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Intraosseous epithelioid haemangioendothelioma of the mandible: A case report and literature review.
Medicine (Baltimore). 2019 Jul; 98(30):e16572.M

Abstract

INTRODUCTION

Epithelioid 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 CONCERNS

A 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.

DIAGNOSIS

Enhanced 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.

INTERVENTIONS

Surgical resection was performed via segmental mandibulectomy and the defect was reconstructed using a vascularized fibular flap.

OUTCOMES

The patient did not exhibit signs of locoregional recurrence after 3 years of follow-up.

CONCLUSIONS

Intraosseous 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.

Authors+Show Affiliations

Department of Stomatology, The Second Affiliated Hospital of Nanchang University, Nanchang, Jiangxi. Department of Stomatology, Huangshi Central Hospital, Huangshi, Hubei.Department of Stomatology, The Second Affiliated Hospital of Nanchang University, Nanchang, Jiangxi.Department of Stomatology, The Second Affiliated Hospital of Nanchang University, Nanchang, Jiangxi.Department of Stomatology, The Second Affiliated Hospital of Nanchang University, Nanchang, Jiangxi. State Key Laboratory Breeding Base of Basic Science of Stomatology (Hubei -MOST) and Key Laboratory of Oral Biomedicine Ministry of Education, School and Hospital of Stomatology, Wuhan University, Wuhan, China.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

31348287

Citation

Yang, Haili, et al. "Intraosseous Epithelioid Haemangioendothelioma of the Mandible: a Case Report and Literature Review." Medicine, vol. 98, no. 30, 2019, pp. e16572.
Yang H, Wang J, Song L, et al. Intraosseous epithelioid haemangioendothelioma of the mandible: A case report and literature review. Medicine (Baltimore). 2019;98(30):e16572.
Yang, H., Wang, J., Song, L., & Zou, H. (2019). Intraosseous epithelioid haemangioendothelioma of the mandible: A case report and literature review. Medicine, 98(30), e16572. https://doi.org/10.1097/MD.0000000000016572
Yang H, et al. Intraosseous Epithelioid Haemangioendothelioma of the Mandible: a Case Report and Literature Review. Medicine (Baltimore). 2019;98(30):e16572. PubMed PMID: 31348287.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Intraosseous epithelioid haemangioendothelioma of the mandible: A case report and literature review. AU - Yang,Haili, AU - Wang,Jun, AU - Song,Li, AU - Zou,Haixiao, PY - 2019/7/27/entrez PY - 2019/7/28/pubmed PY - 2019/8/14/medline SP - e16572 EP - e16572 JF - Medicine JO - Medicine (Baltimore) VL - 98 IS - 30 N2 - INTRODUCTION: Epithelioid 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 CONCERNS: A 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. DIAGNOSIS: Enhanced 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. INTERVENTIONS: Surgical resection was performed via segmental mandibulectomy and the defect was reconstructed using a vascularized fibular flap. OUTCOMES: The patient did not exhibit signs of locoregional recurrence after 3 years of follow-up. CONCLUSIONS: Intraosseous 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. SN - 1536-5964 UR - https://www.unboundmedicine.com/medline/citation/31348287/Intraosseous_epithelioid_haemangioendothelioma_of_the_mandible:_A_case_report_and_literature_review L2 - http://dx.doi.org/10.1097/MD.0000000000016572 DB - PRIME DP - Unbound Medicine ER -