We present a middle-aged woman complaining of weakness, lethargy and weight loss for 6 months. Positron emission tomography (PET)/CT scan revealed huge bilateral adrenal masses with intense 18F-fluorodeoxyglucose avidity. Biopsy and immunohistochemical staining were compatible with mantle cell lymphoma (MCL). The patient received six cycles of rituximab, cyclophosphamide, doxorubicin, vincristine and prednisone (R-CHOP) followed by four cycles of intrathechal methotrexate chemotherapy. Bone marrow aspiration biopsy was performed for initial staging and also before bone marrow transplantation. Follow-up PET/CT scan after completion of chemotherapy and before bone marrow transplantation demonstrated complete metabolic response with no evidence of abnormal metabolic activity in either adrenal gland or bone marrow. MCL has an aggressive nature and is usually considered incurable; however, there have been a few reports indicating favourable treatment response when MCL is rarely arising from the adrenal glands. Dramatic response of a primary adrenal MCL to R-CHOP is documented in this case.
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