Division of Hematology and Oncology, Department of Medicine, Kurume University School of Medicine, 67 Asahi-machi, Kurume, 830-0011, Japan.
Department of Dermatology, Kurume University School of Medicine, 67 Asahi-machi, Kurume, 830-0011, Japan.
Int J Hematol. 2019 Oct;110(4):506-511. doi: 10.1007/s12185-019-02675-1. Epub 2019 May 31.
Allogeneic hematopoietic cell transplantation (allo-HSCT) is considered the curative treatment option in patients with aggressive adult T cell leukemia/lymphoma (ATLL), but the treatment of relapse after allo-HSCT remains a major challenge. We report a case of ATLL that was treated with sequential mogamulizumab (MOG) and lenalidomide (LEN) for early relapse after allo-HSCT. A 73-year-old Japanese male with acute-type ATLL underwent haploidentical-HSCT with post-transplant cyclophosphamide. He attained a complete response. However, ATLL relapse was diagnosed by biopsy of skin lesions that appeared on day 67. Discontinuation of immunosuppressant therapy alone did not result in improvement of ATLL, however, the skin lesions disappeared after an immune response was induced by sequential MOG and LEN. Following MOG and LEN, very serious toxic epidermal necrolysis (TEN) developed requiring high-dose intravenous immunoglobulin and methylprednisolone pulse therapy. Although graft-versus-host disease exacerbated and progressed to TEN, a complete response was achieved after successful treatment of TEN. These agents may thus enhance anti-ATLL activity by immune modulation. Further investigation is necessary to determine the optimal use of MOG and LEN in relapsed ATLL after allo-HSCT.
异基因造血细胞移植（allo-HSCT）被认为是侵袭性成人 T 细胞白血病/淋巴瘤（ATLL）患者的根治性治疗选择，但 allo-HSCT 后复发的治疗仍然是一个主要挑战。我们报告了一例 allo-HSCT 后早期复发的 ATLL 患者，采用莫格利珠单抗（MOG）和来那度胺（LEN）序贯治疗。一名 73 岁的日本男性患有急性型 ATLL，接受了haploidentical-HSCT 联合移植后环磷酰胺治疗。他达到了完全缓解。然而，在第 67 天出现皮肤病变的活检诊断为 ATLL 复发。单独停止免疫抑制剂治疗并未改善 ATLL，但序贯 MOG 和 LEN 诱导免疫反应后皮肤病变消失。在使用 MOG 和 LEN 后，出现非常严重的中毒性表皮坏死松解症（TEN），需要大剂量静脉注射免疫球蛋白和甲基强的松龙脉冲治疗。尽管移植物抗宿主病加重并进展为 TEN，但在成功治疗 TEN 后达到完全缓解。这些药物可能通过免疫调节增强抗 ATLL 活性。需要进一步研究以确定 MOG 和 LEN 在 allo-HSCT 后复发 ATLL 中的最佳应用。
