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BackgroundThe risk of gastrointestinal stromal tumor (GIST) in combination with other primary malignancies is high, which occurs before and after the diagnosis of GIST. Primary pulmonary T-cell lymphoma is a rare type of non-Hodgkin lymphoma.Case presentationWe report a 53-year-old male patient who was admitted to our hospital with fever, cough, and expectoration for 2 weeks. Chest computed tomography (CT) showed a cavitary mass in the left lower lobe with multiple nodules in the upper lobes of both lungs. The patient had a history of surgery for small intestinal stromal tumors and was treated with oral imatinib after surgery. Lung biopsy was diagnosed as lymphomatoid granulomatosis, tending to grade 3. The pathological diagnosis was corrected by surgery and genetic testing for lung non-Hodgkin CD8-positive cytotoxic T-cell lymphoma with Epstein–Barr virus (EBV) infection in some cells. After multiple chemotherapies, the CT scan showed a better improvement than before. The patient is still under follow-up, and no tumor recurrence has been found.ConclusionPatients with a history of GIST should be monitored for other malignancies. The clinical symptoms and imaging examinations of primary pulmonary T-cell lymphoma are not characteristic, and the definite diagnosis still depends on pathological examination. The patient was treated with the CHOP chemotherapy regimen after the operation, the curative effect was good.