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Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor

Inflammatory myofibroblastic tumors (IMTs) are locally aggressive malignancies occurring at various sites. Surgery is the mainstay of treatment and prognosis is generally good. For children with unresectable or metastatic tumors, however, outcome is particularly severe, limited also by the lack of p...

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Autores principales: Bonvini, Paolo, Rossi, Elisabetta, Zin, Angelica, Manicone, Mariangela, Vidotto, Riccardo, Facchinetti, Antonella, Tombolan, Lucia, Affinita, Maria Carmen, Santoro, Luisa, Zamarchi, Rita, Bisogno, Gianni
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8116882/
https://www.ncbi.nlm.nih.gov/pubmed/33996693
http://dx.doi.org/10.3389/fped.2021.652583
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author Bonvini, Paolo
Rossi, Elisabetta
Zin, Angelica
Manicone, Mariangela
Vidotto, Riccardo
Facchinetti, Antonella
Tombolan, Lucia
Affinita, Maria Carmen
Santoro, Luisa
Zamarchi, Rita
Bisogno, Gianni
author_facet Bonvini, Paolo
Rossi, Elisabetta
Zin, Angelica
Manicone, Mariangela
Vidotto, Riccardo
Facchinetti, Antonella
Tombolan, Lucia
Affinita, Maria Carmen
Santoro, Luisa
Zamarchi, Rita
Bisogno, Gianni
author_sort Bonvini, Paolo
collection PubMed
description Inflammatory myofibroblastic tumors (IMTs) are locally aggressive malignancies occurring at various sites. Surgery is the mainstay of treatment and prognosis is generally good. For children with unresectable or metastatic tumors, however, outcome is particularly severe, limited also by the lack of predictive biomarkers of therapy efficacy and disease progression. Blood represents a minimally invasive source of cancer biomarkers for real-time assessment of tumor growth, particularly when it involves the analysis of circulating tumor cells (CTC). As CTCs potentially represent disseminated disease, their detection in the blood correlates with the presence of metastatic lesions and may reflect tumor response to treatment. Herein, we present a case report of a 19-year-old boy with an ALK-positive IMT of the bladder, proximal osteolytic and multiple bilateral lung lesions, who received ALK inhibitor entrectinib postoperatively and underwent longitudinal CTC analysis during treatment. Antitumor activity of entrectinib was demonstrated and was accompanied by regression of lung lesions, elimination of CTCs from the blood and no development of relapses afterwards. Therapy continued without any clinical sign of progression and 24 months since the initiation of treatment the patient remains symptom-free and disease-free.
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spelling pubmed-81168822021-05-14 Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor Bonvini, Paolo Rossi, Elisabetta Zin, Angelica Manicone, Mariangela Vidotto, Riccardo Facchinetti, Antonella Tombolan, Lucia Affinita, Maria Carmen Santoro, Luisa Zamarchi, Rita Bisogno, Gianni Front Pediatr Pediatrics Inflammatory myofibroblastic tumors (IMTs) are locally aggressive malignancies occurring at various sites. Surgery is the mainstay of treatment and prognosis is generally good. For children with unresectable or metastatic tumors, however, outcome is particularly severe, limited also by the lack of predictive biomarkers of therapy efficacy and disease progression. Blood represents a minimally invasive source of cancer biomarkers for real-time assessment of tumor growth, particularly when it involves the analysis of circulating tumor cells (CTC). As CTCs potentially represent disseminated disease, their detection in the blood correlates with the presence of metastatic lesions and may reflect tumor response to treatment. Herein, we present a case report of a 19-year-old boy with an ALK-positive IMT of the bladder, proximal osteolytic and multiple bilateral lung lesions, who received ALK inhibitor entrectinib postoperatively and underwent longitudinal CTC analysis during treatment. Antitumor activity of entrectinib was demonstrated and was accompanied by regression of lung lesions, elimination of CTCs from the blood and no development of relapses afterwards. Therapy continued without any clinical sign of progression and 24 months since the initiation of treatment the patient remains symptom-free and disease-free. Frontiers Media S.A. 2021-04-29 /pmc/articles/PMC8116882/ /pubmed/33996693 http://dx.doi.org/10.3389/fped.2021.652583 Text en Copyright © 2021 Bonvini, Rossi, Zin, Manicone, Vidotto, Facchinetti, Tombolan, Affinita, Santoro, Zamarchi and Bisogno. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Bonvini, Paolo
Rossi, Elisabetta
Zin, Angelica
Manicone, Mariangela
Vidotto, Riccardo
Facchinetti, Antonella
Tombolan, Lucia
Affinita, Maria Carmen
Santoro, Luisa
Zamarchi, Rita
Bisogno, Gianni
Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor
title Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor
title_full Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor
title_fullStr Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor
title_full_unstemmed Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor
title_short Case Report: Circulating Tumor Cells as a Response Biomarker in ALK-Positive Metastatic Inflammatory Myofibroblastic Tumor
title_sort case report: circulating tumor cells as a response biomarker in alk-positive metastatic inflammatory myofibroblastic tumor
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8116882/
https://www.ncbi.nlm.nih.gov/pubmed/33996693
http://dx.doi.org/10.3389/fped.2021.652583
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