Efficacy of pamidronate in children with chronic non-bacterial osteitis using whole body MRI as a marker of disease activity.


Journal

Pediatric rheumatology online journal
ISSN: 1546-0096
Titre abrégé: Pediatr Rheumatol Online J
Pays: England
ID NLM: 101248897

Informations de publication

Date de publication:
04 Jul 2019
Historique:
received: 25 03 2019
accepted: 12 06 2019
entrez: 6 7 2019
pubmed: 6 7 2019
medline: 14 1 2020
Statut: epublish

Résumé

To study the response to pamidronate using whole body magnetic resonance imaging (WB-MRI) in children with chronic non-bacterial osteitis (CNO) in a tertiary health centre. The medical records of children under the age of sixteen with a diagnosis of chronic non-bacterial osteitis between 2005 and 2018 were reviewed. All those who were treated with pamidronate were included and relevant data was collected. Response to therapy was determined based on the status of lesions on WB- MRI. Forty six patients were included in the study. Pre- and post-treatment WB-MRI was available in forty patients. Cumulative lesions pre-treatment were 150 and reduced to 45 (30%) post-treatment. Seventeen patients (42.5%) had a good response with complete resolution of all lesions and nine patients (22.5%) worsened during or following treatment with pamidronate. Vertebral disease had a good response and 82.3% of the lesions resolved completely. Our study describes the experience with pamidronate in a tertiary health centre using WB-MRI as a marker of disease activity. Pamidronate was well tolerated in our cohort and treatment response was fairly good. 1. Bisphosphonates can be used in the treatment of CNO when response to NSAIDs is suboptimal. 2. In the presence of spinal or mandibular lesions bisphosphonates were used as first line. 3. Treatment was escalated to a TNF blocker when response to bisphosphonates was suboptimal.

Sections du résumé

BACKGROUND BACKGROUND
To study the response to pamidronate using whole body magnetic resonance imaging (WB-MRI) in children with chronic non-bacterial osteitis (CNO) in a tertiary health centre.
METHODS METHODS
The medical records of children under the age of sixteen with a diagnosis of chronic non-bacterial osteitis between 2005 and 2018 were reviewed. All those who were treated with pamidronate were included and relevant data was collected. Response to therapy was determined based on the status of lesions on WB- MRI.
RESULTS RESULTS
Forty six patients were included in the study. Pre- and post-treatment WB-MRI was available in forty patients. Cumulative lesions pre-treatment were 150 and reduced to 45 (30%) post-treatment. Seventeen patients (42.5%) had a good response with complete resolution of all lesions and nine patients (22.5%) worsened during or following treatment with pamidronate. Vertebral disease had a good response and 82.3% of the lesions resolved completely.
CONCLUSION CONCLUSIONS
Our study describes the experience with pamidronate in a tertiary health centre using WB-MRI as a marker of disease activity. Pamidronate was well tolerated in our cohort and treatment response was fairly good.
SIGNIFICANCE AND INNOVATION CONCLUSIONS
1. Bisphosphonates can be used in the treatment of CNO when response to NSAIDs is suboptimal. 2. In the presence of spinal or mandibular lesions bisphosphonates were used as first line. 3. Treatment was escalated to a TNF blocker when response to bisphosphonates was suboptimal.

Identifiants

pubmed: 31272461
doi: 10.1186/s12969-019-0340-7
pii: 10.1186/s12969-019-0340-7
pmc: PMC6609396
doi:

Substances chimiques

Bone Density Conservation Agents 0
Pamidronate OYY3447OMC

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

35

Références

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Auteurs

C S Bhat (CS)

Departments of Paediatric Rheumatology and Immunology, Bristol Royal Hospital for Children, Level 6, Education Centre, Upper Maudlin Street, Bristol, BS2 8BJ, UK.

M Roderick (M)

Departments of Paediatric Rheumatology and Immunology, Bristol Royal Hospital for Children, Level 6, Education Centre, Upper Maudlin Street, Bristol, BS2 8BJ, UK.

E S Sen (ES)

Bristol Children's Hospital and Translational Health Sciences Bristol Medical School, University of Bristol, BS81QU, Bristol, UK.
Department of Paediatric Rheumatology, Great North Children's Hospital, NE14LP, Newcastle upon Tyne, UK.

A Finn (A)

Departments of Paediatric Rheumatology and Immunology, Bristol Royal Hospital for Children, Level 6, Education Centre, Upper Maudlin Street, Bristol, BS2 8BJ, UK.

A V Ramanan (AV)

Departments of Paediatric Rheumatology and Immunology, Bristol Royal Hospital for Children, Level 6, Education Centre, Upper Maudlin Street, Bristol, BS2 8BJ, UK. avramanan@hotmail.com.
Bristol Children's Hospital and Translational Health Sciences Bristol Medical School, University of Bristol, BS81QU, Bristol, UK. avramanan@hotmail.com.

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