MANDEL AND PERRINO
malignant behavior. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 93:287, 2002
16. Goldenberg D, Sciubba J, Koch W, et al: Malignant odonto-
genic tumors: A 22-year experience. Laryngoscope 114:1770,
2004
17. Kawano K, Ono K, Yada N, et al: Malignant calcifying epithelial
odontogenic tumor of the mandible: Report of a case with
pulmonary metastasis showing remarkable response to plati-
num derivatives. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 104:76, 2007
18. Kumar M, Fasanmade A, Barrett AW, et al: Metastasising clear
cell odontogenic carcinoma: A case report and review of the
literature. Oral Oncol 39:190, 2003
19. Veness MJ, Morgan G, Collins AP, et al: Calcifying epithelial
odontogenic (Pindborg) tumor with malignant transformation
and metastatic spread. Head Neck 23:692, 2001
20. Sciubba JJ, Eversole LR, Slootweg PJ: Odontogenic/ameloblas-
tic carcinomas, in Barnes L, Eveson J, Reichart P, et al (eds):
World Health Organization Classification of Tumours, Pathol-
Phleboliths and the Vascular
Maxillofacial Lesion
Louis Mandel, DDS,* and Michael A. Perrino, DDS†
Phleboliths are calcified thrombi found within vascu-
lar channels (Fig 1), often in the presence of heman-
giomas or vascular malformations (VMs). They may
originate from injury to a vessel wall or result from
stagnation of the flow of blood.1 The injury can result
in damage to the intima. Healing involves formation of
a protective thrombus that then may calcify as part of
the healing process. Slowing with stagnation of blood
flow also favors thrombus formation, and with calci-
fication of the thrombus, a phlebolith evolves.
The phlebolith consists of a mixture of calcium
carbonate and calcium phosphate salts,2,3 and it has
either a radiolucent or radiopaque core. A fibrous
component attaches itself to the developing phlebo-
lith, and in turn, it becomes calcified.3 Repetition of
the process causes a layering effect such that the
*Director, Salivary Gland Center, and Associate Dean and Clinical
Professor, Department of Oral and Maxillofacial Surgery, Columbia
University College of Dental Medicine, New York, NY.
†Resident, Department of Oral and Maxillofacial Surgery, New
York–Presbyterian Hospital (Columbia Campus), New York, NY.
Address correspondence and reprint requests to Dr Mandel:
Columbia University College of Dental Medicine, 630 W 168th St,
New York, NY 10032; e-mail: lm7@columbia.edu
© 2010 American Association of Oral and Maxillofacial Surgeons
0278-2391/10/6808-0037$36.00/0
doi:10.1016/j.joms.2010.04.002
1973
ogy and Genetics of Head and Neck Tumors. Lyon, IARC Press,
2005, p 287
21. Bang G, Koppang H: Clear cell odontogenic carcinoma, in
Barnes L, Eveson J, Reichart P, et al (eds): World Health Orga-
nization Classification of Tumours, Pathology and Genetics of
Head and Neck Tumors. Lyon, IARC Press, 2005, p 292
22. Smith B, Mulligan J, Wagner E: Ameloblastic carcinoma and
relationships with ameloblastoma. Oral Surg Oral Med Oral
Pathol Oral Radiol Endod 108:526, 2004
23. Gerdes J: Ki-67 and other proliferation markers useful for im-
munohistological diagnostic and prognostic evaluations in hu-
man malignancies. Semin Cancer Biol 1:199, 1990
24. Gorgoulis VG, Vassiliou LV, Karakaidos P, et al: Activation of
the DNA damage checkpoint and genomic instability in human
precancerous lesions. Nature 434:907, 2005
25. Harris CC: p53 tumor suppressor gene: From the basic research
laboratory to the clinic—An abridged historical perspective.
Carcinogenesis 17:1187, 1996
26. Tsantoulis PK, Kastrinakis NG, Tourvas AD, et al: Advances in
the biology of oral cancer. Oral Oncol 43:523, 2007
J Oral Maxillofac Surg
68:1973-1976, 2010
phlebolith usually has a concentric ring or onion-like
appearance, which suggests a growth mechanism.
Phleboliths are incidental findings that by them-
selves cause no subjective symptoms, and they may
be found during routine imaging or during studies of
vascular lesions. In the head and neck area, they are
usually multiple, varied in size, randomly distributed,
and associated with either intramuscular hemangio-
mas (IMHs) or VMs. However, the diagnosis of a head
or neck phlebolith requires its differentiation from
other calcifications that occur in the same area. Sialo-
liths, tonsilloliths, calcified lymph nodes, atheroscle-
rotic plaques in the carotid artery, healed acne le-
sions, cysticercosis, and miliary skin osteomas all
serve to muddy the diagnostic waters.
We wish to illustrate the signs and symptoms of
phlebolithiasis by presenting 2 case reports involving
the head and neck area. The first case depicts the
presence of a phlebolith in an IMH, and the second
case represents an example of phlebolith formation in
a VM.
Report of Cases
CASE 1
A 56-year-old woman in excellent health was referred in
1982 to the Columbia University College of Dental Medi-
cine’s Salivary Gland Center (New York, NY) because of a
tentative diagnosis of right parotid swelling whose etiology
was unknown.
1974
FIGURE 1. Calcified structure (phlebolith) occupying lumen of
arteriole (hematoxylin-eosin stain, original magnification !40).
Mandel and Perrino. Phleboliths and Vascular Maxillofacial Le-
sion. J Oral Maxillofac Surg 2010.
For the previous 2 years, the patient had been aware of a
painless swelling of the right side of her face, and this
prompted her decision to seek medical attention. Question-
ing indicated that the swelling’s exact duration was un-
known, and it did not fluctuate in size during meals.
Extraorally, a somewhat diffuse and indistinct right facial
swelling was visible, occupying the buccal soft tissue area
(Fig 2). There was a slight erythematous appearance to the
overlying skin. Palpation indicated that the swelling was not
well circumscribed, it was soft and painless, and no pulsa-
tions were noted. Close scrutiny showed that the swelling
clearly involved the right buccal tissues anterior to a pain-
less parotid gland. There was no cervical lymphadenopathy.
Intraorally, the right buccal mucosa was slightly dis-
tended and had a bluish tinge. It was painless when pal-
pated. A normal clear salivary volume was observed exiting
from the right parotid duct when the right parotid gland
was aggressively massaged.
Because the relationship of the swelling to the surround-
ing anatomic structures was not certain, sialography of the
right parotid gland was performed (Fig 3). A normal right
FIGURE 2. Moderate swelling in case 1 (right side) causing facial
asymmetry.
Mandel and Perrino. Phleboliths and Vascular Maxillofacial Le-
sion. J Oral Maxillofac Surg 2010.
PHLEBOLITHS AND VASCULAR MAXILLOFACIAL LESION
FIGURE 3. Right parotid sialogram in case 1. Phleboliths (arrows)
are present anterior to the parotid and signal the existence of the
IMH.
Mandel and Perrino. Phleboliths and Vascular Maxillofacial Le-
sion. J Oral Maxillofac Surg 2010.
parotid sialogram clearly ruled out the gland as a source of
the patient’s swelling. However, the presence of several
lamellated opacities, anterior to the gland, signified the
existence of phleboliths. Combined with the information
derived from the history and clinical examination, a diag-
nosis of an IMH involving the buccinator and masseter
muscle area was made.
CASE 2
A 64-year-old woman with no medical problems was
referred because of a large blue-purple growth that involved
the entire right tongue. She stated that the tongue swelling
and discoloration had been present since birth. The lesion
had grown as she aged. Although the lesion was painless, its
continued growth had interfered with her speech pattern.
Furthermore, a tendency to traumatize the mass during
mastication had caused concern because of the excessive
bleeding that ensued.
Intraorally, a gross and startling disfiguring blue-purple
swelling was evident involving the entire right side of the
tongue with a spilling over to the left tongue dorsum and
right mouth floor (Fig 4). Palpation indicated that the
growth was painless and moderately firm and had no dis-
tinguishable borders. An occlusal film showed the existence
of several phleboliths, thus confirming what was obviously
a vascular lesion (Fig 5). The history and clinical appearance
of the lesion supported a diagnosis of a VM.
Discussion
Phleboliths are most frequently found in the pelvic
veins, where they are subject to stress injury during
defecation.1 Of 1,555 consecutive pelvic radiographs,
39% showed the presence of a phlebolith.1 The next
most common site for a phlebolith is the head and
neck area, where they are found in 15% to 25% of
IMHs.4-6 The IMH represents only 1% of all hemangio-
mas, but 15% of these involve the head and neck re-
MANDEL AND PERRINO 1975

mellated calcifications that, with the patient’s history

and clinical findings, clearly pointed to a diagnosis of an
IMH. Alternatively to radiography, a computed tomog-
raphy scan will distinctly illustrate the calcified phlebo-
lith, whereas magnetic resonance imaging, with T2
weighting, will show the hyperintensity of the heman-
gioma. Computed tomography and magnetic resonance
imaging have largely replaced sialography as the pre-
ferred imaging procedure.
Our case 2 lesion is a classic VM. The VM repre-
sents a congenital lesion, clinically present at birth, as
in our patient, and it becomes larger in proportion to
the patient’s growth throughout life.9,10,18,19 The VM
is a structural abnormality and results from errors in
FIGURE 4. VM of tongue in case 2. blood vessel morphogenesis.7,20 Forty percent of VMs
Mandel and Perrino. Phleboliths and Vascular Maxillofacial Le- are found in the head and neck area.19 Occasionally,
sion. J Oral Maxillofac Surg 2010. a phlebolith will develop,19 but no data are available
regarding its incidence. Histologically, the VM usually
contains arterial, venous, and lymphatic elements,
gion.5,6 The masseter and/or buccinator muscles repre- and it is lined by mature endothelial cells that show a
sent the location of 36% of head and neck IMHs.7,8 normal mitotic rate.9,10,18 A predominantly venous
The seminal work of Mulliken and Glowacki9,10 VM is a low-flow lesion, whereas the arteriovenous
defined the hemangioma as a benign neoplasm, the VM flows at a high rate. Because of the indolent
vast majority of which are first noticed at birth or in nature of the blood flow, the venous variety of VM
early infancy. An initial rapid growth phase is fol- more commonly develops a phlebolith.19
lowed by a period of involution that is usually com-
pleted by 12 years of age, when it is replaced by a
Treatment
fibrofatty infiltration.7-13 The IMH is an uncommon
neoplastic vascular lesion that differs from the neo- The therapeutic approach to the IMH and VM is
plastic hemangioma of infancy in that it manifests dependent on a variety of clinical factors. Patient age,
itself in the later decades of life.6,12,14-16 After a period rapidity of growth, size and location, cosmetics, and
of long asymptomatic dormancy, growth acceleration subjective symptoms all must be considered in the
develops such that the signs and symptoms of the
IMH’s presence become evident in adult life. Sponta-
neous regression does not occur.12,16,17 Trauma may
serve as the inciting stimulus.8,12,14-16 It is thought
that the tumor evolves from an abnormally differenti-
ated and proliferating mitotic endothelial cell net-
work.5,15,17 Pulsations and bruits are not apparent
because the lesion is embedded in the surrounding
musculature. Pain can be present, but it is the devel-
oping cosmetic swelling that usually serves as the
driving force for medical care.
Because the IMH represents a mass of tortuous
blood vessels with a sluggish flow of blood, stasis
develops. Platelet aggregation and subsequent throm-
bosis with calcification result and succeed in forming
a phlebolith. It is also possible that the location of our
first patient’s IMH made it susceptible, during masti-
cation, to vascular stretching and distension, which
can lead to vessel wall damage with the creation of a
thrombus and subsequent calcification.1
Diagnosis of an IMH in our first patient was FIGURE 5. Occlusal radiograph in case 2 showing presence of
achieved when the clinical picture of a neoplastic-like multiple phleboliths in VM of tongue.
swelling was factored in with the results of sialo- Mandel and Perrino. Phleboliths and Vascular Maxillofacial Le-
graphic imaging. The radiography readily showed la- sion. J Oral Maxillofac Surg 2010.
1976
therapeutic decision making. Sclerosing agents, ste-
roids, radiotherapy, lasers, cryotherapy, embolization,
and even continued observation represent reasonable
approaches to the vascular aspect of the lesion.6,7
Nevertheless, when feasible, surgical excision is the
treatment method of choice and will also succeed in
eliminating the phlebolith.
Our patient with the IMH opted for no treatment
because she stated that other than a moderate cos-
metic asymmetry, she had no subjective problems.
She did agree to return every 6 months for evaluation,
but she failed to present for further follow-up.
In our patient with the VM, significant symptoms
had developed that prompted her to seek therapy.
Normal masticatory function inevitably caused trauma
with associated hemorrhage. Furthermore, she be-
came increasingly embarrassed about her speech im-
pediment. She was referred to an interventional radi-
ologist who plans to embolize the lesion.
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J Oral Maxillofac Surg
68:1976-1982, 2010
Cervicofacial Subcutaneous Emphysema:
Case Report and Review of Literature
Nishul Patel, DMD,* Stewart K. Lazow, MD, DDS,† and
Julius Berger, DDS‡
Received from Department of Oral and Maxillofacial Surgery, Kings
County Hospital, Brooklyn, NY.
*Chief Resident.
†Director of Service.
‡Chief of Service.
Address correspondence and reprint requests to Dr Patel: De-
partment of Oral and Maxillofacial Surgery, Kings County Hospital,
451 Clarkson Avenue, E-Building, Brooklyn, NY 11203; e-mail:
NishulPatel@Yahoo.com
© 2010 American Association of Oral and Maxillofacial Surgeons
0278-2391/10/6808-0038$36.00/0
doi:10.1016/j.joms.2010.02.018
CERVICOFACIAL SUBCUTANEOUS EMPHYSEMA
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Subcutaneous emphysema is by no means a contem-
porary discovery, having been mentioned more than
180 years ago. Previously described as pneumomedi-
astinum, Laenec, in 1827, termed the disease “inter-
lobar emphysema.”1 Reports described subcutaneous
emphysema in a patient with a violent coughing epi-
sode in 1850.2 The first official report by an anony-
mous investigator of the phenomenon stemmed from
a musician playing the bugle after a dental extraction
at the beginning of the 20th century.3 Macklin in 1939
and 1944 proposed a pathophysiologic clarification,2
which led to the rudimentary 4-tier classification ac-
cording to the etiology of cervicofacial emphysema in
1957, derived by Shovelton. The various etiologies of