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Calcifying epithelial odontogenic tumor of the maxilla with ulcerative stomatitis: A case report
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British Journal of Oral and Maxillofacial Surgery 47 (2009) 222–224
Short communication
Calcifying epithelial odontogenic tumor of the maxilla with ulcerative stomatitis: A case report Hiroyuki Nakano a,∗ , Yoshiyuki Ota b , Yoshiaki Yura b a
Department of Oral and Maxillofacial Surgery, Yamaguchi University Graduate School of Medicine, 1-1-1 Minamikogushi, Ube, Yamaguchi, Japan b Department of Oral and Maxillofacial Surgery II, Osaka University Graduate School of Dentistry, 1-8 Yamadaoka, Suita, Osaka, Japan Accepted 27 July 2008 Available online 14 September 2008
Abstract Calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic tumor, known as Pindborg tumor. Although ulcer formation was reported in one previously involving the peripheral maxilla, such change of the overlying mucosa has been reported in intraosseous CEOT. We report maxillary CEOT in a patient who complained of spontaneous pain due to extensive ulcer formation of the oral mucosa. © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Calcifying epithelial odontogenic tumor; Ulcerative mucositis, Extirpation
Case report A 33-year-old woman complained of a gingival swelling and pain in the left maxilla. She had recognized palatal swelling on the left side 3 years earlier. The swelling had gradually increased over the 3 years without any pain or discomfort. However, starting from 2 days before the patient consulted our hospital, she experienced pain in the left maxilla and a fever of 39.7 centigrade. On clinical examination, there was a swelling from the left first molar to the third molar tooth region. The overlying palato-gingival mucosa was reddish and ulcer formation was observed (Fig. 1). Panoramic radiograph demonstrated a well-demarcated radiopaque mass in the left maxilla extending from the maxillary sinus to the palate. Impacted second molar tooth was included in the lesion. Computed tomography also demonstrated maxillary tumor with calcification and impacted tooth (Fig. 2). The patient was treated with antibiotics and anti-inflammatory drugs. After one week, ulcerative mucositis disappeared. ∗
Corresponding author. +81 836 22 2299. E-mail address: [email protected] (H. Nakano).
The histopathological diagnosis of the biopsy material taken from the palate was CEOT. Under general anesthesia, tumor extirpation and excision of the overlying mucosa was performed. Tumor was easily extirpated from the surrounding bone and the adjacent bone surface was removed for a depth of several millimetres (Fig. 3). The postoperative course was uneventful. The patient has been checked regularly for more than 6 years without any sign of recurrence.
Discussion CEOT is a rare benign odontogenic tumor, described in 1955 by Pindborg and now known as Pindborg tumor. This lesion represent about 0.4–3% of all odontogenic tumors.1–4 Franklin and Pindborg2 examined 113 cases and found that it occurred frequently between 30 and 50 years of age with no gender predilection. The posterior mandible is the most frequently involved site and approximately 25% occurred in the maxilla.3
0266-4356/$ – see front matter © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2008.07.201
H. Nakano et al. / British Journal of Oral and Maxillofacial Surgery 47 (2009) 222–224
Fig. 1. Palatal appearance of CEOT in the maxilla.
Fig. 2. Computed tomographs showing a maxillary tumor with calcification and impacted tooth.
223
Until the reported by Pindborg, CEOT had been reported under various names such as ameloblastoma of an unusual type with calcification. Indeed, CEOT behaves in a manner similar to ameloblastoma; it can recur locally and does not metastasize, although 3 cases have shown features of malignancy.5–8 Most CEOTs are recognized as painless and slow-growing expansion of the jaws and radiolucent lesions on routine radiograms. Peripheral cases account for 5% of CEOT and only one case was reported to have formed an ulcer over the tumor, but the detailed features were not described.9 There have not been any such mucosal changes described in intraosseous CEOT. In the present case, the overlaying mucosa was ulcerated, accompanied by spontaneous pain, and the first impression of the lesion was ulcerative stomatitis. However, panoramic radiograph and Computed tomography demonstrated the presence of tumor under the inflamed palatal mucosa. Although the tumor showed extensive growth in the oral cavity, it arose from the maxilla. In this aspect, we have not seen any previous report describing intraosseous CEOT with extensive ulcer formation. When we removed the tumor, it could be easily detached from the bone, but the border between the tumor and palatal mucosa was unclear. Thus, the mucosa was excised with the tumor. Histopathologically, the tumor showed typical features of CEOT, with calcifying body adjacent to the epithelium. No apparent infiltration of inflammatory cells was observed in the sections of the biopsy material, because the patient had been treated with antibiotic prior to biopsy. It is not known why a strong inflammatory reaction occurred in the mucosa in this patient. The mucosa might have shown necrotic change because of expansive growth of the tumor and became sensitive to inflammatory agents. Furthermore, it is suspected that ulcerative stomatitis is caused by bacterial infection of the palatal mucosa, because anti-inflammatory treatment with antibiotics was effective.
References
Fig. 3. Histological photographs showing numerous concentric calcified bodies in and adjacent to the epithelium (haematoxylin and eosin stain, magnification ×200).
1. Philipsen HP, Reichart PA. Calcifying epithelial odontogenic tumours: Biological profile based on 181 cases from the literature. Oral Oncol 2000;36:17. 2. Franklin CD, Pindborg JJ. The calsifying epithelial odontogenic tumors. A review and analysis of 113 cases. Oral Surg Oral Med Oral Pathol 1976;42:753. 3. Pindborg JJ, Vedtofte P, Reibel J, Praetorius F. The calcifying epithelial odontogenic tumor: A review of recent literature and report of case. APMIS Suppl 1991;23:152. 4. Pindborg JJ. Calcifying epithelial odontogenic tumors. AMPS 1955;111:7. 5. Veness MJ, Morgan G, Collins AP, Walker DM. Calcifying epithelial odontogenic (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 2001;23:692–6. 6. Basu MK, Matthews JB, Shear AJ, Browne RM. Calcifying epithelial odontogenic tumor: a case showing features of malignancy. J Oral Pathol 1984;13:310–9.
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H. Nakano et al. / British Journal of Oral and Maxillofacial Surgery 47 (2009) 222–224
7. Goldenberg D, Scinbba J, Koch W, Tufano RP. Malignant odontogenic tumors: a 22-year experience. Laryngoscope 2004;114:1770–4. 8. Cheng YS, Wright JM, Walstad WR, Finn MD. Calcifying epithelial odontogenic tumor showing microscopic features of potential malig-
nant behavior. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:287–95. 9. Ai-Ru L, Zhen L, Jian S. Calcifying epithelial odontogenic tumors: A clinicopathologic study of nine cases. J Oral Pathol 1982;11:399–406.
British Journal of Oral and Maxillofacial Surgery 47 (2009) 222–224
Short communication
Calcifying epithelial odontogenic tumor of the maxilla with ulcerative stomatitis: A case report Hiroyuki Nakano a,∗ , Yoshiyuki Ota b , Yoshiaki Yura b a
Department of Oral and Maxillofacial Surgery, Yamaguchi University Graduate School of Medicine, 1-1-1 Minamikogushi, Ube, Yamaguchi, Japan b Department of Oral and Maxillofacial Surgery II, Osaka University Graduate School of Dentistry, 1-8 Yamadaoka, Suita, Osaka, Japan Accepted 27 July 2008 Available online 14 September 2008
Abstract Calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic tumor, known as Pindborg tumor. Although ulcer formation was reported in one previously involving the peripheral maxilla, such change of the overlying mucosa has been reported in intraosseous CEOT. We report maxillary CEOT in a patient who complained of spontaneous pain due to extensive ulcer formation of the oral mucosa. © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Calcifying epithelial odontogenic tumor; Ulcerative mucositis, Extirpation
Case report A 33-year-old woman complained of a gingival swelling and pain in the left maxilla. She had recognized palatal swelling on the left side 3 years earlier. The swelling had gradually increased over the 3 years without any pain or discomfort. However, starting from 2 days before the patient consulted our hospital, she experienced pain in the left maxilla and a fever of 39.7 centigrade. On clinical examination, there was a swelling from the left first molar to the third molar tooth region. The overlying palato-gingival mucosa was reddish and ulcer formation was observed (Fig. 1). Panoramic radiograph demonstrated a well-demarcated radiopaque mass in the left maxilla extending from the maxillary sinus to the palate. Impacted second molar tooth was included in the lesion. Computed tomography also demonstrated maxillary tumor with calcification and impacted tooth (Fig. 2). The patient was treated with antibiotics and anti-inflammatory drugs. After one week, ulcerative mucositis disappeared. ∗
Corresponding author. +81 836 22 2299. E-mail address: [email protected] (H. Nakano).
The histopathological diagnosis of the biopsy material taken from the palate was CEOT. Under general anesthesia, tumor extirpation and excision of the overlying mucosa was performed. Tumor was easily extirpated from the surrounding bone and the adjacent bone surface was removed for a depth of several millimetres (Fig. 3). The postoperative course was uneventful. The patient has been checked regularly for more than 6 years without any sign of recurrence.
Discussion CEOT is a rare benign odontogenic tumor, described in 1955 by Pindborg and now known as Pindborg tumor. This lesion represent about 0.4–3% of all odontogenic tumors.1–4 Franklin and Pindborg2 examined 113 cases and found that it occurred frequently between 30 and 50 years of age with no gender predilection. The posterior mandible is the most frequently involved site and approximately 25% occurred in the maxilla.3
0266-4356/$ – see front matter © 2008 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2008.07.201
H. Nakano et al. / British Journal of Oral and Maxillofacial Surgery 47 (2009) 222–224
Fig. 1. Palatal appearance of CEOT in the maxilla.
Fig. 2. Computed tomographs showing a maxillary tumor with calcification and impacted tooth.
223
Until the reported by Pindborg, CEOT had been reported under various names such as ameloblastoma of an unusual type with calcification. Indeed, CEOT behaves in a manner similar to ameloblastoma; it can recur locally and does not metastasize, although 3 cases have shown features of malignancy.5–8 Most CEOTs are recognized as painless and slow-growing expansion of the jaws and radiolucent lesions on routine radiograms. Peripheral cases account for 5% of CEOT and only one case was reported to have formed an ulcer over the tumor, but the detailed features were not described.9 There have not been any such mucosal changes described in intraosseous CEOT. In the present case, the overlaying mucosa was ulcerated, accompanied by spontaneous pain, and the first impression of the lesion was ulcerative stomatitis. However, panoramic radiograph and Computed tomography demonstrated the presence of tumor under the inflamed palatal mucosa. Although the tumor showed extensive growth in the oral cavity, it arose from the maxilla. In this aspect, we have not seen any previous report describing intraosseous CEOT with extensive ulcer formation. When we removed the tumor, it could be easily detached from the bone, but the border between the tumor and palatal mucosa was unclear. Thus, the mucosa was excised with the tumor. Histopathologically, the tumor showed typical features of CEOT, with calcifying body adjacent to the epithelium. No apparent infiltration of inflammatory cells was observed in the sections of the biopsy material, because the patient had been treated with antibiotic prior to biopsy. It is not known why a strong inflammatory reaction occurred in the mucosa in this patient. The mucosa might have shown necrotic change because of expansive growth of the tumor and became sensitive to inflammatory agents. Furthermore, it is suspected that ulcerative stomatitis is caused by bacterial infection of the palatal mucosa, because anti-inflammatory treatment with antibiotics was effective.
References
Fig. 3. Histological photographs showing numerous concentric calcified bodies in and adjacent to the epithelium (haematoxylin and eosin stain, magnification ×200).
1. Philipsen HP, Reichart PA. Calcifying epithelial odontogenic tumours: Biological profile based on 181 cases from the literature. Oral Oncol 2000;36:17. 2. Franklin CD, Pindborg JJ. The calsifying epithelial odontogenic tumors. A review and analysis of 113 cases. Oral Surg Oral Med Oral Pathol 1976;42:753. 3. Pindborg JJ, Vedtofte P, Reibel J, Praetorius F. The calcifying epithelial odontogenic tumor: A review of recent literature and report of case. APMIS Suppl 1991;23:152. 4. Pindborg JJ. Calcifying epithelial odontogenic tumors. AMPS 1955;111:7. 5. Veness MJ, Morgan G, Collins AP, Walker DM. Calcifying epithelial odontogenic (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 2001;23:692–6. 6. Basu MK, Matthews JB, Shear AJ, Browne RM. Calcifying epithelial odontogenic tumor: a case showing features of malignancy. J Oral Pathol 1984;13:310–9.
224
H. Nakano et al. / British Journal of Oral and Maxillofacial Surgery 47 (2009) 222–224
7. Goldenberg D, Scinbba J, Koch W, Tufano RP. Malignant odontogenic tumors: a 22-year experience. Laryngoscope 2004;114:1770–4. 8. Cheng YS, Wright JM, Walstad WR, Finn MD. Calcifying epithelial odontogenic tumor showing microscopic features of potential malig-
nant behavior. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:287–95. 9. Ai-Ru L, Zhen L, Jian S. Calcifying epithelial odontogenic tumors: A clinicopathologic study of nine cases. J Oral Pathol 1982;11:399–406.