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Published on: August 8, 2019
Standardized reporting and quantification of whole-body MRI findings in children with chronic non-bacterial
C M Andreasen1,2,3, R F Klicman4,5, T Herlin6,4
1Department of Rheumatology, Aarhus University Hospital, Aarhus N, Denmark. carand@rm.dk.
Objectives:
The objectives were to assess changes in radiological disease activity in children with chronic non-bacterial osteomyelitis (CNO) receiving pamidronate therapy and to test a modified radiological index for non-bacterial osteitis (mRINBO) in CNO. mRINBO was used for standardized reporting and quantification of whole-body MRI (WBMRI) findings resulting in an individual summary patient score.
Methods:
WBMRI was retrospectively assessed in 18 children with CNO at baseline and after receiving pamidronate therapy for one year. Parameters of interest were: number and anatomic site of radiologically active bone lesions (RAL), size of RAL, extramedullary affection, spinal involvement and changes in mRINBO, which includes both the number and maximal size of RAL (RALmax) in addition to extramedullary and chronic changes.
Results:
At the time of diagnosis, the mean age of the children was 9.8 (sd, 8.7-10.9) years and 11/18 were females. The number of RALs per patient decreased from median [interquartile range] 4.5 [3-8] to 3 [2-5] RALs per patient (p = 0.02) and extramedullary inflammatory changes regressed. Sixty-one percent of all RALs occurring at baseline resolved and three children became without active inflammatory lesions by WBMRI. The median size of RALs did not change when taking new lesions occurring in 7/18 children into account, but RALmax decreased significantly from 39 [29-45] mm at baseline to 28 [20-40] mm (p < 0.01) at year-one with a concomitant decrease of mRINBO from a median of 5 [4-7] to 4 [3-5] (p = 0.05).
Conclusions:
Pamidronate therapy resulted in a decrease of mRINBO from baseline to year one. mRINBO may be a potential scoring method to quantify changes in radiological disease activity in children with CNO. However, further studies are needed to test feasibility and validity of mRINBO.
Insights
Pamidronate therapy reduced radiological disease activity in children with chronic non-bacterial osteomyelitis (CNO). A modified radiological index for non-bacterial osteitis (mRINBO) showed decreased scores after one year, suggesting its potential for assessing CNO progression.
Area of Science:
- Pediatric rheumatology
- Radiology
- Pharmacology
Background:
- Chronic non-bacterial osteomyelitis (CNO) is a rare inflammatory bone disease in children.
- Assessing radiological disease activity in CNO is crucial for treatment monitoring.
- Standardized radiological indices can improve objective disease assessment.
Purpose of the Study:
- To evaluate changes in radiological disease activity in pediatric CNO patients treated with pamidronate.
- To assess the utility of a modified radiological index for non-bacterial osteitis (mRINBO) in CNO.
- To quantify whole-body MRI (WBMRI) findings using mRINBO.
Main Methods:
- Retrospective WBMRI analysis in 18 children with CNO at baseline and after one year of pamidronate therapy.
- Assessment of radiologically active bone lesions (RAL), extramedullary affection, and spinal involvement.
- Application of mRINBO, incorporating RAL number, size, and chronic changes, for scoring.
Main Results:
- Pamidronate therapy led to a significant decrease in the number of RALs and regression of extramedullary inflammatory changes.
- Sixty-one percent of baseline RALs resolved, and three children achieved remission.
- The modified radiological index for non-bacterial osteitis (mRINBO) score decreased significantly, indicating reduced disease activity.
Conclusions:
- Pamidronate therapy effectively reduced radiological disease activity in children with CNO.
- The mRINBO shows promise as a tool for quantifying disease activity changes in CNO.
- Further research is necessary to validate the feasibility and reliability of mRINBO.
