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)

H emangiomas of the jawbone are quite rare.’ A comprehensive review of the literature by Gamez- Araujo and his associates2 in 1974 identified 50 cen- tral hemangiomas of the mandible and even fewer in the maxilla. Although these tumors are benign, they should be approached with great caution as demon- strated by numerous reports of morbidity and mortality.3‘5

There is tremendous variation in the features of central hemangiomas of the jawbone. Because of this variation, no pathognomonic sign exists that can be used to make a definite clinical diagnosis of heman- gioma. In addition, confusion with other conditions can occur since hemangiomas may mimic other lesions clinically, radiographically, and in some cases histologically. Among the clinical features that may be present are pain, spontaneous hemorrhage, asym- metry of the face, mobility of teeth, pulsation, blanch- ing of tissue, expansion of bone, paresthesia, early ex- foliation of primary teeth, delayed eruption, missing teeth, and root resorption. Some central hemangio- mas may be entirely asymptomatic.

aAssistant Professor, Albert Einstein College of Medicine, Monte- fiore Medical Center, Bronx. bAssistant Director, Section of Oral Pathology, Booth Memorial Hospital, New York. CAssociate Clinical Professor, Deputy Director of Oral and Max- illofacial Surgery, Albert Einstein College of Medicine, Montefiore Medical Center, Bronx. dAssociate Professor, Director of Dentistry, Weiler Hospital of The Albert Einstein College of Medicine, Montefiore Medical Center, Bronx. 7/12/12428

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The two most common types of hemangioma are the capillary and the cavernous forms. The capillary hemangioma is composed of small fine blood vessels filled with blood. In contrast, the cavernous hemangi- oma is formed by large thin-walled vessels or sinu- soids 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,13 and embolization.143 ls 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 cases of 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 com- plaint of a slowly enlarging mass in the right maxillary gin- giva that was slightly painful (Fig. 1). According to the pa- tient 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 shaped mass extending from the right maxillary first premolar to the distal of the first molar. The mass involved the buccal surface of the alveolus, as well as

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Fig. 1. Initial appearance of right maxillary lesion. Fig. 3. Photomicrograph demonstrating ulcerated strat- ified 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 aspect of the al- veolus. In the middle of the mass, at the height of the lesion, was a white, raised exophytic structure that remarkably re- sembled the crown of a primary molar, although it consisted entirely of soft tissue. There was no facial asymmetry, bruit, thrill, or lymphadenopathy. Occlusal and periapical radio- graphs demonstrated an increase in the trabecular density of the alveolar bone (Fig. 2). Panoramic views showed that there was no second premolar toothbud evident in the right maxilla. In addition, an increase in the anteroposterior di- mension when compared to the contralateral segment was noted.

With the patient under local anesthesia and nitrous ox- ide/oxygen analgesia, an incisional biopsy was performed. During the procedure, an unexpected amount of hemor- rhage 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 ex- cision of the mass under general anesthesia. This was accomplished in December 1983. Hemorrhage was antici- pated because of the experience at incisional biopsy, so an

acrylic stent was constructed preoperatively. During sur- gery, 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 granu- loma.

The postoperative course was notable for the rapid recurrence of the mass, which clinically resembled the original lesion. Because of the unusual rapidity with which the lesion recurred, representative slides of the biopsy spec- imen and surgical excision were sent for consultation to an oral pathologist.

The consultant pathologist stated that the tissue submit- ted revealed ulcerated stratified squamous epithelium over- lying 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

270 Greene et al. ORAL SURG ORAL MED ORAL PATHOL September 1990

Fig. 5. Photomicrograph demonstrating cancellous bone trabeculae separated by vascular tissue identical to overly- ing soft tissue.

Fig. 6. Angiogram delineating vascular supply to lesion.

throughout the lesional stroma were sparse clusters of lym- phocytes and plasma cells. At the base of the excision, can- cellous bone trabeculea were noted; these were separated by 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.

Because of our clinical impression and the diagnosis from the oral pathologist, we pursued the case further by obtain- ing angiographic studies. These studies demonstrated the exaggerated vascular supply to the tumor via the right de- scending palatine artery and an impressive blush phase (Figs. 6 and 7). The blush phase was 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 catheter- ization during angiography, it was decided that emboliza- tion was a viable treatment alternative. Embolization of the terminal one third of the internal maxillary artery was ac- complished with polyvinyl alcohol, a nonresorbable mate- rial. During recovery, the patient demonstrated only mod-

Fig. 7. Blush (capillary) phase of angiogram.

Figs. 8 and 9. Initial presentation of left anterior max- illary lesion.

erate bilateral ecchymoses at the sites of the femoral artery catheterization. In the immediate postoperative period, there was a noticeable decrease in 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 weeks after embolization, the lesion was again sclerosed. At 4 weeks, evidence of sloughing of the surrounding tissue was seen. One week later, the patient had a 2.0 X 1.5 X 0.5 cm segment of necrotic bone wrapped in a tissue. No oroantral

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Fig. 11. Angiogram of left anterior maxillary lesion.

Fig. 10. Radiographic appearance of left antedor max- illa with diffuse trabecular pattern changes.

fistula was seen, and the surrounding tissues appeared to 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 subsequent hemorrhage or other evidence of recur- rence.

CASE 2

A well-nourished, well-developed lCyear-old Hispanic girl undergoing orthodontic therapy was referred for spon- taneous bleeding and hypertrophied gingiva in the anterior portion of the right maxilla. The medical history was non- contributory. 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 exhib- ited separation when compared with the left side. Radio- graphic examination revealed a distinct change in the tra- becular pattern of the right anterior maxilla as compared to the unaffected contralateral side (Fig. 10).

The biopsy specimen from case 2 showed soft tissue very similar in appearance to the soft tissue seen in case 1. No

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 because of the amount of vascularity, the lack of ulceration, and the sparse inflam- matory 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 pa- tient underwent angiography with superselective catheter- ization. This revealed that the major feeding vessel was the descending palatine 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 redness of the lesion, but there was no spontaneous or easily elicited hemorrhage. This re- currence occurred despite postembolization sclerosing ther- apy. At this time, it was decided to sclerose the 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 re- sembled the adjacent normal mucosa with the exception of small areas of 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 in- volved area have remained slightly more erythematous than the surrounding mucosa. Close observation has continued in concert with the neuroradiologist because reembolization may be necessary if the lesion does not continue to improve.

DISCUSSION

The treatment of hemangiomas of bone varies con- siderably 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

272 Greene et al.

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 neu- roradiologist achieved access to each lesion’s vascular supply, microembolization became an attractive treat- ment alternative.

3. Since our two cases would have required ligation of the external carotid artery for the control of surgi- cal hemorrhage, microembolization offered a distinct advantage. With the use of microembolization, no li- gation was necessary.

4. The effectiveness of microembolization is due to the fact that the embolizing material lodges within small vessels in the depth of the lesion, producing ir- reversible 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 tech- nique, the patients retain intact jaws and dentition.

7. Ablative surgery can still be performed if neces- sary.

SUMMARY

We have presented two cases of intraoral capillary hemangiomas of bone with soft tissue extension. The various treatment modalities have been reviewed. Our reasoning, treatment protocol, and results have been presented. We believe that microembolization is a vi- able treatment alternative and should be considered as a first line therapy in similar vascular tumors.

REFERENCES

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Shklar G, Meyers 1. Vascular tumors of the mouth and jaws. ORAL SURG URAL MED ORAL PATHOL 1965;19:335-57: Gamez-Arauio JJ. Toth BB. Luna MA. Central hemangioma of the mandible and maxilla: review of vascular lesions. ORAL SURG ORAL MED ORAL PATHOL 1974;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, Fried- man JM. Central hemangioma of the mandible. ORAL SURG ORAL MED ORAL PATHOL 1981;52:471-7. Shafer MG, Hine MK, Levy BM. A textbook of oral pathol- ogy. 4th ed. Philadelphia: WB Saunders, 1983:154-257. Edgerton MT. The treatment of hemangiomas with special reference to the role of steroids. Ann Surg 1976;183:517-30. Shafir R, Slutski S, Bornstein LA. Excision of buccal heman- gioma by carbon dioxide laser beam. ORAL SURG ORAL MED ORAL PATHOL 1977;44:347-50. Apfelberg DB, Greene RA, Maser MR, Lash H, Rivers JL, Laub DR. Results of argon laser exposure of capillary heman- giomas of infancy-preliminary report. Plast Reconstr Surg 1981;67:188-92. Chin DC. Treatment of maxillary hemangioma with scleros-

ORAL SURC ORAL MED ORAL PATNOL September 1990

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12. Morgan JF, Schow E. Use of sodium morrhuate in the man- agement 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 cases treated 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;50:618-2 1.

16. Sachatello CR. McSwain B. Regression of cutaneous capillary hemangioma. km J Surg 1968yll6: 113-4.

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17. Zou Z, Wu Y, Sun Q, Zhu X, Meng X, He Z. Clinical appli- cation of angiography of oral and maxillofacial hemangiomas. ORAL SURG ORAL MED ORAL PATHOL 1983;55:437-47.

18. Berenstein A, Kricheff II. Microembolization techniques of vascular occlusion: radiologic, pathologic, and clinical corre- lation. AJNR 1981;2:261-7.

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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 complet- ing orthodontic therapy.

On presentation, there was clear clinical evidence of recurrence. The patient traces the first sign of re- currence 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 com- pared 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 was consistent 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 symptoms- the temperature slightly increased, and the patient stated that she had had a vague awareness in 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-

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nal carotid artery was performed to address the facial artery anomaly; of course, this also included the internal maxillary artery. A conservative resection of the bony lesion of the anterior maxilla was under- taken. At the time of surgery we believed that the in- cisors could be retained because alveolar support was adequate (endodontic therapy had been done preop- eratively).

The bony defect, which extended to the nasal mu- cosa, was packed with hydroxyapatite. The mucosa was closed routinely. The patient tolerated the proce- dures well and was discharged in good condition. Postoperatively, the patient demonstrated a marginal

mandibular nerve weakness and a hypoglossal nerve deficit. Neuropraxia is the most plausible explanation at this early date.

Certainly, this case appears to demonstrate recan- olization 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 re- quired 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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