Case Report

Use of Zoledronic Acid in Paediatric Craniofacial Fibrous Dysplasia

Table 2

Case reports about use of bisphosphonates in paediatric fibrous dysplasia.

SourceSubjectsDiseaseSigns and symptomsTreatmentEfficacy of treatment

Hart et al. [4] Male with cherubism diagnosed when he was 6 years oldCherubismPain localized in mandibleCalcitonin and bisphosphonatesPain reduction

Zumkeller et al. [5]Male child 8 years oldMcCune-Albright syndrome and pituitary adenomaPolyostotic dysplasia, Cafè-au-lait spots, and precocious pubertyPamidronate and sandostatinIncreased mobility

Rastogi et al. [6]15-year-old femaleMcCune-Albright syndromeRecurrent fractures of femur by minor trauma and chronic bone pain (right thigh)Pamidronate i.v. (1 mg/kg/day for 3 days once in 3 months for a period of 1 year), 25-hydroxy-vitamin D, and calcium Pain reduction and no fractures developed over three years of follow-up

Kochar and Kulkarni [7]2- and 8-year-old femaleMcCune-Albright syndromeGeneralized bone pain, recurrent fractures by minor trauma, fatigue, and bone deformitiesPamidronate i.v.Remarkable clinical improvement

Atsali et al. [8]14-year-old femaleMcCune-Albright syndromeCystic lesions of the right humerus incidentally discovered, bone pain, and a history of fracture of the distal humerus by minor traumaAt first, pamidronate i.v. and then i.v. zoledronic acid (5 mg annually)During treatment, pain reduction, no fractures, and increased BMD after 4 years of therapy

Bieniasz et al. [9]12-year-old femaleCraniofacial FDOrbital cavity and sinuses lesionsPamidronate Clinical improvement

Mäkitie et al. [22]12-year-old femaleCraniofacial FDMandibular lesions, swelling, disfigurement, and pain of the right side of the jawPamidronate i.v. (1 mg/kg on 3 consecutive days at 4-month intervals), in the following year, the therapy was switched to zoledronic acid i.v. (0.05 mg/kg once every 6 months)Pain and disfigurement reduction

Khadilkar et al. [11]3-year-old malePolyostotic FDRecurrent fractures of the left femur, pain, and asymmetryOral alendronatePain reduction and no fractures

Chattopadhyay et al. [12]17-year-old femalePolyostotic FD and hypophosphatemiaSwelling of the left forearm, progressive bowing deformity of the lower limb, widening of the wrist, decrease in linear growth with sever short stature, rickets, and osteomalaciaOral alendronate, phosphate, and 25-hydroxy-vitamin DClinical improvement

Jayaraman et al. [13]18-year-old femalePolyostotic FDPain, swelling and fracture in the right middle finger after minor trauma, and expansive lesions in the right hand and in the superior pubic ramusOral alendronate (70 mg weekly), calcium carbonate 1000 mg daily, and 25-hydroxy-vitamin D 400 IU dailyRadiographic improvement of bone lesions