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

J Neurooncol. 2020 May;147(3):619-631. doi: 10.1007/s11060-020-03456-1. Epub 2020 Mar 28.

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[Formula: see text] 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.

Keywords: ATRT; Age; Ethnicity; High-dose chemotherapy; Radiotherapy.

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Combined Modality Therapy
  • Female
  • Humans
  • Male
  • Prognosis
  • Rhabdoid Tumor / drug therapy*
  • Rhabdoid Tumor / radiotherapy*
  • Teratoma / drug therapy*
  • Teratoma / radiotherapy*
  • Treatment Outcome
  • Young Adult

Supplementary concepts

  • Typical Teratoid Rhabdoid Tumor