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Complex regional pain syndrome and dysautonomia in a 14‐year‐old girl responsive to therapeutic plasma exchange
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Jeanne E. Hendrickson Emma T. Hendrickson Eric A. Gehrie Davinder Sidhu Gerd Wallukat Ingolf Schimke Christopher A. Tormey 《Journal of clinical apheresis》2016,31(4):368-374
Reflex sympathetic dystrophy, also known as complex regional pain syndrome (CRPS), has recently been shown to be associated with autoantibodies against β2‐adrenergic and muscarinic M2 receptors. In addition to pain and sudomotor/vasomotor symptoms, dysautonomia is also observed in a subset of CRPS patients. Despite its severity, there are few effective therapies for CRPS described to date. We report a case of a 14‐year‐old girl with CRPS of her right leg and dysautonomia (gastroparesis, postural tachycardia) refractory to multiple therapies, successfully treated with therapeutic plasma exchange (TPE) with albumin replacement. The patient, who has serum anti β2‐adrenergic and muscarinic M2 receptor autoantibodies in addition to nicotinic acetylcholine receptor ganglionic autoantibodies, underwent an initial course of five TPEs over a 2‐week period. She demonstrated a clinical response to TPE as manifested by a rapid improvement in her fatigue and gastroparesis, with a gradual yet significant improvement in her leg pain and sudomotor/vasomotor flares. Following the loading procedures, the patient was treated with rituximab. She continues to require periodic TPE to maintain a remission, with additional immunosuppression being considered long term. Although further studies are needed, TPE (in combination with immunosuppression) may be an appropriate therapy for CRPS patients with detectable autoantibodies, as it is for better characterized diseases with autoantibodies against neuronal surface receptors such as myasthenia gravis or Lambert Eaton myasthenic syndrome. J. Clin. Apheresis 31:368–374, 2016. © 2015 Wiley Periodicals, Inc. 相似文献
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Liane A. Arcinas Weiang Yan Davinder S. Jassal Michael P. Love Michael H. Yamashita Basem Elbarouni 《The Canadian journal of cardiology》2018,34(12):1688.e5-1688.e7
We describe a case of a 54-year-old woman who had a right cardiac mass found on coronary angiography. Echocardiography, computed tomography, and cardiac magnetic resonance imaging characterized it as a thrombosed giant right coronary artery aneurysm. This was confirmed on pathology. We present the role of multimodality cardiovascular imaging in the diagnosis and characterization of a giant coronary artery aneurysm. 相似文献
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OX40/OX40 ligand interactions in T-cell regulation and asthma 总被引:1,自引:0,他引:1
The OX40 receptor is preferentially expressed by T cells, and its cognate ligand OX40L is primarily expressed by antigen-presenting cells such as dendritic cells following activation by thymic stromal lymphopoietin (TSLP). TSLP is released by the bronchial epithelium, airway smooth muscle, and some inflammatory cells in response to numerous insults such as allergens, viruses, and physical damage. OX40L is a costimulatory molecule that plays a sentinel role in the adaptive immune response by promoting T helper (Th) 2 polarization of naive T cells within the lymph node. These polarized T cells produce Th2 cytokines such as IL-4, IL-5, and IL-13, which have been implicated particularly in allergic eosinophilic asthma. Animal models have positioned both TSLP and OX40/OX40L as critical in the development of airway inflammation and hyperreactivity. In human disease, there is good evidence that TSLP is upregulated in asthma, but there are limited data to demonstrate overexpression of OX40 or OX40L in disease. Targeting the OX40/OX40L axis or TSLP presents a novel therapeutic strategy that has the potential of modifying the disease process and, therefore, impacting on its natural history. Whether this approach can demonstrate efficacy in established disease rather than at disease onset is unknown. Biologic therapies directed toward OX40/OX40L are in early phases of development, and results from these studies are eagerly awaited. 相似文献
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Colish J Walker JR Elmayergi N Almutairi S Alharbi F Lytwyn M Francis A Bohonis S Zeglinski M Kirkpatrick ID Sharma S Jassal DS 《Chest》2012,141(3):674-681
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Ducas RA Philipp RK Jassal DS Wassef AW Weldon E Hussain F Schmidt C Khadem A Ducas J Grierson R Tam JW 《The Canadian journal of cardiology》2012,28(4):423-431