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Paraneoplastic Necrotizing Autoimmune Myopathy in a Patient Undergoing Laparoscopic Pancreatoduodenectomy for Distal Cholangiocarcinoma
A 73-year-old male presented with jaundice and severe muscle weakness. He was diagnosed with distal cholangiocarcinoma and paraneoplastic necrotizing autoimmune myopathy (NAM). Treatment of NAM consisted of dexamethasone pulse therapy, prednisone, and single-dose intravenous immunoglobulin. The distal cholangiocarcinoma was resected through a total laparoscopic pancreatoduodenectomy. After hospital discharge, muscle strength initially increased postoperatively; however, pneumonia resulted in the deterioration of his general condition and death 5 months after the diagnosis of paraneoplastic NAM.Case Rep Gastroenterol 2016;10:525 –530
Source: Case Reports in Gastroenterology - October 6, 2016 Category: Gastroenterology Source Type: research

Can Quantitative Muscle Strength and Functional Motor Ability Differentiate the Influence of Age and Corticosteroids in Ambulatory Boys with Duchenne Muscular Dystrophy?
Conclusion The baseline data analysis of this natural history study indicates that the outcomes measures utilized in this study were sensitive to the age related differences in strength and motor function that are characteristic of disease progression boys with DMD; however treatment effects were less likely to be identified. These findings reflect the difficulty inherent in obtaining the statistical power needed to substantiate intervention efficacy in the small, heterogeneous samples sizes that are characteristic of DMD clinical studies. Isokinetic dynamometry revealed variability in the muscles affected, which has been ...
Source: PLOS Currents Muscular Dystrophy - July 8, 2016 Category: Neurology Authors: cbuckon Source Type: research

Corticosteroid Monotherapy Is Usually Insufficient Treatment for Idiopathic Inflammatory Myopathy
Idiopathic inflammatory myopathies (IIM) are a rare group of autoimmune diseases characterized by muscle inflammation. High-dose corticosteroids are conventionally used as the first-line therapy for IIM, but early introduction of noncorticosteroid immunosuppressive agents has become increasingly common in recent years despite a paucity of compelling evidence to support this. Here, we systematically analyze therapeutic practice patterns of IIM in a large municipal safety net medical center. We conducted a retrospective chart review of all adult patients attending rheumatology clinics at the Cook County Health and Hospital S...
Source: American Journal of Therapeutics - September 1, 2015 Category: Drugs & Pharmacology Tags: Original Articles Source Type: research

Efficacy of Anakinra in Refractory Adult-Onset Still's Disease: Multicenter Study of 41 Patients and Literature Review
Abstract: Adult-onset Still's disease (AOSD) is often refractory to standard therapy. Anakinra (ANK), an interleukin-1 receptor antagonist, has demonstrated efficacy in single cases and small series of AOSD. We assessed the efficacy of ANK in a series of AOSD patients. Multicenter retrospective open-label study. ANK was used due to lack of efficacy to standard synthetic immunosuppressive drugs and in some cases also to at least 1 biologic agent. Forty-one patients (26 women/15 men) were recruited. They had a mean age of 34.4 ± 14 years and a median [interquartile range (IQR)] AOSD duration of 3.5 [2–6] years befor...
Source: Medicine - September 1, 2015 Category: Internal Medicine Tags: Research Article: Observational Study Source Type: research

Treatment for inclusion body myositis.
CONCLUSIONS: Trials of interferon beta-1a and MTX provided moderate-quality evidence of having no effect on the progression of sporadic inclusion body myositis. Overall trial design limitations including risk of bias, low numbers of participants, and short duration make it difficult to say whether or not any of the drug treatments included in this review were effective. An open trial of ATG combined with MTX versus MTX provided very low-quality evidence in favour of the combined therapy based on the percentage change data given. We were unable to draw conclusions from trials of IVIg, oxandrolone, and AZA plus MTX versus MT...
Source: Cochrane Database of Systematic Reviews - July 3, 2015 Category: Journals (General) Authors: Rose MR, Jones K, Leong K, Walter MC, Miller J, Dalakas MC, Brassington R, Griggs R Tags: Cochrane Database Syst Rev Source Type: research

Pulse Dose Methylprednisolone Therapy for Adult Idiopathic Inflammatory Myopathy
Idiopathic inflammatory myopathies (IIM) are a rare group of autoimmune diseases characterized by muscle inflammation. Typically high-dose daily oral corticosteroids are used as the first-line therapy of IIM. Pulse dose intravenous methylprednisolone (IVMP) has been used for serious or refractory cases. Here, we systematically analyze the therapeutic effect of pulse dose IVMP in patients with IIM in a large municipal safety net medical center and review the literature on pulse IVMP in adult IIM. We conducted a retrospective chart review of patients who were diagnosed with IIM in the rheumatology clinics of the Cook County ...
Source: American Journal of Therapeutics - July 1, 2015 Category: Drugs & Pharmacology Tags: Original Articles Source Type: research

Cystinosis associated vasculopathy masquerading as atypical migraine headache (P1.032)
Conclusions: Cystinosis, especially if untreated, can lead to neurological complications, more commonly distal myopathy and, rarely as our patient suffered, cerebral vasculopathy. Identifying this vasculopathy is essential and can be treatable, thus preventing further neurologic deterioration.Disclosure: Dr. Smith has nothing to disclose. Dr. Kushlaf has nothing to disclose.
Source: Neurology - April 8, 2015 Category: Neurology Authors: Smith, D., Kushlaf, H. Tags: Cerebrovascular Disease and Interventional Neurology: Case Reports Source Type: research

Neuromyotonia: an Unusual Presentation in Inflammatory Myopathy (P2.045)
CONCLUSIONS. Neuromyotonia and inflammatory myopathy is a very rare association and in our case the patient had atypical clinical manifestations. The diagnosis of neuromyotonia was made with neurophysiologic study. The patient received immunosuppressive treatment, whereas she did not have other clinical manifestations of neuromyotonia. Neuromyotonia is an unusual presentation in inflammatory myopathy.Disclosure: Dr. Scola has nothing to disclose. Dr. Ducci has nothing to disclose. Dr. Lorenzoni has nothing to disclose. Dr. Werneck has nothing to disclose.
Source: Neurology - April 8, 2015 Category: Neurology Authors: Scola, R., Dal-Pra Ducci, R., Lorenzoni, P., Werneck, L. Tags: Muscle Disease: Genetics, Pathogenesis, and Pathology Source Type: research

Necrotizing Autoimmune Myopathy: Comparison of Clinical Features and Treatment Outcomes among Different Etiologies (S17.002)
CONCLUSIONS: NAM is usually idiopathic and often accompanied by SRP-IgG; statin therapy is a common association. SRP-IgG detection did not predict clinical outcome. Treatment with corticosteroids or IVIG alone did not control disease in most patients. Early aggressive immunotherapy promotes better outcome. Relapse risk is high during medication taper.Disclosure: Dr. Kassardjian has nothing to disclose. Dr. Lennon stands to receive royalty payments for commercial assays to detect of Aquaporin 4-specific Autoantibody. Dr. Milone has nothing to disclose.
Source: Neurology - April 8, 2015 Category: Neurology Authors: Kassardjian, C., Lennon, V., Milone, M. Tags: Muscle and Anterior Horn Cell Diseases: Mechanisms and Biomarkers Source Type: research

Clinical course and treatment of anti-HMGCR antibody-associated necrotizing autoimmune myopathy
Conclusions: Recognition of HMGCR antibody–associated NAM is important because these patients are responsive to immunosuppression, and early multiagent therapy and a slow and cautious approach to withdrawing steroids may improve outcomes.
Source: Neurology Neuroimmunology and Neuroinflammation - April 2, 2015 Category: Neurology Authors: Ramanathan, S., Langguth, D., Hardy, T. A., Garg, N., Bundell, C., Rojana-Udomsart, A., Dale, R. C., Robertson, T., Mammen, A. L., Reddel, S. W. Tags: Article Source Type: research

Use of Prednisone With Abiraterone Acetate in Metastatic Castration-Resistant Prostate Cancer.
Abstract Abiraterone acetate, a prodrug of the CYP17A1 inhibitor abiraterone that blocks androgen biosynthesis, is approved for treatment of patients with metastatic castration-resistant prostate cancer (mCRPC) in combination with prednisone or prednisolone 5 mg twice daily. This review evaluates the basis for the effects of prednisone on mineralocorticoid-related adverse events that arise because of CYP17A1 inhibition with abiraterone. Coadministration with the recommended dose of glucocorticoid compensates for abiraterone-induced reductions in serum cortisol and blocks the compensatory increase in adrenocorticot...
Source: The Oncologist - October 31, 2014 Category: Cancer & Oncology Authors: Auchus RJ, Yu MK, Nguyen S, Mundle SD Tags: Oncologist Source Type: research

CNS Toxoplasmosis: A Serious Complication Of Immunotherapy In The Neuromuscular Patient (P6.102)
Conclusions: Immunotherapy with medication such as MM can cause a devastating CNS toxoplasmosis in non-HIV patients with neuromuscular disorders. In contrast to the HIV infected patients, treatment is ineffective implying a different pathogenesis. Early consideration and recognition of this complication is important to possibly prevent unfavorable outcome.Disclosure: Dr. Bernardo has nothing to disclose. Dr. Chahin has nothing to disclose.
Source: Neurology - April 9, 2014 Category: Neurology Authors: Bernardo, D., Chahin, N. Tags: Neuromuscular Disease Source Type: research

IgG4-Related Disease Manifesting as Middle Cranial Fossa Mass: A Case Report (P5.165)
A 62 year old woman presented with progressive right hearing loss and a right middle ear mass. Resection showed lymphoblastic inflammation. 8 months later she presented with right optic neuritis, right facial nerve paresis. Brain MRI demonstrated a dural-based homogenous enhancing mass extending from cavernous sinus into middle cranial fossa. Review of available biopsy showed plasmacytic infiltration, storiform fibrosis and phlebitis with >50 IgG4-staining cells/HPF. Serum IgG4 level was normal. Cerebrospinal fluid (CSF) revealed mild lymphocytic pleocytosis, elevated IgG index (2.2, normal <0.66), and unremarkable c...
Source: Neurology - April 9, 2014 Category: Neurology Authors: Malla, P., Zachariah, J., Katirji, B. Tags: CNS Diseases and Differential Diagnosis Source Type: research

Anti-synthetase syndrome: anti-PL-7, anti-PL-12 and anti-EJ
CONCLUSIONS: ASS (anti-PL-7, PL-12 and EJ) was found to predominantly affect white women. Although the autoantibodies described in the present study are more related to pulmonary than joint involvement, our patients showed a significant percentage of both types of involvement and a high percentage of myopathy. We also observed a low mortality rate.
Source: Revista Brasileira de Reumatologia - November 15, 2013 Category: Rheumatology Source Type: research

Teaching NeuroImages: Hydroxychloroquine-induced vacuolar myopathy
A 58-year-old woman with long-standing mixed connective tissue disorder had proximal leg weakness for 4 months. She had been treated with 400 mg/day of hydroxychloroquine and varying doses of prednisone over 15 years. Creatine kinase was 600 U/mL. MRI of quadriceps showed edema and its biopsy revealed myriad acid-phosphatase–positive autophagic vacuoles indicating increased lysosomal activity (figure). Hydroxychloroquine induces autophagy by reducing lysosomal acidity.1 Autophagic vacuolar myopathy can be seen with chloroquine or colchicine therapy or in inherited disorders (α-glucosidase deficiency, Danon dise...
Source: Neurology - June 3, 2013 Category: Neurology Authors: Ghosh, P. S., Swift, D., Engel, A. G. Tags: Autoimmune diseases, Muscle disease, Other toxicology RESIDENT AND FELLOW SECTION Source Type: research

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