Official Journal of Indian Dental Association Tirunelveli Branch

UPHEAVEL IN STYLOID PROCESS – CASE REPORT

Dr. Bavadharani K1*, Dr. Angeline Deepthi 2, Dr.Kandasamy 3, Dr. 4 Present address: †Post Nelson Graduate, Department of Oral and , Rajas Dental College and Hospital, Kavalkinaru, Tirunelveli, Tamil Abstract Nadu, India.; ‡Professor and Head Of The Department Oral Eagle syndrome is a symptomatic elongation of the styloid process or calcification Medicine and Radiology, Rajas of the stylomandibular ligament. It is a rare entity that causes recurrent throat pain, Dental College and Hospital, neck pain, , difficulty with deglutition, phonation, cervical movement, Kavalkinaru, Tirunelveli, Tamil Nadu, India; §Reader, Oral or facial pain due to an elongated styloid process or calcified stylohyoid ligament Medicine and Radiology, Rajas and the sensation of a foreign body in the oropharynx are all symptoms of this Dental College and Hospital, syndrome. Various theories have been put forward toward the development Kavalkinaru, Tirunelveli, Tamil of Eagle syndrome. Depending on the underlying pathogenetic mechanism Nadu, India; ¶Reader, Oral and the anatomical structures compressed or irritated by the elongated styloid Medicine and Radiology, Rajas Dental College and Hospital, process, symptoms vary greatly, ranging from cervicofacial pain to cerebral Kavalkinaru, Tirunelveli, Tamil . Its treatment consists of partial removal of the styloid process, leaving Nadu, India. it within the range of normality. Clinical findings related to lower cranial nerve

Access this article online compression have also been reported. In some cases, it is reported that carotid https://www.jidati.com/ artery compression or dissection can be seen due to the elongated styloid process Article ID and this is called carotid artery syndrome. Carotid artery compression causes flow JIDATI0101004 reduction and carotidynia or neurological symptoms can be seen, dural sinuses and the jugular vein can also be compressed. Eagle syndrome with neurological symptoms has been rarely reported. Keywords: Eagle Syndrome, Styloid Process, Stylomandibular Ligament, Cervicofacial Pain, Oto-rhino-laryngology

INTRODUCTION Address for correspondence: drbavadharani2015@gmail. The styloid process normally measures 2.5–3 cm in length and is situated at the com (Post Graduate, base of the , immediately posterior to the mastoid apex. When the Department of length of the styloid process exceeds 3 cm, it is considered to be elongated and and Radiology, Rajas Dental can be associated with a wide variety of symptoms that were first described by College and Hospital, Eagle in 1937. Eagle originally described two morbidity forms. The first classic Kavalkinaru, Tirunelveli, Tamil Nadu, India.) styloid process syndrome form is caused by the calcification of the stylohyoid complex resulting in the scar tissue impinging on cranial nerves V, VII, IX, X Submitted: 05-Nov-2020 or XII, all of which are situated near the styloid process[1]. The second carotid Revised: 15-Dec-2020 artery Eagle syndrome form is caused by compression of the sympathetic chain Accepted: 20-Jan-2021 in the carotid sheath[1,2]. As per the theories of various persons, the following Published: 07-Mar-2021 factors may be considered: & the ossification of the stylohyoid ligament complex, causing contraction of the stylopharyngeal muscle and stretching of the XII cranial nerve[2]. & the fracture and medialization of the ossified stylohyoid ligament,

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with incomplete repair due to continuous hyoid bone movements and formation of excessive granulation tissue & the ossification of muscular tendons leading to irritation of the structures nearby & the abnormal length associated with abnormal angulation of the styloid process[3,4,5]. A lateral neck radiograph, orthopantomogram, and modified Towne’s view can confirm the diagnosis, but a three-dimensional (3D) computed tomography (CT) scan is the most valuable diagnostic tool that can define the anatomic relationship between the elongated styloid process and the vascular structures and nerves[6].

CASE REPORT A 27 years old female patient reported to the department of oral medicine and radiology with the complaint of pain in the right and left side of the jaw for the past 5 months. On eliciting the history pain is gradual in onset severe dull aching pain aggravates on chewing and while opening-closing the mouth and by turning the neck. Pain is non-radiating. On past medical history patient had been under medication for hypothyroidism for the past three years and this is her first dental visit and there is no familial and systemic disease running in their family, there is no abnormal habit history. On general examination, the patient was moderately built and nourished well oriented to time, place and personal signs pallor, icterus, clubbing, cyanosis, lymphadenopathy and pedal edema, and all her vital signs are within the normal limits. On extraoral examination of the on inspection no deviation while opening and closing the mouth, on palpation tenderness on right and left side of jaw mouth opening restricted and about 10 mm clicking sound present, difficulty in swallowing, no palpable lymph nodes. On intraoral examination tenderness on palpation in the tonsilar region on the right side while palpating. So the further investigation was done in orthopantomogram there was the elongation of the styloid process more than 3 cm in the right and left side but there is the superimposition of structure so higher imaging modalities like a cone-beam computed tomography is taken in cross-sectional view elongation of the styloid process of size 67.8mm, in 3D reconstruction view elongation of styloid process on right and left side and its segmented type. So with all the history and positive final diagnosis is given as eagle syndrome.

DISCUSSION Eagle syndrome is a rare clinical entity and described as an elongated styloid process or mineralization of the stylohyoid or stylomandibular ligaments that cause cervical and facial pain, the sensation of a foreign body lodged in the pharynx, dysphagia and odynophagia[7,8,9]. The normal length of the adult styloid in an adult is thought to be approximately 2.5 cm while an elongated styloid is considered >3 cm. Elongation can be unilateral or bilateral[10,11]. Two types of Eagle syndrome have been described. An abnormally long styloid process producing autonomic effects gives rise to Eagle syndrome. On a literature search through the Internet, we came across a case of sudden death due to Eagle syndrome. In this particular case, the diagnosis was made by cone-beam computed tomography. Patients often experience a wide variety of symptoms, including pain in the throat and ear, vertigo, voice alteration, cough, dizziness, sinusitis, conjunctival injection, headaches, swallowing problems, and pain when turning the head[7,11]. The first type includes cervicofacial pain aggravated by swallowing and the sensation of a foreign body in the throat after tonsillectomy[10]. The second type is the “stylo-carotid artery syndrome”, and is attributed to impingement of the ICA extracranially by the styloid process[7]. This can cause compression when turning the head, resulting in a transient ischemic accident or [12]. The differential diagnosis of Eagle’s syndrome may include any condition that can result in cervicofacial pain[4]. These include temporomandibular joint (TMJ) diseases; trigeminal, sphenopalatine, and glossopharyngeal neuralgias; myofascial pain; mastoiditis; otitis; temporal arteritis; dental pain[5,6]. Eagle syndrome can cause serious complications such as carotid artery pseudoaneurysm, carotid artery dissection and dural venous thrombosis. Bouzaidi et al.emphasized the importance of radiological examinations and

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especially 3D volume-rendering CT scan for the diagnosis of Eagle syndrome. In all of our cases, CT and 3D volume-rendering CT scans were used for the diagnosis[3]. Dao et al.[7] presented a case of Eagle syndrome resulting in an external carotid artery pseudoaneurysm. Aneurysmectomy and local resection of the styloid process was performed for treatment. Clinical diagnosis rests upon the previous history of trauma or tonsillectomy and palpation of the . Radiologic studies such as an orthopantomogram or lateral skull view with the head slightly extended may help to confirm this di¬agnosis18. A precise history, examination, and imaging stud¬ies also contribute greatly to achieving the correct diagnosis[8,9].

Figure 2. Side Profile Figure 1. Profile

Figure 3. Intraoral Image

Figure 4. Orthopantamogram

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Figure 5. Cone Beam Computed Tomography Cross-Sectional

Figure 6. Cone Beam Computed Tomography 3D Reconstruction

CONCLUSION When dealing with cases of cervical pain or neurological symptoms, Eagle syndrome must be taken into account. Plain radiographs can be helpful, but the most useful examination for diagnosis is a 3D volume-rendering CT scan. Future studies are necessary to find out the relationship between an elongated styloid process and the development of these complications.

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[2] Trauma induced eagle Syndrome A. Koivuma¨ki, M. Marinescu-Gava, J. Ja¨rnstedt, G.K. Sa´ndor, J. Wolff: Trauma induced eagle syndrome. Int. J. Oral Maxillofac. Surg. 2012; 41: 350–353.International Association of Oral and Maxillofacial Surgeons.

[3] Monsour PA, Young WG. Variability of the styloid process and tylohyoid ligament in panoramic radiographs. Oral Surg Oral Med Oral Pathol. 1986;61(5):522Y526.

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[4] Moffat DA, Ramsden RT, Shaw HJ. The styloid process syndrome: aetiological factors and surgical management. J Laryngol Otol. 1977; 91(4):279Y294.

[5] Lindeman P. The elongated styloid process as a cause of throat discomfort. Four case reports. J Laryngol Otol. 1985;99(5):505Y508.

[6] Boddour HM, McAnear JT, Tilson HB. Eagle’s syndrome. Report of a case. Oral Surg Oral Med Oral Pathol. 1978;46(4):486Y494.

[7] Andrade MG, Marchionni AM, Rebello IC: Three-dimensiona identification of vascular compression in Eagle’s syndrome using computed tomography: Case report. J Oral Maxillofac Surg 66: 169-176, 2008

[8] Dao A, Karnezis S, Lane III JS, Fujitani RM, Saremi F: Eagle syndrome presenting with external carotid artery pseudoaneurysm. Emerg Radiol 18: 263-265, 2011

[9] David J, Lieb M, Rahimi SA: Stylocarotid artery syndrome. J Vasc Surg 60: 1661-1663, 2014

[10] Eagle WW: Symptomatic elongated styloid process: Report of two cases of styloid process–carotid artery syndrome with operation. Arch Otolaryngol 49: 490–503, 1949

[11] Al Weteid AS, Miloro M: Transoral endoscopic-assisted styloidectomy: How should Eagle Syndrome be managed surgically? Int J Oral Maxillofac Surg 44: 1181-1187, 2015

[12] Andrade MG, Marchionni AM, Rebello IC: Three-dimensional identification of vascular compression Eagle’s syndrome using computed tomography: Case report. J Oral Maxillofac Surg 66: 169-176, 2008

[13] Chuang WC, Short JH, McKinney AM, Anker L, Knoll B, McKinney ZJ: Reversible left hemispheric ischemia secondary to carotid compression in Eagle syndrome: Surgical and CT angiographic correlation. AJNR Am J Neuroradiol 28: 143-145, 2007

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