Background: Arteriovenous malformations (AVM) are congenital vascular lesions and occur most commonly in the head and neck area [1]. In contrast to hemangiomas, AVM are rarities and show progressive growth and high recurrence-rates [2, 3]. Since more than 90% of the AVM are clinically significant, a treatment is usually necessary [4]. Case Report: We report about a 40-year-old male who was admitted to our clinic center suffering from a manifested and progressive AVM of the left cheek, which initial appeared about ten years ago. In addition to a strong, tough, non-movable swelling of the left cheek, typical signs like pain, overheating and telangiectasia of the affected skin were recognizable. As a result of the extensive growth of the AVM, the upper molars were dislocated and induced an ulceration of the buccal mucosa. Magnetic resonance imaging showed a large, two-parted tumor of the left face, which presented typical characteristics of an AVM in the angiography. The interventional multiple embolization with ethylene-vinyl alcohol copolymer (Onyx) could not achieve a significant reduction of the tumor mass. Therefore, the subtotal resection was performed by protecting the outer skin and the facial nerve. The defect was filled up with a free, microvascular-anastomosed latissimus dorsi muscle flap. Over an observation period of one year, the patient was still free of recurrence and satisfied with the aesthetic result. Conclusion: In cases of extensive AVM in the facial area, an interdisciplinary approach with embolization, subsequent resection, and immediate reconstruction is the goal for adequate functional and aesthetic outcomes. Subtotal resection reduces morbidity even without increasing the risk of recurrence. Keywords: AV- malformation, subtotal resection