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    -  Tripathi M
    -  Singla R
    -  Madan R
    -  Batish A
    -  Ahuja CK
    -  Tewari MK
    -  Mohindra S
    -  Patil NR
    -  Dutta S
    -  Chauhan R
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CASE REPORT
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Primary Hypofractionated Gamma Knife Radiosurgery for Giant Cavernous Sinus Hemangiomas


1 Department of Neurosurgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Radiotherapy, Post Graduate Institute of Medical Education and Research, Chandigarh, India
3 Department of Radiodiagnosis, Post Graduate Institute of Medical Education and Research, Chandigarh, India
4 Department of Anaesthesia and Intensive Care, PGIMER, Chandigarh, India

Date of Submission19-Jan-2021
Date of Decision29-Mar-2021
Date of Acceptance11-Apr-2021
Date of Web Publication31-May-2021

Correspondence Address:
Manjul Tripathi,
Department of Neurosurgery, Post Graduate Institute of Medical Education and Research, Chandigarh
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0028-3886.317234

  Abstract 


Cavernous sinus hemangioma (CSH) are notoriously difficult to excise because of their location, propensity for profuse bleeding during surgery, and relationship to complex neurovascular structures. Radiosurgery offers an alternative treatment modality in cases of small and medium-sized CSH. However, no reports are available in the world literature detailing gamma knife radiosurgery (GKRS) in large (3–4 cm) and giant (>4 cm) CSH. Two patients with giant CSH (Tumor volume was 72.2 and 99.8 cm3, respectively) were treated with frame-based fractionated GKRS (5 Gy × 5 #). The treatment was done with Leksell Perfexion with frame in situ and interfraction interval of 24 h. The tumor was engulfing the optic apparatus, and chiasma could not be separated delineated. The patients were followed at 3 months interval with clinic–radiologic evaluation. Following GKRS, both patients showed remarkable clinical improvement in presenting complaints of headache and visual deterioration. Sixth nerve paresis recovered completely in case no. 1. Significant reduction in tumor volumes (85.1 and 75.6% respectively) was noticed in both the patients at 6 months follow-up radiology. Transient alopecia was noted in case 1 at 3 months follow-up that completely resolved by the 6 months. There was no complication till the last follow-up of 9 months. We report the first account of five fraction frame-based hypo fractionated GKRS for giant CSH. At an interval as short as 3 months, giant CSH shows remarkable clinical improvement. Primary hypofractionated GKRS may be considered an alternative effective modality in these difficult lesions with a favorable safety profile.


Keywords: Complications, cyberknife, hemangioma, neuroradiology, radiosurgery
Key message: Large volume cavernous sinus hemangiomas are safely treated with hypofractionated frame-based gamma knife radiosurgery with a remarkable volumetric reduction in a short span of time. Standalone radiosurgery effectively negates any need of surgical intervention.



How to cite this URL:
Tripathi M, Singla R, Madan R, Batish A, Ahuja CK, Tewari MK, Mohindra S, Patil NR, Dutta S, Chauhan R. Primary Hypofractionated Gamma Knife Radiosurgery for Giant Cavernous Sinus Hemangiomas. Neurol India [Epub ahead of print] [cited 2021 Jun 14]. Available from: https://www.neurologyindia.com/preprintarticle.asp?id=317234




Cavernous sinus hemangioma (CSH) is a rare intracranial extraaxial vascular neoplasm. Microsurgical resection of CSH is fraught with torrential bleeding, high risk of cranial neuropathy, and infrequent risk of death. Large (3–4 cm) and giant (>4 cm) CSH have been treated with microsurgery or radiosurgery as the primary treatment option. There have been several studies showing good responses in small and medium-sized CSH to gamma knife radiosurgery (GKRS) but none for giant CSH.[1] To the best of our knowledge, there is only one series of radiosurgery for giant CSH that describes the role of hypofractionated stereotactic radiosurgery with cyberknife.[2] We present our experience with two cases of giant CSH treated with dose-fractionated GKRS.


  Case Illustrations Top


A 15-year-old male presented with complaints of progressive visual deterioration (blind in the left eye and perception of light in the right eye), diplopia on left lateral gaze (sixth nerve paresis), and holocranial headache. The MRI imaging was suggestive of giant CSH, but computed tomography head showed significant bony destruction and rapid increase in size unlikely for a hemangioma. The patient underwent an endonasal biopsy for the histological diagnosis. Brisk bleeding was encountered, and pathology was suggestive of a Masson's hemangioma of the cavernous sinus. He underwent hypofractionated GKRS in five fractions. At the first follow-up of 3 months, he presented with left temporal alopecia, and complete improvement in sixth nerve paresis [Figure 1], and significant improvement in vision to finger counting at 6 feet in the right eye. CEMRI showed 50.5 and 85.1% reduction in tumor volume with complete decompression of the optic apparatus and medial temporal lobe at 3 and 6 months follow-up, respectively [Figure 2]. At 6 months follow-up, the patient regained complete hair growth [Figure 3].
Figure 1: The left sixth nerve paresis at the time of gamma knife radiosurgery, with complete improvement at 3 months follow-up and persistent effect at 6 months follow-up (Case 1)

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Figure 2: MRI at the time of gamma knife radiosurgery, 3 months, and 6 months follow-up in case 1 with respective volume

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Figure 3: (a) Transient temporal alopecia at 3 months post gamma knife radiosurgery and, (b) complete hair growth at 6 months follow-up in case 1

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Another 43-year-old female presented with headache and progressive visual deterioration for 6 months. Visual acuity was 6/36 in both eyes. Her neurological examination was nonremarkable. The patient had classical imaging findings of giant CSH and was treated with five fractions primary GKRS. At 3 months follow-up, the tumor showed significant reduction in size with decompression of the medial temporal lobe, optic apparatus. There was 53.7 and 75.6% volume reduction at 3 and 6 months, respectively [Figure 4]. No new focal deficits were noted, and serial imaging did not reveal any adverse reaction.
Figure 4: MRI at the time of gamma knife radiosurgery, 3 months, and 6 months follow-up in case 2 with respective volume

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  Treatment Planning Top


We treated both the patients with Leksell Perfexion with Gamma Plan version 10.0 (Elekta Instruments, Norcross, GA, USA). Both the patients gave prior informed consent for the hypofractionated GKRS (approved by Institute Ethics Committee). The patient consented to the publication of his/her image. The aim was to cover the entire tumor while restricting the total radiation delivered to the surrounding neurovascular structures and normal brain parenchyma. The optic apparatus could be partially delineated in both the cases because of the large size of these tumors. With an aim to cover maximum tumor area and minimize radiation fallout to the organs at risk, the tumor margins received a radiation dose of 5 Gy at 50% isodose with forward planning. This was repeated for five fractions with interfraction interval of 24 h. The rationale for decreasing the dose in each fraction to 5 Gy was to minimize the chances of radiation-induced optic neuropathy (RION). Periprocedure steroids were prescribed to both the patients for 5 days. Both the patients tolerated the procedure well.


  Discussion Top


The ideal treatment for large and giant CSH remains under debate with proponents for both microsurgical resection and radiosurgery. While microsurgical resection seems an acceptable treatment option for large CSH, it remains a formidable surgical challenge even among the best hands. Modern microsurgical series report gross total excision rates between 80 and 100% with significant morbidity mortality indices. Sixth nerve palsy is the most common permanent deficit noted in such cases.[3],[4]

There has been a progressive shift to treat CSH by radiosurgery as a primary therapeutic option, especially for small and medium CSH. Ivanov et al.[5] described three cases of CSH, one of which received GKRS as primary treatment. Similarly, Chou et al.[6] described seven cases of CSH with four patients treated with primary GKRS. Both these studies showed good response in terms of tumor shrinkage and long-term persistence of response.

The close proximity of cavernous sinus lesions to optic apparatus, III–VI cranial nerves, pituitary stalk, and medial temporal lobe make them challenging to target in large-sized tumors. Despite the rapid dose fallout in GKRS, the optic apparatus remains at risk due to its low tolerance and the high dose required for vascular lesions. Milano et al.[7] report maximum point doses resulting in <1% RION risk to be 12 Gy in one fraction, 20 Gy in three fractions, and 25 Gy in five fractions. To date, there is only one study of primary stereotactic radiosurgery for large and giant CSH by Wang et al.[2] that reports a median tumor volume reduction of 88.1% (62.3–99.4%) over a median follow-up of 30 months (range 6–78 months) by Cyberknife radiosurgery. They also report significant improvement in CN II, III, IV, V, and VI functions in many cases. They concluded that hypofractionated SRS delivery of 18–22 Gy in 3–4 fractions was effective in reducing tumor volume and improving neurological symptoms without causing any new complications. In our study, the mean tumor reduction rate was 80.4% at 6 months follow-up. No fresh neurological deficits were reported in the patients. Sixth nerve palsy improved in the first case within a month of GKRS. Visual acuity showed significant recovery by 3 months follow-up in both patients. In the short follow-up of the study, the only noted complication was transient temporal alopecia in the first case that has completely resolved by 3 months follow-up.[8] No radiation-induced side effects were noted on MRI imaging at 9 months follow-up.

The hypofractionated GKRS is considered as a treatment option in view of the engulfment of the optic apparatus and the giant size of the lesion. A single session GKRS with the prescription dose of 12–16 Gy exposes surrounding organs at risk (optic apparatus, hypothalamus, etc.) to nonpermissible radiation exposure.[1] Authors have tried five fractions of 5 Gy each regimen to deliver the effective dose while maintaining the radiation exposure to the mentioned OARs in the permissible limits as per the existing literature. We would like to highlight that RION remains a possible complication even with the fractionated regimen and the literature remains fractured in the dose and the length of the exposed portion to predict the chances of RION.[9] A giant CSH pushes the optic apparatus over its capsule rather than its encasement or engulfment. Frequently, it becomes impossible to separately delineate a stretched nerve over the tumor even with sophisticated MR sequences such as CISS. A hypofractionted regimen of 5 Gy with a 24-h interval provides an adequate time frame for the recovery from radiation injury while delivering an effective dose to the target volume. Other cranial neuropathies in cavernous sinus are exceedingly rare even with single-fraction high-dose radiosurgery in view of their radioresistant nature by virtue of them being motor. Seldom, patients may complain of sensory problems in trigeminal nerve distribution, but it largely remains transient and rarely bothersome. Still, such patients should be followed for long term as the safety and complication profile of RION still remain largely nonstandardized. Five-day frame in situ fixation remains another area of criticism against frame-based GKRS. There has been enough literature on its feasibility, safety, and complication profile even from authors' personal published series.[10],[11] With frameless GKRS (ICON), radio surgeons would be more liberal in effectively treating such giant lesions.

Limitations

This report highlights only two cases of giant CSH with 9 months follow-up. It definitely needs long-term evaluation for continued efficacy and safety profile, especially for optic apparatus. The hypofractionated regimens with radiosurgery are still evolving, and we are trying to achieve the best risk-benefit ratio adhering to the Noren's policy. Our aim was to highlight the feasibility and efficacy of frame-based hypofractionated GKRS for the lesions in the close vicinity of the optic apparatus.


  Conclusion Top


While GKRS is a well-established primary treatment modality for small and medium-sized CSH, its role in giant CSH has yet remained undefined. We report the first series of giant CSH successfully treated with fractionated GKRS. Improvement in clinical signs, symptoms, and radiological response is seen as early as 3 months in these cases. Such patients need long-term follow-up for further identification of safety and complication profile.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Tripathi M, Batish A, Kumar N, Ahuja CK, Oinam AS, Kaur R, et al. Safety and efficacy of single-fraction gamma knife radiosurgery for benign confined cavernous sinus tumors: Our experience and literature review. Neurosurg Rev 2020;43:27-40.  Back to cited text no. 1
    
2.
Wang X, Zhu H, Knisely J, Mei G, Liu X, Dai J, et al. Hypofractionated stereotactic radiosurgery: A new treatment strategy for giant cavernous sinus hemangiomas. J Neurosurg 2018;128:60-7.  Back to cited text no. 2
    
3.
Linskey ME, Sekhar LN. Cavernous sinus hemangiomas. Neurosurgery 1992;30:101-7.  Back to cited text no. 3
    
4.
Suri A, Ahmad FU, Mahapatra AK. Extradural transcavernous approach to cavernous sinus hemangiomas. Neurosurgery 2007;60:483-9.  Back to cited text no. 4
    
5.
Ivanov P, Chernov M, Hayashi M, Nakaya K, Izawa M, Murata N, et al. Low-dose gamma knife radiosurgery for cavernous sinus hemangioma: Report of 3 cases and literature review. Minim Invasive Neurosurg 2008;51:140-6.  Back to cited text no. 5
    
6.
Chou C-W, Wu H-M, Huang C-I, Chung WY, Guo WY, Shih YH, et al. Gamma knife surgery for cavernous hemangiomas in the cavernous sinus. Neurosurgery 2010;67:611-6.  Back to cited text no. 6
    
7.
Milano MT, Grimm J, Soltys SG, Yorke E, Moiseenko V, Tomé WA, et al. Single- and multi-fraction stereotactic radiosurgery dose tolerances of the optic pathways. Int J Rad Onc Biophysics 2018:S0360301618301251. doi: 10.1016/j.ijrobp. 2018.01.053.  Back to cited text no. 7
    
8.
Tripathi M, Buddhiraja M, Kumar N, Batish A, Ahuja CK, Kamboj P, et al. Temporary noncicatricial focal alopecia following Gamma knife radiosurgery: Case series and review of literature. Neurol India 2018;66:1469.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Tripathi M, Ahuja C, Mukherjee K, Kumar N, Dhandapani S, Dutta P, et al. The safety and efficacy of bevacizumab for radiosurgery-Induced steroid-resistant brain edema; not the last part in the ship of theseus. Neurol India 2019;67:1292.  Back to cited text no. 9
    
10.
Mukherjee K, Kumar N, Tripathi M, Oinam AS, Ahuja CK, Dhandapani S, et al. Dose fractionated gamma knife radiosurgery for large arteriovenous malformations on daily or alternate day schedule outside the linear quadratic model: Proof of concept and early results. A substitute to volume fractionation. Neurol India 2017;65:826.  Back to cited text no. 10
    
11.
Tripathi M, Rekhapalli R, Batish A, Kumar N, Oinam AS, Ahuja CK, et al. Safety and efficacy of primary multisession dose fractionated gamma knife radiosurgery for jugular paragangliomas. World Neurosurg 2019;131:e136-48.  Back to cited text no. 11
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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