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Stylohyoid Complex Syndrome a New Diagnostic Classification

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ORIGINAL ARTICLE Stylohyoid Complex Syndrome A New Diagnostic Classification Candice C. Colby, MD; John M. Del Gaudio, MD Objective: To describe stylohyoid complex syndrome Intervention: The pathologic areas were surgically ad- (SHCS) as a new diagnostic classification of all lateral neck dressed through transoral or cervical approaches. and/or facial pain conditions resulting from an elon- gated styloid process, ossified stylohyoid ligament, or elon- Main Outcome Measure: Symptoms following sur- gated hyoid bone. All of these pathologic conditions re- gical intervention. sult in tension and reduced distensibility of the stylohyoid complex (SHC), with resultant irritation of the surround- Results: Seven patients (8 sides) were identified as hav- ing cervical structures with movement of the complex. ing SHCS. Computed tomographic findings included elon- gated styloid processes (3 sides), ossified stylohyoid liga- Design: A retrospective medical chart review was per- ments (2 sides), and elongated hyoid bones (3 sides). formed to identify a cohort of patients who underwent Computed tomographic scan, frequently with volume- surgical intervention for lateral neck and/or facial pain rendered 3-dimensional reconstructions, identified the due to pathologic SHCS. Follow-up time of greater than pathologic condition. All patients experienced clini- 1 year is reported in 5 of 7 patients. cally significant relief of presenting symptoms follow- Setting: Tertiary, academic referral center. ing surgical intervention. Patients: Patients included were those given a diagno- Conclusions: Stylohyoid complex syndrome includes all sis of SHCS who underwent surgical intervention from lateral neck and/or facial pain conditions resulting from June 2006 through September 2009. There were 7 pa- an elongated styloid process, ossified stylohyoid liga- tients, 5 of whom were female. The age range was 38 to ment, or elongated hyoid bone. Surgical intervention di- 53 years at time of presentation (mean age, 45.3 years). rected at any pathologic point to disrupt this complex Common presenting complaints were lateral neck and relieves tension and offers patients relief of symptoms. oropharyngeal pain exacerbated by tongue and head movements. Arch Otolaryngol Head Neck Surg. 2011;137(3):248-252 HE STYLOHYOID COMPLEX ated with these structures, which can in- (SHC) consists of the sty- clude an elongated styloid process, ossified loid process, the stylohy- stylohyoid ligament, or elongated hyoid oid ligament, and the lesser bone, can cause cervical and pharyngeal cornu of the hyoid bone. symptoms including lateral neck pain in the TThe SHC arises from 4 cartilage seg- area of the angle of the mandible, subman- ments: the tympanohyal, stylohyal, cera- dibular space, and upper neck that is exac- tohyal, and hypohyal.1 The styloid pro- erbated with speaking, swallowing, chew- cess originates from the temporal bone ing, yawning, head turning, and other oral medial and anterior to the stylomastoid fo- and cervical movements. The symptoms are ramen and runs anteromedially, bor- likely related to irritation of the structures dered on either side by the internal and around the SHC, including the carotid ar- external branches of the carotid artery. teries and the cranial nerves VII, IX, and X. Three muscles (stylopharyngeus, stylohy- We believe that the pathologic conditions oid, and stylomandibular) and 2 liga- of the individual structures result in re- ments (stylohyoid and stylomandibular) duced distensibility of the SHC, resulting in originate at the styloid process, with the compression of the surrounding neurovas- stylohyoid ligament and muscle insert- cular structures with movement of the com- Author Affiliations: 2 Department of ing on the lesser cornu of the hyoid bone. plex. In addition, tightness of the complex Otolaryngology–Head and This complex traverses the area from the can result in a more posterior position of the Neck Surgery, Emory University lateral skull base to the anterolateral neck, greater horn of the hyoid bone, bringing it School of Medicine, Atlanta, forming a band that crosses the upper lat- into closer proximity of the neurovascular Georgia. eral neck. Pathologic conditions associ- structures of the lateral neck. (REPRINTED) ARCH OTOLARYNGOL HEAD NECK SURG/ VOL 137 (NO. 3), MAR 2011 WWW.ARCHOTO.COM 248 Downloaded from www.archoto.com at , on May 18, 2011 ©2011 American Medical Association. All rights reserved. The first description of a syndrome of vague orocervi- ing structures with head and neck movement owing to cofacial pain was in 1937 by Watt Eagle, MD, an otolar- the lack of distensibility of the complex. Surgery to in- yngologist.3 His first 2 cases were patients with elongated terrupt this complex at any point is likely to improve the styloid processes presenting with orocervical facial pain. symptoms by relieving the tension of the SHC. We sug- Eagle considered any styloid process longer than the nor- gest a new diagnostic classification of stylohyoid com- mal adult length of 25 mm to be abnormal4,5 and found plex syndrome (SHCS) to include the previously de- that 4% of the population had elongated styloid pro- scribed Eagle syndrome, stylohyoid syndrome, and cesses, but only 4% of these individuals complained of any pseudostyloid syndrome because all entities originate from symptoms.5 Subsequent studies have estimated the inci- pathologic conditions within the SHC. Herein, we re- dence of elongated styloid process to be as low as 1.4% by view our series of patients with SHCS. Gossman and Tarsitano,6 who analyzed 4200 panoramic radiographs of men 18 to 22 years old, and as high as 30% METHODS by Keur et al7 in a study on 1135 men and women 18 to 91 years old. Keur et al7 found the incidence of elongated styloid processes to be equal in men and women, al- A retrospective medical chart review was performed at an aca- though women displayed symptoms consistent with Eagle demic tertiary care center of patients who presented with lat- syndrome more frequently. Gossman and Tarsitano6 stud- eral neck and/or facial pain and underwent surgical interven- ied only panoramic radiologic films of young males and tion for SHCS from June 2006 through September 2009. Demographic data, presenting symptoms, radiologic findings, acknowledged that most symptomatic patients are women surgical procedures, and outcomes were gathered. Patients were older than 30 years; thus, they stated the incidence of elon- asked to rate the percentage improvement of their pain symp- gated styloid processes is likely closer to 4%. toms at the first postoperative visit approximately 2 weeks af- Eleven years after Eagle first made his diagnosis, he ter surgery and at all subsequent visits. described4 2 classifications of the syndrome: the first with the typical symptoms described herein, and the second as a carotid artery syndrome.8 Patients classified as hav- RESULTS ing the carotid artery syndrome exhibited migraines, clus- ter headaches, or transient neurological symptoms caused Seven patients (8 sides) were identified. The age range by irritation of the periarterial sympathetic nerve plexus was 38 to 53 years at the time of presentation (mean age, overlying the artery in the area of pain distribution. Eagle 45.3 years). Five of the 7 patients were female. Com- syndrome has also been reported in the neurosurgical lit- mon presenting complaints were lateral neck and oro- erature as a cause for transient ischemic attack in a pa- pharyngeal pain exacerbated by tongue and head move- tient presenting with severe reversible narrowing of the ments, with tenderness over the affected structures when internal carotid artery (ICA) on head movement to the palpated during these activities. All patients underwent affected side.8 The patient experienced right-sided weak- computed tomographic (CT) scan of the neck with con- ness and syncope when he turned his head to the left, trast with multiplanar reconstructions, and when nec- and on angiography there was a filling defect of the cer- essary 3-dimensional volume-rendered reconstructions vical ICA at the level C2-3, found to be caused by an os- were performed of the bony anatomy to show spatial re- seous structure on imaging. This patient was treated suc- lationships. The pathologic conditions identified on CT cessfully by transcervical styloid process excision with imaging included 3 elongated styloid processes (1 bilat- resolution of symptoms. eral), 2 ossified stylohyoid ligaments, and 3 elongated hy- Camarda et al9 previously classified this constellation oid bones. One of these patients had bilateral ossified sty- of symptoms into 3 distinct entities: Eagle syndrome as lohyoid ligaments, and 1 had bilateral elongated styloid classically described with prior trauma, stylohyoid syn- processes, but each underwent unilateral resection due drome, and pseudostyloid syndrome. Stylohyoid syn- to unilateral symptoms. Two patients had a history of drome was the most common of the 3, and applied when trauma or tonsillectomy. a patient’s symptoms appeared earlier in life owing to a The surgical approach was determined by the location developmental anomaly of ossified stylohyoid ligaments of the pathologic conditions along the SHC identified on or elongated styloid processes, with no associated trauma. examination and imaging. All operations were per- Pseudostyloid syndrome is caused by tendinosis at the junc- formed in a same-day surgical suite under general anes- tion of the stylohyoid ligament and the lesser cornu of the thesia,
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