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Case Report
Revised

Case Report: Microsurgical excision of grade 5 cerebral AVM

[version 2; peer review: 1 approved, 2 approved with reservations]
PUBLISHED 04 Jul 2016
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Abstract

In this case report, we discuss the microsurgical management of a Spetzler-Martin grade 5 arteriovenous malformation (AVM) in a young boy who presented with a hemorrhagic episode and had a high calculated risk of rebleeding. We also outline the rationale for choosing the management option.

Keywords

arteriovenous malformation, AVM, grading, management

Revised Amendments from Version 1

In this revised version, we have included two new figures

  1. Depicting size of the AVM nidus during early and late phase of preoperative angiography.
     
  2. Postoperative angiography revealing complete excision of the nidus with no remaining feeders
We hope these will have significant additive value in the context of our paper.

To read any peer review reports and author responses for this article, follow the "read" links in the Open Peer Review table.

Introduction

As of now, many tenets exist regarding management of high grade cerebral arterio-venous malformation (AVM) management, making a rigid algorithm impossible to create. In experienced hands, microsurgery proved to have better results, compared to other treatments1,2. Herein, we report a microsurgical management of a grade 5 arteriovenous malformation (AVM) in a young patient with a high predicted risk for rebleeding.

Case report

A 22-year-old Brahmin male from Khaireni, a remote village in Nepal, presented to our emergency room with a sudden-onset severe headaches and left-sided weakness over the last 24 hours. Physical examination revealed a Glasgow coma scale (GCS) of 14/15 with left-sided hemiparesis of 3+/5(Medical research council grading). Medical history was significant for a few episodes of paroxysmal headaches since last couple of years, which improved after taking 500 mg Paracetamol tablet on an ‘as needed’ basis. The frequency and intensity of the headache had worsened in the last few months. There was no significant family history. An urgent head computerized tomogram (CT) revealed evidence of a hyperdense lesion with peripheral stippled calcification on the right side in the territory of posterior limb of internal capsule and the retro-thalamic region (Figure 1). There was also coating of vessel along the middle cerebral artery (MCA) territory (Figure 2) and hyperdensity along the deep venous territory. A four-vessel diagnostic carotid angiography revealed Grade 5 Spetzler-Martin AVM in the right sub corticol region with feeders from lenticulostiates of the middle cerebral artery (Figure 3). Drainage was to the deep draining veins and also to the superior sagittal sinus (Figure 4).

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure1.gif

Figure 1. CT showing presence of hyperdense lesion in the right parietal region with stippled calcification on the periphery.

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure2.gif

Figure 2. CT showing presence of coating along the MCA territory.

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure3.gif

Figure 3. Angiogram showing the nidus of the AVM during angiography.

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure4.gif

Figure 4. Angiogram showing the major deep draining vein and a single vein to the superior sagittal sinus.

Multiple factors such as young age at presentation, the fact that the lesion had bled, presentation of patient with deficits associated with the lesion on the non-dominant side, presence of deep venous drainage and intra-nidal aneurysm led to a high calculated risk for rebleeding in the patient. We therefore decided on surgical management, despite the high grade of the lesion. After explanation of the risks of the treatment and role of adjuvants in the form of radiosurgery and embolisation the patient was taken up for microsurgical excision. Since the facility of radiosurgery is not available in the country, we only had the option of embolisation of the feeders prior to the surgical excision of the lesion. However, since the lesion had only low velocity feeders from the lenticulostriate vessels, we opted for direct microsurgical management. After a liberal craniotomy, basal cisterns were opened to gain access to the M1 branch of the MCA. We identified the major deep draining vein that was looping over the MCA bifurcation with the help of Indocyanine Green (ICG) venography. We placed a temporary clip on M1, then made a minimal corticostomy over the parietal cortex and continued our dissection over the gliotic tissue surrounding the AVM taking care of the minimal bleeders with the help of bipolar cauterization and avoiding inadvertent entry to the nidus. Lastly a clip was applied to the draining vein after completely dissecting the AVM nidus. The lesion was finally excised (Figure 5). Complete hemostasis was confirmed.

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure5.gif

Figure 5. Excised AVM nidus.

Postoperatively his blood pressure was rigorously monitored so as not to overshoot the mean arterial pressure above 100 mm of mercury so as to prevent breakthrough perfusion rebleeding. Patient was started on Sodium Valproate (1 gm stat followed by 300 mg IV 8 hourly) and Nimodipine (60 mg 4 hourly via nasogastric tube) for seizure and vasospasm prophylaxis, respectively. Repeat head CT scan the following morning revealed no cavity hematoma or any evidence of vasospasm (Figure 6). Patient was extubated uneventfully. He had hemiparesis of 3+ in upper limbs and 3 in lower limbs. Patient was started on physiotherapy and finally discharged home on the 7TH post-operative day after removal of sutures. Patient came for follow-up 2 weeks later walking on his own with left upper limb weakness of grade 3+/5. The Nimodipine was tapered off in the subsequent three weeks. The patient was advised to continue Na Valproate 300 mg orally three times a day for at least a year. Post operative angiography revealed complete excision of the AVM with no remaining feeders (Figure 7). The patient followed up in the outpatient clinic 6 months later with minimal pronator drift on the left arm and grade 2 spasticity on his left leg.

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure6.gif

Figure 6. Post-operative scan with no haemorrhage or the evidence of vasospasm.

46ee80a0-6e99-4bd0-8049-efc63fd05fc2_figure7.gif

Figure 7. Post operative angiography revealing complete excision of the AVM with no residual feeders left.

Discussion

Bleeding within the AVM is considered a significant predictor of rebleeding. Other important factors moderating risk of rebleeding include deep venous drainage3. Studies have verified that the risk of rebleeding under these circumstances is as high as 34.4% compared to just 0.9% per year in patients without these risk factors4,5. Another important factor to be considered while calculating the risk of rebleeding is the presence of concurrent aneurysm within the AVM (6.93% with aneurysm Vs 3.99% without aneurysm)3.

Up to 40% of cases with AVM manifest neurological deficits6, mostly attributable to hemorrhage. A minority of only 5% to 15% of such deficits are related to factors such as coronary steal phenomenon and venous hypertension79.

The Spetzler-Martin Scale is used to estimate the risk of surgical resection of an AVM with higher grades being associated with greater surgical morbidity and mortality10. Multivariate studies have shown this grading system to reliably predict permanent major morbidity or mortality at the following levels: Grade I (4%), Grade II (10%), Grade III (18%), Grade IV (31%), and Grade V (37%)11. This data has been further validated prospectively, and this grading system remains the most widely used among neurosurgeons and neurointerventionalists12.

Han et al. reported the management of 73 grade 4 and 5 lesions and found the annual hemorrhage rate for untreated lesions to be only 1.5% versus 10.4% for partially treated lesions13. Grade IV or V lesions are only treated in circumstances of progressive neurological deterioration from hemorrhage, vascular steal, or seizure as seen in our case, which had a high risk of rebleeding because of presentation at young age with hemorrhagic episode, large size of the nidus, deep venous drainage pattern and associated aneurysm within the AVM.

There is time-lag of about two years following radiosurgery for complete obliteration of the nidus in the lesion. The risk of hemorrhage in this time period is around 4.8% per year14 which parallels the natural history of the lesion after bleeding. However there is a risk of inadvertent radiation injury to the adjacent eloquent brain area15 and also a risk of symptomatic radiation necrosis in around 9% of cases15,16.

The main indication for other embolisation options in such a high grade of AVM is in order to downgrade the lesion and to minimise the intraoperative blood loss so as to make the lesion amenable for microsurgical excision, which bears an acceptable complication rate of around just 6.5%17. One study has shown that the deep venous drainage, higher grade of the lesions and the periprocedural hemorrhage are predictors of post procedural complications following the embolisation treatment17.

In our case there were only few feeders from the lenticulostriate branches from MCA: not ideal for embolization. Partial embolisation of the lesion will not reduce the risk of hemorrhage to zero3. Partial embolisation of the high grade lesions are only justified in few circumstances, such as in vascular steal phenomenon or an AVM with associated aneurysm18.

Conclusion

In a few selected cases who have a high calculated risk of rebleeding, microsurgical excision remains a therapeutic option even for a high grade AVM especially in centers with limited resources for intervention and radiosurgery. However, all the patients should be well counseled about the available alternative mode of intervention and the associated risks. The management plan in each patient should be tailored addressing factors such as age of the patient, mode of presentation, grade of the lesion, treatment modalities and expertise availability etc.

Consent

Written, informed consent was sought and attained from the father of the patient as per medical protocol in Nepal.

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Version 2
VERSION 2 PUBLISHED 02 Nov 2015
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Munakomi S, Bhattarai B and Cherian I. Case Report: Microsurgical excision of grade 5 cerebral AVM [version 2; peer review: 1 approved, 2 approved with reservations]. F1000Research 2016, 4:1197 (https://doi.org/10.12688/f1000research.7257.2)
NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article.
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Open Peer Review

Current Reviewer Status: ?
Key to Reviewer Statuses VIEW
ApprovedThe paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approvedFundamental flaws in the paper seriously undermine the findings and conclusions
Version 2
VERSION 2
PUBLISHED 04 Jul 2016
Revised
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2
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Reviewer Report 28 Oct 2016
Guo-Yi Gao, Department of Neurosurgery, Shanghai Jiao Tong University, Shanghai, China 
Approved
VIEWS 2
This is a very successful case of surgical removal of high grade AVM which is not ideal ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Gao GY. Reviewer Report For: Case Report: Microsurgical excision of grade 5 cerebral AVM [version 2; peer review: 1 approved, 2 approved with reservations]. F1000Research 2016, 4:1197 (https://doi.org/10.5256/f1000research.9835.r17066)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
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5
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Reviewer Report 21 Sep 2016
Sunil Kumar Singh, Department of Neurosurgery, King George's Medical University, Lucknow, India 
Approved with Reservations
VIEWS 5
I have seen the revised manuscript. I still see that the AVM ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Singh SK. Reviewer Report For: Case Report: Microsurgical excision of grade 5 cerebral AVM [version 2; peer review: 1 approved, 2 approved with reservations]. F1000Research 2016, 4:1197 (https://doi.org/10.5256/f1000research.9835.r16467)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
Views
3
Cite
Reviewer Report 31 Aug 2016
Kenichiro Kikuta, Department of Neurosurgery, University of Fukui, Fukui, Japan 
Approved with Reservations
VIEWS 3
This is an interesting paper but I do not agree that it was grade 5 AVM. The size is probably over 6cm, and deep drainage can also be confirmed, but it is unclear whether this AVM is located in an eloquent ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Kikuta K. Reviewer Report For: Case Report: Microsurgical excision of grade 5 cerebral AVM [version 2; peer review: 1 approved, 2 approved with reservations]. F1000Research 2016, 4:1197 (https://doi.org/10.5256/f1000research.9835.r15967)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
Version 1
VERSION 1
PUBLISHED 02 Nov 2015
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15
Cite
Reviewer Report 22 Mar 2016
Sunil Kumar Singh, Department of Neurosurgery, King George's Medical University, Lucknow, India 
Not Approved
VIEWS 15
It is a well written case report and the discussion is precise. Main points for not approving this article are:
  1. Grading appears faulty as size of nidus, eloquence and drainage are suspicious on single images. more images might have been helpful. The
... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Singh SK. Reviewer Report For: Case Report: Microsurgical excision of grade 5 cerebral AVM [version 2; peer review: 1 approved, 2 approved with reservations]. F1000Research 2016, 4:1197 (https://doi.org/10.5256/f1000research.7819.r12755)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
Views
19
Cite
Reviewer Report 12 Nov 2015
Kenichiro Kikuta, Department of Neurosurgery, University of Fukui, Fukui, Japan 
Approved with Reservations
VIEWS 19
The authors reported a case of S-M grade 5 AVM successfully treated by microsurgical resection. I have some questions about as below.
  1. How did the authors measure the size of AVM. How did they distinguish hematoma from AVM?  Was the AVM
... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Kikuta K. Reviewer Report For: Case Report: Microsurgical excision of grade 5 cerebral AVM [version 2; peer review: 1 approved, 2 approved with reservations]. F1000Research 2016, 4:1197 (https://doi.org/10.5256/f1000research.7819.r10999)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.

Comments on this article Comments (0)

Version 2
VERSION 2 PUBLISHED 02 Nov 2015
Comment
Alongside their report, reviewers assign a status to the article:
Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions
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