Abstract
A rare case of aberrant tortuous common carotid artery (dolichoarteriopathia) presenting as submucosal swellings in bilateral pyriform sinuses, non-pulsatile, mimicking submucosal growths in an elderly female with complaints of dysphagia for solids, is described. CT with contrast by the radiologist opined the swellings as common carotid dolichoarteriopathia. We discussed the presentation of the anomaly in head and neck briefly by reviewing the literature and the hazards of biopsy in such a situation. Flexible fibreoptic videoendoscopy, CT with contrast of the head and neck, and the radiologist’s opinion gave the clue to diagnosis. The non-pulsatile masses in the pyriform sinuses, is intriguing and minimally invasive procedure like biopsy would have been fatal. One should be careful in dealing with such non-pulsatile submucosal swellings in the laryngopharynx without proper investigations. Anomalies of common carotid artery (CCA) are rare in clinical experience. Choi et al. (Eur Arch Otorhinolaryngol 255(5):269–270, 1998) reported tortuous CCA in neck dissection for cancer but in clinical practice, the abnormal course of CCA presenting in pyriform sinus is extremely rare.
Keywords: Anomalous common carotid artery, Pyriform fossa growth, Dysphagia, Snoring, Foreign body sensation in throat
Introduction
Tortuosity of the CCA (dolichoarteriopathia) is rarely seen in Otorhinolaryngology practice. In this case report, we describe briefly about the tortuous CCA (dolichoarteriopathia) presenting as submucosal, non pulsatile, indenting masses in the pyriform fossae (Fig. 1). It was observed during the routine fibreoptic videolaryngoscopy in a 63 year old lady for evaluation of dysphagia for solids. CT with contrast taken showed aberrant tortuous CCA (dolichoarteriopathia) in both pyriform fossae reported by the radiologist (Fig. 1). This is being documented since it is very rare to see such cases in Otorhinolaryngology practice. Knowledge of this anomaly is required to avoid misinterpretation and iatrogenic bleeding while doing biopsy.
Fig. 1.
Flexible fibreoptic videolaryngoscopy image showing non pulsatile masses indenting bilateral pyriform fossae
Case Report
A 63 year old female from North India came to our attention with symptoms of dysphagia for solids for 6 months. Clinical examination showed no abnormalities in throat and pharynx. There were no obvious swellings or visible pulsations in the neck. A flexible fibreoptic videolaryngoscopy showed dynamic picture of non- pulsatile masses in both pyriform fossae with normal mucosal covering, mimicking a benign lesion (Fig. 1). A biopsy was contemplated. But on second thought, a CT scan of neck with contrast was advised. The radiologist reported aberrant course of common carotid artery (dolichoarteriopathia) on both sides, indenting pyriform sinuses (Fig. 2). We did not offer her any treatment and the patient was informed of the diagnosis and was advised to avoid any surgical manipulations.
Fig. 2.
Radiological imaging of tortuosity of the bilateral common carotid arteries
Discussion
Dolicho in Greek means long. Dolichoarteriopathia of CA means elongated CA. Dolichoarteriopathia can be classified as tortuous, coiling and kinking [2]. It is classified as tortuosity when artery assumes a C or S shape [3]. Coiling occurs by elongation of the vessel in a restricted space causing circular or double circular configuration. Kinking presents as a sharp angulation. The illustration of the different types of dolichoarteriopathia with reference to ICA is presented in Fig. 3 is also applicable to the case described in the text which is rare.
Fig. 3.
An illustration of different types of vessel tortuosities. a Tortuous, b coiling, c kinking.
Source: Yu et al. [10]
Pathology of this condition is controversial and some authors have explained it is due to the consequence of myointimal thickening in atherosclerosis while others think it is secondary to anatomical variations of embryological origin or due to hypertension [4]. These anomalies if missed preoperatively can cause fatal haemorrhage during head and neck surgeries [2].
Ciorba et al. [5] reported two cases of aberrant ICA in the middle ear. Choi et al. [1] reported a case of tortuous CCA which traversed over trachea, encountered during neck dissection. Okami et al. [6] identified three cases presenting as bulging in lateral pharyngeal wall in Magnetic Resonance Angiogram (MRIA) presenting with abnormal throat sensation. There are case reports of tortuous ICA presenting as bulging in oropharynx and hypopharynx causing snoring [7]. Cong et al. [8] analysed 7 cases of tortuosity and kinking of cervical segment of internal carotid artery.
Presentation of dolichoarteriopathia of CCA as non pulsatile mass indenting the pyriform fossa is extremely rare [9]. It is usually seen in older adults, mostly asymptomatic, bilateral and pulsatile. But in this case, dysphagia was present and the swellings were non- pulsatile on flexible fibreoptic videoendoscopy. There are case reports of tortuous common carotid artery anomaly encountered during neck dissection for head and neck cancer crossing the lower cervical trachea [1].
The gold standard investigation is a combination of endoscopic visualisation followed by CT scan of the area. Flexible fibreoptic videoendoscopy gives a better and closer, dynamic view of the lesion. MRIA can also be done [8] but CT scan with contrast is faster and cheaper than MRIA and it also detects atherosclerotic foci. Colour Doppler ultrasonography is also a convenient investigation [10]. Routine flexible fibreoptic videoendoscopy did not find any pulsations in the laryngopharynx in this case.
The most important point to be stressed in the treatment is, avoidance of invasive surgeries and even biopsies. Mostly these anomalies are commonly seen in the course of ICA and ECA, but dolichoarteriopathia of CCA is extremely rare. Dolichoarteriopathia of the ECA are more commonly encountered in head and neck surgeries. Most of the CCA anomalies are seen in cadaver dissections and sometimes cause symptoms, depending on the situation in the head and neck and attempted biopsies are fatal, and should be avoided [2]. Multidisciplinary approach will help in such patients and such patients should be treated conservatively and reassured explaining the hazards associated with the invasive surgical procedures.
Conclusion
Vascular anomalies in the laryngopharynx are rare. The presentation of vascular masses mimicking submucosal growth in the pyriform fossae due to aberrant CCA (dolichoarteriopathia), is extremely rare. Routine fibreoptic videolaryngoscopy dynamic view, and CT with contrast helped us in the diagnosis and biopsy of the lesion was deferred and if done would have been fatal. We discussed the aberrant abnormalities of the ICA and ECA which are commonly encountered in head and neck surgeries, but the dolichoarteriopathia of CCA is extremely rare according to literature review.
A dynamic view with Flexible fibreoptic videolaryngoscopy showed the masses in both sides pyriform sinuses without any pulsations is baffling. A simple CT with contrast, by the radiologist opined as CCA Dolichoarteriopathy, prevented us from doing biopsy, and if done would have been fatal.
It is important to consider the possible presence of these anomalies in otolaryngologist practice and adequate diagnostic protocol should be done before any invasive procedures are performed.
Acknowledgements
We thank the medical and non medical staff of Apollo Hospitals, Chennai for providing support to publish this article.
Compliance with Ethical Standards
Conflict of interest
The authors declare that they have no conflict of interests.
Ethical Approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed Consent
Informed consent was obtained from all individual participants included in the study.
Footnotes
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