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LETTER TO EDITOR
Year : 2011  |  Volume : 59  |  Issue : 6  |  Page : 903-904

Chronic encapsulated intracerebral hematoma associated with cavernous angioma: Expression of vascular endothelial growth factor and its receptor


Department of Neurosurgery, National Defense Medical College, Saitama, Japan

Date of Submission18-Jul-2011
Date of Decision14-Aug-2011
Date of Acceptance26-Aug-2011
Date of Web Publication2-Jan-2012

Correspondence Address:
Satoru Takeuchi
Department of Neurosurgery, National Defense Medical College, Saitama
Japan
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DOI: 10.4103/0028-3886.91375

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How to cite this article:
Takeuchi S, Nawashiro H, Wada K, Minamimura K. Chronic encapsulated intracerebral hematoma associated with cavernous angioma: Expression of vascular endothelial growth factor and its receptor. Neurol India 2011;59:903-4

How to cite this URL:
Takeuchi S, Nawashiro H, Wada K, Minamimura K. Chronic encapsulated intracerebral hematoma associated with cavernous angioma: Expression of vascular endothelial growth factor and its receptor. Neurol India [serial online] 2011 [cited 2014 Oct 21];59:903-4. Available from: http://www.neurologyindia.com/text.asp?2011/59/6/903/91375


Sir,

Chronic encapsulated intracerebral hematoma (CEIH) is rare and behaves as a slowly expanding lesion with an insideous onset. [1] This lesion is commonly associated with vascular malformations and only 11 cases of CEIH associated with cavernous angioma (CA) have been reported. [2],[3],[4],[5],[6],[7],[8],[9],[10] Herein, we report an additional case of CEIH with CA and review the literatures.

A 24-year-old male was admitted for a generalized seizure and history of progressive headache of 1-week duration. He was diagnosed to have acute lymphoblastic leukemia for which he received chemotherapy and whole brain radiation at 6 years of age and in remission. Laboratory findings showed a white blood cell count of 6,600 cells/mm 3 , a hematocrit of 45%, and a platelet count of 265,000/mm 3 . Neurological examination showed no abnormality. Computed tomography (CT) scan showed an intracerebral hematoma with peri-lesional edema in right frontal lobe [Figure 1]a. Magnetic resonance imaging revealed a mixed intensity mass lesion with a peripheral hypointense ring [Figure 1]b and c. Cerebral angiography was essentially normal. A right frontal craniotomy was performed and capsule was opened at a depth of 5 mm from the cortex. The cavity was filled with brownish fluid hematoma exhibiting characteristics of a chronic stage. A lobulated mass was also found on the capsule. The capsule, mass, and hematoma were removed in toto. Histological examination confirmed the diagnosis of CA, and also revealed that the capsule consisted of an outer collagenous layer and an inner granulated layer with deposits of hemosiderin, which was compatible with CEIH [Figure 2]a and b. Immunohistochemical analysis revealed increased expression of vascular endothelial growth factor (VEGF) and the VEGF receptor (VEGFR)-1 in the endothelium of blood cells and fibroblasts [Figure 2]c and d. The postoperative course was uneventful and he was discharged without neurological deficit.
Figure 1: a: Computed tomography scan showing an intracerebral hematoma surrounded by edema in the right frontal lobe. T1- (b) and T2- (c) weighted magnetic resonance images revealing a mass lesion as mixed intensities with a peripheral low-signal ring

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Figure 2: Histopathological findings. (a) Specimen taken from the lobulated mass showing vascular channels of various sizes, suggesting cavernous angioma. (b) Specimen of the capsule showing its structure consisting of an outer collagenous layer and an inner granulated layer with deposits of hemosiderin, which was compatible with CEIH. Immunohistochemistry for VEGF (c) and VEGFR-1 (d) revealing positive immunostaining for VEGF in the endothelium of blood cells (arrow heads) and fibroblasts (arrow)

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The clinicoradiological features of all the 12 patients with CEIH associated and CA are summarized in [Table 1]. [2],[3],[4],[5],[6],[7],[8],[9],[10] The clinical features included: Adult onset except on patient younger than 20 years, male gender, high frequency of headache and seizure, high frequency of frontal or parietal lobe location, and presence of peri-lesional edema. Our patient had a past history of acute lymphoblastic leukemia, which can cause intracranial hemorrhage. [11] However, the patient was in remission with chemotherapy and whole brain radiation. Hematological investigations at this admission were normal. In view of these observations, we feel that there is no causal relation between acute lymphoblastic leukemia and CEIH formation.
Table 1: Summary of patients with chronic encapsulated intracerebral hematoma associated with cavernous angioma

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CEIH is characterized by the presence of a fibrotic capsule which histologically resembles the outer capsule of chronic subdural hematoma, and is thought to develop by chronic progressive growth due to repeated bleeding from the new blood vessels in the capsule. [1],[2] The VEGF pathway has also been implicated in CEIH as it can specifically regulate endothelial cell growth and differentiation, which may promote the enlargement of this type of hematoma. [1] VEGF binds two related receptor tyrosine kinases, VEGFR-1 and VEGFR-2. Nakamizo et al. [1] recently reported that VEGF and VEGFR-1 were expressed in specimens of CEIH developing following radiosurgery for arteriovenous malformation. Further, these authors had hypothesized that the VEGF pathway might lead to neovascularization in the granulation layer of the capsule that serves as a continual source of bleeding and perifocal edema. [1] In the present patient we observed immunopathological expression of high levels of VEGF and VEGFR-1, thus suggesting that the VEGF pathway might also contribute to the development of CEIH associated with CA. Additionally, the activation of VEGF pathway may lead to high rates of edema formation, as VEGF can induce vascular leak. [1]

 
 » References Top

1.Nakamizo A, Suzuki So, Saito N, Shono T, Matsumoto K, Onaka S, et al. Clinicopathological study on chronic encapsulated expanding hematoma associated with incompletely obliterated AVM after stereotactic radiosurgery. Acta Neurochir (Wien) 2011;153:883-93.  Back to cited text no. 1
    
2.Masuzawa T, Saito K, Shimabukuro H, Iwasa H, Sato F. Chronic encapsulated hematomas in the brain. Acta Neuropathol 1985;66:24-8.  Back to cited text no. 2
[PUBMED]    
3.Steiger HJ, Markwalder TM, Reulen HJ. Clinicopathological relations of cerebral cavernous angiomas: Observations in eleven cases. Neurosurgery 1987;21:879-84.  Back to cited text no. 3
[PUBMED]    
4.Monma S, Ohno K, Hata H, Komatsu K, Ichimura K, Hirakawa K. Cavernous angioma with encapsulated intracerebral hematoma: Report of two cases. Surg Neurol 1990;34:245-9.  Back to cited text no. 4
[PUBMED]  [FULLTEXT]  
5.Murakami S, Sotsu M, Morooka S, Suzuki T. Chronic encapsulated intracerebral hematoma associated with cavernous angioma: A case report. Neurosurgery 1990;26:700-2.  Back to cited text no. 5
[PUBMED]    
6.Mabuchi S, Kamiyama H, Iwasaki Y, Abe H. Multilocular encapsulated intracerebral hematoma: A case report. No Shinkei Geka 1991;19:677-9.  Back to cited text no. 6
[PUBMED]    
7.Okuno S, Hisanaga M, Miyasaki A, Tsunoda S, Sakaki T. Chronic encapsulated intracerebral hematoma associated with cavernous angioma: Case report and review of the literature. No Shinkei Geka 1993;21:655-9.  Back to cited text no. 7
[PUBMED]    
8.Roda JM, Carceller F, Pérez-Higueras A, Morales C. Encapsulated intracerebral hematomas: A defined entity. Case report. J Neurosurg 1993;78:829-33.  Back to cited text no. 8
    
9.Motegi H, Kuroda S, Ishii N, Aoyama H, Terae S, Shirato H, et al. De novo formation of cavernoma after radiosurgery for adult cerebral arteriovenous malformation-case report. Neurol Med Chir (Tokyo) 2008;48:397-400.  Back to cited text no. 9
[PUBMED]  [FULLTEXT]  
10.Miyahara K, Fujitsu K, Yagishita S, Ichikawa T, Takemoto Y, Okada T, et al. Chronic encapsulated intracerebral hematoma associated with cavernous angioma-case report. Neurol Med Chir (Tokyo) 2011;51:52-5.  Back to cited text no. 10
[PUBMED]  [FULLTEXT]  
11.Chern JJ, Tsung AJ, Humphries W, Sawaya R, Lang FF. Clinical outcome of leukemia patients with intracranial hemorrhage. J Neurosurg 2011;115:268-72.  Back to cited text no. 11
[PUBMED]  [FULLTEXT]  


    Figures

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    Tables

  [Table 1]

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