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PubMed1985

Advanced osteitis fibrosa cystica in the absence of phalangeal subperiosteal resorption. A case report and review of the literature.

A case of primary hyperparathyroidism with advanced osteitis fibrosa cystica but without any subperiosteal phalangeal bone resorption is described. A review of this unusual radiological feature is presented. High-detail magnification radiography (microradiography) is advocated for the early diagnosis of bony defects in hyperparathyroidism.

Bone Resorption↗
PubMed1993

Pseudohypoparathyroidism with osteitis fibrosa cystica: direct demonstration of skeletal responsiveness to parathyroid hormone in cells cultured from bone.

A young girl had tibial osteotomies at age 14 for genu valgum and then had recurrent tibial cysts over a number of years. Hypocalcemia and hyperphosphatemia were first noted at age 21. The diagnosis of pseudohypoparathyroidism was made at age 28, when elevated plasma PTH was detected. Clinical and biochemical features, including a PTH response test and assay of RBC Gs, established the diagnosis of pseudohypoparathyroidism type 1b. Failure to suppress plasma PTH with vitamin D therapy led to an exacerbation of her cystic bone disease; there were widespread lytic lesions radiologically, most of which took up [99mTc]diphosphonate on bone scan. Microradioscopy revealed evidence of resorption of phalangeal tufts. Bone biopsy showed osteitis fibrosa cystica. During an orthopedic procedure, trabecular bone fragments were taken from her right humerus, and bone-derived cells cultured using an explant technique. The cultured cells were osteoblast-like in morphology, fully responsive to PTH, cholera toxin, forskolin, and PGE1 in vitro, and had an alkaline phosphatase and osteocalcin response to 1,25-dihydroxyvitamin D3 [1,25-(OH)2D3]. Following this examination of skeletal responsiveness, attempts were made to suppress the elevated plasma PTH levels and symptomatic bone disease by optimizing therapy with oral 1,25-(OH)2D3. When bone pain associated with the cystic bone disease failed to resolve, the patient underwent total parathyroidectomy, following which the bone pain gradually resolved. This is the first direct demonstration of PTH responsiveness in cultured bone cells in the syndrome of pseudohypoparathyroidism with osteitis fibrosa cystica.

Adenylyl Cyclases↗
PubMed1993

Osteitis fibrosa cystica simulating metastatic tumor. An almost-forgotten relationship.

The diagnosis of primary hyperparathyroidism most often results from the incidental finding of hypercalcemia. In two recent cases of osteitis fibrosa cystica (OFC), however, patients without adequate access to health care served as graphic reminders that the clinical spectrum of the disease includes bone disease, and that OFC can be the presenting manifestation of long-standing primary hyperparathyroidism. Both patients complained of bone pain and had widespread osteolytic bone lesions in addition to hypercalcemia on a multitest biochemical panel. The presumptive diagnosis of malignancy with bone involvement (metastatic cancer or multiple myeloma) led to random bone marrow trephine biopsies. Examination of the bone marrow biopsy material revealed the characteristic pathology of OFC, leading to appropriate diagnosis and surgical management of large parathyroid adenomas in both patients.

Adult↗
PubMed1980

Vitamin D metabolism during recovery from severe osteitis fibrosa cystica of primary hyperparathyroidism.

Serum concentrations of 1,25-dihydroxyvitamin D [1,25-(OH)2D] and immunoreactive parathyroid hormone were measured before and for 7 months after the removal of a 15-g parathyroid adenoma from a 44-yr-old woman with primary hyperparathyroidism and severe osteitis fibrosa cystica. Despite the fall in parathyroid hormone levels from preoperative levels of 20 to 1--2 ng/ml after surgery (normal, up to 1.2 ng/ml), serum 1,25-(OH)2D concentrations remained markedly elevated (156 pg/ml) preoperatively; 124 pg/ml 17 weeks postoperative), approaching the normal range (18--56 pg/ml) only after 5 months (65 pg/ml). Hypocalcemia and hypophosphatemia persisted despite oral 1,25-(OH)2D3 (1 and 2 micrograms/day) and large doses of (oral and iv) calcium gluconate (up to 30 g/day). Healing of the skeletal lesions, reversal of the myopathy, and return of 1,25-(OH)2D circulating levels to normal corresponded to the time when serum phosphate became normal. The stimulus for the persistently elevated serum 1,25-(OH)2D levels may have been hypocalcemia per se, low serum phosphate, or an unidentified signal that paralleled serum phosphate, as serum PTH levels remained in the upper normal range throughout the recovery period.

Adenoma↗
PubMed2006

Oxyphilic cell adenoma of parathyroid resulting in primary hyperparathyroidism and osteitis fibrosa cystica--a case report.

Adenomas of the parathyroid gland, the majority of which are of the solitary chief cell type, are the most frequent cause of primary hyperparathyroidism (pHPT). Parathyroid adenomas composed predominantly or exclusively of oxyphil cells are rare and most oxyphil cell adenomas of this organ remain clinically silent. We present here a case of hyperfunctioning oxyphil cell adenoma of the parathyroid gland resulting in pHPT, osteitis fibrosa cystica and simultaneous bilateral fractures upper shafts of femora.

Adenoma, Oxyphilic↗
PubMed2004

Pathologic fracture of the distal femur in osteitis fibrosa cystica simulating metastatic disease.

A 32-year-old woman sustained a minor sliding accident with moderate to severe pain about the left distal thigh and hip and the right shoulder. Radiographs showed marked osteopenia, multiple osteolytic bone lesions, and a pathologic fracture of the left distal femur. Surgical intervention was conducted for the pathologic fracture, and the pathological findings confirmed the diagnosis of primary hyperparathyroidism with osteitis fibrosa cystica. We report this rare case here because it may be mistaken for neoplastic metastatic disease on radiographs and bone scan. The pathologic fracture usually heals and the bone mineral density improves significantly within 6 months after parathyroidectomy.

Adult↗
PubMed1999

Osteitis fibrosa cystica generalizata with adult T-cell leukaemia: a case report.

We report on a 62-year-old female initially suffering from extreme pain in both her lower extremities. Plain radiographs revealed multiple osteolytic lesions. Laboratory analyses indicated high levels of serum calcium and parathyroid hormone related protein (PTHrP) and detected HTLV-1 antibody. Histological examination of biopsied specimens which were obtained from osteolytic lesions indicated osteitis fibrosa cystica (OFC) without tumour cells. After the initial biopsy, a subcutaneous mass developed in the left thigh. Microsections of the biopsied mass disclosed adult T-cell leukaemia (ATL) neoplastic cells. Immunohistochemical findings revealed that PTHrP secreted from ATL neoplastic cells induced the OFC.

Female↗
PubMedDate not supplied

Osteitis fibrosa cystica (brown tumor) in a patient with renal transplantation. Report of a case with aspiration cytodiagnosis.

The cytologic findings from fine needle aspiration of a left shoulder mass are described for a woman who had undergone renal transplantation and on whom an orthopedic consultation suggested the clinical possibility of a soft tissue sarcoma. The aspirate samples from multiple sites in the mass showed several multinucleate, osteoclast-type giant cells, spindly or fibrillary cells with ovoid nuclei and a significant amount of granular calcium. The needle aspiration cytodiagnosis was of critical importance since it saved the patient from invasive surgical intervention. This case emphasizes the role of needle aspiration cytology in providing a rapid cytodiagnosis of osteitis fibrosa cystica with a minimally invasive method.

Adult↗