Immune brachial plexus neuropathy

Suárez, Guillermo A.; Giannini, Caterina; Bosch, E. Peter; Barohn, Richard J.; Wodak, Jack; Ebeling, P; Anderson, Ronald; McKeever, Paul E.; Bromberg, Mark B.; Dyck, P. J.

doi:10.1212/wnl.46.2.559

Cited by 176 publications

(101 citation statements)

References 4 publications

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“…To our knowledge, the findings from only five nerve biopsies performed during attacks of neuralgic amyotrophy have been reported in two studies 75,76 . In one of these reports, two patients had typical idiopathic neuralgic amyotrophy, and their brachial plexus biopsy samples revealed conspicuous mononuclear perivascular inflammation in the epineurium and endoneurium; no other histopathological features were detailed.…”

Section: Diabetic Lumbosacral Radiculoplexus Neuropathymentioning

confidence: 96%

The nonsystemic vasculitic neuropathies

2017

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The vasculitides are a clinically diverse group of diseases with the histopathological signature of blood vessel-centred inflammation that results in vascular damage and ischaemic injury to the affected tissues 1 . Vasculitis can be caused by drugs, infections and cancers, but diagnostic workup reveals no trigger in most patients. Autoimmune mechanisms are active in all vasculitides, except those caused by direct infection of vessel walls. Most vasculitides are systemic and involve multiple organs and tissues. Those affecting small-to medium-sized vessels often manifest with a vasculitic neuropathy. For example, neuropathies occur in 60-70% of patients with polyarteritis nodosa, eosinophilic granulomatosis with polyangiitis and cryoglobulinaemic vasculitis, and 40-50% of those with microscopic polyangiitis and rheumatoid vasculitis 2 .Vasculitic neuropathy can also occur without systemic vasculitis. This single-organ vasculitis of the PNS has commonly been referred to as nonsystemic vasculitic neuropathy (NSVN), but other forms of clinically isolated PNS vasculitis are now recognized and can be considered variants of NSVN 3 . Although NSVN is classically an acute, relapsing, multifocal neuropathy, it can present as a slowly progressive neuropathy without distinct asymmetries. As such, it should be considered as a possible cause of any progressive axonal neur opathy. NSVN can be diagnosed only with a nerve biopsy, with or without a concomitant muscle or skin biopsy; as nerve biopsies are seldom performed, the condition is under-recognized. The disorder must be distinguished from many other causes of multifocal neuropathy, the definition of which is itself not standardized and requires clarification.A Peripheral Nerve Society guideline group reviewed the literature on vasculitic neuropathy and published consensus recommendations on the classification, diagnosis and immunosuppressive treatment of NSVN in 2010 (REF. 4) Abstract | Nonsystemic vasculitic neuropathy (NSVN) is an under-recognized single-organ vasculitis of peripheral nerves that can only be diagnosed with a nerve biopsy. A Peripheral Nerve Society guideline group published consensus recommendations on the classification, diagnosis and treatment of NSVN in 2010, and new diagnostic criteria for vasculitic neuropathy were developed by the Brighton Collaboration in 2015. In this Review, we provide an update on the classification, diagnosis and treatment of NSVN. NSVN subtypes include Wartenberg migratory sensory neuropathy and postsurgical inflammatory neuropathy. Variants include diabetic radiculoplexus neuropathy and -arguably -neuralgic amyotrophy. NSVN with proximal involvement is sometimes termed nondiabetic lumbosacral radiculoplexus neuropathy. Cutaneous polyarteritis nodosa and other skin-nerve vasculitides overlap with NSVN clinically. Three patterns of involvement in NSVN have been identified: multifocal neuropathy, distal symmetric polyneuropathy, and overlapping multifocal neuropathy (asymmetric polyneuropathy). These patterns lack standard defi...

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Section: Diabetic Lumbosacral Radiculoplexus Neuropathymentioning

confidence: 96%

The nonsystemic vasculitic neuropathies

2017

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“…17(p315) When evaluating a patient for possible PTS, the history should include a search for recent infection (including Epstein-Barr, Varicella Zoster, Dengue, and Hepatitis E viruses), immunization (tetanus toxoid, influenza), recent childbirth, prescribed medication (antiepileptics, antibiotics, immunosuppressants, antiretrovirals, and botulinum toxin), and all forms of surgery. 6,12,16,17 All of these are well documented to precede the onset of an attack of PTS and offer a valuable pointer toward the diagnosis if present, as was the case for surgery (37%) and infection (5%) in our series. Having decided upon a possible diagnosis of PTS, MRI imaging can be of use in ruling out alternative pathologies, including shoulder pathology or extrinsic nerve compression.…”

Section: Discussionmentioning

confidence: 58%

“…14,18 Effective treatment for PTS remains a distant ideal, but recent works have concentrated on abrogating the disease course with steroid and immunoglobulin therapy. 16,21 In a recent Cochrane update on PTS treatment, only 1 openlabel retrospective study demonstrated a possible benefit from steroid administration if given within 1 month of symptom development, with earlier pain resolution and faster recovery in some patients. 20 The high cost and associated problems with insurance reimbursement precluded the use of pharmacological agents such as immunomodulators in our series.…”

Section: Discussionmentioning

confidence: 99%

“…10 The syndrome is characterized by spontaneous pain arising around the shoulder girdle that typically lasts between 3 to 4 weeks, followed by focal weakness and subsequent muscle wasting that lasts for several months before a variable recovery. 16,17,19 The pattern of nerve involvement is highly variable but typically affects the upper and middle trunks of the brachial plexus with a reported predilection for the suprascapular and long thoracic nerves. 15 Since its original description, PTS (synonymous with brachial plexus neuritis or acute brachial neuropathy) has been found to be a relatively rare condition with an incidence of 2 to 3 cases per 100 000 per year.…”

Section: Introductionmentioning

confidence: 99%

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Parsonage-Turner Syndrome

Milner

Kumar

Iyer

et al. 2016

Hand (New York, N,Y.)

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Background: Parsonage-Turner Syndrome (PTS) is a rare but serious condition characterized by spontaneous paresis of the upper extremity, typically lasting several months with variable recovery. With little literature on the behavior of PTS from a hand surgeon's perspective, accurate diagnosis and subsequent counseling of patients with PTS can be challenging. Methods: This study is a retrospective evaluation of the clinical features of all PTS patients seen over a 9-year period. Data was collected for gender, side affected, handedness, inciting event, clinical presentation, nerve involvement, time taken for recovery, and extent of recovery. Results: Thirty-eight adult cases of PTS were identified, representing an incidence of 0.34 per 1000. Fifty-five percent were female, with predomination of right handedness and dominant upper extremity involvement (60% and 58%, respectively). There was an inciting incident identified in 42% of cases, and 37% of these involved surgery. Twenty-nine percent of cases presented without experiencing typical neuropathic pain. There was a predomination of anterior interosseous nerve (AIN) or posterior interosseous nerve (PIN) involvement (42% overall). Only 44% of patients achieved a complete recovery, taking a mean duration of 10 months. Conclusions: This study highlights both the rarity and atypical spectrum of clinical presentation of PTS, especially considering the more common involvement found for AIN and PIN. This highlights the likelihood that patients presenting with PTS to a hand surgical practice may differ from those typically described in the literature. The poor rate of recovery is in line with other recent reports and contrasts with the more positive outcomes found in earlier studies.

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“…Um estudo mostrou que os linfócitos de pacientes acometidos apresentavam aumento da sua atividade proliferativa (isto é, a transformação de linfócitos pequenos em células maiores que são capazes de sofrer mitose) em culturas com extratos de diferentes nervos do plexo braquial e seus ramos, mas não em culturas com extratos de nervos dos plexos sacrais. 18 Dois estudos mostraram infiltrados mononucleares inflamatórios em biópsias do plexo braquial, 19,20 ao passo que outro estudo mostrou aumento de anticorpos fixadores do complemento para a mielina do nervo periférico, na fase aguda da doença em três pacientes. 21 A forma hereditária da SPT é autossômica dominante e é causada por mutações no gene septin 9 no cromossomo 17q23.…”

Section: Etiologiaunclassified

Síndrome de Parsonage-Turner

Cavalheiro

Costa

Caetano

et al. 2016

Rev. Fac. Ciênc. Méd. Sorocaba

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Parsonage-Turner syndrome RESUMOA síndrome de Parsonage-Turner, neuralgia amiotrófica ou neurite do plexo braquial é uma síndrome rara, cuja causa é desconhecida. Os nervos supraescapular, axilar e seus músculos correspondentes são afetados com maior frequência. A doença apresenta uma ampla gama de manifestações clínicas, e os pacientes geralmente apresentam-se aos médicos de diferentes especialidades com queixa de dor intensa, de início súbito no ombro, que irradia para o braço ou pescoço e dura por horas ou semanas. Quando a dor desaparece, desenvolve-se uma paralisia flácida com fraqueza e atrofia muscular associada à perda sensorial da cintura escapular. O diagnóstico preciso pode ser desafiador e requer uma história completa e exame físico detalhado. Estudos de velocidade de condução nervosa e imagem auxiliam na avaliação. O tratamento consiste no controle sintomático. Os sintomas podem persistir durante mais do que um ano, mas a maioria dos pacientes evolui para resolução ao longo do tempo. Palavras-chave: neurite do plexo braquial; plexo braquial; inflamação. ABSTRACTThe Parsonage-Turner syndrome, amyotrophic neuralgia or neuritis of the brachial plexus is a rare syndrome, of unknown cause. The more frequently affected structures are the suprascapular, axillary nerves and their corresponding muscles. The disease has a wide range of clinical manifestations, and patients usually come to physicians of different specialties with intense and sudden pain complaints in the shoulder, radiating to the arm or neck and that lasts for hours or weeks. When the pain disappears, the patient develops a flaccid paralysis and muscle weakness associated with sensory loss of the shoulder girdle. Accurate diagnosis can be challenging and requires a complete history and detailed physical examination. Nerve conduction velocity and imaging studies help to evaluate the disease. Treatment consists of symptomatic control. The symptoms can persist for more than one year, but most patients recover over time.

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Immune brachial plexus neuropathy

Abstract: 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.

Cited by 176 publications

References 4 publications

The nonsystemic vasculitic neuropathies

The nonsystemic vasculitic neuropathies

Parsonage-Turner Syndrome

Síndrome de Parsonage-Turner

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