• Sumiko Saito, Haruko Tashiro, Ritsu Sumiyoshi, Takuji Matsuo, Tadashi Yamamoto, Kensuke Matsumoto, Jun Ooi, and Naoki Shirafuji.
• Department of Hematology/Oncology, Teikyo University School of Medicine, Tokyo, Japan.
• Medicine (Baltimore). 2021 Apr 16; 100 (15): e25576.

RationaleAnaplastic lymphoma kinase (ALK) + anaplastic large cell lymphoma (ALCL) is considered as a good prognosis lymphoma. However, in an extremely rare subset of patients, ALK+ ALCL with leukemic presentations is known to be chemotherapy-resistant. Although several novel therapies have been tested, the standard therapy for relapsed/refractory ALK+ ALCL has not been established yet.Patient ConcernsAn 18-year-old female patient who had conventional chemotherapy- and Brentuximab Vedotin (BV)-resistant ALK+ ALCL with leukemic presentation. She was successfully treated with an ALK inhibitor, crizotinib. Crizotinib induced complete remission (CR) and bridged to allogeneic bone marrow transplantation (BMT).DiagnosisHowever, her ALCL relapsed on day 60 after BMT and she developed high grade fever and lymphadenopathy.InterventionAlthough crizotinib was given to the patient immediately after relapse, she developed grade 3 nausea and could not continue to take it. Then, we gave alectinib to the patient, which promptly induced sustained CR without any further chemotherapy. The patient received second stem cell transplantation using umbilical cord blood with myeloablative regimen in 2nd CR.OutcomesThe patient has been in CR under maintenance therapy of alectinib for more than 16 months.LessonsBoth ALK inhibitors demonstrated drastic efficacy for our patient who had chemotherapy- and BV-resistant ALK+ ALCL with leukemic presentation. Alectinib showed less gastro-intestinal toxicity than crizotinib and the patient was able to take it even at the relatively early phase of stem cell transplantation.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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