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patients, disease, aosd, article, class, study, stills, google, scholar, pubmed, analysis, adultonset, clinical, survival, latent, relapse, table, onset, risk, data, systemic, pattern, cas, treatment, syndrome, median, mas, years, arthritis, age, ferritin, rates, adult, factors, death, group, japan, rheumatol, subgroups, cohort, model, chiba, information, included, received, criteria, characteristics, relapses, relapsefree, chronic,

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org/hematology/article/2009/1/127/19872/hemophagocytic-lymphohistiocytosis-hlh article download pdf kaplan-meier survival curve treatment-related adverse events relapse-free survival rates maintain long-term remission life-threatening adult-onset cox regression analysis clinical research centre genome-wide association study c-reactive protein full access catastrophic antiphospholipid syndrome shin-ichiro kagami model selection criteria 1093/rheumatology/keaa904 online ahead macrophage activation syndrome rapid reduction/withdrawal privacy choices/manage cookies thrombotic thrombocytopenic purpura elderly-onset aosd group erythrocyte sedimentation rate latent class analysis latent transition analysis kaplan-meier method multivariate model approach long-term prognosis disseminated intravascular coagulation item response probabilities aosd-related specific complications article sugiyama life-threatening complications relapse-free survival reactive haemophagocytic syndrome elevated liver enzymes reactive hemophagocytic syndrome asahi general hospital long-term treatment creative commons licence pediatr blood cancer higher neutrophil count median initial doses high systemic scores median neutrophil count semin arthritis rheum baseline patient/disease characteristics italian group published elderly-onset group adult haemophagocytic syndrome multicenter retrospective analysis

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         headline:Latent class analysis of 216 patients with adult-onset Still’s disease
         description:Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disease which encompasses patients with heterogenous presentation and a wide range of clinical courses. In this study, we aimed to identify potential subgroups of AOSD and reveal risk factors for relapse. We included a total of 216 AOSD patients who received treatment in nine hospitals between 2000 and 2019. All patients fulfilled the Yamaguchi classification criteria. We retrospectively collected information about baseline characteristics, laboratory tests, treatment, relapse, and death. We performed latent class analysis and time-to-event analysis for relapse using the Cox proportional hazard model. The median age at disease onset was 51.6 years. The median follow-up period was 36.8 months. At disease onset, 22.3% of the patients had macrophage activation syndrome. The median white blood cell count was 12,600/μL, and the median serum ferritin level was 7230 ng/mL. Systemic corticosteroids were administered in all but three patients (98.6%) and the median initial dosage of prednisolone was 40mg/day. Ninety-six patients (44.4%) were treated with concomitant immunosuppressants, and 22 (10.2%) were treated with biologics. Latent class analysis revealed that AOSD patients were divided into two subgroups: the typical group (Class 1: 71.8%) and the elderly-onset group (Class 2: 28.2%). During the follow-up period, 13 of 216 patients (6.0%) died (12 infections and one senility), and 76 of 216 patients (35.1%) experienced relapses. Overall and relapse-free survival rates at 5 years were 94.9% and 57.3%, respectively, and those rates were not significantly different between Class 1 and 2 (p=0.30 and p=0.19). Time-to-event analysis suggested higher neutrophil count, lower hemoglobin, and age ≥65 years at disease onset as risk factors for death and age ≥65 years at disease onset as a risk factor for relapse. AOSD patients were divided into two subgroups: the typical group and the elderly-onset group. Although the survival of patients with AOSD was generally good, the patients often experienced relapses. Age ≥65 years at disease onset was the risk factor for relapse.
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      headline:Latent class analysis of 216 patients with adult-onset Still’s disease
      description:Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disease which encompasses patients with heterogenous presentation and a wide range of clinical courses. In this study, we aimed to identify potential subgroups of AOSD and reveal risk factors for relapse. We included a total of 216 AOSD patients who received treatment in nine hospitals between 2000 and 2019. All patients fulfilled the Yamaguchi classification criteria. We retrospectively collected information about baseline characteristics, laboratory tests, treatment, relapse, and death. We performed latent class analysis and time-to-event analysis for relapse using the Cox proportional hazard model. The median age at disease onset was 51.6 years. The median follow-up period was 36.8 months. At disease onset, 22.3% of the patients had macrophage activation syndrome. The median white blood cell count was 12,600/μL, and the median serum ferritin level was 7230 ng/mL. Systemic corticosteroids were administered in all but three patients (98.6%) and the median initial dosage of prednisolone was 40mg/day. Ninety-six patients (44.4%) were treated with concomitant immunosuppressants, and 22 (10.2%) were treated with biologics. Latent class analysis revealed that AOSD patients were divided into two subgroups: the typical group (Class 1: 71.8%) and the elderly-onset group (Class 2: 28.2%). During the follow-up period, 13 of 216 patients (6.0%) died (12 infections and one senility), and 76 of 216 patients (35.1%) experienced relapses. Overall and relapse-free survival rates at 5 years were 94.9% and 57.3%, respectively, and those rates were not significantly different between Class 1 and 2 (p=0.30 and p=0.19). Time-to-event analysis suggested higher neutrophil count, lower hemoglobin, and age ≥65 years at disease onset as risk factors for death and age ≥65 years at disease onset as a risk factor for relapse. AOSD patients were divided into two subgroups: the typical group and the elderly-onset group. Although the survival of patients with AOSD was generally good, the patients often experienced relapses. Age ≥65 years at disease onset was the risk factor for relapse.
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PostalAddress:
      name:Department of Allergy and Clinical Immunology, Chiba University Hospital, Chiba, Japan
      name:Department of Allergy and Clinical Immunology, Chiba University Hospital, Chiba, Japan
      name:Department of Rheumatology and Allergy, Japanese Red Cross Narita Hospital, Chiba, Japan
      name:Department of Allergy and Clinical Immunology, Chiba University Hospital, Chiba, Japan
      name:Biostatistics Section, Clinical Research Centre, Chiba University Hospital, Chiba, Japan
      name:Department of Allergy and Clinical Immunology, Asahi General Hospital, Chiba, Japan
      name:Department of Rheumatology, Yokohama Rosai Hospital, Yokohama, Japan
      name:Third Department of Internal Medicine, University of Yamanashi, Yamanashi, Japan
      name:Department of Internal Medicine, Chiba Aoba Municipal Hospital, Chiba, Japan
      name:Department of Rheumatology, Dokkyo Medical University, Tochigi, Japan
      name:Third Department of Internal Medicine, University of Yamanashi, Yamanashi, Japan
      name:Center for Rheumatic Diseases, Chibaken Saiseikai Narashino Hospital, Chiba, Japan
      name:Biostatistics Section, Clinical Research Centre, Chiba University Hospital, Chiba, Japan
      name:Biostatistics Section, Clinical Research Centre, Chiba University Hospital, Chiba, Japan
      name:Department of Rheumatology, National Hospital Organization Shimoshizu Hospital, Chiba, Japan
      name:Department of Allergy and Clinical Immunology, Chiba University Hospital, Chiba, Japan

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