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British Journal of Radiology (2007) 80, e54-e57
© 2007 British Institute of Radiology
doi: 10.1259/bjr/61848822

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Case report

Transcatheter arterial embolisation of a ruptured pseudoaneurysm of the lingual artery with n-butyl cyanoacrylate

T Matsumoto, MD T Yamagami, MD, PhD T Kato, MD, PhD T Hirota, MD R Yoshimatsu, MD and T Nishimura, MD, PhD

Department of Radiology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, 465 Kajiicho, Kawaramachi Hirokoji, Kamigyo-ku, 602-8566, Kyoto, Japan

Correspondence: Tomohiro Matsumoto, Department of Radiology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, 465 Kajiicho, Kawaramachi Hirokoji, Kamigyo-ku, 602-8566, Kyoto, Japan. E-mail: t-matsu{at}koto.kpu-m.ac.jp


    Abstract
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
Pseudoaneurysms can arise due to localized arterial wall disruption, owing to various factors such as inflammation, trauma, neoplasm, or surgical procedures. Once ruptured, bleeding can be life-threatening. Thus, a treatment for pseudoaneurysm is necessary. We describe a case of post-surgical ruptured pseudoaneurysm of the lingual artery that was successfully treated by transcatheter embolisation with n-butyl cyanoacrylate (NCBA).


    Introduction
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
The transcatheter embolisation technique has recently gained widespread acceptance as the first treatment method for ruptured pseudoaneurysms. Pseudoaneurysms of the external carotid artery and its branches are rare. Paralleling the development of interventional radiology, the number of reports of such pseudoaneurysms treated by interventional radiological methods is rising [1, 2]. We describe a case of ruptured pseudoaneurysm of the lingual artery 5 weeks after pharyngolaryngoesophagectomy for hypopharyngeal carcinoma and oesophageal carcinoma that was successfully treated by transcatheter embolisation with n-butyl cyanoacrylate (NBCA) (Histoacryl-Blue; Braun, Melsungen, Germany).


    Case report
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 Abstract
 Introduction
 Case report
 Discussion
 References
 
A 69-year-old female suffered a post-cricoid squamous cell carcinoma and a middle thoracic oesophageal squamous cell carcinoma. The patient underwent a total pharyngolaryngoesophagectomy with right radical neck dissection, left selective neck dissection and mediastinal lymph node dissection. The pharynx was reconstructed with a gastric pull-up procedure. Intraoperative bleeding was controlled with electrocautery.

5 weeks after surgery, the patient suddenly had fresh major haemoptysis. The patient's haemoglobin had decreased from 10.5 g dl–1 to 7.4 g dl–1. The patient suffered profuse palpitating bleeding. Blood pressure was 60/40 mmHg, heart rate 110 beats min–1. Because the patient was considered to be in a state of severe hypovolaemic shock requiring infusion of 2800 ml of red blood concentrate, 240 ml of fresh frozen plasma, 2500 ml of plasma protein fraction and 1500 ml of Ringer's lactate, she was immediately transferred to the angiography suite to undergo angiography to determine the bleeding source and subsequently to undergo therapeutic transcatheter embolisation to stop the bleeding, after informed consent was obtained from the patient.

Following insertion of a 5-F sheath introducer from the right femoral artery under local anaesthesia, a 5-F catheter (Clinical Supply, Gifu, Japan) was advanced through the sheath introducer with its tip positioned in the left common carotid artery. Left common carotid arteriography revealed extravasation from a pseudoaneurysm of the left lingual artery (Figure 1Go). Superselective catheterization of the left lingual artery was then performed with a 2.5-F microcatheter system (Renegado; Boston Scientific, Japan), which was coaxially advanced through the 5-F catheter. Then, 1.5 ml of NBCA mixed with iodized oil (Lipiodol; Laboratoire Guerbet, Roissy, France) was inserted from the pseudoaneurysm to the left lingual artery, while care was taken not to embolise arterial branches other than the bleeding vessel (Figure 2Go). The ratio of NBCA to Lipiodol was 1:2. On post-procedure left common carotid arteriography there was no extravasation from the left lingual artery (Figure 3Go).


Figure 1
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Figure 1. Left common carotid arteriography from the anteroposterior view, showing a ruptured pseudoaneurysm(arrow) of the left lingual artery.

 

Figure 2
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Figure 2. Post-embolisation spot radiography showing a mixture of n-butyl cyanoacrylate (NBCA) and Lipiodol which had been injected into the ruptured pseudoaneurysm and the inflow vessel (arrows).

 

Figure 3
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Figure 3. Left common carotid arteriography from the lateral view after embolisation, showing stoppage of bleeding from the left lingual artery.

 
A transient brain infarction occurred 3 h after the embolisation procedure and was suspected of being a complication related to the procedure that was caused, for example, by intracranial embolisation by NBCA fragments as the microcatheter was withdrawn, but that was not confirmed. The neurological symptoms observed included mild weakness of the right arm and right facial palsy. Diffusion-weighted MRI of the brain 4 h after the onset of symptoms showed a hyperintensive area in the left frontal cortex, although this finding could not be correlated with the symptoms in view of the site of the lesion (Figure 4Go). The symptoms soon diminished, and functional ability was completely restored several weeks after the procedure.


Figure 4
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Figure 4. Diffusion-weighted MRI of the brain obtained 4 h after onset of mild weakness in the patient's right arm and right facial palsy, showing a hyperintensive area in the left frontal cortex (arrow).

 
Immediately after embolisation, the patient was haemodynamically stable. Blood pressure was 110/60 mmHg, heart rate 90 beats min–1. 1 day after the embolisation, her haemoglobin was 9.7 g dl–1. There was no post-procedure clinical evidence of lingual ischaemia or infarction. Currently, 2.5 years after the episode described, no further bleeding has occurred.


    Discussion
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
Aneurysms of the external carotid artery and its branches are relatively rare. A review by Schechter [3] of 853 cervical carotid aneurysms from 1687 [4] to 1977 [3] revealed only 2.2% to primarily affect the external carotid. Among the branches of the external carotid artery, pseudoaneurysms of the lingual artery are especially uncommon [510].

Once pseudoaneurysms rupture, they cause massive, life-threatening bleeding. Thus, some treatment would be required. Although surgical treatment has traditionally been performed [57], transcatheter embolisation technique has recently gained widespread acceptance as the first treatment method for ruptured pseudoaneurysms [810].

Salgarelli et al [8] reported a case of a pseudoaneurysm of the lingual artery after an odontogenic infection. The patient was treated by transcatheter embolisation with Gelfoam. Transcatheter embolisation of lingual artery pseudoaneurysms secondary to tonsillectomy with platinum coils was reported by Mitchell et al [9] and Simoni et al [10].

In general, however, embolisation with coils necessitates many coils to embolise sufficiently, making the procedure expensive and time-consuming [11]. As an embolic material is to be used when it is required that the embolisation procedure be performed without being time-consuming, as in our present case, Yamagami et al [11] recommended use of NBCA. NBCA is a liquid and permanent adhesive material, which can be injected through a narrow lumen catheter because of its low viscosity. By adding Lipiodol to NBCA, the embolised vessel can be visualized. In addition, the adhesion time can be regulated flexibly according to the ratio of the mixture of NBCA to Lipiodol [1114]. Reported ratios of NBCA to Lipiodol range from 1:2 [13] to 1:5 [14]. Usage of NBCA has been widely accepted as being effective in the treatment of intracranial arteriovenous malformations [1517], and recent reports of NBCA use in other cases such as intra-abdominal arteriovenous fistulae, pseudoaneurysms and pseudoaneurysm of the lower extremity are increasing [13, 14, 1820].

In the present emergent case, the patient was in a state of hypovolaemic shock. Thus, treatment had to be performed as quickly as possible. We therefore chose the treatment involving transcatheter embolisation with NBCA. As a result, the embolisation procedure was performed expediently. After the embolisation procedure, the haemodynamics of this patient recovered rapidly and we were able to save her life. Additionally, we must note that ischaemic change or infarction of the tongue, which was a possible complication, could be prevented by regulating the adhesion time to thereby accelerate it, which was achieved by mixing Lipiodol at a low rate. After embolisation of the left lingual artery exclusively, it was assumed that the tongue was supplied with blood from the ascending pharyngeal arteries and tonsillar branches of the facial artery [2] or the right lingual artery.

To our knowledge, transcatheter embolisation treatment of the lingual artery with NBCA has not been reported previously.

Received for publication December 1, 2005. Revision received February 15, 2006. Accepted for publication March 6, 2006.


    References
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 Abstract
 Introduction
 Case report
 Discussion
 References
 

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This Article
Right arrow Abstract Freely available
Right arrow Figures Only
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Right arrow Similar articles in PubMed
Right arrow Alert me to new issues of the journal
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Right arrow Citing Articles via Google Scholar
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Right arrow Articles by Matsumoto, T
Right arrow Articles by Nishimura, T
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Right arrow Articles by Matsumoto, T
Right arrow Articles by Nishimura, T


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