HRQoL Evaluation of Pediatric Osteogenesis Imperfecta with Zoledronic Acid Therapy
Downloads
Background: Zoledronic acid as bisphosphonates could increase bone mineral density (BMD), which in osteogenesis imperfecta will reduce clinical manifestations. Pediatric patients with osteogenesis imperfecta given zoledronic acid therapy should improve their quality of life.
Methods: A retrospective analytic study was conducted on 16 pediatric osteogenesis imperfecta patients who had received intravenous zoledronic acid. Interview using PedsQL 4.0 regarding the condition before and after therapy assessed the quality of life. A paired t-test was used to assess the contrast in each aspect of PedsQL 4.0. Results were expressed as an adjusted odds ratio with a 95% confidence interval. P-value <0.05 is considered statistically significant.
Results: A significant increase was found in the child report of social performance (+12.083, p=0.023), and proxy report of physical performance, social performance, and total score of the proxy report (+14.844, p=0.006; +10.625, p=0.010; +10.364, p=0.006). An insignificant increase was found in the child report of physical performance, school performance, and total score of the child report (+8.833, p=0.148; +5.000, p=0.359; +7.065, p=0.115), and proxy report of emotional performance and school performance (+2.500, p=0.669; +6.250, p=0.167). An insignificant decrease was found in child reports of emotional performance (-2.500, p=0.669).
Conclusion: After receiving bisphosphonate therapy, pediatric patients with osteogenesis imperfecta experienced an increased quality of life in most aspects within every perception. Physical and social aspects experienced the greatest improvement. Emotional is the aspect that experienced the lowest increase in the child's perception and decreased parent perception of the value of quality of life after bisphosphonate therapy.
Marini JC, Forlino A, Bächinger HP, Bishop NJ, Byers PH, Paepe AD, et al. Osteogenesis imperfecta. Nat Rev Dis Prim 2017;3:17052.
Folkestad L, Hald JD, Hansen S, Gram J, Langdahl B, Abrahamsen B, et al. Bone geometry, density, and microarchitecture in the distal radius and tibia in adults with osteogenesis imperfecta type I assessed by high-resolution pQCT. J. Bone Miner Res 2012;27(6):1405–12.
Imbert L, Aurégan JC, Pernelle K, Hoc T. Mechanical and mineral properties of osteogenesis imperfecta human bones at the tissue level. Bone. 2014;65:18–24.
Prameswari ZT, Sjafei A, Winoto ER. Kelainan Gigi Pada Pasien Osteogenesis Imperfecta [Dental anomaly in osteogenesis imperfecta patients]. Orthod Dental J 2011;2(1):16-25.
Tournis S and Dede AD. Osteogenesis imperfecta – A clinical update. Metabolism 2018;80:27–37.
Kuurila K, Johansson R, Kaitila I, Grénman R. Hearing loss in Finnish adults with osteogenesis imperfecta: A nationwide survey. Ann Otol Rhinol Laryngol 2002;111(10):939–46.
Engelbert RH, Uiterwaal CS, Gerver WJ, Van Der Net JJ, Pruijs HE, Helders PJ. Osteogenesis imperfecta in childhood: Impairment and disability. A prospective study with 4-year follow-up. Arch Phys Med Rehabil 2004;85(5):772–8.
Widmann RF, Laplaza FJ, Bitan FD, Brooks CE, Root L. Quality of life in osteogenesis imperfecta. Int Orthop 2002;26(1):3–6.
Dahan-Oliel N, Oliel S, Tsimicalis A, Montpetit K, Rauch F, Dogba MJ. Quality of life in osteogenesis imperfecta: A mixed-methods systematic review. Am J Med Genet A 2016;170A(1):62–76.
Riswanto BK, Bayusentono S. Quality of Life Evaluation in Children With Osteogenesis Imperfecta After Giving Bisphosphonate Infusion in Dr. Soetomo Hospital Surabaya. Med Health Sci J 2020;4(1):34–8.
Dwan K, Phillipi CA, Steiner RD, Basel D. Bisphosphonate therapy for osteogenesis imperfecta. Cochrane Database Syst Rev 2016;10(10):CD005088.
Kok DHJ, Sakkers RJB, Janse AJ, Pruijs HE, Verbout AJ, Castelein RM, et al. Quality of life in children with osteogenesis imperfecta treated with oral bisphosphonates (Olpadronate): A 2-year randomized placebo-controlled trial. Eur. J. Pediatr 2007;166(11):1155–61.
Löwing K, í…ström E, Oscarsson KA, Söderhäll S, Eliasson AC. Effect of intravenous pamidronate therapy on everyday activities in children with osteogenesis imperfecta. Acta. Paediatr. Int. J. Paediatr 2007;96(8):1180–3.
Seikaly MG, Kopanati S, Salhab N, Waber P, Patterson D, Browne R, et al. Impact of alendronate on quality of life in children with osteogenesis imperfecta. J. Pediatr. Orthop. 2005;25(6):786–91.
DeCarlo DK, Forte E, Gao L, McGwin G Jr, Owsley C. Reliability and validity of the PedsQL 4.0 Generic Core Scales in pediatric vision impairment. J Am Assoc Pediatr Ophthalmol Strabismus 2020;24(2):94.e1-7.
Fenig S, Levav I, Kohn R, Yelin N. Telephone vs face-to-face interviewing in a community psychiatric survey. Am J Public Health 1993;83(6):896-8.
Sitaresmi MN, Mostert S, Gundy CM, Sutaryo, Veerman AJ. Health-related quality of life assessment in Indonesian childhood acute lymphoblastic leukemia. Health Qual Life Outcomes 2008;6:96.
Song Y, Zhao D, Li L, Lv F, Wang O, Jiang Y et al. Health-related quality of life in children with osteogenesis imperfecta: a large-sample study. Osteoporos Int 2019;30(2):461–8.
Situmorang L, Suryawan A, Soemyarso NA. Comparison of Perceived Quality of Life Between Children with Lupus Nephritis and Their Parents. J Health Sci Med Res 2019; 37(4):281-7.
Cuevas-Olivo R, Alejo-Fuentes LJ, Alejo-Fuentes LF, Campos-Angulo G. El tratamiento con bifosfonatos mejora la calidad de vida en pacientes con diagnóstico de osteogénesis imperfecta [Treatment with bisphosphonates improves the quality of life in patients with diagnosis of osteogenesis imperfecta]. Acta Ortopédica Mex 2019;33(2):63–6.
Rochmah N, Mustikasari RI, Faizi M, Irwanto I. Determinant Factors of the Quality of Life in Children with Osteogenesis Imperfecta during the COVID-19 Pandemic. Open Access Maced J Med Sci. 2022;10(B):1137-42.
Rochmah N and Faizi M. Pediatric Quality of Life Inventory in Children with Osteogenesis Imperfect in Dr Soetomo Hospital Surabaya [abstract]. In Proceedings of the 57th Annual European Society of Paediatric Endocrinology (ESPE); 2018 Sep 27-29; Athens, Greece. Horm Res Paediatr 2018; 90 (Suppl. 1): 181.
Copyright (c) 2023 Journal Orthopaedi and Traumatology Surabaya
![Creative Commons License](http://i.creativecommons.org/l/by-nc-sa/4.0/88x31.png)
This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.
- The author acknowledges that the copyright of the article is transferred to the Journal of Orthopaedi and Traumatology Surabaya (JOINTS), whilst the author retains the moral right to the publication.
- The legal formal aspect of journal publication accessibility refers to Creative Commons Attribution-Non Commercial-Share Alike 4.0 International License (CC BY-NC-SA).
- All published manuscripts, whether in print or electronic form, are open access for educational, research, library purposes, and non-commercial uses. In addition to the aims mentioned above, the editorial board is not liable for any potential violations of copyright laws.
- The form to submit the manuscript's authenticity and copyright statement can be downloaded here.
Journal of Orthopaedi and Traumatology Surabaya (JOINTS) is licensed under a Creative Commons Attribution-Non Commercial-Share Alike 4.0 International License.