Inflammatory pseudotumor following cord blood transplantation for adult T-cell leukemia

Adult T-cell leukemia (ATL) is a human malignancy associated with human T-lymphotropic retrovirus type-1 and diagnosed based on clinicopathological findings and the presence of integrated human T-lymphotropic retrovirus type-1 provirus in the DNA of tumor cells.1 Despite recent progress in combination chemotherapy regimens, acute-type ATL usually has a poor prognosis.2 Allogeneic SCT is expected to be another therapeutic option for patients with ATL.3, 4 Recently, cord blood transplantation (CBT) has been used for adult patients with hematological malignancies as donor coordination is not necessary, and the incidence and severity of acute GVHD are lower than those for BMT.5 Inflammatory pseudotumor (IPT) is a rare tumor that can occur in various organs and tissues.6 Although its pathogenesis remains unclear, the terms inflammatory myofibroblastic tumor or inflammatory myofibrohistiocytic proliferation have been proposed as being more descriptive of IPT arising in the lung.7 IPT of different organs associated with hematopoietic SCT (HSCT), although rare, has also been reported.8 We describe a patient who developed a lung IPT after CBT. To the best of our knowledge, this report of lung IPT after CBT is also the first report of any IPT after CBT.

The patient was a 36-year-old Japanese woman with chemorefractory ATL. There were no HLA-identical sibling donors, and therefore unrelated umbilical cord blood was arranged. The patient and the cord blood were confirmed as fully matched at six HLA loci (HLA-A 24/1101, -B 5201/1501, DRB1-1502/0406). CY was administered at 3000 mg/day from days −6 to −5, whereas 4 Gy/day of TBI was given in two fractions from days −3 to −1. For GVHD prophylaxis, the patient received CyA at 150 mg/day as a continuous infusion from day −1, methotrexate 15 mg i.v. on day 1 and 10 mg on days 3 and 6, and 45 mg/day prednisolone starting on day 11. In January 2005, she received 2.70 × 107 cryopreserved maternal cord blood stem cells, at a dose of 0.40 × 105 CD34+ cells per kg body weight. Hematological engraftment was confirmed 28 days after CBT. Complete chimerism was confirmed by the FISH technique using sex chromosomes. On day 30, grade II skin and gastrointestinal acute GVHD occurred transiently. However, the patient developed no clinically significant chronic GVHD or infections. The human T-lymphotropic retrovirus type-1 proviral load in PBMCs from the patient fell to carrier levels in peripheral blood samples. Approximately 18 months post transplant, the patient developed a cough, slight fever and malaise. Slight anemia and thrombocytosis appeared. Computer tomography of the chest revealed a solid mass (Figure 1). Histologic specimens were obtained by percutaneous needle biopsy using an ultrasound echo-guiding technique. On pathological evaluation, variable concentrations of lymphocytes, plasma cells and histiocytes were found, but with minimal fibrous connective tissue. The pathological findings appeared to be compatible with IPT, although atypical.9 Fungal pneumonia was excluded. Anti-inflammatory medication improved the clinical symptoms, and the mass lesion exhibited no obvious change, while the ATL remained stable.

Figure 1
figure1

An axial unenhanced CT scan demonstrates a well-circumscribed mass in the right lower lobe of the lung. No mediastinal lymphadenopathy is noted. Histological analysis confirmed the diagnosis of an inflammatory pseudotumor. CT, computer tomography.

The causes of IPT are unknown; however, some authors believe that this tumor is a low-grade fibrosarcoma with inflammatory cells.6 IPT most commonly involves the lung and the orbit, but has been reported to occur in most sites in the body.6 The case described occurred in the lung. Although the relationship between IPT and HSCT is unclear, many factors are implicated. Infection, chemotherapy and radiotherapy have been conjectured as factors for IPT development.7, 10 All patients with IPT after HSCT have undergone high-dose chemotherapy and some also received TBI. For IPTs developing after HSCT, immunosuppression and GVHD are the two factors most likely to be associated.11, 12 In particular, in the first report of a lung IPT after HSCT, the patient had a lung infection prior to the development of IPT.12 However, our patients had no significant lung infection prior to the development of IPT. In addition, cord blood has been demonstrated to induce less GVHD in the setting of HLA-mismatched grafting. Indeed, our case did not suffer severe GVHD.

The degree to which the various factors may contribute to the development of IPT is unclear. It is known that IPT can develop as a result of a ‘reactive process’ generated in response to noninfectious agents such as trauma, surgery, foreign body retention and smoking. However, our patient experienced none of these events. The patient was treated with chemotherapy, TBI, had a history of a specific virus-associated malignancy, not ATL, and had a transplant with some signs of GVHD. Therefore, each or all of these could be considered as contributing factors. Our experience may suggest that IPT is a rare but containable potential complication of CBT for ATL.

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Nomura, S., Ishii, K., Shimizu, M. et al. Inflammatory pseudotumor following cord blood transplantation for adult T-cell leukemia. Bone Marrow Transplant 42, 493–494 (2008). https://doi.org/10.1038/bmt.2008.191

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