{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T23:03:27Z","timestamp":1792019007164,"version":"4.4.0"},"reference-count":22,"publisher":"MDPI AG","issue":"21","license":[{"start":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T00:00:00Z","timestamp":1761264000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Background\/Objectives: Anterior cervical meningocele (ACM) is a rare congenital condition characterized by the herniation of the meninges through a defect in the anterior vertebral column. ACM clinical management is not standardized because this condition is rare, and guidelines are missing. Hereby, a systematic literature review is performed to determine management options and outcomes. Methods: The case of a 62-year-old patient with incidental diagnosis of C3-C5 ACM is presented. A systematic review was conducted using standard PRISMA (preferred reporting items for systematic reviews and meta-analyses) guidelines for all cases of anterior cervical meningocele from 1837 to 2025. Results: The review provided nine clinical cases and our illustrative case. The median age was 47 years, with a predominance of female patients (70%). The most common presenting symptom was neck pain (60%), followed by paresthesia and hypoesthesia in the upper limbs. Four patients underwent conservative management with clinical and radiological follow-up, while four patients underwent neurosurgical intervention. Surgical treatment was complicated by cerebrospinal fluid (CSF) leak in two patients, and one of them developed meningitis. Conclusions: ACM is typically associated with mesodermal dysplasia and dural ectasia. ACM usually has a benign clinical course, requiring neurological follow-up and conservative management alone. However, a surgical approach should be considered in cases of vertebral instability or symptoms related to upper airway compression or upper gastrointestinal tract compression despite the high risk of CSF leak when surgical repair is attempted.<\/jats:p>","DOI":"10.3390\/jcm14217530","type":"journal-article","created":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T07:41:35Z","timestamp":1761291695000},"page":"7530","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Anterior Cervical Meningocele: Systematic Review of the Literature and Illustrative Case"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-5159-0183","authenticated-orcid":false,"given":"Edoardo","family":"Ricci","sequence":"first","affiliation":[{"name":"Neurology Unit, Department of Medicine, Surgery and Health Sciences, University Hospital and Health Services of Trieste\u2014ASUGI, University of Trieste, Strada di Fiume, 447, 34149 Trieste, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2426-2884","authenticated-orcid":false,"given":"Antonio","family":"Meola","sequence":"additional","affiliation":[{"name":"Neurosurgery Unit, Department of Medicine, Surgery and Health Sciences, University Hospital and Health Services of Trieste\u2014ASUGI, University of Trieste, Strada di Fiume, 447, 34149 Trieste, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilario","family":"Scali","sequence":"additional","affiliation":[{"name":"Neurology Unit, Department of Medicine, Surgery and Health Sciences, University Hospital and Health Services of Trieste\u2014ASUGI, University of Trieste, Strada di Fiume, 447, 34149 Trieste, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paolo","family":"Manganotti","sequence":"additional","affiliation":[{"name":"Neurology Unit, Department of Medicine, Surgery and Health Sciences, University Hospital and Health Services of Trieste\u2014ASUGI, University of Trieste, Strada di Fiume, 447, 34149 Trieste, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leonello","family":"Tacconi","sequence":"additional","affiliation":[{"name":"Neurosurgery Unit, Department of Medicine, Surgery and Health Sciences, University Hospital and Health Services of Trieste\u2014ASUGI, University of Trieste, Strada di Fiume, 447, 34149 Trieste, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2025,10,24]]},"reference":[{"key":"ref_1","first-page":"1105","article-title":"Anterior cervical meningoceles in neurofibromatosis","volume":"7","author":"Kaiser","year":"1986","journal-title":"Am. J. Neuroradiol."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"717","DOI":"10.1097\/01241398-198511000-00018","article-title":"A true anterior thoracic meningocele associated with a congenital kyphoscoliosis","volume":"5","author":"Jaffray","year":"1985","journal-title":"J. Pediatr. Orthop."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"127","DOI":"10.1136\/adc.46.246.127","article-title":"Natural history of spina bifida cystica and cranium bifidum cysticum: Major central nervous system malformations in South Wales. Part IV","volume":"46","author":"Laurence","year":"1971","journal-title":"Arch. Dis. Child."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"59","DOI":"10.1055\/s-2008-1066168","article-title":"Tethered cord with anorectal malformation, sacral anomalies and presacral masses: An underrecognized association","volume":"5","author":"Walton","year":"1995","journal-title":"Eur. J. Pediatr. Surg."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"444","DOI":"10.1097\/BRS.0000000000003296","article-title":"Anterior Sacral Meningocele: A New Classification and Treatment Using the Dorsal Transsacral Approach","volume":"45","author":"Cheng","year":"2020","journal-title":"Spine"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"430","DOI":"10.1177\/000992288202100709","article-title":"Lateral cervical meningocele","volume":"21","author":"Shore","year":"1982","journal-title":"Clin. Pediatr."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"692","DOI":"10.1097\/00004728-198907000-00028","article-title":"Anterolateral cervical meningocele in association with neurofibromatosis: MR and CT studies","volume":"13","author":"So","year":"1989","journal-title":"J. Comput. Assist. Tomogr."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"124","DOI":"10.1007\/s101430050045","article-title":"Multiple anterolateral cervical meningoceles associated with neurofibromatosis","volume":"22","author":"Tuna","year":"1999","journal-title":"Neurosurg. Rev."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"612","DOI":"10.1016\/j.otohns.2008.11.002","article-title":"Multiple cervical anterior meningoceles in a patient with neurofibromatosis type 1 cause dysphagia and dyspnea","volume":"140","author":"Kos","year":"2009","journal-title":"Otolaryngol.\u2013Head Neck Surg."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"205","DOI":"10.1177\/1971400915576308","article-title":"Cervical vertebral fusion with anterior meningocele","volume":"28","author":"Gallagher","year":"2015","journal-title":"Neuroradiol. J."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"834","DOI":"10.1016\/S1474-4422(14)70063-8","article-title":"Neurofibromatosis type 1: A multidisciplinary approach to care","volume":"13","author":"Hirbe","year":"2014","journal-title":"Lancet Neurol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"183","DOI":"10.1097\/01.blo.0000130267.49895.20","article-title":"Klippel-Feil syndrome: Clinical features and current understanding of etiology","volume":"424","author":"Tracy","year":"2004","journal-title":"Clin. Orthop. Relat. Res."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"617","DOI":"10.1097\/01241398-199509000-00014","article-title":"The natural history of Klippel-Feil syndrome: Clinical, roentgenographic, and magnetic resonance imaging findings at adulthood","volume":"15","author":"Guille","year":"1995","journal-title":"J. Pediatr. Orthop."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"116","DOI":"10.1016\/S0012-1606(02)00022-2","article-title":"Multiple joint and skeletal patterning defects caused by single and double mutations in the mouse Gdf6 and Gdf5 genes","volume":"254","author":"Settle","year":"2003","journal-title":"Dev. Biol."},{"key":"ref_15","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":"Page","year":"2021","journal-title":"BMJ"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"82","DOI":"10.1227\/00006123-198307000-00019","article-title":"Spinal Meningoceles in Association with Neurofibromatosis","volume":"13","author":"Whatmore","year":"1983","journal-title":"Neurosurgery"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"463","DOI":"10.1017\/S002221510011984X","article-title":"Cervical meningocele presenting as a neck mass in a patient with neurofibromatosis 1","volume":"106","author":"Freund","year":"1992","journal-title":"J. Laryngol. Otol."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"440","DOI":"10.4103\/jcvjs.jcvjs_20_21","article-title":"Anterior cervical meningocele with craniovertebral junction instability\u2014A case report and literature review","volume":"12","author":"Kumar","year":"2021","journal-title":"J. Craniovertebr. Junction Spine"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"6079","DOI":"10.4081\/ni.2015.6079","article-title":"True Cervicothoracic Meningocele: A Rare and Benign Condition","volume":"7","author":"Pessoa","year":"2015","journal-title":"Neurol. Int."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"40","DOI":"10.4103\/1817-1745.84406","article-title":"Anterior sacral meningocele presenting as constipation","volume":"6","author":"Mohta","year":"2011","journal-title":"J. Pediatr. Neurosci."},{"key":"ref_21","first-page":"842620","article-title":"An asymptomatic large anterior sacral meningocele in a patient with a history of gestation: A case report with radiological findings","volume":"2013","author":"Beyazal","year":"2013","journal-title":"Case Rep. Radiol."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"39","DOI":"10.1001\/jama.1977.03270280041019","article-title":"Anterior sacral meningocele. A presentation of three cases","volume":"237","author":"Anderson","year":"1977","journal-title":"JAMA"}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/14\/21\/7530\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,24]],"date-time":"2025-10-24T07:52:15Z","timestamp":1761292335000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/14\/21\/7530"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,10,24]]},"references-count":22,"journal-issue":{"issue":"21","published-online":{"date-parts":[[2025,11]]}},"alternative-id":["jcm14217530"],"URL":"https:\/\/doi.org\/10.3390\/jcm14217530","relation":{},"ISSN":["2077-0383"],"issn-type":[{"value":"2077-0383","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,10,24]]}}}