Transmaxillary excision of a rare cavernous hemangioma of the infratemnoral fossa.

Abstract

Cavernous hemangiomas that manifest as a primary tumor of the infratemporal fossa are exceedingly rare. We report such a case in a 40-year-old woman. Her lesion was excised via a transantral anterior approach. Although this approach is usually recommended only for biopsy, in selected cases it can be used to excise a lesion without causing any significant complications. We recommend the transantral anterior app roach for relatively small, benign lesions that show no evidence of invasion into surrounding tissues because it does not expose the patient to surgical complications such as transient or permanent facial nerve injury or cosmetic deformity.

Introduction

Hemangiomas are the most common benign tumors in infants; in fact, they are the most common of all birth defects. They are benign vascular malformations that originate in mesodermal nests of vasoformative tissue. Synonyms for hemangioma are benign mesenchymoma, infiltrating angiolipoma, angiofibrolipoma, hemartoma, and cavernous hemangioma. [1,2] Depending on their histology, hemangiomas are classified as capillary (small-vessel), cavernous (large-vessel), and mixed-type. [1] In adults, the most common type is the cavernous hemangioma. There have been several reports of cavernous hemangiomas of the oral cavity, pharynx, and larynx, [3-6] but these neoplasms are exceedingly rare as primary tumors of the infratemporal fossa. [6-10] This article describes our diagnosis and treatment of a cavernous hemangioma that was located in infratemporal fossa.

Case report

A 40-year-old woman was admitted to the Department of Otolaryngology--Head and Neck Surgery at Hacettepe University in Ankara, Turkey, with a history of right facial pain and headache of 2 years' duration. Five months earlier, a neurosurgeon had diagnosed trigeminal neuralgia and had prescribed carbamazepine. Two months prior to her admission to our ENT department, she had noticed that a mass on her right cheek became visible whenever she opened her mouth.

The results of her physical examination, routine laboratory tests, chest x-ray, and panoramic x-ray of the mandible were all normal. Magnetic resonance imaging revealed the presence of a well-circumscribed, cylindrical mass in the right infratemporal fossa whose dimensions were estimated to be 2 x 1.5 x 3 cm (figure 1). Her angiogram revealed a faint tumor blush without a significant feeder vessel.

A transantral anterior surgical approach with a Caldwell-Luc incision was used to gain access to the infratemporal fossa. The right maxillary sinus was normal. The posterior wall of the maxillary sinus was fenestrated with a hammer and chisel until the mass could be seen. Exposure was improved by removing the posterior and lateral walls of the maxillary sinus with a rongeur. The mass was easily detached and removed from the surrounding tissues by blunt dissection. There was no hemorrhage.

On visual inspection, the mass was brown and had regular contours, and its actual measurement was 3 x 2.5 x 1 cm. Microscopic examination with hematoxylin and eosin staining revealed that the lesion contained large vessels with cystically dilated lumina, which were filled with erythrocytes and fibrous tissue interspersed between these vessels (figure 2). These features are characteristic of a cavernous hemangioma.

The patient experienced no postoperative complications, and she was discharged 2 days following surgery. Followup computed tomography obtained 1 year after surgery revealed no evidence of recurrence (figure 3).

Discussion

Hemangiomas are congenital, benign, well-circumscribed malformations of the vasculature. Capillary hemangiomas are more common in infants, while cavernous lesions are more common in adults. Capillary hemangiomas tend to be more superficial than cavernous lesions, as they usually occur in the dermal layer, and they gradually undergo involution. Cavernous hemangiomas appear as globular, compressible, bright-red or deep-purple lesions, which sometimes temporarily enlarge during straining. They tend to involve the deep, subcutaneous tissues. Cavernous hemangiomas are less likely to spontaneously involute. [11]

Nearly all tumors seen in the infratemporal fossa are either metastatic or extensions of intramuscular or osseous lesions. [2,8,12] Primary tumors of the infratemporal fossa are rare, and primary hemangiomas of the infratemporal fossa are exceedingly rare, as only a few cases have been reported in the literature. [6-10]

The treatment options include surgery, irradiation, laser therapy, cryotherapy, and instillation of sclerosing agents. [11] The most effective treatment is a wide surgical excision. Surgery is indicated for large tumors that invade adjacent structures and cause functional derangement, disfigurement, or pain. Postoperative radiation therapy is suggested for troublesome recurrent tumors, but its use as a primary treatment is not recommended. [8,12,13]

The complete removal of an infratemporal fossa lesion can be difficult because of the anatomic complexity and the inaccessibility of some masses. Additionally, complete excision raises concerns with regard to major postoperative complications.

Both lateral and anterior approaches have been used to gain access to the infratemporal fossa. The lateral approaches include the direct, the transparotid, and the transotic procedures. [9,14] The major complications of these operations are facial palsy, trismus, enophthalmus, malunion, and hearing loss. [7] The most common anterior approaches are the transmandibular, the transoral, and the transmaxillary procedures, all of which require an osteotomy. Complications of these procedures include malunion, postoperative infection, neural deficits, dysphagia, and facial scarring. [14,15] In addition, Kornfehl et al have described a relatively new transpalatine approach that does not require an osteotomy. [8]

The transantral anterior approach that we describe involved a Caldwell-Luc procedure and the removal of the posterior wall of the maxillary sinus to gain access to the infratemporal fossa. This technique is usually recommended only for biopsy, but in selected cases it can be performed without any significant complications. The major disadvantage of this approach is the limited exposure of the operative field. The transantral anterior approach is most useful for relatively small, well-circumscribed, benign lesions that show no evidence of invasion into surrounding tissues. The main advantage of this technique is the lack of significant surgical complications.

Patients who have had hemangiomas removed should be followed periodically because the rate of recurrence is high. Our patient has been observed for 30 months post-operatively, and we have not yet seen any sign of recurrence.

References

(1.) Allen PW, Enzinger FM. Hemangioma of skeletal muscle. An analysis of 89 cases. Cancer 1972;29:8-22.

(2.) Knox RD, Pratt MF, Garen PD, Giles WC. Intramuscular hemangioma of the infratemporal fossa. Otolaryngol Head Neck Surg 1990; 103:637-41.

(3.) Bardach J, Panje W. Surgical management of the large cavernous hemangioma. Otolaryngol Head Neck Surg 1981;89:792-6.

(4.) Bridger GP, Nassar VH, Skinner HG. Hemangioma in the adult larynx. Arch Otolaryngol 1970;92:493-8.

(5.) Cocke EW. Cavernous hemangioma of the oral and hypopharynx. Am J Surg 1961;102:798-802.

(6.) Davies HT, Neil-Dwyer G, Evans BT, Lees PD. The zygomaticotemporal approach to the skull base: A critical review of 11 patients. Br J Neurosurg 1992;6:305-12.

(7.) Johnson AT, Maran AG. Extra-cranial tumours of the infratemporal fossa. J Laryngol Otol 1982;96:1017-26.

(8.) Kornfehl J, Gstottner W, Kontrus M, Sedivy R. Transpalatine excision of a cavernous hemangioma of the infratemporal fossa. Eur Arch Otorhinolaryngol 1996;253:172-5.

(9.) Shaheen OH. Swellings of the infratemporal fossa. J Laryngol Otol 1982;96:817-36.

(10.) Toriumi DM, Shermetaro CB, Pecaro BC. Cavernous hemangioma of the infratemporal fossa. Ear Nose Throat J 1989;68:252, 258-9.

(11.) Staindl O. [Clinical aspects and therapy of hemangioma]. HNO 1988;36:259-66.

(12.) Kumar A, Valvassori G, Jafar J, Mafee M. Skull base lesions: A classification and surgical approaches. Laryngoscope 1986;96:252-63.

(13.) Hoehn JG, Farrow GM, Devine KD, Masson JK. Invasive hemangioma of the head and neck. Am J Surg 1970;120:495-500.

(14.) Grime PD, Haskell R, Robertson I, Gullan R. Transfacial access for neurosurgical procedures: An extended role for the maxillofacial surgeon. II. Middle cranial fossa, infratemporal fossa and pterygoid space. Int J Oral Maxillofac Surg 1991;20:291-5.

(15.) Grime PD, Haskell R, Robertson I, Gullan R. Transfacial access for neurosurgical procedures: An extended role for the maxillofacial surgeon. I. The upper cervical spine and clivus. Int J Oral Maxillofac Surg 1991;20:285-90.