Use of Zoledronic Acid in Paediatric Craniofacial Fibrous Dysplasia

Pede, Chiara Di; Congedi, Sabrina; Rossin, Sara; Divisic, Antuan; Gregorio, Alesandra De; Agosto, Caterina; Catalano, Igor; Mazza, Alessandro; Sartori, Leonardo; Masiero, Stefano; Benini, Franca

doi:10.1155/2016/2329483

Cited by 10 publications

(10 citation statements)

References 17 publications

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“…Nevertheless, its efficacy and safety have never been compared to other bisphosphonates in children with FD. One to four cycles of intravenous zoledronic acid in pediatric FD have shown to relieve pain, decreased the markers of bone turnover, and improved radiologic findings (6,(11)(12)(13)(14)(15)(16)(17)(18)(19). In agreement with these studies, our observation revealed that there were significant improvements in pain and radiological findings.…”

Section: Discussionsupporting

confidence: 89%

“…Because of their antiresorptive properties, bisphosphonates are the medical treatment of choice in symptomatic cases similar to conditions associated with bone resorption (5,7,8). Though pamidronate and alendronate are commonly used in various studies for the treatment of FD (9-11), the literature on zoledronic acid use in FD is limited (11)(12)(13)(14)(15). Compared to pamidronate, zoledronic acid requires fewer doses leading to better compliance with minimal side effects (6,16).…”

Section: Introductionmentioning

confidence: 99%

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Response to Zoledronic Acid Infusion in Children With Fibrous Dysplasia

et al. 2020

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Objective: This retrospective study evaluated the outcome and safety of long-term treatment with zoledronic acid, in both polyostotic and mono-ostotic fibrous dysplasia (FD) of children.Methods: The case records of children and adolescents with symptomatic FD who received zoledronic acid (0.1 mg/kg IV infusion over 1 h) and have completed at least 2 years follow-up were analyzed. The relevant details were recorded in a predesigned chart. Clinical assessment [pain assessment by visual analog scale (VAS) and incidence of new fracture], radiological changes (cortical thickening, ossification, and decrease in the diameter of the osteolytic lesions) and biochemical parameters [alkaline phosphatase (ALP)] were used to evaluate the improvement.Results: The mean age of presentation was 9.1 years, with four males and six females. All patients had symptomatic FD in the lower limb with complaints of pain, tenderness, swelling, or deformity. Four children had associated pathological fracture. The radiological evaluation with bone scan revealed polyostotic involvement in eight patients and mono-ostotic involvement in two patients. Three patients had associated systemic features like café-au-lait spots or precocious puberty. The fracture united within 3 months and the radiological improvement was evident in the form of filling of the osteolytic defect. The pain score in six patients showed significant improvement (VAS < 3). The ALP level decreased to 544.12 ± 47.35 IU/L from an initial value of 895.75 ± 79.64 IU/L (p = 0.04) at 12 months. One patient had symptomatic hypocalcaemia after zoledronic acid infusion.Conclusion: The clinical and radiological response of zoledronic acid treatment in FD of children is promising. Further randomized control trials with a larger sample size are required to establish this drug as a first-line medical treatment in FD.

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Section: Discussionsupporting

confidence: 89%

Section: Introductionmentioning

confidence: 99%

Response to Zoledronic Acid Infusion in Children With Fibrous Dysplasia

et al. 2020

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“…Since then, a number of publications reported variable beneficial effects of different bisphosphonates on pain, bone turnover markers and radiological features of FD/MAS lesions largely in adults and children with polyostotic FD/MAS. These publications include 18 case reports , 20 observational case series and a single randomized controlled study (RCT) using oral alendronate .…”

Section: Antiresorptivesmentioning

confidence: 99%

“…Case reports were predominantly on women (11/18), seven on children and six reporting the effects of treatment with zoledronate and pamidronate in patients with isolated craniofacial FD (CFD) . Positive outcome of treatment in the form of decreased pain was reported in all patients, associated by a concomitant normalization in bone turnover markers in six of the 17 patients in whom these markers were measured before and after treatment . A total of 462 patients were included in the 20 published case series.…”

Section: Antiresorptivesmentioning

confidence: 99%

Clinical and translational pharmacological aspects of the management of fibrous dysplasia of bone

Rotman

Hamdy

Appelman‐Dijkstra

2018

Brit J Clinical Pharma

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Fibrous dysplasia (FD) is a genetic, noninheritable rare bone disease caused by a postzygotic activating mutation of the α subunit of the stimulatory G‐protein causing increased abnormal bone formation leading to pain, deformity and fractures. To date, no cure has been identified for FD/McCune–Albright syndrome (MAS) and treatment is symptomatic and aimed at decreasing pain and/or local bone turnover. Various drugs have been used to achieve clinical improvement in FD/MAS patients including bisphosphonates and denosumab, however further translational studies are also warranted to address unresolved pathophysiological issues and explore novel pharmacological targets for the management of FD/MAS. In this article, we review literature on the medical treatment of FD/MAS, discuss the unresolved pathophysiological issues and explore novel pharmacological targets for the management of FD/MAS.

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“…Treatment with pamidronate in six pediatric patients with FD showed improvement in bone pain and radiographic features of FD (22). ZA treatment at the dose of 0.05 mg/kg resulted in resolution of pain and improvement in bone lesions in a 12-year-old boy with craniofacial FD (23). underlying chronic illnesses that adversely affect bone mass accrual and/or maintenance ( Table 1).…”

Section: Primary Osteoporosismentioning

confidence: 99%

Zoledronic acid in pediatric metabolic bone disorders

Bowden¹,

Mahan²

2017

Transl. Pediatr

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Zoledronic acid (ZA), a highly potent intravenous bisphosphonate (BP), has been increasingly used in children with primary and secondary osteoporosis due to its convenience of shorter infusion time and less frequent dosing compared to pamidronate. Many studies have also demonstrated beneficial effects of ZA in other conditions such as hypercalcemia of malignancy, fibrous dysplasia (FD), chemotherapy-related osteonecrosis (ON) and metastatic bone disease. This review summarizes pharmacologic properties, mechanism of action, dosing regimen, and therapeutic outcomes of ZA in a variety of metabolic bone disorders in children. Several potential novel uses of ZA are also discussed. Safety concerns and adverse effects are also highlighted.

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Use of Zoledronic Acid in Paediatric Craniofacial Fibrous Dysplasia

Cited by 10 publications

References 17 publications

Response to Zoledronic Acid Infusion in Children With Fibrous Dysplasia

Response to Zoledronic Acid Infusion in Children With Fibrous Dysplasia

Clinical and translational pharmacological aspects of the management of fibrous dysplasia of bone

Zoledronic acid in pediatric metabolic bone disorders

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