A comparative analysis of clinicopathological features and survival among early adolescents/young adults and children with low-grade glioma: a report from the Children’s Oncology Group

Margol, Ashley; Yeo, Kee Kiat; Xia, Chengcai; Onar, Arzu; Robison, Nathan; Freyer, David R.; Dhall, Girish

doi:10.1007/s11060-018-2983-5

Cited by 8 publications

(5 citation statements)

References 34 publications

(43 reference statements)

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“…In accordance with the reports of Fouladi et al 18 and Fisher et al, 12 most DG2 in our cohort were hemispheric tumors, while other series did not relate histology to site 11,19 . Correspondingly, seizures were among the most frequently reported presenting symptoms comparable to the observation in young adults 33 . Seizures related to better survival in adults 34 .…”

Section: Discussionsupporting

confidence: 92%

“…Although this was not investigated in our cohort directly, patients with DG2 of the cerebral hemispheres, but also of the cerebellum and spinal cord, had a better OS than those with tumors in the supratentorial midline and brainstem. A higher rate of complete/subtotal resection is accepted as the major contributing factor to better PFS and/or OS for all pediatric LGG 4,5,12,13,33 and had been achieved for 48% to 70% for hemispheric, cerebellar and spinal tumors in this cohort. In addition to site, tumor size and the infiltrative nature of DG2 contribute to resectability.…”

Section: Discussionmentioning

confidence: 80%

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Prognostic impact of distinct genetic entities in pediatric diffuse glioma WHO‐grade II—Report from the German/Swiss SIOP‐LGG 2004 cohort

Falkenstein

Gessi

Kandels

et al. 2020

Intl Journal of Cancer

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Reports on pediatric low‐grade diffuse glioma WHO‐grade II (DG2) suggest an impaired survival rate, but lack conclusive results for genetically defined DG2‐entities. We analyzed the natural history, treatment and prognosis of DG2 and investigated which genetically defined sub‐entities proved unfavorable for survival. Within the prospectively registered, population‐based German/Swiss SIOP‐LGG 2004 cohort 100 patients (age 0.8‐17.8 years, 4% neurofibromatosis [NF1]) were diagnosed with a DG2. Following biopsy (41%) or variable extent of resection (59%), 65 patients received no adjuvant treatment. Radiologic progression or severe neurologic symptoms prompted chemotherapy (n = 18) or radiotherapy (n = 17). Multiple lines of salvage treatment were necessary for 19/35 patients. Five years event‐free survival dropped to 0.44, while 5 years overall survival was 0.90 (median observation time 8.3 years). Extensive genetic profiling of 65/100 DG2 identified Histone3‐K27M‐mutation in 4, IDH1‐mutation in 11, BRAF‐V600‐mutation in 12, KIAA1549‐BRAF‐fusions in 6 patients, while the remaining 32 tumor tissues did not show alterations of these genes. Progression to malignant glioma occurred in 12 cases of all genetically defined subgroups within a range of 0.5 to 10.8 years, except for tumors carrying KIAA1549‐BRAF‐fusions. Histone3‐K27M‐mutant tumors proved uniformly fatal within 0.6 to 2.4 years. The current LGG treatment strategy seems appropriate for all DG2‐entities, with the exemption of Histone3‐K27M‐mutant tumors that require a HGG‐related treatment strategy. Our data confirm the importance to genetically define pediatric low‐grade diffuse gliomas for proper treatment decisions and risk assessment.

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Section: Discussionsupporting

confidence: 92%

Section: Discussionmentioning

confidence: 80%

Prognostic impact of distinct genetic entities in pediatric diffuse glioma WHO‐grade II—Report from the German/Swiss SIOP‐LGG 2004 cohort

Falkenstein

Gessi

Kandels

et al. 2020

Intl Journal of Cancer

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“…Long‐term survival rates of 65%‐85% vary by location, histology, and degree of resection 2,3 . The majority of pediatric LGG are pilocytic astrocytoma (75% of patients on CCG9891/POG9130), which have been shown to be independently associated with improved outcomes compared with other histologies 4 . Tuberous sclerosis and neurofibromatosis type‐1 are autosomal‐dominant inherited predispositions for LGG.…”

Section: Gliomasmentioning

confidence: 99%

Gliomas, germ cell tumors, and craniopharyngioma

Aridgides

Janssens

Braunstein

et al. 2020

Pediatric Blood & Cancer

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This report summarizes the current multimodality treatment approaches for children with low‐ and high‐grade gliomas, germinoma, and nongerminomatous germ cell tumors, and craniopharyngiomas used in the Children's Oncology Group (COG) and the International Society of Pediatric Oncology (SIOP). Treatment recommendations are provided in the context of historical approaches regarding the roles of surgery, radiation, and chemotherapy. Future research strategies for these tumors in both COG and SIOP are also discussed.

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“…11 Limitation of this study is the investigated time period from 1991 to 1996 in which today's follow-up imaging techniques using high-field (1.5-or 3.0-T) MRI to assess EoR or tumor progression were not available. Furthermore, a more recent retrospective volumetric study by Margol et al 36 analyzed the progression-free survival and the overall survival in 468 children (age 0-14 years) and 50 early adolescents (15-21 years) after low-grade glioma surgery without iMRI assistance. Rate of GTR in this series investigating lowgrade glioma surgery without iMRI guidance was 63.9% in the children group, whereas 66.0% of the early adolescent group had a GTR of the tumor in the postoperative MRI.…”

Section: Rate Of Gtr In Low-grade Gliomas (Who Grades I and Ii)mentioning

confidence: 99%

Intraoperative MRI-guided Resection in Pediatric Brain Tumor Surgery: A Meta-analysis of Extent of Resection and Safety Outcomes

Wach

Banat

Borger

et al. 2020

J Neurol Surg A Cent Eur Neurosurg

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Background The objective of this meta-analysis was to analyze the impact of intraoperative magnetic resonance imaging (iMRI) on pediatric brain tumor surgery with regard to the frequency of histopathologic entities, additional resections secondary to iMRI, rate of gross total resections (GTR) in glioma surgery, extent of resection (EoR) in supra- and infratentorial compartment, surgical site infections (SSIs), and neurologic outcome after surgery. Methods MEDLINE/PubMed Service was searched for the terms “intraoperative MRI,” “pediatric,” “brain,” “tumor,” “glioma,” and “surgery.” The review produced 126 potential publications; 11 fulfilled the inclusion criteria, including 584 patients treated with iMRI-guided resections. Studies reporting about patients <18 years, setup of iMRI, surgical workflow, and extent of resection of iMRI-guided glioma resections were included. Results IMRI-guided surgery is mainly used for pediatric low-grade gliomas. The mean rate of GTR in low- and high-grade gliomas was 78.5% (207/254; 95% confidence interval [CI]: 64.6–89.7, p < 0.001). The mean rate of GTR in iMRI-assisted low-grade glioma surgery was 74.3% (35/47; 95% CI: 61.1–85.5, p = 0.759). The rate of SSI in surgery assisted by iMRI was 1.6% (6/482; 95% CI: 0.7–2.9). New onset of transient postoperative neurologic deficits were observed in 37 (33.0%) of 112 patients. Conclusion IMRI-guided surgery seems to improve the EoR in pediatric glioma surgery. The rate of SSI and the frequency of new neurologic deficits after IMRI-guided surgery are within the normal range of pediatric neuro-oncologic surgery.

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A comparative analysis of clinicopathological features and survival among early adolescents/young adults and children with low-grade glioma: a report from the Children’s Oncology Group

Abstract: Unlike several other cancers, LGG has similar presenting features and survival for both eAYAs and children. This support continuing a unified treatment approach and enrollment of eAYAs in pediatric clinical trials for LGGs.

Cited by 8 publications

References 34 publications

Prognostic impact of distinct genetic entities in pediatric diffuse glioma WHO‐grade II—Report from the German/Swiss SIOP‐LGG 2004 cohort

Prognostic impact of distinct genetic entities in pediatric diffuse glioma WHO‐grade II—Report from the German/Swiss SIOP‐LGG 2004 cohort

Gliomas, germ cell tumors, and craniopharyngioma

Intraoperative MRI-guided Resection in Pediatric Brain Tumor Surgery: A Meta-analysis of Extent of Resection and Safety Outcomes

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