Control # 209 Title: Life -Threatening Lytic lesion of the Mandible: A Lesson Learned eEdE# eEdE-157

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Life-Threatening Lytic lesion of the Mandible Life-Threatening Lytic lesion of the Mandible A Lesson Learned Nucharin Supakul, MD 1 Juan G Tejada, MD 2 1. Ramathibodi Hospital, Mahidol University Bangkok, Thailand 2. Indiana University School of Medicine, Eskenazi Health Indianapolis, Indiana, USA

Purpose To review the characteristic imaging findings of mandibular vascular malformations and avoid unnecessary and risky biopsies of the mandibular lesions. To demonstrate interventional treatment options for vascular lesions in the mandible in life-threatening conditions and also in the preoperative setting.

Introduction Mandibular Vascular lesions are divided in 2 groups HemangiomaHemangioma Failure of differentiation in the early stages of embryogenesis Appears in childhood and regresses over time Rarely associated with fatal hemorrhage Vascular malformationVascular malformation Disturbance in the late stage of angiogenesis (truncal stage) and results in persistence of arteriovenous anastomosis Present at birth and grows over time Symptoms depend on hemodynamic factors High flow: AVM, AVF Fatal hemorrhage Low flow: Lymphatic, venous, or mixed

Mandibular AVM Abnormal direct communication between arteries and veins, bypassing capillary bed Location:Location: Usually posterior location within the ramus and posterior mandibular body Clinical presentationClinical presentation Gingival bleeding (most common) Massive bleeding with shock following by the extraction of teeth (most common) Soft tissue mass (pulsatile/ non pulsatile), bruit, thrill Painful, alteration of facial morphology Neurosensory deficit

Imaging Findings Cystic radiolucent lesion Honeycomb (multilocular) or soap bubble appearance Resorption of the adjacent bone/ dental root teeth floating in the adjacent alveolar osseous erosion Mimics odontogenic/non-odontogenic lesions Central giant cell granuloma Ossifying fibroma Traumatic bone cyst Ameloblastoma

Imaging Findings Imaging Findings CT and MRI Evaluates the extent of the lesion Bone erosion Involvement of major vessels Catheter angiogram Gold standard in diagnosis and treatment Super-selective arteriography of the external carotid evaluates collaterals and multiple anastomoses of the Internal maxillary artery

Management Sclerosing agents (sodium morrhuate,alcohol, tetracycline etc.) ineffective most of the times Ligation of the external carotid not recommended Numerous anastomoses (internal carotid, ophthalmic, vertebral, cervical, and contralateral external carotid) and collateral vessels Limits further angiography and future embolization Direct trans osseous puncture of the vascular bed and embolization Embolization (Onyx, cyanoacrylate, polyvinyl alcohol particles, Gelfoam, coils, collagen) Pre operative embolization in acute phase then surgery within 48 hours to 2 weeks Usually multiple stages of embolization for curative results

19-MONTH-OLD FEMALE WITH LEFT LOWER GINGIVA BLEEDING

Skull AP and Towns views were performed. No demonstrable lytic lesion within the mandible is noted. This is an inappropriate study to evaluate a mandibular lesion.

MRI and MRA of the head and neck were obtained. A - B: Coronal T2 (A) and axial T1 fat suppression images show an expansile T1 iso/T2 hyperintense bony lesion within the left- sided mandible involving body, angle and ramus (orange arrows). Involvement of the left canine, left premolar and left 1 st molar teeth is noted. Several flow void signals are noted, best seen on T2 images. C - D: Post contrast T1 fat suppression in coronal (C) and axial (D) images show heterogeneous contrast enhancement and increased signal intensity with in the left masseter and let temporalis muscles. E - F: MIP MRA images of the head and neck vessels show enlargement of the left external carotid artery (blue arrows) supplying this mass (pink arrow) with early draining vein to the left external jugular vein (green arrow). FED C B A

Conventional angiogram with left external carotid artery catheterization. A B: There is a vascular blush of the mass (green arrow) within the left buccal/maxillary region supplied by branches of the left internal maxillary artery (orange arrows) and left facial artery (blue arrows) with AV shunting and venous drainage into the external jugular vein (pink arrows). C: Post PVA embolization via the left internal maxillary artery, superficial temporal, and left facial arteries with nearly complete disappearance of the vascular blush. A C B

7-YEAR-OLD MALE WITH RIGHT LOWER GINGIVA MASS WITH INTERMITTENT BLEEDING FOR A MONTH. RECENT HISTORY OF ACTIVE BLEEDING WITH SHOCK

Grossly unremarkable Panoramic radiograph of the mandible

MRI and MRA of the head and neck without and with contrast A-B: Axial T1 fat suppression (A) and T2 fat suppression (B) images show T1/T2 hyperintense expansile lesion within the body of the right-sided mandible (orange arrows). A few signal voids are noted. C-D: Post contrast axial T1 fat suppression (C) and coronal T1 fat suppression (D) images show heterogeneous contrast enhancement within this mass (orange arrows). D-E: Contrasted MRA images show dilation of the right facial vein (blue arrow) and external carotid artery (pink arrows), related to a feeding artery. F: MIP image shows dilation of the right external carotid artery (pink arrow) with no visualized drain vein. G F E D C A B

D C B A Conventional angiogram with right external carotid artery catheterization A-B: Lateral and AP images show abnormal vascular blush with arterial supply from the right facial (orange arrows) and right internal maxillary arteries (blue arrows) and early draining vein to the right external jugular vein (pink arrow). C-D: Post embolization images after gelfoam and NBCA injection show complete occlusion of the mandibular AVM.

12-YEAR-OLD MALE WITH LOWER GINGIVA BLEEDING

CT head and neck with contrast A-B: Axial and coronal CT without contrast at the level of the mandible show well- defined lytic lesion within the posterior body of the right mandible associated with tooth root resorption (orange arrows). C-D: Axial post contrast images show avid enhancement within this lesion (blue arrow) associated with enlargement of the right external carotid artery and right facial artery. Findings are suggestive of AVM. A B C D

Conventional angiogram with right external carotid artery catheterization A-D: AP and lateral images show abnormal vascular blush with arterial supply from the right inferior alveolar (orange arrows) and right facial arteries (blue arrows). Drainage to the right facial vein (pink arrow) is noted. There is a large venous pouch in the right mandibular body (green arrow). E-F: Post embolization images with NBCA demonstrate residual venous pouch (green arrow) and vascular blush lesion. Patient was scheduled for second stage embolization within a month. F E D B C A

Second stage embolization with NBCA Residual small AVM was treated with NBCA. Post embolization angiogram shows marked decreased flow of the AVM and increased venous stagnation.

15-YEAR-OLD MALE WITH VASCULAR MASS FOUND ON DENTAL PROCEDURE

15-year-old boy with intra-oral vascular mass identified incidentally during a dental procedure. A: Axial CT image shows a lytic lesion within the posterior body of the right mandible (orange arrow). B: Axial T1 post contrast image shows an enhancing vascular mass in the right mandibular body (blue arrow). C: Doppler US demonstrates an AVM in the right mandibular body draining into a dilated varix (green star). D: DSA lateral image from the right external carotid artery injection shows an AVM supplied by branches of the facial and internal maxillary arteries (pink arrow) with venous drainage predominantly to the right external jugular vein. E F: Post-embolization lateral images of the right external carotid artery injection show approximately 80% occlusion of the mandibular AVM (yellow arrow = residual AVM).

Conclusion extremely rare potentially life-threatening conditions presenting with intractable hemorrhage after tooth extractions or biopsies.Vascular malformations of the mandible are extremely rare potentially life-threatening conditions presenting with intractable hemorrhage after tooth extractions or biopsies. Occur predominantly during childhood with a variety of symptoms including gingival bleeding, bruit, dental loosening, swelling of the soft tissues of the face, discoloration of the skin and mucosa and sometime neurosensory deficits. Think about vascular AVM in case of gingival bleeding/ lesion in the posterior body of the mandible. Cross-sectional imaging especially CT and MRI with contrast are useful imaging modalities for clarifying the extent of the lesion, the degree of bone erosion, and involvement of major vesselsCross-sectional imaging especially CT and MRI with contrast are useful imaging modalities for clarifying the extent of the lesion, the degree of bone erosion, and involvement of major vessels (feeding arteries and draining veins). Radiologist should be able to recognize the imaging patterns to avoid risky and unnecessary biopsies and suggest prompt treatment in case of life-threatening hemorrhage or the need for preoperative treatment with endovascular or percutaneous embolization

References 1.Scholl, Robert J., et al. "Cysts and Cystic Lesions of the Mandible: Clinical and Radiologic-Histopathologic Review 1." Radiographics 19.5 (1999): 1107-1124. 2.Singh V, Bhardwaj PK. Arteriovenous malformation of mandible: Extracorporeal curettage with immediate replantation technique. Natl J Maxillofac Surg. 2010 Jan- Jun; 1(1): 4549. 3.Kiyosue, Hiro, et al. "Treatment of mandibular arteriovenous malformation by transvenous embolization: a case report." Head & neck 21.6 (1999): 574-577. 4.Noreau, Gatan, Pierre-. Landry, and Dany Morais. "Arteriovenous malformation of the mandible: review of literature and case history." Journal-Canadian Dental Association 67.11 (2001): 646-651. 5.A. Churojana, R. Khumtong, D. Songsaeng, C. Chongkolwatana, and S. Suthipongcha,. Life-Threatening Arteriovenous Malformation of the Maxillomandibular Region and Treatment Outcomes. Interv Neuroradiol. 2012 Mar; 18(1): 4959.

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Control # 209 Title: Life -Threatening Lytic lesion of the Mandible: A Lesson Learned eEdE# eEdE-157