• Huiyang Zhang, Xiang Li, and Yan Jiang.
• Department of Radiology, The Central Hospital of Wuhan, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, P.R. China.
• Medicine (Baltimore). 2025 Jan 31; 104 (5): e41294e41294.

RationalePrimary ectopic meningiomas (PEMs) are extremely rare, with limited literature available on the subject. Understanding their clinical and radiological characteristics is crucial for accurate diagnosis and treatment.Patient ConcernsThis study presents a case of a multicentric PEM involving the lungs and cranial bone barrier, aiming to provide new insights into its clinical and diagnostic features.DiagnosesA 46-year-old woman with no prior history of intracranial meningiomas or other tumors was found to have multiple lobulated nodules in the lungs during a routine physical examination. Computed tomography scans revealed well-defined lesions with mild to moderate heterogeneous enhancement. Magnetic resonance imaging showed a lesion at the cranial bone barrier, which presented as a high-signal area on T2-weighted FLAIR images and mild to moderate enhancement on T1-weighted images. The magnetic resonance spectroscopy displayed a broad Lip peak but lacked N-acetylaspartate or creatine peaks. Histopathological and immunohistochemical analyses confirmed the diagnosis of multicentric PEMs.InterventionsWe performed surgical resection of the lesion on the cranial bone plate and conducted follow-up examinations for the multiple lesions in the lungs.OutcomesThis case highlights the diagnostic challenges of multicentric PEMs involving the lungs and cranial bone barrier. Due to their extremely low incidence and nonspecific clinical manifestations, a comprehensive evaluation combining radiological, pathological, and immunophenotypic data is essential for accurate diagnosis.LessonsThis case underscores the importance of a thorough, multidisciplinary approach to diagnosis and treatment and provides valuable insights for managing similar rare cases.Copyright © 2025 the Author(s). Published by Wolters Kluwer Health, Inc.

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