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### Data Analysis and Statistics |
Data analysis and plotting were performed in R v. 4.4.0. Agreement heatmaps were created using the seaborn package for Python. Kappa statistics with equal weights as well as simple agreement were used for analysis of interobserver agreement. Additionally majority voting and leave‐one‐out approach were used to compare t... |
### Hardware |
The algorithm training was performed on the high‐performance cluster of the University of Cologne equipped with 12x NVIDIA V100 32Gb graphic cards. All tests and large‐scale inference in prognostic cohorts were performed on the AI server with 4x A100 80Gb NVIDIA cards. |
## Author Contributions |
Y.W. was responsible the data preparation, annotation, data management, data analysis, and manuscript drafting. K.L. prepared and analyzed the data. M.H. managed the clinical data. W.A. prepared the data. S.S., A.B., Y.Y., D.J., X.Z., S.C., A.S., A.Q., A.R., J.F., and A.L.M. analyzed the data. R.B. analyzed the data, s... |
## Ethics Statement |
All study steps were performed in accordance with the Declaration of Helsinki. This study was approved by the Ethical committees of the University of Cologne (22‐1233, 20‐1583) and Kameda General Hospital (22‐094). The need for patient consent was waived as only anonymized, retrospective archive materials were used (no... |
## Conflicts of Interest |
Author Sofia Campelos is an employee of IMP Diagnostics but has no relevant financial or non‐financial interests to disclose. The other authors declare no conflicts of interest. |
## Supporting information |
04bca247ae94a24b08695bbf34a3dbea50eca88aae40f0194bd1b04017e55ba4 pmc12415442.txt |
PMCID: PMC12724472 |
TITLE: Angiomatous Meningioma With Postictal Hemiparesis: Clinical Presentation and Surgical Challenges in a Rare Case |
LICENSE: https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution License CC-BY 4.0., which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. |
ABSTRACT: Angiomatous meningioma (AM) is a rare histological variant of meningioma characterized by prominent vascular proliferation, often posing intraoperative hemostatic challenges. We report the case of a 42-year-old man presenting with recurrent generalized tonic-clonic seizures and postictal right hemiparesis. CT... |
## Introduction |
Meningiomas are the most common primary intracranial tumors, arising from the meninges - the protective membranes surrounding the brain. They comprise a heterogeneous group with multiple histological variants. Among these, angiomatous meningioma (AM) is a rare subtype, accounting for approximately 2.1% of all meningiom... |
According to the 2021 World Health Organization (WHO) classification of central nervous system tumors, meningiomas are divided into 15 histological subtypes, with AM recognized as a distinct but uncommon variant [2,3]. Radiologically, AM often mimics other meningioma subtypes, making accurate preoperative differentiati... |
Certain imaging features, such as marked peritumoral edema and prominent vascularity, may suggest the diagnosis; however, histopathological and immunohistochemical confirmation remains essential [3]. Immunohistochemistry typically demonstrates positivity for epithelial membrane antigen (EMA) and progesterone receptors ... |
Surgical resection remains the mainstay of treatment. However, the rich vascular supply of AM poses a significant intraoperative bleeding risk and increases the likelihood of incomplete resection, which in turn may predispose to recurrence. Despite these challenges, AMs are usually classified as WHO grade I tumors and ... |
Here, we report the case of a 42-year-old man who presented with generalized tonic-clonic seizures followed by right-sided postictal hemiparesis, later found to harbor an AM. This case not only illustrates the diagnostic and surgical challenges posed by this rare subtype but also underscores the feasibility of achievin... |
## Case presentation |
A 42-year-old African male, a high school mathematics teacher and chronic smoker (20 pack-years) with no other comorbidities, presented to the neurosurgical outpatient clinic in August 2025 after his third generalized tonic-clonic seizure in three years. Each episode was followed by transient right hemiparesis. Neurolo... |
The patient reported prior seizures in 2022 and 2024, for which he had presented to the emergency department and was treated for presumptive malaria. No neuroimaging studies were performed at those visits. At presentation, he was started on carbamazepine 200 mg twice daily, omeprazole 40 mg daily, methylprednisolone 4 ... |
Initial laboratory studies revealed mild neutrophilia (absolute neutrophil count: 7.98 × 10⁹/L; reference range: 1.20-7.50 × 10⁹/L) with relative lymphopenia (absolute lymphocyte count: 0.61 × 10⁹/L; reference range: 1.50-4.00 × 10⁹/L). Red blood cell count (4.54 × 10¹²/L; reference range: 4.00-5.40 × 10¹²/L), hemoglob... |
A non-contrast CT scan of the head showed a hypodense left parietal mass with significant perilesional edema, compressing the roof, lateral wall, and posterior horn of the left lateral ventricle, and displacing the cingulate gyrus beneath the falx (Figure 1). No adjacent bone hyperostosis was observed. Contrast-enhance... |
Given the unavailability of MRI and cerebral angiography at this rural hospital, a presumptive diagnosis of a low-grade glial tumor was made based on CT findings. Although referral to a more specialized center was considered, the patient’s socioeconomic circumstances prevented transfer. This lack of access to advanced ... |
The patient underwent a conventional left parietal craniotomy under general anesthesia with the head secured in a three-point headrest. After dural opening, a highly vascular, well-circumscribed lesion was encountered (Figure 3). Careful dissection and meticulous hemostasis allowed for gross total resection, achieving ... |
Postoperative recovery was uneventful. The patient remained in the intensive care unit for 48 hours, where he received IV hydration with 0.9% saline, lansoprazole 30 mg daily, ceftriaxone 1 g twice daily, and levetiracetam 500 mg twice daily. His right-sided weakness gradually improved, and no further seizures were obs... |
This case highlights the clinical course of AM presenting with recurrent seizures and progressive focal neurological deficits. Despite the absence of MRI and cerebral angiography, a timely CT-based diagnosis, intraoperative recognition of a hypervascular lesion, and Simpson grade II resection resulted in complete tumor... |
## Discussion |
AM is a rare histological subtype of meningioma, representing approximately 2% of all cases [4]. It is classified as a WHO grade I tumor, a designation that reflects its generally benign biological behavior and favorable prognosis [5]. These tumors predominantly occur in adults, with a mean age of 50-64 years, and, int... |
Histologically, AM are characterized by a predominance of vascular components, with more than 50% of the tumor volume composed of blood vessels interspersed among meningothelial cells. Nuclear atypia and anaplasia are absent, and the proliferative index (Ki-67) is typically low (~2.4%), further supporting their indolen... |
Radiologically, AM may mimic other hypervascular lesions, such as hemangioblastomas or hemangiopericytomas, because of their prominent vascularity [10]. This overlap underscores the importance of histopathological confirmation, which demonstrates meningothelial cells interspersed among abundant vascular channels. Immun... |
Although most meningiomas are effectively treated with surgical resection, published literature on AM remains limited to small case series and individual reports [8,9]. Gross total resection (Simpson grade I or II) is considered the treatment of choice and is associated with excellent outcomes [7]. Nevertheless, the pr... |
In our case, specific strategies were undertaken to mitigate these risks. Preoperative neuroimaging was carefully reviewed to delineate vascular supply and venous anatomy, facilitating differentiation from other hypervascular tumors. A wide craniotomy was performed to optimize exposure and venous control. Early coagula... |
This case contributes to the limited literature on AM by providing a comprehensive clinical, radiological, and histopathological description of this rare variant while illustrating the unique challenges of managing a hypervascular intracranial tumor in a resource-limited setting. Our experience underscores the importan... |
## Conclusions |
AM is a rare but surgically curable tumor whose marked vascularity can present significant intraoperative challenges. This case illustrates that recurrent seizures with postictal deficits may be the first clue to its diagnosis and should prompt timely neuroimaging. In this setting, where MRI and cerebral angiography we... |
94b844943c8e7baa8610ff0b53b19854b3a0c008d44b1aeb6147d1286c863a13 pmc12724472.txt |
PMCID: PMC12777031 |
TITLE: Cervical dumbbell meningioma exhibiting distinct histopathological subtypes in intradural and extradural components: a case report |
LICENSE: Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Com... |
ABSTRACT: BackgroundSpinal dumbbell meningiomas are rare, accounting for only 4–5% of all spinal meningiomas. Histological heterogeneity within the same dumbbell lesion is even rarer, with all previously reported cases involving coexistence of a meningioma with either a schwannoma or a neurofibroma. To our knowledge, t... |
## Background |
Spinal dumbbell meningiomas are rare, accounting for only 4–5% of all spinal meningiomas [1, 2]. They develop a characteristic dumbbell configuration by extending from the intradural compartment through the intervertebral foramen into the extradural and extraforaminal spaces [3]. This growth pattern presents unique ana... |
## Case presentation |
A 57-year-old man presented with progressive cervical myelopathy and radiculopathy, characterized by MRC Grade 4 strength of the left grasp, numbness over the left thumb and index finger (C6 dermatome), positive Hoffman sign, positive Lhermitte and Spurling signs, hyperreflexia and spasticity in both upper and lower ex... |
The patient underwent a staged microsurgical excision. The first procedure involved a left posterior laminotomy and laminoplasty of C4–C6, along with a left C5/6 facetectomy, to remove the intraspinal portion of the tumour while minimising spinal cord injury. A second surgery via a ventrolateral cervical approach was p... |
The tumour was classified as Eden type III, involving intraspinal, foraminal, and paraspinal (extraforaminal) components [5]. Histopathological examination revealed distinct morphologies between the intradural and extraforaminal tumour components. The intradural specimen obtained during the first-stage surgery was diag... |
The postoperative course was uneventful, with no new neurological deficits observed. The patient’s left-hand grip strength gradually improved within 3 months, while tandem gait and spasticity recovered more slowly but progressively, returning to near-normal by 5–6 months postoperatively. He was able to resume normal da... |
No fusion was performed initially following the C5–6 facetectomy, as both preoperative and postoperative images demonstrated stable alignment with preservation of the contralateral facet and posterior tension band. Spinal stability was subsequently monitored through serial radiographs, which revealed progressive C5–6 s... |
### Discussion and conclusions |
This case illustrates a unique cervical dumbbell meningioma with distinct histological subtypes: intradural transitional meningioma and extraforaminal fibrous meningioma. Complete tumour removal was achieved through a two-stage approach utilising posterior laminotomy and laminoplasty, followed by a ventrolateral cervic... |
Meningiomas are a heterogeneous group of tumours that most commonly arise in the intradural extramedullary compartment of the spinal cord [6]. Dumbbell-shaped meningiomas are rare, with a limited number of cases reported in the literature [1, 2]. Unlike conventional spinal meningiomas which may include higher-grade var... |
[TABLE] Summary of reported cases of spinal dumbbell-shaped tumors with concurrent histological heterogeneity |
Authors/Year | Age/Sex | Levels | Surgical Approach | Staging | Histopathology | Extent of resection | Outcomes |
Present case (2025) | 57/M | C5–6 | Posterior (C4–6 laminotomy and laminoplasty with left C5–6 facetectomy) + ventrolateral approach | Two-stage | Intradural transitional meningioma (WHO I) + extraforaminal fibrous meningioma (WHO I) | GTR (Simpson grade II, complete resection of both components) | Complete neurologic ... |
Zheng et al. (2023) [7] – Case 1 | 53/M | C2 | Posterior (details NR) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (intact subcapsular resection) | Good neurologic recovery; no neurologic deficit |
Zheng et al. (2023) [7] – Case 2 | 47/F | C2 | Posterior (details NR) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (intact subcapsular resection) | Improved, ambulatory with walker at 6 months |
Zheng et al. (2023) [7] – Case 3 | 55/M | C2 | Posterior (details NR) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (intact subcapsular resection) | Neurologic improvement; no neurologic deficit at 2.5-year follow-up |
Zheng et al. (2023) [7] – Case 4 | 61/M | C2 | Posterior (details NR) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (intact subcapsular resection) | Neurologic improvement; no neurologic deficit |
Zhan etal. (2019) [8] | 47/F | C1–C2 | Posterior (left suboccipital craniectomy + C1–2 hemilaminectomy) | Single-stage | Intradural neurofibroma + extradural meningioma (WHO I) | GTR (coagulated and resected the adjacent dura mater) | Good neurologic recovery; no recurrence at 3-month follow-up |
Matsuda et al. (2018) [9] – Case 1 | 53/M | C2 | Posterior (C1–2 laminectomy) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (subcapsular extradural + total intradural removal) | Good neurologic recovery; no neurologic deficit |
Matsuda et al. (2018) [9] – Case 2 | 70/M | C2 | Posterior (right suboccipital craniectomy + C1–2 hemilaminectomy) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR | Good neurologic recovery |
Liebelt et al. (2016) [10] | 58/M | C1–Foramen Magnum | Posterior (right suboccipital craniectomy + C1 hemilaminectomy) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR | Good neurologic condition; no new deficits; discharged POD 2 |
Oichi et al. (2015) [11] | 64/M | C2 | Posterior (C1 hemilaminectomy) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (coagulated and resected the adjacent dura mater) | Occipital pain resolved; no recurrence at 6-month follow-up |
Chen et al. (2014) [12] | 72/F | C3–C4 | Posterior (C3–4 laminectomy + posterior instrumentation) | Single-stage | Intradural meningioma (WHO I) + extradural neurofibroma | GTR (extradural removal with C3 root sacrifice + total intradural removal) | Full neurologic recovery; no neurologic deficit; no recurrence at 12-m... |
Nakamizo et al. (2012) [13] | 49/M | C2 | Posterior (C1 laminoplastic laminotomy + C2 partial laminectomy) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (coagulated the dura mater adjacent to the intradural tumor and resected the dura mater around the C2 root exits) | Full neurologic reco... |
Ogihara et al. (2003) [14] | 54/F | C4–C5 | Posterior (details NR) | Single-stage | Intradural meningioma (WHO I) + extradural schwannoma | GTR (coagulated the dura adjacent to the intradural meningiomas) | Sensory and gait improvement; transient right deltoid/biceps weakness with partial recovery by 24 months |
Hokari et al. (2002) [15] | 59/F | C1–2 | Posterior (details NR) | Single-stage | Intradural meningioma (WHO I) + extradural neurofibroma | NR | Symptoms resolved |
These composite or collision tumours pose considerable diagnostic challenges. On preoperative MRI, they often mimic a single dumbbell-shaped mass, obscuring their underlying histological diversity. Intraoperative discrepancies—such as marked differences in tumour colour, texture, or firmness between compartments—should... |
Histological heterogeneity itself did not correlate with adverse outcomes. Postoperative neurological outcome was generally favourable following gross-total resection (GTR) (Table 1). Surgical management, however, requires balancing the objective of complete resection with preservation of neurological function. Reporte... |
Several theories to explain the rare occurrence of concurrent histologically distinct tumours at the same spinal level have been proposed. The tumour microenvironment theory suggests that varying mechanical pressures and anatomical constraints promote the development of different tumour lineages. The intradural compart... |
Neural microenvironments also differ. While intradural tumours are influenced by central nervous system (CNS)-specific growth factors and cell adhesion molecules, extraforaminal tumours are exposed to peripheral neurotrophic factors and Schwann cell-derived cues [16]. Stromal differences are reflected in CNS-specific f... |
Meanwhile, the common progenitor cell theory posits that both tumours originate from a shared mesenchymal progenitor cell that differentiates along multiple lines in response to specific genetic or environmental cues, particularly when tumours are closely intermingled or exhibit mixed growth patterns [7, 8]. The shared... |
In the current case, the transitional meningioma appeared macroscopically elastic, whereas the fibrous meningioma was firm and adhered to the cord, roots, and surrounding structures. The physical consistency of meningiomas varies according to the subtype. Fibroblastic meningiomas are consistently the firmest subtype. T... |
The posterior approach remains standard for most spinal meningiomas; however, complex lesions may require combined or staged strategies [19]. In our case, a two-stage approach was selected for a dumbbell-shaped Eden type III meningioma characterised by substantial extraforaminal extension, firm fibrous consistency, cha... |
Among previously reported cervical dumbbell meningiomas with histological heterogeneity, most were located at the high cervical levels (C1–C2), with only two exceptions (Table 1). Chen et al. described a C3–C4 tumour that achieved GTR at the cost of C3 nerve root sacrifice but without persistent deficit [12], whereas O... |
A six-month interval between procedures permitted complete wound healing, dural maturation, and confirmation of full neurological recovery to establish a clear baseline for the second operation, while enabling interval imaging to rule out delayed complications such as CSF leakage, haematoma, or cord oedema. This period... |
In conclusion, this case highlights the exceptional histopathological heterogeneity that can occur within dumbbell cervical meningiomas, with distinct transitional and fibrous subtypes identified in the intradural and extraforaminal components, respectively. Successful total resection was achieved using a tailored two-... |
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