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      Incidence of Neuralgic Amyotrophy (Parsonage Turner Syndrome) in a Primary Care Setting - A Prospective Cohort Study

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          Abstract

          Objective

          Neuralgic 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.

          Methods

          In 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.

          Results

          Of 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.

          Conclusions

          Our 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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          Most cited references18

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          The clinical spectrum of neuralgic amyotrophy in 246 cases.

          We investigated the symptoms, course and prognosis of neuralgic amyotrophy (NA) in a large group of patients with idiopathic neuralgic amyotrophy (INA, n = 199) and hereditary neuralgic amyotrophy (HNA, n = 47) to gain more insight into the broad clinical spectrum of the disorder. Several findings from earlier smaller-scale studies were tested, and for the first time the potential differences between the hereditary and idiopathic phenotypes and between males and females were explored. Generally, the course of the pain manifests itself in three consecutive phases with an initial severe, continuous pain lasting for approximately 4 weeks on average. Sensory involvement was quite common and found in 78.4% of patients but was clinically less impairing than the initial pain and subsequent paresis. As a typically patchy disorder NA can affect almost any nerve in the brachial plexus, although damage in the upper and middle trunk distribution with involvement of the long thoracic and/or suprascapular nerve occurred most frequently (71.1%). We found no correlation between the distribution of motor and sensory symptoms. In INA recurrent attacks were found in 26.1% of the patients during an average 6 year follow-up. HNA patients had an earlier onset (28.4 versus 41.3 years), more attacks (mean 3.5 versus 1.5) and more frequent involvement of nerves outside the brachial plexus (55.8 versus 17.3%) than INA patients, and a more severe maximum paresis, with a subsequent poorer functional outcome. In males the initial pain tended to last longer than it did in females (45 versus 23 days). In females the middle or lower parts of the brachial plexus were involved more frequently (23.1 versus 10.5% in males), and their functional outcome was worse. Overall recovery was less favourable than usually assumed, with persisting pain and paresis in approximately two-thirds of the patients who were followed for 3 years or more.
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            Clinical and pathophysiological concepts of neuralgic amyotrophy.

            Neuralgic amyotrophy--also known as Parsonage-Turner syndrome or brachial plexus neuritis--is a distinct and painful peripheral neuropathy that causes episodes of multifocal paresis and sensory loss in a brachial plexus distribution with concomitant involvement of other PNS structures (such as the lumbosacral plexus or phrenic nerve) in a large number of patients. The phenotype can be limited or extensive and the amount of disability experienced also varies between patients, but many are left with residual disabilities that affect their ability to work and their everyday life. Both idiopathic and hereditary forms exist. The latter form is genetically heterogeneous, but in 55% of affected families, neuralgic amyotrophy is associated with a point mutation or duplication in the SEPT9 gene on chromosome 17q25. The disease is thought to result from an underlying genetic predisposition, a susceptibility to mechanical injury of the brachial plexus (possibly representing disturbance of the epineurial blood-nerve barrier), and an immune or autoimmune trigger for the attacks. The precise pathophysiological mechanisms are still unclear; treatment is empirical, and preventive measures are not yet available. This Review provides an overview of the current clinical and pathophysiological concepts and research topics in neuralgic amyotrophy.
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              Neuralgic amyotrophy and hepatitis E virus infection.

              To determine whether there is an association between an acute preceding hepatitis E virus (HEV) infection and neuralgic amyotrophy (NA), and if so, whether patients with HEV-related NA differ from patients without an associated HEV infection.
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                Author and article information

                Contributors
                Role: Academic Editor
                Journal
                PLoS One
                PLoS ONE
                plos
                plosone
                PLoS ONE
                Public Library of Science (San Francisco, CA USA )
                1932-6203
                27 May 2015
                2015
                : 10
                : 5
                : e0128361
                Affiliations
                [1 ]Department of Neurology, Donders Centre for Neuroscience, Radboud university medical centre, Nijmegen, The Netherlands
                [2 ]Department of Neurology, Jeroen Bosch Hospital, Den Bosch, The Netherlands
                [3 ]Department of Neurology, Erasmus Medical Center, Rotterdam, The Netherlands
                [4 ]Royal College of Surgeons in Ireland, Dublin, Ireland
                [5 ]Primary Health Centre Oosterhout, Nijmegen, The Netherlands
                [6 ]Department of Rehabilitation, Donders Centre for Neuroscience, Radboud university medical centre, Nijmegen, The Netherlands
                [7 ]Department of Neurology, Canisius Wilhelmina Hospital, Nijmegen, The Netherlands
                [8 ]Department of Primary and Community Care, Radboud Institute for Health Sciences, Radboud university medical centre, The Netherlands & Academic Health Centre Thermion, Lent, The Netherlands
                University of Würzburg, GERMANY
                Author notes

                Competing Interests: The authors have declared that no competing interests exist.

                Conceived and designed the experiments: NvA JG FvdL. Performed the experiments: NvA JvE IN SP FvdL. Analyzed the data: NvA TE SOF. Wrote the paper: NvA JvE TE SOF IN JG SP FvdL.

                Article
                PONE-D-14-58360
                10.1371/journal.pone.0128361
                4445915
                26016482
                a560b6a8-aa14-4309-8b22-5b35796bc034
                Copyright @ 2015

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited

                History
                : 7 January 2015
                : 26 April 2015
                Page count
                Figures: 1, Tables: 4, Pages: 9
                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.
                Categories
                Research Article
                Custom metadata
                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.

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                Uncategorized

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