| Article Access Statistics|
| Viewed||2105 |
| Printed||37 |
| Emailed||0 |
| PDF Downloaded||35 |
| Comments ||[Add] |
Click on image for details.
|LETTERS TO EDITOR
|Year : 2019 | Volume
| Issue : 1 | Page : 281-283
Tumorigenic aneurysmal bleed: Cause of sinister bleed in intracranial metastases
Manish Kumar1, Ravi Bharatbhai Chauhan1, Manjul Tripathi1, Chirag K Ahuja2, Sandeep Mohindra1
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 Publication||7-Mar-2019|
Dr. Manjul Tripathi
Department of Neurosurgery Postgraduate Institute of Medical Education and Research, Chandigarh - 160 012
Source of Support: None, Conflict of Interest: None
|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
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%., 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. 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. ICH is the most frequent mode of presentation of metastatic choriocarcinoma and accounts for two-third of the cerebral metastases presenting with bleed; the other forms of presentation include cerebral arterial or venous thrombosis. 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), 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, but reviewing the literature, we could only find 19 cases of metastatic choriocarcinoma with aneurysm rupture [Table 1].,,,,,,,,,,,,,,,,, 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)|
Click here to view
|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)|
Click here to view
|Figure 3: X-ray chest posterior–anterior view showing bilateral diffuse patchy metastatic infiltration of the lungs at 15 days of follow-up|
Click here to view
|Table 1: Reported cases of intracranial aneurysm due to metastatic deposits of choriocarcinoma|
Click here to view
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
Conflicts of interest
There are no conflicts of interest.
| » References|| |
Evans A, Soper J, Clarke-Pearson D, Berchuck A, Rodriguez G, Hammond C. Gestational trophoblastic disease metastatic to the central nervous system. Gynecol Oncol 1995;59:226-30.
Piura E, Piura B. Brain metastases from gestational trophoblastic neoplasia: Review of pertinent literature. Eur J Gynaecol Oncol 2014;35:359-67.
Giannakopoulos G, Nair S, Snider C, Amenta PS. Implications for the pathogenesis of aneurysm formation: Metastatic choriocarcinoma with spontaneous splenic rupture. Case report and a review. Surg Neurol 1992;38:236-40.
d'Avella D, Cicciarello R, Albiero F, Andrioli G. Scanning electron microscope study of human arachnoid villi. J Neurosurg 1983;59:620-6.
Vaughan HG, Howard RG. Intracranial hemorrhage due to metastatic choriocarcinoma. Neurology 1962;12:771-7.
Cyriac S, Sagar TG, Mahajan V. Choriocarcinoma with arterial and venous thrombosis. Neurol India 2009;57:505-7.
] [Full text]
Suresh TN, Santosh V, ShastryKolluri VR, Jayakumar PN, Yasha TC, Mahadevan A, et al
. Intracranial haemorrhage resulting from unsuspected choriocarcinoma metastasis. Neurol India 2001;49:231.
] [Full text]
Bryce W, Nell M, Bruce M. lntracranial vascular complications of choriocarcinoma. Neurosurgery 1978;2:138-42.
Fujiwara T, Mino S, Nagao S, Ohmoto T. Metastatic choriocarcinoma with neoplastic aneurysms cured by aneurysm resection and chemotherapy. Case report. J Neurosurg 1992;76:148-51.
Hove B, Andersen BB, Christiansen TM. Intracranial oncotic aneurysms from choriocarcinoma. Case report and review of the literature. Neuroradiology 1990;32:526-8.
Huang CY, Chen CA, Hsieh CY, Cheng WF. Intracerebral hemorrhage as initial presentation of gestational choriocarcinoma: A case report and literature review. Int J Gynecol Cancer 2007;17:1166-71.
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.
Momma F, Beck H, Miyamoto T, Nagao S. Intracranial aneurysm due to metastatic choriocarcinoma. SurgNeurol 1986;25:74-6.
Montant J, Hepner H, Tridon P, Picard L, Floquet J, Lepoire J. Aspects pseudovasculaires des mdtastases intracraniennes des choriodpithdliomes. Neurochirurgie 1971;17:119-28.
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.
Noterman J, Verhest A, Baleriaux D, Brotchi J. A ruptured cerebral aneurysm from choriocarcinomatous origin—A case report and a review. Neurosurg Rev 1989;12:71-4.
Olmsted WW, McGee TP. The pathogenesis of peripheral aneurysm of the central nervous system: A subject review of the APFIP. Radiology 1977;123:661-6.
Pullar M, Blumbergs PC, Phillips GE, Carney PG. Neoplastic cerebral aneurysm from metastatic gestational choriocarcinoma. J Neurosurg 1985;63:664-7.
Seigle JM, Caputy AJ, Manz HJ, Wheeler C, Fox JL. Multiple oncotic intracranial aneurysms and cardiac metastases from choriocarcinoma: Case report and review of the literature. Neurosurgery 1987;20:39-42.
Shuangshoti S, Panyathanya R, Wichienkur P. Intracranial metastases from unsuspected choriocarcinoma. Neurology 1974;24:649-54
Stilp TJ, Bucy PC, Brewer JI. Cure of metastatic choriocarcinoma of the brain. JAMA 1972;221:276-9.
Weed JC, Hammond CB. Cerebral metastatic choriocarcinoma: Intensive therapy and prognosis. Obstet Gynecol 1980;55:89-94.
Weir B, MacDonald N, Mielke B. Intracanial vascular complications of choriocarcinoma. Neurosurgery 1978;2:138-42.
[Figure 1], [Figure 2], [Figure 3]