Skip to main content

Advertisement

Log in

Evaluation of the trabecular bone score in 35 children and adults with X-linked hypophosphatemic rickets

  • Original Article
  • Published:
Journal of Bone and Mineral Metabolism Aims and scope Submit manuscript

Abstract

Introduction

 The aim of this study is to evaluate and compare the trabecular bone scores (TBSs) of 11 children and 24 adults with X-linked hypophosphatemic rickets (XLH) and non-XLH subjects from a tertiary center.

Materials and methods

 The areal bone mineral density at the lumbar spine (LS-aBMD) and LS-aBMD Z score were analyzed by dual-energy X-ray absorptiometry. The bone mineral apparent density (BMAD) and LS-aBMD Z score adjusted for height Z score (LS-aBMD–HAZ) were calculated. The TBS was determined using TBS iNsight software based on DXA images from the Hologic QDR 4500 device.

Results

The XLH patients exhibited a higher mean LS-aBMD Z score, BMAD, and TBS than the non-XLH subjects (p < 0.01). LS-aBMD–HAZ and BMAD were greater in the XLH children than those in their corresponding non-XLH subjects (p < 0.01 and p = 0.02), and the XLH children trended toward a greater TBS (p = 0.06). The XLH adults had a higher LS-aBMD Z score, BMAD, and TBS than the non-XLH subjects (p < 0.01). When stratified by metabolic status according to the serum values of bone formation markers, compensated adult patients had a higher LS-aBMD Z score, BMAD, and TBS than non-XLH subjects (p < 0.01). Noncompensated patients had higher LS-aBMD Z scores and BMAD results than non-XLH subjects. However, TBS values did not differ statistically significantly between those groups (p = 0.45).

Conclusion

The higher LS-aBMD Z score, BMAD, and TBS result in the XLH patients compared to non-XLH subjects indicates an increased amount of trabecular bone within the lumbar spine, regardless of extraskeletal calcifications.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Similar content being viewed by others

References

  1. The HYP consortium (1995) A gene (PEX) with homologies to endopeptidases is mutated in patients with X-linked hypophosphatemic rickets. Nat Genet 11:130–136

    Article  Google Scholar 

  2. Haffner D, Emma F, Eastwood DM, Duplan MB, Bacchetta J et al (2019) Clinical practice recommendations for the diagnosis and management of X-linked hypophosphataemia. Nat Rev Nephrol 15:435–455

    Article  PubMed  PubMed Central  Google Scholar 

  3. Carpenter TO, Shaw NJ, Portale AA, Ward LM, Abrams SA, Pettifor JM (2017) Rickets. Nat Rev Dis Primers 3:17101

    Article  PubMed  Google Scholar 

  4. Colares Neto GP, Pereira RM, Alvarenga JC, Takayama L, Funari MF, Martin RM (2017) Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets. Osteoporos Int 28:1685–1692

    Article  CAS  PubMed  Google Scholar 

  5. Cheung M, Roschger P, Klaushofer K, Veilleux LN, Roughley P, Glorieux FH, Rauch F (2013) Cortical and trabecular bone density in X-linked hypophosphatemic rickets. J Clin Endocrinol Metab 98:E954–E961

    Article  PubMed  Google Scholar 

  6. Shanbhogue VV, Hansen S, Folkestad L, Brixen K, Beck-Nielsen SS (2015) Bone geometry, volumetric density, microarchitecture, and estimated bone strength assessed by HR-pQCT in adult patients with hypophosphatemic rickets. J Bone Miner Res 30:176–183

    Article  PubMed  Google Scholar 

  7. Beck-Nielsen SS, Brixen K, Gram J, Molgaard C (2013) High bone mineral apparent density in children with X-linked hypophosphatemia. Osteoporos Int 24:2215–2221

    Article  CAS  PubMed  Google Scholar 

  8. Padlina I, Gonzalez-Rodriguez E, Hans D, Metzger M, Stoll D, Aubry-Rozier B, Lamy O (2017) The lumbar spine age-related degenerative disease influences the BMD not the TBS: the Osteolaus cohort. Osteoporos Int 28:909–915

    Article  CAS  PubMed  Google Scholar 

  9. Anderson KB, Holloway-Kew KL, Mohebbi M, Kotowicz MA, Hans D, Pasco JA (2018) Is trabecular bone score less affected by degenerative-changes at the spine than lumbar spine BMD? Arch Osteoporos. https://doi.org/10.1007/s11657-018-0544-3

    Article  PubMed  Google Scholar 

  10. Shevroja E, Lamy O, Kohlmeier L, Koromani F, Rivadeneira F, Hans D (2017) Use of trabecular bone score (TBS) as a complementary approach to dual-energy X-ray absorptiometry (DXA) for fracture risk assessment in clinical practice. J Clin Densitom 20:334–345

    Article  PubMed  Google Scholar 

  11. Martineau P, Leslie WD (2017) Trabecular bone score (TBS): method and applications. Bone 104:66–72

    Article  CAS  PubMed  Google Scholar 

  12. Caparbo VF, Furlam P, Saad CGS, Alvarenga JC, Aubry-Rozier B, Hans D, de Brum-Fernandes AJ, Pereira RMR (2019) Assessing bone impairment in ankylosing spondylitis (AS) using the trabecular bone score (TBS) and high-resolution peripheral quantitative computed tomography (HR-pQCT). Bone 122:8–13

    Article  PubMed  Google Scholar 

  13. Alvarenga PPM, Silva BC, Diniz MP, Leite MB, da Silva CAM, de Cassia-Mendes-Eleuterio J, Soares MMS, Bilezikian JP, Camargos BM (2019) Trabecular bone score: a useful clinical tool for the evaluation of skeletal health in women of short stature. Endocrine 66:398–404

    Article  CAS  PubMed  Google Scholar 

  14. Silva BC, Leslie WD, Resch H, Lamy O, Lesnyak O, Binkley N, McCloskey EV, Kanis JA, Bilezikian JP (2014) Trabecular bone score: a noninvasive analytical method based upon the DXA image. J Bone Miner Res 29:518–530

    Article  PubMed  Google Scholar 

  15. Leaf DE, Pereira RC, Bazari H, Juppner H (2013) Oncogenic osteomalacia due to FGF23-expressing colon adenocarcinoma. J Clin Endocrinol Metab 98:887–891

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  16. RRID: AB_2782966, http://antibodyregistry.org/AB_2782966. Accessed 8 Jan 2019

  17. RRID: AB_2782967, http://antibodyregistry.org/AB_2782967. Accessed 8 Jan 2019

  18. RRID: AB_2782968, http://antibodyregistry.org/AB_2782968. Accessed 8 Jan 2019

  19. Cvijetić S, Koršić M (2003) Apparent bone mineral density estimated from DXA in healthy men and women. Osteoporos Int 15:295–300. https://doi.org/10.1007/s00198-003-1525-x

    Article  PubMed  Google Scholar 

  20. Zemel BS, Leonard MB, Kelly A, Lappe JM, Gilsanz V, Oberfield S, Kalkwarf HJ (2010) Height adjustment in assessing dual energy X-ray absorptiometry measurements of bone mass and density in children. J Clin Endocrinol Metab 95:1265–1273. https://doi.org/10.1210/jc.2009-2057

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  21. Muschitz C, Kocijan R, Haschka J, Pahr D, Kaider A, Pietschmann P, Hans D, Muschitz GK, Fahrleitner-Pammer A, Resch H (2015) TBS reflects trabecular microarchitecture in premenopausal women and men with idiopathic osteoporosis and low-traumatic fractures. Bone 79:259–266

    Article  PubMed  Google Scholar 

  22. Kindler J, Lappe J, Gilsanz V, Oberfield S, Shepherd J, Kelly A, Winer K, Kalkwarf H, Zemel B (2019) Lumbar spine bone mineral apparent density in children: results from the bone mineral density in childhood study. J Clin Endocrinol Metab 104:1283–1292

    PubMed  Google Scholar 

  23. Duran I, Martakis K, Rehberg M, Semler O, Schoenau E (2018) Individualized evaluation of lumbar bone mineral density and bone mineral apparent density in children and adolescents. Arch Osteoporos 13:117

    Article  PubMed  Google Scholar 

  24. Beck-Nielsen SS, Brusgaard K, Rasmussen LM, Brixen K, Brock-Jacobsen B, Poulsen MR, Vestergaard P, Ralston SH, Albagha OM, Poulsen S, Haubek D, Gjorup H, Hintze H, Andersen MG, Heickendorff L, Hjelmborg J, Gram J (2010) Phenotype presentation of hypophosphatemic rickets in adults. Calcif Tissue Int 87:108–119

    Article  CAS  PubMed  Google Scholar 

  25. Robinson ME, AlQuorain H, Murshed M, Rauch F (2019) Mineralized tissues in hypophosphatemic rickets. Pediatr Nephrol. https://doi.org/10.1007/s00467-019-04290-y

    Article  PubMed  Google Scholar 

  26. Marie PJ, Glorieux FH (1983) Relation between hypomineralized periosteocytic lesions and bone mineralization in vitamin D-resistant rickets. Calcif Tissue Int 35:443–448

    Article  CAS  PubMed  Google Scholar 

  27. Rauch F (2006) Material matters: a mechanostat-based perspective on bone development in osteogenesis imperfecta and hypophosphatemic rickets. J Musculoskelet Neuronal Interact 6:142–146

    CAS  PubMed  Google Scholar 

  28. Malhan D, Muelke M, Rosch S, Schaefer AB, Merboth F, Weisweiler D, Heiss C, Arganda-Carreras I, El Khassawna T (2018) An optimized approach to perform bone histomorphometry. Front Endocrinol (Lausanne) 9:666

    Article  PubMed  Google Scholar 

  29. Kulak CA, Dempster DW (2010) Bone histomorphometry: a concise review for endocrinologists and clinicians. Arq Bras Endocrinol Metabol 54:87–98

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

No benefits in any form have been received or will be received from a commercial party directly or indirectly related to the subject of this article.

Author information

Authors and Affiliations

Authors

Contributions

Conceptualization and methodology: CNGP, RPRMM, and RM. Formal analysis and investigation: CNGP, AJC, TL. FMFA, and MRM. Writing: CNGP and MRM.

Corresponding author

Correspondence to Guido de Paula Colares Neto.

Ethics declarations

Conflict of interest

All authors have no conflicts of interest.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

de Paula Colares Neto, G., Pereira, R.M.R., Alvarenga, J.C. et al. Evaluation of the trabecular bone score in 35 children and adults with X-linked hypophosphatemic rickets. J Bone Miner Metab 41, 666–672 (2023). https://doi.org/10.1007/s00774-023-01442-x

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00774-023-01442-x

Keywords

Navigation