Epithelioid inflammatory myofibroblastic sarcoma arising in the maxilla: first reported case in the jaws

Nayyar, Vivek; Bhutia, Ongkila; Kakkar, Aanchal; Mishra, Deepika

doi:10.1016/j.oooo.2023.03.001

Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology

2023

DOI: 10.1016/j.oooo.2023.03.001

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Epithelioid inflammatory myofibroblastic sarcoma arising in the maxilla: first reported case in the jaws

Vivek Nayyar¹,

Ongkila Bhutia²,

Aanchal Kakkar³

et al.

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2024

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Cited by 2 publications

References 14 publications

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Epithelioid inflammatory myofibroblastic sarcoma with exceptionally long response to lorlatinib—a case report

Becht,

Kiełbowski,

Żychowska

et al. 2024

Ther Adv Med Oncol

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Epithelioid inflammatory myofibroblastic sarcoma (EIMS) is a rare and aggressive subtype of inflammatory myofibroblastic tumor. The disease is associated with rearrangements of the anaplastic lymphoma kinase (ALK). In this paper, we present the clinicopathological features and treatment of a female patient diagnosed with EIMS. In 2019, an 18-year-old female patient was admitted to the hospital with abdominal pain. Radiological examinations confirmed a large pelvic mass which was subsequently resected. After re-evaluation of the initial histologic diagnosis, the final diagnosis of EIMS was established. Consequently, due to the lack of response to chemotherapy and deteriorating clinical condition, she began the therapy with ALK inhibitors. In total, the patient was treated with crizotinib, alectinib, and lorlatinib. As a result, after over 4 years since the initial diagnosis, she is still alive with significantly improved clinical condition and quality of life. This paper demonstrates the clinical benefits of sequential therapy of ALK inhibitors and an exceptionally long response to lorlatinib, a third-generation tyrosine kinase inhibitor.

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Epithelioid inflammatory myofibroblastic sarcoma with exceptionally long response to lorlatinib—a case report

Becht,

Kiełbowski,

Żychowska

et al. 2024

Ther Adv Med Oncol

View full text Add to dashboard Cite

show abstract

Rare giant epithelioid inflammatory myofibroblastic sarcoma of the abdominal cavity in a child: a case report and review of the literature

Li,

Su,

Zhang

et al. 2024

Front. Oncol.

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Epithelioid inflammatory myofibroblastic sarcoma (EIMS) is a distinct subtype of inflammatory myofibroblastoma tumor (IMT) that is recognized as a rare malignant tumor characterized by anaplastic lymphoma kinase (ALK) positivity, significant aggressiveness, treatment challenges, and a poor prognosis. We report on the case of an 8-year-old boy presenting with abdominal pain and vomiting. Computed tomography (CT) of the abdomen revealed a large tumor, and the pathology results following a biopsy confirmed the diagnosis of EIMS. The patient underwent radical tumor resection, and genetic testing identified the presence of the RANBP2–ALK fusion. To our knowledge, this represents the largest pediatric case of abdominal EIMS documented in the literature. Currently, there is no standard therapy for EIMS; however, existing studies advocate for the use of ALK tyrosine kinase inhibitors (TKIs) in its treatment. This case was reported to be in remission following treatment with crizotinib, thereby contributing to the understanding of the specific pathology of EIMS and facilitating accurate diagnosis and targeted therapy.

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Epithelioid inflammatory myofibroblastic sarcoma arising in the maxilla: first reported case in the jaws

Cited by 2 publications

References 14 publications

Epithelioid inflammatory myofibroblastic sarcoma with exceptionally long response to lorlatinib—a case report

Epithelioid inflammatory myofibroblastic sarcoma with exceptionally long response to lorlatinib—a case report

Rare giant epithelioid inflammatory myofibroblastic sarcoma of the abdominal cavity in a child: a case report and review of the literature

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