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Therapeutic options in inoperable ROS1-rearranged inflammatory myofibroblastic tumor of the tongue in a child: a case report and literature review

Therapeutic options in inoperable ROS1-rearranged inflammatory myofibroblastic tumor of the... Inflammatory myofibroblastic tumor (IMT) is a rare borderline malignancy, usually treated with surgery only. Exceedingly rare cases of inoperable, recurrent, or metastatic IMTs pose a therapeutic challenge. We report successful treatment of a 7-year-old girl with an inoperable anaplastic lymphoma kinase (ALK)-negative IMT of the tongue. The patient underwent various anti-inflammatory (steroids, nonsteroidal anti-inflammatory drugs, clarithromycin) and antiproliferative (chemotherapy) therapies to enable tumor regression and complete resection. Ultimately, next-generation sequencing of the tumor revealed a TFG-ROS-1 translocation, allowing for an off-label targeted therapy with crizotinib. Crizotinib treatment caused slight tumor regression but evident change of its structure, allowing for complete non-mutilating resection. Two histopathology examinations revealed complete disappearance of neoplastic cells following therapy. The patient remains disease-free 22 months after the delayed surgery. In children with inoperable ALK-negative IMTs, molecular testing must be performed to identify other targetable oncogenic fusions, including TFG-ROS1. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Anti-Cancer Drugs Wolters Kluwer Health

Therapeutic options in inoperable ROS1-rearranged inflammatory myofibroblastic tumor of the tongue in a child: a case report and literature review

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Publisher
Wolters Kluwer Health
Copyright
Copyright © 2021 Wolters Kluwer Health, Inc. All rights reserved.
ISSN
0959-4973
eISSN
1473-5741
DOI
10.1097/cad.0000000000001099
Publisher site
See Article on Publisher Site

Abstract

Inflammatory myofibroblastic tumor (IMT) is a rare borderline malignancy, usually treated with surgery only. Exceedingly rare cases of inoperable, recurrent, or metastatic IMTs pose a therapeutic challenge. We report successful treatment of a 7-year-old girl with an inoperable anaplastic lymphoma kinase (ALK)-negative IMT of the tongue. The patient underwent various anti-inflammatory (steroids, nonsteroidal anti-inflammatory drugs, clarithromycin) and antiproliferative (chemotherapy) therapies to enable tumor regression and complete resection. Ultimately, next-generation sequencing of the tumor revealed a TFG-ROS-1 translocation, allowing for an off-label targeted therapy with crizotinib. Crizotinib treatment caused slight tumor regression but evident change of its structure, allowing for complete non-mutilating resection. Two histopathology examinations revealed complete disappearance of neoplastic cells following therapy. The patient remains disease-free 22 months after the delayed surgery. In children with inoperable ALK-negative IMTs, molecular testing must be performed to identify other targetable oncogenic fusions, including TFG-ROS1.

Journal

Anti-Cancer Drugs – Wolters Kluwer Health

Published: Nov 17, 2021

References

  • Inflammatory myofibroblastic tumour of the lung: a reactive lesion or a true neoplasm?
    Panagiotopoulos, N; Patrini, D; Gvinianidze, L; Woo, WL; Borg, E; Lawrence, D
  • Inflammatory myofibroblastic tumors in children.
    Dalton, BG; Thomas, PG; Sharp, NE; Manalang, MA; Fisher, JE; Moir, CR
  • Neoadjuvant treatment with cyclooxygenase-2 inhibitor and prednisolone allows conservative surgery for inflammatory myofibroblastic tumor of the bladder.
    Tsuma, Y; Miyachi, M; Ouchi, K; Tsuchiya, K; Iehara, T; Naitoh, Y
  • Recurrent involvement of 2p23 in inflammatory myofibroblastic tumors.
    Griffin, CA; Hawkins, AL; Dvorak, C; Henkle, C; Ellingham, T; Perlman, EJ
  • Crizotinib and surgery for long-term disease control in children and adolescents with ALK-positive inflammatory myofibroblastic tumors.
    Trahair, T; Gifford, AJ; Fordham, A; Mayoh, C; Fadia, M; Lukeis, R
  • Inflammatory myofibroblastic tumors harbor multiple potentially actionable kinase fusions.
    Lovly, CM; Gupta, A; Lipson, D; Otto, G; Brennan, T; Chung, CT
  • ETV6-NTRK3 is expressed in a subset of ALK-negative inflammatory myofibroblastic tumors.
    Alassiri, AH; Ali, RH; Shen, Y; Lum, A; Strahlendorf, C; Deyell, R
  • Inflammatory myofibroblastic tumors-A retrospective analysis of the Cooperative Weichteilsarkom Studiengruppe.
    Kube, S; Vokuhl, C; Dantonello, T; Scheer, M; Hallmen, E; Feuchtgruber, S
  • Durable response to the ALK inhibitor alectinib in inflammatory myofibroblastic tumor of the head and neck with a novel SQSTM1-ALK fusion: a case report.
    Honda, K; Kadowaki, S; Kato, K; Hanai, N; Hasegawa, Y; Yatabe, Y; Muro, K
  • Remission of ALK-negative primary pulmonary inflammatory myofibroblastic tumor on treatment with clarithromycin: a case report and review of the literature.
    Watanabe, H; Uruma, T; Tazaki, G; Tajiri, T; Kikuchi, R; Itoh, M
  • Successful treatment of an unresectable inflammatory myofibroblastic tumor of the frontal bone using a cyclooxygenase-2 inhibitor and methotrexate.
    Kusunoki-Nakamoto, F; Matsukawa, T; Tanaka, M; Miyagawa, T; Yamamoto, T; Shimizu, J
  • The 2020 WHO classification of soft tissue tumours: news and perspectives.
    Sbaraglia, M; Bellan, E; Dei Tos, AP
  • Régression complète d’une tumeur myofibroblastique inflammatoire de vessie après antibiothérapie.
    Alezra, E; Delforge, X; Buisson, P; Gourmel, A; Cordonnier, C; Ricard, J
  • Complete remission of ALK-negative plasma cell granuloma (inflammatory myofibroblastic tumor) of the lung induced by celecoxib: a case report and review of the literature.
    Chavez, C; Hoffman, MA
  • Infantile inflammatory myofibroblastic tumors: clinicopathological and molecular characterization of 12 cases.
    Lopez-Nunez, O; John, I; Panasiti, RN; Ranganathan, S; Santoro, L; Grélaud, D
  • Clinicopathological study of 18 cases of inflammatory myofibroblastic tumors with reference to ALK-1 expression: 5-year experience in a tertiary care center.
    Telugu, RB; Prabhu, AJ; Kalappurayil, NB; Mathai, J; Gnanamuthu, BR; Manipadam, MT
  • Molecular characterization of inflammatory myofibroblastic tumors with frequent ALK and ROS1 gene fusions and rare novel RET rearrangement.
    Antonescu, CR; Suurmeijer, AJ; Zhang, L; Sung, YS; Jungbluth, AA; Travis, WD
  • Expanding the molecular characterization of thoracic inflammatory myofibroblastic tumors beyond ALK gene rearrangements.
    Chang, JC; Zhang, L; Drilon, AE; Chi, P; Alaggio, R; Borsu, L
  • Anaplastic lymphoma kinase expression in inflammatory pseudotumors.
    Chan, JK; Cheuk, W; Shimizu, M
  • Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases.
    Coffin, CM; Hornick, JL; Fletcher, CD
  • Entrectinib in two pediatric patients with inflammatory myofibroblastic tumors harboring ROS1 or ALK gene fusions.
    Ambati, SR; Slotkin, EK; Chow-Maneval, E; Basu, EM
  • Case report: Crizotinib is effective in a patient with ROS1-rearranged pulmonary inflammatory myofibroblastic tumor.
    Mai, S; Xiong, G; Diao, D; Wang, W; Zhou, Y; Cai, R
  • Crizotinib in children and adolescents with advanced ROS1, MET, or ALK-rearranged cancer: results of the AcSé phase II trial.
    Vassal, G; Faivre, L; Geoerger, B; Plantaz, D; Auvrignon, A; Coze, C
  • Partial response to ceritinib in a patient with abdominal inflammatory myofibroblastic tumor carrying a TFG-ROS1 fusion.
    Li, Y; Chen, X; Qu, Y; Fan, JM; Li, Y; Peng, H