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Outcomes in children with central nervous system tumors disseminated at presentation: a large single-center
Matthew A Kirkman1,2, Richard Hayward1, Kim Phipps1
1Department of Neurosurgery, Great Ormond Street Hospital for Children NHS Trust, London, WC1N 3JH, UK.
Insights
Disseminated central nervous system (CNS) tumors in children significantly impact survival and recurrence risk compared to solitary tumors. Early disease spread at diagnosis negatively affects outcomes for pediatric CNS malignancies.
Area of Science:
- Pediatric neuro-oncology
- Central nervous system (CNS) tumors
- Cancer dissemination
Background:
- Disseminated central nervous system (CNS) tumors in children are associated with poorer prognoses than localized disease.
- Systematic evaluation of disease dissemination at initial presentation across different pediatric CNS tumor types is lacking.
- Understanding the impact of initial disease spread is crucial for optimizing management strategies.
Purpose of the Study:
- To evaluate the impact of tumor dissemination at initial presentation on management and clinical outcomes in pediatric CNS tumors.
- To compare outcomes between children with disseminated versus solitary CNS tumors.
- To analyze the association between dissemination status, tumor type, and treatment modalities with patient outcomes.
Main Methods:
- Retrospective review of 361 pediatric patients with CNS tumors (medulloblastoma, PNET, AT/RT, pilocytic astrocytoma, ependymoma) treated between 2003-2016.
- Data analysis included univariate and multivariate assessments of overall survival, event-free survival, and time to recurrence.
- Comparison of outcomes based on disease dissemination status at presentation.
Main Results:
- Tumor type and dissemination status at presentation significantly influenced overall survival, risk of recurrence, and event-free survival.
- Subtotal resection correlated with shorter time to recurrence and worse event-free survival.
- Chemotherapy and radiotherapy were associated with improved overall and event-free survival.
Conclusions:
- Disease dissemination status at initial presentation is a significant prognostic factor for pediatric CNS tumors.
- Treatment modalities like chemotherapy and radiotherapy improve survival outcomes.
- Further research is needed to refine treatment strategies for disseminated pediatric CNS malignancies.
Purpose:
Children with disseminated central nervous system (CNS) tumors have worse outcomes than those with solitary disease, but outcomes of disease dissemination at initial presentation have not been systematically studied and compared across tumor groups to date. We evaluated the impact of tumor dissemination at presentation on management and clinical outcomes in a cohort of consecutively treated children in a single neurosurgical unit over a 14-year period.
Methods:
Method used was a retrospective review of data on children presenting to Great Ormond Street Hospital, London, UK, with medulloblastoma, primitive neuroectodermal tumor, atypical teratoid rhabdoid tumor, pilocytic astrocytoma, and ependymoma between 2003 and 2016 inclusive. Uni- and multi-variate analyses were performed to evaluate a range of outcome measures.
Results:
Three-hundred sixty-one children were identified in total, 53 with disease dissemination at presentation (M:F = 34:19, median age = 3.8 years, range = 7 days-15.6 years) and 308 with solitary tumors (M:F = 161:147, median age = 5.8 years, range = 1 day-16.9 years). Median follow-up was similar irrespective of dissemination status (disseminated tumor 64.0 months, range = 5.2-152.0 months; solitary tumor 74.5 months, range = 4.7-170.1 months; P > 0.05). In multivariate analyses, tumor type and dissemination status at presentation were significantly associated with overall survival (P < 0.0001), risk of recurrence/disease progression (P < 0.01), and event-free survival (P < 0.0001). Subtotal resection was associated with shorter time to recurrence/disease progression (P < 0.01) and worse event-free (P < 0.0001) but not overall survival, whereas treatment with chemotherapy and radiotherapy were associated with improved overall (Ps < 0.0001) and event-free survival (Ps < 0.05). Differences between tumor groups were evident.
Conclusions:
Dissemination status at initial presentation significantly affects outcomes in children with CNS tumors.
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