2023 Volume 10 Pages 21-25
A 78-year-old man underwent a tracheostomy after embolization for a dural arteriovenous fistula. Seventy days after tracheostomy, arterial bleeding appeared through the tracheal stoma. The bleeding stopped spontaneously. However, two days later, arterial bleeding reappeared, and he was diagnosed with a tracheo-innominate artery fistula (TIF). He then underwent urgent endovascular covered stent placement. After the procedure, there was no bleeding. TIF can be a fatal complication after tracheostomy and it is generally treated with open chest surgery. However, a successful endovascular treatment for TIF has recently been reported and may yield better results.
Tracheo-innominate artery fistula (TIF) is a rare complication after tracheostomy that is not well known to neurosurgeons, although neurosurgeons who frequently care for patients after tracheostomy may be aware of this condition. A TIF can form when the tracheal cuff comes into prolonged contact with the tracheal wall, which causes an ulcer and a fistula to form between the tracheal wall and the innominate artery, resulting in massive hemorrhage. TIF occurs in 0.1%-1% of post-tracheostomy cases, and once it occurs, the prognosis is poor, with a mortality rate of almost 100% if no surgical treatment is provided.1,2) Traditionally, TIF is treated by median sternotomy and ligation of the innominate artery, but recently, the development of endovascular treatment has been reported.
We report a case of TIF that was successfully treated with the placement of a covered stent.
A 78-year-old man with a history of embolization for a dural arteriovenous fistula underwent tracheostomy because of postoperative prolonged disturbance of consciousness and repeated pneumonia. Seventy days after the tracheostomy, arterial bleeding suddenly appeared through his tracheal stoma. He was taking apixaban 10 mg/day at the time for arterial fibrillation. Computed tomography angiography (CTA) showed no active bleeding source, but the tip of the tracheal tube was flushed with the innominate artery (Fig. 1A, B). We consulted an otorhinolaryngologist, and a TIF was suspected because of the position of the tip of the tracheal tube and the innominate artery. Nasopharyngolaryngoscopy revealed granulation on the tracheal wall near the tip of the tracheal cannula. Bleeding from the granulation was suspected, but the possibility of a TIF remained. Because the bleeding had stopped spontaneously, the depth of the tracheal cannula was adjusted with a movable cannula (Adjust Fit; Fuji Systems, Tokyo, Japan) so that the tip of the cannula did not contact the bleeding site and the cuff could compress the site.
CT angiography (CTA) at the first bleeding and the second bleeding.
At the first bleeding, CTA did not show any extravasation (A). The tip of the tracheal cannula is touching the innominate artery (arrow: the contact point of the tip of the tracheal cannula and the innominate artery) (B). Two days later, a second bleeding occurred, and CTA still showed no obvious extravasation (C). The position of the tip of the tracheal cannula was adjusted so as not to contact the innominate artery (arrow: the tip of the tracheal cannula) (D).
Two days later, arterial bleeding reappeared. CTA was performed again, but it still revealed no obvious extravasation. The position of the tip of the cannula was adjusted so as not to approach the innominate artery (Fig. 1C, D). The cuff was hyperinflated, and compression hemostasis was attempted, but hemostasis could not be achieved. We diagnosed a TIF from the clinical findings and performed urgent endovascular treatment.
Under general anesthesia, the right brachial artery was exposed, and a 12 Fr long sheath (W. L. Gore & Associates, Flagstaff, AZ, USA) was inserted. A 4 Fr long sheath was inserted from the right femoral artery, and control aortography was performed with a 4 Fr pigtail catheter positioned in the ascending aorta to confirm the origin of the innominate artery, the bleeding point, and the origin of the common carotid artery. A covered stent made of polytetrafluoroethylene (PTFE) (VIABAHN 13*50 mm; W. L. Gore & Associates) was inserted from the right brachial artery and deployed to securely cover the fistula point and avoid the orifice of the common carotid artery. The stent protruded slightly into the aorta, and a small amount of endoleak was observed. However, the attachment to the arterial wall appeared secure, and the procedure was completed. Bleeding disappeared immediately after stent placement (Fig. 2). The use of VIABAHN for the innominate artery was approved by the ethics committee of Iwakuni Clinical Center (Approval number: 0454).
Endovascular procedure.
A 12 Fr long sheath and guide wire were inserted from the right brachial artery, and a pigtail catheter was inserted from the right common femoral artery. Diagnostic imaging showed the entrance of the right brachiocephalic innominate artery and no obvious bleeding source (A). A covered stent made of polytetrafluoroethylene (VIABAHN; 13 mm × 50 mm) was placed so that it did not reach the origin of the common carotid artery and so that it covered the fistula definitively (arrow: distal end of covered stent; arrowhead: proximal end of covered stent) (B, C, D).
Cefazolin was administered to prevent postoperative infection. As an antithrombotic therapy, 100 mg aspirin per day was given to prevent a thromboembolism, and the patient continued to take 10 mg apixaban per day as before the procedure. Two months after stent placement, the patient developed pneumonia and died as a result, but rebleeding never recurred.
TIF is an emergency condition, so prompt diagnosis and management are essential.
Imaging diagnosis such as CTA, angiography, or bronchoscopy often shows no signs of TIF. Sensitivity is reported to be only 20%-30%, so it may be difficult to diagnose TIF by such imaging.3) Over 50% of patients present sentinel bleeding before fatal hemorrhage.2) At this point, the possibility of TIF should be considered, even if there are no other signs of TIF, and fatal bleeding should be prepared for. In our case, we were unable to diagnose TIF by CTA, but we prepared a covered stent for the next bleeding and treated it successfully. The first goal of initial management is to achieve temporary hemostasis and to establish an airway. Hyperinflating the tracheostomy cuff is an effective way to compress the bleeding site, which can provide bleeding control in approximately 70% of cases.2) Digital compression is also effective for compressing the bleeding site.4,5) Bronchoscopy through the tracheostomy is a good way to secure an airway by suctioning blood, and this sometimes allows the physician to confirm the bleeding point.2) In this case, however, these management techniques were temporary, and radical treatment was necessary.
Traditionally, ligation of the innominate artery has been performed as a curative procedure for TIF. This involves having a median sternotomy, cutting the innominate artery, and achieving hemostasis, and sometimes, it requires revascularization by bypass surgery.1,2) Although the direct repair of the artery provides reliable hemostasis, it must be performed under general anesthesia, and open surgery is difficult to perform on unstable patients in emergency situations.1,6) The post-surgical mortality rate is reported to be over 50%,7) and complications such as cerebral ischemia and ischemia of the right upper extremity can be problematic. Previous reports indicate that cranial nerve complications after the ligation of the innominate artery occur in approximately 10% of patients.4) If a patient has severe atherosclerotic changes in the common carotid arteries or the vertebral arteries, the risk of neurologic sequelae can be higher.8) It is also reported that there is no need for revascularization if the stamp pressure at the end of the right common carotid artery is 50 mmHg or greater.9) To avoid the risk of neurological complications, preoperative vascular evaluation or intraoperative stamp pressure measurement can be helpful. Another problem is that emergency surgery is difficult in patients with a history of open chest surgery due to postoperative adhesions.6)
The usefulness of endovascular treatment has been pointed out in recent years. The treatment modality is mainly a covered stent. Endovascular therapy is less invasive than open chest surgery, and it can be performed under local anesthesia. One problem with stent placement is the risk of rebleeding compared to the ligation of the innominate artery. Another problem is that the fistula is an unclean wound, and the insertion of a stent carries the risk of wound infection1,6) (Table 1). According to a literature review by Taechariyakul et al., who compared the results of endovascular surgery to sternotomy, the clinical success rate of each intervention was 76% versus 69%, the post-procedure 30-day survival rate was 70% versus 40%, and the post-procedure complication rate was 30% versus 60%, respectively, indicating that endovascular treatment is superior to direct surgery in these three regards.6) The first endovascular treatment case was reported by Deguchi et al. in 2001,10) and 49% of the sternotomy cases in the literature review were reported before 2000.6) Therefore, the outcomes could have been affected by differences in the historical background, but their findings clearly suggest the effectiveness of endovascular treatment for TIF.
| Endovascular treatment | Open chest surgery | No surgical treatment | |
|---|---|---|---|
| Advantages | ・Able to perform on unstable patient | ・Certain hemostasis ・Less infection |
|
| Disadvantages | ・Rebleeding ・Stent infection |
・Cerebral ischemia ・Difficult in patients with a history of open chest surgery |
・Life-threating |
VIABAHN is one of the covered stents and can be the self-expanding (VIABAHN; W. L. Gore & Associates) or the balloon expandable type (VIABAHN VBX; W. L. Gore & Associates). In general, the self-expanding stent is flexible and has good trackability of the delivery system.11) The stent should be placed to cover the fistula accurately and to not hang over the common carotid artery or extend too far into the aorta, so the accuracy of stent placement is important. Because of its flexibility and kink resistance, it is easy to place in the tortuous vessels. The self-expanding stent does not compress the vessel when it is placed, but it continues to expand in the long term. As such, it exudes pressure in the chronic phase and sometimes injures the vessel. By contrast, the balloon expandable type can crimp and form to match the shape of vessels. It compresses the vessel when it is deployed, but there is no pressure to the vessel in the chronic phase. There is no consensus on which device is better to use for TIF. The general trend is using a balloon expandable stent with the over-the-wire technique.11) According to the data of O'Malley et al., the self-expanding stent is most frequently used.2) Further data accumulation is expected regarding which device is preferred to address the pathogenesis of TIF. We also place the stent retrogradely to adjust the distal end accurately and because of tortuous vessels.
After covered stent placement, double antiplatelet therapy is generally recommended to prevent stent occlusion. However, in patients at high risk of bleeding, it is not always necessary to start two antiplatelet agents and the use of no antiplatelet agents or just a single antiplatelet agent can be considered.11)
In addition, covered stent implantation carries a risk of infection, so an antibiotic should be administered after the procedure. It is generally recommended to administer cefazolin 30 min before vascular access and up to 24 h after the procedure.6)
Because of the high mortality attendant to TIF, preventing a TIF from forming should be a priority. Risk factors for TIF are a high-riding innominate artery, low tracheostomy position, continuous cuff hyperinflation, and factors delaying wound healing, including steroid use or chronic inflammation.1,2) Avoiding these conditions is an effective way to prevent TIF. At some institutions, CTA is performed to visualize the anatomy of the innominate artery. When a patient has a high-riding innominate artery, surgeons can perform the tracheostomy at a higher level than usual, thereby avoiding the risk of TIF. Confirming CTA before tracheostomy is an effective way to prevent TIF.
In our case, the patient developed TIF after tracheostomy, and we placed a covered stent to treat the TIF because the patient was unstable. We placed a self-expanding covered stent because the innominate artery was tortuous, and we wanted to avoid injury to the fistula during balloon expansion. Because the risk of rebleeding was relatively high after endovascular treatment, we started single antiplatelet therapy, and cefazolin was administered for 1 week to prevent infection. There were no complications, including stent occlusion, rebleeding, or stent infection, for 2 months after the procedure. Less invasive endovascular treatment can be a good choice for TIF patients.
TIF is a rare but fatal complication of tracheostomy, and neurosurgeons who care for patients after tracheostomy can encounter this condition. Endovascular treatment can yield better outcomes than open surgery. In this case, the placement of a covered stent was also useful for achieving hemostasis.
TIF: tracheo-innominate artery fistula
CTA: computed tomography angiography
PTFE: polytetrafluoroethylene
Informed consent was obtained from all participants.
No authors have conflicts of interest.
