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.
1327
LEE ET AL
Report of Case On December 11, 1979 a 27-year-old woman was seen with a complaint of a progressive enlargement of the left malar region for the past 18 months, associated with infraorbital dysesthesia. She also complained of difficulty in breathing through the left side of the nose. There were no reports of nasal discharge, epistaxsis, or headache. The past medical history was unremarkable.
FIGURE 2.
Gross specimen after left hemimaxilleetomy.
Clinical examination showed a well-developed, healthy young woman with a firm, nontender mass over the left malar region causing facial asymmetry. In addition, there was proptosis of the left globe, with orbital dystopia. Examination of the neck did not reveal lymphadenopathy. The patient had no difficulty opening her mouth, although there was some discomfort. Intraorally, there was a firm, expansile lesion involving the left hard palate, with obliteration of the maxillary buccal vestibule. The mucosa was intact, without any evidence of a draining fistula. There was no report of tooth sensitivity, and the patient stated that her maxillary first premolar tooth had been extracted without complication. Radiographic examination of the facial bones and maxillary sinus showed a radiopacity of the inferior aspect of the left maxillary sinus as well as a fully developed ectopic tooth located in the posterosuperior aspect (Fig 1). An incisional biopsy was performed on December 14, and a histologic diagnosis of calcifying epithelial odontogenic tumor was made (CEOT). The patient was admitted to the university hospital on December 2 1 for definitive treatment. Preoperative laboratory workup, including electrocardiogram (ECG) and chest radiographs, was within normal limits. During the surgical procedure, the tumor was found to occupy most of the maxillary sinus, with compression of the left lateral nasal wall and roof of the maxillary sinus. The bony floor and lateral wall of the sinus were missing, having been replaced with calcified tissue from the tumor. An intraoral left hemimaxillectomy measuring 4 X 3.5 X 2 cm was performed (Fig 2). The patient’s postoperative course was uneventful and she was discharged from the hospital on the fourth postaperative day. The patient refused maxillofacial reconstructive surgery, and only a maxillary prosthesis was fabricated. MACROSCOPIC
FIGURE I. A. Panoramic radiograph showing the lesion with premolar tooth in the left maxillary sinus. B, Waters view radiograph showing a premolar tooth high in the maxillary sinus associated with the lesion.
AND MICROSCOPIC
FINDINGS
Gross examination showed a tumor encased in bone and calcified material, measuring 25 X 35 X 4 mm. The mucous membrane of the maxillary sinus was attached to the tumor. A maxillary premolar tooth and calcified masses were observed within the tumor. Histologically, the tumor was composed of sheets of epithelial cells, with a fibrovascular stroma. The epithclial cells had an eosinophilic cytoplasm. Intercellular bridges were present. No mitotic figures were noted throughout the specimen. Regions of amorphous and eosinophilic material were
1328
CALCIFYING EPITHELIAL ODONTOGENIC TUMOR
FIGURE 3. A, Low-power view of CEOT composed of sheets of large polyhedral cells in a fibrous stroma (hematoxylin-eosin stain, original magnification X63). B, Higher magnification view showing sheets of polyhedral cells with intercellular bridges (hematoxylin-eosin stain, original magnification x560).
seen within the sheets of epithelial cells (Fig 3). Calcification in the form of Leisegang’s rings was seen in some areas. The patient was followed for 7 years postoperatively and there was no evidence of recurrence of the tumor. Since then, the patient has been lost to follow-up. Discussion
reported by Basu et al, the patient underwent a hemimandibulectomy because the lesion was malignant.14 Lesions of the maxilla are treated more aggressively, because tumors of the maxilla could impinge on vital structures. Maxillectomy has been recommended as the treatment of choice.‘5,‘6 References
Calcifying epithelial odontogenic tumor with extension into the maxillary sinus is uncommon. In the three cases previously reported, in which a CEOT was situated in the maxillary sinus, each CEOT was associated with an embedded tooth. In 1965, Gon’O reported a CEOT in the right maxillary sinus associated with an unerupted maxillary first molar. Stimson in 1968” described a case where the maxillary sinus was completely filled by a CEOT, which invaded the nasal cavity from the medial wall. The tumor was associated with an unerupted third molar. In both cases, the clinical signs were nasal obstruction and swelling of the cheek. In our case, the tumor had extended into the maxillary sinus, was associated with an unerupted second premolar tooth, and the patient complained of nasal obstruction. The CEOT is generally considered a benign tumor. In a few cases, the tumor has been described as being locally aggressive, invading the surrounding soft tissues and bone marrow spaces. ‘**I3Metastasis of this lesion has been reported by Basu and colleagues.‘4 In their case there was evidence of local tissue invasion and lymph node involvement. Surgical management of the CEOT has varied, depending on the site and size of the lesion as well as the amount of bone destruction. In the mandible, the recommended surgical approach is enucleation with vigorous curettage in the early stages. With more advanced bone infiltration, resection of the tumor with a normal margin of bone should be considered.15 In the case
1. Thoma KH, Goldman HM: Odontogenic tumors: Classification based on observations of epithelial, mesenchymal and mixed varieties. Amer J Path01 22:433, 1946 2. Pindborg JJ: Calcifying epithelial odontogenic tumors. Acta Pathol Microbial Stand Ill:7 1, 1955 (suppl) 3. Ivy RH: Unusual case of ameloblastoma of the mandible. Oral Surg 1:1074, 1948 4. Stoopack JC: Cystic odontoma of the mandible. Oral Surg 10: 807, 1957 5. Pindborg JJ: A calcifying odontogenic tumor. Cancer 2:838,1958 6. Krolls SO, Pindborg JJ: Calcifying epithelial odontogenic tumors: A survey of 23 cases and discussion of histomorphological variations. Arch Path01 98206, 1974 7. Franklin CD, Pindborg JJ: The calcifying epithelial odontogenic tumor. A review and analysis of 113cases. Oral Surg 42:753, 1976 8. Pindborg JJ: The calcifying epithelial odontogenic tumor. Review of the literature and report of an extraosseous case. Acta Odont Stand 24:4 19, 1966 9. Baunsgaard P, Lontoft E, Sorensen M: Calcifying epithelial odontogenic tumor (Pindborg tumor): An unusual case. Laryngoscope 93:635, 1983 10. Gon F: The calcifying epithelial odontogenic tumor. Report of a case and study of its histogenesis. Br J Cancer 19:39, 1965 11. Stimson PG, Luna MA, Butler JJ: Seventeen-year history of a calcifying epithelial odontogenic (Pindborg) tumor. Oral Surg 25:204, 1968 12. Smith RA, Roman RS, Hansen LS, et al: The calcifying epithelial odontogenic tumor. J Oral Surg 35: 160, 1977 13. Van DR. Dahlin DC, Turlington EG: The so-called calcifying epithelial odontogenic tumor. Cancer 25:629, 1970 14. Baw MK. Mathews JB, Sear AJ. et al: Calcifying epithelial odontogenic tumor: A case showing features of malignancy. J Oral Path01 I3:3 10, 1984 15. Gargiulo EA, Ziter WD. Mastrocola R: Calcifying epithelial odontogenic tumor: Report of case and review of literature. Oral Surg 29:862, 197 1 16. Sadeghi EM, Hopper TL: Calcifying epithelial odontogenic tumor. J Oral Maxillofac Surg 40~225, 1982
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.
1327
LEE ET AL
Report of Case On December 11, 1979 a 27-year-old woman was seen with a complaint of a progressive enlargement of the left malar region for the past 18 months, associated with infraorbital dysesthesia. She also complained of difficulty in breathing through the left side of the nose. There were no reports of nasal discharge, epistaxsis, or headache. The past medical history was unremarkable.
FIGURE 2.
Gross specimen after left hemimaxilleetomy.
Clinical examination showed a well-developed, healthy young woman with a firm, nontender mass over the left malar region causing facial asymmetry. In addition, there was proptosis of the left globe, with orbital dystopia. Examination of the neck did not reveal lymphadenopathy. The patient had no difficulty opening her mouth, although there was some discomfort. Intraorally, there was a firm, expansile lesion involving the left hard palate, with obliteration of the maxillary buccal vestibule. The mucosa was intact, without any evidence of a draining fistula. There was no report of tooth sensitivity, and the patient stated that her maxillary first premolar tooth had been extracted without complication. Radiographic examination of the facial bones and maxillary sinus showed a radiopacity of the inferior aspect of the left maxillary sinus as well as a fully developed ectopic tooth located in the posterosuperior aspect (Fig 1). An incisional biopsy was performed on December 14, and a histologic diagnosis of calcifying epithelial odontogenic tumor was made (CEOT). The patient was admitted to the university hospital on December 2 1 for definitive treatment. Preoperative laboratory workup, including electrocardiogram (ECG) and chest radiographs, was within normal limits. During the surgical procedure, the tumor was found to occupy most of the maxillary sinus, with compression of the left lateral nasal wall and roof of the maxillary sinus. The bony floor and lateral wall of the sinus were missing, having been replaced with calcified tissue from the tumor. An intraoral left hemimaxillectomy measuring 4 X 3.5 X 2 cm was performed (Fig 2). The patient’s postoperative course was uneventful and she was discharged from the hospital on the fourth postaperative day. The patient refused maxillofacial reconstructive surgery, and only a maxillary prosthesis was fabricated. MACROSCOPIC
FIGURE I. A. Panoramic radiograph showing the lesion with premolar tooth in the left maxillary sinus. B, Waters view radiograph showing a premolar tooth high in the maxillary sinus associated with the lesion.
AND MICROSCOPIC
FINDINGS
Gross examination showed a tumor encased in bone and calcified material, measuring 25 X 35 X 4 mm. The mucous membrane of the maxillary sinus was attached to the tumor. A maxillary premolar tooth and calcified masses were observed within the tumor. Histologically, the tumor was composed of sheets of epithelial cells, with a fibrovascular stroma. The epithclial cells had an eosinophilic cytoplasm. Intercellular bridges were present. No mitotic figures were noted throughout the specimen. Regions of amorphous and eosinophilic material were
1328
CALCIFYING EPITHELIAL ODONTOGENIC TUMOR
FIGURE 3. A, Low-power view of CEOT composed of sheets of large polyhedral cells in a fibrous stroma (hematoxylin-eosin stain, original magnification X63). B, Higher magnification view showing sheets of polyhedral cells with intercellular bridges (hematoxylin-eosin stain, original magnification x560).
seen within the sheets of epithelial cells (Fig 3). Calcification in the form of Leisegang’s rings was seen in some areas. The patient was followed for 7 years postoperatively and there was no evidence of recurrence of the tumor. Since then, the patient has been lost to follow-up. Discussion
reported by Basu et al, the patient underwent a hemimandibulectomy because the lesion was malignant.14 Lesions of the maxilla are treated more aggressively, because tumors of the maxilla could impinge on vital structures. Maxillectomy has been recommended as the treatment of choice.‘5,‘6 References
Calcifying epithelial odontogenic tumor with extension into the maxillary sinus is uncommon. In the three cases previously reported, in which a CEOT was situated in the maxillary sinus, each CEOT was associated with an embedded tooth. In 1965, Gon’O reported a CEOT in the right maxillary sinus associated with an unerupted maxillary first molar. Stimson in 1968” described a case where the maxillary sinus was completely filled by a CEOT, which invaded the nasal cavity from the medial wall. The tumor was associated with an unerupted third molar. In both cases, the clinical signs were nasal obstruction and swelling of the cheek. In our case, the tumor had extended into the maxillary sinus, was associated with an unerupted second premolar tooth, and the patient complained of nasal obstruction. The CEOT is generally considered a benign tumor. In a few cases, the tumor has been described as being locally aggressive, invading the surrounding soft tissues and bone marrow spaces. ‘**I3Metastasis of this lesion has been reported by Basu and colleagues.‘4 In their case there was evidence of local tissue invasion and lymph node involvement. Surgical management of the CEOT has varied, depending on the site and size of the lesion as well as the amount of bone destruction. In the mandible, the recommended surgical approach is enucleation with vigorous curettage in the early stages. With more advanced bone infiltration, resection of the tumor with a normal margin of bone should be considered.15 In the case
1. Thoma KH, Goldman HM: Odontogenic tumors: Classification based on observations of epithelial, mesenchymal and mixed varieties. Amer J Path01 22:433, 1946 2. Pindborg JJ: Calcifying epithelial odontogenic tumors. Acta Pathol Microbial Stand Ill:7 1, 1955 (suppl) 3. Ivy RH: Unusual case of ameloblastoma of the mandible. Oral Surg 1:1074, 1948 4. Stoopack JC: Cystic odontoma of the mandible. Oral Surg 10: 807, 1957 5. Pindborg JJ: A calcifying odontogenic tumor. Cancer 2:838,1958 6. Krolls SO, Pindborg JJ: Calcifying epithelial odontogenic tumors: A survey of 23 cases and discussion of histomorphological variations. Arch Path01 98206, 1974 7. Franklin CD, Pindborg JJ: The calcifying epithelial odontogenic tumor. A review and analysis of 113cases. Oral Surg 42:753, 1976 8. Pindborg JJ: The calcifying epithelial odontogenic tumor. Review of the literature and report of an extraosseous case. Acta Odont Stand 24:4 19, 1966 9. Baunsgaard P, Lontoft E, Sorensen M: Calcifying epithelial odontogenic tumor (Pindborg tumor): An unusual case. Laryngoscope 93:635, 1983 10. Gon F: The calcifying epithelial odontogenic tumor. Report of a case and study of its histogenesis. Br J Cancer 19:39, 1965 11. Stimson PG, Luna MA, Butler JJ: Seventeen-year history of a calcifying epithelial odontogenic (Pindborg) tumor. Oral Surg 25:204, 1968 12. Smith RA, Roman RS, Hansen LS, et al: The calcifying epithelial odontogenic tumor. J Oral Surg 35: 160, 1977 13. Van DR. Dahlin DC, Turlington EG: The so-called calcifying epithelial odontogenic tumor. Cancer 25:629, 1970 14. Baw MK. Mathews JB, Sear AJ. et al: Calcifying epithelial odontogenic tumor: A case showing features of malignancy. J Oral Path01 I3:3 10, 1984 15. Gargiulo EA, Ziter WD. Mastrocola R: Calcifying epithelial odontogenic tumor: Report of case and review of literature. Oral Surg 29:862, 197 1 16. Sadeghi EM, Hopper TL: Calcifying epithelial odontogenic tumor. J Oral Maxillofac Surg 40~225, 1982
