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A case report: Pathological complete response to neoadjuvant lorlatinib for Epithelioid inflammatory myofibroblastic sarcoma with EML4-ALK rearrangement

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Title: A case report: Pathological complete response to neoadjuvant lorlatinib for Epithelioid inflammatory myofibroblastic sarcoma with EML4-ALK rearrangement
Authors: Yang Zheng, Fanfei Zhao, Yaqian Ren, Yaran Xue, Bing Yan, Chun Huang
Source: Frontiers in Pharmacology, Vol 15 (2024)
Publisher Information: Frontiers Media S.A., 2024.
Publication Year: 2024
Collection: LCC:Therapeutics. Pharmacology
Subject Terms: EML4-ALK, Epithelioid inflammatory myofibroblastic sarcoma, neoadjuvant treatment, lorlatinib, pathological complete response, Therapeutics. Pharmacology, RM1-950
More Details: Inflammatory myofibroblastic tumor (IMT) is a rare tumor originating from mesenchymal tissue. Epithelioid inflammatory myofibroblastic sarcoma (EIMS) represents a rare and particularly aggressive variant, associated with a worse prognosis. Almost all EIMS cases exhibits activating anaplastic lymphoma kinase (ALK) gene rearrangements, which suggests that EIMS patients may potentially benefit from treatment with ALK tyrosine kinase inhibitors (TKIs). We presented a case involving a 34-year-old woman who was diagnosed with mediastinal EIMS and had a rare echinoderm microtubule-associated protein-like 4 (EML4) -ALK fusion. Following 15 months of neoadjuvant lorlatinib treatment, the patient underwent a complete surgical resection, resulting in a pathological complete response. Given the heightened risk of postoperative recurrence associated with EIMS, the patient’s treatment plan included ongoing adjuvant therapy with lorlatinib. As of the present moment, the patient has achieved an overall survival of over 2 years with no observed tumor recurrence. Consequently, the case offers valuable clinical evidence supporting the potential benefits of neoadjuvant lorlatinib treatment for ALK-positive locally mediastinal EIMS patients, with a demonstrated tolerable safety profile.
Document Type: article
File Description: electronic resource
Language: English
ISSN: 1663-9812
Relation: https://www.frontiersin.org/articles/10.3389/fphar.2024.1401428/full; https://doaj.org/toc/1663-9812
DOI: 10.3389/fphar.2024.1401428
Access URL: https://doaj.org/article/526d3b3419c244238bd988373ec54efd
Accession Number: edsdoj.526d3b3419c244238bd988373ec54efd
Database: Directory of Open Access Journals
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  Data: A case report: Pathological complete response to neoadjuvant lorlatinib for Epithelioid inflammatory myofibroblastic sarcoma with EML4-ALK rearrangement
– Name: Author
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  Data: <searchLink fieldCode="AR" term="%22Yang+Zheng%22">Yang Zheng</searchLink><br /><searchLink fieldCode="AR" term="%22Fanfei+Zhao%22">Fanfei Zhao</searchLink><br /><searchLink fieldCode="AR" term="%22Yaqian+Ren%22">Yaqian Ren</searchLink><br /><searchLink fieldCode="AR" term="%22Yaran+Xue%22">Yaran Xue</searchLink><br /><searchLink fieldCode="AR" term="%22Bing+Yan%22">Bing Yan</searchLink><br /><searchLink fieldCode="AR" term="%22Chun+Huang%22">Chun Huang</searchLink>
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  Data: Frontiers in Pharmacology, Vol 15 (2024)
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  Data: <searchLink fieldCode="DE" term="%22EML4-ALK%22">EML4-ALK</searchLink><br /><searchLink fieldCode="DE" term="%22Epithelioid+inflammatory+myofibroblastic+sarcoma%22">Epithelioid inflammatory myofibroblastic sarcoma</searchLink><br /><searchLink fieldCode="DE" term="%22neoadjuvant+treatment%22">neoadjuvant treatment</searchLink><br /><searchLink fieldCode="DE" term="%22lorlatinib%22">lorlatinib</searchLink><br /><searchLink fieldCode="DE" term="%22pathological+complete+response%22">pathological complete response</searchLink><br /><searchLink fieldCode="DE" term="%22Therapeutics%2E+Pharmacology%22">Therapeutics. Pharmacology</searchLink><br /><searchLink fieldCode="DE" term="%22RM1-950%22">RM1-950</searchLink>
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  Data: Inflammatory myofibroblastic tumor (IMT) is a rare tumor originating from mesenchymal tissue. Epithelioid inflammatory myofibroblastic sarcoma (EIMS) represents a rare and particularly aggressive variant, associated with a worse prognosis. Almost all EIMS cases exhibits activating anaplastic lymphoma kinase (ALK) gene rearrangements, which suggests that EIMS patients may potentially benefit from treatment with ALK tyrosine kinase inhibitors (TKIs). We presented a case involving a 34-year-old woman who was diagnosed with mediastinal EIMS and had a rare echinoderm microtubule-associated protein-like 4 (EML4) -ALK fusion. Following 15 months of neoadjuvant lorlatinib treatment, the patient underwent a complete surgical resection, resulting in a pathological complete response. Given the heightened risk of postoperative recurrence associated with EIMS, the patient’s treatment plan included ongoing adjuvant therapy with lorlatinib. As of the present moment, the patient has achieved an overall survival of over 2 years with no observed tumor recurrence. Consequently, the case offers valuable clinical evidence supporting the potential benefits of neoadjuvant lorlatinib treatment for ALK-positive locally mediastinal EIMS patients, with a demonstrated tolerable safety profile.
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  Data: https://www.frontiersin.org/articles/10.3389/fphar.2024.1401428/full; https://doaj.org/toc/1663-9812
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        Value: 10.3389/fphar.2024.1401428
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      – Text: English
    Subjects:
      – SubjectFull: EML4-ALK
        Type: general
      – SubjectFull: Epithelioid inflammatory myofibroblastic sarcoma
        Type: general
      – SubjectFull: neoadjuvant treatment
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      – SubjectFull: lorlatinib
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      – TitleFull: A case report: Pathological complete response to neoadjuvant lorlatinib for Epithelioid inflammatory myofibroblastic sarcoma with EML4-ALK rearrangement
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            NameFull: Yang Zheng
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              Y: 2024
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