Pediatric Intracranial Anaplastic Gangliogliomas: Illustrative Case and Systematic Review

Bouali, Sofiene; Said, I. Ben; Zehani, Alia; Drissi, C.; Bouhoula, Asma; Kallel, Jalel; Jemel, Hafedh

doi:10.1016/j.wneu.2018.07.278

Cited by 5 publications

(6 citation statements)

References 35 publications

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“…Mean patient age was 9.18 years, and all underwent surgery. Mean overall survival was 43 months, with 1-and 3-year survival being 76.6% and 45.5%, respectively [56]. These numbers, while not as dire as those reported by the Zanello et al study, still reflect the far poorer prognosis of AGGs as compared to GGs.…”

Section: Anaplastic Ganglioglioma Who Grade IIImentioning

confidence: 60%

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Pathologic and molecular aspects of anaplasia in circumscribed gliomas and glioneuronal tumors

2019

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Many breakthroughs have been made in the past decade regarding our knowledge of the biological basis of the diffuse gliomas, the most common primary central nervous system (CNS) tumors. These tumors as a group are aggressive, associated with high mortality and have a predilection for adults. However, a subset of CNS glial and glioneuronal tumors are characterized by a more circumscribed pattern of growth and occur more commonly in children and young adults. They tend to be indolent, but our understanding of anaplastic changes in these tumors continues to improve as diagnostic classifications evolve in the era of molecular pathology and more integrated and easily-accessible clinical databases. The presence of anaplasia in pleomorphic xanthoastrocytomas and gangliogliomas is assigned a WHO grade III under the current classification, while the significance of anaplasia in pilocytic astrocytomas remains controversial. However, recent data highlight the association of the latter with aggressive clinical behavior, as well as the presence of molecular genetic features of both pilocytic and diffuse gliomas, with the recognition that the precise terminology remains to be defined. We review current concepts and advances regarding histopathology and molecular understanding of pilocytic astrocytomas, pleomorphic xanthoastrocytomas and gangliogliomas, with a focus on their anaplastic counterparts.

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Section: Anaplastic Ganglioglioma Who Grade IIImentioning

confidence: 60%

“…Lastly, a systematic review of pediatric anaplastic gangliogliomas in 2018 included 24 studies following 34 patients [56]. Mean patient age was 9.18 years, and all underwent surgery.…”

Section: Anaplastic Ganglioglioma Who Grade IIImentioning

confidence: 99%

Pathologic and molecular aspects of anaplasia in circumscribed gliomas and glioneuronal tumors

2019

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“…Lucas et al even reported a case in which the anaplastic ganglioglioma developed on a previously resected low-grade astrocytoma, in concordance to the hypothesis that they develop in long lasting lesions [ 39 ]. Another paper, from Bouali et al , reported seven juvenile anaplastic ganglioglioma, due to secondary malignant transformation of WHO grade I ganglioglioma [ 50 ].…”

Section: ⧉ Discussionmentioning

confidence: 99%

“…First of all, it is necessary to confirm the presence of a mixed glioneuronal proliferation and exclude a gangliocytoma or an astrocytoma. To achieve this goal, one must demonstrate the presence of the neuronal component through microtubuleassociated protein 2 (MAP2), synaptophysin, chromogranin A, neuronal nuclei (NeuN), and neurofilaments [2,50], respectively of the glial component through glial fibrillary acidic protein (GFAP) [2].…”

Section: Practical Immunohistochemical Aspectsmentioning

confidence: 99%

Practical aspects regarding the histopathological grading and anaplastic transformation of gangliogliomas – a literature review

Lisievici

Lisievici²,

Pasov³

et al. 2022

Rom J Morphol Embryol

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Ganglioglioma represents a benign central nervous tumor, occurring predominantly in the pediatric population and affecting the temporal lobe. It is also renowned for its epileptogenic potential. However, to date, there are numerous uncertain features about this tumor, especially about its grading system. In the former World Health Organization (WHO) Classification of central nervous tumors system, gangliogliomas could have been attributed one out of three grades: grade I (benign), grade II (atypical), and grade III (anaplastic). The new classification systems have renounced to atypical ganglioglioma nomenclature, due to the lack of histopathological criteria for this entity. Another controversial aspect of grade I ganglioglioma is its potential to transform into a malignant tumor, namely, most frequently an anaplastic ganglioglioma. Based on our knowledge, there are no literature reviews to date focusing on anaplastic transformation potential. The present paper encompasses all anaplastic transformation of gangliogliomas and has analyzed the time frame between the two events, the age of the patients and its relationship to the complete or subtotal resection and administration of radiotherapy. Thirty-three cases of malignant transformation of ganglioglioma have been reported so far in the literature, with 54.54% of them undergoing progression to anaplastic ganglioglioma and 21.21% to anaplastic ganglioglioma. Median age was 26 years, and the cases were evenly distributed between the two genres. Only 27.27% of all evaluated cases had been administrated adjuvant radiotherapy, and only 44% of the latter have had an incomplete tumoral resection.

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“…Based on the data available, patients with aGG fare worse than patients with GG CNS WHO Grade 1. Recurrence was observed in 50% of the aGG patients, and median overall survival ranged from 25 to 44 months [2, 5, 6, 8–12]. Similar to GGs, v‐Raf murine sarcoma viral oncogene homologue B ( BRAF ) V600E mutations also have been described in aGGs.…”

Section: Introductionmentioning

confidence: 99%

Anaplastic ganglioglioma—A diagnosis comprising several distinct tumour types

Reinhardt

Pfister

Schrimpf

et al. 2022

Neuropathology Appl Neurobio

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Aims Anaplastic ganglioglioma is a rare tumour, and diagnosis has been based on histological criteria. The 5th edition of the World Health Organization Classification of Tumours of the Central Nervous System (CNS WHO) does not list anaplastic ganglioglioma as a distinct diagnosis due to lack of molecular data in previous publications. We retrospectively compiled a cohort of 54 histologically diagnosed anaplastic gangliogliomas to explore whether the molecular profiles of these tumours represent a separate type or resolve into other entities. Methods Samples were subjected to histological review, desoxyribonucleic acid (DNA) methylation profiling and next‐generation sequencing. Morphological and molecular data were summarised to an integrated diagnosis. Results The majority of tumours designated as anaplastic gangliogliomas resolved into other CNS WHO diagnoses, most commonly pleomorphic xanthoastrocytoma (16/54), glioblastoma, isocitrate dehydrogenase protein (IDH) wild type and diffuse paediatric‐type high‐grade glioma, H3 wild type and IDH wild type (11 and 2/54), followed by low‐grade glial or glioneuronal tumours including pilocytic astrocytoma, dysembryoplastic neuroepithelial tumour and diffuse leptomeningeal glioneuronal tumour (5/54), IDH mutant astrocytoma (4/54) and others (6/54). A subset of tumours (10/54) was not assignable to a CNS WHO diagnosis, and common molecular profiles pointing to a separate entity were not evident. Conclusions In summary, we show that tumours histologically diagnosed as anaplastic ganglioglioma comprise a wide spectrum of CNS WHO tumour types with different prognostic and therapeutic implications. We therefore suggest assigning this designation with caution and recommend comprehensive molecular workup.

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Pediatric Intracranial Anaplastic Gangliogliomas: Illustrative Case and Systematic Review

Cited by 5 publications

References 35 publications

Pathologic and molecular aspects of anaplasia in circumscribed gliomas and glioneuronal tumors

Pathologic and molecular aspects of anaplasia in circumscribed gliomas and glioneuronal tumors

Practical aspects regarding the histopathological grading and anaplastic transformation of gangliogliomas – a literature review

Anaplastic ganglioglioma—A diagnosis comprising several distinct tumour types

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