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Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports

INTRODUCTION: Pleomorphic xanthoastrocytomas (PXA) are rare, typically benign, slow-growing tumors that commonly occur in the cerebral hemispheres. We describe two cases of clinically aggressive PXA with uncommon locations; one was in the tectal plate, and the other had simultaneous multicentric les...

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Autores principales: Nagaishi, Masaya, Nakae, Ryuta, Fujii, Yoshiko, Inoue, Yuki, Sugiura, Yoshiki, Takano, Issei, Tanaka, Yoshihiro, Suzuki, Kensuke
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7220053/
https://www.ncbi.nlm.nih.gov/pubmed/32011515
http://dx.doi.org/10.1097/MD.0000000000018880
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author Nagaishi, Masaya
Nakae, Ryuta
Fujii, Yoshiko
Inoue, Yuki
Sugiura, Yoshiki
Takano, Issei
Tanaka, Yoshihiro
Suzuki, Kensuke
author_facet Nagaishi, Masaya
Nakae, Ryuta
Fujii, Yoshiko
Inoue, Yuki
Sugiura, Yoshiki
Takano, Issei
Tanaka, Yoshihiro
Suzuki, Kensuke
author_sort Nagaishi, Masaya
collection PubMed
description INTRODUCTION: Pleomorphic xanthoastrocytomas (PXA) are rare, typically benign, slow-growing tumors that commonly occur in the cerebral hemispheres. We describe two cases of clinically aggressive PXA with uncommon locations; one was in the tectal plate, and the other had simultaneous multicentric lesions. PATIENT CONCERNS: The both cases presented with severe headache with no significant past medical history. DIAGNOSIS: PXA World Health Organization grade II were histopathologically diagnosed from surgically resected specimens, and immunohistochemical and sequence analysis revealed a high Ki-67 proliferative index and BRAF V600E mutation in both the cases. INTERVENTIONS: The first case presented with multicentric lesions and underwent partial resection, whereas the second case presented with a tectal plate tumor that was managed by gross total surgical resection. Strong 5-aminolevulinic acid (5-ALA)-induced fluorescence was observed in both the lesions. Postoperative radiotherapy plus concomitant and adjuvant temozolomide was administered to both the patients. OUTCOMES: Despite completing adjuvant chemo-radiotherapy, both the patients had local tumor recurrence at 2 and 5 months after the operation, respectively. CONCLUSION: The progressive clinical courses in our cases suggest that additional postoperative therapy should be considered during the treatment of PXA with a high Ki67 index, and that temozolomide with radiotherapy, followed by temozolomide maintenance therapy, may not prevent recurrence in such tumors. Importantly, our experience implies that unlike other subtypes of low grade gliomas, 5-ALA fluorescence is useful for intraoperative visualization of PXA.
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spelling pubmed-72200532020-06-15 Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports Nagaishi, Masaya Nakae, Ryuta Fujii, Yoshiko Inoue, Yuki Sugiura, Yoshiki Takano, Issei Tanaka, Yoshihiro Suzuki, Kensuke Medicine (Baltimore) 5700 INTRODUCTION: Pleomorphic xanthoastrocytomas (PXA) are rare, typically benign, slow-growing tumors that commonly occur in the cerebral hemispheres. We describe two cases of clinically aggressive PXA with uncommon locations; one was in the tectal plate, and the other had simultaneous multicentric lesions. PATIENT CONCERNS: The both cases presented with severe headache with no significant past medical history. DIAGNOSIS: PXA World Health Organization grade II were histopathologically diagnosed from surgically resected specimens, and immunohistochemical and sequence analysis revealed a high Ki-67 proliferative index and BRAF V600E mutation in both the cases. INTERVENTIONS: The first case presented with multicentric lesions and underwent partial resection, whereas the second case presented with a tectal plate tumor that was managed by gross total surgical resection. Strong 5-aminolevulinic acid (5-ALA)-induced fluorescence was observed in both the lesions. Postoperative radiotherapy plus concomitant and adjuvant temozolomide was administered to both the patients. OUTCOMES: Despite completing adjuvant chemo-radiotherapy, both the patients had local tumor recurrence at 2 and 5 months after the operation, respectively. CONCLUSION: The progressive clinical courses in our cases suggest that additional postoperative therapy should be considered during the treatment of PXA with a high Ki67 index, and that temozolomide with radiotherapy, followed by temozolomide maintenance therapy, may not prevent recurrence in such tumors. Importantly, our experience implies that unlike other subtypes of low grade gliomas, 5-ALA fluorescence is useful for intraoperative visualization of PXA. Wolters Kluwer Health 2020-01-17 /pmc/articles/PMC7220053/ /pubmed/32011515 http://dx.doi.org/10.1097/MD.0000000000018880 Text en Copyright © 2020 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by/4.0 This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0
spellingShingle 5700
Nagaishi, Masaya
Nakae, Ryuta
Fujii, Yoshiko
Inoue, Yuki
Sugiura, Yoshiki
Takano, Issei
Tanaka, Yoshihiro
Suzuki, Kensuke
Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports
title Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports
title_full Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports
title_fullStr Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports
title_full_unstemmed Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports
title_short Rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: Two case reports
title_sort rare clinical presentations of pleomorphic xanthoastrocytoma with a high proliferative index: two case reports
topic 5700
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7220053/
https://www.ncbi.nlm.nih.gov/pubmed/32011515
http://dx.doi.org/10.1097/MD.0000000000018880
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