Childhood’s gliosarcomas: pathological and therapeutical considerations on three cases and critical review of the literature

Salvati, Maurizio; Lenzi, Jacopo; Brogna, Christian; Frati, Alessandro; Piccirilli, Manolo; Giangaspero, Felice; Raco, Antonino

doi:10.1007/s000381-006-0057-z

Cited by 10 publications

(13 citation statements)

References 24 publications

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“…3,9,25 Previously, small series and a large population database study have demonstrated improved survival with adjuvant radiotherapy in patients with GS. 10,13,[20][21][22]24,30 Although our analysis is retrospective, it adds to the existing body of literature and suggests that the addition of chemotherapy to radiotherapy following tumor excision further improves survival.…”

Section: Discussionmentioning

confidence: 96%

Patterns of care and outcomes in gliosarcoma: an analysis of the National Cancer Database

Frandsen¹,

Orton

Jensen

et al. 2018

Journal of Neurosurgery

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OBJECTIVE The authors compared presenting characteristics and survival for patients with gliosarcoma (GS) and glioblastoma (GBM). Additionally, they performed a survival analysis for patients who underwent GS treatments with the hypothesis that trimodality therapy (surgery followed by radiation and chemotherapy) would be superior to nontrimodality therapy (surgery alone or surgery followed by chemotherapy or radiation). METHODS Adults diagnosed with GS and GBM between the years 2004 and 2013 were queried from the National Cancer Database. Chi-square analysis was used to compare presenting characteristics. Kaplan-Meier, Cox regression, and propensity score analyses were employed for survival analyses. RESULTS In total, data from 1102 patients with GS and 36,658 patients with GBM were analyzed. Gliosarcoma had an increased rate of gross-total resection (GTR) compared with GBM (19% vs 15%, p < 0.001). Survival was not different for patients with GBM (p = 0.068) compared with those with GS. After propensity score analysis for GS, patients receiving trimodality therapy (surgery followed by radiation and chemotherapy) had improved survival (12.9 months) compared with those not receiving trimodality therapy (5.5 months). In multivariate analysis, GTR, female sex, fewer comorbidities, trimodality therapy, and age < 65 years were associated with improved survival. There was a trend toward improved survival with MGMT promoter methylation (p = 0.117). CONCLUSIONS In this large registry study, there was no difference in survival in patients with GBM compared with GS. Among GS patients, trimodality therapy significantly improved survival compared with nontrimodality therapy. Gross-total resection also improved survival, and there was a trend toward increased survival with MGMT promoter methylation in GS. The major potential confounder in this study is that patients with poor functional status may not have received aggressive radiation or chemotherapy treatments, leading to the observed outcome. This study should be considered hypothesis-generating; however, due to its rarity, conducting a clinical trial with GS patients alone may prove difficult.

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Section: Discussionmentioning

confidence: 96%

Patterns of care and outcomes in gliosarcoma: an analysis of the National Cancer Database

Frandsen¹,

Orton

Jensen

et al. 2018

Journal of Neurosurgery

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“…It usually occurs in elderly between 5 th and 6 th decades of life and extremely rare in paediatric population. Okami et al[ 5 ] reported eight cases of paediatric gliosarcoma, Sarkar et al[ 6 ] described three cases and Salvati et al[ 7 ] also reported three cases. Malde et al[ 8 ] reported one case of radiation induced gliosarcoma in literature.…”

Section: Discussionmentioning

confidence: 99%

“…Temporal lobe is most commonly involved followed by parietal and frontal lobe and can metastasize to extracranial structures like spinal cord, lung, pleura, liver, bone marrow and lymph nodes with increased frequency as compared to gliobastomas[ 7 ]. Pathogenesis of gliosarcoma is controversial topic and various studies have been proposed.…”

Section: Discussionmentioning

confidence: 99%

Gliosarcoma: A rare variant of glioblastoma multiforme in paediatric patient: Case report and review of literature

Meena¹,

Sharma²,

Chopra³

et al. 2016

WJCC

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Gliosarcoma is rare central nervous system tumour and a variant of glioblastoma multiforme with bimorphic histological pattern of glial and sarcomatous differentiation. It occurs in elderly between 5th and 6th decades of life and extremely rare in children. It is highly aggressive tumour and managed like glioblastoma multiforme. A 12-year-old female child presented with complaints of headache and vomiting from 15 d and blurring of vision from 3 d. Magnetic resonance imaging of brain shows heterogeneous mass in right parieto-occipital cortex. A right parieto-occipito-temporal craniotomy with complete excision of mass revealed a primary glioblastoma on histopathological investigation. Treatment consists of maximum surgical excision followed by adjuvant radiotherapy. The etiopathogenesis, treatment modalities and prognosis is discussed. The available literature is also reviewed.

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“…Newly diagnosed pediatric GS typically occurs in cerebral hemispheres and is treated with total surgical resection, standard RT, followed by chemotherapy 3,4. Survival after total surgical resection without RT and chemotherapy is usually <6 months, but OS after RT and chemotherapy can extend to slightly over 2 years,3,4 making this case study’s report of OS and PFS of >13 years of significant clinical interest.…”

Section: Discussionmentioning

confidence: 87%

“…Survival after total surgical resection without RT and chemotherapy is usually <6 months, but OS after RT and chemotherapy can extend to slightly over 2 years,3,4 making this case study’s report of OS and PFS of >13 years of significant clinical interest. The benefit of TMZ has not yet been established 11.…”

Section: Discussionmentioning

confidence: 89%

Long-term Survival (>13 Years) in a Child With Recurrent Diffuse Pontine Gliosarcoma

Burzynski

Janicki²,

Burzynski³

et al. 2014

Journal of Pediatric Hematology/Oncology

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Pediatric gliosarcoma (GS) is a rare variant of glioblastoma multiforme. The authors describe the case of an unusual pontine location of GS in a 9-year-old boy who was initially diagnosed with low-grade astrocytoma (LGA) that was successfully controlled for 4 years. Subsequently, his brain tumor transformed into a GS. Prior treatment of his LGA included subtotal tumor resection 3 times, standard radiation therapy, and Gamma Knife procedure twice. His LGA was also treated with a standard chemotherapy regimen of carboplatin and vincristine, and his GS with subtotal resection, high-dose cyclophosphamide, and thiotepa with stem cell rescue and temozolomide. Unfortunately, he developed disseminated disease with multiple lesions and leptomeningeal involvement including a tumor occupying 80% of the pons. Upon presentation at our clinic, he had rapidly progressing disease. He received treatment with antineoplastons (ANP) A10 and AS2-1 for 6 years and 10 months under special exception to our phase II protocol BT-22. During his treatment with ANP his tumor stabilized, then decreased, and, ultimately, did not show any metabolic activity. The patient’s response was evaluated by magnetic resonance imaging and positron emission tomography scans. His pathology diagnosis was confirmed by external neuropathologists, and his response to the treatment was determined by central radiology review. He experienced the following treatment-related, reversible toxicities with ANP: fatigue, xerostomia and urinary frequency (grade 1), diarrhea, incontinence and urine color change (grade 2), and grade 4 hypernatremia. His condition continued to improve after treatment with ANP and, currently, he complains only of residual neurological deficit from his previous surgery. He achieved a complete response, and his overall and progression-free survival is in excess of 13 years. This report indicates that it is possible to obtain long-term survival of a child with a highly aggressive recurrent GS with diffuse pontine involvement with a currently available investigational treatment.

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Childhood’s gliosarcomas: pathological and therapeutical considerations on three cases and critical review of the literature

Cited by 10 publications

References 24 publications

Patterns of care and outcomes in gliosarcoma: an analysis of the National Cancer Database

Patterns of care and outcomes in gliosarcoma: an analysis of the National Cancer Database

Gliosarcoma: A rare variant of glioblastoma multiforme in paediatric patient: Case report and review of literature

Long-term Survival (>13 Years) in a Child With Recurrent Diffuse Pontine Gliosarcoma

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