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GLOMUS T U M O R IN THE PAROTID REGION
J Oral Maxillofac Surg 53:830-834, 1995
Glomus Tumor Originating in the Parotid Region HIDEAKI SAKASHITA, DDS, PHD,* MASARU MIYATA, DDS, PHD,1HIZURU MIYAMOTO, DDS,:~ AND HIROSHI KURUMAYA, MD, PHD§ G l o m u s t u m o r is a rare n e o p l a s m that originates from the glomus apparatus, an arteriovenous anastomosis with a specialized vascular structure. The glomus apparatus is c o m p o s e d o f epithelioid glomus cells w h o s e function is thought to be the control o f circulation and b l o o d pressure. G l o m u s tumors form from a proliferation o f g l o m u s cells, and are frequently observed in the fingernails, but occurrence in the parotid region is very rare. The following report describes such a case.
Report of Case A 63-year-old woman was referred to our department on July 26th, 1991, complaining of pain in the right temporomandibular joint (TMJ) area. She had no history of specific illnesses. Approximately 4 years before she had consulted the oral surgery department of another hospital after experiencing pain in the TMJ area; this was subsequently diagnosed as TMJ arthrosis and was treated. Later, after visiting another dentist and then the dental department of yet another hospital, she was referred to us. The patient was experiencing oppressive pain in front of the right TMJ when opening her mouth. However, the range of mouth opening was normal. A mass, approximately 1.5 cm in diameter, was felt in the parotid gland region during palpation. Echo, magnetic resonance imaging, and computerized tomographic scans showed a tumor approximately 1.2 cm in diameter in the right parotid gland. (Figs 1 through 4). After establishing the clinical diagnosis of a tumor in the right parotid gland, the patient was scheduled for surgery. An S- shaped incision was used to approach the lesion (Fig 5). After exposing the main trunk of the facial nerve and the upper main branch, the tumor was found on the parotid capsule under the nerve (Fig 6). The tumor and the surrounding tissue were removed, leaving the facial nerve intact
(Figs 7, 8). Following the operation, the patient experienced temporary facial palsy, but this disappeared after about 3 months. Histologically, there was incomplete encapsulation of the tumor, and it contained many capillaries and a few wider vessels. Numerous round or cubical, medium-sized eosinophilic cells with small, round nuclei could be seen. Typical glomus cells were scattered between these cells. The glomus cells were arranged in organoid and trabecular patterns surrounding vascular lumens (Fig 9). There was no cellular atypia or mitoses seen. A diagnosis of glomus tumor was made. Because there was no parotid tissue around the tumor, it was regarded as a parasialoma rather than as a parotid tumor. Immunoperoxidase stains were performed on the lesion using formalin-fixed paraffin sections. The cytoplasm of the glomus cells was positive for vimentin (Fig 10). S-100 protein, keratin, desmin, epithelial membrane antigen, and chromogranin were negative in glomus cells. The stain for factor VIII-related antigen was positive in the endothelial cells surrounding the vascular channels, but negative in the glomus cells (Fig 11).
Discussion The glomus t u m o r is a distinctive neoplasm, the cells o f which r e s e m b l e the modified smooth m u s c l e
Received from Ishikawa Prefectural Central Hospital, Ishikawa,
Japan. * Chief, Department of Dentistry and Oral Surgery. t Assistant Chief, Department of Denistry and Oral Surgery. ~:Staff, Department of Dentistry and Oral Surgery. § Chief, Department of Pathology. Address correspondence and reprint requests to Dr Sakashita: Department of Dentistry and Oral Surgery, Ishikawa Prefectural Central Hospital, Minami Shinbo, Kanazawa, Ishikawa 920-02, Japan. © 1995 American Association of Oral and Maxillofacial Surgeons 0278-2391/95/5307-001753.00/0
FIGURE 1. Sonogram showing a tumor (arrow) approximately 1.2 cm in diameter.
SAKASHITA ET AL
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FIGURE 2. Axial computerized tomographic scan showing a tumor (arrow) in the right parotid gland. FIGURE 4. Magnetic resonance image (coronal plane) showing the tumor (arrow) in the fight parotid gland.
cells of the normal glomus body. The normal glomus body is a specialized form of arteriovenous anastomosis that controls circulation and blood pressure. It is located in the stratum reticularis of the dermis and is most frequently encountered in the subungual region, the lateral areas of the digits, and the palm. A glomus tumor consists of two basic cells: capillary endothelial cells and pericytes. In addition, there are smooth muscle cells and nonmyelinated nerve fibrils. Tumors of the glomus apparatus are benign, organized hamartomas or hyperplasias of the normal structure. Ultrastruc-
FIGURE 3. Magnetic resonance image (transverse plane) showing the tumor (arrow) in the right parotid gland,
FIGURE 5. Preoperative photograph showing the S- shaped incision used to expose the tumor.
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GLOMUS TUMOR 1N THE PAROTID REGION
tural, immunohistochemical, and tissue culture findings all indicate a smooth-muscle origin] -5 Although glomus tumors form mainly from the glomus apparatus in the nails of the hands and feet, particularly the fingers, they sometimes also originate in the stomach, mediastinum, heart, vagina, uterus, penis, bone, lung, trachea, and eyelid. 6 Makino 7 reported only three glomus tumors in 651 cases of soft tissue tumors, and one of them formed on the face. Tsuneyoshi and Enjoji 2 reported 63 glomus tumors in 11,867 cases of soft tissue tumors, but none were found in the head or neck. Furthermore, Shugart et al 8 reported that none of their 74 cases of this tumor formed in the head and neck. As these reports show, it is very rare for glomus tumors to occur in the oral and facial region. Tokiba et al9 reported that only 16 cases of oral and facial glomus tumors, including their case, had been reported in the European and Japanese literature. In 15 of these cases, four were in the gingiva, four in the hard palate, two in the lip, two in the buccal mucosa, two in the tongue, and only one in the TMJ. According to the imaging and the findings during the operation, our case was originally considered to have started in the parotid, but histologically it was a parasiloma.
FIGURE 7. Intraoperative photograph showing the surgical defect and exposed facial nerve(*).
Generally, this type of tumor forms singly, but in some cases it can form in groups. 1° Solitary tumors are usually accompanied by pain. On the contrary, multiple tumors are not accompanied by pain and appear to
FIGURE 6. Intraoperative photograph showing the tumor (arrows) under the facial nerve.
FIGURE 8.
Surgical specimen.
SAKASHITA ET AL
FIGURE 9. Photomicrograph showing proliferation round and cuboidal eosinophilic cells and typical glomus cells (hematoxylin-eosin stain, original magnification X 100).
occur 10 to 15 years earlier than solitary tumors. Glomus tumors occur in males more frequently than in females. 3 Microscopic examination usually shows an encapsulated tumor with the parenchyma composed of a proliferation of epitheloid glomus cells. Masson H classified these tumors into three types according to the ratio of the cells: vascular type, solid type, and degenerative type. Enzinger and Weiss ~ also classified them into three types: glomus tumor proper, glomangioma, and glomangiomyoma. In our case, there was incomplete encapsulation of the tumor, which corresponded to the solid type according to Masson's classification and the glomus tumor proper according to Enzinger's classification. In general, the parotid gland is a common site for epithelial tumors; most are benign adenomas such as pleomorphic adenoma and basal cell adenoma. Differential diagnosis of glomus tumors from adenomas in
FIGURE 10. The cytoplasm of the glomus cells is positive for vimentin (vimentin stain, original magnification x 100).
833
FIGURE 11. The stain for VIII-related antigen is positive in the endothelial cells surrounding the vascular channels (VIII = related antigen stain, original magnification x 100).
the parotid gland is necessary clinically and histopathologically. In addition, glomus tumors are morphologically similar to hemangiopericytomas. Therefore we conducted immunohistochemical examinations to differentiate the tumor from any type of adenoma and hemangiopericytoma. The immunohistochemical profile of glomus tumors, wherever they are located, includes positive staining for vimentin and muscle-specific actin. Immunostaining for high and low-molecular weight cytokeratins, desmins, myoglobin, S-100 protein, neurofilaments, and factor VIII-related antigen is negative in glomus cells. However, the factor VIII-related antigen stain is positive in the endothelial cells surrounding the vascular channels. Staining for S-100 protein and neurofilaments reveals the presence of nerve fibers in the stroma of the tumor. Hemangiopericytoma stains positively for factor VIII-related antigen and adenomas stain positively for epithelial markers. 4'5 In this case, we found that epithelial markers and factor VIII-related antigen were negative, indicating that the tumor was neither a hemangiopericytoma nor any type of adenoma. This finding is in line with other reports on glomus tumors. 1,4-6 Usually, the glomus tumor is characterized by pain, 9 and our patient experienced such symptoms. Because the patient had pain in the TMJ area, she was inappropriately diagnosed as having TMJ arthrosis. Because the symptoms of TMJ arthrosis can be similar to those of a glomus tumor, misdiagnosis is possible. Proper diagnosis requires considerable care, and should be based on data from magnetic resonance imaging, double-contrast arthrotomography, computerized tomographic scanning, and/or arthroscopy of the TMJ. Glomus tumor can be cured by simple excision as long as the excision is complete. We performed the operation in the same manner as for a parotid gland lesion, excising the tumor and leaving the facial nerve
834
GLANDULAR ODONTOGENIC CYST
intact. No evidence of recurrence has been seen to date.
5. 6.
References 7. 1. Enzinger FM, Weiss SW: Soft Tissue Tumors. St Louis, MO, Mosby, 1983, pp 450-460 2. Tsuneyoshi M, Enjoji M: Glomus tumor; A clinicopathologic and electron microscopic study. Cancer 50:1601, 1982 3. Moody GH, Myskow M, Musgrove C: Glomus tumor of the lip; a case report and immunohistochemical study. Oral Surg 62:312, 1986 4. Garcia-Prats MD, Sotelo-Rodriguez MT, Ballestin C, et al: Glomus tumor of the trachea: Report of a case with microscopic,
8. 9. 10. 11.
ultrastructural and immunohistochemical examination and review of the literature. Histopathology 19:459, 1991 Zolt~inN: Differentiation markers in hemangiopericytoma. Cancer 69:133, 1992 Ficarra G, Merrel PW, Johnston WH, et al: Intraoral solitary glomus tumor (glomangioma); case report and literature review. Oral Surg 62:306, 1986 Makino Y: A clinicopathological study on soft tissue tumors of the head and neck. Acta Pathol Jpn 29:389, 1979 Shugart RR, Soule EH, Johnson EW: Glomus tumor. Surg Gynecol Obstet 117:334, 1963 Tokiba S, Sato A, Sakamaki H, et al: A case of glomus tumor arising in the mandibular gingiva. Jpn J Oral MaxiUofac Surg 36:2295, 1990 Goodman TF,. Abele DC: Multiple glomus tumors; A clinical and electron microscopic study. Arch Dermatol 103:11, 1971 Masson P: Le glomus neuromyo-arteiel des regions tactiles et ses tumeurs. Lyon Chir 21:257, 1924
J Oral MaxillofacSurg 53:834-837, 1995
Glandular Odontogenic Cyst of the Maxilla: Report of Case PANAGIOTA ECONOMOPOULOU, DiPDS, MSc, DRDent,* AND ANNA PATRIKIOU, DBPDS, DRDentlAlthough Gardner ~ first suggested in 1984 that the glandular odontogenic or sialo-odontogenic cyst was a distinct clinicopathologic entity, consensus on terminology, histogenesis, and nature of the lesion remain to be achieved. The first relevant publications, presenting a total of 10 cases, were those of Padayachee and Van Wyk, 2 and Gardner et al. 3 Since then, eight additional cases have appeared in the English language literature. 4-s On this basis, the occurrence of the glandular odontogenic cyst might be considered uncommon. However, the cyst may not be as rare, because various other investigators also claim that they have come across typical examples, although they fail to provide precise data besides radiographs and/or photomicrographs. 9'~°Furthermore, it is entirely possible that cases diagnosed as central mucoepidermoid carcinoma
or lateral periodontal cyst might be reviewed as examples of glandular odontogenic cyst. 9,u Because of the lack of sufficient clinicopathologic information to elucidate the natural history of this enigmatic cyst, reporting of new cases should be encouraged. The following report describes a case of maxillary glandular odontogenic cyst.
Report of Case A 32-year-old white man was referred in May 1993 to the oral and maxillofacial surgery clinic because of a tender
Received from the Department of Oral Surgery and Pathology, School of Dentistry, University of Athens, Athens, Greece. * Assistant Professor in Oral Pathology. t Associate Professor in Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Economopoulou: 6 Serron St, Halandri 152 31, Athens, Greece. © 1995 American Association of Oral and Maxillofacial Surgeons 0278-2391/95/5307-001853.00/0
FIGURE 1. Preoperative panoramic radiograph showing a large irregularly outlined radiolucency in the anterior maxilla.