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Bisphosphonate-induced zebra lines in fibrous dysplasia of bone: histo-radiographic correlation in a case of McCune–Albright syndrome

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Abstract

Bisphosphonates (BPs) are currently used in the treatment of diverse bone diseases including fibrous dysplasia of bone (FD). In pediatric patients, a radiographic consequence of cyclical administration of BPs is the development of apo-, epi-, and meta-physeal sclerotic bands, otherwise known as zebra lines, which result from the temporary inhibition of osteoclastic activity at the time of drug treatment. We report here on a child with McCune–Albright syndrome (FD in addition to hyperfunctioning endocrinopathies and skin hyperpigmentation) treated with cyclical intravenous infusions of pamidronate in which conventional radiography, contact microradiography, histology, and backscattered electron image analysis demonstrated that zebra lines formed only where bone was normal, were arrested at the boundary between FD-unaffected and FD-affected bone where bone is sclerotic, and were absent within the undermineralized FD bone. Moreover, in spite of the treatment, the FD lesions continued to expand. This case report is unique because no previously published studies correlated the radiographic and the histologic features of BP-induced zebra lines in the metaphysis of an FD-affected long bone of the limbs.

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Authors and Affiliations

  1. Department of Molecular Medicine, Sapienza University, Policlinico Umberto I, Viale Regina 324, 00161, Rome, Italy

    Alessandro Corsi & Mara Riminucci

  2. Department of Orthopaedic Surgery, University of Rome Tor Vergata, Rome, Italy

    Ernesto Ippolito

  3. Skeletal Biology Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Department of Health and Human Services, Bethesda, MD, USA

    Pamela G. Robey

  4. Dental Physical Sciences, Queen Mary University of London, London, E1 4NS, UK

    Alan Boyde

Authors
  1. Alessandro Corsi
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  2. Ernesto Ippolito
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  3. Pamela G. Robey
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  4. Mara Riminucci
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  5. Alan Boyde
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Corresponding authors

Correspondence to Alessandro Corsi or Mara Riminucci.

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Conflict of interest

The authors declare that they have no conflicts of interest.

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from all individual participants included in the study.

Funding

This study was funded by Telethon (Grant number: GGP15198), and, in part, by the Division of Intramural Research, National Institute of Dental and Craniofacial Research, a part of the Intramural Research Program, National Institutes of Health, Department of Health and Human Services (ZIA DE000380).

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Corsi, A., Ippolito, E., Robey, P.G. et al. Bisphosphonate-induced zebra lines in fibrous dysplasia of bone: histo-radiographic correlation in a case of McCune–Albright syndrome. Skeletal Radiol 46, 1435–1439 (2017). https://doi.org/10.1007/s00256-017-2698-2

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  • DOI: https://doi.org/10.1007/s00256-017-2698-2

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