American Journal of Emergency Medicine xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
American Journal of Emergency Medicine journal homepage: www.elsevier.com/locate/ajem
Case Report
Acute airway compromise due to ruptured inferior thyroid artery aneurysm Abstract A cervical hematoma secondary to the spontaneous rupture of an aneurysm is an uncommon but catastrophic life-threatening condition because it can potentially obstruct the airway. Inferior thyroid artery aneurysm and rupture is a very rare clinical entity and only a limited number of cases have been reported in the literature. In this article, we present the case of a female patient who suffered from a rapidly enlarging cervical mass followed by a rapid onset of dyspnea as a result of rupture of an inferior thyroid artery aneurysm. The diagnosis was confirmed by magnetic resonance angiography, and delayed surgery resulted in an uneventful outcome. We aim to draw the attention of emergency physicians to this rare condition. A 65-year-old woman was admitted to the emergency department presenting with rapid swelling of the neck and rapid onset of dyspnea. Upon admittance, she expressed anxiety about suffocating. Her vital signs were recorded as follows: a blood pressure of 110/60 mm Hg, a heart rate of 85 beats per minute, a respiratory rate of 16 breaths per minute, and oxygen saturation of 97%. She denied any history of trauma, chronic disease, and the use of any medications, including anticoagulants. Her physical examination was unremarkable except that there was swelling on the left side of the neck with ecchymoses on the anterior chest wall (Fig. 1). The patient underwent a neck and thorax computed tomography (CT) scan with contrast to check for acute dissection of the aorta or its main branches. This diagnosis was excluded, but the CT revealed a hypodense hematoma dislocating the trachea and the left lobe of thyroid gland to the right (Fig. 2). Cervical ultrasonography revealed the same results. The patient was consulted with an otolaryngologist. A laryngoscopic examination revealed ecchymoses on the left arytenoid, the band ventricle, the left vocal cord, and the vallecula in addition to hypokinesis of the left vocal cord (Fig. 3). We performed a magnetic resonance (MR) angiogram of the neck that revealed 2 aneurysms at the proximal and distal portions of the left inferior thyroid artery (ITA), and the diagnosis was confirmed (Fig. 4). The patients’ serial hemoglobin and hematocrit levels were stabilized at 9.5 mg/dL and 28%, respectively. She was hospitalized at the intensive care unit, and on the first day of the week, a team of otolaryngologists performed an operation to excise the aneurysms. The hematoma was also evacuated. The patient was discharged on the postoperative seventh day, and after 1 month, an ambulatory followup examination showed that she was healthy without any sequelae. Among the causes of cervical masses, which include neoplasms, inflammatory lesions, and hematomas, a cervical hemorrhage from the spontaneous rupture of an aneurysm is an uncommon but catastrophic
life-threatening condition because it can potentially obstruct the airway. Aneurysms of the branches of the supra-aortal arteries are extremely rare. The ITA is the most common site for thyrocervical trunk aneurysms [1]. A literature search found only a limited number of spontaneously ruptured ITA aneurysms. The present case is the first to be documented in the emergency medicine literature. Although unruptured ITA aneurysms may be asymptomatic or may cause hoarseness or dysphagia by compressing recurrent laryngeal nerve, ruptured aneurysm may result in a cervical hematoma, the compression of adjacent structures, tracheal displacement, endolaryngeal and subcutaneous bruises, and suffocation [1-6]. Because of the risk of suffocation because of airway compromise, many authors suggest that diagnostic procedures should be undertaken only after securing the airway. Otherwise, airway protection may become extremely difficult; it may need to be secured by means of fiber-optic intubation or tracheotomy, or it may even become impossible [1,2,6]. Unlike in the cases described in the previous literature, our patient was not intubated. Instead, during each imaging procedure, the patient was closely observed by an emergency physician and an otolaryngologist in case endotracheal intubation or tracheostomy became necessary. Computed tomography angiography and selective invasive angiography are commonly used modalities for accurately diagnosing ITA aneurysms. Selective angiography has the advantage of allowing embolization of the lesion [1,7,8]. Magnetic resonance angiography was also used in the workup of a cervical hemorrhage that was then confirmed by selective angiography, but it was unable to confirm the origin of the aneurysm [8]. Computed tomography angiography did not accurately diagnose the origin of the hematoma in our patient. This case is the first to be diagnosed by using MR angiography. Because of the high mortality and morbidity associated with ITA aneurysms, it is suggested to treat them actively at the time of the diagnosis [1,9,10]. The 2 possible treatments for aneurysms are surgery or angiographic embolization [1,6,8,9,11]. Surgical therapy can be used to drain compressive hematomas; otherwise, complete resorption may take as long as several months [1]. Only 1 case was not treated, and authors suggest “wait-and-see” strategy if the patient is hemodynamically stable [5]. As our patient was clinically stable, and we made a clinical decision that the extravasation had spontaneously stopped, the management of the aneurysm was delayed for 24 hours under close observation. The main features that differentiate the present case from previously reported ones are the absence of any risk factors, confirmation of the diagnosis using MR angiography, and the successful uneventful outcome despite the delayed surgery. If invasive angiography is not available, there is no risk of suffocation, and the patient is clinically stable; MR angiography serves as an effective diagnostic tool, and under close observation, delayed surgery may result in an outcome as successful as that of immediate surgery.
0735-6757/© 2015 Elsevier Inc. All rights reserved.
Please cite this article as: Coskun ZO, et al, Acute airway compromise due to ruptured inferior thyroid artery aneurysm, Am J Emerg Med (2015), http://dx.doi.org/10.1016/j.ajem.2015.01.049
2
Z.O. Coskun et al. / American Journal of Emergency Medicine xxx (2015) xxx–xxx
Fig. 1. Swelling of the neck commencing from the left submandibular region and extending to the clavicle and suprasternal notch through the midline. She also had ecchymoses on the anterior chest wall.
Fig. 3. A videolaryngoscopy of the larynx showing ecchymoses on the left arytenoid, the band ventricle, the left vocal cord, and the vallecula.
Zerrin Ozergın Coskun, MD Department of Otorhinolaryngology, Recep Tayyip Erdoğan University, Rize Research and Training Hospital, Rize, Turkey Özcan Yavaşi, MD Department of Emergency Medicine, Recep Tayyip Erdoğan University, Rize Research and Training Hospital E-mail: [email protected] Tugba Durakoglugil, MD Department of Radiology, Recep Tayyip Erdoğan University, Rize Research and Training Hospital, Rize, Turkey Ozlem Celebi Erdivanli, MD Abdulkadir Ozgur, MD Suat Terzi, MD Engin Dursun, MD Department of Otorhinolaryngology, Recep Tayyip Erdoğan University, Rize Research and Training Hospital, Rize, Turkey
http://dx.doi.org/10.1016/j.ajem.2015.01.049
Fig. 4. An MR angiogram showing proximal (arrowhead) and distal (black arrow) aneurysms of the left ITA.
References
Fig. 2. A contrast-enhanced CT scan showing a hematoma (black arrow) dislocating the trachea and the left lobe of the thyroid gland to the right.
[1] Garrett Jr HE, Heidepriem III RW, Broadbent LP. Ruptured aneurysm of the inferior thyroid artery: repair with coil embolization. J Vasc Surg 2005;42: 1226–9. [2] Habib MA. Fatal haemorrhage due to ruptured inferior thyroid artery aneurysm. J Laryngol Otol 1977;91(5):437–40. [3] Seenu V, Baliga S, Misra MC. Aneurysm of the inferior thyroid artery. Postgrad Med J 1994;70(824):452–4. [4] Marrocco-Trischitta MM, Kahlberg A, Calliari F, Chiesa R. Bilateral aneurysm of the inferior thyroid artery. J Vasc Surg 2007;45(3):614. [5] Hoetzenecker K, Topker M, Klepetko W, Ankersmit HJ. Spontaneous rupture of the inferior thyroid artery resulting in mediastinal hematoma. Interact Cardiovasc Thorac Surg 2010;11:209–10. [6] Pop D, Nadeemy S, Venissac N, Aze O, Mouroux J. Ruptured aneurysm of the inferior thyroid artery, which treatment? J Visc Surg 2011;148(3):e227–8. [7] Germain DP. Clinical and genetic features of vascular Ehlers-Danlos syndrome. Ann Vasc Surg 2002;16:391–7.
Please cite this article as: Coskun ZO, et al, Acute airway compromise due to ruptured inferior thyroid artery aneurysm, Am J Emerg Med (2015), http://dx.doi.org/10.1016/j.ajem.2015.01.049
Z.O. Coskun et al. / American Journal of Emergency Medicine xxx (2015) xxx–xxx [8] Kocatürk H, Karaman A, Bayram E, Çolak MC. Unusual presentation of inferior thyroid artery aneurysm: case report. Turkiye Klinikleri J Cardiovasc Sci 2011; 23(3):287–91. [9] Ferrero E, Gaggiano A, Maggio D, Ferri M, Piazza S, Berardi G, et al. Isolated aneurysm of the inferior thyroid artery repair with coil embolization. Minerva Chir 2008;63:547–9.
3
[10] Kos X, Henroteaux D, Dondelinger RF. Embolization of a ruptured aneurysm of the inferior thyroid artery. Eur Radiol 2001;11(7):1285–6. [11] Lee SH, Choi HJ, Yang JS, Cho YJ. Coil embolization in ruptured inferior thyroid artery aneurysm with active bleeding. J Korean Neurosurg Soc 2014;56(4): 353–5.
Please cite this article as: Coskun ZO, et al, Acute airway compromise due to ruptured inferior thyroid artery aneurysm, Am J Emerg Med (2015), http://dx.doi.org/10.1016/j.ajem.2015.01.049
Contents lists available at ScienceDirect
American Journal of Emergency Medicine journal homepage: www.elsevier.com/locate/ajem
Case Report
Acute airway compromise due to ruptured inferior thyroid artery aneurysm Abstract A cervical hematoma secondary to the spontaneous rupture of an aneurysm is an uncommon but catastrophic life-threatening condition because it can potentially obstruct the airway. Inferior thyroid artery aneurysm and rupture is a very rare clinical entity and only a limited number of cases have been reported in the literature. In this article, we present the case of a female patient who suffered from a rapidly enlarging cervical mass followed by a rapid onset of dyspnea as a result of rupture of an inferior thyroid artery aneurysm. The diagnosis was confirmed by magnetic resonance angiography, and delayed surgery resulted in an uneventful outcome. We aim to draw the attention of emergency physicians to this rare condition. A 65-year-old woman was admitted to the emergency department presenting with rapid swelling of the neck and rapid onset of dyspnea. Upon admittance, she expressed anxiety about suffocating. Her vital signs were recorded as follows: a blood pressure of 110/60 mm Hg, a heart rate of 85 beats per minute, a respiratory rate of 16 breaths per minute, and oxygen saturation of 97%. She denied any history of trauma, chronic disease, and the use of any medications, including anticoagulants. Her physical examination was unremarkable except that there was swelling on the left side of the neck with ecchymoses on the anterior chest wall (Fig. 1). The patient underwent a neck and thorax computed tomography (CT) scan with contrast to check for acute dissection of the aorta or its main branches. This diagnosis was excluded, but the CT revealed a hypodense hematoma dislocating the trachea and the left lobe of thyroid gland to the right (Fig. 2). Cervical ultrasonography revealed the same results. The patient was consulted with an otolaryngologist. A laryngoscopic examination revealed ecchymoses on the left arytenoid, the band ventricle, the left vocal cord, and the vallecula in addition to hypokinesis of the left vocal cord (Fig. 3). We performed a magnetic resonance (MR) angiogram of the neck that revealed 2 aneurysms at the proximal and distal portions of the left inferior thyroid artery (ITA), and the diagnosis was confirmed (Fig. 4). The patients’ serial hemoglobin and hematocrit levels were stabilized at 9.5 mg/dL and 28%, respectively. She was hospitalized at the intensive care unit, and on the first day of the week, a team of otolaryngologists performed an operation to excise the aneurysms. The hematoma was also evacuated. The patient was discharged on the postoperative seventh day, and after 1 month, an ambulatory followup examination showed that she was healthy without any sequelae. Among the causes of cervical masses, which include neoplasms, inflammatory lesions, and hematomas, a cervical hemorrhage from the spontaneous rupture of an aneurysm is an uncommon but catastrophic
life-threatening condition because it can potentially obstruct the airway. Aneurysms of the branches of the supra-aortal arteries are extremely rare. The ITA is the most common site for thyrocervical trunk aneurysms [1]. A literature search found only a limited number of spontaneously ruptured ITA aneurysms. The present case is the first to be documented in the emergency medicine literature. Although unruptured ITA aneurysms may be asymptomatic or may cause hoarseness or dysphagia by compressing recurrent laryngeal nerve, ruptured aneurysm may result in a cervical hematoma, the compression of adjacent structures, tracheal displacement, endolaryngeal and subcutaneous bruises, and suffocation [1-6]. Because of the risk of suffocation because of airway compromise, many authors suggest that diagnostic procedures should be undertaken only after securing the airway. Otherwise, airway protection may become extremely difficult; it may need to be secured by means of fiber-optic intubation or tracheotomy, or it may even become impossible [1,2,6]. Unlike in the cases described in the previous literature, our patient was not intubated. Instead, during each imaging procedure, the patient was closely observed by an emergency physician and an otolaryngologist in case endotracheal intubation or tracheostomy became necessary. Computed tomography angiography and selective invasive angiography are commonly used modalities for accurately diagnosing ITA aneurysms. Selective angiography has the advantage of allowing embolization of the lesion [1,7,8]. Magnetic resonance angiography was also used in the workup of a cervical hemorrhage that was then confirmed by selective angiography, but it was unable to confirm the origin of the aneurysm [8]. Computed tomography angiography did not accurately diagnose the origin of the hematoma in our patient. This case is the first to be diagnosed by using MR angiography. Because of the high mortality and morbidity associated with ITA aneurysms, it is suggested to treat them actively at the time of the diagnosis [1,9,10]. The 2 possible treatments for aneurysms are surgery or angiographic embolization [1,6,8,9,11]. Surgical therapy can be used to drain compressive hematomas; otherwise, complete resorption may take as long as several months [1]. Only 1 case was not treated, and authors suggest “wait-and-see” strategy if the patient is hemodynamically stable [5]. As our patient was clinically stable, and we made a clinical decision that the extravasation had spontaneously stopped, the management of the aneurysm was delayed for 24 hours under close observation. The main features that differentiate the present case from previously reported ones are the absence of any risk factors, confirmation of the diagnosis using MR angiography, and the successful uneventful outcome despite the delayed surgery. If invasive angiography is not available, there is no risk of suffocation, and the patient is clinically stable; MR angiography serves as an effective diagnostic tool, and under close observation, delayed surgery may result in an outcome as successful as that of immediate surgery.
0735-6757/© 2015 Elsevier Inc. All rights reserved.
Please cite this article as: Coskun ZO, et al, Acute airway compromise due to ruptured inferior thyroid artery aneurysm, Am J Emerg Med (2015), http://dx.doi.org/10.1016/j.ajem.2015.01.049
2
Z.O. Coskun et al. / American Journal of Emergency Medicine xxx (2015) xxx–xxx
Fig. 1. Swelling of the neck commencing from the left submandibular region and extending to the clavicle and suprasternal notch through the midline. She also had ecchymoses on the anterior chest wall.
Fig. 3. A videolaryngoscopy of the larynx showing ecchymoses on the left arytenoid, the band ventricle, the left vocal cord, and the vallecula.
Zerrin Ozergın Coskun, MD Department of Otorhinolaryngology, Recep Tayyip Erdoğan University, Rize Research and Training Hospital, Rize, Turkey Özcan Yavaşi, MD Department of Emergency Medicine, Recep Tayyip Erdoğan University, Rize Research and Training Hospital E-mail: [email protected] Tugba Durakoglugil, MD Department of Radiology, Recep Tayyip Erdoğan University, Rize Research and Training Hospital, Rize, Turkey Ozlem Celebi Erdivanli, MD Abdulkadir Ozgur, MD Suat Terzi, MD Engin Dursun, MD Department of Otorhinolaryngology, Recep Tayyip Erdoğan University, Rize Research and Training Hospital, Rize, Turkey
http://dx.doi.org/10.1016/j.ajem.2015.01.049
Fig. 4. An MR angiogram showing proximal (arrowhead) and distal (black arrow) aneurysms of the left ITA.
References
Fig. 2. A contrast-enhanced CT scan showing a hematoma (black arrow) dislocating the trachea and the left lobe of the thyroid gland to the right.
[1] Garrett Jr HE, Heidepriem III RW, Broadbent LP. Ruptured aneurysm of the inferior thyroid artery: repair with coil embolization. J Vasc Surg 2005;42: 1226–9. [2] Habib MA. Fatal haemorrhage due to ruptured inferior thyroid artery aneurysm. J Laryngol Otol 1977;91(5):437–40. [3] Seenu V, Baliga S, Misra MC. Aneurysm of the inferior thyroid artery. Postgrad Med J 1994;70(824):452–4. [4] Marrocco-Trischitta MM, Kahlberg A, Calliari F, Chiesa R. Bilateral aneurysm of the inferior thyroid artery. J Vasc Surg 2007;45(3):614. [5] Hoetzenecker K, Topker M, Klepetko W, Ankersmit HJ. Spontaneous rupture of the inferior thyroid artery resulting in mediastinal hematoma. Interact Cardiovasc Thorac Surg 2010;11:209–10. [6] Pop D, Nadeemy S, Venissac N, Aze O, Mouroux J. Ruptured aneurysm of the inferior thyroid artery, which treatment? J Visc Surg 2011;148(3):e227–8. [7] Germain DP. Clinical and genetic features of vascular Ehlers-Danlos syndrome. Ann Vasc Surg 2002;16:391–7.
Please cite this article as: Coskun ZO, et al, Acute airway compromise due to ruptured inferior thyroid artery aneurysm, Am J Emerg Med (2015), http://dx.doi.org/10.1016/j.ajem.2015.01.049
Z.O. Coskun et al. / American Journal of Emergency Medicine xxx (2015) xxx–xxx [8] Kocatürk H, Karaman A, Bayram E, Çolak MC. Unusual presentation of inferior thyroid artery aneurysm: case report. Turkiye Klinikleri J Cardiovasc Sci 2011; 23(3):287–91. [9] Ferrero E, Gaggiano A, Maggio D, Ferri M, Piazza S, Berardi G, et al. Isolated aneurysm of the inferior thyroid artery repair with coil embolization. Minerva Chir 2008;63:547–9.
3
[10] Kos X, Henroteaux D, Dondelinger RF. Embolization of a ruptured aneurysm of the inferior thyroid artery. Eur Radiol 2001;11(7):1285–6. [11] Lee SH, Choi HJ, Yang JS, Cho YJ. Coil embolization in ruptured inferior thyroid artery aneurysm with active bleeding. J Korean Neurosurg Soc 2014;56(4): 353–5.
Please cite this article as: Coskun ZO, et al, Acute airway compromise due to ruptured inferior thyroid artery aneurysm, Am J Emerg Med (2015), http://dx.doi.org/10.1016/j.ajem.2015.01.049
