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A case series of pediatric survivors of anaplastic pleomorphic xanthoastrocytoma. | LitMetric

A case series of pediatric survivors of anaplastic pleomorphic xanthoastrocytoma.

Neurooncol Adv

Division of Pediatric Hematology, Oncology and Bone Marrow Transplantation, British Columbia Children's Hospital, University of British Columbia, Vancouver, British Columbia, Canada.

Published: January 2021

AI Article Synopsis

  • Anaplastic pleomorphic xanthoastrocytoma (APXA) is a rare and aggressive brain tumor in children, often treated similarly to high-grade gliomas, but with unclear best treatment practices.* -
  • In a study of 3 pediatric cases, all patients underwent successful surgeries and were treated with radiation and chemotherapy, showing promising survival rates with no signs of disease over 4 years.* -
  • Genetic profiling indicated specific mutations and chromosomal changes, suggesting that understanding the tumor's genetic makeup could enhance future treatment strategies and prognosis.*

Article Abstract

Background: Anaplastic pleomorphic xanthoastrocytoma (APXA) is a rare subtype of CNS astrocytoma. They are generally treated as high-grade gliomas; however, uncertainty exists regarding the optimal therapy. Here, we report on 3 pediatric cases of APXA.

Methods: Our institutional database was queried for cases of APXA and 3 cases were identified. Surgical samples were processed for methylation profiling and chromosomal microarray analysis. Methylation data were uploaded to the online CNS tumor classifier to determine methylation-based diagnoses to determine copy number variations (CNVs).

Results: Two patients were male, 1 female, and all were aged 12 years at diagnosis. All underwent a gross total resection (GTR) and were diagnosed with an APXA. Immunohistochemical analysis demonstrated that 2 cases were BRAF V600E positive. Methylation-based tumor classification supported the APXA diagnosis in all cases. CNV analyses revealed homozygous deletions in all and chromosome 9p loss in 2 cases. All patients received radiation therapy (54 Gy in 30 fractions) with concurrent temozolomide. Two patients received maintenance chemotherapy with temozolomide and lomustine for 6 cycles as per the Children's Oncology Group ACNS0423. The third patient recurred and went on to receive a second GTR and 6 cycles of lomustine, vincristine, and procarbazine. All are alive with no evidence of disease >4 years post-treatment completion (overall survival = 100%, event free survival = 67%).

Conclusions: The natural history and optimal treatment of this rare pediatric tumor are not well understood. This case series supports the use of adjuvant chemoradiotherapy in the treatment of APXA. The genetic landscape may be informative for optimizing treatment and prognosis.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7849951PMC
http://dx.doi.org/10.1093/noajnl/vdaa176DOI Listing

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