Oral Maxillofac Surg (2014) 18:279–282
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 / Published online: 23 April 2014
# Springer-Verlag Berlin Heidelberg 2014
Abstract Severe hemorrhages of the oral cavity may be
caused by arteriovenous malformations. This case report con-
cerns 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: marcus.kriwalsky@rub.de
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 histopathologi-
cal findings [1]. Hemangiomas and vascular malformations
can be differentiated. Hemangiomas are benign tumors that
increase due to rapid mitotic cellular proliferation. They dem-
onstrate 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 symp-
toms 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 dem-
onstrate no spontaneous regression. Based on the hemody-
namic 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 symp-
toms may be mild, but they usually progress to findings such
as facial asymmetry, pain, otalgia, dental mobility, or sponta-
neous or potentially life-threatening hemorrhages around the
teeth or from the mucosa [3].
This report presents the case of a patient with life-
threatening 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
280 Oral Maxillofac Surg (2014) 18:279–282

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 Fig. 2 Orthopantomogram of the patient showing a periodontally com-
promised tooth 48
malformation prior to the extraction was categorically refused
by the patient. The preoperative diagnostics also included a
transfemoral digital subtraction angiography that revealed the
threatening bleeding after oral surgical procedures have been
right lingual and maxillary artery as feeding vessels (Fig. 3).
reported in the international literature [5–7].
After completing the preoperative diagnostics, the tooth ex-
The present case report describes a life-threatening hemor-
traction was performed under general anesthesia. After the
rhage following tooth extraction in a female patient with an
extraction, massive hemorrhaging developed that could not be
extensive arteriovenous malformation of the intraoral soft
controlled using local techniques. The massive hemorrhage
tissue (tongue, floor of the mouth).
rapidly led to hypovolemic condition and anemia, which re-
Arteriovenous malformations consist of hypertrophic tortu-
quired an immediate intravenous transfusion of packed red
ous arteries and veins that contribute to a common mass of
blood cells. These circumstances required a change in the treat-
numerous aberrant connections, which often include the con-
ment procedure. Following generous intraoral packing, a right
tralateral vessels. They may occur in isolation or together with
submandibular incision was performed, and the branches of the
syndromes such as Osler-Weber-Rendu syndrome, blue rubber
external carotid artery were identified. Initially, the right facial
bleb nevus syndrome, Parkes Weber-syndrome, Bannayan’s
artery and the right lingual artery were ligated, but the bleeding
syndrome, Sturge-Weber-Krabbe syndrome, Klippel-
did not stop. For that reason, the external carotid artery was
Trenaunay syndrome, Servelle-Martorell syndrome, or
ligated, which resulted in cessation of the intraoral hemorrhage
Maffucci syndrome [8–14]. One-third of all arteriovenous
(Fig. 4). In addition, the intraoral wound was packed with gauze.
malformations are found in the maxillofacial region and dem-
At the end of the surgical intervention, no intraoral hemorrhage
onstrate an equal gender distribution. Although malformations
was evident, and the total amount of blood loss was 4,000 ml.
are congenital in nature, they may not be detected at birth. In
The intubated patient was taken to the intensive care unit
contrast to more common, true hemangiomas, these
and could be extubated on the third postoperative day. The
malformations show an increase in size later in life [2]. This
postoperative healing course was uneventful, and no further
was also described by the patient in this case report. The sudden
bleeding was observed.
progression and subsequent recognition of arteriovenous
malformations may be observed after trauma, prior interven-
tions, hormonal changes, or spontaneous internal bleeding
Discussion

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 Fig. 3 Digital subtraction angiography showing the right lingual artery
a vascular malformation on the right side and maxillary artery as feeding vessels
Oral Maxillofac Surg (2014) 18:279–282
A Churojana et al. reviewed a series of five cases of arterio-
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 arterio-
venous 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 ap-
parent [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 find-
ings, no postoperative complications, i.e., ulcers, were ob-
served 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 appar-
ent during an uncomplicated extraction of the left lower
wisdom tooth. The intraoral bleeding could be controlled
initially with local measurements, and embolization was per-
formed immediately [18].
Carneiro et al. reported a case of a spontaneous, massive oral
hemorrhage due to laceration of a mandibular arteriovenous
281
malformation. A transarterial embolization through the right
femoral access with an injection of a 50 % cyanoacrylate
mixture was performed. After repetitive therapeutic interven-
tions, the patient progressed without bleeding [3].
venous malformations of the maxillomandibular region. They
recommended an embolization under general anesthesia. Af-
ter 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 arteriove-
nous malformation of the tongue due to considerable side
effects of the surgical intervention. They found that an embo-
lization 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 interven-
tions are not uncommon. The limitations of this recom-
mended treatment are evident in situations that demon-
strate 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, periopera-
tive morbidity may complicate the treatment. These interven-
tions require a multidisciplinary approach mandatory a hospi-
talization 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.
282
References
1. Mulliken JB, Glowacki J (1982) Hemangiomas and vascular
malformations in infants and children: a classification based on
endothelial characteristics. Plast Reconstr Surg 69:412–422
2. Kohout MP, Hansen M, Pribaz JJ, Mulliken JB (1998) Arteriovenous
malformations of the head and neck: natural history and manage-
ment. Plast Reconstr Surg 102:643–654
3. Carneiro SC, Batista LL, Vasconcelos BC, Maranhao CA, Panazzolo
G, Queiroz IV, Medeiros Junior R (2009) Massive oral hemorrhage
due to mandibular arteriovenous malformation treated with percuta-
neous approach—a case report. J Oral Maxillofac Surg 67:2525–
2528
4. Brauer HU (2009) Unusual complications associated with third molar
surgery: a systematic review. Quintessence Int 40:565–572
5. Moghadam HG, Caminiti MF (2002) Life-threatening hemorrhage
after extraction of third molars: case report and management protocol.
J Can Dent Assoc 68:670–674
6. Heiland M, Weber M, Schmelzle R (2003) Life-threatening bleeding
after dental extraction in a hemophilia A patient with inhibitors to
factor VIII: a case report. J Oral Maxillofac Surg 61:1350–1353
7. Bouloux GF, Perciaccante VJ (2009) Massive hemorrhage during
oral and maxillofacial surgery: ligation of the external carotid artery
or embolization? J Oral Maxillofac Surg 67:1547–1551
8. Gibbon WW, Pooley J (1990) Pathological fracture of the femoral
shaft in a case of Servelle-Martorell syndrome (phleboeclatic
osteohypoplastic angiodysplasia with associated arterio-venous mal-
formation). Br J Radiol 63:574–576
9. Werner JA, Geisthoff UW, Lippert BM, Rudert H (1997) Treatment of
recurrent epistaxis in Rendu-Osler-Weber disease. HNO 45:673–681
10. Greinwald JH Jr, Burke DK, Bonthius DJ, Baumann NM, Smith RJ
(1999) An update on the treatment of hemangiomas in children with
interferon alfa-2a. Arch Otolaryngol Head Neck Surg 125:21–27
11. Aydin A, Cakmakci H, Kovanlikaya A, Dirik E (2000) Sturge-Weber
syndrome without facial nevus. Pediatr Neurol 22:400–402
Oral Maxillofac Surg (2014) 18:279–282
12. Noel AA, Gloviczki P, Cherry KJ Jr, Rooke TW, Stanson AW,
Driscoll DJ (2000) Surgical treatment of venous malformations in
Klippel-Trenaunay syndrome. J Vasc Surg 32:840–847
13. Rodrigues D, Bourroul ML, Ferrer AP, Monteiro Neto H, Goncalves
ME, Cardoso SR (2000) Blue rubber bleb newus syndrome. Rev
Hosp Clin Fac Med Sao Paulo 55:29–34
14. Seckin U, Borman P, Ozoran K (2000) Clinical images: Maffucci’s
syndrome. Arthritis Rheum 43:1672
15. Saydjai R, Saydjari R, Guinto FC Jr, Wolma FJ (1990) Arteriovenous
malformation of the tongue. South Med J 83:1335–1337
16. Slaba S, Herbreteau DJ, Haveri HS, Casasco A, Aymard A, Houdart
E, Aoun N, Riche MC, Enjolras O, Merland JJ (1998) Therapeutic
approach to arteriovenous malformations of the tongue. Eur Radiol 8:
280–285
17. Richter GR, Suen J, North PE, James CA, Waner M, Buckmiller LM
(2007) Arteriovenous malformations of the tongue: a spectrum of
disease. Laryngoscope 117:328–335
18. Kluba S, Meiss A, Prey N, Ernemann U, Reinert S, Hoffmann J
(2007) Arteriovenous malformation of the mandible: life-threatening
manifestation during tooth extraction. Mund Kiefer Gesichtschir 11:
107–113
19. Churojana A, Khumtong R, Songsaeng D, Chongkolwatana C,
Suthipongchai S (2012) Life-threatening arteriovenous malformation
of the maxillomandibular region and treatment outcomes. Interv
Neuroradiol 18:49–59
20. Watzinger F, Gössweiner S, Wagner A, Richling B, Milesi-Schobel
G, Hollmann K (1997) Extensive facial vascular malformations and
haemangiomas: a review of the literature and case reports. J
Craniomaxillofac Surg 25:335–343
21. Ernemann U, Hoffmann J, Grönewäller E, Breuninger H, Rebmann
H, Adam C, Reinert S (2003) Hemangiomas and vascular
malformations in the area of the head and neck. Radiologe 43:958–
966
22. Deng W, Huang D, Chen S, Zhang X, Li X, Li J, Chen X (2010)
Management of high-flow arteriovenous malformation in the maxil-
lofacial region. J Craniofac Surg 21:916–919
Copyright of Oral & Maxillofacial Surgery is the property of Springer Science & Business
Media B.V. and its content may not be copied or emailed to multiple sites or posted to a
listserv without the copyright holder's express written permission. However, users may print,
download, or email articles for individual use.