Complete remission of ALK-negative plasma cell granuloma (inflammatory myofibroblastic tumor) of the lung induced by celecoxib: A case report and review of the literature

Abstract

We report a case in which a 52-year-old female developed a multifocal inflammatory myofibroblastic tumor (IMT) of the lung. The tumor did not overexpress the anaplastic lymphoma kinase (ALK) protein, indicating a lack of ALK rearrangement. The patient required two wedge resections in 15 months due to recurrent disease. Recurrence after the second surgery was treated with corticosteroids, which only led to a transient response (6 months). Introduction of celecoxib, a cyclooxygenase-2 inhibitor, induced a complete remission in the patient. Maintenance on celecoxib further led to a progression-free survival of 34 months. A literature review retrieved a total of eight case reports, comprising ten patients, of IMT of various anatomical sites successfully treated with non-steroidal anti-inflammatory agent (NSAID) therapy. Nine of the ten patients achieved durable complete remission. Remission occurred rapidly and persisted even after termination of NSAID therapy. Although such a successful outcome may only be achieved rarely, a trial of an NSAID should be considered in any patient in whom complete resection is not an option. Our case also demonstrates that NSAID therapy may be successful in a non-ALK rearranged tumor in which ALK inhibition is not an option.

Keywords: cyclooxygenase-2 inhibitors; inflammatory myofibroblastic tumor; non-steroidal anti-inflammatory agents.

Figure 1

Hematoxylin and eosin. Lung wedge resection revealing the typical histology of an IMT. The image depicts an admixture of mesenchymal cells and plasma cells. IMT, inflammatory myofibroblastic tumor.

Figure 2

Left lower lobe nodule prior to celecoxib therapy.

Figure 3

Complete resolution of the left lower lobe nodule following celecoxib therapy.