Whole-body magnetic resonance imaging as surveillance for subsequent malignancies in preadolescent, adolescent, and young adult survivors of germline retinoblastoma: An update Journal Article


Authors: Novetsky Friedman, D.; Hsu, M.; Moskowitz, C. S.; Francis, J. H.; Lis, E.; Fleischut, M. H.; Oeffinger, K. C.; Walsh, M.; Tonorezos, E. S.; Sklar, C. A.; Abramson, D. H.; Dunkel, I. J.
Article Title: Whole-body magnetic resonance imaging as surveillance for subsequent malignancies in preadolescent, adolescent, and young adult survivors of germline retinoblastoma: An update
Abstract: Background: Germline retinoblastoma (Rb) survivors are at lifelong risk for developing subsequent malignancies (SMNs). Optimal surveillance modalities are needed to detect SMN at an early stage in this high-risk cohort. We investigated the use of rapid whole-body magnetic resonance imaging (WB-MRI) as a noninvasive screening modality in this cohort. Procedure: WB-MRI was performed in asymptomatic preadolescent, adolescent, or young adult survivors of germline Rb from February 1, 2008 to December 31, 2018 at a tertiary cancer center. We calculated sensitivity and specificity of WB-MRI and rate of false-positive findings requiring additional evaluation. Results: Overall, 110 WB-MRI were performed in 47 germline Rb survivors (51% female; median age at initial WB-MRI: 15.5 years [range 8-25.3]). Patients received 1-10 annual WB-MRI examinations (median: two). Thirteen patients had an abnormal WB-MRI; three findings were deemed to be likely benign and were not evaluated further. Ten patients required dedicated imaging and three required biopsy; two patients were diagnosed with localized high-grade osteosarcoma, while the other eight had benign findings. One patient was diagnosed with secondary osteosarcoma 3 months after normal WB-MRI. In total, there were 96 true negatives, 11 false positives, two true positives, and one false negative. The sensitivity of WB-MRI in this cohort was 66.7% (95% confidence interval [CI], 14.2-96.0) and the specificity was 89.7% (95% CI, 83.6-93.7). Conclusions: Based on our 10-year experience, surveillance WB-MRI appears to have limited utility as a surveillance modality for SMN in germline Rb survivors. Alternate screening modalities should be investigated. © 2020 Wiley Periodicals, Inc.
Keywords: retinoblastoma; survivorship; surveillance; second cancers; whole-body mri
Journal Title: Pediatric Blood and Cancer
Volume: 67
Issue: 7
ISSN: 1545-5009
Publisher: Wiley Periodicals, Inc  
Date Published: 2020-07-01
Start Page: e28389
Language: English
DOI: 10.1002/pbc.28389
PUBMED: 32386119
PROVIDER: scopus
DOI/URL:
Notes: Article -- Export Date: 1 June 2020 -- Source: Scopus
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MSK Authors
  1. Meier Hsu
    118 Hsu
  2. Ira J Dunkel
    281 Dunkel
  3. Charles A Sklar
    298 Sklar
  4. Eric Lis
    113 Lis
  5. Jasmine Helen Francis
    123 Francis
  6. Chaya S. Moskowitz
    196 Moskowitz
  7. David H Abramson
    283 Abramson
  8. Kevin Oeffinger
    280 Oeffinger
  9. Michael Francis Walsh
    73 Walsh