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Rare mucocutaneous manifestations of ulcerative colitis: A case report of pyostomatitis vegetans and Sweet syndrome

  • Xiamei Zeng
    Affiliations
    Department of Oral Medicine, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Laboratory for Digital and Material Technology of Stomatology & Beijing Key Laboratory of Digital Stomatology & Research Center of Engineering and Technology for Computerized Dentistry Ministry of Health & NMPA Key Laboratory for Dental Materials, Beijing, China

    Stomatological Hospital of Xiamen Medical College, Xiamen Key Laboratory of Stomatological Disease Diagnosis and Treatment, Xiamen, China
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  • Hong Hua
    Affiliations
    Department of Oral Medicine, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Laboratory for Digital and Material Technology of Stomatology & Beijing Key Laboratory of Digital Stomatology & Research Center of Engineering and Technology for Computerized Dentistry Ministry of Health & NMPA Key Laboratory for Dental Materials, Beijing, China
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  • Xiaosheng Hu
    Correspondence
    Corresponding author: Xiaosheng Hu.
    Affiliations
    Department of Oral Medicine, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Laboratory for Digital and Material Technology of Stomatology & Beijing Key Laboratory of Digital Stomatology & Research Center of Engineering and Technology for Computerized Dentistry Ministry of Health & NMPA Key Laboratory for Dental Materials, Beijing, China
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      Inflammatory bowel diseases (IBD) are chronic inflammatory disorders of the digestive tract, and they involve systemic inflammatory diseases known as extra-intestinal manifestations (EIMs). Timely and correct diagnosis of mucocutaneous EIMs could assist with detecting and monitoring IBD. We present a case of 52-year-old male patient of ulcerative colitis with 2 rare EMIs together at the same time: pyostomatitis vegetans in the oral cavity and Sweet syndrome on the skin. They presented as multiple small white or yellow pustules on the surface of the hyperemic fragile oral mucosa and abrupt appearance of painful, swollen, and erythematous papules on the skin, respectively. The final diagnosis was made based on clinical manifestations, skin and oral tissue biopsies, and the ulcerative colitis history. This rare case report may remind dentists of rare mucocutaneous EIMs of IBD that might be overlooked. Dentists and dermatologists could contribute to the early diagnosis and management of systematic diseases.
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      References

        • Baumgart DC
        • Carding SR.
        Inflammatory bowel disease: cause and immunobiology.
        Lancet. 2007; 369: 1627-1640
        • Ng SC
        • Shi HY
        • Hamidi N
        • et al.
        Worldwide incidence and prevalence of inflammatory bowel disease in the 21st century: a systematic review of population-based studies.
        Lancet. 2017; 390: 2769-2778
        • Mak WY
        • Zhao M
        • Ng SC
        • Burisch J.
        The epidemiology of inflammatory bowel disease: East meets West.
        J Gastroenterol Hepatol. 2020; 35: 380-389
        • Magro F
        • Gionchetti P
        • Eliakim R
        • et al.
        Third European evidence-based consensus on diagnosis and management of ulcerative colitis. Part 1: definitions, diagnosis, extra-intestinal manifestations, pregnancy, cancer surveillance, surgery, and ileo-anal pouch disorders.
        J Crohns Colitis. 2017; 11: 649-670
        • Vavricka SR
        • Schoepfer A
        • Scharl M
        • Lakatos PL
        • Navarini A
        • Rogler G.
        Extraintestinal manifestations of inflammatory bowel disease.
        Inflamm Bowel Dis. 2015; 21: 1982-1992
        • Marotto D
        • Atzeni F
        • Ardizzone S
        • Monteleone G
        • Giorgi V
        • Sarzi-Puttini P.
        Extra-intestinal manifestations of inflammatory bowel diseases.
        Pharmacological Res. 2020; 161105206
        • Antonelli E
        • Bassotti G
        • Tramontana M
        • et al.
        Dermatological manifestations in inflammatory bowel diseases.
        J Clin Med. 2021; 10: 364
        • Vavricka SR
        • Rogler G
        • Gantenbein C
        • et al.
        Chronological order of appearance of extraintestinal manifestations relative to the time of IBD diagnosis in the Swiss Inflammatory Bowel Disease Cohort.
        Inflamm Bowel Dis. 2015; 21: 1794-1800
        • Das KM.
        Relationship of extraintestinal involvements in inflammatory bowel disease: new insights into autoimmune pathogenesis.
        Dig Dis Sci. 1999; 44: 1-13
      1. von den Driesch P. Sweet's syndrome (acute febrile neutrophilic dermatosis).
        J Am Acad Dermatol. 1994; 31: 535-556
        • Nofal A
        • Abdelmaksoud A
        • Amer H
        • et al.
        Sweet's syndrome: diagnostic criteria revisited.
        J Dtsch Dermatol Ges. 2017; 15: 1081-1088
        • Lankarani KB
        • Sivandzadeh GR
        • Hassanpour S.
        Oral manifestations in inflammatory bowel disease: a review.
        World J Gastroenterol. 2013; 19: 8571-8579
        • Hegarty AM
        • Barrett AW
        • Scully C.
        Clin Exp Dermatol. 2004; 29 (Pyostomatitis vegetans.): 1-7
        • Clark LG
        • Tolkachjov SN
        • Bridges AG
        • Camilleri MJ.
        Pyostomatitis vegetans (PSV)-pyodermatitis vegetans (PDV): a clinicopathologic study of 7 cases at a tertiary referral center.
        J Am Acad Dermatol. 2016; 75: 578-584
        • Lebwohl B
        • Sanders DS
        • Green PHR.
        Coeliac disease.
        Lancet. 2018; 391: 70-81
        • Rashid M
        • Zarkadas M
        • Anca A
        • Limeback H.
        Oral manifestations of celiac disease: a clinical guide for dentists.
        J Can Dent Assoc. 2011; 77: b39
        • Alsadat FA
        • Alamoudi NM
        • El-Housseiny AA
        • Felemban OM
        • Dardeer FM
        • Saadah OI.
        Oral and dental manifestations of celiac disease in children: a case-control study.
        BMC Oral Health. 2021; 21: 669
        • Ambrose NL
        • Haskard DO.
        Differential diagnosis and management of Behçet syndrome.
        Nat Rev Rheumatol. 2013; 9: 79-89
        • Murrell DF
        • Peña S
        • Joly P
        • et al.
        Diagnosis and management of pemphigus: recommendations of an international panel of experts.
        J Am Acad Dermatol. 2020; 82: 575-585