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• W2885874960 abstract "Question: A 26-year-old Caucasian man presented with a 1-month history of gradually deteriorating ulcerated skin lesions affecting both the inguinal regions (Figure A). He had a long history of ileocolonic Crohn’s disease with perianal involvement. Magnetic resonance imaging of the pelvis showed no underlying abscesses or fistulous tract in the inguinal regions extending to the bowel but active perianal Crohn’s disease. He had undergone a diverting ileostomy 12 months before this presentation for anti-tumor necrosis factor–refractory disease and had been receiving vedolizumab for disease control. Four months into treatment with vedolizumab, he developed a papulopustular rash on his left foot owing to acute bacterial folliculitis. This condition was treated with antibiotics with good response and vedolizumab was not discontinued at this time. Over the last few months, he had no significant change in his intestinal symptoms but had a raised C-reactive protein of 56. Chest radiography was unremarkable. IFN-γ assay was negative. What is the diagnosis? See the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI. Inguinal skin biopsy showed an inflammatory sinus with a lymphoplasmacytic and histiocyte-rich infiltrate including discrete, noncaseating granulomas with abundant multinucleated giant cells in the dermis and adipose tissue (Figure B). There was no evidence of primary follicular or sweat gland involvement and Ziehl Neelson stain was negative. The presence of discrete epithelioid granulomas in the dermis away from the site of active inflammation raised the possibility of a systemic granulomatous disease such as Crohn's disease and hence a diagnosis of metastatic inguinal Crohn’s disease (MCD) was made. Hidradenitis suppurativa does not usually show a granulomatous infiltrate, other than as a foreign body reaction to keratin, which was absent in this case, and there did not seem to be a primary folliculitis or sweat gland involvement, which is the hallmark of hidradenitis. Infections are always a possibility, but the lack of central necrosis was against this. Sarcoidosis does not usually present as such a generalized inflammatory process, in addition to the granulomas.1Siroy A. Wasman J. Metastatic Crohn disease: a rare cutaneous entity.Arch Pathol Lab Med. 2012; 136: 329-332Crossref PubMed Scopus (48) Google Scholar Owing to the inadequate response to vedolizumab, he was started on ustekinumab along with topical wound care. At 12 months after treatment with ustekinumab, his inguinal ulcerations had completely healed. Repeat magnetic resonance imaging of the pelvis showed vast improvement in his perianal inflammation and ileoscopy showed superficial ulcers in the first 10 cm of neoileum. He remains to ustekinumab with the aim to achieve sustained disease remission. MCD is a rare clinical entity with limited reports in literature. Its etiology remains mostly unknown, although it is hypothesized to be an autoimmune response to gut antigens.2Teixeira M. Machado S. Lago P. et al.Cutaneous Crohn's disease.Int J Dermatol. 2006; 45: 1074-1076Crossref Scopus (14) Google Scholar Nodules, ulcerations, pustules, plaques, abscesses, or papules on both extremities and less commonly in the genital area may all be dermatological manifestations of this mind-puzzling mime. Therefore, similar to our case, the definite diagnosis mainly relies on histopathogy.1Siroy A. Wasman J. Metastatic Crohn disease: a rare cutaneous entity.Arch Pathol Lab Med. 2012; 136: 329-332Crossref PubMed Scopus (48) Google Scholar Its diagnosis requires a high index of suspicion, but a previous diagnosis of Crohn’s disease may be of additional benefit along with the presence of noncaseating granulomas for a lesion to be classified as MCD. To date, treatment success has been reported with antibiotics, surgery and immunosuppressives.3Bender-Heine A. Grantham J.T. Zaslau S. et al.Metastatic Crohn disease: a review of dermatologic manifestations and treatment.Cutis. 2017; 99: E33-E40Google Scholar This case was refractory to all these pharmacologic treatments, with ustekinumab being our last possible solution. However, it has proved to be a promising regimen in the management of this patient’s severe MCD and could be considered as an alternative solution for complicated cases, before more invasive surgical interventions." @default.
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• W2885874960 date "2018-11-01" @default.
• W2885874960 modified "2023-09-26" @default.
• W2885874960 title "Multiple Unusual Ulcerated Skin Lesions in a Crohn's Disease Patient" @default.
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• W2885874960 doi "https://doi.org/10.1053/j.gastro.2018.07.042" @default.
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