lateral antebrachial cutaneous nerve lesion). Two weeks later, fever developed and the pain spread to the whole left arm and shoulder. The fever subsided within days, when he noticed the inability to flex the distal phalanx of his left thumb (presumed left partial anterior interosseous nerve lesion). At this point, he received a single dose of 2 mg of intramuscular betamethasone, leading to pain reduction. One month later (6 weeks after onset), fever lasting for 2-3 days developed again, the pain intensified and be...

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Section: Discussionsupporting

confidence: 86%

Section: Discussionmentioning

confidence: 99%

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Multiphasic presentation of neuralgic amyotrophy associated with hepatitis E virus infection

2019

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“…We are not aware of a report about the link between HEV seropositivity and reflexes impairment. Acute and chronic HEV infections may lead to extrahepatic manifestations and has been associated with neurological diseases [24, 25], including for instance, neuralgic amyotrophy [26], acute encephalitis Parkinsonism [25], and Guillain Barré syndrome [27].…”

Section: Discussionmentioning

confidence: 99%

Hepatitis E Virus infection and waste pickers: a case-control seroprevalence study

Alvarado‐Esquivel¹,

Gutierrez-Martinez

Ramírez-Valles

et al. 2019

Preprint

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33Whether waste pickers are a risk group for hepatitis E virus (HEV) infection is largely 34 unknown. Therefore, this study aimed to determine the association between HEV exposure 35 and: 1) the occupation of waste picker; and 2) the work characteristics of waste pickers. An 36 age-and gender-matched case-control seroprevalence study of 86 waste pickers (mean age: 37 35.45 ± 17.15 years) and 86 control subjects of the general population was performed. We 38 determined anti-HEV IgG antibodies in sera of cases and controls using a commercially 39 available enzyme-linked immunoassay. The McNemar's test was used to assess the 40 association between HEV seropositivity and the occupation of waste picker. The 41 association between HEV seropositivity and work characteristics of waste pickers was 42 assessed by bivariate and regression analyses. Anti-HEV IgG antibodies were detected in 43 14 (16.3%) of the 86 waste pickers and in 8 (9.3%) of the 86 control subjects (McNemar´s 44pair test: OR = 13.0; 95% CI: 0.73-230.77; P=0.02). Ill waste pickers had a higher HEV 45 seroprevalence than those who were apparently healthy (6/15: 40% vs 8/71: 11.3%, 46 respectively: P=0.01). Waste pickers with reflexes impairment had a higher HEV 47 seroprevalence than those without this impairment (5/10: 50% and 9/76: 11.8%, 48 respectively; P=0.009). Logistic regression analysis of sociodemographic, work, and 49 behavioral characteristics of waste pickers showed that HEV seropositivity was associated 50 with increasing age (OR = 6.52; 95% CI: 1.95-21.78; P=0.002) and raising pigs (OR = 51 12.01; 95% CI: 1. 48-97.26; P=0.02). This is the first age-and gender-matched case-control 52 study on the association between HEV infection and the occupation of waste picker. Waste 53 pickers represent a risk group for HEV infection. Factors associated with HEV 4 54 seropositivity found in this study may help in the design of optimal planning to avoid HEV 55 infection. 56 57 58 Estimates indicate that there are about 20 million cases of acute hepatitis E and 62 56000 deaths associated with this disease in the world every year [1]. Hepatitis E results 63 from infection with hepatitis E virus that is a single-stranded, positive sense RNA virus [2]. 64 Predominant route of infection with HEV varies depending on the development of the 65 country. In developing countries, HEV transmission occurs via contaminated water and 66 food by the fecal oral route; whereas in developed countries, this infection occurs by 67 zoonotic transmission [3]. Other transmission routes of HEV include blood transfusion [4], 68 hemodialysis [5], and solid organ transplantation [6]. Chronic infections with HEV have 69 been reported in immunocompromised patients, and a high seroprevalence of HEV was 70 found in patients with autoimmune hepatitis [7]. Fulminant hepatitis E has been reported in 71 pregnant women, patients with underlying liver disease [8], and children [9]. Hepatitis E 72 may present as sporadic cases or outbreaks [10]. Consumption of meat from HEV-infected 73 anim...

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“…18 In these cases, motor weakness and sensory disturbances are more often bilateral, asymmetric and are not confined to the brachial plexus compared to non HEV-infected patients. 19,20 The prevalence of HEV infection in patients with GBS is 5%. 21 The mechanisms by which HEV causes PTS or GBS are unknown, but it is probably an immune response induced by the virus.…”

Section: Neurolog I C Al Inj Urymentioning

confidence: 99%

Hepatitis E, what’s the real issue?

Larrue

Abravanel

Péron

2020

Liver International

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Hepatitis E Virus (HEV) infection is a worldwide disease and the primary cause of acute viral hepatitis in the world with an estimated 20 million cases every year and 70 000 deaths. Hepatitis E is a waterborne infection in the developing countries. In these countries, HEV genotypes 1 and 2 cause large outbreaks and affect young subjects, resulting in significant mortality in pregnant women and patients with cirrhosis. In the developed countries, HEV genotypes 3 and 4 are responsible for autochthonous, sporadic hepatitis and transmission is zoonotic. Parenteral transmission by the transfusion of blood products has been identified as a potential new mode of transmission. The prevalence of positive HEV viraemia in blood donors in Europe ranges from 1/600 to 1/2500 in highly endemic European countries. HEV can cause neurological disorders and chronic infections in immunocompromised patients. The progression of acute hepatitis E is usually asymptomatic and resolves spontaneously. Diagnostic tools include anti‐HEV IgM antibodies in serum and/or viral RNA detection in the blood or the stools by PCR. Ribavirin is used to treat chronic infection. A vaccine has been developed in China.

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

Cited by 87 publications

References 23 publications

Multiphasic presentation of neuralgic amyotrophy associated with hepatitis E virus infection

Multiphasic presentation of neuralgic amyotrophy associated with hepatitis E virus infection

Hepatitis E Virus infection and waste pickers: a case-control seroprevalence study

Hepatitis E, what’s the real issue?

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