{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,24]],"date-time":"2025-11-24T21:44:09Z","timestamp":1764020649736,"version":"build-2065373602"},"reference-count":19,"publisher":"MDPI AG","issue":"1","license":[{"start":{"date-parts":[[2023,1,9]],"date-time":"2023-01-09T00:00:00Z","timestamp":1673222400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Brain Sciences"],"abstract":"<jats:p>An encephalocele is a pathological brain herniation caused by osseous dural defects. Encephaloceles are known to be regions of epileptogenic foci. We describe the case of a 44-year-old woman with refractory epilepsy associated with a frontal skull base encephalocele. Epilepsy surgery for encephalocele resection was performed; however, the epilepsy was refractory. A second epilepsy surgery for frontal lobectomy using intraoperative electroencephalography was required to achieve adequate seizure control. Previous reports have shown that only encephalocele resection can result in good seizure control, and refractory epilepsy due to frontal lobe encephalocele has rarely been reported. To the best of our knowledge, this is the first report of frontal encephalocele plus epilepsy in which good seizure control using only encephalocele resection was difficult to achieve. Herein, we describe the possible mechanisms of encephalocele plus epilepsy and the surgical strategy for refractory epilepsy with encephalocele, including a literature review.<\/jats:p>","DOI":"10.3390\/brainsci13010115","type":"journal-article","created":{"date-parts":[[2023,1,9]],"date-time":"2023-01-09T02:09:39Z","timestamp":1673230179000},"page":"115","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Frontal Encephalocele Plus Epilepsy: A Case Report and Review of the Literature"],"prefix":"10.3390","volume":"13","author":[{"given":"Ken","family":"Yamazaki","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9667-9828","authenticated-orcid":false,"given":"Kohei","family":"Kanaya","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"},{"name":"Division of Epilepsy, Shinshu University Hospital, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6422-4140","authenticated-orcid":false,"given":"Takehiro","family":"Uda","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tetsuhiro","family":"Fukuyama","sequence":"additional","affiliation":[{"name":"Division of Epilepsy, Shinshu University Hospital, Matsumoto 390-8621, Japan"},{"name":"Department of Pediatrics, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Makoto","family":"Nishioka","sequence":"additional","affiliation":[{"name":"Division of Epilepsy, Shinshu University Hospital, Matsumoto 390-8621, Japan"},{"name":"Department of Pediatrics, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yumi","family":"Hoshino","sequence":"additional","affiliation":[{"name":"Division of Epilepsy, Shinshu University Hospital, Matsumoto 390-8621, Japan"},{"name":"Department of Medicine (Neurology and Rheumatology), Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tomoki","family":"Kaneko","sequence":"additional","affiliation":[{"name":"Department of Radiology, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ridzky Firmansyah","family":"Hardian","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daisuke","family":"Yamazaki","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Haruki","family":"Kuwabara","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kohei","family":"Funato","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tetsuyoshi","family":"Horiuchi","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shinshu University School of Medicine, Matsumoto 390-8621, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2023,1,9]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"E11","DOI":"10.3171\/FOC.2008.25.12.E11","article-title":"Spontaneous encephaloceles of the temporal lobe","volume":"25","author":"Wind","year":"2008","journal-title":"Neurosurg. Focus."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"200","DOI":"10.2174\/1573396315666191018161535","article-title":"Occipital Encephalocele: Cause, Incidence, Neuroimaging and Surgical Management","volume":"16","author":"Markovic","year":"2020","journal-title":"Curr. Pediatr. Rev."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/j.wneu.2021.04.121","article-title":"Encephalocele-Associated Drug-Resistant Epilepsy of Adult Onset: Diagnosis, Management, and Outcomes","volume":"151","author":"Domingo","year":"2021","journal-title":"World Neurosurg."},{"key":"ref_4","first-page":"17","article-title":"Drug-Resistant Epilepsy and Surgery","volume":"16","author":"Sheng","year":"2018","journal-title":"Curr. Neuropharmacol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"160","DOI":"10.1684\/epd.2010.0308","article-title":"Epilepsy surgery for refractory epilepsy due to encephalocele: A case report and review of the literature","volume":"12","author":"Faulkner","year":"2010","journal-title":"Epileptic Disord."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"E19","DOI":"10.1093\/ons\/opz098","article-title":"Medial Temporal Encephalocele and Medically Intractable Epilepsy: A Tailored Inferior Temporal Lobectomy and Case Report","volume":"18","author":"Mikula","year":"2020","journal-title":"Oper. Neurosurg."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"787","DOI":"10.1111\/j.0013-9580.2004.60203.x","article-title":"Electric stimulation on human cortex suppresses fast cortical activity and epileptic spikes","volume":"45","author":"Kinoshita","year":"2004","journal-title":"Epilepsia"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"884","DOI":"10.1056\/NEJM198909283211308","article-title":"Case records of the Massachusetts General Hospital. Weekly clinicopathological exercises Case 39-1989. A 63-year-old woman with a polypoid nasal mass and a recent grand-mal seizure","volume":"321","author":"Scully","year":"1989","journal-title":"N. Engl. J. Med."},{"key":"ref_9","first-page":"292","article-title":"Encephalocele-related seizures in adulthood: Synergistic contributions of CT and MRI to the anatomic work-up","volume":"98","author":"Guettat","year":"1998","journal-title":"Acta Neurol. Belg."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1113","DOI":"10.1007\/s00415-006-0211-2","article-title":"Symptomatic epilepsy with a tumor in the nose","volume":"253","author":"Eichler","year":"2006","journal-title":"J. Neurol."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1007\/s00701-006-1070-4","article-title":"Acquired encephaloceles and epilepsy in osteopetrosis","volume":"149","author":"Mandl","year":"2007","journal-title":"Acta Neurochir."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"157","DOI":"10.1212\/01.wnl.0000296822.59839.38","article-title":"Frontal encephalocele in a middle-aged woman with first seizure: Smells like a seizure to me","volume":"70","author":"Morley","year":"2008","journal-title":"Neurology"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"bcr2012006749","DOI":"10.1136\/bcr-2012-006749","article-title":"Not the usual sinusitis","volume":"2012","author":"Ammar","year":"2012","journal-title":"BMJ Case Rep."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"386","DOI":"10.3340\/jkns.2015.57.5.386","article-title":"Unusual Position and Presentation of Frontobasal Meningoencephalocela","volume":"57","author":"Busic","year":"2015","journal-title":"J. Korean Neurosurg. Soc."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"902","DOI":"10.1177\/000348940711601206","article-title":"Risk of meningitis with cerebrospinal fluid rhinorrhea","volume":"116","author":"Daudia","year":"2007","journal-title":"Ann. Otol. Rhinol. Laryngol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"1513","DOI":"10.1212\/WNL.42.8.1513","article-title":"Characteristics of intractable seizures following meningitis and encephalitis","volume":"42","author":"Marks","year":"1992","journal-title":"Neurology"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"444","DOI":"10.1093\/brain\/awv372","article-title":"Temporal plus epilepsy is a major determinant of temporal lobe surgery failures","volume":"139","author":"Barba","year":"2016","journal-title":"Brain"},{"key":"ref_18","doi-asserted-by":"crossref","unstructured":"Bannout, F., Harder, S., Lee, M., Zouros, A., Raghavan, R., Fogel, T., De Los Reyes, K., and Losey, T. (2018). Epilepsy Surgery for Skull-Base Temporal Lobe Encephaloceles: Should We Spare the Hippocampus from Resection?. Brain Sci., 8.","DOI":"10.3390\/brainsci8030042"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1007\/s10143-017-0922-0","article-title":"The usefulness of stereo-electroencephalography (SEEG) in the surgical management of focal epilepsy associated with \"hidden\" temporal pole encephalocele: A case report and literature review","volume":"41","author":"Mullin","year":"2018","journal-title":"Neurosurg. Rev."}],"container-title":["Brain Sciences"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2076-3425\/13\/1\/115\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T18:03:53Z","timestamp":1760119433000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2076-3425\/13\/1\/115"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,1,9]]},"references-count":19,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2023,1]]}},"alternative-id":["brainsci13010115"],"URL":"https:\/\/doi.org\/10.3390\/brainsci13010115","relation":{},"ISSN":["2076-3425"],"issn-type":[{"type":"electronic","value":"2076-3425"}],"subject":[],"published":{"date-parts":[[2023,1,9]]}}}