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LETTERS TO EDITOR
Year : 2019  |  Volume : 67  |  Issue : 1  |  Page : 281-283

Tumorigenic aneurysmal bleed: Cause of sinister bleed in intracranial metastases


1 Department of Neurosurgery, Postgraduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Radio-diagnosis and Imaging, Postgraduate Institute of Medical Education and Research, Chandigarh, India

Date of Web Publication7-Mar-2019

Correspondence Address:
Dr. Manjul Tripathi
Department of Neurosurgery Postgraduate Institute of Medical Education and Research, Chandigarh - 160 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.253651

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How to cite this article:
Kumar M, Chauhan RB, Tripathi M, Ahuja CK, Mohindra S. Tumorigenic aneurysmal bleed: Cause of sinister bleed in intracranial metastases. Neurol India 2019;67:281-3

How to cite this URL:
Kumar M, Chauhan RB, Tripathi M, Ahuja CK, Mohindra S. Tumorigenic aneurysmal bleed: Cause of sinister bleed in intracranial metastases. Neurol India [serial online] 2019 [cited 2019 Aug 24];67:281-3. Available from: http://www.neurologyindia.com/text.asp?2019/67/1/281/253651




Sir,

Choriocarcinoma is a well-known entity and its metastatic potential is beyond any doubt. Cerebral involvement with choriocarcinoma either as a primary or a metastatic lesion is a rare occurrence, with an incidence of 3%–21.4% and survival rates as low as 35%–60%.[1],[2] Brain metastasis is an important cause of mortality in patients with choriocarcinoma, and its early recognition is mandatory as the cure rates approach nearly 100%. Choriocarcinoma has a predilection for parietal, frontal, and temporal lobe followed by brainstem and cerebellum.[3] Due to its angioinvasive property, choriocarcinoma gets deposited inside the blood vessels weakening tunica media predisposing to aneurysm formation. We report a case of a 20-year-old female patient, who presented with the history of sudden onset headache with left hemiparesis and altered sensorium. On examination, she was unconscious (Glasgow Coma Scale, E2V2M5) with the pupil dilated on the right side and left hemiparesis (2/5). Computed tomography (CT) of the head showed a large right frontoparietal intracerebral hematoma (ICH) [Figure 1]a. CT angiogram of cerebral vessels suggested an arteriovenous malformation (AVM) [Figure 1]b. Right frontoparietal craniotomy and evacuation of hematoma was done. No AVM was found during surgery. The patient regained consciousness 12 h after surgery with improved motor power on the left side (4/5), but she developed a new deficit in the form of right-side hemiplegia. Immediate plain CT scan of head revealed ICH in the left motor cortex remote from the initial site of ICH [Figure 1]c. Digital subtraction angiogram was performed which showed aneurysm in the left middle cerebral artery (M4 segment) [Figure 1]d. Histopathological examination of the earlier evacuated hematoma revealed a cellular tumor predominantly composed of cytotrophoblasts admixed with many syncytiotrophoblasts signifying choriocarcinoma [Figure 2]. Serum β-human chorionic gonadotropin was reported being more than 7 lakhs IU, and reevaluation of history revealed two abortions. The patient improved on conservative management and was discharged with advice to receive chemotherapy. Within 15 days, the patient returned with respiratory distress. Chest X-ray revealed widespread pulmonary metastases with right pleural effusion [Figure 3]. The patient did not consent for any further treatment and left against medical advice. The remote site ICH in our case might be secondary to sudden hemodynamic changes after evacuation of the primary ICH with the semiology similar to perfusion pressure breakthrough seen with AVMs. Diapedesis through increased permeability of parenchymal blood vessels due to a sudden increase in cerebral blood flow combined with defective vascular autoregulation following the removal of any cerebral mass is the most likely mechanism responsible for this situation, as noticed by d'Avella et al.[4] ICH is the most frequent mode of presentation of metastatic choriocarcinoma and accounts for two-third of the cerebral metastases presenting with bleed;[5] the other forms of presentation include cerebral arterial or venous thrombosis.[6] The tumor cells tend to invade the blood vessels causing partial destruction, which can produce neoplastic pseudo-aneurysms, which may dislodge necrotic tumor embolus. In the latest review about brain metastases from gestational neoplasia (GTN),[2] the outcome of patients with brain metastases from GTN was improved with multimodal therapy including craniotomy, whole-brain radiotherapy, and EMA-EP (etoposide and cisplatin with etoposide, methotrexate, and dactinomycin) or EMA-CO [etoposide-methotrexate-actinomycin (dactinomycin)-cyclophosphamide-vincristine] regime. If not recognized timely, brain metastases with choriocarcinoma are associated with a high mortality. There are several reports of cerebral metastasis with bleed,[7] but reviewing the literature, we could only find 19 cases of metastatic choriocarcinoma with aneurysm rupture [Table 1].[3],[5],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23] Twenty patients with cerebral aneurysms associated with metastatic choriocarcinoma reported in the literature were all females, with their age ranging from 18 to 29 years. In all the cases, the aneurysm was located on the distal middle cerebral artery.
Figure 1: (a) NCCT head showing right frontoparietal hematoma; (b) CT angiogram demonstrating contrast blush (vascular pathology ?AVM) as the underlying cause of hemorrhage; (c) postoperative NCCT showing ICH in contralateral motor strip, and (d) postoperative DSA showing ruptured aneurysm (arrow) in the cortical branch of distal anterior cerebral artery (AVM: arteriovenous malformation; DSA: digital subtraction angiogram; ICH: intracerebral hemorrhage; NCCT: noncontrast computed tomography)

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Figure 2: (a) Photomicrograph showing a cellular tumor with areas of hemorrhagic necrosis (hematoxylin and eosin, ×40); (b) predominant cytotrophoblasts with cytoplasmic vacuoles admixed with many multinucleated syncytiotrophoblasts (hematoxylin and eosin, ×200); and (c) tumor cells showing diffuse and strong positivity for beta hCG (immunohistochemistry, ×200) (hCG: human chorionic gonadotropin)

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Figure 3: X-ray chest posterior–anterior view showing bilateral diffuse patchy metastatic infiltration of the lungs at 15 days of follow-up

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Table 1: Reported cases of intracranial aneurysm due to metastatic deposits of choriocarcinoma

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This case also reminds us of the unfailing need of proper history taking and a detailed clinicoradiological correlation plus solidifying the need for sending the clots for biopsy from every intracranial hematoma which has been operated.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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[PUBMED]  [Full text]  
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[PUBMED]  [Full text]  
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Kalafut M, Vinuela F, Saver JL, Martin N, Vespa P, Verity MA. Multiple cerebral pseudoaneurysms and hemorrhages: The expanding spectrum of metastatic cerebral choriocarcinoma. J Neuroimaging 1998;8:44-7.  Back to cited text no. 12
    
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Nakahara T, Nosaka N, Kinoshita K, Matsukado Y. Subarachnoid hemorrhage and aneurysmal change of cerebral arteries due to metastases of choriocarcinoma. No Shinkei Geka 1975;3:777-82.  Back to cited text no. 15
    
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