Capillary hemangioma of the maxilla

A report of two cases in which angiography

and embolization

were used

Lynn A. Greene, DDSa Paul D. Freedman, DDS,b Joel hf. Friedman, DDS, and
Merwin Wolx DDS,d Bronx and New York, N.Y.
ALBERT EINSTEIN COLLEGE OF MEDICINE, MONTEFIORE MEDICAL CENTER, AND BOOTH
MEMORIAL HOSPITAL
This article presents two clinical cases of capillary hemangiomas of the maxilla. Such lesions are rare, as
demonstrated by the review of the literature included in this article. The presentation, differential diagnosis,
histopathology,
management, and follow-up for each case are discussed. Our rationale for approaching
these types of lesions, as well as our opinion that microembolization
should be considered as a first line
approach to treatment, is presented.
(ORAL SURC ORAL MED O&u, PATHOL 1990;70:268-73)

emangiomas of the jawbone are quite rare. A

comprehensive review of the literature by GamezAraujo and his associates2in 1974 identified 50 central hemangiomas of the mandible and even fewer in
the maxilla. Although these tumors are benign, they
should be approached with great caution as demonstrated by numerous reports of morbidity and
mortality.35
There is tremendous variation in the features of
central hemangiomas of the jawbone. Becauseof this
variation, no pathognomonic sign exists that can be
used to make a definite clinical diagnosis of hemangioma. In addition, confusion with other conditions
can occur since hemangiomas may mimic other
lesions clinically, radiographically, and in somecases
histologically. Among the clinical features that may
be present are pain, spontaneoushemorrhage, asymmetry of the face, mobility of teeth, pulsation, blanching of tissue, expansion of bone, paresthesia, early exfoliation of primary teeth, delayed eruption, missing
teeth, and root resorption. Some central hemangiomas may be entirely asymptomatic.

aAssistant Professor,Albert Einstein College of Medicine, Montefiore Medical Center, Bronx.

bAssistant Director, Section of Oral Pathology, Booth Memorial
Hospital, New York.
CAssociateClinical Professor, Deputy Director of Oral and Maxillofacial Surgery, Albert Einstein College of Medicine, Montefiore
Medical Center, Bronx.
dAssociateProfessor,Director of Dentistry, Weiler Hospital of The
Albert Einstein College of Medicine, Montefiore Medical Center,
Bronx.
7/12/12428
266

The two most common types of hemangioma are

the capillary and the cavernous forms. The capillary
hemangioma is composedof small fine blood vessels
filled with blood. In contrast, the cavernous hemangioma is formed by large thin-walled vessels or sinusoids lined with a single layer of endothelium.7
Treatment modalities vary considerably depending
on the extent and the type of the hemangioma. The
range of treatment includes steroid therapy,* carbon
dioxide9 or argon laserlo therapy, sclerosing
agents, l, l2 surgical excision with or without ligation
of vessels,13and embolization.143ls Angiograms can
be invaluable in the management of hemangiomas by
defining the vascular supply of the tumor and by
identifying the pathway for the embolization of feeder
vessels.
This report describes the features, diagnosis, and
treatment of two casesof capillary hemangioma of the
maxilla with extension into the overlying gingiva.
CASE REPORTS
CASE 1

A well-nourished, well-developed lo-year-old Hispanic

girl came to our clinic in November 1983 with a chief complaint of a slowly enlarging massin the right maxillary gingiva that was slightly painful (Fig. 1). According to the patient and her mother, the lesion, which bled spontaneously
on occasion,was first noticed 3 months before presentation,
There was no history of trauma or previous surgery. The
medical history was essentially negative.
Physical examination showed a lo-year-old girl in good
general health. On the gingiva, there was a red, raised,
somewhat irregularly shapedmassextending from the right
maxillary first premolar to the distal of the first molar. The
massinvolved the buccal surface of the alveolus, as well as

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Number 3

Fig.

1. Initial appearance of right maxillary lesion.

269

Fig. 3. Photomicrograph demonstrating ulcerated stratified squamous epithelium with unencapsulated tumor
composed of numerous thin-walled capillary channels in
edematous stroma.

Fig. 2. Periapical radiograph of area of right maxillary

lesion.
Fig. 4. Photomicrograph demonstrating capillaries with
single layer of uniform endothelial cells.

approximately half the depth of the palatal aspectof the alveolus. In the middle of the mass,at the height of the lesion,
was a white, raised exophytic structure that remarkably resembledthe crown of a primary molar, although it consisted
entirely of soft tissue. There was no facial asymmetry, bruit,
thrill, or lymphadenopathy. Occlusal and periapical radiographs demonstrated an increase in the trabecular density
of the alveolar bone (Fig. 2). Panoramic views showed that
there was no secondpremolar toothbud evident in the right
maxilla. In addition, an increase in the anteroposterior dimension when compared to the contralateral segment was
noted.
With the patient under local anesthesia and nitrous oxide/oxygen analgesia, an incisional biopsy was performed.
During the procedure, an unexpected amount of hemorrhage was encountered. The patient was discharged to home
with routine postoperative instructions. The pathology
report obtained from the hospital pathologist stated that the
diagnosis was consistent with pyogenic granuloma. On
the basis of the diagnosis, the patient was scheduled for excision of the mass under general anesthesia. This was
accomplished in December 1983. Hemorrhage was anticipated becauseof the experience at incisional biopsy, so an

acrylic stent was constructed preoperatively. During surgery, the stent was filled with microfibrillar collagen and
placed under pressure. Hemorrhage was controlled by this
method with a total blood loss of 450 ml. The immediate
postoperative course was uneventful and the patient was
discharged in satisfactory condition. The specimen was
again submitted to the general pathology department,
which confirmed the original diagnosis of pyogenic granuloma.
The postoperative course was notable for the rapid
recurrence of the mass, which clinically resembled the
original lesion. Becauseof the unusual rapidity with which
the lesion recurred, representative slides of the biopsy specimen and surgical excision were sent for consultation to an
oral pathologist.
The consultant pathologist stated that the tissue submitted revealed ulcerated stratified squamousepithelium overlying an unencapsulated tumor composed of numerous
thin-walled capillary channels set in an edematous fibrous
stroma (Fig. 3). The capillaries were lined with a single
layer of uniform endothelial cells (Fig. 4). Scattered

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Greene et al.

ORALSURGORALMEDORAL PATHOL
September1990

Fig.

Fig. 5. Photomicrograph demonstrating cancellous bone

trabeculae separated by vascular tissue identical to overlying soft tissue.

Fig.

7. Blush (capillary) phase of angiogram.

6. Angiogram delineating vascular supply to lesion.

throughout the lesional stroma were sparseclusters of lymphocytes and plasma cells. At the baseof the excision, cancellous bone trabeculea were noted; these were separatedby
vascular tissue that was identical to and in continuity with
the overlying soft tissue lesion (Fig. 5). No cortical bone was
evident. The diagnosis was capillary hemangioma of bone
with extension into overlying soft tissue.
Becauseof our clinical impression and the diagnosis from
the oral pathologist, we pursued the casefurther by obtaining angiographic studies. These studies demonstrated the
exaggerated vascular supply to the tumor via the right descending palatine artery and an impressive blush phase
(Figs. 6 and 7). The blush phasewas consistent with the slow
flow characteristics of a capillary lesion. This confirmed the
microscopic diagnosis of capillary hemangioma.
Because of the relative ease of superselective catheterization during angiography, it was decided that embolization was a viable treatment alternative. Embolization of the
terminal one third of the internal maxillary artery was accomplished with polyvinyl alcohol, a nonresorbable material. During recovery, the patient demonstrated only mod-

Figs.

8 and 9. Initial presentation of left anterior max-

illary lesion.
erate bilateral ecchymosesat the sites of the femoral artery
catheterization. In the immediate postoperative period,
there was a noticeable decreasein the hemorrhagic nature
of the mass.One week after embolization, 1.5 ml of sodium
morrhuate 1.5%was injected into the lesion. This modality
was used to preclude peripheral revascularization. At 3
weeksafter embolization, the lesion was again sclerosed.At
4 weeks,evidenceof sloughing of the surrounding tissue was
seen.One week later, the patient had a 2.0 X 1.5 X 0.5 cm
segmentof necrotic bone wrapped in a tissue. No oroantral

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Number 3

Fig.

Fig. 10. Radiographic appearance of left antedor maxilla with diffuse trabecular pattern changes.

fistula was seen,and the surrounding tissuesappearedto be

within normal limits. The area proceeded to heal by
secondary intention. A small buccal hemorrhagic area was
sclerosed 3 weeks later. At 24 months after treatment, the
area of the lesion appeared clinically normal. There has
been no subsequenthemorrhage or other evidence of recurrence.
CASE 2

A well-nourished, well-developed lCyear-old Hispanic

girl undergoing orthodontic therapy was referred for spontaneous bleeding and hypertrophied gingiva in the anterior
portion of the right maxilla. The medical history was noncontributory. Physical examination revealed a healthy girl
who demonstrated a hemorrhagic, slightly raised mass of
the maxillary anterior labial and palatal attached gingiva,
which blanched on pressure. The lesion extended from the
midline to the right first premolar area (Figs. 8 and 9). In
addition, the right central, lateral, and cuspid teeth exhibited separation when compared with the left side. Radiographic examination revealed a distinct change in the trabecular pattern of the right anterior maxilla as compared to
the unaffected contralateral side (Fig. 10).
The biopsy specimen from case2 showed soft tissue very
similar in appearance to the soft tissue seen in case 1. No

11. Angiogram of left anterior maxillary lesion.

bone was noted in the specimen.The diagnosis rendered by

the oral pathologist at that time was suggestive of pyogenic
granuloma with a recommendation to evaluate the patient
to rule our capillary hemangioma becauseof the amount of
vascularity, the lack of ulceration, and the sparse inflammatory infiltrate.
Symptoms continued to increase in severity with more
frequent spontaneous hemorrhage and epistaxis from the
right nostril. For delineation of the vascular supply, the patient underwent angiography with superselective catheterization. This revealed that the major feeding vesselwas the
descendingpalatine artery (Fig. 11). The distal one third of
the internal maxillary artery was sucessfully embolized with
polyvinyl alcohol.
During the immediate postoperative period, the lesion no
longer blanched on pressure and was noticeably paler in
color. Approximately 3 months after the embolization,
there was an increase in the rednessof the lesion, but there
was no spontaneousor easily elicited hemorrhage. This recurrence occurred despite postembolization sclerosing therapy. At this time, it was decided to sclerosethe lesion again.
The sclerosing therapy involved three applications of 0.8 to
1.0 ml of 1.5% of sodium morrhuate at 2-week intervals.
After this therapy, the appearance of the lesional area resembled the adjacent normal mucosa with the exception of
small areasof redness.Thirty-eight months after treatment,
there has been no bleeding or epistaxis. In addition,
orthodontic therapy was reinstituted and completed without
complication. The teeth in the involved area have decreased
in mobility, but portions of the gingiva in the previously involved area have remained slightly more erythematous than
the surrounding mucosa.Close observation has continued in
concert with the neuroradiologist because reembolization
may be necessaryif the lesion doesnot continue to improve.
DISCUSSION

The treatment of hemangiomas of bone varies considerably depending on the clinical features and the
anatomic considerations. We chose to treat these two
cases by embolization and sclerosing agents for the
following reasons:
1. Neither lesion was life threatening in nature on

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Greene et al.

ORAL SURC ORAL MED ORAL PATNOL

September 1990

presentation and therefore did not require immediate

surgical control of hemorrhage.
2. Angiography was of importance in delineation of
the vascular supply and confirmation of the histologic
diagnosis. Since at the time of angiography the neuroradiologist achieved accessto each lesions vascular
supply, microembolization becamean attractive treatment alternative.
3. Since our two caseswould have required ligation
of the external carotid artery for the control of surgical hemorrhage, microembolization offered a distinct
advantage. With the use of microembolization, no ligation was necessary.
4. The effectivenessof microembolization is due to
the fact that the embolizing material lodges within
small vesselsin the depth of the lesion, producing irreversible tissue necrosis. This makes recanulization
or revascularization unlikely.
5. Microembolization is a procedure that may be
repeated with a high index of success.
6. With the use of the microembolization technique, the patients retain intact jaws and dentition.
7. Ablative surgery can still be performed if necessary.
SUMMARY

We have presented two casesof intraoral capillary

hemangiomas of bone with soft tissue extension. The
various treatment modalities have beenreviewed. Our
reasoning, treatment protocol, and results have been
presented. We believe that microembolization is a viable treatment alternative and should be considered
as a first line therapy in similar vascular tumors.
REFERENCES

1. Shklar G, Meyers 1. Vascular tumors of the mouth and jaws.

ORAL SURGURAL MED ORAL PATHOL1965;19:335-57:
2. Gamez-Arauio JJ. Toth BB. Luna MA. Central hemangioma
of the mandible and maxilla: review of vascular lesions. ORAL
SURGORAL MED ORAL PATHOL1974;37:230-8.
Wilde NJ, et al. Hemangioma of the mandible: report of a case.
J Oral Surg 1966;24:549-52.
Macansh JD, Owen MD. Central cavernous hemangioma of
the mandible: report of case. J Oral Surg 1972;30:293-7.
Shira RB, Guernsey LH. Central cavernous hemangioma of
the mandible: report of a case. J Oral Surg 1965;23:636-42.
Sadowsky D, Rosenberg RD, Kaufman J, Levine BC, Friedman JM. Central hemangioma of the mandible. ORAL SURG
ORAL MED ORAL PATHOL1981;52:471-7.
7. Shafer MG, Hine MK, Levy BM. A textbook of oral pathology. 4th ed. Philadelphia: WB Saunders, 1983:154-257.
8. Edgerton MT. The treatment of hemangiomas with special
reference to the role of steroids. Ann Surg 1976;183:517-30.
9. Shafir R, Slutski S, Bornstein LA. Excision of buccal hemangioma by carbon dioxide laser beam. ORAL SURGORAL MED
ORAL PATHOL1977;44:347-50.
10. Apfelberg DB, Greene RA, Maser MR, Lash H, Rivers JL,
Laub DR. Results of argon laser exposure of capillary hemangiomas of infancy-preliminary report. Plast Reconstr Surg
1981;67:188-92.
11. Chin DC. Treatment of maxillary hemangioma with scleros-

ing agent. ORAL SURGORAL MED ORAL PATHOL 1983;55:

247-9.

12. Morgan JF, Schow E. Use of sodium morrhuate in the management of hemangiomas. J Oral Surg 1974;32:363-6.
13. Shira RB, Guernsey LH. Central cavernous hemangioma of
the mandible: report of case. J Oral Surg 1965;23:636-42.
14. Hoey MF, et al. Management of vascular malformations of the
mandible and maxilla: review and report of two casestreated
by embolization and surgical obliteration. J Oral Surg 1970;
281696-706.

15. Longacre JJ, Benton C, Unterthiner RA. Treatment of facial

hemangioma by intravascular embolization with silicone
spheres. Plast Reconstr Surg 1972;50:618-21.
16. Sachatello CR. McSwain B. Regressionof cutaneouscapillary
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hemangioma. km J Surg 1968yll6: 113-4.
17. Zou Z, Wu Y, Sun Q, Zhu X, Meng X, He Z. Clinical application of angiography of oral and maxillofacial hemangiomas.
ORAL SURGORAL MED ORAL PATHOL1983;55:437-47.
18. Berenstein A, Kricheff II. Microembolization techniques of
vascular occlusion: radiologic, pathologic, and clinical correlation. AJNR 1981;2:261-7.
19. Braun IF, Levy S, Hoffman JC. The use of transarterial microembolization in the management of hemangiomas of the
perioral region. J Oral Surg 1985;43:239-48.
Reprint requests to.

Dr. Lynn A. Greene

Weiler Hospital of the Albert Einstein College of Medicine
1825 Eastchester Rd., Suite 23-65
Bronx, NY 10461
ADDENDUM

Since the completion to this article there have been

new and interesting occurences.Patient 2 returned to
our clinic nearly 5 years after her initial therapy. She
had moved from the area after successfully completing orthodontic therapy.
On presentation, there was clear clinical evidence
of recurrence. The patient traces the first sign of recurrence to about 18 to 20 months before her return.
Clinically, spacing of the maxillary left anterior teeth
and a markedly inferior position of the teeth compared with the unaffected right side were observed.
She also had occasional mild epistaxis. As previously,
there was no palpable bruit or pulse at the affected
area.
The patient underwent diagnostic angiography,
which wasconsistent with capillary hemangioma. The
vascular supply was the same as that of the original
lesion, leading to the conclusion that there had been
recanolization of the lesion. This has been described
by Berenstein.
A second and serendipitous finding was that of an
atriovenous malformation of the left facial artery.
There were minimal clinical signs and symptomsthe temperature slightly increased, and the patient
stated that she had had a vague awarenessin the left
submandibular region. She had attributed it to the
maxillary lesion.
Taking into consideration the two vascular lesions
now presented, we decided first to use superselective
embolization in the left internal maxillary artery as
had been previously done. Ligation of the left exter-

Capillary hemangioma of maxilla

Volume 70
Number 3

nal carotid artery was performed to addressthe facial

artery anomaly; of course, this also included the
internal maxillary artery. A conservative resection of
the bony lesion of the anterior maxilla was undertaken. At the time of surgery we believed that the incisors could be retained becausealveolar support was
adequate (endodontic therapy had been done preoperatively).
The bony defect, which extended to the nasal mucosa, was packed with hydroxyapatite. The mucosa
was closed routinely. The patient tolerated the procedures well and was discharged in good condition.
Postoperatively, the patient demonstrated a marginal

BOUND VOLUMES AVAILABLE

273

mandibular nerve weaknessand a hypoglossal nerve

deficit. Neuropraxia is the most plausible explanation
at this early date.
Certainly, this case appears to demonstrate recanolization after superselective embolization. We chose
to intervene with a surgical approach because the
maxillary lesion had once failed embolization and the
atriovenous malformation of the facial artery required such intervention.
At this time, only weeks after surgery, all appears
well. As clearly demonstrated by this case,only time
will provide the final answer.

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