Pediatric Diencephalic Gliomas – A Review of 18 Cases

Ew, Scott; Jp, Mickle

doi:10.1159/000120335

Cited by 18 publications

(13 citation statements)

References 23 publications

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“…The proportion of low-grade tumors in this series (67%) approximates that observed for pediatric glial tumors in all locations [20,21] and in prior series describing pediatric thalamic gliomas [2,22]. The 4-year PFS among patients with low-grade tumors was 36 B 12 compared to 0% among patients with highgrade tumors.…”

Section: Discussionsupporting

confidence: 68%

Bithalamic Involvement Predicts Poor Outcome among Children with Thalamic Glial Tumors

et al. 1998

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Clinical features and treatment of 36 consecutive pediatric patients with thalamic glial tumors confirmed by histology and characterized by neuroimaging were reviewed to identify prognostic factors. The median age at diagnosis was 10 years (range 1–18 years). Twenty-four patients had low-grade tumors (juvenile pilocytic astrocytoma n = 9, fibrillary astrocytoma n = 6, astrocytomas not otherwise specified n = 6, ganglioglioma n = 2 and oligodendroglioma n = 1) and 12 patients had high-grade tumors (glioblastoma multiforme n = 7, anaplastic astrocytoma n = 4 and unclassified malignant tumor n = 1). With a median follow-up of 4.3 years among survivors, estimates of 4-year progression-free survival (PFS) and overall survival (OS) for the entire group are 28 ± 10 and 37 ± 10%, respectively. Low-grade tumors were associated with a significantly better 4-year PFS (36 ± 12 vs. 0% for the high-grade group; p = 0.03) and OS (52 ± 12 vs. 0%; p < 0.001). This review identified that bithalamic involvement, characterized by neuroimaging, exerted an independent and significant negative impact on PFS and OS for patients with low-grade tumors. Estimates of 4-year PFS and OS among patients with low-grade bithalamic versus monothalamic tumors were 58 ± 15 vs. 0% and 85 ± 11 vs. 0% (p < 0.00001), respectively. The presence of bithalamic involvement did not affect outcome among patients with high-grade tumors. Additionally, age at diagnosis, enhancement with neuroimaging contrast, extension beyond the thalamus and extent of surgical resection did not correlate with overall outcome. Because treatment approaches varied during the study period, the impact of radiation therapy or chemotherapy could not be assessed. This contemporary, single-institution series of pediatric thalamic glial tumors demonstrates, for the first time, the statistical significance of bithalamic involvement as a marker of poor prognosis among patients with low-grade glial lesions.

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Section: Discussionsupporting

confidence: 68%

Bithalamic Involvement Predicts Poor Outcome among Children with Thalamic Glial Tumors

et al. 1998

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“…Even so, one must consider the potential long term toxicities of such treatment, as this group of patients may enjoy survivals of as high a 100-76% at 5-13 years, respectively [9,[21][22][23], We chose not to rely on nitroso urea-based regimens due to their prolonged myelosuppression, and their association with late pulmonary fibro sis [24,25]. Similarly, we feel the nephrotoxicity and oto toxicity associated with cisplatin should be weighed care fully with respect to the quality of life in patients with the potential for long-term survival [26], Likewise, the re peatedly observed association of MOPP chemotherapy with acute leukemia in patients with CNS and other tumors is of concern [27], The least toxic of the effective alternative chemotherapy regimens discussed above may be those containing cyclophosphamide., actinomycin D, and the newer platinum analogue, carboplatin.…”

Section: Discussionmentioning

confidence: 99%

Response of Pediatric Low Grade Gliomas to Chemotherapy

et al. 1993

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Thirteen patients with low grade astrocytomas were treated with alkylating agent or platinum-based chemotherapy regimens. Eleven of these patients received chemotherapy as their initial postoperative treatment modality, and 2 others as treatment for progessive disease postradiation therapy. Responses were objectively determined using CT or MRI. One patient had a complete response (CR), 6 had partial responses (PR), and 3 others had stable disease (SD) as their best response. Clinical responses paralleled those determined objectively. Chemotherapy was well tolerated with the exception of ototoxic-ity in 4 patients treated with cisplatin. Despite good initial responses, 5 of 6 patients who received no further treatment postchemotherapy (4 PR, 1 SD) developed progressive disease 5–13 months after completing chemotherapy. The remaining 4 patients with objective responses or stable disease (1 CR, 2 PR, 1 SD), all received further postchemotherapy treatment with radiation therapy or surgery, and have not demonstrated disease progression. Our experience suggests that alkylator or platinum-based chemotherapy can successfully delay the growth of these locally infiltrative neoplasms, and should be considered in their primary management. Despite the experience of others, further therapy appears necessary in patients with residual CT or MRI abnormalities postchemotherapy.

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“…For patients whose tumors are amenable to a gross total surgical resection, the 10-year survival is greater than 90% (Campbell and Pollack, 1996;Pollack et al, 1995). When a gross total resection (GTR) cannot be accomplished at least initially, there is a less favorable 10-year survival, varying from 50% to 86% (Campbell and Pollack, 1996;Scott and Mickle, 1987;Woo et al, 1988). Approximately 5% of LGNs behave aggressively, as manifested by a higher rate of localized recurrence or a tendency to metastasize.…”

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confidence: 99%

Leptomeningeal dissemination at diagnosis of pediatric low-grade neuroepithelial tumors

Hukin¹,

Siffert²,

Cohen³

et al. 2003

Neuro-Oncology

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The goal of this study was to describe the demographic, histologic, and prognostic features of children with low-grade neuroepithelial tumors (LGN) of the CNS presenting with leptomeningeal metastases (LM) at diagnosis. We identified 528 newly diagnosed LGN children, 13 (3%) of whom had LM at diagnosis. LM was defined by neuroimaging, clinical evidence, and/or biopsy. The charts were reviewed and patients contacted to validate the demographic data, treatment, and clinical status. The distribution of LM patients by primary tumor site was diencephalon, 5; cerebrum, 2; spinal cord, 3; brainstem, 2; and cerebellum, 1. Six of 8 patients with LM had durable objective responses to chemotherapy. The 5-year progression-free survival of patients with LM at diagnosis was 17%, compared to 85% (95% CI, 80%-91%) for those with localized LGN who had a gross total resection and 51% (95% CI, 44%-52%) for those with localized LGN who had less aggressive surgery ( P < 0.0001). Only 1 of these 13 LM patients died. The 5-year overall survival of the localized LGN group with a gross total resection was 97% (95% CI, 92%-99.9%), and that of the localized LGN group with less aggressive surgery was 88% (95% CI, 84%-95%) ( P = 0.004). The 3% frequency of LM at diagnosis is likely an underestimate since patients with newly diagnosed LGN were not routinely staged. We suggest that staging be considered in the following circumstances: diencephalic primary site, unexplained hydrocephalus, clinical features suggestive of LM, and before adjuvant therapy is initiated. The prognosis for children with LM at diagnosis is favorable, and its identification alters therapeutic strategies.

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Pediatric Diencephalic Gliomas – A Review of 18 Cases

Cited by 18 publications

References 23 publications

Bithalamic Involvement Predicts Poor Outcome among Children with Thalamic Glial Tumors

Bithalamic Involvement Predicts Poor Outcome among Children with Thalamic Glial Tumors

Response of Pediatric Low Grade Gliomas to Chemotherapy

Leptomeningeal dissemination at diagnosis of pediatric low-grade neuroepithelial tumors

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