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Squamous odontogenic tumor. Report of a case with lesions in three quadrants.

Squamous odontogenic tumor is a rare, benign, odontogenic lesion. We are presenting the twenty-second case to be reported in the literature and only the fifth case with multiple quadrant lesions. This is the first description of reconstruction with autologous and homologous bone grafts of a discontinuity defect of the mandible resulting from en bloc resection of a squamous odontogenic tumor. Emphasis is placed on the need to consider this rare tumor in the differential diagnosis of ameloblastoma. The correct diagnosis significantly changes the extent of the surgical procedure and may avoid unnecessary morbidity.

Adult↗

Aggressive (malignant?) epithelial odontogenic ghost cell tumor.

Although the calcifying odontogenic cyst (COC) is a well-established pathologic entity, it has been recognized that there is an odontogenic neoplasm with histologic features similar to the COC that has often been diagnosed as COC. Investigators have applied several terms to this neoplasm, but we prefer the term epithelial odontogenic ghost cell tumor (EOGCT). No reports have appeared previously in the literature describing either carcinoma arising in a COC or malignant EOGCT. Three cases are reported here with aggressive growth and histologic features strongly implying a malignant potential, although none is known to have metastasized. Two occurred in the maxilla and one in the mandible. One was discovered 9 years following removal of a typical COC, and another recurred rapidly after excision.

Adolescent↗

Ameloblastic carcinoma: a clinicopathologic study and assessment of eight cases.

The term ameloblastic carcinoma is differentiated from the term malignant ameloblastoma and is defined as an ameloblastoma in which there is histologic evidence of malignancy in the primary tumor or the recurrent tumor (or metastasis), regardless of whether it has metastasized. Eight cases of ameloblastic carcinoma from the Armed Forces Institute of Pathology (AFIP) are reported. The mean age of patients was 30.1 years, with no sex predilection noted. Seven cases involved the mandible and one involved the maxilla, with the posterior regions favored. The most common sign was swelling, although pain, rapid growth, trismus, and dysphonia also occurred. Lesions characteristically were evident as ill-defined destructive radiolucencies, with occasional radiopacities noted. Histologic features generally resembled those of conventional ameloblastoma but with cytologic features of epithelial malignant disease. The clinical course was uniformly aggressive with extensive local destruction and spread, frequent recurrences, and one case of neck node metastasis. The nomenclature and classification of odontogenic carcinomas are discussed, as well as entities that should be included in the differential diagnosis. Further reporting of ameloblastic carcinoma is encouraged.

Adolescent↗

A histopathologic study of 116 ameloblastomas with special reference to the desmoplastic variant.

The histopathologic features of 116 ameloblastomas were reviewed with special reference to the incidence of the recently described desmoplastic variant. The series included 110 intraosseous lesions and 6 extraosseous ameloblastomas. Ninety-six (88%) of the intraosseous tumors were found in the mandible, and 61% involved the molar-ascending ramus area. Fourteen examples of the desmoplastic variant were identified, representing 13% of the intraosseous tumors. The lesions showed distinctive histologic features characterized by an extensive collagenized stroma containing small islands of tumor epithelium with scant tendency to form cystic structures. This histologic variant has a marked tendency to involve the anterior portions of the jaws with 7 of the 14 examples being located in the maxilla. These desmoplastic lesions also showed unusual radiographic findings, often more suggestive of a benign fibro-osseous lesion than of ameloblastoma.

Adolescent↗

Familial gigantiform cementoma: classification and presentation of a large pedigree.

Very few cases of gigantiform cementoma have been reported, and those associated with a positive family history are especially rare. Confusion exists about the relationship of gigantiform cementoma to florid osseous dysplasia, cementifying fibroma, and diffuse chronic sclerosing osteomyelitis. It has been unclear whether gigantiform cementoma should be accorded the status of a separate entity. In this article, we report our findings on a family that, over five generations, has exhibited clinical, radiographic, and/or histologic findings consistent with the designation familial gigantiform cementoma. This pedigree consists of 55 members. Significant heterogeneity in expression of this trait was noted. The pattern of occurrence of the trait is consistent with an autosomal dominant mode of inheritance with variable expressivity of the phenotype. We suggest that familial gigantiform cementoma should be recognized as a separate entity.

Cementoma↗

Multicentric familial squamous odontogenic tumor.

Three cases of multicentric squamous odontogenic tumor, one of which was previously reported, are presented in three black siblings. A review of the literature indicates that 5 of the 26 previously recorded cases arose in multiple sites, but no evidence of familial history has been suggested. Although squamous odontogenic tumor has been described as a relatively rare, benign odontogenic neoplasm, the multicentric familial nature of our present cases suggests this represents either a reactive hyperplasia or hamartoma of residual odontogenic epithelium within the periodontal tissues. Management of previous cases has varied from simple curettage to maxillectomy with only one case of recurrence.

Adult↗

Oral neurofibrosarcoma associated with neurofibromatosis type I.

One of the most feared complications of neurofibromatosis type I (NF-I) is development of cancer, which is estimated to occur in about 5% of cases. The most common associated malignancy is the neurofibrosarcoma (NFS). HOwever, oral NFS in association with NF-I has rarely been reported. We report two cases of oral NFS arising in patients with NF-I. Both patients died of their tumors. Oral NFS arising in association with NF-I appears to have an extremely poor prognosis, as do these tumors at other sites of the body.

Adult↗

Computed tomography of the jaws in familial adenomatosis coli.

Familial adenomatosis coli is known to be associated with tooth impactions, odontomas, and osteomas. Three cases were examined by panoramic radiography and computed tomography. Computed tomography was especially useful for detecting osteomas in the maxilla and also revealed a wavy cortical thickening in the mandible in one instance. The latter finding has been reported for other bones in the body but not previously for the mandible.

Child↗

Appearance of drug-resistant tumor cells in a maxillary cancer patient undergoing chemotherapy and chemoradiotherapy.

In this study, we examine drug resistance and sensitivity in tumor cell lines cultured from a patient undergoing prolonged chemotherapy. Four serially established primary tumor cell lines (KKM-A, -B, -C, and -D) were cultured from a case of maxillary sinus carcinoma: KKM-A from the nontreated primary tumor; KKM-B from the residual tumor after chemotherapy with cisplatin (CDDP) and 5-fluorouracil (5-FU); KKM-C from the local tumor that recurred following complete remission with chemoradiotherapy (combination of 5-FU, vitamin A, and radiation); and KKM-D from a metastatic lymph node tumor after further chemotherapy with CDDP and peplomycin. The sensitivity of the four cell lines to CDDP, 5-FU bleomycin (BLM), carboquone (CQ), and colchicine was assessed according to the reduction in colony formation. The KKM-A cell line was very sensitive to CDDP, BLM, CQ, and colchicine, whereas the posttreatment cell lines were resistant to these four drugs. The resistant cell lines were probably selected as a result of chemotherapy and radiotherapy. The nontreated KKM-A cell line was naturally somewhat resistant to 5-FU. The 5-FU-resistance of the KKM-B, -C, and -D cell lines was reduced in that order, corresponding to their relative degree of treatment. The KKM-D cell line from the metastatic lymph node showed a different sensitivity from the KKM-B and -C cell lines from the primary site, suggesting a heterogeneity in drug sensitivity between primary and metastatic tumor cells.

Adult↗

A survey of tumours of the jawbones in Hong Kong Chinese: 1963-1982.

A total of 114 tumours of the jawbones was confirmed in a survey of 204,583 surgical specimens in Chinese in the University Department of Pathology, Hong Kong from 1963-1982. Odontogenic tumours totalled 82 of which 62 per cent were ameloblastomas. Thus, odontogenic tumours, in particular ameloblastomas, are relatively common in Chinese. In the 51 cases of ameloblastoma, the mean age at presentation in females was significantly younger than in males. Pathological analysis of the ameloblastomas showed the following: (1) connection of tumour epithelium with oral mucosal epithelium suggests a better prognosis; (2) basaloid pattern in the tumour may prognostically indicate a more aggressive biological behaviour; (3) neoplastic infiltration of the grossly normal bone surrounding the tumour mass was frequent; (4) ameloblastomas not uncommonly contained cysts lined by innocuous-looking epithelium; (5) a significant proportion of ameloblastomas appeared grossly as thin-walled unilocular cysts. The implications of these findings in the diagnosis and treatment of ameloblastoma are emphasized.

Adolescent↗

Florid cemento-osseous dysplasia (gigantiform cementoma) in a Caucasian woman.

The present study reports a case of an uncomplicated florid cemento-osseous dysplasia in a 39-year-old Caucasian woman. The extensive dysplastic lesions affected all jaw quadrants and both radio-opaque and lytic lesions were demonstrated radiographically. Histopathological studies revealed calcified, partly acellular cementum-like tissue. All clinical, radiographic, biochemical and histological features were suggestive of the diagnosis of florid cemento-osseous dysplasia.

Adult↗

A central venous catheter complicating head and neck surgery.

A case is reported of a central venous catheter that was placed inadvertently into the internal jugular vein during the anaesthetic preparation for surgery. The catheter tip was sectioned during surgery and was feared to have embolised into the central circulation but it was subsequently identified in the pathological specimen. A summary of the complications associated with central venous catheterisation is reported and a simple protocol is proposed to avoid a similar complication.

Aged↗

Gigantiform cementoma affecting a Caucasian family.

A Caucasian family is presented in which two out of three children showed lesions resembling gigantiform cementoma clinically, radiologically and histopathologically. Both affected children had swellings of the alveolar crest, caused by large, lobular, radio-opaque masses in all four quadrants of the jaws. Histologically, cementum-like masses were surrounded by inflamed fibrous connective tissue. There was evidence indicating that the late father had similar lesions which became malignant following radiation therapy.

Adolescent↗