A metastasising ameloblastoma associated with renal calculi and hypercalcaemia

Abstract

A young male had an assumed dentigerous cyst marsupialized and later a recurrent ameloblastoma resected. Eleven years later he was admitted with renal stones and hypercalcemia and metastases of the ameloblastoma in the left lung were discovered. Death occurred as a result'of spinal and hepatic spread of his tumor and thrombosis of the renal veins. Renal calcification was demonstrated. The possible causes of the hypercalcaemia which was not associated with a raised serum parathormone or affected by parathyroidectomy is discussed.Cancer 36:2277-2285, 1975. ROM TIME TO TIME, CASES ARE REPORTEDThe upper molars bit into the swelling and at this site F of ameloblastomas which have spread ei-the mucosa was inflamed. There was no limitation of ther to the lungs, or to the regional lymph nodes. opening. Such cases are uncommon and frequently the His general health was good, there was no past accounts contain of uncertainty. ~h~~ history of serious illness, and he was a non-smoker. Radiographs revealed a circumscribed bone cavity is reported because metastatic spread of the neo-which involved the right mandible from immediately plasm was accompanied by hypercalcaemia, the distal to the first molar back to the base of the coroproduction of renal calculi a n d metastatic calcifi-noid process and the neck of the condyle (~i~. 1~) cation; a circumstance which, a s far a s we are Only the crown of the third molar, together with aware, has not previously been recorded. about 2 mm of thin, cervical dentine was formed and the tooth was displaced down to the angle of the mandible. The periodontal membrane of was thickened on the distal aspect of the mesial root. Both Of the ramus exhibited quite marked expansion. The appearance was as a dentigerous cyst.At operation a considerable quantity of clear serous fluid was released and a piece of the cyst wall distal to removed, 9/ was extracted and the cavity packed.

Expression of parathyroid hormone‐related protein (PTHrP), osteoclast differentiation factor (ODF)/receptor activator of nuclear factor‐κB ligand (RANKL) and osteoclastogenesis inhibitory factor (OCIF)/osteoprotegerin (OPG) in ameloblastomas

“…Humoral hypercalcemia of malignancy (HHM) is caused most commonly by the humoral action of PTHrP, which is released by tumor cells (9, 10, 12). Malignant ameloblastoma is occasionally accompanied by hypercalcemia with or without metastasis, and several cases have been ascribed to tumor‐derived PTHrP (16, 17, 48, 49). Tumor‐derived PTHrP is also known to participate in bone metastasis and to establish osteolytic bone lesions in metastatic sites, even in the absence of hypercalcemia or increased plasma PTHrP concentration (13–15).…”

“…Production of PTHrP by neoplastic cells has been recognized in malignant tumors commonly associated with HHM, such as adult T cell lymphoma/leukemia (ATLL), pulmonary squamous cell carcinoma, and renal cell carcinoma (9, 10, 12) as well as in malignancies frequently associated with bone metastasis, such as breast and prostate carcinomas (13–15). Several cases of malignant ameloblastoma with hypercalcemia have been attributed to PTHrP (16, 17). Osteoclast differentiation factor (ODF)/receptor activator of nuclear factor‐κB ligand (RANKL) is a cell membrane‐bound and soluble molecule of the tumor necrosis factor (TNF) superfamily (6, 18).…”

Pulmonary metastasis of malignant ameloblastoma: case report and review

“…Death is not so uncommon in cases of malignant ameloblastoma. Seward et al [12] reported a case of death in a patient with ameloblastoma metastasized to the lung 11 years after resection of the primary tumor on the face. According to the authors, death occurred as a result of the spinal and hepatic spread of the tumor, and thrombosis of the renal veins.…”

Ameloblastoma of the maxilla

“…Recently, Seward et al (1975) have reported a case of ameloblastoma associated with renal calculi and hypercalcemia. In our cases no evidence of real aggressive growth or distant metastasis was found, nor was there any sign of renal involvement and hypercalcemia.…”