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Journal of Peking University (Health Sciences) ›› 2020, Vol. 52 ›› Issue (2): 373-377. doi: 10.19723/j.issn.1671-167X.2020.02.028

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Kaposi sarcoma combined with severe ulcerative colitis: A case report and literature review

Jun LI1,△(),Zhan-yue NIU1,Yan XUE1,Xue-ying SHI2,Bo ZHANG2,Yuan WANG3   

  1. 1. Department of Gastroenterology
    2. Department of Pathology
    3. Department of Dermatology, Peking University Third Hospital, Beijing 100191, China
  • Received:2020-02-24 Online:2020-04-18 Published:2020-04-18
  • Contact: Jun LI E-mail:yanwt2003@163.com

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Abstract:

SUMMARY　Kaposi’s sarcoma (KS) is an unusual vascular tumor associated with human herpesvirus-8 (HHV-8) infection, which is common in immunosuppressors. Although extremely rare, iatrogenic (drug-related) KS can occur in human immunodeficiency virus (HIV)-negative patients under immunosuppressive therapy. We report a 64-year-old male diagnosed with ulcerative colitis for 1 year. He was treated with methylprednisolone because of an acute severe disease flare. He presented with several popular violet lesions on the body 4 months after steroid therapy. Histological examination of skin biopsies showed Kaposi’s sarcoma associated with HHV-8. The skin lesions regressed after steroid withdrawal and chemotherapy. Two key words “Kaposi’s sarcoma” and “inflammatory bowel disease” were searched in Wanfang data and CNKI, but no relevant articles were found. Thirty-eight articles in English were retrieved on PubMed with the key words of (“ulcerative colitis” OR “Crohn’s disease” OR “inflammatory bowel disease”) AND (Kaposi sarcoma). Twenty-five cases of Kaposi’s sarcoma related to inflammatory bowel disease (IBD) were reported. Including this case, the majority of 26 Kaposi’s sarcoma related IBD patients were male (80.8%, 21/26). The average age was (51.1 ± 16.4) years. Twenty cases were ulcerative colitis and 6 were Crohn’s disease. All the patients received immunomodulatory therapy, including glucocorticoid, azathioprine/mercaptopurine, methotrexate, cyclosporin and anti tumor necrosis factor α antibody. Thirteen cases were positive for HHV-8. There were 18 cases involving the distal ileum and colorectum only, 3 cases involving skin only, and 5 cases involving both skin and colorectum at the same time. Overall, the prognosis was good. Three patients only stopped immunosuppressive therapy, 1 received radiotherapy, 1 received chemotherapy, and 20 received surgery. Kaposi’s sarcoma could be seen in IBD patients with immunomodulatory therapy. It is very important to distinguish from the skin lesions related to IBD or drug treatment. The adverse reactions of immunomodulatory therapy should not be ignored. In addition, attention should be paid to the cooperation of multi-disciplinary team, which can diagnose and treat rare cases earlier and more accurately.

Key words: Kaposi's sarcoma, Inflammatory bowel disease, Human herpes virus-8, Immunomodulatory therapy

CLC Number: 

  • R574.1

Figure 1

Skin lesions of left lower extremityA, several popular violet lesions on the left lower extremtity after 4 months of steroid therapy; B,skin lesions became more elevated 2 weeks later; C, skin lesions regressed after 5 months of steroid withdrawal and chemotherapy."

Figure 2

Histological examination of skin biopsies showed Kaposi's sarcoma, immunohistochemistry for the detection of HHV-8 proved positiveA, ×20; B, ×100; C, ×100."

[1] Dal Maso L, Polesel J, Ascoli V, et al.Classic Kaposi's sarcoma in Italy, 1985-1998[J]. Br J Cancer, 2005, 92(1): 188-193.
[2] Di Lorenzo G.Update on classic Kaposi sarcoma therapy: new look at an old disease[J]. Crit Rev OncolHematol, 2008, 68(3): 242-249
[3] Kumar V, Soni P, Garg M, et al.Kaposi sarcoma mimicking acute flare of ulcerative colitis[J]. J Investig Med High Impact Case Rep, 2017, 5: 2324709617713510.
[4] Goedert JJ, Vitale F, Lauria C, et al.Risk factors for classical Kaposi's sarcoma[J]. J Natl Cancer Inst, 2002, 94: 1712-1718.
[5] Anderson LA, Lauria C, Romano N, et al.Risk factors for classical Kaposi sarcoma in a population-based case-control study in Sicily[J]. Cancer Epidemiol Biomarkers Prev, 2008, 17(12): 3435-3443.
[6] Semango GP, Charles RM, Swai CI, et al.Prevalence and associated risk factors for Kaposi's sarcoma among HIV-positive patients in a referral hospital in Northern Tanzania: a retrospective hospital-based study[J]. BMC Cancer, 2018, 18(1): 1258.
[7] Stolka K, Ndom P, Hemingway-Foday J, et al.Risk factors for Kaposi's sarcoma among HIV-positive individuals in a case control study in Cameroon[J]. Cancer Epidemiol, 2014, 38(2): 137-143.
[8] Neff R, Kremer S, Voutsinas L, et al.Primary Kaposi's sarcoma of the ileum presenting as massive rectal bleeding[J]. Am J Gastroenterol, 1987, 82(3): 276-277.
[9] Penn I.Kaposi's sarcoma in transplant recipients[J]. Transplantation, 1997, 64(5): 669-673.
[10] Gordon HW, Rywlin AM.Kaposi's sarcomaof the large intestine associated with ulcerativecolitis. A hitherto unreported occurrence[J].Gastroenterology, 1966, 50: 248-253.
[11] Adlersberg R.Kaposi's sarcoma complicating ulcerative colitis: Report of a case[J]. Am J ClinPathol, 1970, 54(1): 143-146.
[12] Meltzer SJ, Rotterdam HZ, Korelitz BI.Kaposi's sarcoma occurring inassociation with ulcerative colitis[J].Am J Gastroenterol, 1987, 82(4): 378-381.
[13] Koop HO, Holodniy M, List AF.Fulminant Kaposi's sarcoma complicatinglong-term corticosteroid therapy[J]. Am J Med, 1987, 83(4): 787-789.
[14] Thompson GB, Pemberton JH, Morris S, et al.Kaposi's sarcoma of the colon in a young HIV-negative man with chroniculcerative colitis. Report of a case[J]. Dis Colon Rectum, 1989, 32(1): 73-76.
[15] Puy-Montbrun T, Pigot F, Vuong PN, et al.Kaposi's sarcoma of the colon in a young HIV-negative woman with Crohn's disease[J]. Dig Dis Sci, 1991, 36(4): 528-531.
[16] Tedesco M, Benevolo M, Frezza F, et al.Colorectal Kaposi's sarcoma in an HIV-negative male in association withulcerative rectocolitis: a case report[J]. Anticancer Res, 1999, 19(4B): 3045-3048.
[17] Cohen RL, Tepper RE, Urmacher C, et al.Kaposi's sarcoma and cytomegaloviralileocolitis complicating long-standing Crohn's disease in an HIV-negativepatient[J]. Am J Gastroenterol, 2001, 96(10): 3028-3031.
[18] Pedulla F, Sisteron O, Chevallier P, et al.Kaposi's sarcoma confined to the colorectum: A case report[J]. Clin Imaging, 2004, 28: 33-35.
[19] Kang MJ, Namgung KY, Kim MS, et al.A case of Kaposi's sarcoma associated with ulcerative colitis[J]. Korean J Gastroenterol, 2004, 43(5): 316-319.
[20] Bursics A, Morvay K, Abrah’m K, et al.HHV-8 positive, HIV negative disseminated Kaposi's sarcoma complicating steroid dependent ulcerative colitis: A successfully treated case[J]. Gut, 2005, 54(7): 1049-1050.
[21] Girelli CM, Serio G, Rocca E, et al.Refractory ulcerative colitis andiatrogenic colorectal Kaposi's sarcoma[J]. Dig Liver Dis, 2009, 41(2): 170-174.
[22] Svrcek M, Tiret E, Bennis M, et al.KSHV/HHV8-associated intestinal Kaposi's sarcoma in patient with ulcerative colitis recei-ving immunosuppressive drugs: Report of a case[J]. Dis Colon Rectum, 2009, 52(1): 154-158.
[23] Rodríguez-Pel’ez M, Fern’ndez-García MS, Gutiérrez-Corral N, et al.Kaposi's sarcoma: Anopportunistic infection by human herpesvirus-8 in ulcerative colitis[J]. J Crohns Colitis, 2010, 4(5): 586-590.
[24] Cetın B, Büyükberber S, Yilmaz IB, et al.Kaposi's sarcoma in patients with ulcerative colitis receiving immunosuppressive drugs:report of a case[J]. Turk J Gastroenterol, 2011, 22(6): 621-625.
[25] Pioche M, Boschetti G, Cotte E, et al.Human herpesvirus 8-associated colorectal Kaposi's sarcomaoccurring in a drug-induced immunocompromised patient with refractory ulcerative colitis: report of a new case and review of the literature[J]. Inflamm Bowel Dis, 2013, 19(2): E12-E15.
[26] Hamzaoui L, Kilani H, Bouassida M, et al.Iatrogenic colorectal Kaposi sarcoma complicating a refractory ulcerative colitis in a human immunodeficiency negative-viruspatient[J]. Pan Afr Med J, 2013, 15: 154.
[27] Kilincalp S, Akıncı H, Hamamci M, et al.Kaposi's sarcoma developing in a HIV-negative Crohn's disease patient shortly after azathioprineand corticosteroid treatment[J]. J Crohns Colitis, 2014, 8(6): 558-559.
[28] Herculano R, Barreiro P, Hann A, et al.Drug-induced colonic Kaposi's sarcoma in a HIV-negativepatient with ulcerative colitis: a case report and review of the literature[J]. Int J Colorectal Dis, 2014, 29(11): 1441-1442.
[29] Windon AL, Shroff SG.Iatrogenic Kaposi's sarcoma in an HIV-negative youngmale with Crohn's disease and IgA nephropathy: A case report and brief review of the literature[J]. Int J Surg Pathol, 2018, 26(3): 276-282.
[30] Shah N, Lidofsky S, Laskiewicz L.Colorectal Kaposi sarcoma in an immunosuppressed ulcerative colitis patient[J]. J Gastrointest Surg, 2018, 22(7): 1301-1302.
[31] Duh E, Fine S.Human herpesvirus-8 positive iatrogenic Kaposi's sarcoma in the setting of refractory ulcerative colitis[J]. World J Clin Cases, 2017, 5(12): 423-427.
[32] Chtourou L, Ayedi L, Rejab H, et al.Iatrogenic colorectal Kaposi's sarcoma complicating arefractory ulcerative colitis in a human immunodeficiency negative-virus patient[J]. Pathologica, 2017, 109(4): 371-374.
[33] Stasi E, De Santis S, Cavalcanti E, et al.Iatrogenic Kaposi sarcoma of the terminal ileum following short-term treatment with immunomodulators for Crohndisease: A case report[J]. Medicine (Baltimore), 2019, 98(20): e15714.
[34] Billon E, Stoppa AM, Mescam L, et al.Reversible rituximab-induced rectal Kaposi's sarcoma misdiagnosed as ulcerativecolitis in a patient with HIV-negative follicular lymphoma[J]. Clin Sarcoma Res, 2018, 8: 11.
[35] Weber JN, Carmichael DJ, Boylston A, et al.Kaposi's sarcoma of the bowel: presenting as apparent ulcerative colitis[J]. Gut, 1985, 26(3): 295-300.
[36] Biggs BA, Crowe SM, Lucas CR, et al.AIDS related Kaposi's sarcoma presenting as ulcerative colitis and complicated by toxic megacolon[J]. Gut, 1987, 28(10): 1302-1306.
[37] Greuter T, Navarini A, Vavricka SR.Skin manifestations of inflammatory bowel disease[J]. Clinic Rev AllergImmunol, 2017, 53: 413-427.
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