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https://www.readbyqxmd.com/read/29168664/pulmonary-arterial-hypertension-and-acute-respiratory-distress-syndrome-in-a-patient-with-adult-onset-stills-disease
#1
Grant H Lowther, Jason Chertoff, Jessica Cope, Hassan Alnuaimat, Ali Ataya
Adult-onset Still's disease (AOSD) is an inflammatory disorder characterized by recurrent fevers, arthralgia, leukocytosis, and a salmon-colored rash. Diagnosis is made based on the Yamaguchi criteria. Various cardiac and pulmonary manifestations have been described in association with AOSD, including acute respiratory distress syndrome (ARDS) and pulmonary arterial hypertension (PAH). We describe the first case of both PAH and ARDS in a patient with AOSD who, despite aggressive therapy, declined rapidly and ultimately died...
October 2017: Pulmonary Circulation
https://www.readbyqxmd.com/read/29159471/evidence-for-genetic-overlap-between-adult-onset-still-s-disease-and-hereditary-periodic-fever-syndromes
#2
R Sighart, J Rech, A Hueber, N Blank, S Löhr, A Reis, H Sticht, U Hüffmeier
OBJECTIVE: Adult onset Still's disease (AOSD) is a severe, autoimmune disease that can be challenging to treat with conventional therapeutics and biologicals in a considerable number of cases. Therefore, there is a high need to understand its pathogenesis better. As major clinical symptoms overlap between AOSD and hereditary periodic fever syndromes (HPFS), we analysed four known HPFS genes in AOSD. METHODS: We performed Sanger sequencing and quantitative analysis of all coding regions of MEFV, TNFRSF1A, MVK and NLRP3 in 40 AOSD patients...
November 20, 2017: Rheumatology International
https://www.readbyqxmd.com/read/29147588/diagnostic-challenge-a-report-of-two-adult-onset-still-s-disease-cases
#3
Sakunee Niranvichaiya, Daranporn Triwongwaranat
This study reports two adult-onset Still's disease (AOSD) cases that met both Yamaguchi's and Fautrel's criteria and that presented with notable clinical manifestations. One case presented with atypical dermographism-like rash with an extremely high ferritin level. The other case presented with typical salmon-pink maculopapular rash but had atypical positive rheumatoid factor. This suggests that although negative rheumatoid factor is one of the criteria used for the diagnosis of AOSD, a positive rheumatoid factor result does not exclude AOSD...
2017: Case Reports in Dermatological Medicine
https://www.readbyqxmd.com/read/29094181/cardiovascular-disease-in-patients-with-autoinflammatory-syndromes
#4
REVIEW
Rainer Hintenberger, Agnes Falkinger, Kathrin Danninger, Herwig Pieringer
Autoinflammatory syndromes (AIS) are characterized by recurring events of inflammation, leading to a variety of organ manifestations and fever attacks. A subgroup of AIS is commonly referred to as hereditary periodic fever syndromes (HPFS). There is substantial evidence that autoimmune diseases such as rheumatoid arthritis and systemic lupus erythematosus are strongly associated with cardiovascular morbidity and mortality. The link between AIS and cardiovascular disease is not that clear, even if the concept of continuous inflammation as a risk factor for cardiovascular disease is widely accepted...
November 1, 2017: Rheumatology International
https://www.readbyqxmd.com/read/29044212/a-pilot-study-on-tocilizumab-for-treating-refractory-adult-onset-still-s-disease
#5
Ting Li, Liyang Gu, Xiaodong Wang, Li Guo, Hui Shi, Chengde Yang, Sheng Chen
To investigate the efficacy and safety of Tocilizumab (TCZ) in patients with refractory adult-onset Still's disease (AOSD). We enrolled 8 female patients from October 2013 to July 2014. All patients fulfilled Japan's Yamaguch AOSD classification and recognized as refractory AOSD. All Patients received TCZ treatment 4-8 mg/kg every 4 weeks. Evaluation of efficacy was conducted after 3 months and 6 months, including clinical manifestations of AOSD patients, improvement of inflammatory markers as well as glucocorticoids dosage adjustments...
October 18, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28899403/effects-of-hla-drb1-alleles-on-susceptibility-and-clinical-manifestations-in-japanese-patients-with-adult-onset-still-s-disease
#6
Tomoyuki Asano, Hiroshi Furukawa, Shuzo Sato, Makiko Yashiro, Hiroko Kobayashi, Hiroshi Watanabe, Eiji Suzuki, Tomoyuki Ito, Yoshifumi Ubara, Daisuke Kobayashi, Nozomi Iwanaga, Yasumori Izumi, Keita Fujikawa, Satoshi Yamasaki, Tadashi Nakamura, Tomohiro Koga, Toshimasa Shimizu, Masataka Umeda, Fumiaki Nonaka, Michio Yasunami, Yukitaka Ueki, Katsumi Eguchi, Naoyuki Tsuchiya, Shigeto Tohma, Koh-Ichiro Yoshiura, Hiromasa Ohira, Atsushi Kawakami, Kiyoshi Migita
BACKGROUND: HLA-DRB1 alleles are major determinants of genetic predisposition to rheumatic diseases. We assessed whether DRB1 alleles are associated with susceptibility to particular clinical features of adult onset Still's disease (AOSD) in a Japanese population by determining the DRB1 allele distributions. METHODS: DRB1 genotyping of 96 patients with AOSD and 1,026 healthy controls was performed. Genomic DNA samples from the AOSD patients were also genotyped for MEFV exons 1, 2, 3, and 10 by direct sequencing...
September 12, 2017: Arthritis Research & Therapy
https://www.readbyqxmd.com/read/28870100/biologic-drugs-in-adult-onset-still-s-disease-a-systematic-review-and-meta-analysis-of-observational-studies
#7
Piero Ruscitti, Francesco Ursini, Paola Cipriani, Giovambattista De Sarro, Roberto Giacomelli
BACKGROUND: Biological drugs, mainly interleukin (IL)-1 and IL-6 antagonists, but also tumor necrosis factor (TNF) inhibitors, have been used in the treatment of adult onset Still's disease patients (AOSD). METHODS: We summarised the available evidence for the effectiveness of biologic drugs in AOSD. A systematic review of the literature was performed in order to identify all the available data concerning the effectiveness of biologic drugs in AOSD. The proportion of patients achieving complete remission or any clinical response was calculated...
November 2017: Expert Review of Clinical Immunology
https://www.readbyqxmd.com/read/28838228/red-blood-cell-distribution-width-is-useful-in-discriminating-adult-onset-still-s-disease-and-sepsis-within-24-hours-after-hospitalization
#8
Hee-Jin Park, Jungsik Song, Yong-Beom Park, Soo-Kon Lee, Sang-Won Lee
Background/Aims: Red blood cell distribution width (RDW) is a value representing the heterogeneity in the size of red blood cell, and it is usually used in distinguishing types of anaemia. Recently, it was reported that it could reflect the burden of inflammation in diverse diseases and their prognosis. Hence, in this study, we investigated whether RDW may contribute to discriminating adult onset Still's disease (AOSD) from sepsis in serious febrile patients within 24 hours after hospitalization...
August 25, 2017: Korean Journal of Internal Medicine
https://www.readbyqxmd.com/read/28781105/adult-s-onset-still-disease-occurring-during-pregnancy-case-report-and-literature-review
#9
Léo Plaçais, Arsène Mekinian, Marie Bornes, Armelle Poujol-Robert, Naiké Bigé, Eric Maury, Olivier Fain
INTRODUCTION: Adult onset Still's disease is a rare affection classified among non-hereditary autoinflammatory diseases. We here report a case of AOSD revealed during pregnancy with a life-threatening presentation along with a review of 19 cases from literature. CASE: A 38-years old woman was treated in our department for diffuse systemic sclerosis and associated Sjögren syndrome. She was pregnant and presented with acute fever and arthralgias. Laboratory data revealed mild liver cytolysis but a large screening for infectious and auto-immune diseases was negative and hepato-biliar imaging was normal...
July 10, 2017: Seminars in Arthritis and Rheumatism
https://www.readbyqxmd.com/read/28765926/imaging-characteristics-of-adult-onset-still-s-disease-demonstrated-with-18f-fdg-pet-ct
#10
Lei Jiang, Yan Xiu, Taoying Gu, Caihong Dong, Bing Wu, Hongcheng Shi
The diagnosis of adult onset Still's disease (AOSD) is non‑specific, and requires the exclusion of other diseases including infectious, inflammatory and malignant diseases. The current study aimed to summarize the imaging characteristics of fluorodeoxyglucose (18F‑FDG) positron emission tomography (PET)/computerized tomography (CT) in patients with AOSD. The 18F‑FDG PET/CT characteristic observations of 32 patients with definite AOSD were retrospectively reviewed based on visual interpretation and the semi‑quantitative index of standard uptake value of maximum (SUVmax)...
September 2017: Molecular Medicine Reports
https://www.readbyqxmd.com/read/28760536/validation-of-the-fautrel-classification-criteria-for-adult-onset-still-s-disease
#11
Delphine Lebrun, Stéphanie Mestrallet, Monique Dehoux, Jean Louis Golmard, Benjamin Granger, Sophie Georgin-Lavialle, Laurent Arnaud, Gilles Grateau, Jacques Pouchot, Bruno Fautrel
OBJECTIVES: To validate the Fautrel classification criteria for adult-onset Still's disease (AOSD) and to compare the discriminative performance to that of the Yamaguchi criteria. METHODS: We retrospectively reviewed the medical charts of 426 patients who had serum ferritin level and percentage glycosylated ferritin assayed at the biochemistry laboratory of Bichat Hospital. Medical data were extracted by use of a standardized form. All clinical, biological, and imaging features were collected, as well, evidence favoring an alternative diagnosis, specifically symptoms suggestive of other immune-mediated inflammatory diseases (IMID) or active infections...
July 12, 2017: Seminars in Arthritis and Rheumatism
https://www.readbyqxmd.com/read/28757235/adult-onset-still-s-disease-the-evidence-that-anti-interleukin-1-treatment-is-effective-and-well-tolerated-a-comprehensive-literature-review
#12
REVIEW
Guido Junge, June Mason, Eugen Feist
The literature contains many reports of the use of commercially available anti-IL-1 agents (anakinra/Kineret(®), canakinumab/Ilaris(®), or rilonacept/Arcalyst(®)) in treatment-resistant adult-onset Still's disease (AOSD). These have been widely summarized in many review articles, but a full account of all reports with each of the agents used is not available. This literature review includes all reports of treatment outcomes in patients treated for AOSD with any commercially available anti-IL-1 agent (excluding cases of unconfirmed or atypical AOSD or treatments only for rare AOSD complications)...
June 23, 2017: Seminars in Arthritis and Rheumatism
https://www.readbyqxmd.com/read/28755070/-still-s-disease-in-children-and-adults
#13
C M Hedrich, C Günther, M Aringer
Systemic juvenile idiopathic arthritis (sJIA) is characterized by fever, arthritis, and other signs of systemic inflammation. Historically, sJIA was named Still's disease after George Frederic Still, who first reported patients. Individuals who manifest after the 16(th) birthday are diagnosed with adult onset Still's disease (AOSD). The pathophysiology of sJIA and AOSD are incompletely understood. Increased activation of inflammasomes and the expression of proinflammatory cytokines play a central role. S100 proteins, which can activate Toll-like receptors, thus, maintaining positive feedback loops, have also been detected at increased levels in sera from sJIA patients...
September 2017: Zeitschrift Für Rheumatologie
https://www.readbyqxmd.com/read/28725546/pulmonary-involvement-in-adult-still-s-disease-case-report-and-brief-review-of-literature
#14
Aldo Guerrieri, Giulia Angeletti, Massimiliano Mazzolini, Ilaria Bassi, Stefano Nava
BACKGROUND: Adult onset Still disease (AOSD) is a rare condition characterized by elevated fever along with arthritic symptoms, elevated polymorphonuclear neutrophil count, evanescent rash, and hyperferritinemia. Diagnosis can be made only after have ruled out more frequent conditions, and Yamagouchi or Fautrel criteria should be applied. Parenchimal lung involvement (PLI) is present in less than 5% of AOSD cases and ranges from aspecific reticular interstitial opacities to life threatening conditions, such as acute respiratory distress syndrome (ARDS)...
2017: Respiratory Medicine Case Reports
https://www.readbyqxmd.com/read/28723802/tocilizumab-for-uncontrollable-systemic-inflammatory-response-syndrome-complicating-adult-onset-still-disease-case-report-and-review-of-literature
#15
REVIEW
Asami Masui-Ito, Ryuji Okamoto, Kaoru Ikejiri, Mika Fujimoto, Muneyoshi Tanimura, Shiro Nakamori, Tomohiro Murata, Eiji Ishikawa, Norikazu Yamada, Hiroshi Imai, Masaaki Ito
RATIONALE: Adult-onset Still disease (AOSD) is a rare systemic inflammatory disease of unknown etiology characterized by evanescent salmon-pink rash, fever spikes, arthralgia, and lymphadenopathy. AOSD usually has a good prognosis, but it can sometimes be fatal, especially when it is complicated by systemic inflammatory response syndrome (SIRS) and multiple organ failure. PATIENT CONCERNS: A previously healthy 26-year-old woman was referred to our hospital for persistent high fever and mild systemic edema...
July 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28723775/the-neutrophil-to-lymphocyte-ratio-could-be-a-good-diagnostic-marker-and-predictor-of-relapse-in-patients-with-adult-onset-still-s-disease-a-strobe-compliant-retrospective-observational-analysis
#16
Ji-Yeoun Seo, Chang-Hee Suh, Ju-Yang Jung, Ar-Reum Kim, Ji Won Yang, Hyoun-Ah Kim
The neutrophil-to-lymphocyte ratio (NLR) is the proportion of absolute neutrophil count to lymphocytes on routine complete blood count (CBC) tests, and has been studied as a simple marker of the systemic inflammatory response. This study was performed to investigate whether the NLR could be used as a tool to diagnose and predict prognosis in cases of adult-onset Still's disease (AOSD).We retrospectively reviewed 164 patients with suspected AOSD. Among 164 patients with suspected AOSD, 37 patients received another diagnosis (such as viral infection) and were compared with the 127 patients who received a diagnosis of AOSD...
July 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28720215/autoimmune-meningitis-and-encephalitis-in-adult-onset-still-disease-case-report
#17
Milena Bożek, Magdalena Konopko, Teresa Wierzba-Bobrowicz, Grzegorz Witkowski, Grzegorz Makowicz, Halina Sienkiewicz-Jarosz
INTRODUCTION: Adult-onset Still disease (AOSD) is a rare systemic inflammatory disease of unknown cause. Its symptoms usually include persistent fever, fugitive salmon-colored rash, arthritis, sore throat (not specific), but it may also lead to internal organs' involvement, which presents with enlargement of the liver and spleen, swollen lymph nodes, carditis or pleuritis - potentially life-threatening complications. In rare cases, AOSD can cause aseptic meningitis or/and encephalitis...
July 8, 2017: Neurologia i Neurochirurgia Polska
https://www.readbyqxmd.com/read/28690954/delay-in-the-diagnosis-of-adult-onset-still-s-disease
#18
Stella Pak, Cindy Pham
Adult-onset Still's disease (AOSD) is a systemic autoinflammatory disease characterized by symptoms including spiking fever, arthralgia, myalgia, maculopapular rash, and pharyngitis. The lack of diagnostic biomarker, non-specific clinical presentation, and the rarity of AOSD often result in a significant delay in diagnosis and treatment. While the average time of initial presentation to diagnosis is four months, we present a case of AOSD diagnosis three years after initial onset of classical symptoms. By reporting the case of delayed diagnosis for AOSD, we hope to raise awareness in our medical community about the diagnostic difficulty in AOSD...
June 7, 2017: Curēus
https://www.readbyqxmd.com/read/28690235/-diagnosis-and-treatment-for-one-case-of-elderly-diabetes-complicated-with-still-s-disease
#19
Tuanyu Fang, Kaining Chen, Eryuan Liao
Adult onset Still's disease (AOSD) is a clinical syndrome with multiple organ failure. The patients normally show intermittent high fever for a long time, a transient rash, arthritis or joint pain as the main performance, accompanied by an increase in granulocytes and enlargement in liver, spleen and lymph node. A 71-years-old female patient with type 2 diabetes admitted hospital because of high fever, skin rash, joint pain and increased granulocyte. After review of the iron protein, she was diagnosed as AOSD...
June 28, 2017: Zhong Nan da Xue Xue Bao. Yi Xue Ban, Journal of Central South University. Medical Sciences
https://www.readbyqxmd.com/read/28659802/response-to-interleukin-1-inhibitors-in-140-italian-patients-with-adult-onset-still-s-disease-a-multicentre-retrospective-observational-study
#20
Serena Colafrancesco, Roberta Priori, Guido Valesini, Lorenza Argolini, Elena Baldissera, Elena Bartoloni, Daniele Cammelli, Giovanni Canestrari, Luca Cantarini, Elena Cavallaro, Giulio Cavalli, Lucia Cerrito, Paola Cipriani, Lorenzo Dagna, Ginevra De Marchi, Salvatore De Vita, Giacomo Emmi, Gianfranco Ferraccioli, Micol Frassi, Mauro Galeazzi, Roberto Gerli, Roberto Giacomelli, Elisa Gremese, Florenzo Iannone, Giovanni Lapadula, Giuseppe Lopalco, Raffaele Manna, Alessandro Mathieu, Carlomaurizio Montecucco, Marta Mosca, Ilaria Piazza, Matteo Piga, Irene Pontikaki, Micol Romano, Silvia Rossi, Maurizio Rossini, Piero Ruscitti, Elena Silvestri, Chiara Stagnaro, Rosaria Talarico, Angela Tincani, Ombretta Viapiana, Gianfranco Vitiello, Francesca Fabris, Sara Bindoli, Leonardo Punzi, Paola Galozzi, Paolo Sfriso
Background: Interleukin (IL)-1 plays a crucial role in the pathogenesis of Adult onset Still's disease (AOSD). Objectives: To evaluate the efficacy and safety of anakinra (ANA) and canakinumab (CAN) in a large group of AOSD patients. Methods: Data on clinical, serological features, and concomitant treatments were retrospectively collected at baseline and after 3, 6, and 12 months from AOSD patients (Yamaguchi criteria) referred by 18 Italian centers. Pouchot's score was used to evaluate disease severity. Results: One hundred forty patients were treated with ANA; 4 were subsequently switched to CAN after ANA failure...
2017: Frontiers in Pharmacology
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