Case Report/Clinical Techniques
Gingival Squamous Cell Carcinoma Mimicking a Dentoalveolar Abscess: Report of a Case Jang-Jaer Lee, DDS, MS, Shi-Jung Cheng, DDS, MS, Sze-Kwan Lin, DDS, MS, PhD, Chun-Pin Chiang, DDS, DMSc, Chuan-Hang Yu, DDS, MS, and Sang-Heng Kok, DDS, PhD Abstract Because of its close proximity to teeth and periodontium, gingival squamous cell carcinoma (SCC) can sometimes mimic tooth-related benign inflammatory conditions, resulting in misdiagnosis. In this study we report a case of gingival SCC that mimicked a dentoalveolar abscess of endodontic origin in its early presentation. The course and treatment of this case is discussed and a brief review of the literature is presented. Because many patients with gingival SCC visit dentists as their initial professional contact, it is hoped that the case can serve as a reminder for dentists to keep the possibility of carcinoma in mind when examining intraoral lesions. (J Endod 2007;33:177–180)
Key Words Dentoalveolar abscess, gingival squamous cell carcinoma, misdiagnosis, pulp necrosis
From the Department of Dentistry, Division of Oral and Maxillofacial Surgery, School of Dentistry, National Taiwan University Medical Center, Taipei, Taiwan. Address requests for reprints to Dr. Sang-Heng Kok, Department of Dentistry, Division of Oral and Maxillofacial Surgery, National Taiwan University Hospital, No. 1 Chang-Te Street, Taipei, Taiwan, 10016. E-mail address:
[email protected]. 0099-2399/$0 - see front matter Copyright © 2007 by the American Association of Endodontists. doi:10.1016/j.joen.2006.08.005
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O
ral squamous cell carcinoma (SCC) is a common malignancy worldwide (1). The main risk factors for oral SCC are tobacco usage, alcohol consumption (2, 3), and chewing of areca (betel) quid (4 – 6). The most common sites for SCC lesions within the oral cavity are the tongue and the floor of the mouth in Western populations, but buccal SCC is also one of the common cancers in South to Southeast Asia, possibly related to the practice of chewing areca quid (2, 4, 7, 8). In contrast, SCC of the gingiva is less common. In Taiwan, about 7% of oral SCC lesions occur on the gingiva and a predilection toward males is prominent, possibly because of the fact that most areca quid chewers are men (7, 9 –11). Like SCC lesions in other sites within the oral cavity, gingival SCC is often preceded by long-standing leukoplakia. It usually appears as an exophytic mass with a granular, papillary, or verrucous surface or presents as an ulcerative lesion (9). Gum pain is the most predominant symptom, but the early stages may be asymptomatic (3, 12). It generally occurs in the premolar and molar regions, the lower jaw being more often affected than the upper, and usually arises in edentulous areas but may also occur in areas where teeth are present (3, 12, 13). Gingival SCC is one of the most serious malignancies of the oral cavity and early invasion of underlying bone is common (14). Because of its close proximity to teeth and periodontium, many patients with gingival SCC visit dentists as their first professional contact. Therefore, dentists play an important role in the early detection and management of gingival SCC. In this study, we report a case of gingival SCC that mimicked a dentoalveolar abscess of endodontic origin in its early presentation. It is hoped that the case can serve as a reminder to keep the possibility of carcinoma in mind when examining intraoral lesions.
Case Report A 46-year-old housewife presented to the screening clinic of the Department of Dentistry, National Taiwan University Hospital with the chief complaint of a gumboil on the labial gingiva of the left maxillary incisor region. The lesion was not preceded by leukoplasia or erythroplasia. The history of her problem can be traced back to 3 months earlier when she was taking a meal and accidentally bit into some hard materials with her left maxillary lateral incisor (tooth #10). Severe pain on tooth #10 was noted at that time, but the symptom gradually improved, and she did not pay much attention to that after a few days. About 2 months after the accident, she found a painless swelling on the labial gingiva of tooth #10, and blood-tinted discharge from the swelling was sometimes noticed. The lesion was observed for 1 month and showed no sign of resolution. Her past medical history was noncontributory, and she denied any habitual usage of tobacco, alcohol, or areca quid. However, she did not have a history of malignant tumors in her family. Clinical examination revealed an erythematous papule on the labial attached gingiva of teeth #9 to #10 near the mucogingival junction (Fig. 1). The lesion was about 6 ⫻ 6 mm in size, soft, and smooth-surfaced. Its boundary was not clearly defined, but no marginal induration was noted. On palpation, serosanguineous discharge from a small crater at the distal margin of the lesion was found (Fig. 1). Her oral hygiene was fair, but early gingivitis in the upper anterior area was noted, especially around teeth #8 and #9, which were restored with splinted metal-ceramic crowns (Fig. 1). No pocket formation or tooth mobility in that area was found. Slight tenderness of tooth #10 was induced by percussion, and pulp vitality test showed that the tooth was nonvital.
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Figure 1. Clinical examination showing a soft, smooth-surfaced, and erythematous papule on the labial gingiva of teeth #9 to #10 near the mucogingival junction.
Periapical radiography revealed that tooth #9 was endodontically treated, but no periodontal or periapical bone destruction around teeth #9 and #10 could be defined (Fig. 2). No root resorption, root fracture, or widening of the periodontal ligament was noted for these teeth on the radiograph (Fig. 2). With a tentative diagnosis of dentoalveolar abscess secondary to pulp necrosis of tooth #10, the patient was referred to the Department of Endodontics for treatment. Pulp necrosis of tooth #10 was confirmed during root canal therapy, but no pus was obtained after incision of the gingival lesion, and no perforation of the labial bone plate could be detected by dental probe exploration. After two visits for root canal therapy, the lesion showed no sign of improvement. The patient was
Figure 2. Periapical radiography showing no periodontal or periapical bone destruction around teeth #9 and #10.
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Figure 3. (A) Histopathological examination of the biopsy specimen. Note hyperchromatic and pleomorphic cells devoid of intercellular bridges and keratin formation (H&E, 100⫻) (B) Immunohistochemical study of the tumor cells demonstrating strong positivity for cytokeratin AE1/AE3 (100⫻).
then referred to the Department of Oral and Maxillofacial Surgery for further evaluation and incisional biopsy was then performed. Microscopic examination showed that the lesion was covered by stratified squamous epithelium. Hyperchromatic and pleomorphic cells forming strands and islands infiltrating the connective tissue were found. Mitoses were noted occasionally. Differentiation of the cells was not evident as intercellular bridges and keratin formation were absent (Fig. 3A). Immunohistochemical study revealed that the cells were strongly positive for cytokeratin AE1/AE3 (Fig. 3B). The pathological diagnosis was poorly differentiated SCC. The patient was admitted and metastatic workup showed no evidence of regional or distant metastasis. According to the 2002 AJCC staging criteria (15), the patient had clinical T1N0M0, stage I disease. She then accepted surgical treatment, which included excision of the tumor via partial maxillectomy from teeth #7 to #11 (Fig. 4) and prophylactic supraomohyoid neck dissection. Intraoperative frozen-section analysis found that two of the three submitted lymph nodes (all movable, soft, and ⬍1 cm in diameter) from the upper third of the deep cervical chain (level IIb) contained metastatic tumor; therefore, a radical neck dissection was performed subsequently in the same operation (Fig. 5). Histopathological study of the surgical specimens confirmed that the lesion was a poorly differentiated SCC. The section margins were free, and there was no evidence of bone invasion. The two metastatic lymph nodes discovered on frozen-section were found to have extracapsular tumor spread (Fig. 6). However, all of the other dissected nodes (a total of 65) were negative for tumor. Pathological stage of the patient’s disease proved to be T1N2b, stage IV. The patient’s wound healing was uneventful, and 1 month after the surgery she received concomitant chemoradiotherapy (CCRT) that included irradiation of
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Figure 4. The specimen of partial maxillectomy extending from teeth #7 to #11.
Figure 6. Histopahological analysis of a metastatic lymph node showing extracapsular tumor spread (H&E, 40⫻).
66 Gy in 33 fractions and 45 mg cisplatin infusion every week for 6 weeks. No evidence of recurrence was found after a 6-month follow-up.
periapical bone destruction or widening of periodontal membrane. No pus discharge or perforation of the labial bone plate could be detected. The clinical appearance of oral SCC is typically described as ulcerative swellings and tumors with a granular, papillary, or verrucous surface. However, early disease can also present as an innocuous erythematous lesion with a smooth surface, occasionally resulting in misdiagnosis by clinicians. Shafer and Waldron (20) found that 91% of lesions clinically described as erythroplasia represented severe dysplasia, carcinoma in situ, or carcinoma. This corresponds with the study by Mashberg et al. (21, 22) in which more than 90% of 158 asympotomatic early squamous cell carcinomas had an erythroplastic component, and they felt that this is the earliest visible sign of an asymptomatic lesion. Metastases to cervical lymph nodes were reported in 25 to 35% of gingival SCC patients, with a higher rate for larger primary tumors (12, 23, 24). Another unusual feature of our case is that lymph node metastasis was found for such a small primary tumor with no evidence of bone invasion. The metastatic nodes were not enlarged, but extracapsular tumor spread was noted by histopathological analysis. The aggressive behavior may relate to its degree of differentiation. Poorly differentiated oral SCC is uncommon (3, 25, 26) but more likely to have early cervical metastasis (23, 27). Surgery, including excision of primary tumor and neck dissection, is the major treatment modality for gingival SCCs. The 5-year survival rates for gingival SCCs ranged from 77% for stage I to 24% for stage IV diseases (3). Regional metastasis usually denoted a poorer prognosis (23, 24). Postoperative CCRT such as that performed in our patient can effectively increase the rate of 5-year survival and decrease the chance of cervical recurrence and metastasis to distant organs (28). In a 20-year survey conducted by Cady and Catlin (12), ⬎60% of gingival cancer patients were initially seen by dentists. In another study of 595 patients with oral cancer, 8% were gingival carcinomas, and of these patients, 52% saw a dentist as their initial professional contact (29). However, only half of the patients in Cady and Catlin’s study were properly handled by prompt referral to a treatment center. One-third had teeth extracted before referral, and one-sixth had various therapies applied for periods ranging from 1 month to ⬎1 year before the patients were referred for treatment of carcinoma, as a result of lack of suspicion that the lesion was cancer (12). Soo and associates (3) also found that in 384 patients with carcinomas of the gingiva, dental extraction had been performed before diagnosis in 20% of the cases. Obviously, dentists should raise their level of suspicion when examining oral lesions to avoid misdiagnosis of serious diseases.
Discussion Gingival SCCs with atypical clinical appearance have been reported by a few authors (Table 1) (16 –19). Most of the reported cases had an initial presentation mimicking periodontal diseases, making correct diagnoses difficult. The affected sites in these cases were often not the usual site for gingival SCC, the lower posterior region. Their common clinical features were erythematous gingiva with radiographic evidence of alveolar bone destruction. However, gingival SCC with an initial presentation resembling dentoalveolar abscess of endodontic origin has seldom been mentioned in the English literature. The case reported in this article was a middle-aged housewife having no habits of smoking, alcohol use, or areca quid chewing. The smooth-surfaced erythematous gingival swelling with serosanguineous discharge was not unlike a dentoalveolar abscess, considering that it was confounded by a history of occlusal trauma to a tooth in that region. In fact, the pulp of the lateral incisor was found to be necrotic during root canal therapy. Furthermore, anterior maxilla was not a common location for SCC. However, clinical features not typical for a periapical abscess were also noted. Radiographic examination failed to reveal any
Figure 5. Classical radical neck dissection on left neck.
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Case Report/Clinical Techniques TABLE 1. Previous case reports of atypical gingival squamous cell carcinoma Case Report Gallagher et al., 1984 (16) Kirkham et al., 1985 (17)
Age/ Gender 53/M 37/F
Oral Habits
Syptoms and Signs
Location
Lesion Size
Radiographic Findings
Drinking and smoking N/A
Painful and erythematous gingiva, easily bled Painless
#22 to 26
N/A
#14
5 mm
Loss of alveolar bone N/A
#22 to 23
N/A
Subgingival external root resorption of #24
#30
N/A
Angular loss of alveolar bone
Craig et al., 1989 (18)
70/M
Occasionally drank
Enlarged and erythematous labial marginal gingival
Heller et al., 1991 (19)
66/F
Smoking
Abscess (red tissue) with mild pain and 9 mm pocket, no ulceration/ induration
Clinical Diagnosis Periodontal disease 1. Periodontal abscess 2. Irritation fibroma 3. Traumatic ulcer 4. Neurofibroma Periodontal disease
Periodontal abscess
Pathological Diagnosis and Findings WD-SCC WD-SCC
1. MD-SCC 2. No involvement of underlying bone or area of #24 root. resorption WD-SCC
N/A, not available; WD, well-differentiated; MD, moderately differentiated; SCC, squamous cell carcinoma.
In conclusion, dentists play an important role in early detection of gingival cancer. If an erythroplastic lesion near the teeth failed to heal 2 weeks after the removal of all obvious causative factors, the dentist should be alerted and take a biopsy to establish a definitive diagnosis. Delay in diagnosis and treatment or repeated manipulation of these lesions may adversely affect the prognosis.
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