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JOURNAL ARTICLE
Takahito Harada, Yoshikazu Hirata, Hayato Kawamura, Yuki Yamamoto, Yuka Kobayashi, Daisuke Kumai, Akihisa Adachi, Yoshihito Nagura, Hirokazu Ikeuchi, Noriyuki Hayashi, Hiroki Takada, Satoshi Sobue, Megumi Yoshida
A 51-year-old woman with fever was admitted to our hospital. A computed tomography (CT) scan showed thickened colonic walls. Colonoscopy revealed erosion in the ileum and colon. Adult-onset Still's disease (AOSD) was diagnosed due to a subsequent sore throat and skin rash. Following AOSD treatment, methylprednisolone pulse therapy, followed by prednisolone and cyclosporine, was initiated. Despite achieving a temporary improvement, relapse occurred with fever, abdominal pain, with worsening CT and endoscopic findings...
April 16, 2024: Internal Medicine
#2
P V Akhila Arya, Erica Marnet, Madhumita Rondla, Jia Wei Tan, Dileep Unnikrishnan, Gregory Buller
OBJECTIVE: We aimed to review the literature on the clinical presentation, renal pathology, treatment, and outcome of renal manifestations in adult-onset Still's disease (AOSD). METHODS: We used PRISMA guidelines for our systematic review and included all English-language original articles from inception till September 15, 2023, on AOSD and kidney involvement in any form. Data on patient demographics, diagnostic criteria, clinical presentation, renal pathology, treatment employed including dialysis, outcome, cause of death were collected and analyzed...
April 16, 2024: Rheumatology International
#3
JOURNAL ARTICLE
Antonio Gallardo-Pizarro, Valerio Campos-Rodríguez, Daniel Martín-Iglesias, Guillermo Ruiz-Irastorza
AIM: This study addresses the challenge of predicting the course of Adult-onset Still's disease (AoSD), a rare systemic autoinflammatory disorder of unknown origin. Precise prediction is crucial for effective clinical management, especially in the absence of specific laboratory indicators. METHODS: We assessed the effectiveness of combining traditional biomarkers with the k-medoids unsupervised clustering algorithm in forecasting the various clinical courses of AoSD-monocyclic, polycyclic, or chronic articular...
April 2024: International Journal of Rheumatic Diseases
#4
JOURNAL ARTICLE
Yuri Shirahama, Ayako Kokuzawa, Yusuke Yamauchi, Yohei Kirino, Hideto Nagai, Yasushi Inoue, Toshiyuki Ota, Yutaka Chifu, Hiroki Mitoma, Mitsuteru Akahoshi, Mariko Sakai, Akihito Maruyama, Akihide Ohta, Masahiro Iwamoto, Yoshifumi Tada
OBJECTIVES: To define groups and characterize differences in the prognosis of patients with adult-onset Still's disease (AOSD). METHODS: We performed a retrospective cohort study. Patients with AOSD were grouped using hierarchical unsupervised cluster analysis according to age, sex, clinical features, and laboratory data. The primary endpoints were overall survival and drug-free remission rate. RESULTS: A total of 153 patients with AOSD were placed into four clusters...
March 6, 2024: Modern Rheumatology
#5
JOURNAL ARTICLE
Hiroya Tamai, Yasushi Kondo, Tsutomu Takeuchi, Yuko Kaneko
OBJECTIVES: Tocilizumab, an IL-6 inhibitor, has been proven effective in patients with adult-onset Still's disease (AOSD). This study aimed to clarify whether tocilizumab can be discontinued after achieving remission and to identify factors relevant to its successful discontinuation. METHODS: Consecutive patients with AOSD diagnosed according to Yamaguchi's criteria from April 2012 to July 2022, who were treated with tocilizumab, were retrospectively reviewed. RESULTS: Forty-eight patients with AOSD treated with intravenous tocilizumab, with sufficient information, were included...
March 20, 2024: Rheumatology
#6
JOURNAL ARTICLE
Piero Ruscitti, Francesco Masedu, Antonio Vitale, Valeria Caggiano, Ilenia Di Cola, Paola Cipriani, Marco Valenti, Henrique A Mayrink Giardini, Isabele Parente de Brito Antonelli, Marilia Ambiel Dagostin, Giuseppe Lopalco, Florenzo Iannone, Morrone Maria, Ibrahim A Almaghlouth, Kazi Nur Asfina, Hebatallah Hamed Ali, Francesco Ciccia, Daniela Iacono, Ilenia Pantano, Daniele Mauro, Petros P Sfikakis, Maria Tektonidou, Katerina Laskari, Onorina Berardicurti, Lorenzo Dagna, Alessandro Tomelleri, Abdurrahman Tufan, Rıza Can Kardas, Andrea Hinojosa-Azaola, Eduardo Martín-Nares, Perla Ayumi Kawakami-Campos, Gaafar Ragab, Mohamed Tharwat Hegazy, Haner Direskeneli, Fatma Alibaz-Oner, Lampros Fotis, Paolo Sfriso, Marcello Govoni, Francesco La Torre, Maria Cristina Maggio, Carlomaurizio Montecucco, Ludovico De Stefano, Serena Bugatti, Silvia Rossi, Joanna Makowska, Emanuela Del Giudice, Giacomo Emmi, Elena Bartoloni, José Hernández-Rodríguez, Giovanni Conti, Alma Nunzia Olivieri, Alberto Lo Gullo, Gabriele Simonini, Ombretta Viapiana, Ewa Wiesik-Szewczyk, Sukran Erten, Francesco Carubbi, Amato De Paulis, Armin Maier, Samar Tharwat, Stefania Costi, Annamaria Iagnocco, Gian Domenico Sebastiani, Antonio Gidaro, Antonio Luca Brucato, Anastasios Karamanakos, Nurullah Akkoç, Francesco Caso, Luisa Costa, Marcella Prete, Federico Perosa, Fabiola Atzeni, Giuliana Guggino, Claudia Fabiani, Bruno Frediani, Roberto Giacomelli, Luca Cantarini
OBJECTIVES: To evaluate the clinical usefulness of the systemic score in the prediction of life-threatening evolution in Still's disease. To assess the clinical relevance of each component of the systemic score in predicting life-threatening evolution and to derive patient subsets accordingly. METHODS: A multicenter, observational, prospective study was designed including patients included in the GIRRCS (Gruppo Italiano Di Ricerca in Reumatologia Clinica e Sperimentale) AOSD-study group and AIDA (AutoInflammatory Disease Alliance) Network Still's Disease Registry...
March 18, 2024: Arthritis & Rheumatology
#7
JOURNAL ARTICLE
Dehao Zhu, Longfang Chen, Jianfen Meng, Mengyan Wang, Yuning Ma, Xia Chen, Yu Xiao, Da Yi, Hui Shi, Yue Sun, Honglei Liu, Xiaobing Cheng, Yutong Su, Junna Ye, Huihui Chi, Zhuochao Zhou, Chengde Yang, Jialin Teng, Jinchao Jia, Qiongyi Hu
Macrophage activation syndrome (MAS) is a potentially fatal consequence of adult-onset Still's disease (AOSD), driven by a cytokine storm. Efficient early diagnosis of AOSD-associated MAS requires a sensitive and specific biomarker. In this study, we demonstrated that pentraxin 3 (PTX3), an acute phase protein, was associated with AOSD disease activity and served as a biomarker for AOSD-MAS. PTX3 levels were significantly increased in AOSD patients compared to other autoimmune diseases and healthy controls...
March 8, 2024: Journal of Autoimmunity
#8
Noriharu Nakagawa, Ai Fujii, Yoshimichi Ueda, Masahide Yamazaki
KEY CLINICAL MESSAGE: The present case indicates that cryoglobulinemia vasculitis should be considered in the differential diagnosis of purpura in patients with adult-onset Still's disease (AOSD). ABSTRACT: The presence of purpura is suggested in adult-onset Still's disease (AOSD) hematological complications of hemophagocytic syndrome, disseminated intravascular coagulation, or thrombotic microangiopathy. We herein report a case of AOSD complicated by cryoglobulinemia vasculitis presenting with purpura...
March 2024: Clinical Case Reports
#9
REVIEW
Sara Bindoli, Chiara Baggio, Andrea Doria, Paolo Sfriso
Adult-onset Still's disease (AOSD) is a multisystemic complex disorder clinically characterised by episodes of spiking fever, evanescent rash, polyarthritis or diffuse arthralgias; multiorgan involvement may develop according to the hyper-inflammatory extent. The pathogenesis of AOSD is not completely recognised. The central role of macrophage activation, which results in T helper 1 (Th1) cell cytokine activation, is well established. Pro-inflammatory cytokines such as interleukin (IL)-1, IL-6 and IL-18 play a fundamental role in disease onset and progression...
March 5, 2024: Drugs
#10
Daniele Sola, Carlo Smirne, Francesco Bruggi, Chiara Bottino Sbaratta, Aubin Cardin Tamen Njata, Guido Valente, Maria Cristina Pavanelli, Rosetta Vitetta, Mattia Bellan, Lorenzo De Paoli, Mario Pirisi
Adult-onset Still's disease (AOSD) is a rare systemic inflammatory disorder. Diagnosis can take a long time, especially in the presence of confounding factors, and it is, to some extent, a process of exclusion. AOSD has life-threating complications ranging from asymptomatic to severe, such as macrophage activation syndrome (MAS), which is also referred to as hemophagocytic lymphohistocytosis (HLH). This condition is correlated with cytokine storm production and monocyte/macrophage overactivation and typically occurs with rash, pyrexia, pancytopenia, hepatosplenomegaly and systemic involvement...
January 29, 2024: Life
#11
JOURNAL ARTICLE
Valentina Myachikova, Igor Kudryavtsev, Artem Rubinstein, Arthur Aquino, Dmitry Isakov, Alexey Golovkin, Alexey Maslyanskiy
Adult-onset Still's disease (AOSD) is a complex systemic inflammatory disorder, categorized as an 'IL-1 driven' inflammasomapathy. Despite this, the interaction between T and B cells remains poorly understood. We conducted a study, enrolling 7 patients with relapsing AOSD and 15 healthy control subjects, utilizing deep flow cytometry analysis to examine peripheral blood T- and B-cell subsets. T-cell and B-cell subsets were significantly altered in patients with AOSD. Within CD4+ T cells, Th2 cells were decreased...
February 1, 2024: Current Issues in Molecular Biology
#12
Yoichi Kurosawa, Sayuri Takamura, Ayako Wakamatsu, Daisuke Kobayashi, Ichiei Narita
Adult-onset Still's disease (AOSD) causes fever, rash, pharyngalgia, and arthralgia through autoinflammation. Its complement titer has not previously received attention because this usually increases during the inflammatory process. Our female patient in her 60s was admitted to the hospital with fever, rash, arthralgia, and pharyngalgia. Her white blood cell count was 19,130/μL, hemoglobin was 11.0 g/dL, platelet count was 26.0 × 104 /μL, and ferritin titer was 6,175 ng/mL. Anti-nuclear antibodies and anti-neutrophil cytoplasmic antibodies were negative...
January 2024: Curēus
#13
Anh N Le, Nino Gudushauri, Irene J Tan, Rekha Bhat, Rasha Khan
Adult-onset Still's disease (AOSD) is a rare multi-systemic inflammatory disorder characterized by high spiking fevers, nonpruritic, salmon-colored rash, and severe polyarthralgia. Laboratory features typically include elevation in white blood cells, liver enzymes, and ferritin. Central nervous system and cardiac involvements, particularly myocarditis, are rare. Macrophage activation syndrome (MAS) is a well-described complication of AOSD, leading to a high mortality rate. Herein, we describe a case of AOSD complicated by MAS in a 32-year-old male presenting with atypical clinical manifestations, including recurrent seizures, scaly, pruritic, and hyperpigmented rash, and right heart failure due to lymphocytic myocarditis...
January 2024: Curēus
#14
REVIEW
Satoshi Suzuki, Yuko Kataoka, Tomoya Otani, Yukino Taniguchi, Keigo Ikeda, Naoto Tamura, Shinji Morimoto
Adult-onset still's disease is a rare condition that is generally treated by glucocorticoids. Importantly, due to the limited established treatments, glucocorticoid-refractory cases are particularly difficult to treat. Between December 2009 and August 2022, nine patients with adult-onset Still's disease were treated with tocilizumab (tocilizumab group). The therapeutic efficacy and safety of tocilizumab initiation in the acute phase were evaluated in cases of initial onset and recurrence. We also compared the efficacy of tocilizumab with that of methotrexate (methotrexate group, n = 13), which has been the drug of choice for adjunctive therapy...
February 12, 2024: Clinical Rheumatology
#15
Ken Nagahata, Kazuyuki Murase, Masatoshi Kanda, Hiroki Takahashi
A 27-year-old man presented with quotidian fever, rash, knee arthralgia, sore throat, and bloody diarrhea. Laboratory findings showed neutrophilia, elevated CRP, ferritin, and liver enzyme levels, and decreased hemoglobin levels. Radiological investigations revealed splenomegaly, systemic lymphadenopathy, thickening of the descending colon wall, and an abnormal uptake in the bone marrow and spleen as seen in F-fluorodeoxyglucose positron emission tomography. Malignant lymphoma was initially suspected, but biopsies showed no malignant findings...
January 15, 2024: JMA journal
#16
Hiroaki Nishioka, Shogo Shirota
Adult-onset Still's disease (AOSD) is an uncommon autoinflammatory disorder without a clear etiology that primarily affects young adults. New-onset disease at > 80 years of age is uncommon. We present the case of an 82-year-old woman with AOSD which developed after receiving a messenger ribonucleic acid (mRNA) coronavirus disease 2019 (COVID-19) vaccine. COVID-19 vaccines are known to cause overproduction of cytokines, systemic inflammation, and some immune-mediated adverse events, such as rheumatoid arthritis, systemic lupus erythematosus, dermatomyositis, vasculitis, and polymyalgia rheumatica after the vaccination has been reported...
January 2024: Curēus
#17
JOURNAL ARTICLE
Piero Ruscitti, Dennis McGonagle, Viviam Canon Garcia, Hilde Rabijns, Katrin Toennessen, Mary Chappell, Mary Edwards, Paul Miller, Neil Hansell, Joe Moss, Sara Graziadio, Eugen Feist
OBJECTIVE: To conduct a systematic review of the effectiveness and safety of pharmacological treatments for adult-onset Still's disease (AOSD). METHODS: Six databases, two trial registries and conference abstracts were searched from 2012 to February 2023 for studies of pharmacological interventions in people with AOSD. Outcomes were rates of remission and response, discontinuation of concurrent treatments, complications of AOSD and treatment-related adverse events...
February 1, 2024: Journal of Rheumatology
#18
JOURNAL ARTICLE
Nadia Riera, Cecilia Salazar, Bernardina Rivera, Antonio Galiana, Rosario Durán, María Magdalena Portela, Virginia Antelo, Beatriz Pi, Óscar González, Gregorio Iraola
Here we report a case of septic arthritis associated with a genetically divergent Francisella philomiragia strain in a patient with chronic rheumatoid arthritis and Adult-onset Still's disease (AOSD) in Maldonado, Uruguay. In this study mass spectrometry together with whole-genome sequencing using Oxford Nanopore technology allowed for the correct identification of the etiologic agent.
March 2024: New Microbes and New Infections
#19
JOURNAL ARTICLE
Marie-Christin Hoffmann, Giulio Cavalli, Natalie Fadle, Eleonora Cantoni, Evi Regitz, Octavian Fleser, Philipp Klemm, Marina Zaks, Elisabeth Stöger, Corrado Campochiaro, Alessandro Tomelleri, Elena Baldissera, Jörg Thomas Bittenbring, Vincent Zimmer, Jochen Pfeifer, Yvan Fischer, Klaus-Dieter Preuss, Moritz Bewarder, Bernhard Thurner, Sabrina Fuehner, Dirk Foell, Lorenzo Dagna, Christoph Kessel, Lorenz Thurner
BACKGROUND: Adult-onset Still's disease (AOSD) and systemic juvenile idiopathic arthritis (sJIA) resemble a continuum of a rare, polygenic IL-1β-driven disease of unknown etiology. OBJECTIVE: In the present study we sought to investigate a potential role of recently described autoantibodies neutralizing the interleukin-1(IL-1)-receptor antagonist (IL-1-Ra) in the pathogenesis of Still's disease. METHODS: Serum or plasma samples from Still's disease patients (AOSD, n = 23; sJIA, n = 40) and autoimmune and/or inflammatory disease controls (n = 478) were analyzed for autoantibodies against progranulin (PGRN), IL-1Ra, IL-18 binding protein (IL-18BP), and IL-36Ra, as well as circulating IL-1Ra and IL-36Ra levels by ELISA...
January 17, 2024: Journal of Clinical Immunology
#20
JOURNAL ARTICLE
Xinyue Hong, Xiaoming Wang, Ningqi Dai, Yue Sun, Honglei Liu, Xiaobing Cheng, Junna Ye, Hui Shi, Qiongyi Hu, Jianfen Meng, Zhuochao Zhou, Chengde Yang, Jialin Teng, Yutong Su, Huihui Chi
INTRODUCTION: This study aimed to characterize the morbidity, hospitalization, and mortality rates among patients with adult-onset Still's disease (AOSD) affected by coronavirus disease 2019 (COVID-19) and explore the impact of COVID-19 on the disease activity of AOSD. METHODS: Data on the clinical and demographic characteristics, COVID-19-related symptoms, and outcomes were retrospectively collected. Patients were stratified according to COVID-19 severity and associations between risk factors and outcomes were analyzed using multivariate logistic regression...
January 6, 2024: Rheumatology and Therapy
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