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BMJ Case Rep. 2016 Sep 26;2016. doi: 10.1136/bcr-2016-216930.

Lymphomatoid granulomatosis associated with azathioprine use for immune-mediated neuropathy.

BMJ case reports

Nicholas Burwick, Sarah A Buckley, Zhao Ming Dong, Robert E Richard

Affiliations

  1. VA Puget Sound Health Care System, Seattle, Washington, USA Division of Hematology, University of Washington School of Medicine, Seattle, Washington, USA.
  2. Division of Hematology, University of Washington School of Medicine, Seattle, Washington, USA.
  3. VA Puget Sound Health Care System, Seattle, Washington, USA.

PMID: 27671987 PMCID: PMC5051443 DOI: 10.1136/bcr-2016-216930

Abstract

Lymphomatoid granulomatosis (LG) is a rare Epstein-Barr virus-driven lymphoproliferative disorder that generally arises in immunosuppressed patients and which can be life-threatening. Here we describe the development of pulmonary LG in a patient on long-term azathioprine for immune-mediated neuropathy. Although azathioprine carries a boxed warning for malignancy, its association specifically with LG, an otherwise rare entity, is poorly recognised. Early recognition of drug-induced LG is critical, since discontinuation of the offending agent, and implementation of effective therapy can provide rapid clinical benefit in some patients. In this case, rituximab was used as an effective treatment for LG, which also provided an additional benefit of controlling the patient's underlying neuropathy. Further research is needed to identify vulnerable patients who are at high risk of developing drug-induced LG.

2016 BMJ Publishing Group Ltd.

References

  1. Gastroenterology. 2002 Jan;122(1):72-7 - PubMed
  2. Hum Pathol. 1972 Dec;3(4):457-558 - PubMed
  3. Clin Adv Hematol Oncol. 2009 Jan;7(1):65-8 - PubMed
  4. BMC Gastroenterol. 2014 Jul 14;14:127 - PubMed
  5. J Pediatr Gastroenterol Nutr. 2010 May;50(5):559-61 - PubMed
  6. Clin Microbiol Rev. 2010 Apr;23(2):350-66 - PubMed
  7. J Clin Invest. 2003 Apr;111(8):1133-45 - PubMed
  8. Springerplus. 2014 Dec 18;3:751 - PubMed
  9. Cancer J. 2012 Sep-Oct;18(5):469-74 - PubMed
  10. Cancer. 1979 Jan;43(1):360-73 - PubMed

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