Bilateral Stylohyoid Syndrome: A Rare Cause of Cervical Pain

Abstract

The first description of a syndrome of vague orocervicofacial pain was reported in 1937 by Watt Eagle, an otolaryngologist. Described as Eagle syndrome, it is a clinical condition in which the patient has a painful sensation in the head and neck region caused by elongation of the styloid process or calcification of the styloid ligament [1,2]. When the patient has no history of trauma or surgery, it is called stylohyoid syndrome [3]. Baugh and Stocks [4] defined an elongated styloid process as being 5-7 cm. Although an elongated styloid process and a calcified styloid ligament are found in 4% of the general population, only a small percentage are symptomatic [5]. Stylohyoid syndrome is an uncommon entity that appears primarily in the otolaryngology and oral surgery literature. Physiatrists may encounter patients with stylohyoid syndrome and should be aware of the clinical and radiographic presentation. The styloid process is a slender outgrowth at the base of the temporal bone immediately posterior to the mastoid apex. The styloid process arises embryonically from the second brachial arch, or Reichert cartilage. This cartilage can be divided into 4 sections based on the subsequent development of the stylohyoid complex. Mineralization may occur in various sites along the course of the stylohyoid ligament. Pseudoarticulations related to the embryologic development of stylohyoid ligament may form. Three types of pseudoarticulation have been reported [6]. Type I represents an uninterrupted, elongated styloid process. Type II is characterized by the styloid process apparently being joined to the stylohyoid ligament by a single pseudoarticulation, which gives the appearance of an articulated elongated styloid process. Type III consists of interrupted segments of the mineralized ligament, creating the appearance of multiple pseudoarticulations within the ligament. Camarda et al [3] classified the constellation of symptoms into 3 distinct entities: Eagle syndrome as classically described with previous trauma, stylohyoid syndrome, and pseudostyloid syndrome. Stylohyoid syndrome is the most common of the 3 entities. It is applied when a patient’s symptoms appear earlier in life as the result of a developmental anomaly of ossified stylohyoid ligaments or when a patient has elongated styloid processes with no associated trauma and symptoms that develop later in life. Pseudostyloid syndrome is caused by tendinosis at the junction of the stylohyoid ligament and the lesser cornu of the hyoid in older persons with no history of trauma and no radiologic evidence of styloid process elongation or stylohyoid ligament ossification. This case describes a 24-year-old male welder with a 2-year history of bilateral lateral neck pain. He reported severe neck pain with increasing intensity within a few months after he starting a full-time job as a welder. His work occasionally involved sudden movements of neck to either side that precipitated dizziness and, at times, facial pain below the eyes. He had no history of trauma or surgery. Active cervical range of motion demonstrated reduction in flexion and extension 15° 0° 10° and lateral flexion 20° 0° 20°, the latter producing pain and dizziness. The findings of the neurologic examination were normal. The oropharyngeal examination revealed a bilateral elongated styloid process that could be palpated intraorally posterior to the tonsillar fossa and elicited a painful sensation. Conventional anteroposterior and lateral radiographs of the cervical spine (Figure 1; digital radiograph, CURA Health Care Pvt Ltd, Tamilnadu, India) revealed no major pathologic findings within the spine but showed bilateral, elongated, excessively ossified styloid processes. Towne’s view of the skull (Figure 2) showed bilateral, elongated, ossified styloid processes. Maximum intensity projection computed tomography (CT) of the neck