94
A DESMOPLASTIC
17. Balogh G, Inovay J: Recurrent mandibular myxoma. J Oral Surg 30:121, 1972 18. Harrison JB: Odontogenic myxoma. Ultrastructural and histochemical studies. J Clin Path01 26:570, 1973 19. Byrd DL, Kindrick RD, Dunsworth AR: Myxoma of the maxilla. J Oral Surg 31:123, 1973 20. Wood NK, Goaz PW (eds): Differential Diagnosis of Oral Lesions (ed 2). St Louis, MO, Mosby, 1980, p 409 2 1. McClure DK, Dahlin DC: Myxoma of bone. Report of three cases. Mavo Clin Proc 52:249. 1977 22. David RB, Baker RD. Alling CC: Odontogenic myxoma. J Oral Surg 36:610, 1978 23. Pindborg JJ, Kramer IRH (eds): Histological typing of odontogenic tumours, jaw cysts, and allied lesions. Intemational Histological Classification of Tumours (no. 5). Geneva, World Health Organization, 1971 24. Alderson GL: Myxoma of the maxilla and mandible. Paper presented at the Thirtieth Annual Meeting of AAOP, Atlanta, GA, April 1976 25. Ebling H, Wagner JE: Recurrent myxoma. Oral Surg 21:94, 1966 26. Kangur TT, Dahlin DC, Turlington EG: Myxomatous tumours of the iaws. J Oral Sura 33:523. 1975 27. Harder F: Myxomas of the jaws.-Int J Oral Surg 7:148, 1978 28. Sveen K, Band G, Gilhuus-Moe 0: Myxom i Kjevene. Nor Tandlaegeforen Tid 6:230, 1975 29. Buchner A, Ramon Y: Fibromyxoma of the maxilla. J Oral Surg 23: 145, 1965 30. Schneider LL, Weisinger E: Odontogenic tibromyxoma aris-
J Oral Maxillofac 49:94-97,
VARIANT OF AMELOBLASTOMA
ing from the periodontal ligament. J Periodontol 46:493, 1975 3 1. Stewart SS, Baum SM, Arlen M, et al: Myxoma of the lower jaw. Oral Surg 36:800, 1973 32. Westwood RM, Alexander RW, Bennet DE: Giant odontogenie myxofibroma. Oral Surg 37:83, 1974 33. Slootweg PJ, Van De Bos T, Straks W. Glycosaminoglycans in myxoma of the jaws. A biochemical study. J Oral Path01 14:299, 1985 34. Prout RES, Hodson JJ: Analysis of the mucopolysaccharide of a myxoma of the mandible. Nature 21899, 1968 35. Auriol M, Chomette G, Martin0 R, et al: Myxome odontogene. Etude histolenzymoloqiue et ultrastructurale. A propos de 5 observations. J Biol Buccale 14:215, 1986 36. Goldblatt LI: Ultrastructural study of an odontogenic myxoma. Oral Surg 42:206, 1976 37. White DK, Chen SY, Mohnac AM, et al: Odontogenic myxoma. A clinical and ultrastructural study. Oral Surg 39901, 1975 38. Simes RJ, Barros RE, Klein-Szanto AJP, et al: Ultrastructure of an odontogenic myxoma. Oral Surg 39:640, 1975 39. Le Doussal V, Mahe E, Hebert H: Les fibromyxomes odontogbnes. Etude histologique, histochimique et ultrastructurale d’un cas avec revue de la litttrature. Arch Anat Cytol Path01 29:325, 1981 40. Mori M, Murakami M, Hirose I, et al: Histochemical studies of myxoma of the jaws. J Oral Surg 33529, 1975 41. Hendler BH, Abaza NA, Quinn P: Odontogenic myxoma. Surgical management and an ultrastructural study. Oral Surg 47:203, 1979
Surg
1991
A Case of Desmoplastic Variant of a Mandibular Ameloblastoma KEIJI TANIMOTO, DDS, PHD,* TAKASHI TAKATA, DDS, PHD,t YOSHIKAZU SUEI, DDS,$ AND TAKURO WADA, DDS, PHD§
Radiographically, typical ameloblastomas have a multilocular or unilocular radiolucent appearance with fairly well-defined margins. Adjacent teeth may be tilted or displaced and root resorption is commonly observed. This article reports a case of an ameloblastoma having an unusual
Received from Hiroshima University, School of Dentistry, Japan. * Associate Professor, Department of Oral Radiology. t Assistant Professor, Clinical Laboratory, Dental Hospital. $ Research Associate, Department of Oral Radiology. 8 Professor and Chairman, Department of Oral Radiology. 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 1991 American geons
Association
0278-2391/91/4901-0019$3.00/O
of Oral and Maxillofacial
Sur-
radiographic tology.
appearance
in spite of the typical his-
Report of a Case A 24-year-old Japanese woman noticed a painless swelling of the alveolar region of the left mandible, gradually enlarging during the past 3 years. She was referred to our hospital for treatment. Intraoral examination (Fig 1) showed a buccolingual swelling of the alveolar process that extended from the left lateral incisor to the first molar region. The lingual expansion was more prominent. The lingual and occlusal gingiva was red-colored and had a granular surface. The first premolar was displaced bucally and separated from the adjacent teeth by the tumorous swelling. Half of the crown of the first premolar was buried in the tumor. The canine and premolars were all vital, and exhibited slight mobility but no pain on percussion. Radiographic examination (Fig 2) demonstrated an illdefined
radiolucency
extending
from the left canine to the
95
TANIMOTO ET AL
FIGURE 1. Clinical photograph showing lingual and occlusal expansion of the tumor with a red-colored granular surface. molar region, completely surrounding the first premolar. The lesion lacked a honeycombed, soap-bubble or unicystic appearance. Partial resorption of the alveolar crest was seen between the canine and the second premolar. The lamina dura and periodontal space of the involved teeth were partially obliterated, but root resorption was not present. Lingual expansion with perforation was seen in the occlusal view, and increased radiolucency from the left central incisor to the first molar also was seen (Fig 3). The lesion was treated by the marginal resection, with extraction of the left incisors, canine, premolars and hemisection of the fist molar. One-year follow-up shows no evidence of recurrence (Fig 4). HISTOLOGY Proliferating islands of an epithelial tumor were seen infiltrating the alveolar bone adjacent to the involved teeth (Fig 5). Although bony trabeculae were extensively destroyed and replaced by tumor tissue in some areas, tumor islands often infiltrated marrow spaces between intact bone trabeculae. Focally, cortical bone was perforated by the tumor tissue. Tooth resorption was not seen.
FIGURE 2. Panoramic radiograph showing increased radiolucency between canine and first molar. Tooth divergence and displacement are seen.
FIGURE 3. Occlusal view showing lingual cortical plate destruction with tumor expansion. A radiolucent zone also is seen from labial side of left central incisor to first molar (arrows). Note the displacement
of first premolar.
The infiltrating tumor, composed of many epithelial islands, showed typical features of desmoplastic ameloblastoma (Fig 6). Tumor islands were surrounded by densely fibrosed stromal tissue. Although some of the tumor islands showed the typical structure of follicular ameloblastoma, most of them were composed of peripheral cuboidal cells and central spindle or polygonal cells. Cystic change of both the parenchymal and stromal tissues was minimal. The tumor tissue fused with the gingival epithelium at the surface and showed papillary epithelial proliferation resembling the histologic features of peripheral ameloblastoma.
Discussion The radiographic presentation of ameloblastoma is not always pathognomonic, but is frequently sug-
96
A DESMOPLASTIC
VARIANT OF AMELOBLASTOMA
FIGURE 4. Panoramic view taken at 1 year after operation. A smooth intact cortex is seen and there is no evidence of recurrence.
gestive. Most present as cystic lesions with either a unilocular or multilocular pattern, with or without bony expansion. l-4 Root resorption is also common.5 In the present case, neither a cystic appear-
FIGURE 5. Low-power photomicrographic scan showing extensive destruction of the alveolar process by tumor islands in a desmoplastic stroma. No root resorption is seen (insert). The gingival surface shows papillary proliferation resembling peripheral ameloblastoma(*).
ante nor root resorption were observed, but rather, an ill-defined, infiltrative radiolucency. These findings did not suggest ameloblastoma, and were more suggestive of other tumors such as a
TANIMOTO ET AL
FIGURE 6. The tumor tissue showing the features of desmoplastic ameloblastoma. Tumor islands were surrounded by densely fibrosed stromal tissue. Although some of tumor islands showed the typical structure of follicular ameloblastoma(*), most of tumor islands were composed of peripheral cuboidal cells and central spindle or polygonal cells.
fibro-osseous lesion, malignant tumor of hematopoietic origin, or chondrosarcoma. Eversole et al reported three cases of an atypical form of ameloblastoma characterized by extensive desmoplasia and unusual radiographic findings.6 Recently, Waldron et al7 reviewed 116 cases of ameloblastoma and found 14 cases of this type. They named it desmoplastic variant of ameloblastoma and described the histologic features characterized by an extensive collagenized stroma containing small islands of tumor epithelium with scant tendency to form cystic structures. This variant has a marked tendency to involve the anterior portion of the jaws and present unusual radiographic findings more suggestive of a benign fibro-osseous lesion than an ameloblastoma. These characteristics are well represented in the present case. A case of ameloblastoma showing unique radiographic features reported by Okada et al8 also seems to be a desmoplastic variant. The question arises as to why this lesion showed such unusual radiographic features. The surface growth of the lesion could lead one to suspect that the origin of the tumor was peripheral and that the tumor secondarily infiltrated underlying bone. However, the main portion of the lesion involved the alveolar bone and body of mandible. The biologic behavior of the peripheral ameloblastoma lacks the persistent invasiveness of the intraosseous
lesion.’ Furthermore, unusual radiographic features of this tumor do not suggest a peripheral type. Therefore, it is reasonable to consider that the tumor originated centrally within the alveolar bone and fused with the surface gingival epithelium. Minimal cystic change of both the parenchymal and desmoplastic stromal tissue and tumor infiltration into the marrow spaces within intact bone trabeculae may explain the unique radiographic features of this tumor. References 1. Pindborg JJ, Kramer IRH: Histological typing of odontogenit tumours, jaw cysts, and allied lesions, in International Histological Classification of Tumors, no 5. Geneva, Switzerland, World Health Organization, 1971, p 24 2. Thunthv KH: Dental Radioaranhic Diaanosis. Snrinafield. IL, Charles C Thomas, 1988:~ 351 _ 3. Gibilisco JA: Stafne’s Oral Radiographic Diagnosis (ed 5). Philadelphia, PA, Saunders, 1985, p 181 4. Goaz PW, White SC: Oral Radiology principles and interpretation (ed 2). St Louis, MO, Mosby, 1987, p 520 5. St&hers P, Shear M: Root resorption by ameloblastomas and cvsts of the iaws. Int J Oral Sura 5:128. 1976 6. Eversole LR, Leider AS, Hansen LS: Ameloblastoma with pronounced desmoplasia. J Oral Maxillofac Surg 42:734, 1984 7. Waldron CA, El-Mofty SK: A histopathologic study of 116 ameloblastomas with special reference to the desmoplastic variant. Oral Surg 63:441, 1987 8. Okada Y, Sugimura M, Ishida T: Amelobiastoma accompanied by prominent bone formation. J Oral Maxillofac Surg 44:555, 1986 9. Shafer WG, Hine MK, Levy BN: A textbook of oral pathology (ed 4). Philadelphia, PA, Saunders, 1983, p 276
A DESMOPLASTIC
17. Balogh G, Inovay J: Recurrent mandibular myxoma. J Oral Surg 30:121, 1972 18. Harrison JB: Odontogenic myxoma. Ultrastructural and histochemical studies. J Clin Path01 26:570, 1973 19. Byrd DL, Kindrick RD, Dunsworth AR: Myxoma of the maxilla. J Oral Surg 31:123, 1973 20. Wood NK, Goaz PW (eds): Differential Diagnosis of Oral Lesions (ed 2). St Louis, MO, Mosby, 1980, p 409 2 1. McClure DK, Dahlin DC: Myxoma of bone. Report of three cases. Mavo Clin Proc 52:249. 1977 22. David RB, Baker RD. Alling CC: Odontogenic myxoma. J Oral Surg 36:610, 1978 23. Pindborg JJ, Kramer IRH (eds): Histological typing of odontogenic tumours, jaw cysts, and allied lesions. Intemational Histological Classification of Tumours (no. 5). Geneva, World Health Organization, 1971 24. Alderson GL: Myxoma of the maxilla and mandible. Paper presented at the Thirtieth Annual Meeting of AAOP, Atlanta, GA, April 1976 25. Ebling H, Wagner JE: Recurrent myxoma. Oral Surg 21:94, 1966 26. Kangur TT, Dahlin DC, Turlington EG: Myxomatous tumours of the iaws. J Oral Sura 33:523. 1975 27. Harder F: Myxomas of the jaws.-Int J Oral Surg 7:148, 1978 28. Sveen K, Band G, Gilhuus-Moe 0: Myxom i Kjevene. Nor Tandlaegeforen Tid 6:230, 1975 29. Buchner A, Ramon Y: Fibromyxoma of the maxilla. J Oral Surg 23: 145, 1965 30. Schneider LL, Weisinger E: Odontogenic tibromyxoma aris-
J Oral Maxillofac 49:94-97,
VARIANT OF AMELOBLASTOMA
ing from the periodontal ligament. J Periodontol 46:493, 1975 3 1. Stewart SS, Baum SM, Arlen M, et al: Myxoma of the lower jaw. Oral Surg 36:800, 1973 32. Westwood RM, Alexander RW, Bennet DE: Giant odontogenie myxofibroma. Oral Surg 37:83, 1974 33. Slootweg PJ, Van De Bos T, Straks W. Glycosaminoglycans in myxoma of the jaws. A biochemical study. J Oral Path01 14:299, 1985 34. Prout RES, Hodson JJ: Analysis of the mucopolysaccharide of a myxoma of the mandible. Nature 21899, 1968 35. Auriol M, Chomette G, Martin0 R, et al: Myxome odontogene. Etude histolenzymoloqiue et ultrastructurale. A propos de 5 observations. J Biol Buccale 14:215, 1986 36. Goldblatt LI: Ultrastructural study of an odontogenic myxoma. Oral Surg 42:206, 1976 37. White DK, Chen SY, Mohnac AM, et al: Odontogenic myxoma. A clinical and ultrastructural study. Oral Surg 39901, 1975 38. Simes RJ, Barros RE, Klein-Szanto AJP, et al: Ultrastructure of an odontogenic myxoma. Oral Surg 39:640, 1975 39. Le Doussal V, Mahe E, Hebert H: Les fibromyxomes odontogbnes. Etude histologique, histochimique et ultrastructurale d’un cas avec revue de la litttrature. Arch Anat Cytol Path01 29:325, 1981 40. Mori M, Murakami M, Hirose I, et al: Histochemical studies of myxoma of the jaws. J Oral Surg 33529, 1975 41. Hendler BH, Abaza NA, Quinn P: Odontogenic myxoma. Surgical management and an ultrastructural study. Oral Surg 47:203, 1979
Surg
1991
A Case of Desmoplastic Variant of a Mandibular Ameloblastoma KEIJI TANIMOTO, DDS, PHD,* TAKASHI TAKATA, DDS, PHD,t YOSHIKAZU SUEI, DDS,$ AND TAKURO WADA, DDS, PHD§
Radiographically, typical ameloblastomas have a multilocular or unilocular radiolucent appearance with fairly well-defined margins. Adjacent teeth may be tilted or displaced and root resorption is commonly observed. This article reports a case of an ameloblastoma having an unusual
Received from Hiroshima University, School of Dentistry, Japan. * Associate Professor, Department of Oral Radiology. t Assistant Professor, Clinical Laboratory, Dental Hospital. $ Research Associate, Department of Oral Radiology. 8 Professor and Chairman, Department of Oral Radiology. 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 1991 American geons
Association
0278-2391/91/4901-0019$3.00/O
of Oral and Maxillofacial
Sur-
radiographic tology.
appearance
in spite of the typical his-
Report of a Case A 24-year-old Japanese woman noticed a painless swelling of the alveolar region of the left mandible, gradually enlarging during the past 3 years. She was referred to our hospital for treatment. Intraoral examination (Fig 1) showed a buccolingual swelling of the alveolar process that extended from the left lateral incisor to the first molar region. The lingual expansion was more prominent. The lingual and occlusal gingiva was red-colored and had a granular surface. The first premolar was displaced bucally and separated from the adjacent teeth by the tumorous swelling. Half of the crown of the first premolar was buried in the tumor. The canine and premolars were all vital, and exhibited slight mobility but no pain on percussion. Radiographic examination (Fig 2) demonstrated an illdefined
radiolucency
extending
from the left canine to the
95
TANIMOTO ET AL
FIGURE 1. Clinical photograph showing lingual and occlusal expansion of the tumor with a red-colored granular surface. molar region, completely surrounding the first premolar. The lesion lacked a honeycombed, soap-bubble or unicystic appearance. Partial resorption of the alveolar crest was seen between the canine and the second premolar. The lamina dura and periodontal space of the involved teeth were partially obliterated, but root resorption was not present. Lingual expansion with perforation was seen in the occlusal view, and increased radiolucency from the left central incisor to the first molar also was seen (Fig 3). The lesion was treated by the marginal resection, with extraction of the left incisors, canine, premolars and hemisection of the fist molar. One-year follow-up shows no evidence of recurrence (Fig 4). HISTOLOGY Proliferating islands of an epithelial tumor were seen infiltrating the alveolar bone adjacent to the involved teeth (Fig 5). Although bony trabeculae were extensively destroyed and replaced by tumor tissue in some areas, tumor islands often infiltrated marrow spaces between intact bone trabeculae. Focally, cortical bone was perforated by the tumor tissue. Tooth resorption was not seen.
FIGURE 2. Panoramic radiograph showing increased radiolucency between canine and first molar. Tooth divergence and displacement are seen.
FIGURE 3. Occlusal view showing lingual cortical plate destruction with tumor expansion. A radiolucent zone also is seen from labial side of left central incisor to first molar (arrows). Note the displacement
of first premolar.
The infiltrating tumor, composed of many epithelial islands, showed typical features of desmoplastic ameloblastoma (Fig 6). Tumor islands were surrounded by densely fibrosed stromal tissue. Although some of the tumor islands showed the typical structure of follicular ameloblastoma, most of them were composed of peripheral cuboidal cells and central spindle or polygonal cells. Cystic change of both the parenchymal and stromal tissues was minimal. The tumor tissue fused with the gingival epithelium at the surface and showed papillary epithelial proliferation resembling the histologic features of peripheral ameloblastoma.
Discussion The radiographic presentation of ameloblastoma is not always pathognomonic, but is frequently sug-
96
A DESMOPLASTIC
VARIANT OF AMELOBLASTOMA
FIGURE 4. Panoramic view taken at 1 year after operation. A smooth intact cortex is seen and there is no evidence of recurrence.
gestive. Most present as cystic lesions with either a unilocular or multilocular pattern, with or without bony expansion. l-4 Root resorption is also common.5 In the present case, neither a cystic appear-
FIGURE 5. Low-power photomicrographic scan showing extensive destruction of the alveolar process by tumor islands in a desmoplastic stroma. No root resorption is seen (insert). The gingival surface shows papillary proliferation resembling peripheral ameloblastoma(*).
ante nor root resorption were observed, but rather, an ill-defined, infiltrative radiolucency. These findings did not suggest ameloblastoma, and were more suggestive of other tumors such as a
TANIMOTO ET AL
FIGURE 6. The tumor tissue showing the features of desmoplastic ameloblastoma. Tumor islands were surrounded by densely fibrosed stromal tissue. Although some of tumor islands showed the typical structure of follicular ameloblastoma(*), most of tumor islands were composed of peripheral cuboidal cells and central spindle or polygonal cells.
fibro-osseous lesion, malignant tumor of hematopoietic origin, or chondrosarcoma. Eversole et al reported three cases of an atypical form of ameloblastoma characterized by extensive desmoplasia and unusual radiographic findings.6 Recently, Waldron et al7 reviewed 116 cases of ameloblastoma and found 14 cases of this type. They named it desmoplastic variant of ameloblastoma and described the histologic features characterized by an extensive collagenized stroma containing small islands of tumor epithelium with scant tendency to form cystic structures. This variant has a marked tendency to involve the anterior portion of the jaws and present unusual radiographic findings more suggestive of a benign fibro-osseous lesion than an ameloblastoma. These characteristics are well represented in the present case. A case of ameloblastoma showing unique radiographic features reported by Okada et al8 also seems to be a desmoplastic variant. The question arises as to why this lesion showed such unusual radiographic features. The surface growth of the lesion could lead one to suspect that the origin of the tumor was peripheral and that the tumor secondarily infiltrated underlying bone. However, the main portion of the lesion involved the alveolar bone and body of mandible. The biologic behavior of the peripheral ameloblastoma lacks the persistent invasiveness of the intraosseous
lesion.’ Furthermore, unusual radiographic features of this tumor do not suggest a peripheral type. Therefore, it is reasonable to consider that the tumor originated centrally within the alveolar bone and fused with the surface gingival epithelium. Minimal cystic change of both the parenchymal and desmoplastic stromal tissue and tumor infiltration into the marrow spaces within intact bone trabeculae may explain the unique radiographic features of this tumor. References 1. Pindborg JJ, Kramer IRH: Histological typing of odontogenit tumours, jaw cysts, and allied lesions, in International Histological Classification of Tumors, no 5. Geneva, Switzerland, World Health Organization, 1971, p 24 2. Thunthv KH: Dental Radioaranhic Diaanosis. Snrinafield. IL, Charles C Thomas, 1988:~ 351 _ 3. Gibilisco JA: Stafne’s Oral Radiographic Diagnosis (ed 5). Philadelphia, PA, Saunders, 1985, p 181 4. Goaz PW, White SC: Oral Radiology principles and interpretation (ed 2). St Louis, MO, Mosby, 1987, p 520 5. St&hers P, Shear M: Root resorption by ameloblastomas and cvsts of the iaws. Int J Oral Sura 5:128. 1976 6. Eversole LR, Leider AS, Hansen LS: Ameloblastoma with pronounced desmoplasia. J Oral Maxillofac Surg 42:734, 1984 7. Waldron CA, El-Mofty SK: A histopathologic study of 116 ameloblastomas with special reference to the desmoplastic variant. Oral Surg 63:441, 1987 8. Okada Y, Sugimura M, Ishida T: Amelobiastoma accompanied by prominent bone formation. J Oral Maxillofac Surg 44:555, 1986 9. Shafer WG, Hine MK, Levy BN: A textbook of oral pathology (ed 4). Philadelphia, PA, Saunders, 1983, p 276
