Startseite A case of a severe reaction following the use of bisphosphonates in a patient with osteogenesis imperfecta
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A case of a severe reaction following the use of bisphosphonates in a patient with osteogenesis imperfecta

  • Julie Park EMAIL logo , Andrew Riordan , Nicholas J. Bishop , Poonam Dharmaraj und Renuka Ramakrishnan
Veröffentlicht/Copyright: 11. Juli 2019

Abstract

We present a case of an unusual delayed multi-systemic reaction, following treatment with pamidronate. Although serious adverse reactions have been reported with pamidronate use, such a severe reaction, late in the course of pamidronate treatment, has not been described before. An 11-month-old boy with severe and complex osteogenesis imperfecta (OI) presented with hyperpyrexia and respiratory distress 10 days after his fifth cycle of pamidronate. He had significant derangement of his biochemical parameters including a positive urine myoglobin. His respiratory distress was out of proportion to his chest radiograph changes. Bilevel positive airway pressure (BiPAP) and paediatric intensive care (PICU) admission were required. He was extensively investigated to exclude other diagnoses, but all of these investigations were negative. The reaction resembled rhabdomyolysis. He made a full recovery with only supportive management.


Corresponding author: Dr. Julie Park, Alder Hey Children’s NHS Foundation Trust, East Prescott Road, Liverpool L14 5AB, UK

  1. Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.

  2. Research funding: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

  3. Employment or leadership: None declared.

  4. Honorarium: None declared.

  5. Competing interests: The funding organisation(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.

  6. Conflicts of interest: None.

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Received: 2019-01-07
Accepted: 2019-05-04
Published Online: 2019-07-11
Published in Print: 2019-08-27

©2019 Walter de Gruyter GmbH, Berlin/Boston

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