J Oral Maxillofac 50:1324-1326,
Surg 1992
Calcifying Epithelial Odontogenic Tumor (Pindborg Tumor): Report of Case MAURICIO ROBERTO
ANDRADE, DDS,* PAUL0 J. MEDEIROS, PRADO, DDS,* AND RENATO SAMPAIO,
porated within the lesion. There was cortical expansion in all directions, mainly toward the superior and lingual of the lesion (Fig 3). In consideration of the benign radiographic appearance and the size of the lesion, intraoral surgical excision with moderate curettage of the adjacent bone under general anesthesia was planned. The lesion was easily detached from the surrounding bone and was removed intact. There were no intraoperative complications. The surgical specimen measured 3 cm in diameter. The consistency was firm and the color was pale pink (Fig 4). Histologically there was a fibrous connective-tissue capsule surrounding epithelial tissue composed of polyhedral cells of different sizes, containing nuclei of varying forms and sizes, as well as an extracellular eosinophilic material, probably amyloid. There were concentric calcifications in the form of Liesegang rings (Fig 5). The histologic diagnosis was calcifying epithelial odontogenic tumor. The postoperative course was uneventful and the patient is being recalled regularly. There are no signs of recurrence 24 months after surgery.
The calcifying epithelial odontogenic tumor (CEOT) is a rare benign epithelial odontogenic lesion that comprises from 0.2% to 1% of all odontogenic tumors.’ Clinically it manifests as a bony lesion, accounting for the majority of cases (95%), or as the extremely rare peripheral type (5%). In the past a number of different names have been given to this lesion, such as calcifying ameloblastoma, ’ malignant odontoma,’ adamantoblastoma,2 adenoid adamantoblastoma,3 cystic complex odontoma,3 uncommon ameloblastoma with calcifications,’ and others. In 1956, Pindborg4 delineated this lesion as a distinct entity and named it the calcifying epithelial odontogenic tumor. In 1963, along with a comprehensive review of the literature, Shafer et al* suggested the eponym “Pindborg tumor.” The purpose of this article is to report an additional case of the CEOT and discuss important aspects of the radiographic appearance, histologic features, clinical behavior, and surgical management.
Discussion
Report of Case
It is commonly accepted that the CEOT has its origin from odontogenic epithelium.‘,3-6 Pindborg6 believes that the origin of this lesion is the enamel epithelial because there is a large number of cases associated with unerupted teeth. He also states that the origin of the peripheral lesions is still uncertain. There is agreement among several authors regarding the clinical features of this tumor. In the most comprehensive review of the literature by Franklin and Pindborg,’ 113 cases have been studied. There was a predilection for occurrence in the premolar-molar region of the mandible. There is no sex predilection, with the mean age of occurrence around 40 years. Most cases manifested as an intraoral or extraoral mass without other symptoms.‘,3,7,8 The varying radiographic features have been described as 1) pericoronal radiolucent; 2) pericoronal radiolucent with small radiopacities; 3) mixed radiolucent-radiopaque lesion not associated with an unerupted tooth; 4) “falling snow” appearance; 5) dense
A 3 1-year-old white man was seen for consultation because of an asymptomatic, 3-cm-diameter mass on the left mandibular alveolar ridge (Fig 1). The patient had first noticed the lesion 4 years before. The lesion was indurated and was expanded toward the lingual side. The color of the overlying oral mucosa was normal. No facial asymmetry could be noticed, and there was no regional lymphadenopathy. The panoramic radiograph showed features of a benign lesion (Fig 2). The lesion was radiolucent with well-defined borders and extended from the second premolar to the third molar region. There were several radiopaque masses and the crown of a molar tooth, probably the second molar, incorReceived from Rio de Janeiro State University, Brazil. * Staff Member, Oral and Maxillofacial Surgery Service. t Chief. Oral and Maxillofacial Sumerv _ _ Service; Director, Residency Training Program. $ Associated Professor; Head, Oral Pathology Service. Address correspondence and reprint requests to Dr Medeiros: Av NS Copacabana 680-9 10, 22.050 Rio de Janeiro, R3 Brazil. 0 1992 American
Association
of Oral and Maxillofacial
DDS, MS,t DDS, MS*
Surgeons
0278-2391/92/5012-0015$3.00/O
1324
1325
ANDRADE ET AL
FIGURE I. ridge.
The intraoral view showing the mass on the alveolar
radiopacity. Franklin and Pindborg’ found the two most common presentations to be pericoronal radiolucent and diffuse radiopacities in a radiolucent area. The histologic pattern is quite typical and very well defined. However, it may show subtle changes in cellular pattern related to the occurrence of amyloid and calcifications.‘*5,9-” No author believes the CEOT has a malignant biologic behavior. Some authors believe that the safest treatment consists of a wide marginal resection.’ However, some cases have been treated by simple surgical excision.‘q7.‘2 Sadeghi and HoperI believe that the treatment for the CEOT should be guided by the size of the lesion, its location, and the histologic features. According to these authors, maxillary lesions should be treated more aggressively because they grow faster and because of the proximity to important structures. They recommend resection of the maxillary lesions and enucleation of the mandibular tumors. They also state that the more amyloid tissue and calcification there is, the less aggressive is the lesion.
FIGURE 2. Panoramic radiograph showing the lesion, radiopaque masses, and the tooth displaced toward the inferior border.
FIGURE 3. Occlusal view showing expansion of the cortical plates that is more evident on the lingual side.
In the case presented, the lesion was well defined radiographically, slow-growing, and easily enucileated. For these reasons a conservative approach was ohosen. We believe that the treatment should be individualized for each lesion because the radiographic and histologic features may differ from one lesion to another. Although it has not been established in the literature, we believe that 5 years should be the minimum follow-up
FIGURE 4.
Surgical specimen with the associated tooth.
1326
CALCIFYING EPITHELIAL ODONTOGENIC
FIGURE 5. Photomicrograph showing epithelium with polyhedral cells of varying size, nuclei of different shape and size, and demonstration of possible amyloid tissue (hematoxylin-eosin stain, original magnification X200).
necessary to assess cure for this type of odontogenic tumor. References 1. Franklin CD, Pindborg JJ: The calcifying epithelial odontogenic tumor. A review and analysis of 113 cases. Oral Surg 42:753, 1976
J Oral Maxillofac 50:1326-1326,
TUMOR
2. Smith RA, Roman RS, Hansen IS, et al: The calcifying epithelial odontogenic tumor. Oral Surg 35: 160, 1977 3. Gargiulo EA, Ziter WD, Matrocola E: Calcifying epithelial odontogenic tumor: Report of case and review of the literature. Oral Surg 29862, 197 1 4. Pindborg JJ: Calcifying epithelial odontogenic tumor Acta Path Microbial Stand Ill:7 1, 1956 apud (suppl) Vap DR, Dahlin DC, Turlington EG: Pindborg tumor: The so-called calcifying epithelial odontogenic tumor. Cancer 25:629, 1970 5. Shafer WC, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 3). Philadelphia, PA, Saunders, 1974, pp 258-26 1 6. Pindborg JJ: The calcifying epithelial odontogenic tumor: Review of the literature and report of an extra-osseous case. Acta Odontol Stand 24:4 19, 1966 7. Krolls SO, Pindborg JJ: Calcifying epitbelial odontogenic tumor. A survey of 23 cases and discussion of histomorphologic variations. Arch Path01 98206, 1974 8. Liu AR, Liu Z, Shao J: Calcifying epithelird odontogenic tumors. Clinicopathologic study of nine cases. J Oral Path01 11:399, 1982 9. Mori M, Makino M, Imai K: The histochemical nature of homogeneous amorphous materials in odontogenic epithelial tumors. J Oral Surg 38:96, 1980 10. Mainwaring AR, Ahmed A, Hopkinson JM, et al: A clinical and electron microscopic study of a calcifying epithelial odontogenie tumor. J Clin Path01 24: 152, 1971 11. Solomon MP, Vuletin JC, Pertschuk LP, et al: Calcifying epithelial odontogenic tumor: A histologic, histochemical, fluorescent and ultrastructural study. Oral Surg 4Ck522, 1975 12. Franklin CD, Hindle MO: The calcifying epithelial odontogenic tumor. Report of four cases; two with long-term follow-up. Br J Oral Surg 13:230, 1976 13. Sadegui EM, Hoper TL: Calcifying epithelial odontogenic tumor. J Oral Maxillofac Surg 40:225, 1982
Surg 1992
Calcifying Epithelial Odontogenic Tumor of the Maxillary Sinus CAMERON Y.S. LEE, DMD,* HOSSEIN MOHAMMADI, REZA MOSTOFI, DMD, MS,* AND ATA HABIBI, The calcifying epithelial odontogenic tumor (CEOT), a rare odontogenic neoplasm arising from odontogenic epithelium, was first identified by Thoma and Goldman.’ It was not until 1955, however, that the CEOT was recognized as a separate entity by Pindborg.2 The CEOT has been reported under a variety of different Received from the Department of Oral and Maxillofacial Surgery, University of Illinois College of Dentistry, Chicago. * Formerly, Chief Resident; currently, in private practice, Honolulu. 7 Assistant Professor and Director of Undergraduate Oral and Maxillofacial Surgery. $ Clinical Assistant Professor. 8 In private practice. Address correspondence and reprint requests to Dr Lee: The Oral Surgery Group, 868 S Beretania St, Honolulu, HI 968 13. 0 1992 AmericanAssociationof Oral and MaxillofacialSurgeons 0278-2391/92/5012-0015$3.00/0
DDS, MS,t DOSS
terms, such as “adamantoblastoma,“’ “unusual ameloblastoma,“3 and “cystic complex odontoma.“4 The CEOT is a lesion of the jaws that accounts for about 1% of all tumors of odontogenic origin5,” In the study by Franklin and Pindborg,7 the CEOT was found most frequently in the premolar-molar region of the mandible. However, peripheral lesions have been reported, usually located in the anterior mandibular or maxillary gingiva.8 Unerupted teeth are associated with the CEOT in 52% of cases. The mean age of patients is 40 years, with no sex predilection.’ A calcifying epithelial odontogenic tumor arising within the maxillary sinus is extremely rare, with only three cases previously reported since the original description in 1946.9-” We describe an additional case of a calcifying epithelial odontogenic tumor in this anatomic location.
Surg 1992
Calcifying Epithelial Odontogenic Tumor (Pindborg Tumor): Report of Case MAURICIO ROBERTO
ANDRADE, DDS,* PAUL0 J. MEDEIROS, PRADO, DDS,* AND RENATO SAMPAIO,
porated within the lesion. There was cortical expansion in all directions, mainly toward the superior and lingual of the lesion (Fig 3). In consideration of the benign radiographic appearance and the size of the lesion, intraoral surgical excision with moderate curettage of the adjacent bone under general anesthesia was planned. The lesion was easily detached from the surrounding bone and was removed intact. There were no intraoperative complications. The surgical specimen measured 3 cm in diameter. The consistency was firm and the color was pale pink (Fig 4). Histologically there was a fibrous connective-tissue capsule surrounding epithelial tissue composed of polyhedral cells of different sizes, containing nuclei of varying forms and sizes, as well as an extracellular eosinophilic material, probably amyloid. There were concentric calcifications in the form of Liesegang rings (Fig 5). The histologic diagnosis was calcifying epithelial odontogenic tumor. The postoperative course was uneventful and the patient is being recalled regularly. There are no signs of recurrence 24 months after surgery.
The calcifying epithelial odontogenic tumor (CEOT) is a rare benign epithelial odontogenic lesion that comprises from 0.2% to 1% of all odontogenic tumors.’ Clinically it manifests as a bony lesion, accounting for the majority of cases (95%), or as the extremely rare peripheral type (5%). In the past a number of different names have been given to this lesion, such as calcifying ameloblastoma, ’ malignant odontoma,’ adamantoblastoma,2 adenoid adamantoblastoma,3 cystic complex odontoma,3 uncommon ameloblastoma with calcifications,’ and others. In 1956, Pindborg4 delineated this lesion as a distinct entity and named it the calcifying epithelial odontogenic tumor. In 1963, along with a comprehensive review of the literature, Shafer et al* suggested the eponym “Pindborg tumor.” The purpose of this article is to report an additional case of the CEOT and discuss important aspects of the radiographic appearance, histologic features, clinical behavior, and surgical management.
Discussion
Report of Case
It is commonly accepted that the CEOT has its origin from odontogenic epithelium.‘,3-6 Pindborg6 believes that the origin of this lesion is the enamel epithelial because there is a large number of cases associated with unerupted teeth. He also states that the origin of the peripheral lesions is still uncertain. There is agreement among several authors regarding the clinical features of this tumor. In the most comprehensive review of the literature by Franklin and Pindborg,’ 113 cases have been studied. There was a predilection for occurrence in the premolar-molar region of the mandible. There is no sex predilection, with the mean age of occurrence around 40 years. Most cases manifested as an intraoral or extraoral mass without other symptoms.‘,3,7,8 The varying radiographic features have been described as 1) pericoronal radiolucent; 2) pericoronal radiolucent with small radiopacities; 3) mixed radiolucent-radiopaque lesion not associated with an unerupted tooth; 4) “falling snow” appearance; 5) dense
A 3 1-year-old white man was seen for consultation because of an asymptomatic, 3-cm-diameter mass on the left mandibular alveolar ridge (Fig 1). The patient had first noticed the lesion 4 years before. The lesion was indurated and was expanded toward the lingual side. The color of the overlying oral mucosa was normal. No facial asymmetry could be noticed, and there was no regional lymphadenopathy. The panoramic radiograph showed features of a benign lesion (Fig 2). The lesion was radiolucent with well-defined borders and extended from the second premolar to the third molar region. There were several radiopaque masses and the crown of a molar tooth, probably the second molar, incorReceived from Rio de Janeiro State University, Brazil. * Staff Member, Oral and Maxillofacial Surgery Service. t Chief. Oral and Maxillofacial Sumerv _ _ Service; Director, Residency Training Program. $ Associated Professor; Head, Oral Pathology Service. Address correspondence and reprint requests to Dr Medeiros: Av NS Copacabana 680-9 10, 22.050 Rio de Janeiro, R3 Brazil. 0 1992 American
Association
of Oral and Maxillofacial
DDS, MS,t DDS, MS*
Surgeons
0278-2391/92/5012-0015$3.00/O
1324
1325
ANDRADE ET AL
FIGURE I. ridge.
The intraoral view showing the mass on the alveolar
radiopacity. Franklin and Pindborg’ found the two most common presentations to be pericoronal radiolucent and diffuse radiopacities in a radiolucent area. The histologic pattern is quite typical and very well defined. However, it may show subtle changes in cellular pattern related to the occurrence of amyloid and calcifications.‘*5,9-” No author believes the CEOT has a malignant biologic behavior. Some authors believe that the safest treatment consists of a wide marginal resection.’ However, some cases have been treated by simple surgical excision.‘q7.‘2 Sadeghi and HoperI believe that the treatment for the CEOT should be guided by the size of the lesion, its location, and the histologic features. According to these authors, maxillary lesions should be treated more aggressively because they grow faster and because of the proximity to important structures. They recommend resection of the maxillary lesions and enucleation of the mandibular tumors. They also state that the more amyloid tissue and calcification there is, the less aggressive is the lesion.
FIGURE 2. Panoramic radiograph showing the lesion, radiopaque masses, and the tooth displaced toward the inferior border.
FIGURE 3. Occlusal view showing expansion of the cortical plates that is more evident on the lingual side.
In the case presented, the lesion was well defined radiographically, slow-growing, and easily enucileated. For these reasons a conservative approach was ohosen. We believe that the treatment should be individualized for each lesion because the radiographic and histologic features may differ from one lesion to another. Although it has not been established in the literature, we believe that 5 years should be the minimum follow-up
FIGURE 4.
Surgical specimen with the associated tooth.
1326
CALCIFYING EPITHELIAL ODONTOGENIC
FIGURE 5. Photomicrograph showing epithelium with polyhedral cells of varying size, nuclei of different shape and size, and demonstration of possible amyloid tissue (hematoxylin-eosin stain, original magnification X200).
necessary to assess cure for this type of odontogenic tumor. References 1. Franklin CD, Pindborg JJ: The calcifying epithelial odontogenic tumor. A review and analysis of 113 cases. Oral Surg 42:753, 1976
J Oral Maxillofac 50:1326-1326,
TUMOR
2. Smith RA, Roman RS, Hansen IS, et al: The calcifying epithelial odontogenic tumor. Oral Surg 35: 160, 1977 3. Gargiulo EA, Ziter WD, Matrocola E: Calcifying epithelial odontogenic tumor: Report of case and review of the literature. Oral Surg 29862, 197 1 4. Pindborg JJ: Calcifying epithelial odontogenic tumor Acta Path Microbial Stand Ill:7 1, 1956 apud (suppl) Vap DR, Dahlin DC, Turlington EG: Pindborg tumor: The so-called calcifying epithelial odontogenic tumor. Cancer 25:629, 1970 5. Shafer WC, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 3). Philadelphia, PA, Saunders, 1974, pp 258-26 1 6. Pindborg JJ: The calcifying epithelial odontogenic tumor: Review of the literature and report of an extra-osseous case. Acta Odontol Stand 24:4 19, 1966 7. Krolls SO, Pindborg JJ: Calcifying epitbelial odontogenic tumor. A survey of 23 cases and discussion of histomorphologic variations. Arch Path01 98206, 1974 8. Liu AR, Liu Z, Shao J: Calcifying epithelird odontogenic tumors. Clinicopathologic study of nine cases. J Oral Path01 11:399, 1982 9. Mori M, Makino M, Imai K: The histochemical nature of homogeneous amorphous materials in odontogenic epithelial tumors. J Oral Surg 38:96, 1980 10. Mainwaring AR, Ahmed A, Hopkinson JM, et al: A clinical and electron microscopic study of a calcifying epithelial odontogenie tumor. J Clin Path01 24: 152, 1971 11. Solomon MP, Vuletin JC, Pertschuk LP, et al: Calcifying epithelial odontogenic tumor: A histologic, histochemical, fluorescent and ultrastructural study. Oral Surg 4Ck522, 1975 12. Franklin CD, Hindle MO: The calcifying epithelial odontogenic tumor. Report of four cases; two with long-term follow-up. Br J Oral Surg 13:230, 1976 13. Sadegui EM, Hoper TL: Calcifying epithelial odontogenic tumor. J Oral Maxillofac Surg 40:225, 1982
Surg 1992
Calcifying Epithelial Odontogenic Tumor of the Maxillary Sinus CAMERON Y.S. LEE, DMD,* HOSSEIN MOHAMMADI, REZA MOSTOFI, DMD, MS,* AND ATA HABIBI, The calcifying epithelial odontogenic tumor (CEOT), a rare odontogenic neoplasm arising from odontogenic epithelium, was first identified by Thoma and Goldman.’ It was not until 1955, however, that the CEOT was recognized as a separate entity by Pindborg.2 The CEOT has been reported under a variety of different Received from the Department of Oral and Maxillofacial Surgery, University of Illinois College of Dentistry, Chicago. * Formerly, Chief Resident; currently, in private practice, Honolulu. 7 Assistant Professor and Director of Undergraduate Oral and Maxillofacial Surgery. $ Clinical Assistant Professor. 8 In private practice. Address correspondence and reprint requests to Dr Lee: The Oral Surgery Group, 868 S Beretania St, Honolulu, HI 968 13. 0 1992 AmericanAssociationof Oral and MaxillofacialSurgeons 0278-2391/92/5012-0015$3.00/0
DDS, MS,t DOSS
terms, such as “adamantoblastoma,“’ “unusual ameloblastoma,“3 and “cystic complex odontoma.“4 The CEOT is a lesion of the jaws that accounts for about 1% of all tumors of odontogenic origin5,” In the study by Franklin and Pindborg,7 the CEOT was found most frequently in the premolar-molar region of the mandible. However, peripheral lesions have been reported, usually located in the anterior mandibular or maxillary gingiva.8 Unerupted teeth are associated with the CEOT in 52% of cases. The mean age of patients is 40 years, with no sex predilection.’ A calcifying epithelial odontogenic tumor arising within the maxillary sinus is extremely rare, with only three cases previously reported since the original description in 1946.9-” We describe an additional case of a calcifying epithelial odontogenic tumor in this anatomic location.
