Abstract
BackgroundLymphomatoid 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 presentationA 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.ConclusionsThiopurine 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.
Highlights
Lymphomatoid granulomatosis (LYG) is a rare Epstein-Barr virus-associated lymphoproliferative disorder
Thiopurine therapy for inflammatory bowel disease is associated with an increased incidence of lymphoproliferative disorders
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
Summary
We present the first case in the literature of lymphomatoid granulomatosis in the setting of azathioprine therapy for Crohn disease. The atypical features in our case underscore the importance of maintaining a high degree of clinical suspicion and diagnostic vigilance when evaluating non-specific abdominal symptoms frequently encountered in CD patients. This case highlights that even limited exposure to such therapy may induce LYG. Consent Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. 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
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