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Multi-institutional Characterization of Outcomes for Pediatric and Young Adult Patients With High-Risk Myxopapillary Ependymoma After Radiation Therapy.
Liu, Kevin X; Indelicato, Daniel J; Paulino, Arnold C; Looi, Wen S; Catalano, Paul J; Chintagumpala, Murali M; Gallotto, Sara L; Marcus, Karen J; Haas-Kogan, Daphne A; Tarbell, Nancy J; MacDonald, Shannon M; Mahajan, Anita; Yock, Torunn I.
Affiliation
  • Liu KX; Department of Radiation Oncology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts; Department of Radiation Oncology, Brigham and Women's Hospital, Dana-Farber Cancer Institute, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts.
  • Indelicato DJ; Department of Radiation Oncology, University of Florida College of Medicine, Jacksonville, Florida.
  • Paulino AC; Department of Radiation Oncology, MD Anderson Cancer Center, Houston, Texas.
  • Looi WS; Department of Radiation Oncology, University of Florida College of Medicine, Jacksonville, Florida.
  • Catalano PJ; Department of Biostatistics, Harvard T.H. Chan School of Public Health, Boston, Massachusetts; Department of Biostatistics and Computational Biology, Dana-Farber Cancer Institute, Boston, Massachusetts.
  • Chintagumpala MM; Department of Pediatrics, Division of Hematology-Oncology, Texas Children's Cancer Center, Baylor College of Medicine, Houston, Texas.
  • Gallotto SL; Department of Radiation Oncology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts.
  • Marcus KJ; Department of Radiation Oncology, Brigham and Women's Hospital, Dana-Farber Cancer Institute, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts.
  • Haas-Kogan DA; Department of Radiation Oncology, Brigham and Women's Hospital, Dana-Farber Cancer Institute, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts.
  • Tarbell NJ; Department of Radiation Oncology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts; Inspire Exercise Medicine, Naples, Florida.
  • MacDonald SM; Department of Radiation Oncology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts.
  • Mahajan A; Department of Radiation Oncology, Mayo Clinic, Rochester, Minnesota.
  • Yock TI; Department of Radiation Oncology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts. Electronic address: tyock@mgh.harvard.edu.
Int J Radiat Oncol Biol Phys ; 117(5): 1174-1180, 2023 Dec 01.
Article in En | MEDLINE | ID: mdl-37437812
ABSTRACT

PURPOSE:

Myxopapillary ependymoma (MPE) is a rare, typically slow-growing subtype of spinal ependymomas. There are no standard guidelines for radiotherapy and long-term outcomes after radiation, particularly patterns of relapse, for pediatric and young adult (YA) patients with MPE remain under-characterized. METHODS AND MATERIALS This is an Institutional Review Board-approved multi-institutional retrospective cohort study of 60 pediatric and YA patients diagnosed with MPE and received radiotherapy between 2000-2020. Clinical and treatment characteristics, and long-term outcomes were recorded. Site(s) of progression was compared to radiation fields. Survival outcomes were analyzed using Kaplan-Meier method. Cumulative incidence of local in-field progression (CILP) after initial radiotherapy was analyzed using Gray's method with out-of-field-only progression as a competing risk. Univariate analyses were performed using Cox proportional hazard's model.

RESULTS:

The median age at radiation was 14.8 years (range 7.1-26.5). At time of radiotherapy, 45 (75.0%) and 35 (58.3%) patients had gross residual and multifocal disease, respectively. Forty-eight (80.0%), seven (11.7%) and five (8.3%) patients received involved field radiotherapy, craniospinal irradiation, and whole spine radiation, respectively. Median follow-up from end of radiotherapy was 6.2 years (range 0.6-21.0). Five-year overall survival, progression-free survival, and CILP were 100%, 60.8% and 4.1%, respectively. Both local recurrences were at sites of gross residual disease. Of the eighteen out-of-field first recurrences after radiotherapy, all were superior to the initial treatment field and nine had intracranial relapse. On univariate analyses, distant-only recurrence before radiation (HR 4.00, 95% CI 1.54-10.43, p = 0.005) was significantly associated with shorter time to progression.

CONCLUSIONS:

While the risk of recurrence within the radiation field is low, pediatric and YA patients with high-risk MPE remain at risk for recurrences in the spine above the radiation field and intracranially after radiotherapy. Future prospective studies are needed to investigate the appropriate radiation field and dose based on the extent of metastases.
Subject(s)

Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Spinal Cord Neoplasms / Ependymoma Type of study: Etiology_studies / Guideline / Observational_studies / Prognostic_studies / Risk_factors_studies Limits: Adolescent / Adult / Child / Humans Language: En Journal: Int J Radiat Oncol Biol Phys Year: 2023 Document type: Article

Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Spinal Cord Neoplasms / Ependymoma Type of study: Etiology_studies / Guideline / Observational_studies / Prognostic_studies / Risk_factors_studies Limits: Adolescent / Adult / Child / Humans Language: En Journal: Int J Radiat Oncol Biol Phys Year: 2023 Document type: Article