Conflict of interest: Nothing to declare.
Whole-body magnetic resonance imaging (WB-MRI) as surveillance for subsequent malignancies in survivors of hereditary retinoblastoma: A pilot study
Article first published online: 1 NOV 2013
© 2013 Wiley Periodicals, Inc.
Pediatric Blood & Cancer
Volume 61, Issue 8, pages 1440–1444, August 2014
How to Cite
Friedman, D. N., Lis, E., Sklar, C. A., Oeffinger, K. C., Reppucci, M., Fleischut, M. H., Francis, J. H., Marr, B., Abramson, D. H. and Dunkel, I. J. (2014), Whole-body magnetic resonance imaging (WB-MRI) as surveillance for subsequent malignancies in survivors of hereditary retinoblastoma: A pilot study. Pediatr. Blood Cancer, 61: 1440–1444. doi: 10.1002/pbc.24835
- Issue published online: 10 JUN 2014
- Article first published online: 1 NOV 2013
- Manuscript Accepted: 6 AUG 2013
- Manuscript Received: 3 JUL 2013
- Perry's Promise Fund
- Clinical and Translational Science Center at Weill Cornell Medical College. Grant Number: UL1TR000457
- National Institutes of Health. Grant Number: K05CA160724
- pediatric oncology;
- whole-body MRI
Individuals with hereditary retinoblastoma (RB) are at very high risk of developing subsequent malignant neoplasms (SMNs) of which osteosarcoma (OS) is one of the most common. We hypothesized that annual surveillance using whole-body magnetic resonance imaging (WB-MRI) in asymptomatic survivors of hereditary RB would detect SMN of the bone and soft tissues at an early stage.
Retrospective review of the results of a WB-MRI screening program in hereditary RB survivors from February 2008 to August 2012. The primary outcome was to determine the sensitivity and specificity of WB-MRI in detecting SMNs.
Twenty-five patients had at least one WB-MRI performed (range: 1–5). First WB-MRI was performed at a median age of 16 years (range: 8–25 years). WB-MRI detected new osseous abnormalities suspicious for malignancy in five patients: two were diagnosed with localized high-grade OS of the extremity and three were found to have benign osseous abnormalities after dedicated imaging (n = 5/5) and/or biopsy (n = 3/5). One patient was diagnosed with secondary OS 3 months after a normal screening WB-MRI exam. Among a total of 41 WB-MRI screening tests performed in survivors of hereditary RB, the sensitivity of detecting SMN was 66.7% and the specificity was 92.1%.
Preliminary results suggest that annual WB-MRI surveillance detects SMN in survivors of hereditary RB, but with modest sensitivity. Further study is needed to assess the performance of annual surveillance WB-MRIs and whether this modality decreases SMN-related mortality in RB survivors. Pediatr Blood Cancer 2014; 61:1440–1444. © 2013 Wiley Periodicals, Inc.