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      Neuralgia amiotrófica posterior a la vacunación contra la leptospirosis humana Translated title: Neuralgic amyotrophy following vaccination against human leptospirosis

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          Abstract

          Introducción: la neuralgia amiotrófica o síndrome de Parsonage-Turner se asocia a vacunaciones en 15 % de los casos. No existen reportes que vinculen esta condición con la vacuna contra la leptospirosis humana. Objetivo: describir las características clínicas de un paciente que presentó una plexopatía braquial aguda posterior a la inmunización contra la leptospirosis humana. Presentación del caso: un varón de 31 años de edad, obrero agrícola en 2008 recibió una dosis de la vacuna cubana contra la leptospirosis humana (vax-Spiral); 3 semanas después presentó dolor intenso localizado en ambos hombros, a la semana siguiente aparece debilidad muscular y posteriormente atrofia del músculo deltoides derecho. La evaluación neurológica era compatible con afección aguda asimétrica de la porción superior del plexo braquial, con mayor intensidad en el lado derecho, en el cual también estaba involucrado el diafragma. Se comprobó el compromiso de la porción superior del plexo braquial con estudios de neuroconducción y electromiografía. El paciente es tratado con analgésicos y fisioterapia, evolucionando de modo favorable. Conclusiones: el evento ocurrido en este caso sugiere que la vacuna cubana contra leptospirosis humana pudiera producir inmunorreactividad cruzada contra antígenos del sistema nervioso periférico.

          Translated abstract

          Introduction: neuralgic amyotrophy or Parsonage-Turner syndrome is associated with vaccination in 15% of cases. There are no reports linking this condition to the vaccine against human leptospirosis. Objective: describe the clinical characteristics of a patient who developed acute brachial plexopathy after vaccination against human leptospirosis. Case presentation: a male 31 year-old agricultural worker received a dose of the Cuban vaccine against human leptospirosis (vax-SPIRAL) in 2008. Three weeks later he presented intense pain in both shoulders. The following week he had muscular weakness, and then atrophy of the right deltoid muscle. Neurological evaluation showed acute asymmetric damage to the upper brachial plexus, with greater intensity on the right side, and involvement of the diaphragm. Nerve conduction examination and electromyography revealed involvement of the upper brachial plexus. The patient was treated with analgesics and physical therapy, and was found to evolve favorably. Conclusions: the events described suggest that the Cuban vaccine against human leptospirosis might produce immunological cross-reactivity against antigens of the peripheral nervous system.

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          Most cited references20

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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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            Treatment for idiopathic and hereditary neuralgic amyotrophy (brachial neuritis).

            Neuralgic amyotrophy (also know as Parsonage-Turner syndrome or brachial plexus neuritis) is a distinct peripheral nervous system disorder characterised by episodes (attacks) of extreme neuropathic pain and rapid multifocal weakness and atrophy in the upper limbs. Neuralgic amyotrophy has both an idiopathic and hereditary form, with similar clinical symptoms but generally an earlier age of onset and more episodes in the hereditary form. The current hypothesis is that the episodes are caused by an immune-mediated response to the brachial plexus. Recovery is slow, in months to years, and many patients are left with residual pain and decreased exercise tolerance of the affected limb(s). Anecdotal evidence suggests that corticosteroids may relieve pain or help improve functional recovery. The objective was to provide a systematic review of all randomised clinical trials of treatment in neuralgic amyotrophy. We searched the Cochrane Neuromuscular Disease Group Trials Register (April 2 2009), MEDLINE (January 1966 to April 2 2009), EMBASE (January 1980 to April 2 2009), CINAHL (January 1982 to April 2 2009), and LILACS (January 1982 to April 2 2009) for randomised controlled trials of treatment for neuralgic amyotrophy. Any randomised or quasi-randomised trial of any intervention for neuralgic amyotrophy would be included in the review. Two review authors extracted the data (RH, NvA) and two authors assessed study quality and performed data extraction independently (NvA, BvE). No randomised or quasi-randomised trials were identified. In 30 articles anecdotal evidence was found on treatment for neuralgic amyotrophy. Only three of these articles contained more than 10 treated cases, with one providing sufficient details to calculate the primary and secondary outcome measures for this review. At this moment there is no evidence from randomised trials on any form of treatment for neuralgic amyotrophy. Evidence from one open-label retrospective series suggests that oral prednisone given in the first month after onset can shorten the duration of the initial pain and leads to earlier recovery in some patients. Randomised clinical trials are needed to establish the efficacy of treatment with corticosteroids or other immune-modulating therapies.
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              Immune brachial plexus neuropathy: suggestive evidence for an inflammatory-immune pathogenesis.

              We report brachial plexus biopsy findings from two Australian and two American patients with brachial plexus neuropathy. There were florid multifocal mononuclear inflammatory cell infiltrates. Present evidence suggests that these brachial neuropathies have an immune basis.
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                Author and article information

                Journal
                mtr
                Revista Cubana de Medicina Tropical
                Rev Cubana Med Trop
                Centro Nacional de Información de Ciencias Médicas (Ciudad de la Habana, , Cuba )
                0375-0760
                1561-3054
                September 2013
                : 65
                : 3
                : 398-402
                Affiliations
                [01] La Habana orgnameHospital Hermanos Ameijeiras Cuba
                Article
                S0375-07602013000300014 S0375-0760(13)06500314
                7c7c79e6-abbc-41ea-875a-da6a8b7340ac

                This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

                History
                : 17 January 2013
                : 29 May 2013
                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 13, Pages: 5
                Product

                SciELO Cuba

                Self URI: Texto completo solamente en formato PDF (ES)
                Categories
                PRESENTACIÓN DE CASO

                neuralgic amyotrophy,leptospirosis humana,vacunación,neuralgia amiotrófica,human leptospirosis,vaccination

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