Own experience in the diagnosis and treatment of elongated styloid process syndrome

ABBREVIATIONS

3D-CT – 3D computed tomography reconstruction CBCT – cone beam computed tomography CT – computed tomography

INTRODUCTION

Eagle’s syndrome represents a cluster of symptoms associated with the elongated styloid process of the temporal bone, which is often accompanied by a calcified stylohyoid ligament. It is a rare disease entity and its prevalence is difficult to determine, however previ- ous studies suggest it occurs in 2% to 11.8% of the total population with only 4% of this group reporting symptoms [1–4]. The styloid process is an osseous bone structure located on the surface of the inferior aspect of the temporal bone and ranges from 20 to 30 mm in length in Caucasian adults [5]. The facial nerve extends forward and medially from the styloid process while the accessory nerve and the vagus nerve extend medially. The glossopharyngeal nerve

Own experience in the diagnosis and

treatment of elongated styloid process

syndrome

Magdalena Sylwia Wacławek

, Piotr Pietkiewicz

, Piotr Niewiadomski

, Jurek Olszewski

1Department of Otolaryngology, Laryngological Oncology, Audiology and Phoniatrics, USK WAM, Lodz, Poland;

Head: prof. Jurek Olszewski MD PhD

2II Department of Otolaryngology, Medical University of Lodz, Poland; Head: prof. Jurek Olszewski MD PhD

Article history: Received: 31.07.2020 Accepted: 24.08.2020 Published: 09.09.2020

SUMMARY: Introduction: The aim of the study was to present selected cases with Eagle’s syndrome diagnosed and treated at the Department of Otolaryngology, Laryngological Oncology, Audiology and Phoniatrics, Medical University of Lodz, in the years 2016–2020.

Material and method: Five selected clinical cases with Eagle’s syndrome are presented, including three male patients and two female patients, aged 28 to 42 years.

Results: The prevalence of Eagle’s syndrome was similar for both female and male patients. The carotid artery syndrome, which is characterized by: visual disturbances, unilateral pain located along cervical vessels, headache and migraine, was more common.

Lack of proper diagnosis of Eagle’s syndrome often significantly delays the implementation of proper treatment and thus exposes patients to long-term struggle with pain. Our observations have shown that the time between the onset of symptoms and correct diagnosis in patients averaged about five years. 3D-CT scan is the gold standard for detecting Eagle’s syndrome. An intraoral approach was used in surgical treatment. Although this approach offers shorter treatment time and better cosmetic effect, there is also a greater risk of complications associated with limited visibility of the surgical field and infection.

Conclusions: Although styloid syndrome usually occurs bilaterally, these patients reported unilateral symptoms. No correlations were found between the prevalence of Eagle’s syndrome and sex, the length of the styloid process or age, nor side of the body. The best healing effect is obtained by surgical correction/reduction of the elongated styloid process.

KEYWORDS: diagnostics, elongated styloid process syndrome, treatment

Authors’ Contribution:

A – Study Design B – Data Collection C – Statistical Analysis D – Manuscript Preparation E – Literature Search F – Funds Collection

also runs in a short distance. The tip of the styloid process is located between the internal and external carotid artery [6, 7]. Anatomi- cal location is important in the pathophysiology of the disease.

There are three main classifications of Eagle’s syndrome [8], with the oldest introduced by Eagle being used most often [9]. It distin- guishes a classic form associated with tonsillectomy and another so-called carotid artery syndrome. The former occurs as a compli- cation following tonsillectomy – shifting of the scar tissue towards the elongated styloid process causes irritation of the facial, glosso- pharyngeal and vagus nerves. The latter is caused by mechanical compression of the elongated styloid process on the sympathetic fibers of the carotid plexus [10, 11]. Other classifications concern alterations of the styloid process structure based on the result of radiological examination [12] and the location of the styloid pro- cess tip in relation to the mandibular foramen [13].

Patients with Eagle’s syndrome report various nonspecific symp- toms. These may include: earache, sore throat and lateral neck pain occurring when opening the mouth or turning the head, swallowing

with increased salivation, periodical trismus, the sensation of hav- ing a foreign body in the throat as well as neurological symptoms such as facial dysesthesia, unilateral headaches, visual disturbances, Horner’s syndrome (miosis, ptosis, enophtalmus) or even a tran- sient ischemic attack [14–17]. Hence, Eagle’s syndrome is most often diagnosed following the exclusion of other diseases.

After visiting numerous specialists (primary care physician, den- tist, otolaryngologist, neurologist) patients typically acquire con- siderable medical documentation and have undergone a number of imaging tests. However, the key to diagnosing elongated styloid process syndrome is the use of 3D computed tomography recon- struction (3D-CT), which allows accurate visualization with the as- sessment of the styloid process angle and its surroundings [18, 19].

Treatment of styloid syndrome may be conservative or surgical, however the literature reports that surgical methods demonstrate higher efficacy with about 80% of cases demonstrating complete regression of symptoms [20]. Conservative treatment, mainly used in the classic form of Eagle’s syndrome, involves injecting anes- thetic or glucocorticosteroids into the tonsillar fossa [14, 21–22].

The aim of the study was to present selected cases with Eagle’s syn- drome diagnosed and treated at the Department of Otolaryngol- ogy, Laryngological Oncology, Audiology and Phoniatrics, Medi- cal University Teaching Hospital Lodz, in the years 2016–2020.

CASE REPORTS

The first 28-year-old male patient (K.P.) was admitted to the De- partment of Otolaryngology, Laryngological Oncology, Audiol- ogy and Phoniatrics due to left neck pain radiating to the left ear.

The symptoms had occurred in varying severity for the previous three years. The pain intensified when moving the head, especially when bending the neck and swallowing. During the previous three years, the patient had been diagnosed and treated in another cen- ter. Upon first examination by an ENT specialist in 2013, a tumor of left submandibular gland was diagnosed and left submandibu- lar tumor resection was performed. Histopathological examina- tion revealed reactive lymph node enlargement.

In 2014, due to persistent pain and detection of inflammatory changes in the paranasal sinuses, the patient underwent bilateral medial antrostomy, left-sided frontoethmoidectomy and bilateral middle turbinate correction. Eagle’s syndrome was later diagnosed in 2015 in the same center. The patient was qualified for surgery by intraoral approach and after tonsillectomy (the first stage), sub- sequent stages of the procedure were abandoned. The patient was advised to continue treatment in another center.

Fig. 1. CT of the neck – 3D reconstruction before surgery. Styloid process – right side, 45 mm long, styloid process – left side, including calcification of the stylohyoid ligament about 39 mm long.

Fig. 2. CT of the neck – 3D reconstruction after surgery. Left styloid process after partial resection – presently 20 mm in length.

Fig. 3. CT of the neck – 3D reconstruction before surgery. Styloid process: 35 mm in length (right side), 32 mm in length (left side).

Fig. 4. CT of the neck – 3D reconstruction before surgery. Elongated styloid process on the left side measures 60 mm in length.

The patient underwent further clinical observation at the Depart- ment of Otolaryngology, Laryngological Oncology, Audiology and Phoniatrics at the Medical University Teaching Hospital in Lodz.

On the basis of clinical observation, after analyzing the test re- sults, with particular consideration of CT with 3D reconstruction of styloid processes (Fig. 1.), the patient was qualified for surgery under general endotracheal anesthesia.

On February 25, 2016, the resection of left styloid process was per- formed by intraoral approach. The surgery and postoperative pe- riod were uneventful. On the second postoperative day neck CT was performed (Fig. 2.) and the patient was discharged in good general and local condition.

CT of the neck was performed with 3D reconstruction after sur- gery (Fig. 2.), i.e. partial resection of the left styloid process. The styloid process measured 20 mm. Soft tissue swelling and numerous small gas bubbles were observed along the stylohyoid ligament on the left side, with secondary asymmetry at the level of oropharynx – these were believed to be postoperative changes. On the right side, the styloid process and calcification of the stylohyoid liga- ment were about 39 mm in length.

Within the next few days following surgery, after wound healing in the throat, the patient reported complete resolution of pain. He was asymptomatic at follow-up one year later.

A 42-year old female patient (A.C.) was admitted to the Department of Otolaryngology, Laryngological Oncology, Audiology and Pho- niatrics at the Medical University Teaching Hospital in November 2018 with persistent sore throat radiating to the right ear, right sided headache, tongue numbness and hoarseness. These symptoms had been of varying severity for the previous nine years. Therefore, the patient underwent bilateral tonsillectomy in October 2016. Due to persistent hoarseness, the patient was also qualified for micro- direct laryngoscopy with excision of vocal cord nodules (the pa- tient was a professional singer). The procedure was performed in March of 2017, but the pain persisted despite treatment. To aid the diagnosis, in September 2018 a neck CT scan with 3D recon- struction was performed (Fig. 3.). CT revealed elongation of the

styloid process, particularly on the right side (about 35 mm on the right side and about 32 mm on the left side). Furthermore, MRI examination of the temporomandibular joints a was performed as well as ultrasound of the neck, but no deviations were detected.

On the basis of the medical history, clinical observation, and after analysis of the test results with particular consideration of neck CT-3D reconstruction, the patient was diagnosed with right-sid- ed Eagle’s syndrome, and qualified for surgery under endotracheal anesthesia. On November 15, 2018 a partial resection of the ab- normally elongated right styloid process was performed by intra- oral approach. The surgery and postoperative period were un- eventful. After the throat wound had healed, the patient reported complete pain relief.

Seven months later, the patient returned to the Department of Oto- laryngology, Laryngological Oncology, Audiology and Phoniatrics at the Medical University Teaching Hospital, this time with a sore throat occurring when swallowing; she reported pain radiating to the left ear and numbness around the base of the tongue on the left side. Based on the imaging tests provided earlier, the patient was qualified for partial removal of the excessively elongated left sty- loid process by intraoral approach. The surgery was performed on June 6, 2019. No complications were observed at short and long- term follow up. Upon follow-up at three weeks after surgery, the patient reported complete resolution of pain.

Another patient (34-year-old female A.G.) presented a similar lengthy medical interview before a proper diagnosis was made.

She was admitted to the Department of Otolaryngology, Laryn- gological Oncology, Audiology and Phoniatrics at the Medical University Teaching Hospital in April 2019 for surgical treat- ment. She reported persistent headache in the parietal, temporal region on the right side radiating to the right retropharyngeal space. In addition, the patient reported a sensation of foreign body at the height of the thyroid cartilage on the right side and non-specific neck pain radiating down to the chest, also on the right side. Symptoms had been of varying severity for about four years. Previous diagnostics included numerous imaging tests, including neck ultrasound, cervical spine X-ray, contrast and no contrast MRI of the head, cervical spine and neck MRI, angio- CT of the head and head CT scan with 3D reconstruction. The patient was mainly diagnosed neurologically – chronic head- ache was recognized as the leading symptom. The sensation of the foreign body in the throat was explained by gastroesoph- ageal reflux and gastritis. Diagnostics were performed at the Department of Internal Medicine in January 2019 (esogastro- duodenoscopy, gastrointestinal X-ray, laboratory tests). Non- specific neck pain radiating to the right side of the chest was diagnosed in the pulmonary ward in December 2018, however, although bronchoscopy, chest X-ray, CT of the lungs were per- formed, the cause of the symptoms was not found. The patient reported that she had been consulted with the Thoracic Surgery Department because of suspected persistent thymus, but this disease entity had been excluded. The doctors treating the pa- tient suggested degenerative changes of the cervical spine, but that would not explain all the reported symptoms. The break- through in diagnostics occurred in January 2019, when a CT

scan with 3D reconstruction of the head was performed and right-sided Eagle’s syndrome was diagnosed (about 36 mm on the right side and about 31 mm on the left side).

The patient was referred to the Department of Otolaryngology, Lar- yngological Oncology, Audiology and Phoniatrics for surgery. On the basis of the medical history and clinical observation, as well as after analysis of test results, with particular consideration of neck CT scan with 3D reconstruction, the patient was qualified for sur- gical procedure under general endotracheal anesthesia. On April 11, 2019 resection of the elongated styloid process was performed by intraoral approach. In the immediate postoperative period, the patient complained of a feeling of fluid entering the nasopharynx and pain on swallowing. After wound healing in the oropharynx, the patient reported complete regression of symptoms. After about seven months, due to recurrence of pain, the patient had a consulta- tion at another ENT center, where she was also qualified for surgical treatment. This time, the styloid process was resected by external approach. Unfortunately, the patient’s general and local condition did not improve.

Unlike other individuals, a 39-year old male patient (W.J.) was cor- rectly diagnosed quite quickly. He reported to the Department of Otolaryngology, Laryngological Oncology, Audiology and Phoniat- rics at the Medical University Teaching Hospital in October 2019, in a planned mode. He complained of persistent pain located along the left sternocleidomastoid muscle, intensifying during head move- ments, foreign body sensation in the throat and pain in the lateral wall of the throat on the left side occurring during swallowing. The pain began to increase in 2017; it had not previously been bother- some for the patient. Previous diagnostics included neck ultrasound and head CT scan with 3D reconstruction (Fig. 4.). According to the patient, he owed the rapid diagnostics to self-studying of medical knowledge and to an experienced ENT doctor whom he had found himself. Noteworthy is the fact that the elongated styloid process was easily palpated intraorally.

On the basis of the medical history and clinical observation, af- ter analysis of the test results, and with particular consideration of neck CT scan with 3D reconstruction, the patient was qualified for

surgical procedure under general endotracheal anesthesia. On Oc- tober 7, 2019 partial removal of the abnormally elongated left sty- loid process was performed by intraoral approach. An approximately 30-mm fragment of the distal portion of the left styloid process was excised with a bone nibbling rongeur. The surgery and postopera- tive period were uneventful. No complications were observed. At two-month follow-up, the patient was completely asymptomatic.

A 29-year old male individual (G.G.) was the next patient. He was admitted to the Department of Otolaryngology, Laryngological Oncology, Audiology and Phoniatrics at the Medical University Teaching Hospital in January 2020 with a complaint of a left-sided headache located in the projection of the ethmoidal and maxillary sinuses, radiating to the neck on the left side. The patient also re- ported a periodic sensation of numbness and tingling on the left side of the face. The symptoms had gradually increased for over three years and at the time of admission to the Department, they were extremely bothersome. The first symptoms included tooth- ache on the left side of the jaw, therefore the patient had reported to the dentist. A number of imaging tests were performed, includ- ing a pantomographic image and CBCT (cone beam computed to- mography) as well as comprehensive dental treatment (endodontic and extraction of tooth 28), but no improvement was obtained. In the meantime, blockage of the left sinus passage and rhinorrhea oc- curred, especially on the left side. The patient reported to the ENT office, where the a craniofacial CT scan was performed, which ex- cluded sinus headache and nasal steroids were administered due to suspected vasomotor rhinitis. Despite the applied therapies, left craniofacial pain intensified, and following the occurrence of peri- odic sensation disorders in the left craniofacial region, the patient was referred to the Department of Neurology. Numerous imaging tests were performed: targeted x-ray of the cervical spine, head MRI scan with contrast, cervical spine MRI, MRA of the cerebral arteries and a neck CT scan with contrast, which revealed an abnormally long styloid process on the left side (Fig. 5.).

On the basis of the medical history and clinical observation, after analysis of the test results, and with particular consideration of the neck CT scan with 3D reconstruction, the patient was qualified for surgical procedure under general endotracheal anesthesia. On January 16, 2020 partial removal of the abnormally elongated left styloid process was performed by intraoral approach. An approxi- mately 30-mm fragment of the distal portion of the styloid process was excised with a bone nibbling rongeur. The surgery was un- eventful. In the postoperative period, there were major edematous changes observed in the left neck area and periodic bleeding from the postoperative niche in the middle part of the throat on the left side. Steroid, antibiotic and antihemorrhagic treatment was applied.

The symptoms resolved and the patient was discharged after seven days of observation at the Department in good general and local condition. At two week follow-up, the pain reported by the patient before the surgery began to subside gradually.

DISCUSSION

The term Eagle’s syndrome derives from the name of an American otolaryngologist, Watt Weems Eagle, who in 1937 was the first to Fig. 5. CT of the neck – 3D reconstruction before surgery. Left side styloid process

elongated by 50 mm, right side styloid process 22 mm in length.

as confirmed by the fact that a neurologist had made the diagnosis in the case of patient G.G.

Computed tomography with 3D reconstruction is the gold standard in recognizing Eagle’s syndrome, however some reports indicate that cone beam computed tomography (CBCT) is also a good diagnostic tool [18, 31]. CBCT creates the possibility of precise visualization of the elongated styloid process and calcified ligaments as well as accurate linear measurements. One of the cases above (G.G.) underwent this examination but the results were not sufficient for a correct diagnosis.

When discussing the diagnosis of Eagle’s syndrome, it is impossible not to mention what we forget as 21st century physicians. Patients are referred for numerous specialized imaging tests and sometimes it is sufficient to carry out a well performed physical examination, as was the case for patient W.J., in whom the abnormally elongated styloid process was easily palpable in the oral cavity. The patient was referred by a laryngologist for head CT scan with 3D reconstruc- tion, on the basis of which he was subsequently qualified for surgery.

Intraoral and extraoral approaches are both used for surgical treat- ment. The intraoral approach offers shorter treatment time and better cosmetic effect. Unfortunately, these advantages are accompanied by a greater risk of complications associated with the limited visibility of the surgical site and infections [7, 32, 33]. All patients hospital- ized at the Department of Otolaryngology, Laryngological Oncol- ogy, Audiology and Phoniatrics of the Medical University Teaching Hospital as part of the presented study were operated on intraoral- ly. Apart from the discomfort associated with the surgery (painful swallowing, surgical site edema), no complications were observed.

Pain complaints typically subsided two to four weeks after surgery.

CONCLUSIONS

1. Although the styloid process syndrome occurs bilaterally, the symptoms reported by the patient are mostly one-sided;

2. The prevalence of Eagle’s syndrome is not influenced by sex, age, styloid process length or side of the body;

3. Computed tomography with 3D reconstruction is the gold stan- dard for detecting Eagle’s syndrome;

4. Best healing effects are given by surgery/shortening of the elon- gated styloid process.

distinguish a separate disease entity for symptoms associated with abnormal elongation of the styloid process of the temporal bone [9].

The incidence of diagnosed styloid syndrome is believed to range from 0.4 to 84.4% [10, 23, 24]. Although almost all cases demon- strate bilateral elongation, the symptoms reported by the patient are unilateral. This was the case with patient A.C. After surgery, she noticed that the symptoms on the operated side had resolved, how- ever, similar less severe symptoms arose on the other side, which had resolved after second surgery.

There is a dispute in the available literature whether Eagle’s syndrome is more prevalent in men [2, 14, 23, 28] or women [25–27]. Our ob- servations show similar prevalence of Eagle’s syndrome among male and female population.

Bożyk et al. [18] found no correlation between the prevalence of Ea- gle’s syndrome and gender and no correlation between the length of the styloid process and age. Similarly, no previous study has identi- fied any significant preference for the right or left side (among the described cases, four patients were operated on the left side and two on the right side).

The age range for patients who report to physicians is 30–50 years of life [22, 28, 29]. In line with this, the youngest case described herein was 28 years old, while the oldest was 42 years old.

Determination of the prevalence of the classic form compared to ca- rotid artery syndrome is difficult. Our observations to date suggest that carotid artery syndrome may be more common. It is character- ized by visual disturbances, unilateral pain located along the cervi- cal vessels, headache and migraine [15, 30]. Two of the five patients described in the present study underwent tonsillectomy, however, as the symptoms had occurred before the procedure, they cannot be classified as a typical classic form of the syndrome. The existing literature reports do not address this subject.

Lack of proper diagnosis of Eagle’s syndrome often significantly delays the implementation of proper treatment, and thus exposes patients to long-term struggle with pain [7]. We found the mean time from the onset of symptoms to correct diagnosis to be about 5 years. During this time, the patients had visited numerous spe- cialists, including dentists, primary care physicians, internal medi- cine physicians, neurologists, medical rehabilitation physicians, and physiotherapists before approaching an otolaryngologist. Obviously, Eagle’s syndrome can be diagnosed by any of the above specialists,

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prof. Jurek Olszewski MD PhD (ORCID: 0000-0002-8868-9679); Department of Otolaryngology, Laryngological Oncology, Audiology and Phoniatrics, USK WAM, Lodz; Żeromskiego street 113, 90-549 Lodz, Poland;

E-mail: jurek.olszewski@umed.lodz.pl

Waclawek M.S., Pietkiewicz P., Niewiadomski P., Olszewski J.: Own experience in the diagnosis and treatment of elongated styloid process syndrome; Otolaryngol Pol 2020; 74 (1-7); DOI: 10.5604/01.3001.0014.3366 (Advanced online publication)