Profuse bleeding from a pseudoaneurysm of the right superior thyroid artery after tracheostomy

Abstract

Case

A 70‐year‐old man was brought to our hospital emergency department with accidental thermal burns. Surgical tracheostomy was carried out on day 8 after admission, followed by several profuse bleeding episodes from the orifice. Contrast‐enhanced computed tomography of the neck revealed a small nodule with arterial phase enhancement that was suspected to be a pseudoaneurysm. During emergency angiography, the nodule was revealed to be a pseudoaneurysm arising from the right superior thyroid artery with contrast medium extravasation.

Outcome

The patient underwent transcatheter arterial embolization, which resolved bleeding from the tracheostomy orifice.

Conclusion

Pseudoaneurysm of the superior thyroid artery is an extremely rare and life‐threatening tracheostomy complication. All clinicians certified to perform tracheostomy should be acquainted with the various complications and methods for managing life‐threatening post‐tracheostomy complications.

Introduction

Tracheostomy is a common critical care procedure. Although it is associated with various complications, major post‐procedural bleeding from the tracheostomy orifice, such as that caused by a tracheo‐innominate artery fistula, is rare and life threatening. We encountered a case of major bleeding from the orifice due to a tracheostomy‐related pseudoaneurysm of the superior thyroid artery.

Case

A 70‐year‐old man was brought to our hospital emergency department with deep, accidental thermal burns on his head, face, dorsal neck, and shoulder (12% of the total body surface area). His nasal hair was singed, and his nasal and oral mucosae were edematous. He presented with hypoxia and a depressed mental status. Following a diagnosis of inhalation burns, he was intubated to protect his airway and underwent artificial respiration following admission to our intensive care unit. Although his respiratory status gradually improved and stabilized, his face and mouth swelled severely, and we carried out surgical tracheostomy to avoid airway obstruction and wean him from artificial ventilation on day 8 after admission.

Tracheostomy was carried out in the operation room, with the patient under general anesthesia. The patient's neck was extended over a shoulder roll. A 4–5 cm transverse skin incision was created 1–2 cm above the suprasternal notch. After dissection of the s.c. tissues and muscles, we divided the thyroid isthmus. An inverted U‐shaped flap was made between the second and third tracheal rings, and a tracheostomy tube was inserted into the distal trachea under visual control. The patient's short neck and edema presented challenges during this procedure. However, no significant bleeding was encountered.

On postoperative days 2, 6, 16, and 30, we confirmed bleeding from the tracheostomy orifice (Fig. 1). Bleeding detected on postoperative day 2 achieved spontaneous hemostasis; however, profuse bleeding was observed on postoperative day 6. Therefore, we undertook an unsuccessful surgical re‐exploration to determine the bleeding source. We considered the end of the divided thyroid as a likely bleeding source, and ligated the divided end of the thyroid isthmus.

Figure 1.

Clinical course of a 70‐year‐old man after tracheostomy, treated for deep, accidental thermal burns on his head, face, dorsal neck, and shoulder. CECT, contrast enhanced computed tomography; POD, postoperative day; TAE, transcatheter artery embolization.

Despite ligation, we again confirmed profuse bleeding from the tracheostomy orifice on postoperative day 16. Blood flowed from the orifice into the trachea, where the clotted blood temporarily obstructed the patient's airway. The clotted blood was removed, an endotracheal intubation tube was inserted just above the tracheal bifurcation from the tracheostomy orifice to secure a clear airway, and the bleeding achieved spontaneous hemostasis. We suspected bleeding from a tracheo‐innominate artery fistula and performed neck and chest contrast‐enhanced computed tomography (CECT) and flexible tracheoscopy. However, we could not clearly detect the source of bleeding.

When profuse bleeding recurred on postoperative day 30, we reassessed the neck CECT images from postoperative day 16 and found a small nodule (approximately 5.0 mm × 5.5 mm) with arterial phase enhancement on the right side of the tracheostomy tube (Fig. 2A, B). A pseudoaneurysm was the suspected source of recurrent bleeding, and the patient underwent emergency angiography, which revealed a pseudoaneurysm arising from the right superior thyroid artery with contrast medium extravasation (Fig. 2C, D). The patient subsequently underwent transcatheter arterial embolization (TAE) to treat this pseudoaneurysm. A catheter was inserted in the right superior thyroid artery, through which Serescue (Nippon Kayaku, Tokyo, Japan) and an Interlocking Detachable Coil (Boston Scientific, Marlborough, MA, US) were released. After TAE, the pseudoaneurysm was occluded completely and the contrast medium extravasation disappeared (Fig. 2E, F). Bleeding from the tracheostomy orifice also resolved after TAE. No evidence of pseudoaneurysm was visible on neck CECT carried out on postoperative day 45 (Fig. 2G, H). No thyroid functional abnormalities were observed after TAE.

Figure 2.

A, B, Neck contrast enhanced computed tomography (CECT) images taken on postoperative day (POD) 16. A nodule of approximately 5.0 mm × 5.5 mm in size with enhancement is visible at the arrow tip. C, D, Neck angiography images on POD 30. Right common arteriography and right superior thyroid arteriography are shown in C and D, respectively. These images show the pseudoaneurysm arising from the right superior thyroid artery, indicated by arrows, and contrast medium extravasation, indicated by arrowheads. E, F, Neck angiography images after transcatheter artery embolization on POD 30. A sagittal view and coronary view are shown in E and F, respectively. The arrows indicate the Interlocking Detachable Coil in the right superior thyroid artery. These images show a completely occluded pseudoaneurysm and the disappearance of contrast medium extravasation from the pseudoaneurysm. G, H, Neck CECT images taken on POD 45. No pseudoaneurysm was observed on the neck CECT carried out on this day. Rt. CCA, right common carotid artery; Rt. ECA, right external carotid artery; Rt. FA, right facial artery; Rt. ICA, right internal carotid artery; Rt. STA, right superior thyroid artery.

Discussion

Although minor bleeding is common after tracheostomy, profuse bleeding is considered a less frequent complication; for example, tracheo‐innominate artery fistula, one of the best‐known lethal hemorrhagic complications, occurs in less than 1% of all patients undergoing tracheostomy.1 Briefly, a loss of airway patency and profuse bleeding are commonly reported lethal complications of tracheostomy. A loss of airway patency usually results from accidental decannulation, tracheal tube displacement, a blood clot, or a mucous plug. Profuse bleeding causes not only lethal blood losses, but also airway obstruction due to clotted blood. Pseudoaneurysm of the superior thyroid artery is rarer still, with an estimated incidence lower than that of tracheo‐innominate artery fistula. To our knowledge, only nine cases of pseudoaneurysm of the superior thyroid artery have been reported worldwide, including the present case (Table 1),2, 3, 4, 5, 6, 7, 8, 9 and only one case has involved a tracheotomy‐related pseudoaneurysm of the superior thyroid artery.4

Table 1.

Chart review of reported pseudoaneurysms of the superior thyroid artery

Author (years)	Age/sex	Side	Cause	Diagnostic imaging	Treatment	Outcome
Present case	70/M	R	Tracheostomy	CECT Angiography	TAE	Successful
Khera et al. (2015)2	50/F	R	Placement of IJV catheter	CECT DS Angiography	TAE	Successful
Varetto et al. (2013)3	78/F	R	Surgical procedure for primary hyperparathyroidism	CECT DS	TAE→Surgical excision	Successful
Harrison et al. (2012)4	56/M	L	Tracheostomy	CECT Angiography	TAE	Successful
Chang et al. (2010)5	66/M	R	Carotid blowout syndrome	Angiography	Direct percutaneous puncture (embolized by NBCA and lipiodol)	Successful
Celik et al. (2004)6	39/F	R	Fine needle aspiration biopsy	DS	Ultrasonographically guided compression therapy	Successful
Ernemann et al. (2003)7	54/M	Unknown	Carotid blowout syndrome	CECT Angiography	TAE	Successful
Perona et al. (1999)8	71/F	L	Ultrasonographically guided chemical parathyroidectomy	DS Angiography	TAE	Successful
Sharma et al. (1994)9	19/M	L	Penetrating iron splinter injury	99mTc scanning Angiography	TAE	Successful

CECT, contrast enhanced computed tomography; DS, duplex sonography; F, female; IJV, internal jugular vein; L, left; M, male; NBCA, N‐butyl cyanoacrylate; R, right; TAE, transcatheter arterial embolization.

Pseudoaneurysms result from tangential injury to the arterial vessel wall, with subsequent hemorrhage into the defect and formation of a hematoma comprising adventitia or perivascular tissue.6 In seven of the eight previous cases, the causes were iatrogenic complications following a procedure or treatment:2, 3, 4, 5, 6, 7, 8 three cases were related to thyroid or parathyroid disease treatment,3, 6, 8 two involved carotid blowout syndrome after radiation therapy to the neck area,5, 7 one was a complication of internal jugular venous dialysis access catheter placement, and one involved tracheostomy‐related trauma.4 The non‐iatrogenic cause in the remaining case was a penetrating iron splinter injury.9 In our case, the exact cause of the pseudoaneurysm remains uncertain. However, given the timing of bleeding from the tracheostomy orifice and difficulties with the tracheostomy procedure, the cause is suspected intraoperative injury to the patient's right superior thyroid artery.

In the present case, diagnosis of the pseudoaneurysm of the superior thyroid artery was difficult. This condition is extremely rare and unprecedented as a complication of tracheostomy, whereas tracheo‐innominate fistula is among the most well‐known life‐threatening complications. Therefore, our strong suspicion of a tracheo‐innominate fistula, supported by recurrent bleeding that reflected intermittent sentinel bleeding, led to a delayed diagnosis of pseudoaneurysm of the superior thyroid artery.

Pseudoaneurysms of the superior thyroid artery are detected using various imaging methods, including CECT, duplex sonography, and angiography. Angiography is usually carried out to ascertain the precise locations of these pseudoaneurysms following initial imaging studies. However, Nadig et al. reported that CT alone was not adequately sensitive for the detection of pseudoaneurysms of the external carotid artery and could lead to the initial detection of normal findings.10 Similarly, our assumption and ignorance led to an initial failure to detect this condition. However, the neck CECT images obtained on postoperative day 16 revealed a pseudoaneurysm of the superior thyroid artery on the right side of the tracheostomy tube. As the resolution and performance of CT have recently improved, we consider CECT to be useful for the early detection of bleeding sources in the tracheostomy orifice.

Although TAE is the most frequently selected treatment,2, 3, 4, 7, 8, 9 surgical excision,3 ultrasonography‐guided compression therapy,6 and direct percutaneous puncture and embolization via spinal injection of N‐butyl cyanoacrylate (Histoacryl; B. Braun Melsungen, Melsungen, Germany) and lipiodol oil have been used.5 We selected TAE to treat the pseudoaneurysm of the superior thyroid artery in this case. Transcatheter arterial embolization is safe and effective for bleeding lesions and allows a direct, minimally invasive approach to the responsible artery or lesion. This therapy is useful and time conserving, as it can be carried out following angiographic diagnosis. Before treatment, airway protection is imperative to avoid suffocation from bleeding, and vascular access must be prepared in case of hypovolemic shock.

This is only the second reported case of tracheostomy‐related pseudoaneurysm of the superior thyroid artery worldwide. Additional cases will further clarify the causes, risks, and management of this condition.

Conflict of Interest

None.