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      Disfiguring Annular Sarcoidosis Improved by Adalimumab

      case-report

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          Abstract

          Depending on the location, dermatoses can produce blemishes that severely impair quality of life and require highly effective treatment that is otherwise used for extensive skin involvement. We report the case of a 39-year-old, otherwise healthy male disfigured by an 8 × 7-cm hypopigmented and centrally atrophic annular plaque with erythematous indurated borders in an area of scar tissue on his forehead. Skin biopsies revealed non-caseating granulomas, and hilar involvement was identified, leading to the diagnosis of systemic sarcoidosis stage II with cutaneous involvement. The lesions proved resistant to multiple therapies, but responded within 4 months to adalimumab with regression of the lesion and inflammatory infiltrate. The visual analogue scale of disease activity decreased from 7/10 to 3.5/10, and the Dermatology Life Quality Index from 16/30 to 3/30 points. In conclusion, TNF-α inhibition can control inflammation and disfigurement by cutaneous sarcoidosis and restore quality of life.

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          Most cited references5

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          Tumour necrosis factor alpha restores granulomas and induces parasite egg-laying in schistosome-infected SCID mice.

          Schistosomiasis (bilharzia) is a parasitic disease caused by several species of schistosome worms (blood flukes). The key pathogenic event in this disease is the formation of granulomas around schistosome eggs trapped in portal venules of the liver. Granulomas are a distinctive form of chronic inflammation characterized by localized aggregation of activated macrophages around an inciting stimulus. Each granuloma evolves to form a fibrous scar; in schistosomiasis, the result is widespread hepatic fibrosis and portal hypertension. To identify the specific immune signal molecules necessary for granuloma formation, we studied schistosome infections in severe combined immunodeficient (SCID) mice, which have normal macrophages but lack functional B or T lymphocytes. Here we report that the immunoregulatory cytokine tumour necrosis factor alpha is necessary and sufficient to reconstitute granuloma formation in schistosome-infected SCID mice. Moreover, we find that the parasitic worms require tumour necrosis factor alpha for egg-laying and for excretion of eggs from the host. The implication of this latter result is that the parasite has adapted so successfully to its host that it uses a host-derived immunoregulatory protein as a signal for replication and transmission.
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            Psoriasis induced by tumor necrosis factor-alpha antagonist therapy: a case series.

            Although tumor necrosis factor-alpha (TNF-alpha) antagonists are effective in the treatment of refractory psoriasis, some cases have suggested that psoriasis might be induced as a result of treatment prescribed mainly for rheumatoid arthritis, ankylosing spondylitis, and Crohn's disease. To investigate anti-TNF-alpha induced psoriasis, we conducted a systematic analysis of the 6 cases we observed among our inflammatory patient cohort treated with anti-TNF-alpha (infliximab or etanercept). We report 6 cases of psoriasis with onset during TNF-alpha antagonist therapy (infliximab and etanercept); characteristics and skin lesions are described. No patient had a personal or family history of psoriasis. The development of psoriasis was seen in all the types of inflammatory diseases we treated with TNF-alpha antagonists. There was great variation in the age of affected patients and in the onset of psoriasis after initiation of TNF-alpha antagonists. Both TNF-alpha antagonists studied were associated with development of psoriasis. In 2 cases psoriasis was associated with 2 different TNF-alpha antagonists in the same patient. In half our patients, skin lesions started in the inguinal and pubic regions, but palmoplantar pustulosis was also common. In half the cases, skin lesions responded favorably with topical agents despite continuation of TNF-alpha antagonist therapy. In light of previously published cases describing psoriasis or psoriasiform lesions after TNF-alpha antagonist therapy, our series strongly confirms that TNF-alpha antagonists may induce psoriasis in some patients. Further studies are needed to identify risk factors for TNF-alpha antagonist induced psoriasis.
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              Lung sarcoidosis induced by TNF antagonists in rheumatoid arthritis: a case presentation and a literature review.

              We report on a 72 year-old woman with long-standing rheumatoid arthritis diagnosed as granulomatosis due to pulmonary sarcoidosis after 49 months of treatment with etanercept. A clinical and radiological improvement was seen after tumor necrosis factor (TNF) antagonist withdrawal plus a course of steroids. Currently, 27 cases of histological proven sarcoidosis with pulmonary involvement have been reported in relation to anti-TNF therapy, with etanercept being more frequent in comparison with the anti-TNF monoclonal antibodies infliximab and adalimumab. Potential pathogenic mechanisms of the paradoxical effect of anti-TNF treatment is discussed. It is important for clinicians to be aware of this potential and uncommon complication of biological therapy with TNF antagonists. Copyright © 2010 SEPAR. Published by Elsevier Espana. All rights reserved.
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                Author and article information

                Journal
                Case Rep Dermatol
                CDE
                Case Reports in Dermatology
                S. Karger AG (Allschwilerstrasse 10, P.O. Box · Postfach · Case postale, CH-4009, Basel, Switzerland · Schweiz · Suisse, Phone: +41 61 306 11 11, Fax: +41 61 306 12 34, karger@karger.ch )
                1662-6567
                May-Aug 2011
                17 May 2011
                17 May 2011
                : 3
                : 2
                : 103-106
                Affiliations
                Department of Dermatology, University Hospital of Zurich, Zurich, Switzerland
                Author notes
                *Dr. Alexander Navarini, Department of Dermatology, University Hospital of Zurich, Gloriastrasse 31, CH-8091 Zurich (Switzerland), Tel. +41 44 255 1111, E-Mail Alexander.Navarini@ 123456usz.ch
                Article
                cde0003-0103
                10.1159/000328796
                3104865
                21677887
                368f80b3-6f94-448e-800d-f964e03844cb
                Copyright © 2011 by S. Karger AG, Basel

                This is an Open Access article distributed under the terms of the Creative Commons Attribution-Noncommercial-No-Derivative-Works License ( http://creativecommons.org/licenses/by-nc-nd/3.0/). Users may download, print and share this work on the Internet for noncommercial purposes only, provided the original work is properly cited, and a link to the original work on http://www.karger.com and the terms of this license are included in any shared versions.

                History
                Page count
                Figures: 1, References: 5, Pages: 4
                Categories
                Published: May 2011

                Dermatology
                tnf-α antagonist,systemic sarcoidosis,sarcoidosis treatment,adalimumab,cutaneous sarcoidosis

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