Dear Sir, Sweet’s syndrome (SS) is a reactive process that is characterized by the abrupt appearance of tender erythematous to violaceous plaque-forming papules and nodules, accompanied by fever and dermal neutrophil infiltrate in the absence of vasculitis. The association with inflammatory bowel disease (IBD) is known; however, there are few published cases in the literature. A 25-year-old man with ulcerative colitis presented to our hospital with diarrhoea, hematochezia and fevers. He had been diagnosed with ulcerative colitis 9 months prior to his admission. His outpatient medications were mesalazine and azathioprine due to steroid dependence. He had discontinued azathioprine due to intolerance. Initial laboratory evaluation demonstrated anaemia, leukocytosis and elevated C-reactive protein (364mg/L). Intravenous methylprednisolone (1mg/kg/day) was initiated for suspected ulcerative colitis flare. On Hospital Day 3, numerous violet oedematous plaques appeared all over the surface of the body except in mucous membranes, palms and feet (Figure 1A and B). These cutaneous lesions were biopsied, revealing neutrophilic dermatitis with discrete oedema in the absence of vasculitis, confirming the clinical suspicion of SS (Figure 1D). Upon excluding possible opportunistic infections, superinfection of the skin lesions and lymphoproliferative disorders and due to the lack of response to treatment with corticosteroids, infliximab 5mg/kg was started for induction. Two days after receiving infliximab, the patient’s violet plaques and blisters began to heal (Figure 1C). Figure 1. View largeDownload slide (A, B) Painful erythematous edematous plaques all over the surface of the body except in mucous membranes, palms and feet, 48h after admission. (D) Histopathological study shows neutrophilic dermatitis with a discrete edema in the absence of vasculitis. (C) Blisters show a favourable development towards the healing after to begin Infliximab 5mg/kg. Figure 1. View largeDownload slide (A, B) Painful erythematous edematous plaques all over the surface of the body except in mucous membranes, palms and feet, 48h after admission. (D) Histopathological study shows neutrophilic dermatitis with a discrete edema in the absence of vasculitis. (C) Blisters show a favourable development towards the healing after to begin Infliximab 5mg/kg. SS is classified into subgroups: classic/idiopathic, parainflammatory, paraneoplastic, pregnancy-induced and drug-induced. Up to 70% of SS cases are idiopathic, and it occurs predominantly in young women after a mild respiratory illness and may be linked IBD. SS is a rare extra-intestinal manifestation of IBD, and occurs more commonly in middle-aged women with Crohn’s disease and in parallel with IBD flares.1,2 It is important to note that SS can manifest as a drug-induced injury, as well, with the most common culprit being granulocyte colony-stimulating factor (G-CSF). Something to keep in mind in the differential diagnosis is azathioprine hypersensitivity syndrome3; this is an idiosyncratic reaction and tends to occur in the first 4 weeks of azathioprine therapy. The typical lesions are cutaneous eruptions with neutrophilic dermatosis in the biopsy. Literature highlights that about half of these patients suffer from IBD or multiple sclerosis While there are case reports of SS occurring in patients on anti-TNF therapy,4 our study, similar to others that have been published, supports the use of anti-TNF therapy as a therapeutic option in Sweet’s syndrome in patients who do not respond to corticosteroid therapy or who are intolerant to immunomodulators.5 Funding No specific funding has been received. Conflict of Interest The authors declare that they have no conflict of interest. Author Contributions All authors have contributed equally to writing the manuscript. References 1. Harbord M , Annese V , Vavricka SR et al. ; European Crohn’s and Colitis Organisation . The first European evidence-based consensus on extra-intestinal manifestations in inflammatory bowel disease . J Crohns Colitis 2016 ; 10 : 239 – 54 . Google Scholar CrossRef Search ADS PubMed 2. Pintado JCA , Abad AS , Zapata A , Campos EM . Síndrome de Sweet y enfermedad inflamatoria intestinal . Aportación de un nuevo caso y revisión de la literatura . 2002 ; 19 : 419 – 22 . 3. Tien V , Jones AD , Aronowitz PB . Drug-induced Sweet’s syndrome . J Gen Intern Med 2017 ; 32 :953– 4 . 4. Banse C , Sobocinski V , Savoye G , Avenel G , Vittecoq O . Occurrence of Sweet syndrome under anti-TNF . Clin Rheumatol 2015 ; 34 : 1993 – 4 . Google Scholar CrossRef Search ADS PubMed 5. Foster EN , Nguyen KK , Sheikh RA , Prindiville TP . Crohn’s disease associated with Sweet’s syndrome and Sjögren’s syndrome treated with infliximab . Clin Dev Immunol 2005 ; 12 : 145 – 9 . Google Scholar CrossRef Search ADS PubMed Copyright © 2018 European Crohn’s and Colitis Organisation (ECCO). Published by Oxford University Press. All rights reserved. For permissions, please email: firstname.lastname@example.org This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/about_us/legal/notices)
Journal of Crohn's and Colitis – Oxford University Press
Published: Mar 21, 2018
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