J Oral MaxillofacSurg 50:190-193.1992
Odontogenic Cyst Giving Rise to an Adenomatoid Odontogenic Tumor: Report of a Case With Peculiar Features YOSHIFUMI TAJIMA, DDS,* EIICHI SAKAMOTO, AND YOSHIRO YAMAMOTO, DDS*
DDS,t
It has been reported that some odontogenic cysts occur in association with odontogenic tumors.’ Because neoplastic and hamartomatous abberations can occur at any stage of odontogenesis2 combined features of odontogenic tumors with epithelial and mesenchymal components may arise within odontogenic cysts. The authors report an unusual example of an odontogenic cyst that contained focal areas of odontogenic epithelial and mesenchymal proliferation exhibiting a combination of adenomatoid odontogenic tumor (AOT) and dentinoid components. Emphasis is placed on the clinical, radiographic, and microscopic features of this case.
mass was identified as dentinoid material (Fig 3C,D). Abutting the dentinoid structure at one end was a small, but distinct, proliferation of epithelium showing features of an adenomatoid odontogenic tumor (Fig 3B). The proliferating epithelium was in the form of strands and whorled masses with rosette and ductlike structures. Polyhedral-shaped cells forming organoid clumps and spindle-shaped cells were found at the periphery (Fig 3B). Tall columnar epithelium resembling ameloblasts and an immature enamellike substance (Fig 3D) were also noted adjacent to the dentinoid structure, The diagnosis was an AOT arising in an odontogenic (dentigerous) cyst.
Report of Case
In 195 1, Miles3 reported a case of a cystic complex composite odontoma. Later, Dunlap and Fritzlen reported a cystic odontoma with a soft-tissue component resembling an AOT. A similar example was also reported by Miller.’ These lesions, as well as this case, may represent examples of AOTs arising within the wall of odontogenic cysts. Giansanti and colleague@ found in their review of the literature that 74% of AOTs were associated with dentigerous cysts or with the crowns of unerupted teeth. Both the odontoma and AOT have been regarded by some investigators to be hamartomas rather than neoplasms.7,8 Odontomas may be occasionally associated with a dentigerous cyst.’ The AOT has also been reported to occur with cystic features.r”“’ We believe that this case represents an odontogenic cyst that developed “hamartomatous” proliferations containing both epithelial and mesenchymal components. It is interesting to speculate whether this lesion had the potential of developing into a more aggressive odontogenic neoplasm. It has been claimed that ameloblastomas may originate from a preexisting ameloblastic fibro-odontoma or odontoma.” Odontogenic cysts showing aggressive growth with neoplastic potential have been reported by Eversole et al,13 Van der
Discussion
The patient, a 15-year-old boy, had an asymptomatic lesion involving the left maxillary sinus. The lesion contained the crown of an unerupted tooth and an irregularly-formed, calcified mass beneath the nasal side of the orbit with expansion of the overlying bone (Fig lA,B). The lesion was completely enucleated; there was no adhesion to the sinus wall. Grossly, the lesion (4.0 X 4.0 cm) appeared cystic and contained a malformed, craterlike calcified mass and the crown of an unerupted tooth (Figs lC, 2). Follow-up for a period of 5 years has shown no recurrence. Microscopic examination exhibited a thick-walled cystic structure (Fig 3A) lined by flattened squamous epithelium in the majority of the lesion (Fig 3C). The malformed calcified
Received from Meikai University School of Dentistry, Sakado, Saitama, Japan. * Associate Professor, Department of Oral Pathology. t Lecturer, Department of Oral and Maxillofacial Surgery. $ Professor and Chairman, Department of Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Tajima: Department of Oral Pathology, Meikai University School of Dentistry, Sakado, Saitama 350-02, Japan. 0 1992 American
Association
of Oral and Maxillofacial
Surgeons
0278-2391/92/5002-0018$3.00/O
190
TAJIMA, SAKAMOTO, AND YAMAMOTO
191
FIGURE 1. Water’s (A) and panoramic (B) radiographs showing a relatively well-defined radiopaque mass (arrows) and crown of an unerupted tooth (arrowheads) in the superior portion of the maxillary sinus beneath the orbit. C, Radiograph of the excised specimen showing an irregularly formed radiopacity in close proximity to the embedded tooth.
FIGURE 2. The gross appearance of specimen, showing a cystic configuration with a craterlike calcified mass at the center.
CYSTOGENIC ADENOMATOID
ODONTOGENIC
TUMOR
FIGURE 3. A, Low-power photomicrograph of the entire specimen. The lesion is composed of a cystic structure exhibiting dentinoid material (Dn) and a small focus of epithelial mass (large arrow) (hematoxylin-eosin stain. original magnification X5). B, The epithelial components consist of swirling polyhedral and spindle-shaped cells forming ductlike structures. Tall columnar cells (Ab), resembling ameloblasts, are seen (hematoxylin-eosin stain, original magnification X 100). C, The cyst is lined in large part by flattened squamous epithelium (arrows). Dentinoid material (Dn) is present adjacent to the cystic lumen (L) (hematoxylin-eosin stain, original magnification X200). D. Photomicrograph of the area showing immature enamellike material (Em) closely apposed to the columnar ameloblastlike cells and dentinoid material (hematoxylineosin stain, original magnification X200).
Waal et a1,14and by Waldron and Mustoe.” Whether a lesion of the type shown in this case has the potential to develop into a frank AOT is unknown. The differentiation between an odontogenic cyst and neoplasm is usually distinct. However, one might occasionally encounter difficulty in separating these two entities from microscopic examination alone. This case
emphasizes the importance of interpreting the clinical and radiologic features in conjunction with the microscopic appearance. References 1. Verbin RS, Barnes L: Surgical pathology of the head and neck. New York, NY, Dekker, 1985, p 1233
TAJIMA, SAKAMOTO, AND YAMAMOTO
2. Waldron CA: Pathology of the head and neck. Contemporary issues in surgical pathology, vol 10. New Ybtk, NY, Churchill Livingstone, 1988, p 403 3. Miles AEW: A cystic complex composite odontome. Proc R Sot Med 44:51, 1951 4. Dunlap CL, Fritzlen TJ: Cystic odontoma with concomitant adenoameloblastoma (adenoameloblastic odontoma). Oral Surg Oral Med Oral Path01 34450, 1972 5. Miller WA: Cystic odontoma or so-called adenoameloblastoma. J Path01 98:75, 1969 6. Giansanti JS. Someren A, Waldron CA: Odontogenic adenomatoid tumor (adenoameloblastoma)-Survey of I I 1cases. Oral Surg Oral Med Oral Pathol 30:69, 1970 7. Abrams AM. Melrose RJ. Howell FV: Adenoameloblastoma: A clinical pathologic study of ten new cases. Cancer 22:175, 1968 8. Slootweg PJ: An analysis of interrelationship of mixed odontogenie tumors-Ameloblastic fibroma, ameloblastic fibroodontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 51:266, 1981 9. Gorlin RJ. Chaudhry AP, Pindborg JJ: Odontogenic tumors.
193
Classification, histopathology, and clinical behavior in man and’domestic animals. Cancer 14:73. 1961 10. Freedman PD. Lumerman H, Gee JK: Calcifying odontogenic cyst. A review and analysis of seventy cases. Oral Surg Oral Med Oral Path01 40:93, 1975 11. International Association of Oral Pathologists: Slide Seminar on odontogenic cysts and tumors, case 2 (contributed by Daniels TE and Hansen LS). Tokvo. Japan. International Association of Oral Pathologists 5th Riennial Congress, 1990 12. Eversole LS, Tomich CE, Cherrick HM: Histogenesis of odontogenic tumors. Oral Surg Oral Med Oral Pathol32:569, 197 1 13. Eversole LS, Sabes WR, Rovin S: Aggressive growth and neoplastic potential of odontogenic cysts. With special reference to central epidermoid and mucoepidermoid carcinomas. Cancer 35:270, 1975 14. Van der Waal 1, Rauhamaa R, Van der Kwast WAM, et al: Squamous cell carcinoma arising in the lining of odontogenic cysts. Report of 5 cases. Int J Oral Sung 14:146, 1985 15. Waldron CA, Mustoe TA: Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst. Oral Surg Oral Med Oral Path01 67:7 16, I989
Odontogenic Cyst Giving Rise to an Adenomatoid Odontogenic Tumor: Report of a Case With Peculiar Features YOSHIFUMI TAJIMA, DDS,* EIICHI SAKAMOTO, AND YOSHIRO YAMAMOTO, DDS*
DDS,t
It has been reported that some odontogenic cysts occur in association with odontogenic tumors.’ Because neoplastic and hamartomatous abberations can occur at any stage of odontogenesis2 combined features of odontogenic tumors with epithelial and mesenchymal components may arise within odontogenic cysts. The authors report an unusual example of an odontogenic cyst that contained focal areas of odontogenic epithelial and mesenchymal proliferation exhibiting a combination of adenomatoid odontogenic tumor (AOT) and dentinoid components. Emphasis is placed on the clinical, radiographic, and microscopic features of this case.
mass was identified as dentinoid material (Fig 3C,D). Abutting the dentinoid structure at one end was a small, but distinct, proliferation of epithelium showing features of an adenomatoid odontogenic tumor (Fig 3B). The proliferating epithelium was in the form of strands and whorled masses with rosette and ductlike structures. Polyhedral-shaped cells forming organoid clumps and spindle-shaped cells were found at the periphery (Fig 3B). Tall columnar epithelium resembling ameloblasts and an immature enamellike substance (Fig 3D) were also noted adjacent to the dentinoid structure, The diagnosis was an AOT arising in an odontogenic (dentigerous) cyst.
Report of Case
In 195 1, Miles3 reported a case of a cystic complex composite odontoma. Later, Dunlap and Fritzlen reported a cystic odontoma with a soft-tissue component resembling an AOT. A similar example was also reported by Miller.’ These lesions, as well as this case, may represent examples of AOTs arising within the wall of odontogenic cysts. Giansanti and colleague@ found in their review of the literature that 74% of AOTs were associated with dentigerous cysts or with the crowns of unerupted teeth. Both the odontoma and AOT have been regarded by some investigators to be hamartomas rather than neoplasms.7,8 Odontomas may be occasionally associated with a dentigerous cyst.’ The AOT has also been reported to occur with cystic features.r”“’ We believe that this case represents an odontogenic cyst that developed “hamartomatous” proliferations containing both epithelial and mesenchymal components. It is interesting to speculate whether this lesion had the potential of developing into a more aggressive odontogenic neoplasm. It has been claimed that ameloblastomas may originate from a preexisting ameloblastic fibro-odontoma or odontoma.” Odontogenic cysts showing aggressive growth with neoplastic potential have been reported by Eversole et al,13 Van der
Discussion
The patient, a 15-year-old boy, had an asymptomatic lesion involving the left maxillary sinus. The lesion contained the crown of an unerupted tooth and an irregularly-formed, calcified mass beneath the nasal side of the orbit with expansion of the overlying bone (Fig lA,B). The lesion was completely enucleated; there was no adhesion to the sinus wall. Grossly, the lesion (4.0 X 4.0 cm) appeared cystic and contained a malformed, craterlike calcified mass and the crown of an unerupted tooth (Figs lC, 2). Follow-up for a period of 5 years has shown no recurrence. Microscopic examination exhibited a thick-walled cystic structure (Fig 3A) lined by flattened squamous epithelium in the majority of the lesion (Fig 3C). The malformed calcified
Received from Meikai University School of Dentistry, Sakado, Saitama, Japan. * Associate Professor, Department of Oral Pathology. t Lecturer, Department of Oral and Maxillofacial Surgery. $ Professor and Chairman, Department of Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Tajima: Department of Oral Pathology, Meikai University School of Dentistry, Sakado, Saitama 350-02, Japan. 0 1992 American
Association
of Oral and Maxillofacial
Surgeons
0278-2391/92/5002-0018$3.00/O
190
TAJIMA, SAKAMOTO, AND YAMAMOTO
191
FIGURE 1. Water’s (A) and panoramic (B) radiographs showing a relatively well-defined radiopaque mass (arrows) and crown of an unerupted tooth (arrowheads) in the superior portion of the maxillary sinus beneath the orbit. C, Radiograph of the excised specimen showing an irregularly formed radiopacity in close proximity to the embedded tooth.
FIGURE 2. The gross appearance of specimen, showing a cystic configuration with a craterlike calcified mass at the center.
CYSTOGENIC ADENOMATOID
ODONTOGENIC
TUMOR
FIGURE 3. A, Low-power photomicrograph of the entire specimen. The lesion is composed of a cystic structure exhibiting dentinoid material (Dn) and a small focus of epithelial mass (large arrow) (hematoxylin-eosin stain. original magnification X5). B, The epithelial components consist of swirling polyhedral and spindle-shaped cells forming ductlike structures. Tall columnar cells (Ab), resembling ameloblasts, are seen (hematoxylin-eosin stain, original magnification X 100). C, The cyst is lined in large part by flattened squamous epithelium (arrows). Dentinoid material (Dn) is present adjacent to the cystic lumen (L) (hematoxylin-eosin stain, original magnification X200). D. Photomicrograph of the area showing immature enamellike material (Em) closely apposed to the columnar ameloblastlike cells and dentinoid material (hematoxylineosin stain, original magnification X200).
Waal et a1,14and by Waldron and Mustoe.” Whether a lesion of the type shown in this case has the potential to develop into a frank AOT is unknown. The differentiation between an odontogenic cyst and neoplasm is usually distinct. However, one might occasionally encounter difficulty in separating these two entities from microscopic examination alone. This case
emphasizes the importance of interpreting the clinical and radiologic features in conjunction with the microscopic appearance. References 1. Verbin RS, Barnes L: Surgical pathology of the head and neck. New York, NY, Dekker, 1985, p 1233
TAJIMA, SAKAMOTO, AND YAMAMOTO
2. Waldron CA: Pathology of the head and neck. Contemporary issues in surgical pathology, vol 10. New Ybtk, NY, Churchill Livingstone, 1988, p 403 3. Miles AEW: A cystic complex composite odontome. Proc R Sot Med 44:51, 1951 4. Dunlap CL, Fritzlen TJ: Cystic odontoma with concomitant adenoameloblastoma (adenoameloblastic odontoma). Oral Surg Oral Med Oral Path01 34450, 1972 5. Miller WA: Cystic odontoma or so-called adenoameloblastoma. J Path01 98:75, 1969 6. Giansanti JS. Someren A, Waldron CA: Odontogenic adenomatoid tumor (adenoameloblastoma)-Survey of I I 1cases. Oral Surg Oral Med Oral Pathol 30:69, 1970 7. Abrams AM. Melrose RJ. Howell FV: Adenoameloblastoma: A clinical pathologic study of ten new cases. Cancer 22:175, 1968 8. Slootweg PJ: An analysis of interrelationship of mixed odontogenie tumors-Ameloblastic fibroma, ameloblastic fibroodontoma, and the odontomas. Oral Surg Oral Med Oral Pathol 51:266, 1981 9. Gorlin RJ. Chaudhry AP, Pindborg JJ: Odontogenic tumors.
193
Classification, histopathology, and clinical behavior in man and’domestic animals. Cancer 14:73. 1961 10. Freedman PD. Lumerman H, Gee JK: Calcifying odontogenic cyst. A review and analysis of seventy cases. Oral Surg Oral Med Oral Path01 40:93, 1975 11. International Association of Oral Pathologists: Slide Seminar on odontogenic cysts and tumors, case 2 (contributed by Daniels TE and Hansen LS). Tokvo. Japan. International Association of Oral Pathologists 5th Riennial Congress, 1990 12. Eversole LS, Tomich CE, Cherrick HM: Histogenesis of odontogenic tumors. Oral Surg Oral Med Oral Pathol32:569, 197 1 13. Eversole LS, Sabes WR, Rovin S: Aggressive growth and neoplastic potential of odontogenic cysts. With special reference to central epidermoid and mucoepidermoid carcinomas. Cancer 35:270, 1975 14. Van der Waal 1, Rauhamaa R, Van der Kwast WAM, et al: Squamous cell carcinoma arising in the lining of odontogenic cysts. Report of 5 cases. Int J Oral Sung 14:146, 1985 15. Waldron CA, Mustoe TA: Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst. Oral Surg Oral Med Oral Path01 67:7 16, I989
