title: Epstein Barr virus-positive mucocutaneous ulcer of the colon associated Hodgkin lymphoma in Crohn's disease.
abstract: Epstein Barr virus (EBV) positive mucocutaneous ulcers (EBVMCU) form part of a spectrum of EBV-associated lymphoproliferative disease. They have been reported in the setting of immunosenescence and iatrogenic immunosuppression, affecting the oropharyngeal mucosa, skin and gastrointestinal tract (GIT). Case reports and series to date suggest a benign natural history responding to conservative management, particularly in the GIT. We report an unusual case of EBVMCU in the colon, arising in the setting of immunosuppression in the treatment of Crohn's disease, with progression to Hodgkin lymphoma 18 mo after cessation of infliximab. The patient presented with multiple areas of segmental colonic ulceration, histologically showing a polymorphous infiltrate with EBV positive Reed-Sternberg-like cells. A diagnosis of EBVMCU was made. The ulcers failed to regress upon cessation of infliximab and methotrexate for 18 mo. Following commencement of prednisolone for her Crohn's disease, the patient developed widespread Hodgkin lymphoma which ultimately presented as a life-threatening lower GIT bleed requiring emergency colectomy. This is the first report of progression of EBVMCU to Hodgkin lymphoma, in the setting of ongoing iatrogenic immunosuppression and inflammatory bowel disease.
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: Neil R
authors.initials: NR
authors.lastname: Moran
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: Bradley
authors.initials: B
authors.lastname: Webster
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: Kenneth M
authors.initials: KM
authors.lastname: Lee
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: Judith
authors.initials: J
authors.lastname: Trotman
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: Yiu-Lam
authors.initials: YL
authors.lastname: Kwan
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: John
authors.initials: J
authors.lastname: Napoli
authors.affiliation: Neil R Moran, Rupert gW Leong, Gastroenterology and Liver Services, Concord Repatriation General Hospital, Sydney, NSW 2137, Australia.
authors.forename: Rupert W
authors.initials: RW
authors.lastname: Leong
mesh_terms: D000971:Antineoplastic Combined Chemotherapy Protocols / Q000627:therapeutic use; D001706:Biopsy; D003082:Colectomy; D003110:Colonic Neoplasms / Q000175:diagnosis / Q000188:drug therapy / Q000276:immunology / Q000821:virology*; D003113:Colonoscopy; D003424:Crohn Disease / Q000175:diagnosis / Q000188:drug therapy* / Q000276:immunology; D018450:Disease Progression; D020031:Epstein-Barr Virus Infections / Q000175:diagnosis / Q000276:immunology / Q000821:virology*; D005260:Female; D006471:Gastrointestinal Hemorrhage / Q000209:etiology / Q000601:surgery; D004854:Herpesvirus 4, Human / Q000235:genetics / Q000302:isolation & purification*; D006689:Hodgkin Disease / Q000175:diagnosis / Q000188:drug therapy / Q000276:immunology / Q000821:virology*; D006801:Humans; D007081:Ileostomy; D016867:Immunocompromised Host; D007166:Immunosuppressive Agents / Q000009:adverse effects*; D017403:In Situ Hybridization; D008875:Middle Aged; D013997:Time Factors; D016896:Treatment Outcome; D014456:Ulcer / Q000175:diagnosis / Q000276:immunology / Q000821:virology*
chemical_list: D007166:Immunosuppressive Agents
keywords: Crohn’s disease; Epstein Barr virus; Hodgkin lymphoma; Inflammatory bowel disease; Mucocutaneous ulcer