Τίτλος:
Profound hypocalcemia following effective response to zoledronic acid treatment in a patient with juvenile Paget's disease
Γλώσσες Τεκμηρίου:
Αγγλικά
Περίληψη:
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. © 2010 The Japanese Society for Bone and Mineral Research and Springer.
Συγγραφείς:
Polyzos, S.A.
Anastasilakis, A.D.
Litsas, I.
Efstathiadou, Z.
Kita, M.
Arsos, G.
Moralidis, E.
Papatheodorou, A.
Terpos, E.
Περιοδικό:
Journal of Bone and Mineral Metabolism
Λέξεις-κλειδιά:
alfacalcidol; alkaline phosphatase; calcitonin; calcium; calcium carbonate; colecalciferol; etidronic acid; gluconate calcium; risedronic acid; zoledronic acid, adolescent disease; adult; alkaline phosphatase blood level; article; bone bowing; bone mineral; bone pain; bone scintiscanning; bone turnover; case report; clinical feature; disease severity; dose response; drug effect; drug efficacy; drug megadose; female; hearing impairment; human; hypertelorism; hypocalcemia; kyphoscoliosis; macrocephaly; Paget bone disease; priority journal; secondary hyperparathyroidism; treatment duration; walking difficulty, Adult; Bone and Bones; Bone Density Conservation Agents; Diphosphonates; Drug Monitoring; Female; Humans; Hyperparathyroidism, Secondary; Hypocalcemia; Imidazoles; Osteitis Deformans
DOI:
10.1007/s00774-010-0198-8