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https://www.readbyqxmd.com/read/29784018/adding-colchicine-to-immunosuppressive-treatments-a-potential-option-for-biologics-refractory-adult-onset-still-s-disease
#1
Tomoyuki Asano, Makiko Yashiro Furuya, Shuzo Sato, Hiroko Kobayashi, Hiroshi Watanabe, Eiji Suzuki, Kiyoshi Migita
BACKGROUND: Adult-onset Still's disease (AOSD) is a rare inflammatory disorder characterized by the classical triad of daily spiking fever, arthritis, and typical salmon-colored rash. Resistance to first-line corticosteroids and second-line disease modified anti-rheumatic-drugs defines refractory AOSD, which mostly includes the polycyclic or chronic courses of the disease. Anti-cytokine therapies are recommended in AOSD patients who are refractory to traditional treatments. This is the first report on the efficacy of colchicine in a patient with AOSD which was refractory to immunosuppressive treatments including biologics...
May 21, 2018: BMC Research Notes
https://www.readbyqxmd.com/read/29733534/adult-onset-still-s-disease-initially-thought-to-be-an-odontogenic-infection-a-case-report
#2
Shunsuke Hino, Satoshi Nakamura, Takahiro Kaneko, Norio Horie, Tetsuo Shimoyama
OBJECTIVE: To present a case of Adult-onset Still's disease (AOSD) initially suspected to be odontogenic inflammation. BACKGROUND: Adult-onset Still's disease is a rare, complex autoinflammatory disease and a known cause of fever of unknown origin. MATERIALS AND METHODS: The patient had both a fever and dental pain. Following meticulous examination, the patient was diagnosed with AOSD. CONCLUSION: Clinicians should keep in mind that a patient such as AOSD may visit their clinics...
June 2018: Gerodontology
https://www.readbyqxmd.com/read/29658384/complications-of-adult-onset-still-s-disease-and-their-management
#3
Stéphane Mitrovic, Bruno Fautrel
Adult-onset Still's disease (AOSD) is a rare systemic auto-inflammatory disorder in which management and treatment have considerably progressed over the past decade. Despite wide use of interleukin (IL)-1 or IL-6 inhibitors, serious complications remain possible. Areas covered: A comprehensive literature search in MEDLINE via Pubmed was performed to review AOSD's severe and sometimes life-threatening complications: reactive hemophagocytic lymphohystiocytosis, coagulation disorders, fulminant hepatitis, cardiac or pulmonary complications and amyloid A amyloidosis...
April 26, 2018: Expert Review of Clinical Immunology
https://www.readbyqxmd.com/read/29657144/macrophage-activation-syndrome-in-patients-affected-by-adult-onset-still-disease-analysis-of-survival-rates-and-predictive-factors-in-the-gruppo-italiano-di-ricerca-in-reumatologia-clinica-e-sperimentale-cohort
#4
Piero Ruscitti, Daniela Iacono, Francesco Ciccia, Giacomo Emmi, Paola Cipriani, Rosa Daniela Grembiale, Federico Perosa, Lorenzo Emmi, Giovanni Triolo, Roberto Giacomelli, Gabriele Valentini
OBJECTIVE: Macrophage activation syndrome (MAS) is a reactive form of hemophagocytic lymphohistiocytosis, which can complicate adult-onset Still disease (AOSD). We investigated AOSD clinical features at the time of diagnosis, to assess predictors of MAS occurrence. Further, we analyzed the outcomes of patients with AOSD who experience MAS. METHODS: Patients with AOSD admitted to any Gruppo Italiano di Ricerca in Reumatologia Clinica e Sperimentale center were retrospectively analyzed for features typical of AOSD, MAS occurrence, and their survival rate...
April 15, 2018: Journal of Rheumatology
https://www.readbyqxmd.com/read/29651907/evidence-based-clinical-practice-guideline-for-adult-still-s-disease
#5
Toshihide Mimura, Yuya Kondo, Akihide Ohta, Masahiro Iwamoto, Akiko Ota, Nami Okamoto, Yasushi Kawaguchi, Hajime Kono, Yoshinari Takasaki, Shuji Takei, Norihiro Nishimoto, Manabu Fujimoto, Yu Funakubo Asanuma, Akio Mimori, Naoko Okiyama, Shunta Kaneko, Hiroyuki Takahashi, Masahiro Yokosawa, Takayuki Sumida
OBJECTIVES: Using an expert- and data-driven methodology, we have constructed the first clinical practice guidelines (CPGs) for adult Still's disease (ASD) after complete systematic review (SR) of the literature based upon the Medical Information Network Distribution Service (Minds) procedure. METHODS: The CPG committee for ASD organized by the Research Team for Autoimmune Diseases, the Research Program for Intractable Disease of the Japanese Ministry of Health, Labour, and Welfare has developed CPG for ASD 2017, according to the procedure proposed by Minds...
May 9, 2018: Modern Rheumatology
https://www.readbyqxmd.com/read/29642928/diagnostic-and-management-of-life-threatening-adult-onset-still-disease-a-french-nationwide-multicenter-study-and-systematic-literature-review
#6
Antoine Néel, Anaïs Wahbi, Benoit Tessoulin, Julien Boileau, Dorothée Carpentier, Olivier Decaux, Laurence Fardet, Guillaume Geri, Pascal Godmer, Cécile Goujard, Hervé Maisonneuve, Arnaud Mari, Jacques Pouchot, Jean-Marc Ziza, Cédric Bretonnière, Mohamed Hamidou
BACKGROUND: Adult-onset Still disease (AOSD) is a rare systemic inflammatory disorder. A few patients develop organ complications that can be life-threatening. Our objectives were to describe the disease course and phenotype of life-threatening AOSD, including response to therapy and long-term outcome. METHODS: A multicenter case series of intensive care medicine (ICU) patients with life-threatening AOSD and a systematic literature review. RESULTS: Twenty patients were included...
April 11, 2018: Critical Care: the Official Journal of the Critical Care Forum
https://www.readbyqxmd.com/read/29600942/serum-epstein-barr-virus-dna-detected-by-droplet-digital-pcr-correlates-with-disease-activity-in-patients-with-rheumatoid-arthritis
#7
Elina Kuusela, Vesa-Petteri Kouri, Juri Olkkonen, Riitta Koivuniemi, Leena Äyräväinen, Kristiina Rajamäki, Heikki Valleala, Dan Nordström, Marjatta Leirisalo-Repo, Mari Ainola, Kari K Eklund
OBJECTIVES: To study the prevalence of asymptomatic activation of Epstein-Barr virus (EBV) in patients with rheumatoid arthritis (RA) and to analyse the correlation of serum EBV DNA with the disease activity. METHODS: The level of EBV DNA was determined by droplet digital PCR assay from the serum of 46 DMARD naive early RA (ERA) and 22 chronic RA (CRA)-patients at study onset. Follow-up samples from 31 ERA and 16 CRA patients were obtained after starting or modifying the anti-rheumatic treatment...
March 20, 2018: Clinical and Experimental Rheumatology
https://www.readbyqxmd.com/read/29566725/interleukin-37-is-increased-in-adult-onset-still-s-disease-and-associated-with-disease-activity
#8
Huihui Chi, Dongzhou Liu, Yue Sun, Qiongyi Hu, Honglei Liu, Xiaobing Cheng, Junna Ye, Hui Shi, Yufeng Yin, Mengru Liu, Xinyao Wu, Zhuochao Zhou, Jialin Teng, Chengde Yang, Yutong Su
BACKGROUND: Interleukin (IL)-37 has been known to play an immunosuppressive role in various inflammatory disorders, but whether it participates in the regulation of pathogenesis of adult-onset Still's disease (AOSD) has not been investigated. In this study, we examined serum IL-37 levels and their clinical association with AOSD, and we explored the anti-inflammatory effects of IL-37 on peripheral blood mononuclear cells (PBMCs) from patients with AOSD. METHODS: Blood samples were collected from 62 patients with AOSD and 50 healthy control subjects (HC)...
March 22, 2018: Arthritis Research & Therapy
https://www.readbyqxmd.com/read/29565301/efficacy-of-denosumab-for-osteoporosis-in-two-patients-with-adult-onset-still-s-disease-denosumab-efficacy-in-osteoporotic-still-s-disease-patients
#9
Daiki Kumaki, Yukio Nakamura, Takako Suzuki, Hiroyuki Kato
Adult-onset Still's disease (AOSD) is an autoimmune inflammatory disorder. Glucocorticoids are often used for AOSD, which may induce complicating glucocorticoid-induced osteoporosis (GIO). An anti-resorption drug, denosumab, has recently been approved for osteoporosis treatment in Japan. However, the drug's efficacy for GIO in AOSD is largely unknown. This retrospective, consecutive case series investigated two patients with GIO in AOSD to examine the effects of denosumab on bone metabolism. Bone turnover markers, and bone mineral density (BMD) of the lumbar 1-4 spine (L-BMD) and bilateral total hips (H-BMD) were followed for six months in a male patient and for twelve months in a female patient...
March 22, 2018: Journal of Clinical Medicine
https://www.readbyqxmd.com/read/29533755/efficacy-and-safety-of-canakinumab-in-patients-with-still-s-disease-exposure-response-analysis-of-pooled-systemic-juvenile-idiopathic-arthritis-data-by-age-groups
#10
Eugen Feist, Pierre Quartier, Bruno Fautrel, Rayfel Schneider, Paolo Sfriso, Petros Efthimiou, Luca Cantarini, Karine Lheritier, Karolynn Leon, Chetan S Karyekar, Antonio Speziale
OBJECTIVES: To describe the efficacy, safety, and exposure-response relationship of canakinumab in a subgroup of patients with systemic juvenile idiopathic arthritis (SJIA) aged ≥16 years, representative of adult-onset Still's disease (AOSD) patients, and to compare this subgroup with those of children and young adolescents with SJIA by pooling clinical data collected during the development programme of canakinumab. METHODS: Safety and efficacy data on canakinumab-treated patients were pooled from 4 SJIA studies (NCT00426218, NCT00886769, NCT00889863, and NCT00891046)...
March 2, 2018: Clinical and Experimental Rheumatology
https://www.readbyqxmd.com/read/29472362/open-label-multicentre-dose-escalating-phase-ii-clinical-trial-on-the-safety-and-efficacy-of-tadekinig-alfa-il-18bp-in-adult-onset-still-s-disease
#11
Cem Gabay, Bruno Fautrel, Jürgen Rech, François Spertini, Eugen Feist, Ina Kötter, Eric Hachulla, Jacques Morel, Thierry Schaeverbeke, Mohamed A Hamidou, Thierry Martin, Bernhard Hellmich, Peter Lamprecht, Hendrik Schulze-Koops, Delphine Sophie Courvoisier, Andrew Sleight, Eduardo Jorge Schiffrin
OBJECTIVES: Adult-onset Still's disease (AOSD) is a rare systemic autoinflammatory disease; its management is largely empirical. This is the first clinical study to determine if interleukin (IL)-18 inhibition, using the recombinant human IL-18 binding protein, tadekinig alfa, is a therapeutic option in AOSD. METHODS: In this phase II, open-label study, patients were ≥18 years with active AOSD plus fever or C reactive protein (CRP) levels ≥10 mg/L despite treatment with prednisone and/or conventional synthetic disease-modifying antirheumatic drugs (DMARDs)...
June 2018: Annals of the Rheumatic Diseases
https://www.readbyqxmd.com/read/29468168/severe-pulmonary-hypertension-due-to-adult-onset-still-s-disease
#12
Ankur Sinha, Ravikaran Patti, Paurush Ambesh, Chukwudi Obiagwu, Namrita Malhan, Kabu Chawla
A 29-year-old female with adult-onset Still's disease (AOSD) presented with progressive shortness of breath both on rest and on exertion, increased abdominal girth, and swelling in both legs. She was on oral prednisone and was recently started on canakinumab (interleukin-1 antagonist) for joint pain and rash of AOSD. Echocardiogram showed severely dilated right ventricle, dilated pulmonary artery, moderately reduced right ventricular systolic function, but with normal left ventricular systolic function. Computed tomography with contrast ruled out pulmonary embolism...
January 2018: Journal of Investigative Medicine High Impact Case Reports
https://www.readbyqxmd.com/read/29420734/convergent-pathways-of-the-hyperferritinemic-syndromes
#13
Grant S Schulert, Scott W Canna
Hyperferritinemia and pronounced hemophagocytosis help distinguish a subset of patients with a particularly inflammatory and deadly systemic inflammatory response syndrome. Two clinically similar disorders typify these hyperferritinemic syndromes: hemophagocytic lymphohistiocytosis (HLH) and macrophage activation syndrome (MAS). HLH is canonically associated with a complete disturbance of perforin/granzyme-mediated cytotoxicity, whereas MAS occurs in the context of the related rheumatic diseases systemic juvenile idiopathic arthritis and adult-onset Still's disease, with associated IL-1 family cytokine activation...
April 25, 2018: International Immunology
https://www.readbyqxmd.com/read/29416600/impaired-autophagic-flux-and-its-related-inflammation-in-patients-with-adult-onset-still-s-disease
#14
Chia-Wei Hsieh, Chun-Yu Chang, Yi-Ming Chen, Hsin-Hua Chen, Wei-Ting Hung, Ning-Rong Gung, Shiow-Jiuan Wey, Der-Yuan Chen
The pathogenic role of autophagic immune regulation in adult-onset Still's disease (AOSD) is unclear. We investigated the relative levels of autophagy in AOSD patients and healthy controls, its association with disease activity or course, and the change in autophagy after 6 months of therapy. Autophagosome levels were determined from the mean fluorescence intensity of autophagosomotropic dye incorporated into circulating immune cells. The fluorescent signal from lymphocytes, monocytes, and granulocytes from AOSD patients was greater than from controls...
January 2, 2018: Oncotarget
https://www.readbyqxmd.com/read/29416343/biological-therapy-of-traditional-therapy-resistant-adult-onset-still-s-disease-an-evidence-based-review
#15
REVIEW
Sha Zhou, Jianjun Qiao, Juan Bai, Yinhua Wu, Hong Fang
Background: Biotherapy is becoming increasingly important in the treatment of adult-onset Still's disease (AOSD). The aim of our study was to evaluate the efficacy and safety of biological therapy for AOSD resistant to traditional therapy. Patients and methods: Database of Library of Congress, the PubMed, and Web of Science Core Collection were used to retrieve relevant articles published in English language until March 2017. Only studies published in English language were included, and the additional references quoted in these articles were also checked...
2018: Therapeutics and Clinical Risk Management
https://www.readbyqxmd.com/read/29390286/purulent-constrictive-pericarditis-caused-by-salmonella-enteritidis-in-a-patient-with-adult-onset-still-s-disease-a-case-report
#16
Chia-Cheng Kuo, Wen-Liang Yu, Chen-Hui Lee, Nan-Chun Wu
RATIONALE: Purulent pericarditis is a rare and usually fatal disease. Immunodeficiency state and preexisting pericardial effusion can predispose patients to infections. However, we are not aware of similar cases in patients with adult-onset Still's disease (AOSD). In addition, it is seldom caused by Salmonella bacteria. PATIENT CONCERNS: We report a 30-year-old woman with dyspnea on exertion and epigastric fullness. She was newly diagnosed with AOSD 4 months previously and medicated with prednisolone...
December 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29384877/acute-appendicitis-complicated-with-necrotizing-fasciitis-in-a-patient-with-adult-onset-still-s-disease-a-case-report
#17
Zheng-Hao Huang, Yu-Chen Chiu, Li-Lu Ho, Hsiu-Lung Fan, Chun-Chi Lu
RATIONALE: Adult-onset Still disease (AOSD) is a rare systemic inflammatory disease of unknown etiology characterized by evanescent salmon-pink rash, spiking fever, arthralgia/ arthritis, and lymphadenopathy. AOSD sometimes was fatal when it is complicated by macrophage activation syndrome (MAS) or hemophagocytic lymphohistiocytosis (HLH). Nonetheless, the literature provides no recommendations for treatment of AOSD patients with severe sepsis. PATIENT CONCERNS: A previously healthy 65-year-old man with history of AOSD was referred to our hospital for persistent right lower quadrant abdominal pain for 2 days...
February 2018: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29370863/application-of-the-international-league-against-rheumatism-classification-criteria-for-systemic-juvenile-idiopathic-arthritis-as-a-prognostic-factor-in-patients-with-adults-onset-still-s-disease
#18
Ji Won Yang, Eunyoung Lee, Ji-Yeon Seo, Ju-Yang Jung, Chang-Hee Suh, Hyoun-Ah Kim
BACKGROUND: Adult-onset Still's disease (AOSD) is an adult form of systemic juvenile idiopathic arthritis (JIA) that differs from the latter in its classification. This study evaluated the concordance between the International League Against Rheumatism (ILAR) criteria for systemic JIA and the Yamaguchi criteria and then compared their possible prognostic value in patients with AOSD. METHODS: In a retrospective review of 169 adults with suspected AOSD, patients were classified according to the Yamaguchi or ILAR criteria...
January 25, 2018: Pediatric Rheumatology Online Journal
https://www.readbyqxmd.com/read/29278009/beneficial-use-of-serum-ferritin-and-heme-oxygenase-1-as-biomarkers-in-adult-onset-still-s-disease-a-multicenter-retrospective-study
#19
Yohei Kirino, Yasushi Kawaguchi, Yoshifumi Tada, Hiroshi Tsukamoto, Toshiyuki Ota, Masahiro Iwamoto, Hiroki Takahashi, Kohei Nagasawa, Shuji Takei, Takahiko Horiuchi, Hisae Ichida, Seiji Minota, Atsuhisa Ueda, Akihide Ohta, Yoshiaki Ishigatsubo
BACKGROUND: Heme oxygenase (HO)-1 is a heme-degrading enzyme highly expressed in monocyte/macrophage, serum levels of which may be promising biomarker for adult-onset Still's disease (AOSD). We here report data on the use of serum ferritin and HO-1 levels in AOSD. METHODS: Under the Hypercytokinemia Study Group collaboration, we collected sera from a total of 145 AOSD patients. Three independent experts judged whether the patients were definite AOSD depending on the clinical information...
January 11, 2018: Modern Rheumatology
https://www.readbyqxmd.com/read/29251027/efficacy-and-safety-of-tocilizumab-with-inhibition-of-interleukin-6-in-adult-onset-still-s-disease-a-meta-analysis
#20
Yubo Ma, Meng Wu, Xu Zhang, Qing Xia, Jiajia Yang, Shengqian Xu, Faming Pan
OBJECTIVES: Tocilizumab (TCZ) is the only available biologics inhibiting interleukin-6 presently, and emerging evidences have figured that elevated serum level of interleukin-6 is a crucial link of the pathogenesis of adult-onset Still's disease (AOSD). However, evidence about the efficacy and safety of TCZ in AOSD with strong power is still scarce. Thus, this meta-analysis was conducted to synthetically assess the efficacy and safety of TCZ on AOSD. METHODS: PubMed, Web of Science, Cochrane Library, Chinese National Knowledge Infrastructure, and Chinese Biological Medical Literature database were comprehensively searched up to 31 July 2017 for the pertinent studies...
February 8, 2018: Modern Rheumatology
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