Whole-body magnetic resonance imaging (WB-MRI) as surveillance for subsequent malignancies in survivors of hereditary retinoblastoma: A pilot study Journal Article

Authors: Friedman, D. N.; Lis, E.; Sklar, C. A.; Oeffinger, K. C.; Reppucci, M.; Fleischut, M. H.; Francis, J. H.; Marr, B.; Abramson, D. H.; Dunkel, I. J.
Article Title: Whole-body magnetic resonance imaging (WB-MRI) as surveillance for subsequent malignancies in survivors of hereditary retinoblastoma: A pilot study
Abstract: Background: 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. Procedure: 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. Results: 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%. Conclusions: 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. © 2013 Wiley Periodicals, Inc.
Keywords: osteosarcoma; adolescent; adult; child; clinical article; cancer staging; sensitivity and specificity; cancer screening; retinoblastoma; cancer survivor; survivors; pilot study; screening; malignant neoplastic disease; pediatric oncology; predictive value; nuclear magnetic resonance scanner; diagnostic test accuracy study; chondroblastoma; whole-body mri; whole body mri; human; male; female; priority journal; article; spondylolysis; subsequent malignant neoplasm
Journal Title: Pediatric Blood and Cancer
Volume: 61
Issue: 8
ISSN: 1545-5009
Publisher: Wiley Periodicals, Inc  
Date Published: 2014-08-01
Start Page: 1440
End Page: 1444
Language: English
DOI: 10.1002/pbc.24835
PROVIDER: scopus
PMCID: PMC4007376
PUBMED: 24402721
Notes: Pediatr. Blood Cancer -- Cited By (since 1996):1 -- Export Date: 8 July 2014 -- CODEN: PBCEA -- Source: Scopus
Citation Impact
MSK Authors
  1. Brian Marr
    111 Marr
  2. Ira J Dunkel
    284 Dunkel
  3. Charles A Sklar
    298 Sklar
  4. Eric Lis
    113 Lis
  5. Jasmine Helen Francis
    124 Francis
  6. David H Abramson
    284 Abramson
  7. Kevin Oeffinger
    280 Oeffinger