Polypoid mass of the gingiva

Vol. 88 No. 2 August 1999

ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY

CLINICOPATHOLOGIC CONFERENCE

Editor: John R. Kalmar

Polypoid mass of the gingiva Kenji Izumi, DDS, PhD,a Tamio Nakajima, DDS, PhD,b Takeyasu Maeda, DDS, PhD,c Tarou Irie, DDS, PhD,d Masashi Murata, DDS, PhD,d and Takashi Saku, DDS, PhD,e Niigata, Japan NIIGATA UNIVERSITY

(Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;88:117-21)

A 61-year-old Japanese man was referred because of a rapidly growing gingival swelling in his left maxillary premolar region. The clinical examination showed a loss of the nasolabial fold on the left side of the face and enlarged fixed cervical lymph nodes bilaterally. Intraorally, there was a well-circumscribed polypoid 3.5 × 2.5-cm mass located on the buccal aspect of the gingiva of the left maxillary canine and extending to the left first molar (Fig 1). The lesion had a granular surface, was partially covered with yellowish-grey membrane, and was rubbery on palpation. Computed tomography showed a mass with partial destruction of the alveolar bone and the base of the maxillary sinus. Enlarged lymph nodes with central lucency were noted bilaterally in the submandibular and upper neck regions. Serum carcinoembryonic antigen (CEA) was 9.3 ng/mL (normal range, 0.7-4.2 ng/mL).

DIFFERENTIAL DIAGNOSIS The rapid clinical onset and evidence of lymphadenopathy with fixation were features strongly suggestive of malignancy. The differential diagnosis of a malignant process that could manifest itself as a large aAssistant

Professor, First Department of Oral and Maxillofacial Surgery, School of Dentistry. bProfessor, First Department of Oral and Maxillofacial Surgery, School of Dentistry. cProfessor, Second Department of Oral Anatomy, School of Dentistry. dResident, Department of Pathology, School of Dentistry. eProfessor, Department of Pathology, School of Dentistry. Received for publication Nov 6, 1998; returned for revision Feb 20, 1999; accepted for publication Apr 16, 1999. Copyright © 1999 by Mosby, Inc. 1079-2104/99/$8.00 + 0 7/14/99405

Fig 1. Photograph shows well-defined, large tumor with necrotic surface involving left maxilla.

gingival mass included squamous cell carcinoma, sarcomas, malignant salivary gland tumors, and metastatic tumors. Considering the location, a lesion of maxillary sinus origin was also considered.

DIAGNOSIS Histopathologically, the tumor was composed of solid tumor cell nests of varying sizes and fibrous connective tissue stroma (Fig 2, A). These cells were

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Fig 2. A, Photomicrograph shows solid tumor nests consisting of polygonal and clear cells containing large, pleomorphic, and clear nuclei and small spindle-shaped cells (top, left) in fibrous connective tissue stroma (hematoxylin-eosin, original magnification ×80). B, Photomicrograph shows intracytoplasmic, PAS–positive, diastase-resistant materials (arrows) in solid tumor nest (PAS, original magnification ×600).

polygonal, had foamy or ground glass–like cytoplasm, and had large, pleomorphic, and occasionally clear nuclei with conspicuous nucleoli. Histochemically, dot-like, periodic acid–Schiff (PAS)–positive, diastaseresistant material was found within the cytoplasm of the tumor cells (Fig 2, B). Immunohistochemically, the tumor cells showed a weakly positive reaction for CEA but were negative for keratin, vimentin, desmin, and S100 protein. Ultrastructurally, scattered, intercellular,

canaliculus-like structures sealed with intermediate junctions were noted. Microprojections protruding into the lumen were associated with actin cores (Fig 3). In addition, glycocalyceal bodies were observed within the lumen. Chest radiography and radioisotope scintigraphy with 67Ga revealed no evidence of a distant primary or systemic metastasis. Radical resection of the tumor combined with 2-stage radical neck dissection after a

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Fig 3. Electron photomicrograph shows intercellular, canaliculus-like structure sealed with intermediate junctions (arrowheads). Microprojections with actin cores are seen in luminal portion of tumor cells fronting lumen (L) of intercellular, canaliculus-like structure; in addition, glycocalyceal bodies (arrow) are observed in lumina (osmium tetroxide postfixation and uranyl acetate/lead citrate, original magnification ×7000).

single course of induction chemotherapy (cis-platinum, 5-fluorouracil, and bleomycin) was planned. At the initiation of treatment, serum CEA had reached 33.8 ng/mL. A slight reduction in tumor size was noted after chemotherapy, and the mass was removed by resection of the left maxilla with left radical neck dissection. In the surgical specimen, the bulk of the lesion was composed of solid sheets of plump cells, as in the biopsy specimen. However, small foci of apparent glandular structures with papillary projections into luminal spaces were found in areas adjacent to the maxillary sinus (Fig 4, A and B). One cervical and 2 submandibular lymph nodes were positive for metastatic disease; in these nodes, focal papillotubular growth was recognized among solid tumor nests. Final diagnosis. Primary adenocarcinoma, not otherwise specified (NOS), although a metastatic lesion could not totally be excluded.

FOLLOW-UP The right radical neck dissection was canceled because metastatic involvement of the right fifth cervical and lumbar vertebra was found. Serum CEA and CA 19-9 levels increased dramatically after surgery. Colonoscopy, abdominal ultrasonography, and aerodigestive fiberoscopy revealed no abnormalities. Palliative radiation with 60Co to a total dose of 30 Gy to the cervical and lumbar vertebrae was performed. Bilateral pleural effusion and interstitial reticular shadows of the lungs accompanied by cardiomegaly were noted on the chest radiograph. Six months after initial treatment, the patient died of respiratory failure. At autopsy, no residual tumor was found within the maxilla or the ipsilateral cervical lymph nodes. However, extensive metastases were identified in the lungs, liver, kidneys, and thoracic and lumbar mesenteric and inguinal lymph nodes. Microscopically, metastatic foci in the liver and lung showed focal tubu-

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Fig 4. A, Photomicrograph shows one frontal section of whole surgical specimen at left maxillary second premolar (PM). Tumor (T) forms polypoid mass at buccal aspect of maxillary gingiva. Main tumor nests incline toward oral cavity but partly extend into maxillary sinus with bony destruction. Maxillary buccal vestibule is not involved in tumor mass. Tumor nests predominantly show solid growth pattern. Portion enclosed by small square indicates only small focus showing papillotubular growth pattern (hematoxylineosin, original magnification ×8). B, Higher-magnification photomicrograph shows portion of Fig 4, A enclosed by small square (hematoxylin-eosin, original magnification ×20).

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loglandular differentiation (Fig 5), but solid nests without definite glandular architecture predominated. Because no other focus suggestive of a primary lesion was identified, these findings reaffirmed the gingiva as the most probable origin for this adenocarcinoma.

DISCUSSION Minor salivary gland tumors of the gingiva are uncommon, a fact that is attributed to the relative absence of minor salivary glands in the gingiva.1 However, several reports indicate that salivary gland structures and their associated lesions may occur in the gingiva.2,3 In the present case, origination from the adjacent vestibular mucosa could also be considered, especially inasmuch as minor salivary glands are more plentiful in this area. However, the clinical presentation and gross findings seemed to indicate that the polypoid mass arose from the buccal aspect of the maxillary gingiva, without obvious involvement of the buccal vestibule. Finally, an origin from the maxillary sinus could be considered. Because the clinical signs and symptoms were confined to the oral cavity, the clinical impression did not suggest initial sinus involvement. Furthermore, although destruction of the alveolar bone and sinus base were evident in the surgical specimen, tumor involvement of the maxillary sinus was not remarkable. Therefore, we felt that the evidence most strongly supported a buccal gingival origin for this neoplasm. The essential approach to making a diagnosis of adenocarcinoma, NOS, is by excluding other, more well-defined salivary gland tumors.4,5 However, some recognizable histologic criteria are emphasized for its diagnosis. Besides the absence of characteristics of any other type of salivary gland adenocarcinoma, it is important to identify the formation of glandular or ductlike structures and the presence of infiltrating growth with some degree of cytologic atypia. Some tumor cells may show pleomorphic and hyperchromatic nuclei with severe atypia and consist of solid nests of polygonal epithelial cells analogous to undifferentiated carcinoma.4,5 In the present case, histologic examination of the entire primary tumor revealed a limited yet recognizable papillotubular growth pattern that enabled us to distinguish it from undifferentiated carcinoma. Using the aforementioned criteria, we emphasize that adenocarcinoma, NOS, can be a valid and useful diagnostic category of salivary gland neoplasia. We thank Ms M. Minami and Messers K. Maeda and M. Hoshino for their technical assistance.

Fig 5. Photomicrograph shows metastatic focus of tumor cells from lung. Apparent tubuloglandular differentiation is observed (hematoxylin-eosin, original magnification ×125). REFERENCES 1. Gates GA. Malignant neoplasms of the minor salivary glands. New Engl J Med 1982;306:718-22. 2. Brannon RB, Houston GD, Wampler HW. Gingival salivary gland choristoma. Oral Surg Oral Med Oral Pathol 1986;61:185-8. 3. Moss-Salentijn L, Applebaum E. A minor salivary gland in human gingiva. Arch Oral Biol 1972;17:1373-4. 4. Dardick I. Adenocarcinoma, not otherwise specified (NOS). In: Dardick I, editor. Color atlas/text of salivary gland tumor pathology. New York: Igaku-Shoin; 1996. p. 244-6. 5. Ellis GL, Auclair PL. Adenocarcinoma not otherwise specified. In: Ellis GL, Auclair PL, editors. Atlas of tumor pathology: tumors of the salivary glands. Washington DC: Armed Forces Institute of Pathology; 1996. p. 175-83.

Reprint requests: Kenji Izumi, DDS, PhD First Department of Oral and Maxillofacial Surgery Niigata University School of Dentistry Gakkocho-dori 2-5274, Niigata City 951-8514 Japan