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Craniospinal irradiation for treatment of metastatic pediatric low-grade glioma.

ABSTRACT: Patients with disseminated pediatric low-grade glioma (LGG) initially treated with chemotherapy frequently experience disease progression, with 5-year event-free survival (EFS) of <20% and 10-year overall survival (OS) of approximately 70%. This study aimed to describe outcomes of metastatic pediatric LGG treated with craniospinal irradiation (CSI). A retrospective study was performed of all patients with metastatic pediatric LGG treated with CSI at a single institution. EFS was defined as survival without disease progression or secondary high-grade glioma. Dates were counted from the first day of irradiation. We identified 12 eligible patients; all had histologically confirmed LGG. Metastatic disease was present at initial presentation in 9 patients. The median age at CSI was 9.3 years. The 5-year EFS and OS were 71% (95% CI 33.7-89.5) and 70% (95% CI 32.9-89.2), respectively. No deaths were observed among the patients who underwent subtotal resection (STR) before radiotherapy, whereas 3 patients who had undergone biopsy died (OS log-rank P = 0.01). EFS may be longer among patients who underwent STR before RT (EFS log-rank P = 0.03), with a hazard ratio for biopsy of 8.4 (vs. STR; 95% CI 0.8-84.0, P = 0.07). No patient experienced acute toxicity of grade 3 or higher. Patients with metastatic pediatric LGG treated with CSI experienced longer EFS than historical cohorts treated with chemotherapy alone, with similar OS. CSI may be considered in the management of metastatic pediatric LGG, particularly in older children experiencing progression after chemotherapy.

SUBMITTER: Tsang DS

PROVIDER: S-EPMC8719438 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:PurposeChildren treated for brain tumors with conformal radiation therapy experience preserved cognitive outcomes. Early evidence suggests that adaptive functions or independent-living skills may be spared. This longitudinal investigation prospectively examined intellectual and adaptive functioning during the first 5 years following irradiation for childhood craniopharyngioma and low-grade glioma (LGG). The effect of visual impairment on adaptive outcomes was investigated.Methods and materialsChildren with craniopharyngioma (n=62) and LGG (n=77) were treated using conformal or intensity modulated radiation therapy. The median age was 8.05 years (3.21-17.64 years) and 8.09 years (2.20-19.27 years), respectively. Serial cognitive evaluations including measures of intelligence quotient (IQ) and the Vineland Adaptive Behavior Scales (VABS) were conducted at preirradiation baseline, 6 months after treatment, and annually through 5 years. Five hundred eighty-eight evaluations were completed during the follow-up period.ResultsBaseline assessment revealed no deficits in IQ and VABS indices for children with craniopharyngioma, with significant (P<.05) longitudinal decline in VABS Communication and Socialization indices. Clinical factors associated with more rapid decline included females and preirradiation chemotherapy (interferon). The only change in VABS Daily Living Skills correlated with IQ change (r=0.34; P=.01) in children with craniopharyngioma. Children with LGG performed below population norms (P<.05) at baseline on VABS Communication, Daily Living Indices, and the Adaptive Behavior Composite, with significant (P<.05) longitudinal decline limited to VABS Communication. Older age at irradiation was a protective factor against longitudinal decline. Severe visual impairment did not independently correlate with poorer adaptive outcomes for either tumor group.ConclusionsThere was relative sparing of postirradiation functional outcomes over time in this sample. Baseline differences in functional abilities before the initiation of irradiation suggested that other factors influence functional outcomes above and beyond the effects of irradiation.

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Craniospinal irradiation for treatment of metastatic pediatric low-grade glioma.

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