{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,5,14]],"date-time":"2025-05-14T01:45:52Z","timestamp":1747187152552,"version":"3.40.5"},"reference-count":16,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2022,9,22]],"date-time":"2022-09-22T00:00:00Z","timestamp":1663804800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Neuropathology"],"published-print":{"date-parts":[[2022,12]]},"abstract":"<jats:p>Neurenteric cyst (NC) shows benign histopathology and rarely demonstrate malignant transformation. We herein describe a case of NC that exhibited malignant transformation. A 65\u2010year\u2010old female presented with gait disturbance due to compression by a cystic mass on the dorsal surface of the medulla oblongata. Partial resection was performed twice, leading to improvement of  her symptoms. Two years after the second surgery, gadolinium\u2010perfused T1\u2010weighted magnetic resonance imaging revealed an invasive lesion with contrast enhancement at the trigone of the left lateral ventricle for which partial resection followed by radiotherapy was performed. However, mass regrowth was observed, with the patient eventually succumbing to her disease 11 months after her third surgery. Histopathological analyses of the first and second surgical specimens identified pseudostratified cuboidal epithelial cells, with no nuclear or cellular atypia resembling gastrointestinal mucosa, lining the inner surface of the cystic wall. Based on these findings the lesion was diagnosed as NC. The third surgical specimen exhibited apparent malignant features of the epithelial cells with elongated and hyperchromatic nuclei, several mitotic figures, small necrotic foci, and a patternless or sheet\u2010like arrangement. Based on these findings, the lesion was diagnosed as NC with malignant transformation. Next\u2010generation sequencing revealed <jats:italic>KRAS<\/jats:italic> p.G12D mutation in all specimens. Additionally, the third surgical specimen harbored the following 12 <jats:italic>de novo<\/jats:italic> gene alterations: <jats:italic>ARID1A<\/jats:italic> loss, <jats:italic>BAP1<\/jats:italic> p.F170L, <jats:italic>CDKN1B<\/jats:italic> loss, <jats:italic>CDKN2A<\/jats:italic> loss, <jats:italic>CDKN2B<\/jats:italic> loss, <jats:italic>FLCN<\/jats:italic> loss, <jats:italic>PTCH1<\/jats:italic> loss, <jats:italic>PTEN<\/jats:italic> loss, <jats:italic>PTPRD<\/jats:italic> loss, <jats:italic>SUFU<\/jats:italic> loss, <jats:italic>TP53<\/jats:italic> loss, and <jats:italic>TSC1<\/jats:italic> loss. The aforementioned results suggest that <jats:italic>KRAS<\/jats:italic> mutation is associated with the development of the NC, and that the additional gene alterations contribute to malignant transformation of the NC.<\/jats:p>","DOI":"10.1111\/neup.12822","type":"journal-article","created":{"date-parts":[[2022,9,23]],"date-time":"2022-09-23T05:55:30Z","timestamp":1663912530000},"page":"519-525","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Elucidating the multiple genetic alterations involved in the malignant transformation of a <scp><i>KRAS<\/i><\/scp> mutant neurenteric cyst. A case report"],"prefix":"10.1111","volume":"42","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4471-7858","authenticated-orcid":false,"given":"Shoji","family":"Saito","sequence":"first","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"},{"name":"Department of Pathology Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Manabu","family":"Natsumeda","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Makoto","family":"Sainouchi","sequence":"additional","affiliation":[{"name":"Department of Pathology Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toru","family":"Takino","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kohei","family":"Shibuya","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jotaro","family":"On","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"},{"name":"Department of Pathology Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yu","family":"Kanemaru","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryosuke","family":"Ogura","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masayasu","family":"Okada","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Makoto","family":"Oishi","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Niigata University Brain Research Institute  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshifumi","family":"Shimada","sequence":"additional","affiliation":[{"name":"Division of Digestive and General Surgery Niigata University Graduate School of Medical and Dental Sciences  Niigata Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toshifumi","family":"Wakai","sequence":"additional","affiliation":[{"name":"Division of Digestive and General Surgery Niigata University Graduate School of 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