Clinical outcomes of pediatric patients receiving multimodality treatment of second central nervous system relapse of neuroblastoma Journal Article

  


Authors: Tringale, K. R.; Wolden, S. L.; Casey, D. L.; Kushner, B. H.; Luo, L.; Pandit-Taskar, N.; Souweidane, M.; Cheung, N. K. V.; Modak, S.; Basu, E. M.; Kramer, K.
Article Title: Clinical outcomes of pediatric patients receiving multimodality treatment of second central nervous system relapse of neuroblastoma
Abstract: Background: In high-risk neuroblastoma, multimodality therapy including craniospinal irradiation (CSI) is effective for central nervous system (CNS) relapse. Management of post-CSI CNS relapse is not clearly defined. Procedure: Pediatric patients with neuroblastoma treated with CSI between 2000 and 2019 were identified. Treatment of initial CNS disease (e.g., CSI, intraventricular compartmental radioimmunotherapy [cRIT] with 131I-monoclonal antibodies targeting GD2 or B7H3) and management of post-CSI CNS relapse (“second CNS relapse”) were characterized. Cox proportional hazards models to evaluate factors associated with third CNS relapse and overall survival (OS) were used. Results: Of 128 patients (65% male, median age 4 years), 19 (15%) received CSI with protons and 115 (90%) had a boost. Most (103, 81%) received cRIT, associated with improved OS (hazard ratio [HR] 0.3, 95% confidence interval [CI]: 0.1–0.5, p <.001). Forty (31%) developed a second CNS relapse, associated with worse OS (1-year OS 32.5%, 95% CI: 19-47; HR 3.8; 95% CI: 2.4–6.0, p <.001), and more likely if the leptomeninges were initially involved (HR 2.5, 95% CI: 1.3–4.9, p =.006). Median time to second CNS relapse was 6.8 months and 51% occurred outside the CSI boost field. Twenty-five (63%) patients underwent reirradiation, most peri-operatively (18, 45%) with focal hypofractionation. Eight (20%) patients with second CNS relapse received cRIT, associated with improved OS (HR 0.1; 95% CI: 0.1–0.4, p <.001). Conclusions: CNS relapse after CSI for neuroblastoma portends a poor prognosis. Surgery with hypofractionated radiotherapy was the most common treatment. Acknowledging the potential for selection bias, receipt of cRIT both at first and second CNS relapse was associated with improved survival. This finding necessitates further investigation. © 2022 Wiley Periodicals LLC.
Keywords: child; preschool child; child, preschool; multimodality cancer therapy; combined modality therapy; neoplasm recurrence, local; radiotherapy; central nervous system; neuroblastoma; tumor recurrence; radioimmunotherapy; therapy; craniospinal irradiation; humans; human; male; female; pediatric cancers; intraventricular compartmental radioimmunotherapy
Journal Title: Pediatric Blood and Cancer
Volume: 70
Issue: 2
ISSN: 1545-5009
Publisher: Wiley Periodicals, Inc  
Date Published: 2023-02-01
Start Page: e30075
Language: English
DOI: 10.1002/pbc.30075
PUBMED: 36349892
PROVIDER: scopus
DOI/URL:

https://www.scopus.com/inward/record.uri?eid=2-s2.0-85141724172&doi=10.1002%2fpbc.30075&partnerID=40&md5=4eada0f8d3eeba727a50eaf0798aa0fa
Notes: Article -- MSK corresponding author is Kim Kramer -- Export Date: 3 January 2023 -- Source: Scopus
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MSK Authors
  1. Brian Kushner
    311 Kushner
  2. Nai-Kong Cheung
    648 Cheung
  3. Kim Kramer
    236 Kramer
  4. Shakeel Modak
    249 Modak
  5. Mark Musa Souweidane
    219 Souweidane
  6. Neeta Pandit-Taskar
    342 Pandit-Taskar
  7. Ellen Marlese Basu
    101 Basu
  8. Dana Casey
    55 Casey
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