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Trabecular bone scores in children with osteogenesis imperfecta respond differently to bisphosphonate treatment
Natsuko Futagawa1,2, Kosei Hasegawa2, Hiroyuki Miyahara1
1Department of Pediatrics, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan.
Insights
Bisphosphonate (BIS) treatment improves bone mineral density (BMD) and trabecular bone score (TBS) in children with osteogenesis imperfecta (OI). The impact of BIS on TBS is more pronounced in moderate to severe OI cases, suggesting stratified treatment approaches.
Area of Science:
- Pediatric Endocrinology
- Skeletal Dysplasias
- Bone Biology
Background:
- Osteogenesis imperfecta (OI) is a genetic disorder causing brittle bones.
- Bisphosphonates (BISs) are the primary treatment for pediatric OI, aiming to increase bone mineral density (BMD).
- Bone quality, assessed by trabecular bone score (TBS), is crucial for bone strength but less studied during BIS therapy.
Purpose of the Study:
- To investigate the changes in TBS during BIS treatment in children with OI.
- To compare the effects of BIS on BMD and TBS across different OI severity groups.
Main Methods:
- Retrospective analysis of dual-energy x-ray absorptiometry (DXA) images from 29 children with OI (mild vs. moderate to severe).
- Calculation of areal BMD (aBMD), height-adjusted BMD (BMDHAZ), TBS, and their respective Z-scores.
- Assessment of correlations between these parameters and BIS treatment duration.
Main Results:
- In mild OI, aBMD and BMDHAZ Z-scores significantly correlated with treatment duration; TBS Z-score showed a non-significant increasing trend.
- In moderate to severe OI, TBS Z-score significantly increased with treatment duration, unlike aBMD Z-score.
- BMDHAZ Z-score showed only a weak positive correlation with treatment duration in the moderate to severe group.
Conclusions:
- Bisphosphonates impact BMD and TBS differently depending on OI severity.
- Treatment strategies for OI using BIS may require stratification based on disease severity to optimize outcomes for both bone density and quality.
Introduction:
Osteogenesis imperfecta (OI) is a congenital skeletal disorder characterized by bone fragility. Bisphosphonates (BISs) have become the mainstream treatment in children with OI. However, an optimal treatment protocol has not yet been established, while BIS treatment tends to be administered to normalize bone mineral density (BMD). Bone quality is an important component of bone strength. The trabecular bone score (TBS) is a quantitative measure of the microstructure that affects bone quality. This study investigated the TBS during BIS treatment in children with OI.
Materials And Methods:
Twenty-nine children with OI were enrolled and classified into two groups: mild (type 1) and moderate to severe (types 3 and 4). Dual-energy x-ray absorptiometry images were retrospectively analyzed for TBS calculation. The relationship between the areal BMD (aBMD), its Z-score, height-adjusted BMD (BMDHAZ) Z-score, TBS, and TBS Z-score with the treatment duration was assessed for each group.
Results:
In the mild group, the aBMD, its Z-score, and BMDHAZ Z-score showed a significant positive correlation with treatment duration (r = 0.68, 0.68, 0.72, respectively, p < 0.01). The TBS Z-score tended to increase with treatment duration, albeit without reaching significance. In the moderate to severe group, the TBS Z-score showed a significant positive correlation with treatment duration (r = 0.48, p < 0.01), in contrast to the aBMD Z-score, which did not increase. Finally, the BMDHAZ Z-score only showed a weak positive correlation with treatment duration (r = 0.37, p < 0.01).
Conclusion:
Because BIS affect the BMD and TBS differently based on the severity of OI, treatment goals may need to be stratified by disease severity.
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