Skip to main content

ORIGINAL RESEARCH article

Front. Pediatr.
Sec. Pediatric Endocrinology
Volume 12 - 2024 | doi: 10.3389/fped.2024.1500023

Trabecular bone scores in children with osteogenesis imperfecta respond differently to bisphosphonate treatment depending on disease severity

Provisionally accepted
Natsuko Futagawa Natsuko Futagawa 1,2Kosei Hasegawa Kosei Hasegawa 2*Hiroyuki Miyahara Hiroyuki Miyahara 1Hiroyuki Tanaka Hiroyuki Tanaka 3Hirokazu Tsukahara Hirokazu Tsukahara 1
  • 1 Department of Pediatrics, Graduate School of Medicine Dentistry and Pharmaceutical Sciences, Okayama University, Okayama, Okayama, Japan
  • 2 Department of Pediatrics, Okayama University Hospital, Okayama University, Okayama, Japan
  • 3 Department of Pediatrics, Okayama Saiseikai General Hospital, Okayama, Okayama, Japan

The final, formatted version of the article will be published soon.

    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.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).Because BIS affect the BMD and TBS differently based on the severity of OI, treatment goals may need to be stratified by disease severity.

    Keywords: Bone Density, Osteoporosis, Bone Diseases, Connective Tissue, Child

    Received: 22 Sep 2024; Accepted: 13 Nov 2024.

    Copyright: © 2024 Futagawa, Hasegawa, Miyahara, Tanaka and Tsukahara. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

    * Correspondence: Kosei Hasegawa, Department of Pediatrics, Okayama University Hospital, Okayama University, Okayama, Japan

    Disclaimer: All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.