title: Complete and Repeated Response of a Metastatic ALK-rearranged Inflammatory Myofibroblastic Tumor to Crizotinib in a Teenage Girl.
abstract: Inflammatory myofibroblastic tumors (IMT) are rare tumors in children and young adults, considered by the World Health Organization to be intermediate malignancies and rarely metastasizing, with the presence of an anaplastic lymphoma kinase rearrangement in about 50% of the cases. We report the case of a teenager who presented with a metastatic aggressive IMT that was life-threatening despite multiple treatments, and which responded repeatedly to anaplastic lymphoma kinase-targeted crizotinib therapy. Crizotinib induced drastic primary tumor regression, which was sufficient to allow surgical resection and to control distant disease. This case shows that crizotinib is a promising therapy in IMT, even in adolescents and young adults.
authors.affiliation: *Oncology-Hematology Unit Departments of §Maxillofacial and Plastic Surgery ∥Thoracic Surgery, Caen University Hospital ‡Department of Pathology, Centre François Baclesse, Caen †Pediatric, Adolescent and Young Adult Oncology Department, Institut Curie, Paris, France.
authors.forename: Jérémie
authors.initials: J
authors.lastname: Gaudichon
authors.forename: Corinne
authors.initials: C
authors.lastname: Jeanne-Pasquier
authors.forename: Marianna
authors.initials: M
authors.lastname: Deparis
authors.forename: Alexis
authors.initials: A
authors.lastname: Veyssière
authors.forename: Maxime
authors.initials: M
authors.lastname: Heyndrickx
authors.forename: Odile
authors.initials: O
authors.lastname: Minckes
authors.forename: Daniel
authors.initials: D
authors.lastname: Orbach
mesh_terms: D000293:Adolescent; D000077548:Anaplastic Lymphoma Kinase; D000077547:Crizotinib; D005260:Female; D015321:Gene Rearrangement; D006801:Humans; D007249:Inflammation; D009362:Neoplasm Metastasis; D009379:Neoplasms, Muscle Tissue / Q000188:drug therapy* / Q000473:pathology / Q000601:surgery; D047428:Protein Kinase Inhibitors / Q000627:therapeutic use; D011720:Pyrazoles / Q000008:administration & dosage / Q000627:therapeutic use; D011725:Pyridines / Q000008:administration & dosage / Q000627:therapeutic use; D020794:Receptor Protein-Tyrosine Kinases / Q000037:antagonists & inhibitors / Q000235:genetics
chemical_list: D047428:Protein Kinase Inhibitors; D011720:Pyrazoles; D011725:Pyridines; D000077547:Crizotinib; C000626173:ALK protein, human; D000077548:Anaplastic Lymphoma Kinase; D020794:Receptor Protein-Tyrosine Kinases