Advertisement

Raising the Barr: An Unexpected Lesion at Ileal–Cecal Resection

  • V. Mitsialis
  • Y. Wu
  • B. Gewurz
  • R. Bleday
  • L. A. Doyle
  • R. W. Winter
  • M. J. Hamilton
Multicenter Seminars: IBD (MUSE: IBD)

Case Presentation

Background Data

A 24-year-old man with stricturing, ileocolonic Crohn’s disease with recent partial small bowel obstruction (SBO) due to terminal ileal stenosis, was evaluated 1 day prior to previously scheduled elective ileocecal resection with complaints of fevers, myalgias, and fatigue.

His Crohn’s diagnosis occurred at age 13 (2006) when he was first evaluated with complaints of diarrhea and weight loss for the prior month. The initial diagnostic colonoscopy demonstrated inflammatory changes characterized by superficial and deep ulcerations with luminal narrowing in the terminal ileum and ascending colon. Initiation of infliximab in 2007 led to clinical remission until 2011 when response to the treatments waned. A switch to certolizumab in 2011 recaptured clinical remission until 2017 when he began to experience breakthrough symptoms. He was ultimately switched to adalimumab in 2017, which required titration from every other week to every 10-day dosing in order to...

Notes

Compliance with ethical standards

Conflict of interest

The authors do not have any relevant conflicts of interest with respect to this case presentation.

References

  1. 1.
    Lopes S, Andrade P, Conde S, et al. Looking into enteric virome in patients with IBD: defining guilty or innocence? Inflamm Bowel Dis. 2017;23:1278–1284.CrossRefPubMedPubMedCentralGoogle Scholar
  2. 2.
    Ryan JL, Shen YJ, Morgan DR, et al. Epstein–Barr virus infection is common in inflamed gastrointestinal mucosa. Dig Dis Sci. 2012;57:1887–1898.CrossRefPubMedPubMedCentralGoogle Scholar
  3. 3.
    Shimada T, Nagata N, Okahara K, et al. PCR detection of human herpesviruses in colonic mucosa of individuals with inflammatory bowel disease: comparison with individuals with immunocompetency and HIV infection. PLoS ONE. 2017;12:e0184699.CrossRefPubMedPubMedCentralGoogle Scholar
  4. 4.
    Siegel CA, Marden SM, Persing SM, et al. Risk of lymphoma associated with combination anti-tumor necrosis factor and immunomodulator therapy for the treatment of Crohn’s disease: a meta-analysis. Clin Gastroenterol Hepatol. 2009;7:874–881.CrossRefPubMedPubMedCentralGoogle Scholar
  5. 5.
    Beaugerie L, Brousse N, Bouvier AM, et al. Lymphoproliferative disorders in patients receiving thiopurines for inflammatory bowel disease: a prospective observational cohort study. Lancet. 2009;374:1617–1625.CrossRefPubMedGoogle Scholar
  6. 6.
    Virdis F, Tacci S, Messina F, et al. Hemophagocytic lymphohistiocytosis caused by primary Epstein–Barr virus in patient with Crohn’s disease. World J Gastrointest Surg. 2013;5:306–308.CrossRefPubMedPubMedCentralGoogle Scholar
  7. 7.
    Biank VF, Sheth MK, Talano J, et al. Association of Crohn’s disease, thiopurines, and primary Epstein–Barr virus infection with hemophagocytic lymphohistiocytosis. J Pediatr. 2011;159:808–812.CrossRefPubMedPubMedCentralGoogle Scholar
  8. 8.
    Connors W, Griffiths C, Patel J, et al. Lymphomatoid granulomatosis associated with azathioprine therapy in Crohn disease. BMC Gastroenterol. 2014;14:127.CrossRefPubMedPubMedCentralGoogle Scholar
  9. 9.
    Juan A, Lobatón T, Tapia G, et al. Epstein–Barr virus-positive mucocutaneous ulcer in Crohn’s disease. A condition to consider in immunosuppressed IBD patients. Dig Liver Dis. 2017;49:934–937.CrossRefPubMedGoogle Scholar
  10. 10.
    Dojcinov SD, Venkataraman G, Raffeld M, et al. EBV positive mucocutaneous ulcer—a study of 26 cases associated with various sources of immunosuppression. Am J Surg Pathol. 2010;34:405–417.CrossRefPubMedGoogle Scholar
  11. 11.
    Roberts TK, Chen X, Liao JJ. Diagnostic and therapeutic challenges of EBV-positive mucocutaneous ulcer: a case report and systematic review of the literature. Exp Hematol Oncol. 2016;5:13.CrossRefPubMedPubMedCentralGoogle Scholar
  12. 12.
    Matnani R, Peker D. Azathioprine induced Epstein Barr virus-positive mucocutaneous ulcer arising in perianal fistula and abscess associated with Crohn’s disease. J Crohns Colitis. 2014;8:1747–1748.CrossRefPubMedGoogle Scholar
  13. 13.
    Moran NR, Webster B, Lee KM, et al. Epstein Barr virus-positive mucocutaneous ulcer of the colon associated Hodgkin lymphoma in Crohn’s disease. World J Gastroenterol. 2015;21:6072–6076.CrossRefPubMedPubMedCentralGoogle Scholar
  14. 14.
    Hart M, Thakral B, Yohe S, et al. EBV-positive mucocutaneous ulcer in organ transplant recipients: a localized indolent posttransplant lymphoproliferative disorder. Am J Surg Pathol. 2014;38:1522–1529.CrossRefPubMedGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Authors and Affiliations

  • V. Mitsialis
    • 1
  • Y. Wu
    • 1
  • B. Gewurz
    • 1
  • R. Bleday
    • 1
  • L. A. Doyle
    • 1
  • R. W. Winter
    • 1
  • M. J. Hamilton
    • 1
  1. 1.Crohn’s and Colitis CenterBrigham and Women’s HospitalBostonUSA

Personalised recommendations