The use of a covered stent graft for obliteration of high-flow carotid cavernous fistula presenting with life-threatening epistaxis
- 1Department of Neurology, Medical College of Wisconsin and Froedtert Hospital, Milwaukee, Wisconsin, USA
- 2Department of Neurosurgery, Medical College of Wisconsin and Froedtert Hospital, Milwaukee, Wisconsin, USA
- 3Department of Radiology, Medical College of Wisconsin and Froedtert Hospital, Milwaukee, Wisconsin, USA
- Correspondence to Dr O O Zaidat, Associate Professor of Neurology, Radiology and Neurosurgery, Director, Neurointerventional Program, Medical College of Wisconsin and Froedtert Hospital West, 9200 W. Wisconsin Ave, Milwaukee, WI 53226, USA;
- Received 9 July 2009
- Accepted 10 July 2009
- Published Online First 30 October 2009
Background We present a rare complication of trans-sphenoidal adenectomy (TSA) for pituitary macroadenoma: carotid cavernous fistula (CCF) that was treated with endovascular therapy. The incidence of internal carotid artery (ICA) injury following TSA is 1% and may spontaneously heal by packing and rarely manifest as symptomatic CCF/aneurysm. Treatment of post-TSA CCF may be challenging due to the breach of nasal floor and may be prone to recurrence.
Presentation/intervention Uncontrolled intra-operative bleeding during a TSA led to an emergent angiogram to show slow-flow left CCF. Due to clinical deterioration with nasal bleeding, angiography was repeated after 4 h; the fistula had transformed into high flow with significant increase in size, and was therefore embolized using stent-assisted coiling. The fistula recanalized in a month with massive epistaxis and was re-treated using a covered stent graft.
Conclusion This case represents several unique learning points: (1) CCF as a complication of TSA due to close anatomical proximity; (2) the role of endovascular management post-TSA complication; (3) stent-assisted coil embolization of high-flow fistula with moderate ICA laceration; (4) recanalization of CCF causing massive epistaxis; (5) rare use of covered stent graft stent in distal intracranial circulation maintaining integrity and patency of ICA; (6) long-term results after covered stent graft with no in-stent restenosis.
- Trans-sphenoidal adenectomy (TSA)
- carotid cavernous fistula (CCF)
- pituitary macroadenoma
- coil embolization
- Wingspan stent
- endovascular coiling
- Jomed stent
- graft repair
Pituitary macroadenoma accounts for about 10% of all intracranial neoplasm.1 Invasive adenomas may compress and invade surrounding structures and carotid sinus invasion is reported as between 6% and 10% in the literature.2 3 4 The trans-sphenoidal adenomectomy (TSA) is commonly performed because it offers a lower morbidity and mortality and can often remove the tumor without affecting other parts of the brain.5 6 7 8 The procedure still carries certain risks. Here we present a rare complication (∼1%) of the TSA—internal carotid artery (ICA) injury.9 The complication can usually be managed conservatively using packing; although rarely it requires more aggressive management especially with evidently symptomatic carotid cavernous fistula (CCF), aneurysm or massive hemorrhage.10 11 Cases have been reported in the literature for management of carotid injuries using an open surgical approach.12 Here we report the use of emergent endovascular therapy for CCF secondary to acute ICA injury post-TSA.
We encountered the case at our practice where we were consulted and the patient was transferred from operative room to angio-suite for management of lacerated ICA during TSA when hemorrhage could not be controlled with conservative measures. The patient was embolized using stent-assisted coiling with complete obliteration only to return after a month with recanalization and was eventually re-treated with remodeling of the ICA using a covered stent graft stent to augment the integrity of the lumen while maintaining its patency.
A patient in her 50s presented with prolonged atypical headaches and was diagnosed with a large invasive pituitary macroadenoma with left cavernous sinus extension. TSA was planned and performed to remove the tumor. The surgery was initially uneventful; however, toward the end of the resection, brisk arterial bleeding began to occur after bone was removed from the left lateral portion of the exposure. The wound was packed and homeostasis achieved, but removal of the packing continued to show active bleeding. The surgery could not be continued without identifying the cause of the bleeding. Therefore, the sphenoid sinus was liberally packed with Gelfoam (Johnson & Johnson, Somerville, New Jersey, USA) and bilateral Vaseline gauze nasal packs and finger cots and the patient was transported under anesthesia to the neurointerventional suite.
Emergency bilateral carotid artery angiograms were performed immediately. A small dissection of the carotid siphon with left direct carotid cavernous fistula was noted (figure 1A,B). It was felt that treatment was not warranted due to low flow and small size and that it would spontaneously heal. Unfortunately, due to worsening proptosis and chemosis another angiogram was performed after 4 h and selective left ICA angiogram showed left CCF significantly increased in size and flow velocity (figure 1C,D). Stent-assisted coil embolization was performed by the neurointerventional team with complete occlusion using two overlapping Wingspan stent (4.5 mm×20 mm and 4.0 mm×20 mm, Boston Scientific Neurovascular, Fremont, California, USA) and detachable platinum coils (figure 1E,F). The patient continued to improve neurologically while in the hospital. Her proptosis and ptosis decreased. Her vision improved. She was eventually discharged home.
One month after discharge, she presented to the emergency room with acute onset of massive brisk epistaxis and was close to hemorrhagic shock. The epistaxis was controlled with emergency bilateral placement and inflation of balloon catheters in the nasopharynx and fluid resuscitation. She complained of bifrontal headache, diplopia and fatigue. Because of the patient's past medical record, recanalized CCF was suspected.
Selective left ICA angiogram revealed recanalization of the fistula (figure 2A,B). Transarterial and transvenous embolizations were attempted for several hours but failed in spite of adding more coils; finally a Jomed covered stent graft (4.5×16 mm, Jomed, Abbott Labs, Abbott Park, Illinois, USA) was used to reconstruct and restore ICA wall integrity and lumen patency with complete obliteration of the CCF (figure 2C,D). The balloon-mounted Jomed covered stent was placed over 0.14″ Transcend (Boston Scientific Neurovascular) micro guide wire after obtaining triaxial support using 7 French×70 cm Super-Arrow Flex sheath (Arrow International, Reading, Pennsylvania, USA) with 7 French guide catheter into the distal cervical/proximal petrous ICA.
On her most recent clinical and angiographic follow-up at 2.5 years she had no recurrent symptoms, no change on angiography from post-procedure images, no in-stent restenosis or CCF recurrence.
Pituitary adenomas with mass effect usually compress surrounding structures resulting in sellar enlargement and suprasellar extension and carotid sinus invasion is reported at between 6% and 10% in the literature.2 3 4 The high functional importance of carotid sinus with limited surgical exposure lends it to be highly challenging.13 Most pituitary adenoma extends through the medial corridor and may even overlap the intra-cavernous carotid artery in a tongue-like fashion. To access this area it is important to remove the parasellar portion of the medial wall of the cavernous sinus.14 15 Given the close proximity of the medial surface of the ICA to the lateral surface of the pituitary gland (usually varies between 1 mm and 3 mm),16 the carotid artery is likely at risk of injury and potential massive hemorrhage during TSA as described by Kitano et al.11 In reviewing figure 3A, one may notice that the hypophysis is located medially to the carotid artery and cavernous sinus.
We encountered a CCF post-TSA; to our knowledge this has scarcely been reported.12 16 17 Despite the rarity (∼1%),9 vascular injury to the carotid artery during TSA in treating pituitary macroadenoma with invasion to the carotid sinus is anatomically very plausible (figure 3A).
The emergency angiograms showed a barely visible fistula with very low flow and therefore decision was taken not to treat aggressively, and spontaneous thrombosis of the CCF was expected. However, the same fistula enlarged tremendously and the velocity of the flow also increased dramatically in just 4 h. This is likely because the initial angiograms were performed shortly after Gelfoam packing. The molecular components of the Gelfoam may have partially masked the laceration on the ICA or the high degree of packing may also have impeded the flow thus masking the actual impact of laceration till later. Because of this potential association of Gelfoam packing and deceptive initial emergency angiograms, we believe it is more prudent to perform repeated angiograms after the initial emergency ones. In all cases, it should be kept in mind that initial angiograms may not convey the intensity of bleed or the exact degree of the vascular injury and repeat angiograms with close monitoring should be provided to all patients with traumatic vascular injury.
In a first procedure, our patient was treated with stent-assisted coil embolization. While coil embolization of CCF usually results in a high obliteration rate (∼90%),18 rare recanalization does occur. One month after the discharge she recanalized the fistula and presented with massive epistaxis. Several things may have occurred in the interim. The sellar floor integrity was compromised, and was not sealed post-procedure due to the carotid injury, preventing mechanical support to the hemostasis mechanism. Additionally, the patient was on aspirin and clopidogrel, which may have contributed to recurrent CCF and epistaxis.
Our patient then presented at the emergency department with massive uncontrollable epistaxis. While not as common as the classic symptoms, such as exophthalmus, ophthalmoplegia, diplopia and headache, massive epistaxis can be a rare lethal symptom of CCF.19 20 The epistaxis in our case was emergently managed with an inflating balloon catheter bilaterally in nasopharynx.
Following a prolonged attempt at transarterial and transvenous embolization to obtain CCF obliteration, we used a Jomed graft stent, a covered stent, to address the laceration. The Jomed is designed and commonly used in coronary artery graft repairs and vessel perforation. To our knowledge, it has not been used in CCF treatment before. The Jomed uses a completely different system and it was difficult to track the stent to the correct intracranial site; however, the use of the triaxial system provided the needed support. Moreover, the anatomy of the distal left ICA in this patient was favorable and very unusual with straightening of the cavernous angulation when compared with her right ICA (figure 3B). Fewer left cavernous ICA angulations made it relatively more trackable to reach such a distal location. Interestingly, this anatomical variation may have been related to her pituitary tumor, which has left cavernous extensions, or to the laceration itself, which is hard to discern since there is no prior vessel imaging.
While stent restenosis is always a higher concern with a covered stent than with a bare metal stent, it has been suggested that the layer of 75 μm polytetrafluoroethylene graft material may contribute to the decreased in-stent restenosis rate in the Jomed.21 In our case, we had a long-term 2.5-year follow-up angiogram post-Jomed placement showing no signs of in-stent restenosis.
In conclusion, due to the paucity of the literature we contribute this case to underscore the association between TSA and potential complication with CCF with emphasis on early recognition and urgent referral for endovascular cure of the fistula. The emergency angiograms may be deceptive and repeated angiograms should always be performed to correctly assess the degree of vascular injury. Coil embolization of TSA-related CCF may be more susceptible to recanalization; therefore, multimodal embolization perhaps with a liquid embolic agent should be considered. We remind emergency personnel to be more aware that acute uncontrollable epistaxis is a symptom of CCF, albeit rare. Lastly, the Jomed, a covered stent, can be used as a last resort to treat CCF with great result despite the difficulty of positioning the stent in patients with suitable intracranial carotid anatomy. The long-term patency of the covered stent to repair vessel injury needs to be tested; however, our single case report showed no restenosis at the 30 month angiographic follow-up.
Competing interests None.
Ethics approval This study was conducted with the approval of the Medical College of Wisconsin; IRB: Institutional Review Board.
Patient consent Obtained.
Provenance and peer review Not commissioned; not externally peer reviewed.