International Journal of Clinical Oral and Maxillofacial Surgery

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Low Grade Myofibroblastic Sarcoma of the Oral Cavity: A Case Report and Review of the Literature

Received: Apr. 26, 2022    Accepted: May 10, 2022    Published: Aug. 10, 2022
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Abstract

Low-grade myofibroblastic sarcoma (LGMS) is a rare malignant neoplasm of the soft tissue with a predilection for the head and neck. To date, there are no optimal treatment strategies. This report describes a case of LGMS of the left side retromolar pad region in a 26-year-old otherwise healthy woman with initial presentation of a two-month history of a painless and progressively growing soft tissue mass. Incisional biopsy was completed revealing “fragments of granulation tissue with marked acute and chronic inflammation, and fibrosis”. Based on history and physical examination, the decision was made to complete an excisional biopsy in the operating room due to the location of the lesion and the proximity of the lingual nerve. Pathology report then revealed “cellular myofibroblastic neoplasm, favor low grade myofibroblastic sarcoma”. TNM staging was completed based on NCCN guidelines with subsequent imaging. The treatment plan included marginal resection of the left posterior mandible via trans-oral incisions with oncological margins, resection of lingual nerve with subsequent allogenic nerve grafting, extraction of left mandibular first and second molar, and complex primary closure. The patient is planned to have a secondary reconstruction via anterior iliac crest bone graft completed with simultaneous implant placement. We discuss the differential diagnosis, clinical, histological and therapeutic features of LGMS as it is understood in the literature.

DOI 10.11648/j.ijcoms.20220802.11
Published in International Journal of Clinical Oral and Maxillofacial Surgery ( Volume 8, Issue 2, December 2022 )
Page(s) 16-20
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2024. Published by Science Publishing Group

Keywords

Low Grade Myofibroblastic Sarcoma, Oral Cavity, Head and Neck

References
[1] Ghosh A, Bandopadhyay A, Sarkar R. Low-grade myofibroblastic sarcoma of maxillary sinus and buccal mucosa: Two rare cases and review of the literature. Indian J Pathol Microbiol 2019; 62: 119-21.
[2] Covello R, Licci S, Pichi B, Spriano G, Vidiri A, Morelli L and Rosenberg AE: Low-grade myofibroblastic sarcoma of the larynx. Int J Surg Pathol 2011; 19: 822-26.
[3] Mentzel T, Dry S, Katenkamp D, Fletcher CD. Low-grade myofibroblastic sarcoma: analysis of 18 cases in the spectrum of myofibroblastic tumors. Am J Surg Pathol 1998; 22: 1228–38.
[4] Maruyama T, Nakasone T, Nimura F, Matayoshi A, Kawano T, Nishihara K and Arasaki A. Indolent growth of low-grade myofibroblastic sarcoma of the cheek mimics benign lesions: A case report and literature review. Oncology Letters 2017; 13: 4307-14.
[5] Leitner C, Hoffman J, Kröber S, Reinert S. Low-grade malignant fibrosarcoma of the dental follicle of an untrusted third molar without clinical evidence of any follicular lesion. Journal of Craniofacial-Maxillofacial Surgery 2007; 35: 48-51.
[6] Xu Y, Xu G, Wang X, Mao M, Wu H, Baklaushev VP, Chekhonin VP, Peltzer K, Wang G, Zhang C. Is there a role for chemotherapy and radiation in the treatment of patients with low-grade myofibroblastic sarcoma? Clin Transl Oncol. 2020 Jun 30.
[7] Suit HD, Mankin HJ, Wood WC, Gebhardt MC, Harmon DC, Rosenberg A, Tepper JE, Rosenthal D. Treatment of patients with stage M0 soft tissue sarcomas. Dept of Radiation Medicine, MGH cancer center, Harvard Medical School, Boston.
[8] Gabbiani G, Ryan GB, Majno G. Presence of modified fibroblasts in granulation tissue and their possible role in wound contraction. Experientia. 1971; 27: 549-50.
[9] NCCN Guidlines for Physicians: Soft Tissue Sarcoma 2020.
[10] Wang, L., Li, LX., Chen, DQ. et al. Low-grade Myofibroblastic sarcoma: clinical and imaging findings. BMC Med Imaging 19, 36 (2019). https://doi.org/10.1186/s12880-018-0287-z
[11] Xu Y, Xu G, Wang X, Mao M, Wu H, Baklaushev VP, Chekhonin VP, Peltzer K, Wang G, Zhang C. Is there a role for chemotherapy and radiation in the treatment of patients with low-grade myofibroblastic sarcoma? Clin Transl Oncol. 2021 Feb; 23 (2): 344-352. doi: 10.1007/s12094-020-02425-4. Epub 2020 Jun 30. PMID: 32607812.
[12] Julien Hamon, Amélie Bourkis and Guy Le Toux. Low-grade myofibroblastic sarcoma: a case report of a child. Julien Hamon, Amélie Bourkis and Guy Le Toux. J Oral Med Oral Surg, 25 3 (2019) 27. DOI: https://doi.org/10.1051/mbcb/2019012
[13] Wu X, Guo L, Li S, Zheng Y, Fan B, Zhou C. Low-grade myofibroblastic sarcoma with abdominal pain, a stuffy nose, hearing loss, and multiple cavity effusion: a case report and literature review. Journal of International Medical Research. January 2020. doi: 10.1177/0300060519895661.
[14] Baranov, E., & Hornick, J. L. (2020). Soft Tissue Special Issue: Fibroblastic and Myofibroblastic Neoplasms of the Head and Neck. Head and neck pathology, 14 (1), 43–58. https://doi.org/10.1007/s12105-019-01104-3
[15] Kim JH, Choi W, Cho HS, Lee KS, Park JK, Kim BK. Surgical treatment and long-term outcomes of low-grade myofibroblastic sarcoma: a single-center case series of 15 patients. World J Surg Oncol. 2021; 19 (1): 339. Published 2021 Dec 7. doi: 10.1186/s12957-021-02454-5.
Cite This Article
  • APA Style

    Justin MacRae, Ramiz Mogannam, Hillel Ephros, Amer Akmal. (2022). Low Grade Myofibroblastic Sarcoma of the Oral Cavity: A Case Report and Review of the Literature. International Journal of Clinical Oral and Maxillofacial Surgery, 8(2), 16-20. https://doi.org/10.11648/j.ijcoms.20220802.11

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    ACS Style

    Justin MacRae; Ramiz Mogannam; Hillel Ephros; Amer Akmal. Low Grade Myofibroblastic Sarcoma of the Oral Cavity: A Case Report and Review of the Literature. Int. J. Clin. Oral Maxillofac. Surg. 2022, 8(2), 16-20. doi: 10.11648/j.ijcoms.20220802.11

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    AMA Style

    Justin MacRae, Ramiz Mogannam, Hillel Ephros, Amer Akmal. Low Grade Myofibroblastic Sarcoma of the Oral Cavity: A Case Report and Review of the Literature. Int J Clin Oral Maxillofac Surg. 2022;8(2):16-20. doi: 10.11648/j.ijcoms.20220802.11

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  • @article{10.11648/j.ijcoms.20220802.11,
      author = {Justin MacRae and Ramiz Mogannam and Hillel Ephros and Amer Akmal},
      title = {Low Grade Myofibroblastic Sarcoma of the Oral Cavity: A Case Report and Review of the Literature},
      journal = {International Journal of Clinical Oral and Maxillofacial Surgery},
      volume = {8},
      number = {2},
      pages = {16-20},
      doi = {10.11648/j.ijcoms.20220802.11},
      url = {https://doi.org/10.11648/j.ijcoms.20220802.11},
      eprint = {https://download.sciencepg.com/pdf/10.11648.j.ijcoms.20220802.11},
      abstract = {Low-grade myofibroblastic sarcoma (LGMS) is a rare malignant neoplasm of the soft tissue with a predilection for the head and neck. To date, there are no optimal treatment strategies. This report describes a case of LGMS of the left side retromolar pad region in a 26-year-old otherwise healthy woman with initial presentation of a two-month history of a painless and progressively growing soft tissue mass. Incisional biopsy was completed revealing “fragments of granulation tissue with marked acute and chronic inflammation, and fibrosis”. Based on history and physical examination, the decision was made to complete an excisional biopsy in the operating room due to the location of the lesion and the proximity of the lingual nerve. Pathology report then revealed “cellular myofibroblastic neoplasm, favor low grade myofibroblastic sarcoma”. TNM staging was completed based on NCCN guidelines with subsequent imaging. The treatment plan included marginal resection of the left posterior mandible via trans-oral incisions with oncological margins, resection of lingual nerve with subsequent allogenic nerve grafting, extraction of left mandibular first and second molar, and complex primary closure. The patient is planned to have a secondary reconstruction via anterior iliac crest bone graft completed with simultaneous implant placement. We discuss the differential diagnosis, clinical, histological and therapeutic features of LGMS as it is understood in the literature.},
     year = {2022}
    }
    

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    AU  - Justin MacRae
    AU  - Ramiz Mogannam
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    JO  - International Journal of Clinical Oral and Maxillofacial Surgery
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    AB  - Low-grade myofibroblastic sarcoma (LGMS) is a rare malignant neoplasm of the soft tissue with a predilection for the head and neck. To date, there are no optimal treatment strategies. This report describes a case of LGMS of the left side retromolar pad region in a 26-year-old otherwise healthy woman with initial presentation of a two-month history of a painless and progressively growing soft tissue mass. Incisional biopsy was completed revealing “fragments of granulation tissue with marked acute and chronic inflammation, and fibrosis”. Based on history and physical examination, the decision was made to complete an excisional biopsy in the operating room due to the location of the lesion and the proximity of the lingual nerve. Pathology report then revealed “cellular myofibroblastic neoplasm, favor low grade myofibroblastic sarcoma”. TNM staging was completed based on NCCN guidelines with subsequent imaging. The treatment plan included marginal resection of the left posterior mandible via trans-oral incisions with oncological margins, resection of lingual nerve with subsequent allogenic nerve grafting, extraction of left mandibular first and second molar, and complex primary closure. The patient is planned to have a secondary reconstruction via anterior iliac crest bone graft completed with simultaneous implant placement. We discuss the differential diagnosis, clinical, histological and therapeutic features of LGMS as it is understood in the literature.
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Author Information
  • Department of Oral and Maxillofacial Surgery, St. Joseph’s University Medical Center, Paterson, USA

  • Department of Oral and Maxillofacial Surgery, St. Joseph’s University Medical Center, Paterson, USA

  • Department of Oral and Maxillofacial Surgery, St. Joseph’s University Medical Center, Paterson, USA

  • Department of Pathology, St. Joseph’s University Medical Center, Paterson, USA

  • Section