Solitary neurofibroma of the maxilla: Report of a case

SKOUTERIS

701

AND SOTEREANOS

fissure to enter the ocular surface of the lateral rectus muscle. The patient described in this report did not have any prior history of abducens nerve palsy. The abducens neuropraxia most likely occurred in the region of the cavernous sinus because of the displacement of a fragment of the fracture through the sella tursica onto the medial surface of the cavernous sinus where the sixth cranial nerve is more medial than other cranial nerves that enter the sinus. Although the cause of the abducens nerve palsy was a fracture of the sphenoid bone, the reason why the fracture occurred was not clear. The most probable explanations are either increased force or inappropiate direction of force from the osteotome while separating the pterygoid plates, or the placement of the osteotome too high on the pterygoid plates. It is evident that the direction and force of the osteotomes are extremely important in preventing unwanted fractures. The osteotome should always be directed anteriorly, medially, and inferiorly at the pterygoid plates. Summary A case of transient abducens nerve palsy following Le Fort I maxillary osteotomy is reported. The results of the CT scan strongly suggest the cause was a fracture of the body of the sphenoid bone but the cause of the fracture is unclear. The palsy oc-

J Oral Maxillofac 46:701-705.

curred on the first postoperative day and recovery took approximately 5 months. The most likely explanation for the complication is a transmittal of force from the osteotome used to fracture through the pterygoid plates extending superiorly through the medial surface of the cavernous sinus. This case clearly demonstrates the importance of care in positioning of the pterygomaxillary osteotome. References 1. Pames EI, Becker ML: Necrosis of the anterior maxilla following osteotomy. Oral Surg 33:326, 1972 2. West RA, Epker BN: Posterior maxillary surgery: Its place in the treatment of dentofacial deformities. J Oral Surg 30562. 1972 3. Westwood RM, Tilson HB: Complications associated with maxillary osteotomies. J Oral Surgery 33: 104, 1975 4. Mainous EG, Crowell NT: Nasal septum perforation following total maxillary osteotomy: Report of a case. J Oral Surg 31:869, 1973 5. Tomasetti BJ, Broutsas M, Gormely M, et al: Lack of tearing after Le Fort I osteotomy. J Oral Surgery 34:1095, 1976 6. Stringer DE, Polwick M, Steed DL, et al: Subcutaneous emphysema after Le Fort I osteotomy: Report of two cases. J Oral Surg 37: 115, 1979 7. Stem NS, Shensa DR, Trop RC, et al: Cavernous sinus thrombosis: A complication of maxillary surgery. J Oral Surg 39:436, 1981 8. Lanigan DT, West RA: Management of postoperative hemorrhage following the Le Fort I maxillary osteotomy. J Oral Surg 42:367, 1984 9. Watts PG: Unilateral abducent nerve palsy: A rare complication following a Le Fort I maxillary osteotomy. Br J Oral Surg 22:212, 1984

Surg

1966

Solitary Neurofibroma

of the Maxilla:

Report of a Case CHRIS A. SKOUTERIS,

DMD, PHD,* AND GEORGE C. SOTEREANOS,

Received from the Department of Oral and Maxillofacial Surgery, Pres#byterian University Hospital, Pittsburgh, Pennsylvania. * Chief Resident. t Director of Graduate Education in Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr. Skouteris: Department of Oral and Maxillofacial Surgery, Presbyterian University Hospital, DeSoto at O’Hara Streets, Room 2600, Pittsburgh, PA 15213. 0 1988 American geons

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DMD, MST

Benign neural sheath neoplasms are not uncommon in the maxillofacial region. These lesions can occur as solitary tumors, or they can affect many sites in the form of multiple neurofibromatosis. The tongue represents the most common intraoral site for these tumors.’ Osseous involvement is uncommon; the mandible appears to be affected most often.* The maxilla remains an unusual location for these neoplasms. The purpose of this paper is to report a case of a large solitary neurolibroma of the maxilla with an unusual presentation. A review of

702

SOLITARY NEUROFIBROMA

OF MAXILLA

Report of a Case A 16-year-old white girl presented to the oral and maxillofacial surgical service at Presbyterian University Hospital for evaluation of a mass in the left posterior maxilla.

FIGURE 1. Appearance of the mass protruding into the left posterior maxillary vestibule. Note the absence of the 1st and 2nd maxillary molars.

the literature relative to the occurrence of solitary maxillary neurofibromas is also presented in an effort to identify the characteristics of such tumors in this osseous location.

The patient had noted lack of eruption of teeth on the affected site. The mass had been present for 6 months and had been slowly increasing in size. Physical examination showed a well developed and nourished female in no acute distress. Oral examination revealed a painless swelling in the left posterior maxillary vestibule (Fig. 1). The first and second left maxillary molars were not visible. The mass was firm and covered by an intact, slightly hyperemic mucosa. The premolars adjacent to the mass tested vital to thermal and electrical stimuli. The remaining dentition was normal. The medical history of the patient was noncontributory. and the findings of the remainder of the physical examination were unremarkable. The results of the laboratory tests were within normal limits. Panoramic radiographic evaluation showed an illdefined radiolucent lesion in the left maxillary tuberosity region. The lesion was associated with unerupted and malposed first and second maxillary molars (Fig. 2). An attempt at aspiration yielded no blood or other fluid. The clinical impression was that of an odontogenic tumor or fibro-osseous lesion. An incisional biopsy was

FIGURE 2 (fop). Panoramic radiograph shows an ill-defined radiolucency in the left posterior maxilla associated with impacted and malposed 1st and 2nd maxillary molars. FIGURE 3 (bortom). Low-power photomicrograph showing a myxomatous, collagenous stroma with numerous spindleshaped tumor cells. (Hematoxylin and eosin. Original magnification, X25.)

SKOLJTERIS AND SOTEREANOS

performed through a small vestibular incision. The lesional tissue was characterized histologically by a myxomatous stroma with numerous spindle-shaped cells (Fig. 3). The mlclei were polarized towards one end of the cell, with a long delicate cytoplasmic process extending from the opposite pole. The histologic findings were compatible with neurofibroma. In view of this finding a more detailed evaluation of the patient was done, but it failed to produce evidence of multiple neurofibromatosis. The patient was scheduled for excision of the lesion

under general anesthesia. The mass was approached intraorally by way of a mucosal, interpapillary incision that extended from the left maxillary lateral incisor to an area posterior to the left maxillary tuberosity. The lesion was easily dissected from the overlying mucosa. The mass occupied the entire left zygomatic buttress and maxillary tuberosity area. The unerupted, malposed first molar could be seen surrounded by the lesion (Fig. 4). The mass was removed, together with the contained teeth. The excised specimen measured 3 x 2.5 x 2 cm. Macroscopitally, the cut surface had a glistening, grayish-white color, and a firm, fleshy texture. Histologically, the entire specimen consisted of a moderately cellular, dense, collagenous tissue, with well oriented fibers (Fig. 5). The cells had uniform, spindle-shaped nuclei, and thin, elongated cytoplasmic processes (Fig. 6). There was no evidence of either mitotic figures or malignant metaplasia in the specimen. The final histologic diagnosis was neurotibroma. The patient had an uneventful hospital course. At 10 months postoperatively no evidence of recurrent disease was found.

Discussion Intraosseous nerve sheath neoplasms are unusual. This fact was demonstrated by Fawcett and

FIGURE 4. Intraoperative view of the lesion, which is seen occupying the left zygomatic buttress and maxillary tuberosity area. Part of the crown of the unerupted 1st molar is seen surrounded by the mass.

703 Dahlin, who identified only seven of these tumors in a series of 3987 primary bone neoplasms.3 In a review of the literature, however, Gordon reported an increased incidence of intraosseous nerve sheath neoplasms as the result of reclassification with appropriate histologic designation.4 Nonetheless, the incidence of the solitary neurofibroma of the maxilla remains rare. A review of the literature by Ellis et al. on the intraosseous, benign neural sheath neoplasms of the jaws, included only one case of solitary maxillary neurofibroma.5 However, in the original description of this lesion, only a simple mention was made regarding the histology of the tumor.6 The article did not include any photomicrographs to support the diagnosis of neurofibroma and, therefore, we believe that this cannot be accepted as a welldocumented case of solitary maxillary neurofibroma. Our search of the English-language literature produced three cases of solitary maxillary neurofibroma. These cases, together with the present report, bring the total number of well-documented cases of solitary maxillary neurofibroma to four (Table I), however, more maxillary neoplasms might have been reported under a different name due to the haphazard and often interchangeable use of the terms neurofibroma, neurilemmoma, schwannoma, and neurinoma. Although the true cell of origin of these tumors still remains a point of controversy, the confusion that existed for some time in recognizing the neurofibroma and the schwannoma as two distinct pathologic entities has been cleared in the recent years. The neurofibroma is usually a nonencapsulated, well-delineated mass composed of spindleshaped cells in a myxomatous or collagenous stroma. In contradistinction, the schwannoma is an encapsulated tumor that exhibits Antoni type A tissue, acellular Verocay bodies, palisaded nuclei, and/or Antoni type B tissue. These aforementioned histologic criteria are now well defined, and these two entities should not be confused.’ Although the number of solitary maxillary neurofibromas is small, certain observations can be made regarding their natural behavior. It appears that young adults are usually affected, with a mean age of 27.2 years. There is no sex predilection. The lesions usually present as swellings associated with symptoms that can include pain, dysphagia, and nasal obstruction. These tumors show a strong predilection for the posterior maxilla. Extension into the maxillary sinus has occurred in one case.’ whereas primary involvement of the maxillary sinus has also been demonstrated.’ Radiographically, the majority of the lesions appeared as radiolucencies, with the

704

SOLITARY NEUROFIBROMA

OF MAXILLA

FIGURE 5 (top). Photomicrograph showing a moderately cellular, dense, wavy collagenous tissue. The collagen fibers are well-oriented. (Hematoxylin and eosin. Original magnification, X40.1 FIGURE 6 (bottnml. High-power photomicrograph showing numerous cells with spindle-shaped nuclei, and delicate, elongated, cytoplasmic processes. (Hematoxylin and eosin. Original magnification, X 125.1

exception of one case that presented as a radiopacity.’ From the histologic standpoint, all cases showed the standard features of the neurofibroma with the Table 1.

exception of one case that, in addition, contained delicate, concentric lamellations of pacinian corpuscle origin.’ In general, the clinical behavior of the solitary

Solitary Neurofibromas of the Maxilla

Authors

Age/Sex

Site

Toth et al.’ Agarwal et al9

28/M 45/M

PM* MSt

Brady et al8 Skouteris and Sotereanos

20/F 16/F

PM/MS PM

PM*: Posterior Maxilla MS**: Maxillary Sinus NED + : No Evidence of Disease

Signs and Symptoms

Radiology

Treatment

Follow-up

Dysphagia Nasal obstruction, swelling Pain/swelling Swelling

Lucency Opacity

Excision Curretage

NED + -6 months NA

Lucency Lucency

Excision Excision

NED-1 year NED-10 months

CUESTAS-CARNERO

ET AL.

705

maxillary neurofibromas appears to be similar to that of the other benign nerve sheath tumors. They usually grow slowly, behave in an indolent manner, and rarely recur following excision. However, there appears to be a difference in the incidence of malignant transformation between the neurofibroma and the schwannoma.8 The malignant potential of the schwannoma is low.” The neurofibroma, on the other hand, exhibits a higher frequency of malignant change, particularly when it is associated with multiple neurofibromatosis.” The solitary maxillary neurofibromas have a benign course and total excision is curative . References 1. Toth BB, Long WH, Pleasants JE: Central pacinian neurofibroma of the maxilla. Oral Surg 39:630, 1975

J Oral Maxillofac 46:706-709.

2. Sharawy A, Springer J: Central neurotibroma occurring in the mandible. Oral Surg 25:817, 1968 3. Fawcett KJ, Dahlin DC: Neurilemmoma of bone. Am J Clin Path01 47:759, 1967 4. Gordon EJ: Solitary intraosseous neurilemmoma of the tibia. Clin Orthop 117:271, 1976 5. Ellis GL. Abrams AM, Melrose RJ: Intraosseous benign neural sheath neoplasms of the jaws: Report of seven new cases and review of the literature. Oral Surg 4473 1, 1977 6. Stillman FS: Neurofibromatosis. Oral Surg 10: 112, 1952 7. Shafer WG, Hine MK, Levy GM: A Textbook of Oral Pathology, 3rd ed. Philadelphia, WB Saunders, 1983, pp 208-209 8. Brady GL, Schaffner DL, Joy ED Jr, et al: Solitary Neurofibroma of the Maxilla. J Oral Maxillofac Surg 40:453, 1982 9. Agarwal MK, Gupta OP, Saman HC, et al: Neurotibroma of the maxillary antrum. Oral Surg 48: 150, 1979 10. Carstons HB, Schrodt RG: Malignant transformation of a benign encapsulated neurilemmoma. Am J Clin Path01 51: 144, 1969 11. Hosoi K: Multiple neurotibromatosis (von Recklinghausen’s disease) with special reference to malignant transformation. Arch Surg 22:258, 1931

Surg

1986

Odon togenic Mvxoma: Report of a &se RICARDO CUESTAS-CARNERO, DDS,* RICARDO 0. BACHUR,* HECTOR GENDELMANt

The odontogenic myxoma of the jaw bone is a benign, locally aggressive, and nonmetastisizing neoplasm that probably arises from the primitive mesenchymal structures of a developing tooth including the dental follicle, dental papilla, or periodontal ligament. lm3This view is supported by the close histologic similarity of the tumor to the dental papilla of the tooth germ.4 It is an uncommon lesion and is unusual for a bone other than the maxilla or mandible to be the site of origin of a true odontogenie myxoma.5-7 In the jaws, it usually occurs in adolescents and young adults (67%) although children and older persons can also be affected.8-‘0

Received from the National University of Cordoba, Cordoba, Argentina. * Department of Oral Surgery III, School of Dentistry. t Department of Oral Pathology, School of Dentistry. Address correspondence reprint requests to Dr. CuestasCarnero: General Bustos 401, Cordoba (5000) Argentina. 0 1988 American geons

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AND

However, it rarely occurs before 10 or after 50 years of age.” Distribution of reported cases between the sexes is similar and the maxilla and mandible are equally affected.“-I3 Most cases of early odontogenic myxomas are asymptomatic and the rate of growth is usually slow, but some tumors have grown rapidly. ‘w6 Like most other odontogenit neoplasms, pain and paresthesia do not usually become prominent until considerable enlargement of the mass has occurred. Loosening and displacement of the teeth are likely to occur in time. Radiographically, the tumor is usually a radiolucent, multilocular lesion, often demonstrating a fine soap-bubble or honeycombed trabeculation in the central area and with poorly defined margins.” Occasionally, a well defined sclerotic margin is scalloped between the teeth.‘* The roots of the teeth may show resorption. The myxoma may also be a unilocular lesion and behave less destructively than the larger, multilocular variety.21*22 These lesions may be extensive and involve a condyle or an entire ramus,” or perhaps half of the mandible or maxilla.20 The cortex is at first thinned and ex-