Abstract
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.