Primary CNS Immunomodulatory Therapy-Induced Lymphoproliferative Disorder in a Patient with Ulcerative Colitis: a Case Report and Review of the Literature.
World Neurosurg. 2015 Jul 11;
Authors: Alobaid A, Torlakovic E, Kongkham P
Abstract
BACKGROUND: Immunosuppression and immunomodulatory therapy-induced lymphoproliferative disorders (ILPD) represent a heterogeneous group of lymphoid cell disorders that occur secondary to iatrogenic immune dysfunction, best described in the post-transplant setting.
CASE DESCRIPTION: We describe, to the best of our knowledge, the first reported case of a primary central nervous system (CNS) ILPD in a patient with ulcerative colitis (UC) treated chronically with the immunomodulatory agents infliximab and azathioprine. This 52-year old female presented with a one-month history of left sided weakness and paresthesias. Neuroimaging identified multiple heterogeneously-enhancing lesions in her cerebrum. Extensive systemic infectious and malignancy-related investigations were negative, prompting neurosurgical referral to obtain a tissue diagnosis. Pathologic assessment of her open excisional biopsy specimen confirmed the diagnosis of a polymorphic lymphoproliferative disorder. She was treated by withdrawal of infliximab and azathioprine, along with a prolonged course of prednisone. At early 6-month follow up she demonstrated both clinical and radiologic improvement.
CONCLUSION: ILPD should be considered in the differential diagnosis in patients with iatrogenic immunodeficiency presenting with neurological symptoms and intra-axial mass lesions on neuroimaging investigations. A standard treatment regimen for ILPD remains to be determined, however withholding the immunomodulatory agents and trial of corticosteroids may be tried as one first-line option prior to the use of more aggressive chemotherapy and/or radiotherapy.
PMID: 26171889 [PubMed – as supplied by publisher]
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