Vertebral fracture assessment (VFA) for monitoring vertebral reshaping in children and adolescents with osteogenesis imperfecta treated with intravenous neridronate

Diacinti, D.; Pisani, D.; Cipriani, C.; Celli, M.; Zambrano, A.; Diacinti, D.; Kripa, E.; Iannacone, A.; Colangelo, L.; Nieddu, L.; Pepe, J.; Minisola, S.

Affiliations

¹Department of Radiological Sciences, Oncology and Pathology, Sapienza University of Rome, viale Regina Elena 324, 00161 Rome, Italy

²Department of Clinical and Molecular Medicine, Saint'Andrea Hospital, Sapienza University of Rome, via Grottarossa, 1035, Rome, Italy

³Department of Clinical Internal, Anaesthesiologic and Cardiovascular Sciences, Sapienza University of Rome, Viale del Policlinico 155, 00161,Rome, Italy

⁴Department Materno-Infantile, Center of Rare Disease and Skeletal Dysplasia, Policlinico Umberto I Hospital, Viale del Policlinico, 155, 00161 Rome, Italy

⁵Department of Oral and Maxillo-Facial Sciences, Policlinico Umberto I Hospital, Sapienza University of Rome, via Caserta 6, 00161, Rome, Italy

⁶Department of Diagnostic and Molecular Imaging, Radiology and Radiotherapy, University Foundation Hospital Tor Vergata, Rome, Italy

⁷Faculty of Economics, UNINT University, Via Cristoforo Colombo 200, 00147, Rome, Italy

Abstract and keywords

Purpose The study was aimed at monitoring vertebral bodies changes with the use of Vertebral Fracture Assessment (VFA) in children and adolescents affected by osteogenesis imperfecta (OI) during treatment with intravenous neridronate.

Methods: 60 children and adolescents (35 males and 25 females; age 1–16 years) with OI type I, III and IV were included in the study. Intravenous neridronate was administered at the dose of 2 mg/kg every 3 months in all patients. Lumbar spine (LS) bone mineral density (BMD) and VFA by dual X-ray absorptiometry (DXA) were assessed every 6 months up to 24 months during treatment. VFA with vertebral morphometry (MXA) was used to calculate the three indices of vertebral deformity: wedging, concavity and crushing. Serum calcium, phosphate, parathyroid hormone (PTH), 25-hydroxy-vitamin D [25(OH)D], total alkaline phosphatase (ALP), bone alkaline phosphatase (BALP) and urinary C-terminal telopeptide of type 1 collagen (CTx) were measured at any time point.

Results: Mean LS BMD values significantly increased at 24 months compared to baseline (p < 0.0001); the corresponding Z-score values were −1.28 ± 1.23 at 24 months vs −2.46 ± 1.25 at baseline; corresponding mean Bone Mineral Apparent Density (BMAD) values were 0.335 ± 0.206 vs 0.464 ± 0.216. Mean serum levels of ALP, BALP and CTx significantly decreased from baseline to 24 months. By MXA, we observed a significant 19.1% reduction of the mean wedging index of vertebral reshaping at 12 months, and 38.4% at 24 months (p < 0.0001) and of the mean concavity index (16.3% at 12 months and 35.9% at 24 months; p < 0.0001). Vertebral reshaping was achieved for 66/88 (75%) wedge fractures and 59/70 (84%) concave fractures, but there were 4 incident mild fractures. Finally, VF rate was reduced at 24 months compared to baseline: 37/710 (5.2%) vs 158/710 (22.2%).

Conclusion: Our study demonstrates the utility of VFA as a safe and alternative methodology in the follow-up of children and adolescents with OI.

Keywords: Vertebral fracture assessment (VFA); Vertebral morphometry; Osteogenesis imperfecta; Children; Neridronate

Links

DOI: 10.1016/j.bone.2020.115608

PubMed: 32829035

WoS: 000604733200017

History

Received: 2020-04-24

Revised: 2020-08-15

Accepted: 2020-08-15

Online: 2020-08-20

Cited Works (3)

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