British
of
Journal
fQ 1991 The
Oral
British
and Mar;l/ofaciol
Association
of Oral
Surgery
(1991) 29, 20%-210
and Maxillofacial
Surgeons
Neurilemmoma in the maxillary alveolar bone: report of a case Y. Takeda Department
of Oral Pathology,
School of Dentistry, Iwate Medical
University, Morioka,
Japan
SUMMARY. Neurilemmoma located in the maxillary alveolar bone of a 64-year-old female is presented. review of the literature yields only two previous cases of central neurilemmoma of the maxilla.
A
lated, and was composed of densely cellular and irregularly arranged, elongated spindle tumour cells (Fig. 4). In places, tumour cells were aligned to form a palisading pattern (Fig. 5). Immunohistochemical study showed that tumour cells were positive for S-100 protein and neuron specific enolase. It was not possible to identify a peripheral nerve related to this tumour.
INTRODUCTION
The neurilemmoma, also called schwannoma or neurinoma, is a benign tumour of peripheral nerve sheath and it is believed that this tumour is derived from Schwann cells. Whereas the head and neck region is the most common site for benign nerve sheath tumours, intra-oral lesions are unusual and only a very small number occur intra-osseously. The commonest site for the intra-oral neurilemmoma is the tongue. Central bony neurilemmomas occur most often in the mandible. Only two previous cases of central neurilemmoma of the maxilla appear to have been published (Friedman, 1964; Ord & Rennie, 1981). This paper reports a rare case of neurilemmoma arising in the maxillary alveolar bone, clinically and radiographically mimicking chronic periodontitis.
DISCUSSION
This paper reports an intra-bony neurilemmoma arising in the maxilla. Central neurilemmoma are uncommon, comprising less than 1% of all primary central osseous lesions (Fawcett & Dahlin, 1967). The mandible is the most common intra-bony site with the majority of tumours arising directly from the inferior dental nerve, and the molar and angle regions of the mandible are the intra-oral sites usually involved. There are only two reported cases of maxillary intra-osseous neurilemmoma. The first case, reported by Friedman (1964), was in an 18-year-old female, and the site of the lesion was the maxillary incisor area. Ord and Rennie (1981) reported the second case in a 21-year-old male whose lesion occurred in the maxillary tuberosity. The present case was in a 64-year-old female, and the location of the lesion was the maxillary premolarmolar area. Murphy and Giunta (1985) reviewed the literature of intra-osseous neurilemmoma of the jaws. There was a female predilection, with a 1.6: 1 female to male ratio. The age range was 7 to 84 years, with an average age of 37 years. Seventy-seven percent of the patients were below the age of 50 years at the time of diagnosis and 46% were below the age of 30 years. This sex ratio and age distribution are similar to those in neurilemmomas of soft tissue origin (Cherrick & Eversole, 1971; Ellis et al., 1977). The tumour is usually asymptomatic, but occasional tumours causing paraesthesia and pain have been reported. Radiographs usually reveal a unilocular or multilocular radiolucency which may be bordered by a dense sclerotic margin of bone. A few cases with focal calcification appearing on radiographs have
Case report
A 64-year-old Japanese female was referred for evaluation of a painless swelling of the right maxillary alveolar region of 11 months duration. Clinical examination revealed expansion of the palatal bone of the maxillary right premolar-molar region (Fig. 1). The overlying mucosa was intact and was of normal colour, except for the palatal gingivawhich showed severe chronic marginal periodontitis. The second premolar and first molar teeth were mobile, and the molar did not respond to pulp testing. Radiographic examination of the lesion suggested complete bone loss around the roots of the molar (Fig. 2). Aspiration was attempted, but the lesion was found to be solid. The general physical examination was otherwise non-contributory, and routine laboratory tests revealed no abnormalities. A biopsy was undertaken and showed the feature of a neurilemmoma, with cellular atypia which was probably due to inflammatory change. Under general anaesthesia, the lesion was surgically resected. The postoperative healing process was uneventful, and there has been no sign of recurrence 12 months later. The resected specimen showed a well-circumscribed, intrabony, unilocular mass, greyish-white in colour and rubbery in consistency, around the roots of the first molar tooth (Fig. 3). Histologically, the lesion was not encapsu208
210 British Journal of Oral and Maxillofacial Surgery
examination. The extent of the lesion at diagnosis may indicate more radical procedures, including removal of teeth and bone resection. Very rarely, malignant transformation of an encapsulated neurilemmoma has been reported (Carstens & Schrodt , 1969). References Carstens, P. H. B. & Schrodt, G. R. (1969). Malignant transformation of a benign encapsulated neurilemmoma. American Journal of Clinical Pathology, 51,144.
Cherrick. H. M. & Eversole, L. R. (1971). Benign neural sheath neoplasms of the oral cavity; report of 37 cases. Oral Surgery, Oral Medicine, Oral Pathology, 32,900.
Ellis, G. L., Abrams, A. M. XLMelrose, R. J. (1977). Intraosseous benign neural sheath neoplasms of the jaws. Oral Surgery, Oral Medicine. Oral Pathology, 44,731.
Fawcett, K. J. & Dahlin, D. C. (1967). Neurilemmoma of bone.
Murphy, J. & Giunta, J. L. (1985). Typical central neurilemmoma of the mandible. Oral Surgery, Oral Medicine, Oral Pathology, 59,275.
Ord, R. A. & Rennie, J. S. (1981). Central neurilemmoma of the maxilla. international Journal of Oral Surgery, 10,137. Rengaswamy, V. (1978). Central neurilemmoma of the jaws+ review of the literature and case report. lnternational Journal of Oral Surgery, I, 300.
The Author Yasunori Takeda DDS, DDSc
Department of Oral Pathology School of Dentistry Iwate Medical University Uchimaru 19-1, Morioka, Iwate 020 Japan Correspondence Takeda.
and requests for offprints to Dr. Yasunori
American Journal of Clinical Pathology, 47,759.
Friedman, M. (1964). Intraosseous schwannoma: a report of a case. Oral Surgery, Oral Medicine, Oral Pathology, 18,90.
Paper received 24 October 1990 Accepted 2 January 1991
Neurilemmoma
in the maxillary aveolar bone
209
Fig. 1 - Preoperative view of the lesion revealing palatal expansion of the premolar-molar region of the right maxilla.
Fig. 2 - Radiograph suggesting complete bone loss around the
roots of the first molar tooth.
Fig. 3 - Cut view of the resected specimen. A well circumscribed, intrabony, unilocular mass (arrows) in the first molar region.
Fig. 4 - The lesion is composed of irregularly and densely arranged, elongated spindle cells (H&E. Original magnification x 150).
Fig. 5 - Tumour cells aligned in places to form a characteristic
palisading pattern (H&E. Original magnification x300).
been reported (Ellis et al., 1977; Rengaswamy, 1978). The nerve from which the neurilemmoma arises may be seen related to the capsule in 7% of cases (Ord & Rennie, 1981). Although a nerve bundle was
not detected in the present case, the nerve of origin was most likely a small branch of the middle superior alveolar nerve. The treatment of central neurilemmoma is complete surgical excision with periodic follow-up
of
Journal
fQ 1991 The
Oral
British
and Mar;l/ofaciol
Association
of Oral
Surgery
(1991) 29, 20%-210
and Maxillofacial
Surgeons
Neurilemmoma in the maxillary alveolar bone: report of a case Y. Takeda Department
of Oral Pathology,
School of Dentistry, Iwate Medical
University, Morioka,
Japan
SUMMARY. Neurilemmoma located in the maxillary alveolar bone of a 64-year-old female is presented. review of the literature yields only two previous cases of central neurilemmoma of the maxilla.
A
lated, and was composed of densely cellular and irregularly arranged, elongated spindle tumour cells (Fig. 4). In places, tumour cells were aligned to form a palisading pattern (Fig. 5). Immunohistochemical study showed that tumour cells were positive for S-100 protein and neuron specific enolase. It was not possible to identify a peripheral nerve related to this tumour.
INTRODUCTION
The neurilemmoma, also called schwannoma or neurinoma, is a benign tumour of peripheral nerve sheath and it is believed that this tumour is derived from Schwann cells. Whereas the head and neck region is the most common site for benign nerve sheath tumours, intra-oral lesions are unusual and only a very small number occur intra-osseously. The commonest site for the intra-oral neurilemmoma is the tongue. Central bony neurilemmomas occur most often in the mandible. Only two previous cases of central neurilemmoma of the maxilla appear to have been published (Friedman, 1964; Ord & Rennie, 1981). This paper reports a rare case of neurilemmoma arising in the maxillary alveolar bone, clinically and radiographically mimicking chronic periodontitis.
DISCUSSION
This paper reports an intra-bony neurilemmoma arising in the maxilla. Central neurilemmoma are uncommon, comprising less than 1% of all primary central osseous lesions (Fawcett & Dahlin, 1967). The mandible is the most common intra-bony site with the majority of tumours arising directly from the inferior dental nerve, and the molar and angle regions of the mandible are the intra-oral sites usually involved. There are only two reported cases of maxillary intra-osseous neurilemmoma. The first case, reported by Friedman (1964), was in an 18-year-old female, and the site of the lesion was the maxillary incisor area. Ord and Rennie (1981) reported the second case in a 21-year-old male whose lesion occurred in the maxillary tuberosity. The present case was in a 64-year-old female, and the location of the lesion was the maxillary premolarmolar area. Murphy and Giunta (1985) reviewed the literature of intra-osseous neurilemmoma of the jaws. There was a female predilection, with a 1.6: 1 female to male ratio. The age range was 7 to 84 years, with an average age of 37 years. Seventy-seven percent of the patients were below the age of 50 years at the time of diagnosis and 46% were below the age of 30 years. This sex ratio and age distribution are similar to those in neurilemmomas of soft tissue origin (Cherrick & Eversole, 1971; Ellis et al., 1977). The tumour is usually asymptomatic, but occasional tumours causing paraesthesia and pain have been reported. Radiographs usually reveal a unilocular or multilocular radiolucency which may be bordered by a dense sclerotic margin of bone. A few cases with focal calcification appearing on radiographs have
Case report
A 64-year-old Japanese female was referred for evaluation of a painless swelling of the right maxillary alveolar region of 11 months duration. Clinical examination revealed expansion of the palatal bone of the maxillary right premolar-molar region (Fig. 1). The overlying mucosa was intact and was of normal colour, except for the palatal gingivawhich showed severe chronic marginal periodontitis. The second premolar and first molar teeth were mobile, and the molar did not respond to pulp testing. Radiographic examination of the lesion suggested complete bone loss around the roots of the molar (Fig. 2). Aspiration was attempted, but the lesion was found to be solid. The general physical examination was otherwise non-contributory, and routine laboratory tests revealed no abnormalities. A biopsy was undertaken and showed the feature of a neurilemmoma, with cellular atypia which was probably due to inflammatory change. Under general anaesthesia, the lesion was surgically resected. The postoperative healing process was uneventful, and there has been no sign of recurrence 12 months later. The resected specimen showed a well-circumscribed, intrabony, unilocular mass, greyish-white in colour and rubbery in consistency, around the roots of the first molar tooth (Fig. 3). Histologically, the lesion was not encapsu208
210 British Journal of Oral and Maxillofacial Surgery
examination. The extent of the lesion at diagnosis may indicate more radical procedures, including removal of teeth and bone resection. Very rarely, malignant transformation of an encapsulated neurilemmoma has been reported (Carstens & Schrodt , 1969). References Carstens, P. H. B. & Schrodt, G. R. (1969). Malignant transformation of a benign encapsulated neurilemmoma. American Journal of Clinical Pathology, 51,144.
Cherrick. H. M. & Eversole, L. R. (1971). Benign neural sheath neoplasms of the oral cavity; report of 37 cases. Oral Surgery, Oral Medicine, Oral Pathology, 32,900.
Ellis, G. L., Abrams, A. M. XLMelrose, R. J. (1977). Intraosseous benign neural sheath neoplasms of the jaws. Oral Surgery, Oral Medicine. Oral Pathology, 44,731.
Fawcett, K. J. & Dahlin, D. C. (1967). Neurilemmoma of bone.
Murphy, J. & Giunta, J. L. (1985). Typical central neurilemmoma of the mandible. Oral Surgery, Oral Medicine, Oral Pathology, 59,275.
Ord, R. A. & Rennie, J. S. (1981). Central neurilemmoma of the maxilla. international Journal of Oral Surgery, 10,137. Rengaswamy, V. (1978). Central neurilemmoma of the jaws+ review of the literature and case report. lnternational Journal of Oral Surgery, I, 300.
The Author Yasunori Takeda DDS, DDSc
Department of Oral Pathology School of Dentistry Iwate Medical University Uchimaru 19-1, Morioka, Iwate 020 Japan Correspondence Takeda.
and requests for offprints to Dr. Yasunori
American Journal of Clinical Pathology, 47,759.
Friedman, M. (1964). Intraosseous schwannoma: a report of a case. Oral Surgery, Oral Medicine, Oral Pathology, 18,90.
Paper received 24 October 1990 Accepted 2 January 1991
Neurilemmoma
in the maxillary aveolar bone
209
Fig. 1 - Preoperative view of the lesion revealing palatal expansion of the premolar-molar region of the right maxilla.
Fig. 2 - Radiograph suggesting complete bone loss around the
roots of the first molar tooth.
Fig. 3 - Cut view of the resected specimen. A well circumscribed, intrabony, unilocular mass (arrows) in the first molar region.
Fig. 4 - The lesion is composed of irregularly and densely arranged, elongated spindle cells (H&E. Original magnification x 150).
Fig. 5 - Tumour cells aligned in places to form a characteristic
palisading pattern (H&E. Original magnification x300).
been reported (Ellis et al., 1977; Rengaswamy, 1978). The nerve from which the neurilemmoma arises may be seen related to the capsule in 7% of cases (Ord & Rennie, 1981). Although a nerve bundle was
not detected in the present case, the nerve of origin was most likely a small branch of the middle superior alveolar nerve. The treatment of central neurilemmoma is complete surgical excision with periodic follow-up
