Gliosarcoma with Chromosome Abnormalities in a Neonate Exposed to Heptachlor

Chadduck, William M.; Gollin, Susanne M.; Gray, Brian A.; Norris, James S.; Araoz, C.; Tryka, A. Francine

doi:10.1227/00006123-198710000-00021

Cited by 19 publications

(5 citation statements)

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“…The number of patients included in each reviewed paper ranged from a single patient in a case report to 250 patients in a literature review 24,25 . These 44 papers yielded 468 cases of intracranial tumors in children 6 months of age and less 1,3–5,8–10,12,13,15,17–19,21,26,27,29–31,33–35,37,38,40–46,51–55,57–59,61,62,65–67 . There was significant variability in brain tumor histologies (Table 2).…”

Section: Resultsmentioning

confidence: 99%

Surgical Treatment of Brain Tumors in Infants Younger than Six Months of Age and Review of the Literature

et al. 2012

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Object Brain tumors are rare in infants under 6-months of age. These tumors can be challenging to treat surgically. We analyzed a modern series of patients treated by a multidisciplinary team at a tertiary care center and performed a literature review of this unique population. Methods Retrospective clinical data was collected for patients surgically treated for intracranial mass lesions at The Children’s Hospital of Philadelphia from 1998 to 2007. Dermoid cysts and other skull-based lesions were excluded from the analysis. Results Sixteen patients under 6-months of age underwent surgery for primary intracranial mass lesions. The median age of the patients at surgery was 5.2 months (range 1.4 to 6 months of age). Children most often presented with a bulging fontanelle, hydrocephalus, or macrocephaly (7 patients). Vomiting was seen in 5 patients, cranial nerve palsies in 1 patient, and seizures in 3 patients. All patients had tumor resections and post-operatively were monitored in the intensive care unit. The final pathology consisted of atypical teratoid/rhabdoid tumor (3 cases), primitive neuroectodermal tumor/medulloblastoma (3 cases), choroid plexus papilloma (2 cases), astrocytoma (2 cases), ganglioglioma (2 cases), desmoplastic infantile ganglioglioma (2), glioblastoma multiforme (1), and choroid plexus carcinoma (1). Two intra-operative deaths occurred. Of the surviving 14, a gross total resection was achieved in 4. Adjuvant therapy was determined by a multidisciplinary team composed of neuro-oncology, neurosurgery, and radiation oncology. Seven patients were treated with chemotherapy, 1 patient had proton beam therapy. Five-year overall survival was 45%. The eight surviving patients had neurological sequelae, and developmental outcome was variable. Conclusions Brain tumors are uncommon in children under 6-months of age. Patients present with a variety of tumor pathologies. Children who survive have neurological sequelae. More studies are necessary to understand the impact that different treatment options, tumor pathology, and tumor location have on neurological outcome.

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Section: Resultsmentioning

confidence: 99%

Surgical Treatment of Brain Tumors in Infants Younger than Six Months of Age and Review of the Literature

et al. 2012

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“…gliosarcoma in a two-month-old female whose mother was exposed to the insecticide heptachlor via ingested dairy milk during pregnancy. 34 This girl presented with projectile vomiting at two weeks of age and had and Radkowski et al 18 Cases pertinent to the above differential diagnosis, and not previously recognised to be gliosarcomas, are also present in the literature.…”

Section: Chadduck Et Al Published a Subsequent Instance Of Congenitalmentioning

confidence: 84%

“…With the exception of the cytogenetic results described by Chadduck et al (see above), the genetics of congenital gliosarcomas is poorly understood. 34 However, some insight into the genetics of congenital gliosarcomas may be drawn from the recent analysis of six congenital glioblastomas by Brat et al 1 Clinically, these congenital glioblastoma patients ranged from four days to 12 weeks of age. All tumours were supratentorial and all, save for one, involved the cerebral hemispheric lobes.…”

Section: Chadduck Et Al Published a Subsequent Instance Of Congenitalmentioning

confidence: 99%

Gliosarcoma – Clinico-pathology, Genetics and a Review of Rare Congenital Cases

Dunham¹

2009

European Oncology &Amp; Haematology

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Congenital brain tumours are rare. In general, they are preferentially located in the supratentorial compartment, and despite the occurrence of low-grade entities, these tumours are associated with a very poor prognosis. When strictly defined, the most common forms of congenital neoplasia are teratomas and astrocytomas. Among the astrocytomas, all grades (World Health Organization [WHO] I–IV) and many of the different subtypes are represented. This includes the most prognostically worrisome ‘diffusely infiltrating’ astrocytomas. Gliosarcomas are a variant of glioblastoma (i.e. WHO grade IV astrocytoma) that exhibits both malignant astrocytic (i.e. glioblastoma) and mesenchymal (i.e. sarcoma) components. Despite their bi-phasic histology, genetic analyses of adult gliosarcoma cases suggest not only a molecular profile similar to glioblastoma, but also a monoclonal histogenesis for the glial and sarcomatous elements. Congenital gliosarcomas are extremely rare, with only a handful of cases being described in the literature. Not surprisingly, therefore, detailed clinical, pathological and genetic data are limited. However, based on a recent analysis of congenital glioblastomas, congenital gliosarcomas may constitute an entity that is genetically and prognostically distinct from adult cases.

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“…Our case of congenital gliosarcoma is one of a very few that has been documented in the literature [6,[12][13][14]. Notably, the neuropathologic details of these previously reported cases are extremely limited.…”

Section: Discussionmentioning

confidence: 95%

“…To date, the literature includes only rare examples of congenitally occurring gliosarcomas [6,[12][13][14]. We present a case of a very rare congenital gliosarcoma causing the death of a 1-day-old male.…”

Section: Introductionmentioning

confidence: 99%

Congenital Gliosarcoma: Detailed Clinicopathologic Documentation of a Rare Neoplasm

et al. 2009

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Congenital brain tumors are rare. Clinically, they often result in macrocrania, hydrocephalus, and focal neurologic deficits. Fetal onset may result in dystocia and stillbirth. Antenatal detection is becoming more common as the result of neuroimaging, and modalities such as magnetic resonance imaging can assist in narrowing the pathologic differential diagnoses. Teratomas and astrocytomas appear to be the most common congenital neoplasms. Amongst the latter, all grades and many subtypes are represented in the congenital time period, including the diffusely infiltrative forms of astrocytoma. Gliosarcoma is currently considered a variant of glioblastoma (i.e., astrocytoma, World Health Organization grade IV) that exhibits genetically similar yet phenotypically separate histologic regions of high-grade astrocytoma and sarcoma. Only rare instances of congenital gliosarcoma have been reported. We detail the case of a 1-day-old term male who presented with macrocrania, hydrocephalus, and signs of increased intracranial pressure. Pathology revealed evidence of a classic gliosarcoma.

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Gliosarcoma with Chromosome Abnormalities in a Neonate Exposed to Heptachlor

Cited by 19 publications

References 0 publications

Surgical Treatment of Brain Tumors in Infants Younger than Six Months of Age and Review of the Literature

Surgical Treatment of Brain Tumors in Infants Younger than Six Months of Age and Review of the Literature

Gliosarcoma – Clinico-pathology, Genetics and a Review of Rare Congenital Cases

Congenital Gliosarcoma: Detailed Clinicopathologic Documentation of a Rare Neoplasm

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