Abstract:
Context: Parathyroidectomy is the only effective therapy for osteitis fibrosa cystica in hyperparathyroidism. Objective: The objective of this study was to describe the changes of skeletal and nonskeletal manifestations in a patient with hyperparathyroidism and renal failure after oral vitamin D therapy. Design: This was a descriptive case report. Setting: The patient was followed up in a referral center. Patient: A 55-yr-old male patient with moderate renal failure was referred for expansile lytic lesions affecting several ribs and the spinous process of T12. His creatinine was 1.8 mg-dl; calcium, 8.9 mg-dl; PTH, 666 pg-ml; and 1,25 dihydroxy-vitamin D, 27 pg-ml. Bone mineral density (BMD) Z-scores by dual-energy x-ray absorptiometry were -4.1 at the spine, -1.7 at the hip, and -4.3 at the forearm. Main Outcome Measures: The main outcome measures were the skeletal manifestations of hyperparathyroidism. Results: At 10 months of therapy, calcium level was 10 mg-d, PTH level declined to 71 pg-ml, and BMD increased by 12percent at the spine and 18percent at the hip. Computerized tomography (CT) cuts revealed marked regression in the lytic lesions. At 2 yr, BMD increased by an additional 6percent at the spine, and there were no further changes in the lytic lesions by CT. The vitamin D receptor genotype using the restriction enzymes Bsm1, Taq1, and Apa1 was Bb, tt, and AA. Conclusions: We showed regression of severe skeletal abnormalities of hyperparathyroidism documented by serial CT images in response to oral vitamin D therapy. It is possible that the vitamin D receptor genotype of the patient modulated this response. Copyright © 2006 by The Endocrine Society.