Abstract
Abstract OBJECTIVE In a cohort of patients who were treated with resection and adjuvant radiotherapy (RT) for adamantinomatous craniopharyngioma (ACP), we explored whether gross tumor volume (GTV) at the initiation of RT was associated with the risk of progressive disease (PD) following treatment. METHODS Pediatric and adolescent patients who received surgery and RT for ACP at a single institution from 1998-2021 were identified. Univariable Cox regression analyses (UVA) were performed to assess the association between pre-RT GTV and PD after RT. Multivariable analyses (MVA) were used to control for potential confounders. Two different endpoints were used to define PD. The first definition was based on radiographic tumor growth and the second definition was the requirement for an additional tumor-directed intervention following the completion of RT. RESULTS 48 patients were eligible for inclusion. The median age at diagnosis was 7.9 years (range: 2.1-17.4). All patients were treated with surgery and RT with median dose of 52.2 Gy (range: 45-55.8 Gy) and median GTV of 9.86 cm3 (range 0.7-117.7 cm3). Upon follow-up, 8 patients experienced PD based on both definitions. Five-year EFS was 85.4% (95% CI: 74.1 -98.3%). On both UVA and MVA, GTV was significantly associated with a small increased likelihood of PD (HR 1.02, 95% CI 1.00 - 1.04, p=0.022). However, after exclusion of a single outlier with GTV of 117.7 cm3 prior to RT, a second analysis identified no significant association between GTV and PD (UVA: HR 1.03, p=0.4; MVA: HR 1.06, p=0.2). CONCLUSIONS For most patients with ACP, the GTV at the initiation of RT is not associated with the risk of PD. This finding suggests that aggressive tumor debulking for the purpose of improving the efficacy of RT may not be necessary. For giant ACPs, however, novel strategies may be necessary for tumor control.
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