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antisynthetase syndrome

Angela Ceribelli, Natasa Isailovic, Maria De Santis, Elena Generali, Micaela Fredi, Ilaria Cavazzana, Franco Franceschini, Luca Cantarini, Minoru Satoh, Carlo Selmi
This study aims to characterize myositis-specific antibodies in a well-defined cohort of patients with idiopathic inflammatory myopathy and to determine their association with cancer. Sera from 40 patients with polymyositis, dermatomyositis, and controls were tested by protein and RNA immunoprecipitation to detect autoantibodies, and immunoprecipitation-Western blot was used for anti-MJ/NXP-2, anti-MDA5, and anti-TIF1γ/α identification. Medical records were re-evaluated with specific focus on cancer. Anti-MJ/NXP-2 and anti-TIF1γ/α were the most common antibodies in dermatomyositis...
October 20, 2016: Clinical Rheumatology
Leah J Witt, James J Curran, Mary E Strek
Anti-synthetase syndrome is an autoimmune condition, characterized by antibodies directed against an aminoacycl transfer RNA synthetase along with clinical features that can include interstitial lung disease, myositis, Raynaud's phenomenon, and arthritis. There is a higher prevalence and increased severity of interstitial lung disease in patients with anti-synthetase syndrome, as compared to dermatomyositis and polymyositis, inflammatory myopathies with which it may overlap phenotypically. Diagnosis is made by a multidisciplinary approach, synthesizing rheumatology and pulmonary evaluations, along with serologic, radiographic, and occasionally muscle and/or lung biopsy results...
September 2016: Clinical Pulmonary Medicine
Ana C Zamora, Sumedh S Hoskote, Beatriz Abascal-Bolado, Darin White, Christian W Cox, Jay H Ryu, Teng Moua
BACKGROUND: Interstitial lung disease (ILD) is a common extra-muscular manifestation of antisynthetase (AS) syndrome. ILD prevalence is higher with anti-Jo-1 antibody positivity. Data on long-term outcomes in these patients are lacking. METHODS: Over 15 years, we identified subjects with anti-Jo-1 positive AS syndrome and ILD. Demographics, pulmonary function testing (PFT), high-resolution computed tomography (HRCT), histopathology, and long-term survival were analyzed...
September 2016: Respiratory Medicine
Joanna Collison
No abstract text is available yet for this article.
October 2016: Nature Reviews. Rheumatology
Baptiste Hervier, Mikaël Perez, Yves Allenbach, Hervé Devilliers, Fleur Cohen, Yurdagül Uzunhan, Hanane Ouakrim, Karim Dorgham, Jean-François Méritet, Elisabeth Longchampt, Werner Stenzel, Isabelle Cremer, Olivier Benveniste, Vincent Vieillard
Antisynthetase syndrome (aSS) is characterized by the association of interstitial lung disease and myositis with anti-tRNA synthetase autoantibodies. Immune mechanisms leading to aSS could be initiated in the lungs, but the role of NK cells has not yet been studied. Both extensive NK cell phenotype and functions were compared between 33 patients and 26 controls. Direct and redirected polyfunctionality assays (degranulation and intracellular production of TNF-α and IFN-γ) were performed spontaneously or after IL-12 plus IL-18 stimulation in the presence of K562 or P815 target cells, respectively...
September 1, 2016: Journal of Immunology: Official Journal of the American Association of Immunologists
Gurveen Malhotra, Nitya Ramreddy, Serafin Chua, Mira Iliescu, Tanjeev Kaur
Antisynthetase (AS) syndrome is a major subgroup of inflammatory myopathies seen in a minority of patients with dermatomyositis and polymyositis. Although it is usually associated with elevated creatine phosphokinase level, some patients may have amyopathic dermatomyositis (ADM) like presentation with predominant skin involvement. Interstitial lung disease (ILD) is the main pulmonary manifestation and may be severe thereby determining the prognosis. It may rarely present with a very aggressive course resulting in acute respiratory distress syndrome (ARDS)...
2016: Case Reports in Critical Care
Seo-Hyun Kim, I-Nae Park
Antisynthetase syndrome has been recognized as an important cause of autoimmune inflammatory myopathy in a subset of patients with polymyositis and dermatomyositis. It is associated with serum antibody to aminoacyl-transfer RNA synthetases and is characterized by a constellation of manifestations, including fever, myositis, interstitial lung disease, mechanic's hand-like cutaneous involvement, Raynaud phenomenon, and polyarthritis. Lung disease is the presenting feature in 50% of the cases. We report a case of a 60-year-old female with acute respiratory distress syndrome (ARDS), which later proved to be an unexpected and initial manifestation of anti-Jo-1 antibody-positive antisynthetase syndrome...
July 2016: Tuberculosis and Respiratory Diseases
Yuko Waseda, Takeshi Johkoh, Ryoko Egashira, Hiromitsu Sumikawa, Keigo Saeki, Satoshi Watanabe, Ryo Matsunuma, Hazuki Takato, Yukari Ichikawa, Yasuhito Hamaguchi, Akira Shiraki, Yoshinao Muro, Masahide Yasui, Helmut Prosch, Christian Herold, Kazuo Kasahara
OBJECTIVES: To describe the pulmonary CT findings in patients with anti-ARS-antibody-positive interstitial lung disease (anti-ARS-ILD) METHODS: The CT findings of 64 patients with anti-ARS-ILD were retrospectively reviewed. The images were retrospectively reviewed independently by 2 chest radiologists, and the final decision on the CT findings was made by a third chest radiologist. RESULTS: There were 16 male and 48 female patients, aged 54.2±13.4 years. Sixteen patients had anti Jo-1, 24 had anti-EJ, 9 had anti-PL-7, 7 had anti-PL-12, 5 had anti-KS, and 3 had anti-OJ antibodies...
August 2016: European Journal of Radiology
Khaled Eissa, Jaime Palomino
Interstitial lung disease (ILD) is a major cause of morbidity and mortality in patients with idiopathic inflammatory myopathies (IIM). Glucocorticoids are the initial standard treatment. However, many patients fail to respond and continue to progress despite treatment with high dose glucocorticoids. The efficacy of rituximab has been suggested in case reports and case series of refractory antisynthetase (AS) syndrome, but data on patients without auto-antibodies or with rapidly progressive ILD are scarce. We report a case of rapidly progressive dermatomyositis (DM) associated ILD treated successfully with B-cell depletion therapy...
May 2016: Journal of the Louisiana State Medical Society: Official Organ of the Louisiana State Medical Society
Maki Tateyama, Satoshi Shibuya, Hitoshi Sato, Kazuo Fujihara, Masashi Aoki
No abstract text is available yet for this article.
August 2016: Neuromuscular Disorders: NMD
I E Lundberg, F W Miller, A Tjärnlund, M Bottai
The idiopathic inflammatory myopathies (IIMs) are a heterogeneous group of diseases, collectively termed myositis, sharing symptoms of muscle weakness, fatigue and inflammation. Other organs are frequently involved, supporting the notion that these are systemic inflammatory diseases. The IIMs can be subgrouped into dermatomyositis, polymyositis and inclusion body myositis. The myositis-specific autoantibodies (MSAs) identify other and often more distinct clinical phenotypes, such as the antisynthetase syndrome with antisynthetase autoantibodies and frequent interstitial lung disease and anti-SRP and anti-HMGCR autoantibodies that identify necrotizing myopathy...
July 2016: Journal of Internal Medicine
S Kobayashi, H Shoda, Y Seri, Y Nagafuchi, O Sasaki, K Kubo, K Fujio, K Yamamoto
No abstract text is available yet for this article.
June 16, 2016: Scandinavian Journal of Rheumatology
Jutta Bauhammer, Norbert Blank, Regina Max, Hanns-Martin Lorenz, Ulrich Wagner, Dietmar Krause, Christoph Fiehn
OBJECTIVE: Rituximab (RTX) has been used successfully for the treatment of severe Jo1 antibody-associated antisynthetase syndrome. The aim of this retrospective study was to evaluate the effect of RTX in severe Jo1 antisynthetase syndrome and determine predictive factors for response. METHODS: There were 61 patients with Jo1 antisynthetase syndrome identified; 18 of these received RTX. One patient was lost to followup. The remaining 17 patients and 30 out of 43 patients who were treated with conventional immunosuppressive (IS) drugs were followed for a mean of 35 months and 84 months, respectively...
August 2016: Journal of Rheumatology
Mineto Ota, Mari Kihara, Akito Takamura, Hitoshi Kohsaka
  Antisynthetase syndrome is characterized by the presence of anti-aminoacyl-tRNA synthetase antibodies and characteristic clinical features. We report an anti-EJ antibody-positive case presenting an ILD with slight hyperkeratotic skin changes on the fingertips that appeared simultaneously with respiratory symptoms. We suspected those skin changes of a disease manifestation of antisynthetase syndrome, and thus investigated anti-synthetase antibodies. This case implies that broader spectrum of the patients should fall in antisynthetase syndrome even though the present diagnostic criteria call for mechanic's hand as a skin manifestation...
2016: Nihon Rinshō Men'eki Gakkai Kaishi, Japanese Journal of Clinical Immunology
Mehwish Asif Qureshi, Edward Hoey, Timothy Fletcher, Zubair Ahmed
The case is about a 48-year-old man with one week history of progressively worsening shortness of breath and reduced exercise tolerance. Laboratory investigations revealed raised ESR, creatine kinase (CK) and positive anti-aminoacyl-transfer RNA synthetase. High resolution computed tomography (HRCT) chest showed prominent mediastinal lymph nodes and scattered lung nodules. These findings along with clinical features and investigations confirmed the diagnosis of antisynthetase syndrome (ASS). The radiological investigations proved to be the key step in diagnosis and management of this case...
April 2016: Quantitative Imaging in Medicine and Surgery
Ernesto Trallero-Araguás, Josep María Grau-Junyent, Anne Labirua-Iturburu, Francisco José García-Hernández, Manuel Monteagudo-Jiménez, Guadalupe Fraile-Rodriguez, Iñigo Les-Bujanda, Mónica Rodriguez-Carballeira, Luis Sáez-Comet, Albert Selva-O'Callaghan
OBJECTIVE: To evaluate the clinical manifestations, long-term clinical outcome and longitudinal pulmonary function in a large cohort of Spanish patients with anti-Jo1 antibodies. METHODS: We retrospectively analyzed the clinical data and lung function parameters of 148 anti-Jo1 patients recruited from a multicentre registry including 18 Spanish hospitals. A composite endpoint was defined, comprising death due to respiratory failure directly related to antisynthetase syndrome (ASS), the need for long-term oxygen therapy or lung transplantation...
October 2016: Seminars in Arthritis and Rheumatism
Hj Gayathri Devi, Md Majeed Pasha, Mantha Sathya Padmaja, Sujith Halappa
Anti-Synthetase Syndrome (ASS) is a rare autoimmune disorder characterized by Interstitial Lung Disease (ILD), inflammatory myositis, fever, Raynaud's phenomenon, mechanic's hand, and inflammatory polyarthritis in the setting of antibodies against amino acyl-transfer RNA synthetases, with anti-Jo-1 antibody being the most common. It can sometimes present as interstitial lung disease without any other expression of the syndrome. Clinical and radiological features can be similar to atypical pneumonia and could be a challenge to diagnose at an early stage...
March 2016: Journal of Clinical and Diagnostic Research: JCDR
Charles Sharp, Melanie McCabe, Nick Dodds, Anthony Edey, Lloyd Mayers, Huzaifa Adamali, Ann B Millar, Harsha Gunawardena
OBJECTIVE: CTD-associated interstitial lung disease (ILD) often fails to respond to conventional immunomodulatory agents. There is now considerable interest in the use of rituximab in systemic autoimmune CTD in patients refractory to standard treatments. The aim of this study was to review the experience of North Bristol NHS Trust managing patients with CTD-associated ILD with rituximab and explore possible associations with treatment response. METHODS: We conducted a retrospective analysis of all patients who received rituximab under the Bristol CTD-ILD service, having failed to respond to other immunomodulatory treatments...
July 2016: Rheumatology
Katalin Szabó, Melinda Nagy-Vincze, Levente Bodoki, Katalin Hodosi, Katalin Dankó, Zoltán Griger
INTRODUCTION: In idiopathic inflammatory myopathies, the presence of anti-Jo-1 antibody defines a distinct clinical phenotype (myositis, arthritis, interstitial lung disease, Raynaud's phenomenon fever, mechanic's hands), called antisynthetase syndrome. AIM: To determine the demographic data as well as clinical, laboratory and terapeutical features of anti-Jo1 positive patients, followed by the department of the authors. METHOD: The medical records of 49 consecutive anti-Jo1 patients were reviewed...
April 10, 2016: Orvosi Hetilap
Helena Andersson, Trond Mogens Aaløkken, Anne Günther, Georg Karl Mynarek, Torhild Garen, May Brit Lund, Øyvind Molberg
OBJECTIVE: Interstitial lung disease (ILD) is a major component of the antisynthetase syndrome, but quantitative data on longterm pulmonary outcome in antisynthetase syndrome are limited. In this study, the main aims were to compare pulmonary function tests (PFT) and the 6-min walking distance (6MWD) between patients with antisynthetase syndrome and healthy sex- and age-matched controls, to evaluate the extent of ILD by lung high-resolution computed tomography (HRCT), and to assess correlations between PFT measures and ILD extent...
June 2016: Journal of Rheumatology
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