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Case Reports
. 2023 Jul 4:14:1190210.
doi: 10.3389/fimmu.2023.1190210. eCollection 2023.

Case Report: Immunotherapy for low-grade myofibroblastic sarcoma of the pharynx

Bao Sun  1   2 , Zhiying Luo  1   2 , Ping Liu  3 , Yan He  3 , Shasha He  3 , Wenhui Liu  1   2
Affiliations
Case Reports

Case Report: Immunotherapy for low-grade myofibroblastic sarcoma of the pharynx

Bao Sun et al. Front Immunol. .

Abstract

Low-grade myofibroblastic sarcoma (LGMS) characterized by the increased proliferation of myofibroblasts is a rare type of malignant myofibroblastic tumor that frequently occurs in the head and neck region. Presently, there is no consensus regarding the treatment of LGMS. Here, we report a rare case of LGMS of the pharynx in a 40-year-old male admitted to our hospital. The patient underwent resection for a right metastatic lesion and parapharyngeal mass. However, he had recurrence and multiple metastases without a surgical indication. Then the patient received the treatment of anlotinib plus pembrolizumab for 4 cycles, and there was a partial response (PR) to the treatment. Due to the adverse reaction of anlotinib, the patient subsequently received monotherapy of pembrolizumab for 22 cycles and achieved a complete response (CR). As the first case report of the immunotherapy for LGMS, our study highlights that this strategy may be of great significance to the treatment of LGMS.

Keywords: immunotherapy; low-grade myofibroblastic sarcoma; pembrolizumab; pharynx; recurrence and multiple metastases.

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Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

Figure 1
Figure 1
Positron emission tomography and computed tomography (PET/CT) and magnetic resonance images (MRI) of lesion and mass. (A–C) PET/CT revealed a mass in right lateral wall of the pharynx, along with lung metastases. (D) MRI showed a soft tissue mass with a size of 7.5 cm × 7.5 cm × 3 cm.
Figure 2
Figure 2
Histopathological and Immunohistochemical results of the excisional biopsy. (A) The tumor cells were fusiform, arranged in fascicles or storiform growth patterns (HE, ×100). (B) A few mitoses and atypical cells with irregular nuclei were observed (HE, ×400). (C, D) Immunohistochemical results showed positive staining for α-SMA and Ki-67 with a 40% proliferation index.
Figure 3
Figure 3
The magnetic resonance images (MRI) of the LGMS. (A) In March 2020, new multiple lymphatic metastases were observed. (B) In August 2020, the patient obtained a PR after the combined treatment of pembrolizumab and anlotinib. The patient obtained a PR (C) and CR (D) after the monotherapy of pembrolizumab.
Figure 4
Figure 4
Chest computed tomography (CT) scans. (A, E) In March 2020, multiple metastases of lung and mediastinal window were observed. (B, F) In August 2020, a PR was revealed in the lung and mediastinal window of the patient after the combined treatment of pembrolizumab and anlotinib. The patient obtained a PR (C, G) and CR (D, H) in the lung and mediastinal window after the monotherapy of pembrolizumab.
See this image and copyright information in PMC

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