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Mandibular central leiomyosarcoma with high telomerase activity: a case report
Tomoki Sumida1, Akiko Ishikawa2, Hiroyuki Nakano1
1Department of Oral and Maxillofacial Surgery, Kyushu University Fukuoka, Japan.
International Journal of Clinical and Experimental Pathology
|January 16, 2020
Summary
This case report details an extremely rare intraosseous leiomyosarcoma of the mandible in a young man. Despite initial treatment, the aggressive tumor recurred and metastasized, leading to a fatal outcome.
Area of Science:
- Oral pathology
- Surgical oncology
- Oncologic imaging
Background:
- Leiomyosarcoma, a malignant smooth muscle tumor, is exceptionally rare in the oral region.
- Intraosseous tumors of the mandible require prompt diagnosis and management due to potential for rapid destruction.
Observation:
- A 29-year-old male presented with left mandibular pain and paresthesia, with imaging revealing osteolytic destruction and cortical perforation.
- Biopsy confirmed a non-epithelial malignant tumor, leading to surgical resection and reconstruction.
Findings:
- Histopathological analysis of the surgical specimen confirmed leiomyosarcoma, characterized by hyperchromatic spindle cells positive for smooth muscle markers (α-SMA, calponin, HHF35, desmin).
- Immunohistochemistry ruled out epithelial and neural origins (S-100, EMA, cytokeratin negative).
Implications:
- This case highlights the diagnostic challenges and aggressive nature of intraosseous leiomyosarcoma of the mandible.
- Despite surgical intervention and adjuvant chemotherapy, the patient experienced recurrence and metastasis, underscoring the need for novel therapeutic strategies for rare oral malignancies.
