Short abstract
This letter to the editor responds to questions about the results of the authors’ recently reported study of chemotherapy in inflammatory myofibroblastic tumors.
We are glad to answer P. Schöffski's comments [1] on our article on cytotoxic chemotherapy in advanced inflammatory myofibroblastic tumor (IMT) [2].
With regard to pathologic diagnosis, we believe that in our retrospective study, the risk of misclassification was minimized by involving three major sarcoma reference centers in the U.K. and France and centralizing pathologic review of cases from the other six Italian centers on a network basis. ALK status by immunohistochemistry and/or of fluorescence in situ hybridization was required for all cases.
With regard to the “informed consent to treatment,” this was actually written, whereas each institutional review board approved the retrospective case series analysis according to local rules.
We understand that our response rate was “in sharp contrast with retrospective data collected in the EORTC trial CREATE” [3]. Indeed, in both cases we are talking of a retrospective assessment, with all the limitations thereof (also, in our study, responses were retrospectively evaluated by local radiologists and not centrally reviewed, although this happened at sarcoma reference centers, having a special expertise about the many difficulties of response assessment in sarcomas). All other published data are retrospective as well. However, although scanty and confined to children or young adults, these data look more consistent with our findings, especially if one focuses on anthracyclines or methotrexate/vinca alkaloids [4, 5, 6, 7, 8].
This said, we are all aware of the many limitations of the concept of tumor response in oncology. In this sense, probably the main observation made in our effort was the prolonged progression‐free survival (PFS) of 13 patients with IMT treated with methotrexate/vinca alkaloids, with a median PFS not reached at a follow‐up of 56 months. If confirmed, this would compare favorably with the 12 patients with ALK‐positive IMT treated with crizotinib in the CREATE study.
Indeed, we do believe that a prospective study thereon would be worthwhile. In such a rare disease, prospective efforts like the CREATE study, which enrolled 19 evaluable patients with IMT in 5 years, should be deeply congratulated, and possibly replicated by means of collaborations as large as possible within the international sarcoma community.
Disclosures
Silvia Stacchiotti: Bayer, Bavarian Nordic, Deciphera, Daiichi, Eli Lilly & Co, Epizyme, Karyopharm, MaxiVax, Pharmamar (SAB), Eli Lilly & Co, Pharmamar (H), Pharmamar (Other), Advenchen, Amgen Dompé, AROG, Bayer, Blueprint Medicines, Daiichi Sankyo Pharma, Deciphera, Eli Lilly & Co, Epizyme, GlaxoSmithKline, Karyopharm, Novartis, Pfizer, PharmaMar, SpringWorks (institutional RF); Giacomo Giulio Baldi: Eli Lilly & Co, Eisai, PharmaMar (H), PharmaMar, Pfizer, Eli Lilly & Co (Other), AboutEvents, EditaMed, Eli Lilly & Co (SAB); Paolo Giovanni Casali: Bayer, Glaxo, Novartis (SAB), Advenchen, Amgen Dompé, AROG, Bayer, Blueprint Medicines, Daiichi Sankyo Pharma, Deciphera, Eli Lilly & Co, Epizyme, GlaxoSmithKline, Karyopharm, Novartis, Pfizer, PharmaMar, SpringWorks (institutional RF).
(C/A) Consulting/advisory relationship; (RF) Research funding; (E) Employment; (ET) Expert testimony; (H) Honoraria received; (OI) Ownership interests; (IP) Intellectual property rights/inventor/patent holder; (SAB) Scientific advisory board
This article related to “Regarding “The Activity of Chemotherapy in Inflammatory Myofibroblastic Tumors”” by Patrick Schöffski on page e2017 in this issue.
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