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01.12.2014 | Case report | Ausgabe 1/2014 Open Access

BMC Gastroenterology 1/2014

Lymphomatoid granulomatosis associated with azathioprine therapy in Crohn disease

Zeitschrift:
BMC Gastroenterology > Ausgabe 1/2014
Autoren:
William Connors, Cameron Griffiths, Jay Patel, Paul J Belletrutti
Wichtige Hinweise

Electronic supplementary material

The online version of this article (doi:10.​1186/​1471-230X-14-127) contains supplementary material, which is available to authorized users.
William Connors, Cameron Griffiths, Jay Patel contributed equally to this work.

Competing interests

The authors declare that they have no competing interests.

Authors’ contributions

WC and CG wrote the manuscript and performed the literature review. JP and PJB conceived of the report, participated in expert review and editing of the final manuscript. All authors read and approved the final manuscript.

Abstract

Background

Lymphomatoid granulomatosis (LYG) is a rare Epstein-Barr virus-associated lymphoproliferative disorder. It most often occurs in patients with immunodeficiency and the clinical course ranges from indolent behavior to that of an aggressive malignancy. Pulmonary, central nervous system and dermatological manifestations are most common. To our knowledge this is the first reported case of LYG related to azathioprine therapy in Crohn disease.

Case presentation

A twenty-six year old Caucasian woman with colonic Crohn disease on maintenance azathioprine therapy presented with right upper quadrant pain and fever. Diagnostic imaging revealed extensive liver, pulmonary and cerebral lesions. A diagnosis of LYG was made based on the pattern of organ involvement and the immunohistochemical features on liver and lung biopsy.

Conclusions

Thiopurine therapy for inflammatory bowel disease is associated with an increased incidence of lymphoproliferative disorders. This report highlights the diagnostic challenges associated with LYG. As long-term thiopurine therapy remains central to the management of inflammatory bowel diseases it is essential that both patients and clinicians are aware of this potential adverse outcome.
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