The neuralgic amyotrophy may be of difficult diagnosis, due to phenotypic variability, with different initial presentations (upper plexus multiple mononeuropathy, lumbosacral involvement, distal reached, phrenic involvement). To date, there is little guidance on these patients’ therapeutic management, especially those for which neuralgic amyotrophy is triggered by hepatitis E virus (HEV-NA). The study aims to identify specific features that characterize patients bearing the neuralgic amyotrophy triggered by HEV. We first describe a new case report of HEV-neuralgic amyotrophy, with delayed diaphragmatic reach. Then, the literature was searched for reports of HEV-NA (n = 39), and neuralgic amyotrophy with phrenic paresis (n = 42) from 1999 to June 2016. Relevant data were retrieved, analyzed and compared with the parameters of idiopathic neuralgic amyotrophy (n = 199) of the largest cohort, described by Van Alfen and Van Engelen in 2006. Compared to the published cohort, HEV-NA patients were more likely to be men (M/F 34/5 vs. 136/63, p = 0.017), with more frequent bilateral symptoms (86.8% cases vs. 28.5%, p < 0.0001) as well as phrenic paresis (18.0 vs. 6.6%, p = 0.028). The clinical improvement is poor, with 15.6% of cases with remission only. A particular phenotype characteristic of the HEV-induced neuralgic amyotrophy has arisen. Our findings call for action in validating the above-mentioned features that illustrate the HEV-NA cases as an early diagnosis would prevent complications, especially the phrenic damage often associated with a worse functional outcome.
Brachial neuritis Parsonage–Turner syndrome Peripheral neuropathies HEV Phrenic paresis
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Conception and design: QS and SMD. Collection data (case report): QS, TP, FC, OO, AL, PYH, ML and SMD. Collection and assembly of data (review): QS. Data analysis: QS. Interpretation: QS and SM. Manuscript drafting: QS and SMD. Final approval of manuscript: QS, TP, FC, OO, AL, PYH, ML and SMD.
The authors thank Laura Ravasi MD-PhD. who provided medical writing services and Hélène Béhal for statistical advice on behalf of the University of Lille.
They are grateful to the National Reference Center for Hepatitis E Virus in Toulouse, France, for HEV genotype analysis.
Compliance with ethical standards
Conflicts of interest
All authors report no disclosures relevant to the manuscript.
The study was performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinski and its later amendments.
First and last authors had received consent from the patient whose case is reported.
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