• Haili Yang, Jun Wang, Li Song, and Haixiao Zou.
• Department of Stomatology, The Second Affiliated Hospital of Nanchang University, Nanchang, Jiangxi.
• Medicine (Baltimore). 2019 Jul 1; 98 (30): e16572.

IntroductionEpithelioid haemangioendothelioma (EHE) is a rare vascular neoplasm that primarily occurs in the soft tissues of the extremities, with "intermediate" or "borderline" malignant biological characteristics and a propensity for recurrence and metastasis. Oral cavity involvement is extremely rare, with only 5 reported cases of intraosseous EHE of the mandible. Herein, we report an additional case and review the related literature.Patient ConcernsA 44-year-old man was referred to our institution with a 2-month history of left lower lip numbness and a 2-week history of painful swelling of the left mandible. He had received antibiotic treatment, which failed to ameliorate his symptoms. His medical and family histories were not significant.DiagnosisEnhanced computed tomography revealed a poorly defined, 4.5 × 3.5 × 1.0 cm osteolytic lesion in the angle and ramus of the mandible, as well as an eroded lingual cortex. Histopathological examination confirmed a diagnosis of EHE.InterventionsSurgical resection was performed via segmental mandibulectomy and the defect was reconstructed using a vascularized fibular flap.OutcomesThe patient did not exhibit signs of locoregional recurrence after 3 years of follow-up.ConclusionsIntraosseous EHE of the mandible is an unpredictable lesion with a relatively benign course; however, the few reported cases of this disease do not highlight any factors that can predict the risk of locoregional recurrence or metastasis. There is also no consensus regarding the optimal treatment for intraosseous EHE; however, we recommend extensive local excision with close clinical follow-up.

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