{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T00:48:28Z","timestamp":1791852508163,"version":"4.3.4"},"reference-count":38,"publisher":"Georg Thieme Verlag KG","issue":"04","license":[{"start":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T00:00:00Z","timestamp":1791763200000},"content-version":"vor","delay-in-days":11,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Neurol Surg Rep"],"published-print":{"date-parts":[[2026,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Introduction Meningiomas account for approximately 41% of primary CNS neoplasms, a minority of which are associated with paracrine-induced cranial hyperostosis. We report a unique systematic review of this rare phenomenon alongside an illustrative case of a woman with 14 years of progressive pan-calvarial hyperostosis attributable to a bilateral parasagittal meningioma.<\/jats:p>\n                  <jats:p>Methods A novel case of massive cranial hyperostosis treated surgically and an associated PRISMA-compliant systematic review are presented. Searches of OVID MEDLINE and EMBASE (keywords \u201cHyperostosis\u201d and \u201cMeningioma\u201d) on April 2, 2025 yielded 384 sources. All studies reporting calvarial hyperostosis associated with a convexity meningioma were included. Studies prior to the year 2000, non-human, or those that reported hyperostosis in other skull-base regions were excluded.<\/jats:p>\n                  <jats:p>Results A woman in her mid-40s presented with more than 14 years of enlarging pan-calvarial skull deformity predominantly involving the bilateral frontal bones. MRI revealed an enhancing midline mass obliterating the superior sagittal sinus consistent with meningioma. She underwent bifrontal craniotomy with en bloc hyperostotic bone removal and microsurgical meningioma resection. Reconstruction used dural grafting, titanium mesh cranioplasty, and scalp excision. Pathology confirmed a meningothelial meningioma with brain invasion (WHO Grade 2) with a solitary TRAF7 mutation. Systematic review yielded 25 studies (107 patients) showing a high prevalence of bifrontal bony involvement and meningothelial histology in cranial hyperostosis.<\/jats:p>\n                  <jats:p>Conclusion Mutations in TRAF7, a regulator of NF-\u03baB, MAPK, and related pathways, are linked to calvarial hyperostosis and meningothelial histopathology. This systematic review and uniquely severe case highlight the potentially profound impact of TRAF7 mutation on paracrine hyperostosis.<\/jats:p>","DOI":"10.1055\/a-2967-3259","type":"journal-article","created":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T00:08:37Z","timestamp":1791850117000},"page":"e208-e213","source":"Crossref","is-referenced-by-count":0,"title":["Beyond the Dura \u2015 Massive Hyperostotic Response from a Convexity Meningioma: Systematic Review &amp; Illustrative Case Example"],"prefix":"10.1055","volume":"87","author":[{"ORCID":"https:\/\/orcid.org\/0009-0008-3204-9606","authenticated-orcid":false,"given":"MacKenzie Steven","family":"Thomas","sequence":"additional","affiliation":[{"name":"The University of Oklahoma Health Sciences Center, Oklahoma, United States","place":["Oklahoma City"],"department":["Department of Neurosurgery"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-5090-2322","authenticated-orcid":false,"given":"MacGregor C.","family":"Thomas","sequence":"additional","affiliation":[{"name":"The University of Oklahoma Health Sciences Center, Oklahoma, United States","place":["Oklahoma City"],"department":["Department of Neurosurgery"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kari","family":"O'Toole","sequence":"additional","affiliation":[{"name":"The University of Oklahoma Health Sciences Center, Oklahoma, United States","place":["Oklahoma City"],"department":["Department of Neurosurgery"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark M.","family":"Mims","sequence":"additional","affiliation":[{"name":"The University of Oklahoma Health Sciences Center, Oklahoma, United States","place":["Oklahoma City"],"department":["Department of Otolaryngology-Head and Neck Surgery"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5314-1067","authenticated-orcid":false,"given":"Christopher Salvatore","family":"Graffeo","sequence":"additional","affiliation":[{"name":"The University of Oklahoma Health Sciences Center, Oklahoma, United States","place":["Oklahoma City"],"department":["Department of Neurosurgery"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"194","published-online":{"date-parts":[[2026,10,12]]},"reference":[{"issue":"10","key":"ref1","doi-asserted-by":"crossref","first-page":"1742","DOI":"10.1093\/neuonc\/noae082","article-title":"International Consortium on Meningiomas (ICOM). Meningioma: International Consortium on Meningiomas consensus review on scientific advances and treatment paradigms for clinicians, researchers, and patients","volume":"26","author":"J Z Wang","year":"2024","journal-title":"Neuro-Oncology"},{"key":"ref2","doi-asserted-by":"crossref","first-page":"iv1","DOI":"10.1093\/neuonc\/noy131","article-title":"CBTRUS statistical report: primary brain and other central nervous system tumors diagnosed in the United States in 2011-2015","volume":"20","author":"Q T Ostrom","year":"2018","journal-title":"Neuro-Oncology"},{"issue":"01","key":"ref3","doi-asserted-by":"crossref","first-page":"53","DOI":"10.1007\/s00701-010-0838-8","article-title":"Hyperostosing meningiomas of the cranial vault with and without tumor mass","volume":"153","author":"A Talacchi","year":"2011","journal-title":"Acta Neurochir"},{"issue":"02","key":"ref4","first-page":"591","article-title":"Osseous changes in meningioma en plaque","volume":"31","author":"J Matschke","year":"2011","journal-title":"Anticancer Res"},{"issue":"01","key":"ref5","doi-asserted-by":"crossref","first-page":"86","DOI":"10.4103\/2152-7806.114796","article-title":"Sphenoid wing en plaque meningiomas: surgical results and recurrence rates","volume":"4","author":"N M Simas","year":"2013","journal-title":"Surg Neurol Int"},{"issue":"01","key":"ref6","doi-asserted-by":"crossref","first-page":"22","DOI":"10.1136\/jnnp.20.1.22","article-title":"The recurrence of intracranial meningiomas after surgical treatment","volume":"20","author":"D Simpson","year":"1957","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"ref7","doi-asserted-by":"crossref","first-page":"1137849","DOI":"10.3389\/fonc.2023.1137849","article-title":"Intracranial meningiomas: an update of the 2021 World Health Organization classifications and review of management with a focus on radiation therapy","volume":"13","author":"V Yarabarla","year":"2023","journal-title":"Front Oncol"},{"issue":"71","key":"ref8","doi-asserted-by":"crossref","first-page":"n71","DOI":"10.1136\/bmj.n71","article-title":"The PRISMA 2020 statement: an updated guideline for reporting systematic reviews","volume":"372","author":"M J Page","year":"2021","journal-title":"BMJ"},{"issue":"08","key":"ref9","doi-asserted-by":"crossref","first-page":"e796","DOI":"10.1097\/SCS.0000000000008663","article-title":"Giant skull tumor: a benign meningioma","volume":"33","author":"B Abuzayed","year":"2022","journal-title":"J Craniofac Surg"},{"issue":"02","key":"ref10","doi-asserted-by":"crossref","first-page":"148","DOI":"10.1097\/MPH.0b013e3182830d56","article-title":"Focal cranial hyperostosis from meningioma: a complication from previous radiation treatment for childhood T-cell acute lymphoblastic leukemia","volume":"36","author":"N Songdej","year":"2014","journal-title":"J Pediatr Hematol Oncol"},{"issue":"01","key":"ref11","first-page":"50","article-title":"Does bony hyperostosis in intracranial meningioma signify tumor invasion?","volume":"60","author":"N Goyal","year":"2012","journal-title":"A radio-pathologic study Neurol India"},{"issue":"04","key":"ref12","doi-asserted-by":"crossref","first-page":"222","DOI":"10.5334\/jbr-btr.647","article-title":"Hyperostotic meningioma mimicking skull osteoma","volume":"94","author":"M Eyselbergs","year":"2011","journal-title":"J Belg Soc Radiol"},{"issue":"02","key":"ref13","first-page":"210","article-title":"Giant calvarial hyperostosis with biparasagittal en plaque meningioma","volume":"54","author":"S K Gupta","year":"2006","journal-title":"Neurol India"},{"issue":"04","key":"ref14","doi-asserted-by":"crossref","first-page":"606","DOI":"10.1001\/archneur.63.4.606","article-title":"A head bulge","volume":"63","author":"Y Karmon","year":"2006","journal-title":"Arch Neurol"},{"issue":"09","key":"ref15","doi-asserted-by":"crossref","first-page":"480","DOI":"10.2176\/nmc.45.480","article-title":"Hyperostotic meningioma with minimal tumor invasion into the skull","volume":"45","author":"J H Min","year":"2005","journal-title":"Neurol Med Chir"},{"issue":"02","key":"ref16","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1016\/S0090-3019(03)00521-4","article-title":"Parasagittal meningioma en plaque with extracranial extension presenting diffuse massive hyperostosis of the skull","volume":"61","author":"H Akutsu","year":"2004","journal-title":"Surg Neurol"},{"issue":"02","key":"ref17","first-page":"102","article-title":"Primary ectopic meningioma","volume":"99","author":"Y R Yadav","year":"2001","journal-title":"J Indian Med Assoc"},{"key":"ref18","doi-asserted-by":"crossref","first-page":"150","DOI":"10.25259\/SNI_130_2024","article-title":"Akinetic mutism following bilateral parasagittal meningioma occupied supplementary motor area removal and the spontaneous recovery of symptoms","volume":"15","author":"T Wangapakul","year":"2024","journal-title":"Surg Neurol Int"},{"key":"ref19","doi-asserted-by":"crossref","first-page":"100001","DOI":"10.1016\/j.wnsx.2018.100001","article-title":"Co-existence of atypical meningioma, intratumoral lipometaplasia and extensive hyperostosis of calvarium: a rare entity","volume":"1","author":"B M Aage","year":"2018","journal-title":"World Neurosurg X"},{"key":"ref20","doi-asserted-by":"crossref","first-page":"e497 EP","DOI":"10.1016\/j.wneu.2019.03.183","article-title":"Does putting back hyperostotic bone flap in meningioma surgery cause tumor recurrence? An observational prospective study","volume":"127","author":"B L Lau","year":"2019","journal-title":"World Neurosurg"},{"key":"ref21","doi-asserted-by":"crossref","first-page":"101374","DOI":"10.1016\/j.inat.2021.101374","article-title":"Expression of IL-6 and matrix metalloproteinases in a convexity meningiomas with hyperostosis: case report","volume":"27","author":"P Guadarrama-Ortiz","year":"2022","journal-title":"Interdiscip Neurosurg"},{"issue":"06","key":"ref22","doi-asserted-by":"crossref","first-page":"821 EP","DOI":"10.3340\/jkns.2020.0020","article-title":"Extent of hyperostotic bone resection in convexity meningioma to achieve pathologically free margins","volume":"63","author":"H Fathalla","year":"2020","journal-title":"J Korean Neurosurg Soc"},{"issue":"04","key":"ref23","doi-asserted-by":"crossref","first-page":"707 EP","DOI":"10.1055\/s-0039-3399600","article-title":"Fibrous meningioma with skull invasion","volume":"10","author":"S Ichimura","year":"2019","journal-title":"J Neurosci Rural Pract"},{"issue":"04","key":"ref24","doi-asserted-by":"crossref","first-page":"639 EP","DOI":"10.4103\/jnrp.jnrp_559_17","article-title":"Giant bilateral calvarial hyperostosis secondary to small unilateral frontal meningioma","volume":"9","author":"S Munjal","year":"2018","journal-title":"J Neurosci Rural Pract"},{"issue":"03","key":"ref25","doi-asserted-by":"crossref","first-page":"225 EP","DOI":"10.5835\/jecm.omu.29.03.011","article-title":"Giant calvarial meningioma which obliterated superior sagittal sinus: case report","volume":"29","author":"M Altas","year":"2012","journal-title":"J Exp Clin Med Turk"},{"key":"ref26","doi-asserted-by":"crossref","first-page":"264","DOI":"10.25259\/SNI_337_2020","article-title":"Meningioma-related subacute subdural hematoma: a case report","volume":"11","author":"D Matos","year":"2020","journal-title":"Surg Neurol Int"},{"key":"ref27","doi-asserted-by":"crossref","first-page":"65 EP","DOI":"10.1016\/j.wneu.2022.11.075","article-title":"Middle meningeal artery aneurysm in a giant meningioma","volume":"170","author":"L P Carlstrom","year":"2023","journal-title":"World Neurosurg"},{"issue":"10","key":"ref28","doi-asserted-by":"crossref","first-page":"878 EP","DOI":"10.3109\/00207454.2015.1077833","article-title":"Papillary meningioma: an aggressive variant meningioma with clinical features and treatment: a retrospective study of 10 cases","volume":"126","author":"B Li","year":"2016","journal-title":"Int J Neurosci"},{"issue":"06","key":"ref29","doi-asserted-by":"crossref","first-page":"e542","DOI":"10.1097\/SCS.0000000000006472","article-title":"Protruding meningioma of the forehead with extensive hyperostosis mimicking skull osteoma","volume":"31","author":"D-H Seo","year":"2020","journal-title":"J Craniofac Surg"},{"issue":"01","key":"ref30","doi-asserted-by":"crossref","first-page":"281","DOI":"10.3892\/ol.2014.2074","article-title":"Rare giant bilateral calvarial hyperostosis across the superior sagittal sinus secondary to brain meningioma: a case report","volume":"8","author":"H Zhang","year":"2014","journal-title":"Oncol Lett"},{"issue":"03","key":"ref31","doi-asserted-by":"crossref","first-page":"735 EP","DOI":"10.3171\/2018.4.JNS1850","article-title":"Removal of giant intraosseous meningioma followed by cranioplasty using a custom-made bioceramic implant: case report","volume":"131","author":"J Sundblom","year":"2019","journal-title":"J Neurosurg"},{"issue":"05","key":"ref32","doi-asserted-by":"crossref","first-page":"1345","DOI":"10.3171\/2019.8.JNS191266","article-title":"Correlations between genomic subgroup and clinical features in a cohort of more than 3000 meningiomas","volume":"133","author":"M W Youngblood","year":"2019","journal-title":"J Neurosurg"},{"issue":"6123","key":"ref33","doi-asserted-by":"crossref","first-page":"1077","DOI":"10.1126\/science.1233009","article-title":"Genomic analysis of non-NF2 meningiomas reveals mutations in TRAF7, KLF4, AKT1, and SMO","volume":"339","author":"V E Clark","year":"2013","journal-title":"Science"},{"issue":"17","key":"ref34","doi-asserted-by":"crossref","first-page":"17278","DOI":"10.1074\/jbc.C400063200","article-title":"TRAF7 potentiates MEKK3-induced AP1 and CHOP activation and induces apoptosis","volume":"279","author":"L G Xu","year":"2004","journal-title":"J Biol Chem"},{"issue":"06","key":"ref35","doi-asserted-by":"crossref","first-page":"1233","DOI":"10.1002\/jcp.25676","article-title":"The emerging role of TRAF7 in tumor development","volume":"232","author":"T Zotti","year":"2017","journal-title":"J Cell Physiol"},{"issue":"02","key":"ref36","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1007\/s11060-021-03819-2","article-title":"Type of bony involvement predicts genomic subgroup in sphenoid wing meningiomas","volume":"154","author":"L Jin","year":"2021","journal-title":"J Neuro-Oncol"},{"issue":"06","key":"ref37","doi-asserted-by":"crossref","first-page":"662","DOI":"10.1227\/ons.0000000000001052","article-title":"Epidemiology, genetics, and DNA methylation grouping of hyperostotic meningiomas","volume":"26","author":"G Umbach","year":"2024","journal-title":"Oper Neurosurg"},{"issue":"05","key":"ref38","doi-asserted-by":"crossref","first-page":"783","DOI":"10.1093\/neuonc\/noaa226","article-title":"Associations of meningioma molecular subgroup and tumor recurrence","volume":"23","author":"M W Youngblood","year":"2021","journal-title":"Neuro-Oncology"}],"container-title":["Journal of Neurological Surgery Reports"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/www.thieme-connect.de\/products\/ejournals\/pdf\/10.1055\/a-2967-3259.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T00:08:42Z","timestamp":1791850122000},"score":1,"resource":{"primary":{"URL":"http:\/\/www.thieme-connect.de\/DOI\/DOI?10.1055\/a-2967-3259"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10]]},"references-count":38,"journal-issue":{"issue":"04","published-online":{"date-parts":[[2026,10,6]]},"published-print":{"date-parts":[[2026,10]]}},"URL":"https:\/\/doi.org\/10.1055\/a-2967-3259","archive":["Portico","CLOCKSS"],"relation":{},"ISSN":["2193-6358","2193-6366"],"issn-type":[{"value":"2193-6358","type":"print"},{"value":"2193-6366","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10]]}}}