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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 21  |  Issue : 2  |  Page : 47-51

Aberrant carotid bulb in bilateral tonsillar fossae: An exceptional discovery during tonsillectomy


1 Department of Otorhinolaryngology and Head and Neck Surgery, Al Jahra Hospital, Ministry of Health, Al Jahra, Kuwait
2 Department of ENT, Jahra Hospital, Al Jahra, Kuwait
3 Department of Radiology, SRM Medical College Hospital and Research Centre, Chennai, Tamil Nadu, India

Date of Submission03-Feb-2019
Date of Decision20-Mar-2019
Date of Acceptance14-Apr-2019
Date of Web Publication22-Oct-2019

Correspondence Address:
Dr. Ramiya Ramachandran Kaipuzha
Department of ENT, Jahra Hospital, Al Jahra
Kuwait
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/SJOH.SJOH_5_19

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  Abstract 


Tonsillectomy, though a popular surgery, has now downsized to limited indications. Although rare, this surgery may cause trauma to the great vessels leading to life-threatening hemorrhage. The congenitally tortuous internal carotid artery (ICA) is an unusual but important anomaly, especially when it is situated in the tonsillar fossae. About 1%–16% of patients have a surgically vulnerable ICA manifested clinically. Here, we present the first reported case of carotid bulb in the right tonsillar fossa. A 7-year-old female child underwent tonsillectomy for obstructive sleep apnea syndrome. Intraoperatively, she was found to have a carotid bulb in both tonsillar fossae, right more prominent than left one. Computed tomography angiogram confirmed the presence of a prominent carotid bulb in the right tonsillar fossa at the C2level. This case report highlights the importance of a heightened awareness of the anatomic variations of carotid artery, in particular, the carotid bulb. This is of critical significance for a safe tonsillectomy as well as any procedure performed in the oropharyngeal region.

Keywords: Aberrant internal carotid artery, carotid bulb, posttonsillectomy hemorrhage, tonsillar fossae, tonsillectomy


How to cite this article:
Aylele BA, Kaipuzha RR, Suvindran A, Alhajri N. Aberrant carotid bulb in bilateral tonsillar fossae: An exceptional discovery during tonsillectomy. Saudi J Otorhinolaryngol Head Neck Surg 2019;21:47-51

How to cite this URL:
Aylele BA, Kaipuzha RR, Suvindran A, Alhajri N. Aberrant carotid bulb in bilateral tonsillar fossae: An exceptional discovery during tonsillectomy. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2019 [cited 2019 Dec 12];21:47-51. Available from: http://www.sjohns.org/text.asp?2019/21/2/47/269712




  Introduction Top


Tonsillectomy is one of the most common surgeries performed by otorhinolaryngologists. It is successful in almost all patients. However, anatomic variations in the location of the surrounding structures may well cause fatal complications during this procedure.[1] Hence, understanding these variations is imperative for a successful surgery.

The congenitally tortuous internal carotid artery (ICA) is an unusual but important anomaly for the otorhinolaryngologists and head and neck surgeons to recognize. Of the general population, 1%–16% have a surgically vulnerable ICA. A tortuous ICA is most commonly found incidentally, as part of a surgery or investigation. However, the literature suggests that patients may present with symptoms such as dysphagia, dysphonia, snoring, foreign-body sensation in the throat, and difficulties in breathing.[2]

Anomalous course of ICA is often diagnosed on the basis of radiological examinations such as contrast-enhanced computed tomography (CT), magnetic resonance imaging (MRI), magnetic resonance angiography, and digital subtraction angiography.[3]

Here, we discuss a case report of the right carotid bulb in the right tonsillar fossae. It was an incidental finding during of tonsillectomy surgery. We were unable to find any similar case reports despite an extensive search of literature in English, and hence, we believe to the best of our knowledge that this may be the first case of presentation of carotid bulb in the tonsillar fossa. This is the first case of ICA anomaly in the tonsillar fossa reported from Kuwait.


  Case Report Top


A 7-year-old female presented to the outpatient department of otorhinolaryngology and head and neck surgery of a tertiary care center in Kuwait with complaints of recurrent episodes of throat pain (five or more episodes in 1 year) and few episodes of obstructive sleep apnea. The infection showed an increasingly unsatisfactory response to antibiotics and caused poor school attendance and performance. There was no dysphagia, dysphonia, or foreign-body sensation in the throat.

Clinical examination was unremarkable except the tonsils which were enlarged (Grade IV – Friedman's classification). She was diagnosed to have chronic tonsillitis as per the scottish intercollegiate guidelines network (SIGN) guidelines.[4] The patient was posted for cold tonsillectomy under general anesthesia after routine blood investigations and anesthesia fitness. Intraoperatively, she was found to have a markedly pulsatile tonsil, and hence, a careful dissection of the tonsillar tissue was done exposing the loose areolar tissue. To the astonishment of the surgeon, a large vessel was found to be pulsating inside the tonsillar fossa. The tonsillar tissue was freed from the vessel with the utmost care to avoid an impending vascular injury [Figure 1], [Figure 2], [Figure 3], [Figure 4].
Figure 1: The postoperative status of the left tonsillar fossa

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Figure 2: The postoperative status of the right tonsillar fossa

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Figure 3: A schematic intraoperative diagram of the oropharynx with marked bulging of the right carotid bulb in the right tonsillar fossa and relatively less conspicuous left carotid artery in the left tonsillar fossa

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Figure 4: Schematic representation of the carotid bulb at the C2 level in the neck at the bifurcation of the common carotid artery

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When the same procedure of tonsillectomy was repeated on the left side, though the pulsation was discernible, the bulge was relatively conspicuous. A provisional diagnosis of aberrant course of ICA in the tonsillar fossae was made, and the child was monitored closely after the procedure for few days as an inpatient. A contrast-enhanced CT angiogram from the skull base to the thoracic inlet was performed 6 weeks after the surgery, and she was found to have an aberrant carotid bulb in the right tonsillar fossa. There was a normal filling of both cervical carotids and vertebral arteries. The arterial walls were reported to be normal. There was marked proximity of the right carotid bulb to the pharynx at the C2 level. There was no evidence of carotid dissection or pseudoaneurysm [Figure 5], [Figure 6], [Figure 7].
Figure 5: Variations in the course of the internal carotid artery as described by Weibel and Fields (1965) (a)Mild kinking (b) Moderate kinking (c) Severe kinking (d) Coiling

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Figure 6: Computed tomography angiography from the skull base to the thorax: maximum intensity projection image showing high bifurcation of the common carotid artery at the C2 level and right carotid bulb medialized to the right side of pharynx

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Figure 7: Computed tomography angiography from the skull base to the thorax: showing high bifurcation of bilateral carotid arteries with medial deviation of right internal carotid artery just proximal to the right carotid bifurcation

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The condition was explained to the parents. The child has been on regular follow-up every month. After 6 months of follow–up, the tonsillar fossa was found to have healed well with no bulging of the carotid bulb into the fossa. The surgery had a noticeable beneficial impact on her quality of life, vitality, and school performance. She radiated vigor.


  Discussion Top


The ICA originates from the carotid bifurcation at the level of C3–C4 (C2–C3 in children) as a branch of the common carotid artery. It passes through the carotid triangle in the carotid sheath together with the internal jugular vein (laterally) and vagus nerve (located between these vessels). It ascends toward the base of the skull.[5] The ICA is situated 2.5 cm away from the tonsillar bed.

The incidence of anomalous course on the cervical part of the ICA is found to be 10%–43%. There is no literature that supports the incidence of the carotid bulb in the tonsillar fossae. Tortuosity of the ICA has long been recognized as an uncommon anomaly encountered during routine head and neck examinations. It is usually an incidental finding during the time of imaging. Otolaryngologists were the first clinicians to focus attention on this condition.[3] There are many variations in the course of ICA noted in the literature. It was first described by Metz et al. in 1961[6] [Table 1]. Weibel and Fields (1965) introduced a classification which is still valid today [Table 2]. An angiography which based the classification of the course of the ICA was established by the modified Paulsen's criteria[7] [Table 3] and [Figure 8].
Table 1: Variations in the course of the Internal Carotid Artery as described by Mets et al. (1961)

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Table 2: Variations in the course of the Internal Carotid Artery as described by Weibel and Fields (1965)

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Table 3: Angiography based classification of the course of the Internal Carotid Artery established by modified Paulsen's criteria

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Figure 8: Virtual reality image showing the right and left carotid bifurcation at C2 level with the right carotid bulb being medialized

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The carotid bulb contains baroreceptors and chemoreceptors to detect acute changes in arterial pressure and arterial oxygen saturation, respectively. Both the receptors communicate with brain stem and through reflexes regulate homeostasis of these vital parameters. Literature on the details of the anatomical variation of carotid bulb has generally been scarce.

Aberrant ICA with regard to the pharynx may present with a wide range of subthreshold symptoms. The latter includes speech and swallowing difficulties and increasing sensation of a foreign body. Further, anomalies of ICA may masquerade as a parapharyngeal or retropharyngeal tumor. This situation may make the ICA more exposed to trauma due to a possible trial of diagnostic puncture or biopsy since masses in most occasions arouse suspicion of malignant disease.[8]

Mere clinical examination may not give a substantial evidence of anomalous course of ICA. This is especially true in a Grade IV (Friedman's Classification) tonsillar hypertrophy, where the tonsils are touching each other. The transmitted pulsation from the artery to the tonsillar parenchyma could be missed. In smaller tonsils, its diagnostic value may still be questioned due to anatomic variation around the peripharyngeal structures.

Imaging studies have been frequently used to map the course of carotid arteries accurately. CT angiography (CTA) is pivotal in the evaluation of the tonsillar blood supply and in the detection of possible aberrant courses of great vessels. MRI provides a right assessment of the soft tissues and has been strongly advised by some authors to assess carotid aberrancy. However, comparing to CTA, this method is less accessible, more expensive, and requires a longer time. Both CTA and MRI of the brain also give information about the ischemic lesions.[9]

Doppler ultrasound can be used in screening tests as an economic, common, and noninvasive tool. It enables to evaluate the hemodynamic state of the carotid arteries. Nonetheless, the results may not be satisfying because of reproducibility and precise visualization of the vessel's course.

In the majority of cases, the aberrant course of the great vessels is an incidental finding during radio imaging. The remainders are anomalies that are discovered unexpectedly during the course of surgery. Our case belongs to the second group; the finding was made on the table during tonsillectomy, and hence, further evaluation of radio imaging was performed.

The anomalous course of ICA may be vulnerable in a wide spectrum of surgeries. This includes tonsillectomy, drainage of peritonsillar abscess, soft palate impalement injuries, as well as adenoidectomy and velopharyngoplasty.[5] The mortality rates of adenotonsillectomy range from about 1 in 16,000 to 1 in 35,000 cases.[9] Standard cold dissection method is still used worldwide for tonsillectomy in spite of the advent of sophisticated methods such as radiofrequency and coblation. It is the authors belief that it bears fewer risks related to ICA anomalies compared to a hot method of tonsillectomy that could have easily lead to the ICA injury leading to torrential bleeding.

The treatment options for posttonsillectomy hemorrhage include surgical ligation and endovascular embolization of the bleeding vessels. Endovascular treatment of hemorrhage has three main advantages: concurrent diagnosis and treatment, selective embolization and less mutilation and less risk of injury to surrounding structures. The materials available for use in endovascular treatment in the head and neck area include coils, polyvinyl alcohol particles, and gelatin sponges.[8]

When indicated, partial tonsillectomy or intracapsular tonsillotomy is preferred in cases with already established diagnosis of anomalous course of the great vessel in close proximity with tonsillar parenchyma.[10] In our case, the aberrant course of the vessel was discovered intraoperatively, after performing dissection of the superior pole of the right tonsil. Hence, we proceeded with meticulous, classic cold tonsillectomy not to leave a residual infected tissue reservoir that may, in turn, be a potential threat to the great vessels.


  Conclusion Top


There is an appreciable body of literature on the various anomalous courses of the ICA. Nevertheless, to our knowledge, there is no study to date that has reported the possibility of carotid bulb in the tonsillar fossa. It is to be underscored that this anomaly requires uttermost vigilance, in particular, in hot tonsillectomies. The young surgeon is expected to be well equipped with the essentials of emergency preparedness in case of an accidental life-threatening arterial breech. The long-term prognosis of carotid bulb in the tonsillar fossae is yet to be studied, and hence, an awareness of this condition among the otorhinolaryngologist and head and neck surgeons is crucial.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Greig SR. Current perspectives on the role of tonsillectomy. J Paediatr Child Health 2017;53:1065-70.  Back to cited text no. 1
    
2.
Hosokawa S, Mineta H. Tortuous internal carotid artery presenting as a pharyngeal mass. J Laryngol Otol 2010;124:1033-6.  Back to cited text no. 2
    
3.
Burton MJ, Glasziou PP, Chong LY, Venekamp RP. Tonsillectomy or adenotonsillectomy versus non-surgical treatment for chronic/recurrent acute tonsillitis. Cochrane Database Syst Rev 2014:CD001802.  Back to cited text no. 3
    
4.
Scottish Intercollegiate Guidelines Network (SIGN). Management of sore throat and indications for tonsillectomy: A national clinical guideline. SIGN 117. Edinburgh: Scottish Intercollegiate Guidelines Network; 2010.  Back to cited text no. 4
    
5.
Banjar AA, Hussain SA, Haoumi A, Shamani MR. Aberrant course of the internal carotid artery in surgery of adenoids and tonsils. Ann Saudi Med 2002;22:344-5.  Back to cited text no. 5
    
6.
Metz H, Murray-Leslie RM, Bannister RG, Bull JW, Marshall J. Kinking of the internal carotid artery in relation to cerebrovascular disease. Lancet 1961;1:424-6.  Back to cited text no. 6
    
7.
Paulsen F, Tillmann B, Christofides C, Richter W, Koebke J. Curving and looping of the internal carotid artery in relation to the pharynx: Frequency, embryology and clinical implications. J Anat 2000;197 Pt 3:373-81.  Back to cited text no. 7
    
8.
Juszkat R, Korytowska A, Lukomska Z, Zarzecka A. Facial artery pseudoaneurysm and severe bleeding after tonsillectomy – Endovascular treatment with PVA particle embolization. Pol J Radiol 2010;75:88-91.  Back to cited text no. 8
    
9.
Kendrick D, Gibbin K. An audit of the complications of paediatric tonsillectomy, adenoidectomy and adenotonsillectomy. Clin Otolaryngol Allied Sci 1993;18:115-7.  Back to cited text no. 9
    
10.
Ziolkowska K, Bachvarov C, Sapundzhiev N, Genova P. Bilateral tortuous internal carotid arteries – A case report, otorhinolaryngologic and general clinical implications. J IMAB 2017;23:1657-66.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]



 

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