Recurrent uterine inflammatory myofibroblastic tumor previously managed as leiomyosarcoma has sustained response to alectinib

Carballo, Erica V.; Pham, Tra V.; Turashvili, Gulisa; Hanley, Krisztina; Starbuck, Kristen; Meisel, Jane L.

doi:10.1016/j.gore.2022.101062

Gynecologic Oncology Reports

2022

DOI: 10.1016/j.gore.2022.101062

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Recurrent uterine inflammatory myofibroblastic tumor previously managed as leiomyosarcoma has sustained response to alectinib

Erica V. Carballo

Tra V. Pham

Gulisa Turashvili

et al.

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Cited by 4 publications

References 18 publications

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Inflammatory myofibroblastic tumors: recent progress and future of targeted therapy

Nakano

2023

Japanese Journal of Clinical Oncology

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An inflammatory myofibroblastic tumor is a rare component of bone and soft-tissue sarcomas that has distinct pathological features as a lymphoplasmacytic inflammatory infiltrate. As is the case for other non-small round cell sarcomas, surgical resection remains the standard treatment strategy for inflammatory myofibroblastic tumors, but recurrence is possible. Concerning systemic therapy, the available data for conventional chemotherapy (such as those of doxorubicin-based regimens) are limited, and case reports of anti-inflammatory inflammatory myofibroblastic tumor treatments describe some degree of symptom relief and efficacy against tumor progression. However, as more information about cancer genomics accumulates, the potential for molecularly targeted therapies for inflammatory myofibroblastic tumors has become more promising. Approximately half of inflammatory myofibroblastic tumors harbor anaplastic lymphoma kinase (ALK) fusion genes, and the other half could have potentially targetable fusion genes or mutations such as ROS1, NTRK and RET; case reports demonstrating the clinical efficacy of treatments targeted to inflammatory myofibroblastic tumor have been published, as have several prospective clinical trials. Few drugs are approved for the treatment of inflammatory myofibroblastic tumor, and most of them were approved for tumor-agnostic indications. Drugs that could be used for pediatric indications and dosing in inflammatory myofibroblastic tumor have also not been established. To provide effective targeted therapy for rare diseases such as inflammatory myofibroblastic tumor, it is necessary to obtain clinical evidence by designing and performing clinical trials and to find a path toward regulatory approval.

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Inflammatory myofibroblastic tumors: recent progress and future of targeted therapy

Nakano

2023

Japanese Journal of Clinical Oncology

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The “Other” Uterine Mesenchymal Neoplasms: Recent Developments and Emerging Entities

Bennett,

Pinto

2024

Advances in Anatomic Pathology

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Uterine mesenchymal neoplasms are a challenging group of tumors that often show overlapping morphologic features and immunohistochemical profiles. The increasing use of molecular testing in these tumors has enabled a better appreciation of their pathobiology, resulting in a wave of emerging neoplasms and improved characterization of ones previously considered exceptionally rare. Identification of specific molecular alterations has permitted targeted therapy options in tumors that were typically unresponsive to conventional therapies, as well as recognition that a subset can have a hereditary basis. This review will discuss the more “common” of the uncommon uterine mesenchymal neoplasms, including inflammatory myofibroblastic tumor, perivascular epithelioid cell tumor, uterine tumor resembling ovarian sex cord tumor, and embryonal rhabdomyosarcoma. This will be followed by an overview of emerging entities, including NTRK-rearranged uterine sarcoma, SMARCA4-deficient uterine sarcoma, KAT6B/A::KANSL1 fusion uterine sarcoma, and MEIS1::NCOA2/1 fusion sarcoma.

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Pneumothorax during Alectinib Treatment for a Uterine Inflammatory Myofibroblastic Tumor with Lung Metastasis

Liu

Lin

et al. 2023

Journal of Cancer Research and Practice

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An inflammatory myofibroblastic tumor (IMT) is an uncommon sarcoma subtype with histopathological features, including inflammatory infiltrates. Anaplastic lymphoma kinase (ALK) gene rearrangement has been reported in half of the patients with IMTs; therefore, crizotinib, an ALK inhibitor, may achieve a response rate of 50% in these patients. We present a case with an initial diagnosis of uterine sarcoma and multiple lung metastases. After the failure of doxorubicin-based chemotherapy, revised pathology from a palliative hysterectomy revealed an IMT with ALK gene rearrangement. Treatment with alectinib achieved an excellent tumor response. The accurate differential diagnosis of uncommon sarcoma subtypes is crucial because a specific targeted therapy may considerably alter treatment outcomes.

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Recurrent uterine inflammatory myofibroblastic tumor previously managed as leiomyosarcoma has sustained response to alectinib

Cited by 4 publications

References 18 publications

Inflammatory myofibroblastic tumors: recent progress and future of targeted therapy

Inflammatory myofibroblastic tumors: recent progress and future of targeted therapy

The “Other” Uterine Mesenchymal Neoplasms: Recent Developments and Emerging Entities

Pneumothorax during Alectinib Treatment for a Uterine Inflammatory Myofibroblastic Tumor with Lung Metastasis

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