Anaplastic lymphoma kinase inhibitor therapy in the treatment of inflammatory myofibroblastic tumors in pediatric patients: Case reports and literature review.

Autor: Craig E; Department of Surgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States., Wiltsie LM; Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States., Beaupin LK; Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences and Department of Pediatrics, Roswell Park Comprehensive Cancer Center, Buffalo, NY, United States., Baig A; Division of Pediatric Gastroenterology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States., Kozielski R; Department of Pathology, John R. Oishei Children's Hospital, Buffalo, NY, United States., Rothstein DH; Department of Pediatric Surgery John R. Oishei Children's Hospital and Department of Surgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States. Electronic address: drothste@uw.edu., Li V; Department of Pediatric Neurosurgery, John R. Oishei Children's Hospital and Department of Neurosurgery, University at Buffalo Jacobs School of Medicine and Biomedical Sciences, Buffalo, NY, United States., Twist CJ; Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences and Department of Pediatrics, Roswell Park Comprehensive Cancer Center, Buffalo, NY, United States., Barth M; Division of Pediatric Hematology/Oncology, University at Buffalo Jacobs School of Medicine and Biomedical Sciences and Department of Pediatrics, Roswell Park Comprehensive Cancer Center, Buffalo, NY, United States.
Jazyk: angličtina
Zdroj: Journal of pediatric surgery [J Pediatr Surg] 2021 Dec; Vol. 56 (12), pp. 2364-2371. Date of Electronic Publication: 2021 Feb 11.
DOI: 10.1016/j.jpedsurg.2021.02.004
Abstrakt: Background: Inflammatory myofibroblastic tumors (IMTs) are a rare subtype of inflammatory pseudotumor frequently associated with rearrangement of the anaplastic lymphoma kinase (ALK) gene. Their treatment has historically relied on at-times challenging and morbid surgical excision. Recent studies have shown that neo/adjuvant therapy with ALK inhibitors can significantly enhance outcomes in select patients.
Methods: A systematic literature review was performed to characterize comprehensive treatment of ALK-positive IMTs in the pediatric population. This report also includes two patients from our home institutions not previously reported in the literature.
Results: We identified a total of 27 patients in 12 studies in addition to 2 patients from the senior authors' institution for a total of 29 patients (median age, 7 years; 52% male). The IMTs comprised a wide range of anatomic locations. Almost half (12, 41.3%) were treated with ALK-inhibitors alone and felt to be in remission. The remainder was treated with ALK-inhibitors either before or after surgery and had a curative response.
Conclusions: ALK-positive IMTs can be successfully treated with ALK-inhibition alone or in combination with surgical resection. Further genetic characterization may be helpful in determining more precise treatment and defining needed durations thereof.
(Copyright © 2021. Published by Elsevier Inc.)
Databáze: MEDLINE
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