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Post-transplantation lymphoproliferative disorder (PTLD) is a life-threatening complication following lung transplantation. We report a PTLD case of high-grade, B-cell lymphoma following contralateral single-lung transplantation. The disease involved the liver, right kidney and right native lung. While the PTLD affecting the abdominal organs regressed with rituximab chemotherapy, the native lung disease progressed and was treated surgically (right pneumonectomy). Some aspects are unique in this case: (i) different response to medical treatment between lung and abdominal organs; (ii) absolute absence of involvement of the native lung and (iii) surgical treatment with a pneumonectomy, still very rarely described in the literature. We hypothesized that a different morphotype of the disease involved the abdominal organs or the penetrance of rituximab, and chemotherapy could have been impaired by the presence of pulmonary fibrosis., (© The Author 2016. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved.)