Primary intraosseous ACC of mandible of possible salivary origin: A rare clinical entity

Zhen H Ren; Daminee Chickooree; Jin B Liu; Han J Wu

doi:10.1016/j.ijscr.2014.01.024

Primary intraosseous ACC of mandible of possible salivary origin: A rare clinical entity

Int J Surg Case Rep. 2014;5(5):222-5. doi: 10.1016/j.ijscr.2014.01.024. Epub 2014 Mar 13.

Authors

Zhen H Ren¹, Daminee Chickooree¹, Jin B Liu¹, Han J Wu²

Affiliations

¹ Department of Oral and Maxillofacial Surgery, The Second Xiangya Hospital of Central South University, Renmin Road, No. 139, Changsha, Hunan 410011, China.
² Department of Oral and Maxillofacial Surgery, The Second Xiangya Hospital of Central South University, Renmin Road, No. 139, Changsha, Hunan 410011, China. Electronic address: drwuhj@hotmail.com.

Abstract

Introduction: Adenoid cystic carcinoma (ACC) is a malignant tumor mainly of salivary origin which is well known for its deceptively benign histologic appearance characterized by indolent, locally invasive growth with high propensity for local recurrence and distant metastasis.

Presentation of case: An unusual case of a 23-year-old woman was reported in our hospital. After investigations, it showed that it is a second primary intraosseous lesion of mandible that occurred subsequently after ACC of parotid gland. After diagnosis was established, resection of tumor and reconstruction with a free fibula flap was performed. Ten months follow-up showed no signs of recurrence or metastasis.

Discussion: Among the salivary neoplasms, adenoid cystic carcinoma is very rare and intraosseous lesions are even rarer. We found a total of 26 cases of primary ACC of the mandible reported in the literature. Pain and swelling were the most frequent symptoms.

Conclusion: This case illustrates two key facts. First, not all cystic lesions are necessarily metastatic or recurrence. Second is, even though the exact origin of this tumor is unknown, central salivary gland tumors should be considered in the differential diagnosis of cystic lytic lesions in the mandible.

Keywords: Fibula osteocutaneous flap; Intraosseous ACC; Mandible; Parotid gland.