Ozkan Alan1, Okan Kuzhan2, Sinan Koca3, Tugba Akin Telli1, Tugba Basoglu1, Ozlem Ercelep1, Deniz Filinte4, Yildiz Sengul5, Huseyin Arikan6, Serap Kaya1, Nalan Akgul Babacan1, Faysal Dane1, Perran Fulden Yumuk1. 1. Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey. 2. Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Yeditep University, Istanbul, Turkey. 3. Division of Medical Oncology, Umraniye Research and Training Hospital, Istanbul, Turkey. 4. Department of Pathology, Faculty of Medicine, Marmara University, Istanbul, Turkey. 5. Department of Radiology, Faculty of Medicine, Marmara University, Istanbul, Turkey. 6. Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey.
Abstract
INTRODUCTION: Inflammatory myofibroblastic tumor is a rare disease which is typically seen in children and young adults. Approximately half of the inflammatory myofibroblastic tumors contain translocations that result in over-expression of anaplastic lymphoma kinase gene. Herein, we present two anaplastic lymphoma kinase-positive cases with long-term remission with crizotinib. We do not know how long these therapies need to be continued. CASE REPORTS: We present two cases of inflammatory myofibroblastic tumor treated with anaplastic lymphoma kinase inhibitor therapies: an 8-year-old Turkish boy and a 21-year-old Caucasian man. MANAGEMENT AND OUTCOME: Two cases, both with good tumor control under crizotinib, but one who progressed on drug holiday, responded again to the same drug, and had a very short period of response after restarting crizotinib. CONCLUSION: A molecular-targeted drug (anaplastic lymphoma kinase inhibitor) was found to be extremely effective as selective therapy for inflammatory myofibroblastic tumor with anaplastic lymphoma kinase translocation. Here, we want to emphasize the continuation of this treatment after achieving a good response until progression or a major side effect.
INTRODUCTION: Inflammatory myofibroblastic tumor is a rare disease which is typically seen in children and young adults. Approximately half of the inflammatory myofibroblastic tumors contain translocations that result in over-expression of anaplastic lymphoma kinase gene. Herein, we present two anaplastic lymphoma kinase-positive cases with long-term remission with crizotinib. We do not know how long these therapies need to be continued. CASE REPORTS: We present two cases of inflammatory myofibroblastic tumor treated with anaplastic lymphoma kinase inhibitor therapies: an 8-year-old Turkish boy and a 21-year-old Caucasian man. MANAGEMENT AND OUTCOME: Two cases, both with good tumor control under crizotinib, but one who progressed on drug holiday, responded again to the same drug, and had a very short period of response after restarting crizotinib. CONCLUSION: A molecular-targeted drug (anaplastic lymphoma kinase inhibitor) was found to be extremely effective as selective therapy for inflammatory myofibroblastic tumor with anaplastic lymphoma kinase translocation. Here, we want to emphasize the continuation of this treatment after achieving a good response until progression or a major side effect.