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      Adult Onset Still's Disease as a Cause of Acute Severe Mitral and Aortic Regurgitation

      case-report

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          Abstract

          Adult onset Still's disease (AOSD) is an uncommon acute systemic inflammatory disease of unknown origin. The clinical features include high spiking fever, arthralgia or arthritis, transient maculopapular rash, lymphadenopathy, hepatosplenomegaly, and serositis. Pericarditis is the most common cardiac manifestation of AOSD and occurs in approximately 30% of cases. A simultaneous occurrence of rapidly progressive bi-valvular regurgitation associated with AOSD has not been previously described. We report a case of a 55-year old woman who underwent mitral valve replacement and Bentall's operation due to acute severe mitral and aortic regurgitation associated with AOSD.

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          Still's disease in the adult.

          E Bywaters (1971)
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            Adult Still's disease: review of 228 cases from the literature.

            A Ohta, H Kaneoka, T Nagayoshi … (1987)
            To clarify the clinical pictures of adult Still's disease, 228 cases reported in the past 15 years since Bywaters' first description were reviewed. These included our 9 new cases and an additional 25 cases from the Japanese literature, none of which had been described in previous English reviews. Most of the patients with long followup showed frequent recurrences. About one third developed deforming arthritis with ankylosis. There were 6 deaths. Of interest was the remarkably elevated levels of serum ferritin and prostaglandin E1 in some patients.
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              Methotrexate treatment in patients with adult onset Still's disease--retrospective study of 13 Japanese cases.

              H Kameda, T. Fujii, M Akizuki … (1997)
              To evaluate methotrexate treatment in patients with active adult onset Still's disease (AOSD). Methotrexate was initially given as a single weekly oral dose of 5 mg and adjusted individually afterwards in 13 patients with active AOSD. Symptoms and laboratory findings were investigated. Signs of AOSD activity disappeared (remission) in eight patients between 3 and 16 weeks after starting methotrexate. In these patients, significant improvements in C reactive protein, erythrocyte sedimentation rate, white blood count, and serum ferritin were observed at 8, 12, 14, and 16 weeks after starting methotrexate, respectively. In six of these eight patients, steroids or non-steroidal anti-inflammatory drugs could be reduced or discontinued. In four patients methotrexate was not effective despite 12 or 16 weeks of treatment, and one patient discontinued treatment after 2 weeks because of severe nausea. Five patients suffered from adverse reactions, including acute interstitial pneumonia (one patient) and liver toxicity (two patients). Five out of eight patients successfully treated with methotrexate were HLA-DR4 positive (four homozygotes), and all the unsuccessfully treated patients were DR2 positive. Methotrexate is useful for controlling disease activity in AOSD, not only for refractory patients but also for patients who have never taken steroids or for those with steroid associated toxicity. However, serious adverse reactions can occur, as with rheumatoid arthritis. It is important to determine the critical factors, such as the immunogenetic background, that are associated with response to methotrexate treatment.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Korean J Intern Med
                Journal ID (iso-abbrev): Korean J. Intern. Med
                Journal ID (publisher-id): KJIM
                Title: The Korean Journal of Internal Medicine
                Publisher: The Korean Association of Internal Medicine
                ISSN (Print): 1226-3303
                ISSN (Electronic): 2005-6648
                Publication date (Print): September 2005
                Publication date (Electronic): 30 September 2005
                Volume: 20
                Issue: 3
                Pages: 264-267
                Affiliations
                Department of Internal Medicine, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.
                [2 ]Department of Cardiovascular Surgery, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.
                Author notes
                Correspondence to: Chang-Keun Lee, M.D., Ph.D., Department of Internal Medicine, University of Ulsan, College of Medicine, Asan Medical Center, 388-1 Pungnap-dong, Songpa-gu, Seoul 138-736, Korea. Tel: 82-2-3010-3284, Fax: 82-2-3010-6969, cklee@ 123456amc.seoul.kr
                Article
                DOI: 10.3904/kjim.2005.20.3.264
                PMC ID: 3891165
                PubMed ID: 16295789
                SO-VID: e11868ec-590a-4e72-99bd-521d734c25d8
                Copyright © Copyright © 2005 The Korean Association of Internal Medicine
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 27 January 2005
                Date accepted : 18 March 2005
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Internal medicine
                Keywords: adult onset still's disease,mitral regurgitation,aortic regurgitation
                Data availability:
                ScienceOpen disciplines: Internal medicine
                Keywords: adult onset still's disease, mitral regurgitation, aortic regurgitation

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