Surgical management of patients with Eagle syndrome

Abstract

Objective

Eagle syndrome, a spectrum of disease resulting from an elongated styloid process and/or calcified stylohyoid ligament, lacks standardized recommendations regarding indications for surgical intervention and approach.

Study design

Retrospective cohort study.

Setting

Single tertiary care institution.

Subjects

Patients treated surgically for Eagle syndrome between January 2011 and June 2017.

Methods

Patients were diagnosed with Eagle syndrome based on thorough clinical workup and assessment. The primary outcome was improvement in pain severity following surgery, with complete resolution of pain being considered clinically meaningful. Wilcoxon rank-sum tests and Fisher's exact were used to compare numerical and categorical variables, respectively.

Results

Twenty-one patients were diagnosed with Eagle syndrome and underwent surgical resection of the styloid process. Patients most often complained of neck pain (81%), throat pain (62%), and ear pain (48%). Among these patients, 57% of procedures featured a transcervical approach, while the remaining 43% were transoral. The vast majority (90%) of patients experienced improvement in pain severity from a median of 6.0 before surgery to 0.0 afterwards (p < 0.01) as 62% experienced complete resolution. Using multivariable linear regression to model changes in pain severity, neck pain (β = −1.69, p < 0.01) and jaw pain (β = −0.93, p = 0.03) predicted greater relief, while headache (β = 0.82, p = 0.04) predicted an inferior response. Adverse events were uncommon and typically resolved within three months, with 24% experiencing first bite syndrome and 19% reporting numbness.

Conclusions

Transcervical and transoral styloidectomy are effective treatments for Eagle syndrome with minimal adverse effects. Patients with classic symptoms of neck or jaw pain benefit most from surgery.

Introduction

Eagle syndrome, first described by Watt Eagle in 1937, is the symptomatic elongation of the styloid process and/or calcification of the stylohyoid ligament [1]. An elongated styloid process is defined as a length of at least 25 mm, though only 4–10% of patients with elongated processes report pain, and it is often an incidental finding on imaging [2]. While there is no correlation between the degree of elongation of the styloid process and the severity of the symptoms, Okur et al. reports that an increase in medial angulation of the styloid process may be responsible for symptomatic presentations [3,4]. The frequency of this syndrome is estimated to be 4–8 per 10,000 people [5]. It is characterized by a pattern of pain in the head and neck region that commonly includes several, but not all of the following symptoms: odynophagia, dysphagia, tinnitus, cervicofacial pain, and globus sensation [6]. The etiology of pain associated with Eagle syndrome can be multifactorial, but in most cases is the result of compression of various cranial nerves (V, VII, IX, and X) or local chronic inflammation leading to osteitis, periostitis, and/or tendinosis [7,8]. One rare but well-characterized contributing factor is post-tonsillectomy fibrosis at the styloid apex that can compress neighboring nervous structures [9].

Eagle syndrome is a diagnosis of exclusion that can be corroborated by a thorough history and presentation with typical symptoms, physical exam that reveals a palpable styloid process in the tonsillar fossa, and a neck CT scan or panorex that reveals consistent anatomy [3,10]. Injection of lidocaine along the styloid process can be useful in establishing the diagnosis [6]. The diagnosis is confirmed if the lidocaine injection leads to temporary, quick-onset relief of symptoms. While the literature does address the surgical management of Eagle syndrome, there is no definitive consensus on whether the transoral or transcervical approach is superior [[11], [12], [13]]. Furthermore, the indications for surgical intervention are not standardized; thus, surgical treatment of Eagle syndrome has led to mixed results.

The objective of this study was to evaluate patients diagnosed with Eagle syndrome treated surgically at our tertiary care institution. We sought to characterize this patient population and assess their outcomes following surgical intervention, such as changes in pain severity and frequency of adverse events. Finally, we aimed to identify patient characteristics that predict better responses to surgery.