Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2020 Mar;14(1):199-202.
doi: 10.1007/s12105-019-01017-1. Epub 2019 Feb 13.

Vascular Floor of Mouth Mass with Unanticipated Intracranial, Orbital, and Vertebral Associated Involvements

Affiliations
Case Reports

Vascular Floor of Mouth Mass with Unanticipated Intracranial, Orbital, and Vertebral Associated Involvements

Ho-Hyun Brian Sun et al. Head Neck Pathol. 2020 Mar.

Abstract

We report on a case in which a blanching, unobtrusive oral growth proved to be a systemic threat. A blind, epileptic child presented with a bleeding oral floor mass of 4 weeks. Biopsy showed small, dilated vascular spaces with reactive fibroblasts. MRI indicated distribution of expansile lesions in the mandible, cranial base, and right orbit that had possibly contributed to the patient's years-long neurologic deficits. A subsequent bone scan indicated lesions in multiple axial bones. Histologic markers confirmed the presentation of a rare cystic vascular pathology. Cystic Angiomatosis is a disease of intraosseous vascular malformations with occasional visceral involvements. Oral and craniomaxillofacial cases are especially rare and presentations can involve neuropsychiatric deficits, sensory issues, and mucosal bleeding. While clinicians are often dismissive of intraoral bleeding because of the prevalence of periodontal disease, careful evaluation is nonetheless critical to rule out underlying diseases with a possibly significant systemic involvement.

Keywords: Blindness; Cystic angiomatosis; Epilepsy; Vascular neoplasm.

PubMed Disclaimer

Conflict of interest statement

The authors declare that they have no conflict of interest.

Figures

Fig. 1
Fig. 1
Smooth, soft, immobile, and pink mass of the right floor of the mouth without overt surface changes
Fig. 2
Fig. 2
Multicystic, gadolinium contrast-enhancing lesion occupying almost the entirety of the intracortical space in the right mandible
Fig. 3
Fig. 3
Variously sized cystic masses in the right maxillary alveolus and sinus, orbital bones, and the cranial chamber with displacement of neuronal tissue

Similar articles

References

    1. Najm A, Soltner-Neel E, Le Goff B, Guillot P, Maugars Y, Berthelot J-M. Cystic angiomatosis, a heterogeneous condition: four new cases and a literature review. Medicine. 2016;95:e5213. doi: 10.1097/MD.0000000000005213. - DOI - PMC - PubMed
    1. Hossein Mortazavi S, Khodayari A, Khojasteh A, Abbas FM, Mehrdad L, Kiani MT, et al. Massive craniofacial intraosseous vascular malformation resembling cystic angiomatosis: report of 2 cases and review of the literature. J Oral Maxillofac Surg. 2011;69:204–214. doi: 10.1016/j.joms.2010.07.072. - DOI - PubMed
    1. Beeram RK, Seelam S. Massive craniofacial cystic angiomatosis and extra-skeletal angiomatosis: a unique case report and brief review of literature. J Oral Maxillofac Surg, Med, Pathol. 2016;28:122–127. doi: 10.1016/j.ajoms.2015.08.008. - DOI
    1. Shivaram GM, Pai RK, Ireland KB, Stevens KJ. Temporal progression of skeletal cystic angiomatosis. Skelet Radiol. 2007;36:1199–1204. doi: 10.1007/s00256-007-0378-3. - DOI - PubMed
    1. Bauschard MJ, Hatch JL, Liogier-Weyback LER, Vandergrift WA, McRackan TR, Zheng X, Bi C, Marissa Brooks and DSH Intracranial complications from temporal bone cystic angiomatosis. Otol Neurotol. 2017;38:e1. doi: 10.1097/MAO.0000000000001294. - DOI - PMC - PubMed

Publication types

LinkOut - more resources