Background and Importance
Association between cranial meningiomas and intracranial vascular malformations (aneurysms and arteriovenous malformations (AVMs)) is a very rare occurrence. From a Pubmed-MEDLINE review, 50 cases of AVMs associated with primary intracranial tumors have been previously reported. Among these, only 14 cases involved the association between AVMs and meningiomas (1). Coexistence of an aneurysm and a meningioma was shown approximately in 100 cases (2). The association between AVMs and aneurysm according to their similar vascular nature is well known. The case we reported on is the first in English literature since the only other case in the whole literature was published in Japanese. 

Case Presentation
A 62-year-old man was admitted to our hospital presenting with confusion, ideomotor slowing down and right hand tremor accentuated with the efforts. These symptoms appeared about two months before admission, and worsened gradually. In patient's medical history, the presence of chronic obstructive pulmonary disease (COPD) was detected, and arterial hypertension with previous myocardial infarction (MI), diabetes, and the presence of a pulmonary lesion were diagnosed during hospitalization. At neurological examination, the patient was cooperative, slightly confused with Glasgow coma scale (GCS) =15. We did not find any evident neurological deficit, except for an ideomotor slowing down. Also, encephalic nerves did not show any evident deficit.

Pre-operative Neuroimanging
An MRI with gadolinium showed a right massive extra-axial lesion compressing the fronto-temporal parts of cerebrum and the right cerebral peduncle displacing the ventricular system (Figures 1.A-B). In its posterior side, the lesion was in contact with an AVM. The AGF revealed that the AVM’s nidus had a diameter of three centimeters with the feeders coming from right middle cerebral artery (MCA) and anterior choroidal artery (AChA) and two large venous drainage areas from the vein of Labbé and the vein of Trolard from the same side (Figures 1.C-D), thus configurating an AVM Spetzler-Martin 2. The angiography had also revealed an aneurysm located near the rise of the ophthalmic artery with a diameter of two millimeters (Figures 1.C-D).

 

Surgery 
The patient was placed in supine position with his head turned to the left. Afterwards, a point mark skin incision was made and a large fronto-temporal craniotomy was performed. Then, the dura was opened and a large meningioma occupying large part of the craniotomy perimeter was found. Total removal of the lesion was performed after isolation of the sylvian artery with the help of microdoppler. Subsequently, we exposed the AVM, by transuncal approach. The AVM was then isolated according to the Hashimoto technique and removed. Due to the very small dimension of the aneurysm, the lesion was not treated. 

Post-operative Course
After surgery, the patient showed a progressive improvement of the pre-operative symptoms. The post-operative examination displayed a complete removal of the lesions, meningioma and AVM (Figures 2-3). The post-operative imaging discovered a neoplastic lesion of the right pulmonary apex whereby he is now following a specific therapeutic procedures.
 

 

We reviewed PubMed and Medline publications about the association between meningiomas, AVMs and aneurysms. Therefore, we decided to exclude the association between only two lesions. Our inclusion criteria regarded only patients that presented all three lesions at diagnosis.  These publications were analyzed by patient age, site of lesion, and treatment options.

Discussion
We found only another case, the one described by Fukawa et al. in 1977 (3). However the article is in Japanese (Table 1). Considering the rarity of the case, there is no possibility of a pathophysiology correlation among all these lesions. The contemporary presence of an AVM and aneurism is frequent. The hemodynamic alterations, caused by the arteriovenous shunt, are widely recognized as triggers (4,5). The simultaneous presence of AVM and meningiomas is instead very rare. There are only 14 cases (1) described in literature, excluding our observation. The reason of this association is not clear, although there are some hypotheses. Cushing and Heisenhardt were the first to suggest that chronic flogosis of the arachnoid cells with the typical high-flow rate on arteriovenous shunt of AVMs can be the key factor to facilitate the arising of benign brain tumors (6). Another interesting theory is based on the central pathophysiological role of AVM: Kasanticul et al. speculated that tumor angiogenesis factor (TAF), plus other cytokines released from AVM cells pose a favorable environment for tumor growing (7).
 

 

Conclusion
Our case is the first report in English and the second in the whole literature. However, it is quite hard to find a pathophysiological association among these three lesions. The main reason is the lack of literature on it. There is no doubt about the reason why an AVM and an aneurysm can be found together. The notion neurosurgeons have to be rather focused on is the simultaneous presence of AVM and meningioma. 

None.

Conflicts of Interest
The authors report no conflicts of interest concerning the materials or methods used in this study or the findings specified in this paper.


References