Methotrexate has been reported as an immunosuppressive agent associated with lymphoproliferative disorders. This report describes the case of a cardiac methotrexate-induced lymphoproliferative disorder that could be differentiated from a sinus of Valsalva aneurysm rupture by cardiac magnetic resonance imaging and fluorine-18 (F)-fluorodeoxyglucose positron emission tomography combined with computed tomography. The definitive diagnosis was made by a tissue biopsy that was concomitantly performed with sinus of Valsalva aneurysm repair. Significant regression was seen in response to methotrexate withdrawal. To the best of our knowledge, this is the first case of a cardiac methotrexate-induced lymphoproliferative disorder.
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