J Oral Mawillofac 50:282-285,
Surg
1992
Mandibular Adenomatoid Odontogenic Tumor With Unusual Clinicopathologic Features MASAHISA
NOMURA, DDS, PHD,* KElJl TANIMOTO, DDS, PtiD,t TAKASHI TAKATA, DDS, PHD,$ AND TSUNEHIRO SHIMOSATO, MD, DDS, PHD$
Adenomatoid odontogenic tumor (AOT) is a relatively uncommon odontogenic tumor characterized by the formation of ductlike structures by the epithelial component of the lesion.’ According to the reports of Giansanti et al,* Toida et al,3 and Philipsen et ak4 cases occur predominantly in the second and third decades of life, with few cases occurring in people over 50 years old. The usual site is the anterior maxillary region. The most common radiographic appearance of AOT is a sharply circumscribed, unilocular radiolucency, often associated with the crown of an unerupted anterior maxillary tooth (especially canine), resembling a dentigerous cyst. Detectable radiopacities were reported in two thirds of the case~.~The AOT reported here showed unusual clinicopathologic features.
1) disclosed a well-defined, 5 X 3%cm, granulomatous mass in the left mandibular region. On palpation, a bony defect filled with a pulsatory soft-tissue mass, which had a bleeding tendency, was noted in the region. A panoramic radiograph (Fig 2) showed a mixed radiolucent-radiopaque lesion with an ill-defined border extending from the right central incisor to the left second molar region. The left lower premolars and molars were missing. The inferior alveolar canal was displaced inferiorly by the lesion. An irregular type of root resorption was observed on the distal surface of the left lower canine. Protrusion and slight displacement of the teeth from the right central incisor to the left canine also were seen. Occlusal radiographs (Fig 3) showed destruction of the buccal plate. These clinical and radiographic findings suggested a malignant mesenchymal tumor such as osteosarcoma or chondrosarcoma. However, an aggressive type of fibro-osseous lesion, or an odontogenic tumors such as Pindborg’s tumor and Gorlin’s cyst, could not be excluded. Biopsy showed an AOT without evidence of malignancy. Therefore a partial en bloc resection of the mandible and immediate reconstruction with an iliac bone graft was performed. The patient had an uneventful postoperative course. A panoramic radiograph (Fig 4) taken 2% years postoperatively showed good healing. Soft x-ray radiographs of the resected mandible revealed that the buccal plate was completely destroyed by the tumor. Multilocularity was evident in some areas, which could not
Report of a Case A 64-year-old Japanese man was referred to the Hiroshima University Dental Hospital with a history of swelling in the left mandible and hypesthesia of the left mental region of 3 months’ duration. The patient noticed a slowly growing, painless gingival swelling in the left lower premolar-molar region for the past 2 years after the extraction of two lower teeth. The patient’s medical history was noncontributory. At the time of examination, the patient’s face was asymmetrical due to a diffuse swelling of the left mandible. There was no regional lymphadenopathy. Intraoral examination (Fig
Received from Hiroshima University School of Dentistry, Hiroshima, Japan. * Research Associate, Department of Oral and Maxillofacial Surgery II. t Associate Professor, Department of Oral Radiology. $ Assistant Professor, Clinical Laboratory, Dental Hospital. 4 Professor and Chairman, Department of Oral and Maxillofacial Surgery II. Address correspondence and reprint requests to Dr Tanimoto: Department of Oral Radiology, Hiroshima University School of Dentistry, l-2-3, Kasumi, Minami-ku, Hiroshima, 734 Japan. 0 1992 American
Association
of Oral and Maxillofacial
Surgeons
FIGURE 1. Clinical photograph of vascular-appearing granulomatous tumor.
0278-2391/92/5003-0013$3.00/O
282
NOMURA ET AL
Panoramic radiograph showing a mixed radiolucentradiopaque lesion from the right central incisor to the left second molar region. Note the large amorphous radiopacity in the left mandible and the crowding and protrusion of the remaining left anterior
FIGURE 2.
teeth.
be recognized on routine radiographs (Figs 3,5A). The mandibular canal was displaced and surrounded by tissue with increased radiolucency. The radiopacity in the mandibular bone resembled a cemental mass (Fig 5B). Histologically, the tumor was without a defined fibrous capsule and extensively occupied the mandibular body. The lesion had proliferated intraorally into overlying gingiva and
FIGURE 3. Occlusal radiographs showing buccal plate destruction (A) and a Codman’s trianglelike pexiosteal reaction (B; white arrow). Small radiopacities are scattered in the buccal soft-tissue mass.
FIGURE 4. Panoramic radiograph taken 2% years postoperation showing good healing.
destroyed the buccal cortical plate of the mandible. The tumor was composed of solid sheets of polyhedral and spindleshaped epithelial cells. Variable sized ductlike structures lined by columnar cells, and whorled spheroidal masses of tumor cells, were observed in some nests (Fig 6). Lumina of the ductlike structures sometimes contained fibrillar mucoid material. Eosinophilic droplets stained with periodic acidSchiff were seen in the tumor cells. In the periphery of the epithelial sheets, the tumor cells often differentiated into columnar cells similar to ameloblasts. Homogeneous eosinophilic structures, thought to be dysplastic dentin, were often
284
MANDIBULAR
ADENOMATOID
ODONTOGENIC
TUMOR
FIGURE 5. Soft x-ray radiographs showing (A) multilocularity and (B) deformity of the mandibular canal (arrow) surrounded by increased radiolucency and a large sclerotic mass in the body of the mandible.
observed between the columnar cells and stromal connective tissues, which sometimes showed odontoblastlike differentiation (Fig 7). At the periphery of the tumor, an extensive amount of osteodentin or cementumlike tissue was seen in close relation to the lesion (Fig 7). The stromal connective tissue was generally scanty, but it was often cellular and vascular in the extraosseous gingival portion of the tumor. Although there were mitotic figures in both the epithelial tumor cells and stromal cells, the lesion was not considered to be malignant.
FIGURE 6. Variable-sized ductlike structures (*) lined by columnar cells and whorled spheroidal masses of tumor cells (arrows), with intervening areas of spindle-shaped cells (hematoxylin-eosin stain, original magnification X300).
Discussion The case presented is of particular interest because of the noted differences from a typical AOT4; ie, age, gender, location, size, and clinical, radiographic, and histologic findings. A tumor reported by Meyer et al’
FIGURE 7. A hyalinized band of dysplastic dentinlike tissue (HD) and a mass of osteodentin or cementumlike tissue (OD/C) are noted at the periphery of the tumor. Tumor cells in the periphery of the epithelial sheets are often differentiated into columnar cells similar to ameloblasts (arrowheads). *Stromal connective tissue shows odontoblastic differentiation (hematoxylin-eosin stain, original magnification X 150).
UEDA AND KANEDA
was in an 82-year-old woman; however, only three other cases, in addition to the present one, have been recorded in patients over the age of 64.4 The lesion in our patient was located in the mandibular molar region and had no relation to an unerupted tooth. It appeared as a vascular-rich tumor mass intraorally, and caused hypesthesia. Massive calcification and mitotic figures were also peculiarities seen in this case. In a review of the radiographic literature,2‘6 almost all AOTs were reported to have a cystic appearance, and the prevalent location was in the anterior maxillary region in association with an impacted tooth. In our case, there was no cystic radiolucency and no impacted tooth, but there was a mixed radiolucent-radiopaque area in the mandible. The distribution of the specks of radiopacity within the soft-tissue mass was suggestive of the extraosseous type of Pindborg’s tumor, Gorlin’s cyst, or fibro-osseous lesion. The massive radiopacity in the mandibular body gave an impression of gigantiform cementoma. The ill-defined radiolucency and the perforation of the buccal plate suggested a malignant tumor. Toida et al3 reported 126 cases of AOT, but no preoperative diagnosis of malignancy was made. Therefore, we believe the radiographic findings in our case are quite unique. Radiopacity at the periphery of the tumor is also atypical in AOT and confused the clinical diagnosis of the lesion. The dense mass was suspected to be of odontogenic origin because of the close relationship of the osteodentin or cementumlike tissue and the tumor tissue. Only normal bone marrow was seen in the area of the increased radiolucency.
J Oral Maxillofac 5O:285-287.
Pulsation and a bleeding tendency were observed in our case. These findings led the oral surgeons to believe the lesion might be a vascular tumor. Philipsen and Bim6 found a pronounced vascularity in the stroma of the lesions reported by them. They considered it to be a secondary phenomenon, with no indication of the vascular proliferation being neoplastic. An increased vascularity of the stroma was observed in the extraosseous gingival portion of the present tumor. Enucleation and curettage2*3 are recommended treatment for AOT; however, a mandibular resection has been reported.5 In our case, an en bloc resection of the mandible was performed because of the extensive involvement of the mandible and the high risk of fracture.
References
1. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983. p 289 2. Giansanti JS, Someren A, Waldron CA: Odontogenic adenomatoid tumor (adenoameloblastoma). Survey of I 11 cases. Oral Surg Oral Med Oral Path01 30:69, 1970 3. Toida M, Hyodo 1, Okuda T, et al: Adenomatoid odontogenic tumor: Report of two cases and survey of 126 cases in Japan. J Oral Maxillofac Surg 48:404, 1990 4. Philipsen HP, Reichart PA, Zhang KH, et al: Adenomatoid odontogenic tumor: Biologic profile based on 499 cases. J Oral Path01 Med 20: 149, 1991 5. Meyer I. Giunta JL: Adenomatoid odontogenic tumor (adenoameloblastoma): Report of case. J Oral Surg 32:448, 1974 6. Philipsen HP, Bim H: The adenomatoid odontogenic tumour. Ameloblastic adenomatoid tumour or adeno-ameloblastoma. Acta Path01 Microbial Stand 75:375, 1969
Surg
1992
Maxillary Sinusitis Caused by Dental Implants: Report of Two Cases MINORU
UEDA, DDS, PHD,* AND TOSHIO
Received from the Department of Oral Surgery, Nagoya University School of Medicine. Nagova. Janan. * Associate Professor- . _ t Professor. Address correspondence and reprint requests to Dr Ueda: Department of Oral Surgery, Nagoya University School of Medicine, 65 Tsuruma-cho, Showa-ku, Nagoya, 466, Japan. 0 1992 American
Association
0278-2391/92/5003-0014$3.00/O
of Oral and Maxillofacial
Surgeons
KANEDA,
DDS, PHD~
Many types of complications have been reported following treatment with dental implants. IV3Local infection of the per&implant tissue is the most common complication, and in such cases there can be extensive resorption of the bone surrounding the implant. Implants inserted close to the maxillary sinus also provide a route for spread of infection from the mouth following poor oral hygiene. When the maxillary dental im-
Surg
1992
Mandibular Adenomatoid Odontogenic Tumor With Unusual Clinicopathologic Features MASAHISA
NOMURA, DDS, PHD,* KElJl TANIMOTO, DDS, PtiD,t TAKASHI TAKATA, DDS, PHD,$ AND TSUNEHIRO SHIMOSATO, MD, DDS, PHD$
Adenomatoid odontogenic tumor (AOT) is a relatively uncommon odontogenic tumor characterized by the formation of ductlike structures by the epithelial component of the lesion.’ According to the reports of Giansanti et al,* Toida et al,3 and Philipsen et ak4 cases occur predominantly in the second and third decades of life, with few cases occurring in people over 50 years old. The usual site is the anterior maxillary region. The most common radiographic appearance of AOT is a sharply circumscribed, unilocular radiolucency, often associated with the crown of an unerupted anterior maxillary tooth (especially canine), resembling a dentigerous cyst. Detectable radiopacities were reported in two thirds of the case~.~The AOT reported here showed unusual clinicopathologic features.
1) disclosed a well-defined, 5 X 3%cm, granulomatous mass in the left mandibular region. On palpation, a bony defect filled with a pulsatory soft-tissue mass, which had a bleeding tendency, was noted in the region. A panoramic radiograph (Fig 2) showed a mixed radiolucent-radiopaque lesion with an ill-defined border extending from the right central incisor to the left second molar region. The left lower premolars and molars were missing. The inferior alveolar canal was displaced inferiorly by the lesion. An irregular type of root resorption was observed on the distal surface of the left lower canine. Protrusion and slight displacement of the teeth from the right central incisor to the left canine also were seen. Occlusal radiographs (Fig 3) showed destruction of the buccal plate. These clinical and radiographic findings suggested a malignant mesenchymal tumor such as osteosarcoma or chondrosarcoma. However, an aggressive type of fibro-osseous lesion, or an odontogenic tumors such as Pindborg’s tumor and Gorlin’s cyst, could not be excluded. Biopsy showed an AOT without evidence of malignancy. Therefore a partial en bloc resection of the mandible and immediate reconstruction with an iliac bone graft was performed. The patient had an uneventful postoperative course. A panoramic radiograph (Fig 4) taken 2% years postoperatively showed good healing. Soft x-ray radiographs of the resected mandible revealed that the buccal plate was completely destroyed by the tumor. Multilocularity was evident in some areas, which could not
Report of a Case A 64-year-old Japanese man was referred to the Hiroshima University Dental Hospital with a history of swelling in the left mandible and hypesthesia of the left mental region of 3 months’ duration. The patient noticed a slowly growing, painless gingival swelling in the left lower premolar-molar region for the past 2 years after the extraction of two lower teeth. The patient’s medical history was noncontributory. At the time of examination, the patient’s face was asymmetrical due to a diffuse swelling of the left mandible. There was no regional lymphadenopathy. Intraoral examination (Fig
Received from Hiroshima University School of Dentistry, Hiroshima, Japan. * Research Associate, Department of Oral and Maxillofacial Surgery II. t Associate Professor, Department of Oral Radiology. $ Assistant Professor, Clinical Laboratory, Dental Hospital. 4 Professor and Chairman, Department of Oral and Maxillofacial Surgery II. Address correspondence and reprint requests to Dr Tanimoto: Department of Oral Radiology, Hiroshima University School of Dentistry, l-2-3, Kasumi, Minami-ku, Hiroshima, 734 Japan. 0 1992 American
Association
of Oral and Maxillofacial
Surgeons
FIGURE 1. Clinical photograph of vascular-appearing granulomatous tumor.
0278-2391/92/5003-0013$3.00/O
282
NOMURA ET AL
Panoramic radiograph showing a mixed radiolucentradiopaque lesion from the right central incisor to the left second molar region. Note the large amorphous radiopacity in the left mandible and the crowding and protrusion of the remaining left anterior
FIGURE 2.
teeth.
be recognized on routine radiographs (Figs 3,5A). The mandibular canal was displaced and surrounded by tissue with increased radiolucency. The radiopacity in the mandibular bone resembled a cemental mass (Fig 5B). Histologically, the tumor was without a defined fibrous capsule and extensively occupied the mandibular body. The lesion had proliferated intraorally into overlying gingiva and
FIGURE 3. Occlusal radiographs showing buccal plate destruction (A) and a Codman’s trianglelike pexiosteal reaction (B; white arrow). Small radiopacities are scattered in the buccal soft-tissue mass.
FIGURE 4. Panoramic radiograph taken 2% years postoperation showing good healing.
destroyed the buccal cortical plate of the mandible. The tumor was composed of solid sheets of polyhedral and spindleshaped epithelial cells. Variable sized ductlike structures lined by columnar cells, and whorled spheroidal masses of tumor cells, were observed in some nests (Fig 6). Lumina of the ductlike structures sometimes contained fibrillar mucoid material. Eosinophilic droplets stained with periodic acidSchiff were seen in the tumor cells. In the periphery of the epithelial sheets, the tumor cells often differentiated into columnar cells similar to ameloblasts. Homogeneous eosinophilic structures, thought to be dysplastic dentin, were often
284
MANDIBULAR
ADENOMATOID
ODONTOGENIC
TUMOR
FIGURE 5. Soft x-ray radiographs showing (A) multilocularity and (B) deformity of the mandibular canal (arrow) surrounded by increased radiolucency and a large sclerotic mass in the body of the mandible.
observed between the columnar cells and stromal connective tissues, which sometimes showed odontoblastlike differentiation (Fig 7). At the periphery of the tumor, an extensive amount of osteodentin or cementumlike tissue was seen in close relation to the lesion (Fig 7). The stromal connective tissue was generally scanty, but it was often cellular and vascular in the extraosseous gingival portion of the tumor. Although there were mitotic figures in both the epithelial tumor cells and stromal cells, the lesion was not considered to be malignant.
FIGURE 6. Variable-sized ductlike structures (*) lined by columnar cells and whorled spheroidal masses of tumor cells (arrows), with intervening areas of spindle-shaped cells (hematoxylin-eosin stain, original magnification X300).
Discussion The case presented is of particular interest because of the noted differences from a typical AOT4; ie, age, gender, location, size, and clinical, radiographic, and histologic findings. A tumor reported by Meyer et al’
FIGURE 7. A hyalinized band of dysplastic dentinlike tissue (HD) and a mass of osteodentin or cementumlike tissue (OD/C) are noted at the periphery of the tumor. Tumor cells in the periphery of the epithelial sheets are often differentiated into columnar cells similar to ameloblasts (arrowheads). *Stromal connective tissue shows odontoblastic differentiation (hematoxylin-eosin stain, original magnification X 150).
UEDA AND KANEDA
was in an 82-year-old woman; however, only three other cases, in addition to the present one, have been recorded in patients over the age of 64.4 The lesion in our patient was located in the mandibular molar region and had no relation to an unerupted tooth. It appeared as a vascular-rich tumor mass intraorally, and caused hypesthesia. Massive calcification and mitotic figures were also peculiarities seen in this case. In a review of the radiographic literature,2‘6 almost all AOTs were reported to have a cystic appearance, and the prevalent location was in the anterior maxillary region in association with an impacted tooth. In our case, there was no cystic radiolucency and no impacted tooth, but there was a mixed radiolucent-radiopaque area in the mandible. The distribution of the specks of radiopacity within the soft-tissue mass was suggestive of the extraosseous type of Pindborg’s tumor, Gorlin’s cyst, or fibro-osseous lesion. The massive radiopacity in the mandibular body gave an impression of gigantiform cementoma. The ill-defined radiolucency and the perforation of the buccal plate suggested a malignant tumor. Toida et al3 reported 126 cases of AOT, but no preoperative diagnosis of malignancy was made. Therefore, we believe the radiographic findings in our case are quite unique. Radiopacity at the periphery of the tumor is also atypical in AOT and confused the clinical diagnosis of the lesion. The dense mass was suspected to be of odontogenic origin because of the close relationship of the osteodentin or cementumlike tissue and the tumor tissue. Only normal bone marrow was seen in the area of the increased radiolucency.
J Oral Maxillofac 5O:285-287.
Pulsation and a bleeding tendency were observed in our case. These findings led the oral surgeons to believe the lesion might be a vascular tumor. Philipsen and Bim6 found a pronounced vascularity in the stroma of the lesions reported by them. They considered it to be a secondary phenomenon, with no indication of the vascular proliferation being neoplastic. An increased vascularity of the stroma was observed in the extraosseous gingival portion of the present tumor. Enucleation and curettage2*3 are recommended treatment for AOT; however, a mandibular resection has been reported.5 In our case, an en bloc resection of the mandible was performed because of the extensive involvement of the mandible and the high risk of fracture.
References
1. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983. p 289 2. Giansanti JS, Someren A, Waldron CA: Odontogenic adenomatoid tumor (adenoameloblastoma). Survey of I 11 cases. Oral Surg Oral Med Oral Path01 30:69, 1970 3. Toida M, Hyodo 1, Okuda T, et al: Adenomatoid odontogenic tumor: Report of two cases and survey of 126 cases in Japan. J Oral Maxillofac Surg 48:404, 1990 4. Philipsen HP, Reichart PA, Zhang KH, et al: Adenomatoid odontogenic tumor: Biologic profile based on 499 cases. J Oral Path01 Med 20: 149, 1991 5. Meyer I. Giunta JL: Adenomatoid odontogenic tumor (adenoameloblastoma): Report of case. J Oral Surg 32:448, 1974 6. Philipsen HP, Bim H: The adenomatoid odontogenic tumour. Ameloblastic adenomatoid tumour or adeno-ameloblastoma. Acta Path01 Microbial Stand 75:375, 1969
Surg
1992
Maxillary Sinusitis Caused by Dental Implants: Report of Two Cases MINORU
UEDA, DDS, PHD,* AND TOSHIO
Received from the Department of Oral Surgery, Nagoya University School of Medicine. Nagova. Janan. * Associate Professor- . _ t Professor. Address correspondence and reprint requests to Dr Ueda: Department of Oral Surgery, Nagoya University School of Medicine, 65 Tsuruma-cho, Showa-ku, Nagoya, 466, Japan. 0 1992 American
Association
0278-2391/92/5003-0014$3.00/O
of Oral and Maxillofacial
Surgeons
KANEDA,
DDS, PHD~
Many types of complications have been reported following treatment with dental implants. IV3Local infection of the per&implant tissue is the most common complication, and in such cases there can be extensive resorption of the bone surrounding the implant. Implants inserted close to the maxillary sinus also provide a route for spread of infection from the mouth following poor oral hygiene. When the maxillary dental im-
