CASE REPORT

Stylohyoid Ligament Syndrome Solving the riddle with 3D Computed Tomography

Renu Tanwar1, Chandrashekhar L.2, Asha R. Iyengar2 K.S.Nagesh2, Subhash BV2

1 Department of Oral Medicine and Radiology SGT Dental College Budhera, Gurgaon 123505 U.P India ., 2 Department of Oral Medicine and Radiology, D A P M R V Dental College Bangalore 560078 Karnataka, India

ABSTRACT
Orofacial pain can be associated with stylohyiod ligament calcification or enlargement of styloid process. Calcification or ossification of the stylohyoid ligament is infrequent, often incidental finding on radiographs, however when the source of pain is from the styloid process or calcified stylohyoid ligaments it is referred to as Eagle's syndrome. This case report discusses the pain pattern, clinical presentation, radiologic findings of stylohyoid ligament syndrome. Keywords: Stylohyoid Ligament Syndrome, 3D-CT, Eagle's Syndrome

Journal of Dental Sciences and Research Vol. 2, Issue 2, Pages 1-5

INTRODUCTION Pain in the orofacial region can result due to presence of elongated styloid process unilaterally or bilaterally due to pressure exerted on various vital structures in the neck region. In conditions of hemifacial pain of obscure causation, the oral diagnostician should consider Stylohyoid syndrome as a possible diagnosis. Stylohyoid syndrome occurs due to elongation of stylohyoid process or calcification of stylohyoid ligament. In such cases, imaging helps in identifying abnormally elongated styloid process or calcified stylohyoid ligament. Recent imaging modalities including three dimensional computed tomography aid in assessing the length and anatomical relationship of elongated styloid process with vital structures and for outlining the plane of incision for surgical treatment. CASE REPORT A 32 year old female patient came to the Department of Oral Medicine and Radiology, with chief complaint of pain on right side of inside of the mouth since last three months. She was apparently well three months ago when she first experienced severe pain in right side of the mouth on swallowing the food while having dinner. Pain was of primary incidence, severe in intensity paraoxysmal in nature and it lasted for two to three minutes after swallowing ,and radiated towards the right
Address for correspondence: Dr Renu Tanwar E-mail: renuomdr@gmail.com, drashaiyengar@yahoo.co.in Access this article online Website: http://www.ssdctumkur.org/jdsr.php.

temporal region and side of the neck below the lower jaw on the right side. Patient experienced similar episodes of pain on swallowing and turning the head to the left side at that time. The patient did not report any change in the nature of the pain since its first occurence and symptoms of pain were initiated on turning the head towards left side. Patient did not gave history of decreased salivation, dryness of the mouth, trauma in head and neck region or surgery with respect to the neck or tonsillar region. On extraoral and intraoral examination no significant findings were observed. On intraoral palpation, mild tenderness was observed on bidigital palpation of floor of the mouth on the posterior side. A provisional diagnosis of Stylohyoid syndrome was arrived at. Radiographic examinations included conventional radiographs such as Mandibular true occlusal view, Panoramic radiograph, Lateral oblique view of the ramus of the mandible and advanced imaging as Computed Tomography of the head and neck including 3D Computed Tomography. Panoramic radiograph showed the elongated styloid processes bilaterally, with the right styloid process measuring 37 mm and the left styloid process measuring 38 mm. The right styloid process showed uninterrupted elongation with calcified outline and the left styloid process showed uninterrupted elongation with nodular complex pattern of calcification (Figure 1). The lateral oblique view of of ramus of right side of the mandible showed elongated styloid process with the tip of styloid process extending nearly upto the angle of the mandible on the right side (Figure 2).

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Fig. 1: Panoramic radiograph showing enlargement of styloid process on either sides

Fig. 3: coronal and sagittal sections demonstrating prominent styloid process Fig. 2: Lateral oblique view of the ramus of mandible right side

three times a day for five days and was referred to oral surgeon for surgical management. Computed Tomography was done and volume scans were performed from skull base down to the level of C6 employing 0.625mm sections. Multiplanar reconstructions were also performed for 3D reconstruction (Figure 3 & 4). The following observations were made : 1) Presence of elongated styloid process were seen bilaterally. 2) Right styloid process measured 3.9mm. 3) Left styloid process measuerd 4.2mm. 4) No obvious evidence of calcification of stylohyoid ligament was found on CT images. Based on Patient's history,clinical and radiological findings,a final diagnosis of Stylohyoid Syndrome was arrived at. Patient was advised non steroidal analgesics DISCUSSION The stylohyoid chain consists of the styloid process, the lesser cornu of the hyoid bone and its connecting ligament. The stylohyoid chain is derived from Second branchial arch or Hyoid arch known as Reichert's cartilage. The styloid process is a small, tapering projection of the temporal bone located anterior to the stylomastoid foramen..The styloid process lies between the internal and external carotid arteries, posterior to the tonsillar fossa and lateral to the pharyngeal wall. The styloid process has attachments to three muscles and two ligaments. The stylohyoid ligament itself, extends from the tip of the styloid process to the lesser cornu of the hyoid bone. The stylomandibular ligament extends from the styloid process to the angle of the mandible. The three muscles include the stylopharyngeous, stylohyoid, and styloglossus. The nerve supply comes from the glossopharyngeal, facial, and hypoglossal nerves, respectively. The internal jugular vein and the accessory,
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hypoglossal, vagus, and glossopharyngeal nerves are located medial to the styloid process. The glossopharyngeal nerve emerges from the anterior part of the jugular foramen, medial to the styloid process, where it then curves around the posterior border at the level of the origin of the stylohyoid muscle. This anatomic relationship is important as a cause of glossopharyngeal neuralgia in reported cases with an elongated and ,or fractured styloid process as the etiologic cause[1] . In 1937,Eagle proposed that the average length of the styloid process ranges from 2.5 to 3.0 centimeters[2]. In 1964, Developmental theory was proposed by Lengele and Dhem3 for the elongation of styloid process based on morphogenesis of of Reichert's cartilage. According to them, elongation of styloid process should be congenital. However it was also agreed by them that further growth was possible through the cartilaginous cap of the tip of the styloid process. Langlais et al4 proposed a radigraphic classification of

elongated and mineralized stylohyoid ligament complex.This classification was based on types of elongation and patterns of calcification of stylohyoid ligament. (Table 1 & 2) (Figure 4). Because of an elongated styloid process or a calcified stylohyoid ligament, a patient with Eagle's syndrome may develop non-specific pain, which may change with head movements at the ear or neck. Additionally, a patient with an elongated styloid process may have referred pain to the jaw joint or upper extremities, or dysphagia or foreign body-like irritation throughout the pharynx[5]. There are several different theories, which try to explain the etiopathology of Eagle's syndrome such as congenital elongation of the styloid process and calcification and ossification of the stylohyoid ligament6. Fini et al. reported that past tonsillectomy is related to Eagle's syndrome[7]. In differential diagnosis, laryngopharyngeal dysesthesia has to be considered as well as dental malocclusion,

TABLE 1: MORPHOLOGIC CHARACTERISTICS OF STYLOID PROCESS TYPES I II NOMENCLATURE ELONGATED PSEUDOARTICULATED RADIOGRAPHIC APPEARANCES Uninterrupted integrity of styloid image(>25-28mm). Styloid process is joined to the mineralized stylomandibular or stylohyoid ligament by a single pseudoarticulation, usually located superior to inferior border of the mandible. Short or long continuous portions of the styloid process or uninterrupeted segments of mineralized ligament.

III

SEGMENTED

TABLE 2 : PATTERNS OF CALCIFICATIONS PATTERNS CALCIFIED OUTLINE RADIOGRAPHIC APPEARANCES Thin radiopaque cortex and a central lucency that constitutes most of the process. Thicker radiopaque outline with almost complete opacification as well as small and occasionally discontinuous radiolucent core. Knobby or scalloped outline which may be partially calcified with varying degree of central radiolucency. Totally radiopaque with no evidence of a radiolucent interior.

PARTIALLY CALCIFIED

NODULAR COMPLEX

COMPLETELY CALCIFIED
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REFERENCES

Fig. 4: Three dimensional reconstruction demonstrating enlargement of styoid process on either sides

neuralgia of sphenopalatine ganglia, temporomandibular arthritis, glossopharyngeal and trigeminal neuralgia, chronic tonsillo-pharyngitis, hyoid bursitis, Sluder's syndrome, histamine cephalgia, cluster type headache, esophageal diverticula, temporal arteritis, cervical vertebral arthritis, benign or malign neoplasms, and migraine type headache[8, 9]. Several imaging modalities have been used for the diagnosis of Eagle's syndrome thus far, including lateral head and neck radiograph, Towne radiograph, panoramic radiograph, lateral-oblique mandible plain film, anteroposterior head radiograph, and CT. Also, barium swallow studies can show the indentation of the elongated styloid process as a filling defect[10] . Superimposition of several osseous structures, and distortion and magnifications secondary to angulations are the potential disadvantages of conventional radiographs and, in particular, panoramic films. 3D-CT images reformatted from the raw data obtained with a spiral scanner provide all the information about the styloid process, including its length, direction, and anatomical relations. 3D-CT is an objective diagnostic tool to outline the anatomy, tailor the surgical plan, and offer a detailed explanation to the patients as well. Another advantage of the 3D-CT images is three dimensional length measurements, which are impossible in 2D images[11,12]. In conclusion, 3D-CT is a valuable diagnostic imaging tool in patients with Eagle's syndrome that allows clinicians to evaluate the styloid process in spatial geometry, make accurate length measurements, and explain the problem in detail to patients, all of which make this technique superior to conventional imaging modalities.

Frommer J. Anatomic variations in the stylohyoid chain and their possible clinical significance. Oral Surg 1974; 38:659667. 2) Eagle WW.Elongated styloid process :Report of two cases.Arch Otolaryngol 1937;25:584-587. 3) Lengele B,Dhem A.Microradiographic and histological study of styloid process of temporal bone.Acta Anat1989;135;193199. 4) L a n g l a i s R P, L a n g l a n d O E , N o r t j e C J . S o f t t i s s u e radiopacities.Chapter 19. Diagnostic imaging of the jaws.Philadelphia : A Lea and Febiger.1995. p.617-621 ' 5) K.C.Prasad et al, Elongated styloid process (Eagle's syndrome): a clinical study, Journal of Oral and Maxillofacial Surgery, vol. 60, no. 2, pp. 171175, 2002.. 6) Balbuena L, Hayes D, Ramirez SG, Johnson R. Eagle's syndrome (elongated styloid process). South Med J 1997; 90:331-334. 7) Fini et al. The long styloid proc- ess syndrome or Eagle's syndrome J Craniomaxillofac Surg 2000; 28:123-127. 8) Harma R. Stylalgia: clinical experiences of 52 cases. Acta Otolaryngol 1966; 224:149. 9) Politi M, Toro C, and Tenani G.A Rare Cause for Cervical Pain: Eagle's Syndrome. International Journal of Dentistry Volume 2009, Article ID 781297, 1-3. 10) A Savranlar, L Uzun, M Birol Uur, T zer.Threedimensional CT of Eagle's syndrome. Diagn Interv Radiol 2005; 11:206-209. 11) Chiang C, Liao Y, Yang W. Three-Dimensional Reconstruction CT in Diagnosis of Eagle's Syndrome: a Retrospective Study. Chin J Radiol 2006; 31: 221-225. 12) LEE S K. Eagle's syndrome with 3-D Reconstructed CT:two cases report .Chin J Radiol 2004; 29: 353-357.

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