Giant cell tumor of bone with secondary aneurysmal bone cyst-like change producing β-human chorionic gonadotropin

Abstract

Giant cell tumor of bone is a benign, locally aggressive neoplasm that is composed of sheets of neoplastic mononuclear cells interspersed amongst non-neoplastic, uniformly distributed, osteoclast-like giant cells. They represent approximately 4-5% of primary bone tumors. Rarely, bone tumors have been noted to produce human chorionic gonadotropin, a finding most often reported in osteosarcoma. We present the case of a young woman who presented with a low-level human chorionic gonadotropin level which, after resection of her recurrent giant cell tumor of bone with secondary aneurysmal bone cyst-like change, became undetectable in her blood. Furthermore, cells within the aneurysmal bone cyst component were immunohistochemically positive for β-human chorionic gonadotropin. This is the first report of such a finding in the literature.

Beta-Human Chorionic Gonadotropin Expression in Recurrent and Metastatic Giant Cell Tumors of Bone

“…6 Although beta-hCG immunoreactivity and secretion have not been studied in a larger series of bone tumors, case reports have described markedly higher levels of serum beta-hCG elevation in osteosarcomas than in benign giant cell tumors, which raises the question of whether malignant bone neoplasms have a greater potential toward paraneoplastic hormone production than benign tumors and whether immunohistochemical expression could potentially be used as an marker of malignant phenotype. 9,10,21 The discrepancy between immunolabeling for beta-hCG and its clinical function (serum hormone elevation) seen in several of our patients can be explained in several ways. It has been proposed that neoplastic cells may secrete hormone at a rate that precludes hormone accumulation to a detectable level by immunohistochemical methods.…”

“…6 Although beta-hCG immunoreactivity and secretion have not been studied in a larger series of bone tumors, case reports have described markedly higher levels of serum beta-hCG elevation in osteosarcomas than in benign giant cell tumors, which raises the question of whether malignant bone neoplasms have a greater potential toward paraneoplastic hormone production than benign tumors and whether immunohistochemical expression could potentially be used as an marker of malignant phenotype. 9,10,21…”

“…[3][4][5][6][7][8] Increased serum levels of beta-hCG have been reported in a number of nontrophoblastic neoplasms, with several case reports of elevated serum beta-hCG in osteosarcoma and 1 case report of slightly increased serum levels in a primary giant cell tumor of bone. 3,4,7,9,10 At our institution, we have observed multiple cases of markedly elevated urine and serum beta-hCG levels in patients with giant cell tumors, which has led to diagnostic difficulty and in one instance, concern for metastatic choriocarcinoma. The objective of this study was to report a case of beta-hCG-secreting giant cell tumor of bone and to study the expression and significance of beta-hCG in an institutional cohort of giant cell tumors of bone.…”

Human Chorionic Gonadotropin Regulates the Smad Signaling Pathway by Antagonizing TGF-β in Giant Cell Tumor of Bone

“…Fitzhugh et al . found that the development of GCTB is usually accompanied by the production of β-HCG [ 14 ]. Lawless et al .…”