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cancer-associated myositis

C Calabrese, E Kirchner, K Kontzias, V Velcheti, L H Calabrese
Immunotherapy of cancer with checkpoint inhibitors has been associated with a spectrum of autoimmune and systemic inflammatory reactions known as immune-related adverse events (irAEs). Rheumatic irAEs are infrequently reported and extensively described. Here, we report our experience over an 18-month period with 15 patients evaluated in the rheumatology department for rheumatic irAEs. We identified 13 patients without pre-existing autoimmune disease (AID) who subsequently developed rheumatic irAEs, and two with established AID referred pre-emptively...
2017: RMD Open
Iago Pinal-Fernandez, Maria Casal-Dominguez, Julio A Huapaya, Jemima Albayda, Julie J Paik, Cheilonda Johnson, Leann Silhan, Lisa Christopher-Stine, Andrew L Mammen, Sonye K Danoff
Objective.: The aim was to study the prevalence, rate of appearance and severity of clinical features in patients with different anti-synthetase syndrome (ASyS) autoantibodies. Methods.: All Johns Hopkins Myositis Longitudinal Cohort subjects positive for any ASyS autoantibodies were included. Clinical information, including symptoms, signs, strength, creatine kinase concentrations and pulmonary function tests, were prospectively collected. The standardized mortality and cancer rates and the rate of appearance and intensity of the different organ manifestations were assessed using univariate and multivariate analysis and compared between ASyS autoantibodies...
March 4, 2017: Rheumatology
Maria Giovanna Danieli, Chiara Gelardi, Veronica Pedini, Francesco Logullo, Armando Gabrielli
The efficacy of subcutaneous immunoglobulin is reported in several neurological disorders and, more recently, its use has been extended to other inflammatory diseases, such as the idiopathic inflammatory myopathies, including polymyositis and dermatomyositis. Due to the rarity of these disorders, the role of immunoglobulin, administered intravenously or subcutaneously, remains unclear and poorly investigated. We report our experience about the use of subcutaneous immunoglobulin in myositis spectrum disorders, from idiopathic inflammatory myopathies to more complex conditions, such as overlap and cancer-associated myositis or pregnancy...
March 14, 2017: Current Rheumatology Reviews
Natalia L Gonzalez, Araya Puwanant, Angela Lu, Stanley M Marks, Saša A Živković
Immune checkpoint molecules are potent regulators of immunologic homeostasis that prevent the development of autoimmunity while maintaining self-tolerance. Inhibitors of immune checkpoint molecules are used as immunotherapy in the treatment of melanoma and different types of refractory cancer, and can trigger various autoimmune complications including myositis and myasthenia gravis. We describe a case of generalized myasthenia gravis induced by pembrolizumab and review 11 other cases. Five patients also had elevated serum CK levels ranging from 1200 to 8729 IU/L, and biopsy showed myositis in one...
March 2017: Neuromuscular Disorders: NMD
Eleni Tiniakou, Iago Pinal-Fernandez, Thomas E Lloyd, Jemima Albayda, Julie Paik, Jessie L Werner, Cassie A Parks, Livia Casciola-Rosen, Lisa Christopher-Stine, Andrew L Mammen
OBJECTIVE: To study disease severity and response to therapy in a large cohort of patients with anti-hydroxy-3-methylglutaryl-coenzyme A reductase (HMGCR)-associated myositis. METHODS: Muscle strength, creatine kinase levels and treatments were assessed in anti-HMGCR-positive patients at each clinical visit. Univariate and multivariate analyses were used to analyse the influence of clinical characteristics on strength and the change in strength over time. Whole exome sequencing was performed in a subset of patients...
January 17, 2017: Rheumatology
Jose Luis Ramírez-Bellver, Elena Macías, Claudia Bernárdez, Joaquín López-Robles, Maria Del Carmen Vegas-Sánchez, Jose Luis Díaz-Recuero, Hernán Quiceno, Luis Requena
BACKGROUND: Paraneoplastic syndromes consist of a group of disorders that are not related to the extension of the primary tumor or its metastases and that might be the first manifestation of a hidden neoplasm. It is a well-known association between dermatomyositis (DM) and cancer, especially gynecological tumors in women and lung cancer in men. METHODS: We describe the case of a 67-year-old male who developed muscular weakness and pruritic skin lesions. Skin biopsies were performed and histologic findings were consistent with DM...
January 2017: American Journal of Dermatopathology
Douglas B Johnson, Justin M Balko, Margaret L Compton, Spyridon Chalkias, Joshua Gorham, Yaomin Xu, Mellissa Hicks, Igor Puzanov, Matthew R Alexander, Tyler L Bloomer, Jason R Becker, David A Slosky, Elizabeth J Phillips, Mark A Pilkinton, Laura Craig-Owens, Nina Kola, Gregory Plautz, Daniel S Reshef, Jonathan S Deutsch, Raquel P Deering, Benjamin A Olenchock, Andrew H Lichtman, Dan M Roden, Christine E Seidman, Igor J Koralnik, Jonathan G Seidman, Robert D Hoffman, Janis M Taube, Luis A Diaz, Robert A Anders, Jeffrey A Sosman, Javid J Moslehi
Immune checkpoint inhibitors have improved clinical outcomes associated with numerous cancers, but high-grade, immune-related adverse events can occur, particularly with combination immunotherapy. We report the cases of two patients with melanoma in whom fatal myocarditis developed after treatment with ipilimumab and nivolumab. In both patients, there was development of myositis with rhabdomyolysis, early progressive and refractory cardiac electrical instability, and myocarditis with a robust presence of T-cell and macrophage infiltrates...
November 3, 2016: New England Journal of Medicine
Enrique J deAndrés-Galiana, Juan L Fernández-Martínez, Stephen T Sonis
INTRODUCTION: It has become clear that noise generated during the assay and analytical processes has the ability to disrupt accurate interpretation of genomic studies. Not only does such noise impact the scientific validity and costs of studies, but when assessed in the context of clinically translatable indications such as phenotype prediction, it can lead to inaccurate conclusions that could ultimately impact patients. We applied a sequence of ranking methods to damp noise associated with microarray outputs, and then tested the utility of the approach in three disease indications using publically available datasets...
December 2016: Journal of Biomedical Informatics
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...
February 2017: Clinical Rheumatology
Tomoko Noda, Masahiro Iijima, Seiya Noda, Shinya Maeshima, Hirotaka Nakanishi, Seigo Kimura, Haruki Koike, Shinsuke Ishigaki, Yohei Iguchi, Masahisa Katsuno, Gen Sobue
Objective An association has been reported between inflammatory myopathies (IMs), which include polymyositis (PM) and dermatomyositis (DM), and malignancy, and the concept of cancer-associated myositis (CAM) was recently proposed. We herein attempted to determine the features and etiologies of these myopathies. Methods We analyzed the gene expression levels via microarray and real-time quantitative reverse transcription polymerase chain reaction analyses to identify genes that were specifically upregulated or downregulated with suspected inflammatory involvement and verified the microarray data via an immunohistochemical (IHC) analysis in additional cases...
2016: Internal Medicine
Laura Nuño, Beatriz Joven, Patricia Carreira, Valentina Maldonado, Carmen Larena, Irene Llorente, Eva Tomero, María Carmen Barbadillo, Paloma García-de la Peña, Lucía Ruiz, Juan Carlos López-Robledillo, Henry Moruno, Ana Pérez, Tatiana Cobo-Ibáñez, Raquel Almodóvar, Leticia Lojo, Indalecio Monteagudo, María Jesús García-De Yébenes, Francisco Javier López-Longo
OBJECTIVE: To analyze clinical characteristics, survival and causes of death of patients diagnosed with autoimmune inflammatory myositis in the REMICAM registry from the Society of Rheumatology in the Community of Madrid (SORCOM). METHODS: Multicenter cohort of patients diagnosed with autoimmune inflammatory myopathy with follow-up between January 1980 and December 2014. A total of 313 variables concerning demographic, clinical and morbidity data were collected, and a comparison was performed between clinical subgroups...
September 5, 2016: Reumatología Clinica
Olivier Benveniste, Werner Stenzel, Yves Allenbach
PURPOSE OF REVIEW: Inflammatory myopathies are rare diseases. Their diagnosis criteria are historically based on their clinical phenotype (topography of the muscle weakness, presence of skin lesions and/or of extra-skin/muscle signs) and the presence of inflammatory infiltrates on muscle biopsy. However, the recent discovery of different myositis-specific antibodies (MSA) or myositis-associated antibodies (MAA) permitted to revisit these old classifications. This review covers recent findings in clinical and pathological phenotypes regarding prognosis, associated cancer and response to the treatment based on MSA/MAA categorization...
October 2016: Current Opinion in Neurology
Shin Ohta, Ki-Ichi Unoda, Hideto Nakajima, Soichiro Ikeda, Yasuhito Hamaguchi, Fumiharu Kimura
Myositis-specific autoantibodies (MSAs) are associated with myositis. Anti-nuclear matrix protein 2 (NXP-2) antibody was recently identified as a major MSA and was observed mostly in juvenile dermatomyositis. We report the case of a 44-year-old man who presented with myopathy with anti-NXP-2 antibody and large cell carcinoma of the lung. He was hospitalized because of myalgia and edema of limbs. Neurological examination revealed mild proximal-dominant weakness in all four extremities, and laboratory studies showed elevated creatine kinase level (6,432 IU/l)...
August 31, 2016: Rinshō Shinkeigaku, Clinical Neurology
Chikara Ebisutani, Isao Ito, Masanori Kitaichi, Naoya Tanabe, Michiaki Mishima, Seizo Kadowaki
We report the first case of non-specific interstitial pneumonia (NSIP) in a patient with cancer-associated myositis (CAM) that emerged along with the recurrence of the cancer. A 60-year-old woman, with a history of partial gastrectomy for gastric cancer 11 years ago, presented with exertional dyspnea with anti-Jo-1 antibody-positive myositis. Surgical lung biopsy showed NSIP with metastatic gastric cancer. Accordingly, her condition was diagnosed as CAM with cancer recurrence. In patients with a history of cancer, development of myositis may indicate cancer recurrence; therefore, careful observation would be necessary...
July 2016: Respiratory Investigation
Anna Masiak, Julia Kulczycka, Zenobia Czuszyńska, Zbigniew Zdrojewski
OBJECTIVES: Inflammatory myopathies are a group of idiopathic, heterogeneous systemic diseases affecting predominantly skeletal muscles, though they can also involve the skin and internal organs. The association between cancer and idiopathic inflammatory myopathies, particularly dermatomyositis, which is termed cancer-associated myositis (CAM), has been reported in the medical literature. A newly described autoantibody to a 155-kDa nuclear protein, identified as transcription intermediary factor 1-gamma (TIF1-γ), has proven useful for cancer screening in patients with dermatomyositis...
2016: Reumatologia
Ayumi Hida, Takenari Yamashita, Yuji Hosono, Manami Inoue, Kenichi Kaida, Masato Kadoya, Yusuke Miwa, Nobuyuki Yajima, Reika Maezawa, Satoko Arai, Kazuhiro Kurasawa, Kazuhiro Ito, Hiroyuki Shimada, Tomoko Iwanami, Masahiro Sonoo, Yuki Hatanaka, Shigeo Murayama, Ayumi Uchibori, Atsuro Chiba, Hitoshi Aizawa, Takayuki Momoo, Yoshiharu Nakae, Yasuhisa Sakurai, Yasushi Shiio, Hideji Hashida, Toshihiro Yoshizawa, Yoshio Sakiyama, Aya Oda, Kiyoharu Inoue, Sousuke Takeuchi, Nobue K Iwata, Hidetoshi Date, Naoki Masuda, Takashi Mikata, Yasufumi Motoyoshi, Yoshikazu Uesaka, Meiko Hashimoto Maeda, Ran Nakashima, Shoji Tsuji, Shin Kwak, Tsuneyo Mimori, Jun Shimizu
OBJECTIVE: We aimed to analyze the clinical and histopathologic features of cancer-associated myositis (CAM) in relation to anti-transcriptional intermediary factor 1 γ antibody (anti-TIF1-γ-Ab), a marker of cancer association. METHODS: We retrospectively studied 349 patients with idiopathic inflammatory myopathies (IIMs), including 284 patients with pretreatment biopsy samples available. For the classification of IIMs, the European Neuromuscular Center criteria were applied...
July 19, 2016: Neurology
Yosuke Sasaki, Hiroshige Shimizu, Tetsuo Nemoto, Yoshihisa Urita
The strong association between myositis and malignancy has been well recognised. Cancer-associated myositis (CAM) is thought to be a cross-reaction to regenerating muscle tissue similar to tumour antigen. We report a case of CAM due to oesophageal adenocarcinoma arising in Barrett's oesophagus without elevation of myogenic enzymes, diagnosed by MRI and repeated endoscopy. Elderly onset, prominent symptoms, lack of interstitial pneumonia, poorer response to immunosuppressive therapies, and the combination of negative conventional myositis-related antibodies and positive anti-p155/140 antibody may help to distinguish CAM from idiopathic inflammatory myopathy...
April 21, 2016: BMJ Case Reports
Yves Allenbach, Jeremy Keraen, Anne-Marie Bouvier, Valérie Jooste, Nicolas Champtiaux, Baptiste Hervier, Yoland Schoindre, Aude Rigolet, Laurent Gilardin, Lucile Musset, Jean-Luc Charuel, Olivier Boyer, Fabienne Jouen, Laurent Drouot, Jeremie Martinet, Tanya Stojkovic, Bruno Eymard, Pascal Laforêt, Antony Behin, Emmanuelle Salort-Campana, Olivier Fain, Alain Meyer, Nicolas Schleinitz, Kuberaka Mariampillai, Aurelie Grados, Olivier Benveniste
Cancer can occur in patients with inflammatory myopathies. This association is mainly observed in dermatomyositis, and myositis-specific antibodies have allowed us to delineate patients at an increased risk. Malignancy is also reported in patients with necrotizing autoimmune myopathies, but the risk remains elusive. Anti-signal recognition particle or anti-HMGCR antibodies have been specifically associated with necrotizing autoimmune myopathies. We aimed at screening the incidence of cancer in necrotizing autoimmune myopathies...
August 2016: Brain: a Journal of Neurology
Matthew M Florczynski, Michael S Sanatani, Lauren Mai, Barbara Fisher, Dwight E Moulin, Jeffrey Cao, Alexander V Louie, Janet E Pope, Eric Leung
BACKGROUND: The use of neoadjuvant radiation therapy and chemotherapy in the treatment of locally advanced rectal adenocarcinoma has been shown to reduce disease recurrence when combined with surgery and adjuvant chemotherapy. We report a case of a patient who developed a debilitating bilateral myopathy of the hip flexors after successful treatment for rectal cancer. To the best of our knowledge, this is the first such complication from radiation therapy reported in a patient with colorectal cancer...
March 22, 2016: BMC Cancer
J-J Hauw, S Haïk, C Duyckaerts
Protein misfolding and spreading ("transconformation") are being better understood. Described in Prions diseases, this new paradigm in the field of neurodegenerative disorders and brain aging also implies sporadic inclusion myositis, type 2 diabetes, some cancers, sickle cell disease... Misfolding is transmitted from a protein or peptide to a normally folded one. Often associated with a stress of the endoplasmic reticulum, it may spread along the neurites, following anterograde or retrograde axonal transport...
December 2015: Revue Neurologique
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