Oral Maxillofac Surg DOI 10.1007/s10006-014-0448-3

REVIEW ARTICLE

Life-threatening bleeding after tooth extraction due to vascular malformation: a case report and literature review Marcus Stephan Kriwalsky & Dimitrios Papadimas & Peter Maurer & Martin Brinkmann & Jochen Jackowski & Martin Kunkel

Received: 5 December 2013 / Accepted: 7 April 2014 # Springer-Verlag Berlin Heidelberg 2014

Abstract Severe hemorrhages of the oral cavity may be caused by arteriovenous malformations. This case report concerns a 52-year-old healthy female who presented with a painful lower third molar and an extensive arteriovenous high-flow malformation of the floor of the right side of the mouth. During the extraction of the right lower wisdom tooth, an episode of massive life-threatening bleeding occurred. Since the therapy for intraoral arteriovenous malformations of the soft tissue is complex and often difficult to perform, the modus operandi of the present case is presented, and a review of the literature is included. Keywords Life-threatening bleeding . Hemorrhage . Tooth extraction . Arteriovenous malformation

Introduction Angiodysplastic maxillofacial lesions are rarely encountered in clinical practice. They were classified according to M. S. Kriwalsky (*) : D. Papadimas : M. Kunkel Department of Oral and Plastic Maxillofacial Surgery, Ruhr-University Bochum, In der Schornau 23-25, 44892 Bochum, Germany e-mail: [email protected] P. Maurer Private Practice of Oral and Maxillofacial Surgery, Wendalinusstr. 5-7, 66606 St. Wendel, Germany M. Brinkmann Department of Radiology and Nuclear Medicine, Ruhr-University, In der Schornau 23-25, 44892 Bochum, Germany J. Jackowski Department of Oral Surgery, University of Witten/ Herdecke, Alfred-Herrhausen-Str. 45, 58455 Witten, Germany

Mulliken and Glowacki based on clinical and histopathological findings [1]. Hemangiomas and vascular malformations can be differentiated. Hemangiomas are benign tumors that increase due to rapid mitotic cellular proliferation. They demonstrate a typical and well-defined life cycle of proliferation and involution. Vascular malformations are present at the time of birth but may not be clinically evident [2]. The first symptoms of angiodysplastic lesions may occur during childhood or puberty and are less frequent later in life. Vascular malformations are characterized by the quantity of endothelial cells and structural abnormities of the vessels (arteries, veins, lymphatic vessels, or a combination of these) [1]. They demonstrate no spontaneous regression. Based on the hemodynamic criteria, “high-flow” and “low-flow” malformations can be distinguished. “High-flow” malformations are lesions that demonstrate an increased blood flow rate due to collateral vessel formation and arteriovenous shunts. The initial symptoms may be mild, but they usually progress to findings such as facial asymmetry, pain, otalgia, dental mobility, or spontaneous or potentially life-threatening hemorrhages around the teeth or from the mucosa [3]. This report presents the case of a patient with lifethreatening bleeding after wisdom tooth extraction due to an adjacent arteriovenous malformation, and it evaluates the international literature on this subject.

Case report A 52-year-old otherwise healthy female patient was referred by an oral surgeon for removal of the decayed right lower wisdom tooth to prevent the development of an abscess. Initially, the referring dentist was thinking about extracting the tooth under outpatient conditions but finally decided to refer the patient to the hospital. Since birth, an extensive arteriovenous malformation of the right tongue, floor of the

Oral Maxillofac Surg

mouth, and lower lip was present. She underwent radiotherapy of the lesion during childhood, but she was not able to provide further information. Intraorally, a deeply decayed tooth 48 and an extensive arteriovenous malformation of the tongue, floor of the mouth, lower lip, and buccal region on the right side of her face was found. The malformation was a hyperemic, superficially soft, compressible mass with a speckled bluish discoloration (Fig. 1). An intraosseous lesion of the mandible could not be detected (Fig. 2). The proposed treatment for the arteriovenous malformation prior to the extraction was categorically refused by the patient. The preoperative diagnostics also included a transfemoral digital subtraction angiography that revealed the right lingual and maxillary artery as feeding vessels (Fig. 3). After completing the preoperative diagnostics, the tooth extraction was performed under general anesthesia. After the extraction, massive hemorrhaging developed that could not be controlled using local techniques. The massive hemorrhage rapidly led to hypovolemic condition and anemia, which required an immediate intravenous transfusion of packed red blood cells. These circumstances required a change in the treatment procedure. Following generous intraoral packing, a right submandibular incision was performed, and the branches of the external carotid artery were identified. Initially, the right facial artery and the right lingual artery were ligated, but the bleeding did not stop. For that reason, the external carotid artery was ligated, which resulted in cessation of the intraoral hemorrhage (Fig. 4). In addition, the intraoral wound was packed with gauze. At the end of the surgical intervention, no intraoral hemorrhage was evident, and the total amount of blood loss was 4,000 ml. The intubated patient was taken to the intensive care unit and could be extubated on the third postoperative day. The postoperative healing course was uneventful, and no further bleeding was observed.

Discussion

Fig. 2 Orthopantomogram of the patient showing a periodontally compromised tooth 48

threatening bleeding after oral surgical procedures have been reported in the international literature [5–7]. The present case report describes a life-threatening hemorrhage following tooth extraction in a female patient with an extensive arteriovenous malformation of the intraoral soft tissue (tongue, floor of the mouth). Arteriovenous malformations consist of hypertrophic tortuous arteries and veins that contribute to a common mass of numerous aberrant connections, which often include the contralateral vessels. They may occur in isolation or together with syndromes such as Osler-Weber-Rendu syndrome, blue rubber bleb nevus syndrome, Parkes Weber-syndrome, Bannayan’s syndrome, Sturge-Weber-Krabbe syndrome, KlippelTrenaunay syndrome, Servelle-Martorell syndrome, or Maffucci syndrome [8–14]. One-third of all arteriovenous malformations are found in the maxillofacial region and demonstrate an equal gender distribution. Although malformations are congenital in nature, they may not be detected at birth. In contrast to more common, true hemangiomas, these malformations show an increase in size later in life [2]. This was also described by the patient in this case report. The sudden progression and subsequent recognition of arteriovenous malformations may be observed after trauma, prior interventions, hormonal changes, or spontaneous internal bleeding

Life-threatening bleeding after wisdom tooth removal is a very rare complication [4]. A few case reports of life-

Fig. 1 Intraoral view prior to treatment, the tongue of the patient showing a vascular malformation on the right side

Fig. 3 Digital subtraction angiography showing the right lingual artery and maxillary artery as feeding vessels

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A

B

Fig. 4 Intraoperative view after the ligation of the feeding vessels on the right side of the neck. a Common carotid artery. b External carotid artery (ligated)

[15–17]. Without intervention, these lesions can develop to extremely large sizes and infiltrate the adjacent tissues, resulting in an esthetic and functional impairment. Clinical symptoms may include palpable pulsation, a spongy texture, warmth, a compressible soft tissue mass with brisk vascular rebound, and a bluish discoloration of the mucosa. The lesion can be detected clinically, radiographically, or histologically. Few international publications concerning lingual arteriovenous malformations were found. Malformations involving the tongue or the floor of the mouth were found to be diffuse and spongy with rebound and indefinable boundaries, and often, bilateral feeding arteries (lingual and facial) were apparent [15–17]. Bouloux and Perciaccante described a similar case in which an upper third molar was surgically removed in a patient with a vascular malformation. After the uneventful removal, severe bleeding occurred that could not be controlled by repetitive embolizations of the maxillary artery, ascending pharyngeal artery, and facial artery. Therefore, the external carotid artery was ligated using a cervical approach. This procedure stopped the intraoral hemorrhage. The authors evaluated the ligation of the external carotid artery in cases of severe oral bleeding. The authors clearly identified a great benefit of this procedure in emergencies and observed no postoperative complications. In accordance with those findings, no postoperative complications, i.e., ulcers, were observed in the present case [7]. Kluba et al. treated an arteriovenous malformation of the left mandible and the adjacent soft tissue with repeated transfemoral embolization. The vascular lesion became apparent during an uncomplicated extraction of the left lower wisdom tooth. The intraoral bleeding could be controlled initially with local measurements, and embolization was performed immediately [18]. Carneiro et al. reported a case of a spontaneous, massive oral hemorrhage due to laceration of a mandibular arteriovenous

malformation. A transarterial embolization through the right femoral access with an injection of a 50 % cyanoacrylate mixture was performed. After repetitive therapeutic interventions, the patient progressed without bleeding [3]. Churojana et al. reviewed a series of five cases of arteriovenous malformations of the maxillomandibular region. They recommended an embolization under general anesthesia. After that, oral surgery was accomplished. In one case, two embolizations were necessary. Another case developed an osteonecrosis of the mandible after multiple embolizations during 7 years [19]. Slaba et al. recommended a “wait and see strategy” for clinically asymptomatic patients with an extensive arteriovenous malformation of the tongue due to considerable side effects of the surgical intervention. They found that an embolization alone was sufficient in 10 out 12 treated patients; in two patients, surgical intervention was necessary due to prolonged bleeding [16]. In general, a multidisciplinary approach that includes embolization and complete surgical removal of the nidus within 48 to 72 h is regarded as the treatment of choice although recurrences that necessitate repetitive interventions are not uncommon. The limitations of this recommended treatment are evident in situations that demonstrate unclear boundaries, including vital anatomical structures, extensive expansion of the malformation or in growing patients. For that reason, the management of arteriovenous malformations and their complications is patient-specific in almost every case [20–22]. Due to the emergency situation in the present case, the recommended therapeutic strategy could not be followed. A multidisciplinary curative approach was strongly advised to the patient prior to the tooth extraction, but she vehemently refused this procedure.

Conclusion In the present case, an arteriovenous malformation of the mouth caused a routine dental extraction to result in a nearly lethal situation. Although such lesions are rare, clinicians who perform this type of surgery should be aware of the potential associated risks. A multidisciplinary approach that includes a superselective embolization and surgery after 2 days should be regarded as the treatment of choice. Although the diagnosis of such lesions is easy, perioperative morbidity may complicate the treatment. These interventions require a multidisciplinary approach mandatory a hospitalization to react adequately to complications. In the present case, a very fast response to the complication was possible, because the tooth extraction was performed under general anesthesia.

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Life-threatening bleeding after tooth extraction due to vascular malformation: a case report and literature review.

Severe hemorrhages of the oral cavity may be caused by arteriovenous malformations. This case report concerns a 52-year-old healthy female who present...
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