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Effect of early radiotherapy initiation and high-dose chemotherapy on the prognosis of pediatric atypical teratoid rhabdoid tumors in different age groups

  • Clinical Study
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Abstract

Purpose

The optimal treatment strategy for pediatric atypical teratoid rhabdoid tumor (ATRT) is inconclusive. This study evaluated the prognostic value of early radiotherapy (RT) and high-dose chemotherapy with autologous stem cell rescue (HDC/ASCR) in pediatric ATRT.

Methods

This pooled analysis included ATRT patients treated at our institution and from other studies who were identified by a search of the PubMed electronic database. The effect of patient demographics and treatment profiles on progression-free survival (PFS) and overall survival (OS) were analyzed using Cox regression.

Results

Overall, 34 patients from our institution and 436 patients from 35 published studies were included. In multivariable analysis, patients with gross total resection (GTR), early RT (time to RT interval < 2 months), and HDC/ASCR had both better PFS [hazard ratio (HR) 0.46, p\(<\) 0.001; HR 0.64, p = 0.011; and HR 0.51, p = 0.005, respectively] and OS (HR 0.55, p = 0.002; HR 0.48, p = 0.004; and HR 0.42, p < 0.001, respectively). For patients aged < 3 years, both RT and HDC/ASCR were significant favorable factors for PFS (HR 0.32 and 0.46, respectively) and OS (HR 0.40 and 0.36, respectively), while early RT was not prognostic. For patients aged ≥ 3 years, early RT was significantly associated with better PFS (HR 0.51) and HDC/ASCR did not affect PFS, and neither was related to OS.

Conclusion

Both early RT initiation and HDC/ASCR were important components in the treatment of pediatric ATRT. However, the optimal treatment strategies might differ by age.

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Author information

Authors and Affiliations

  1. Division of Radiation Oncology, Department of Oncology, Taipei Veterans General Hospital, No. 201, Sec. 2, Shipai Road, Beitou District, Taipei, 112, Taiwan (ROC)

    Wan-Chin Yang, Yu-Wen Hu & Yi-Wei Chen

  2. School of Medicine, National Yang-Ming University No, 155, Sec.2, Linong Street, Taipei, 112, Taiwan (ROC)

    Wan-Chin Yang, Hsiu-Ju Yen, Muh-Lii Liang, Hsin-Hung Chen, Yi-Yen Lee, Feng-Chi Chang, Shih-Chieh Lin, Yu-Wen Hu & Yi-Wei Chen

  3. Division of Pediatric Hematology and Oncology, Department of Pediatrics, Taipei Veterans General Hospital, No. 201, Sec. 2, Shipai Road, Beitou District, Taipei, 112, Taiwan (ROC)

    Hsiu-Ju Yen

  4. Division of Pediatric Neurosurgery, The Neurological Institute, Taipei Veterans General Hospital, No. 201, Sec. 2, Shipai Road, Beitou District, Taipei, 112, Taiwan (ROC)

    Muh-Lii Liang, Hsin-Hung Chen & Yi-Yen Lee

  5. Department of Radiology, Taipei Veterans General Hospital, No. 201, Sec. 2, Shipai Road, Beitou District, Taipei, 112, Taiwan (ROC)

    Feng-Chi Chang

  6. Department of Pathology and Laboratory Medicine, Taipei Veterans General Hospital, No. 201, Sec. 2, Shipai Road, Beitou District, Taipei, Taiwan (ROC)

    Shih-Chieh Lin

  7. Department of Neurosurgery, Taipei Medical University Hospital, No.252, Wuxing St, Xinyi District, Taipei, Taiwan (ROC)

    Tai-Tong Wong

  8. Institute of Public Health, National Yang-Ming University, No.155, Sec.2, Linong Street, Taipei, 112, Taiwan (ROC)

    Yu-Wen Hu

Authors
  1. Wan-Chin Yang
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  2. Hsiu-Ju Yen
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  3. Muh-Lii Liang
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  4. Hsin-Hung Chen
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  5. Yi-Yen Lee
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  6. Feng-Chi Chang
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  7. Shih-Chieh Lin
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  8. Tai-Tong Wong
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  9. Yu-Wen Hu
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  10. Yi-Wei Chen
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Corresponding authors

Correspondence to Yu-Wen Hu or Yi-Wei Chen.

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The authors declare that they have no conflict of interest.

Ethical approval

The data of this study was extracted from database of pediatric brain tumors in Taipei Veterans General Hospital approved by Institutional Review Board of the Taipei Veterans General Hospital. The IRB number is 97-07-05A.

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Informed consent to this study was obtained from the parents of all patients.

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Yang, WC., Yen, HJ., Liang, ML. et al. Effect of early radiotherapy initiation and high-dose chemotherapy on the prognosis of pediatric atypical teratoid rhabdoid tumors in different age groups. J Neurooncol 147, 619–631 (2020). https://doi.org/10.1007/s11060-020-03456-1

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  • DOI: https://doi.org/10.1007/s11060-020-03456-1

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