Profound Hypocalcemia Following Effective Response to Zoledronic Acid Treatment in a Patient with Juvenile Paget's Disease

Overview

Journal J Bone Miner Metab

Specialty Endocrinology

Date 2010 Jun 10

PMID 20533067

Citations 14

Authors

Stergios A Polyzos

Athanasios D Anastasilakis

Ioannis Litsas

Zoe Efstathiadou

Marina Kita

Georgios Arsos

Efstratios Moralidis

Athanasios Papatheodorou

Evangelos Terpos

Affiliations

Soon will be listed here.

Abstract

Juvenile Paget's disease (JPD) is a rare, autosomal recessive osteopathy. Although it has phenotypic overlap with Paget's disease of bone (PDB), it is probably a distinct entity. Because of its rarity, optimal disease management has not yet been established by randomized controlled trials. However, clinical, biochemical, and radiographic improvement has been reported after treatment with antiresorptive agents, including calcitonin and bisphosphonates (BPs). Compared with other BPs, zoledronic acid (ZOL) has a higher affinity to bone mineral and is a stronger inhibitor of the enzyme farnesyl pyrophosphate synthase (the main target of nitrogen-containing BPs), properties that explain the prolonged effect of ZOL on bone turnover and render it a therapeutic option for JPD, similar to PDB. We describe hereby, for the first time in the literature, the case of a patient with JPD who developed severe hypocalcemia and secondary hyperparathyroidism following effective treatment with ZOL.

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