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Sporadic meningioangiomatosis with and without meningioma: analysis of clinical differences and risk factors for poor seizure outcomes

  • Clinical Article - Brain Tumors
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Abstract

Background

Meningioangiomatosis (MA) is a rare cerebral lesion. Sporadic MA occasionally combines with meningioma (MA-M). The aim of the present study was to clarify whether MA-M and pure MA have clinical differences and to determine risk factors for unsatisfactory seizure outcomes in sporadic MA.

Methods

We reported 14 sporadic MA cases in our center and conducted a literature review. We compared the demographic, clinical, imaging, electrophysiological and pathological features and surgical outcomes. Logistic regression analysis was performed to evaluate the risk factors for poor seizure outcomes.

Results

MA-M cases showed a more prominent male predilection (4.2 times vs. 1.6 times, p = 0.04), a shorter duration of symptoms (2.8 ± 0.8 years vs. 5.2 ± 0.6 years, p = 0.02), and a lower seizure incidence (53.6 % vs. 89.3 %, p < 0.001) as compared to pure MA. A gyriform alteration on imaging was exclusively associated with pure MA. The Ki-67 was higher in the meningioma component than in the MA component in MA-M (1.2 ± 0.3 % vs. 6.1 ± 1.1 %, p < 0.001). Lesions located in the temporal lobe predicted poor seizure outcomes (p = 0.02, OR = 4.4, 95 % confidence interval, 1.24–15.89).

Conclusion

Clinical differences may be caused by the different biological natures. MA-M seems to be a neoplastic lesion, while pure MA seems to be a non-neoplastic lesion. Long-term follow-up is required for MA-M. Because the coexistence of hippocampal sclerosis may explain the poor seizure outcomes of MA located in the temporal lobe, it is important to identify underlying hippocampal sclerosis and to perform complete resection.

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Acknowledgments

This project was funded by grants from a high-level specialist training project of the Beijing Health Bureau (no. 2011-3-029) and Beijing Municipal Administration of Hospitals Clinical Medicine Development of Special Funding (no. ZYLX201305).

Conflicts of interest

All authors certify that they have no affiliations with or involvement in any organization or entity with any financial interest (such as honoraria; educational grants; participation in speakers’ bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and expert testimony or patent-licensing arrangements) or non-financial interest (such as personal or professional relationships, affiliations, knowledge or beliefs) in the subject matter or materials discussed in this manuscript.

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Authors and Affiliations

  1. Department of Neurosurgery, Beijing Tian Tan Hospital, Capital Medical University, No. 6, Tian Tan Xi Li, Beijing, 100050, China

    Chao Zhang, Xiu Wang, Jian-Guo Zhang & Kai Zhang

  2. Stereotactic and Functional Neurosurgery Laboratory, Beijing Neurosurgical Institute, Capital Medical University, Beijing, 100050, China

    Chao Zhang, Jian-Guo Zhang, Wen-Han Hu & Kai Zhang

  3. Beijing Key Laboratory of Neurostimulation, Beijing, 100050, China

    Chao Zhang, Jian-Guo Zhang, Wen-Han Hu & Kai Zhang

  4. Beijing Jingmei Group General Hospital, Beijing, 102300, China

    Yao Wang

  5. Department of Pathology, Beijing Neurosurgical Institute, Capital Medical University, Beijing, 100050, China

    Jing-Jun Li

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  1. Chao Zhang
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Correspondence to Kai Zhang.

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Zhang and colleagues analyzed the clinical and morphological data and treatment results of 14 patients affected by a rare cerebral disease characterized by a leptomeningeal meningothelial proliferation called “meningioangiomatosis.” Such meningeal abnormality was identified by the authors in seven cases as an isolated disease and in seven further cases associated with a meningioma. The authors also analyzed 160 similar cases reported in the literature to date. According to the results presented in the study, meningioangiomatosis should be considered a non-neoplastic lesion that may, however, be associated with epilepsy. Nonetheless, the contextual presence of other epileptogenic foci (i.e., mesiotemporal sclerosis) is common, and some of the lesions may be associated with gyriform alteration or enhancement. Accurate epilepsy preoperative assessment is therefore mandatory to gain treatment success. Meningiomas associated with menigioangiomatosis are typical neoplastic lesions, requiring standard follow-up. The study provides interesting information on the histopathological features of these lesions and the differences from meningiomas. On such bases, it can be suggested that when meningioangiomatosis is associated with meningiomas, these tumors could arise from the non-neoplastic meningioangiomatosis lesions by additional genetic alterations, which may confer a greater proliferation potential.

Alfredo Conti

Messina, Italy

Chao Zhang and Yao Wang contributed equally to this work

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Zhang, C., Wang, Y., Wang, X. et al. Sporadic meningioangiomatosis with and without meningioma: analysis of clinical differences and risk factors for poor seizure outcomes. Acta Neurochir 157, 841–853 (2015). https://doi.org/10.1007/s00701-015-2375-y

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