Phleboliths, not Sialoliths: A Report of Submandibular Gland Arteriovenous Malformation with Numerous Calcifications: Analysis of Cine Images and Literature Review for the 54 Years

Abstract

Here, we provide a case report of a 28-year-old woman diagnosed with arteriovenous malformation (AVM) of the submandibular gland (SMG). A 14 phleboliths were the visualized on the multi-slice computed tomography (MSCT) within the AVM and two calcifications were located 7-mm distant from AVM margins. Such AVMs and venous malformations are so-called in the literature as tumor-like vascular formations or “hemangiomas.” Ultrasonography (USG) and non-/post-contrast MSCT, which helped to make a correct pre-operative diagnosis, are presented. In total, in this report the 129 MSCT images are cinematically demonstrated. Also, the present case is enhanced by the pre-/intraoperative images, photographs of the specimen, removed phleboliths, and multiple histopathological images. Based on the literature review for the last 54 years and present case, the 19 cases with SMG malformations/hemangiomas were reported in 18 patients which were published in 15 articles. In one patient, a bilateral SMG venous malformation was reported. At the same time, our case report is a first case study of the SMG AVM with phleboliths that highlights this rare pathology in three videos (as cine loops). Advantages of integration of the cine images into case studies are analyzed. Also, the “submandibular gland-arteriovenous malformation conglomerate” was proposed by our team as a term for description of similar cases.