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This report is dealing with simultaneous diagnosis of Myelodysplastic Syndrome with Excess of Blasts 2 (MDS‐EB2) with isolated 5q deletion and Smoldering Multiple Myeloma (SMM) with typical changes, namely t(4;14) and monosomy 13. I‐FISH and FICTION techniques showed distinct specific clones underlie the two hematological malignancies.

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Introduction: Posttransplant lymphoproliferative disorders (PTLDs) refer to a group of diseases, including diffuse large B-cell lymphoma (DLBCL), that develop after solid organ transplantation or hematopoietic stem cell transplantation. Extranodal involvement in PTLDs is common. Reports about exclusive bone marrow involvement are rare. Case description: A 70-year-old woman, who had undergone kidney transplantation in 2018, was diagnosed with exclusively extranodal, Epstein-Barr virus–negative DLBCL, with bone marrow and spleen involvement, during long-term immunosuppression. She achieved complete remission with combined immunochemotherapy and temporary hold of immunosuppression. Conclusions: This case shows an uncommon clinical presentation of DLBCL, which was challenging to diagnose, being entirely extranodal. The favorable clinical course relied on timely diagnosis and a multidisciplinary approach. Long-term consequences of posttransplant immunosuppression require a high level of suspicion for an appropriate management, aimed at preserving the graft while eradicating the lymphoproliferative disorder.