SURGICAL ONCOLOGY AND RECONSTRUCTION

Eagle’s Syndrome Associated With Lingual Nerve Paresthesia: A Case Report Zhiwei Dong, PhD,* Haihong Bao, MD,y Li Zhang, PhD,z and Zequan Hua, MDx Eagle’s syndrome is characterized by a variety of symptoms, including throat pain, sensation of a foreign body in the pharynx, dysphagia, referred otalgia, and neck and throat pain exacerbated by head rotation. Any styloid process longer than 25 mm should be considered elongated and will usually be responsible for Eagle’s syndrome. Surgical resection of the elongated styloid is a routine treatment and can be accomplished using a transoral or an extraoral approach. We report a patient with a rare giant styloid process that was approximately 81.7 mm. He complained of a rare symptom: hemitongue paresthesia. After removal of the elongated styloid process using the extraoral approach, his symptoms, including the hemitongue paresthesia, were alleviated. We concluded that if the styloid process displays medium to severe elongation, the extraoral approach will be appropriate. Ó 2014 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 72:886.e1-886.e4, 2014 Styloid process syndrome (Eagle’s syndrome) was first reported in 1937.1 It is characterized by a variety of symptoms, including throat pain, pharyngeal foreign body sensation, dysphagia, hemifacial pain, headache, referred otalgia, and neck and throat pain that is exacerbated by head rotation. The cause of these symptoms has been thought to be an elongated styloid process, which can result in pressure or impingement on the muscles or ligaments near the styloid process. Because the symptoms vary and are nonspecific— similar signs can appear with diseases other than Eagle’s syndrome—the diagnosis can be difficult. The differential diagnosis of Eagle’s syndrome should include all diseases causing cervicofacial pain, including trigeminal, sphenopalatine, and glossopharyngeal neuralgias, among others. Eagle thought that any styloid process greater than 25 mm should be considered elongated and would usually be responsible for Eagle’s syndrome.2-4 Exacerbation of pain during palpation of the tonsillar fossa could alert clinicians to the possible diagnosis of Eagle’s syndrome. Thus, it is often

diagnosed by radiography and physical examination, including palpation of the tonsillar fossa. Several radiographic examinations can be helpful for diagnosing Eagle’s syndrome, including panoramic radiography, conventional coronal and axial computed tomography (CT), and 3-dimensional CT (3D-CT). Some researchers have found that 3D-CT has advantages compared with other radiographic methods.5,6 Eagle’s syndrome can be treated pharmacologically or surgically, or both. Resection of an elongated styloid process has been thought to be the method of choice for treating Eagle’s syndrome. However, a question exists about which approach is better: the transoral or extraoral approach.7-9 Each has advantages and disadvantages. The choice of treatment usually depends on the clinical situation and surgeon experience. We report a case of a huge elongated styloid process in a patient with the rare symptom of hemitongue paresthesia. The styloid process was so large it would have been difficult to remove using the transoral approach. Thus, extraoral resection was

Received from Department of Oral and Maxillofacial Surgery,

Address correspondence and reprint requests to Dr Hua: Depart-

General Hospital of Shenyang Military Command, Shenhe District, Shenyang, People’s Republic of China.

ment of Oral and Maxillofacial Surgery, General Hospital of Shenyang Military Command, No 83 Wenhua Rd, Shenhe District, Shenyang

*Attending Physician.

110840, People’s Republic of China; e-mail: [email protected]

yAttending Physician.

Received December 4 2013

zAssociate Professor.

Accepted February 1 2014

xDepartment Head.

Ó 2014 American Association of Oral and Maxillofacial Surgeons

Drs Hua and Dong contributed equally to this work and should be

0278-2391/14/00173-6$36.00/0

regarded as joint corresponding authors.

http://dx.doi.org/10.1016/j.joms.2014.02.011

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performed, avoiding the disadvantages of the transoral approach.

Case Report A 59-year-old man presented to our hospital with a 12-year history of left neck pain radiating to the temporal and posterior cervical regions. The pain was exacerbated when he turned his head to the left or raised his left arm. He also complained of paresthesia on the left side of his tongue that had been present for more than 1 decade. He experienced blunt pain when we pierced the tongue with a syringe needle. He also reported the sensation of a foreign body in his left pharynx. During the physical examination, a hard mineralized process was palpable in the left submandibular region and was associated with tenderness. The pain was exacerbated during palpation of the tonsillar fossa. 3D Computed tomography angiography (3D-CTA) was undertaken. Quantitative measurements showed a left styloid process (81.7 mm; Fig 1) that was longer

than that on the right side (31.5 mm). Also, many vessels were present around the left styloid process. Because of the patient’s symptoms, he was diagnosed with Eagle’s syndrome. The patient was so informed, and a surgical solution was recommended. Because the styloid process was huge, the transoral approach was not advocated. The operation was performed with the patient under general anesthesia, and the patient was placed in the supine position with the neck extended to the right. The Risdon approach was used. The tissue in the submandibular space was carefully separated using a vascular clamp to avoid vascular injury. Palpation was occasionally used during surgery to confirm the location of the styloid process. After exposure of the submandibular gland, we found that the inferior one third of the huge styloid process was underneath the space between the submandibular gland and the sternocleidomastoid muscle. Additionally, the lingual artery and lingual nerve were near the styloid process. The periosteum was incised at the tip of the styloid process and then stripped from the tip to the base.

FIGURE 1. Quantitative measurement of the computed tomography angiography (CTA) scan showed that the left styloid process (arrows) was approximately 81.7 mm. The CTA scan also showed that the styloid process passed between the internal and external carotid arteries. A, anterior; F, foot; H, head; P, posterior. Dong et al. Eagle’s Syndrome and Lingual Nerve Paresthesia. J Oral Maxillofac Surg 2014.

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FIGURE 2. The resected portion of the styloid process was approximately 74 mm. The arrows showed the superior and inferior part of the styloid process. Dong et al. Eagle’s Syndrome and Lingual Nerve Paresthesia. J Oral Maxillofac Surg 2014.

The styloid process, which was approximately 74 mm long, was excised using a bone-nibbling rongeur (Fig 2). After resection of the styloid process, the incision was sutured layer by layer. The follow-up examinations showed an uneventful recovery with no complications. One week postoperatively, the patient reported that he had no pain in the cervical or temporal region (including when he had turned his head left or raised his left arm). The left pharyngeal foreign body sensation had also disappeared, and the hemitongue paresthesia had been alleviated.

Discussion The symptoms of Eagle’s syndrome are varied and nonspecific, making it difficult to diagnose precisely. The common complaints are throat pain, the sensation of a pharyngeal foreign body, dysphagia, hemifacial pain, headache, referred otalgia, and neck and throat pain exacerbated by head rotation. Additional symptoms can include neck or throat pain, with radiation to the ipsilateral ear and temporal regions. More rarely, such as in our case, the patient will present with the rare symptom of paresthesia of the hemitongue. The differential diagnosis of Eagle’s syndrome should include all diseases that cause cervicofacial pain. The differential diagnosis should also include all diseases causing paresthesia of the tongue (such as in our patient), including trigeminal, sphenopalatine, and glossopharyngeal neuralgias, temporomandibular joint diseases, otitis media,

external otitis, dental pain, mastoiditis, submandibular sialadenitis or sialolithiasis, and tumors of the pharynx or tongue base.10 Patients with Eagle’s syndrome who are misdiagnosed can undergo unnecessary treatment and experience unnecessary pain. 3D-CTA is an effective tool for diagnosing Eagle’s syndrome. It has many advantages compared with other radiographic methods (eg, panoramic radiography, conventional coronal and axial CT, 3D-CT). It visualizes and can calculate data associated with the styloid process, including its length and direction. In our case, 3D-CTA not only provided precise anatomic images and the relation between the styloid process and adjacent tissues, especially the vessels, but it was also useful for planning the surgery to remove this huge styloid process. Once the diagnosis of Eagle’s syndrome has been established, one must determine whether conservative or surgical treatment will be appropriate. Conservative management has included analgesics and local corticosteroid or anesthetic administration—with short-lived results.11 To obtain long-lasting symptom relief, surgical removal of the long portion of the styloid process has generally been accepted as the primary treatment modality for Eagle’s syndrome. Several transoral and extraoral cervical approaches have been described.7,12 One advantage of the extraoral approach is that it provides adequate anatomic exposure of the styloid process and its adjacent tissues. A clear surgical view is important to avoid neurovascular injury. Thus, if major vessel hemorrhage is encountered, it can be

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managed in a well-visualized, controlled manner. The sterility of the surgical method greatly reduces the risk of surgical site infection. The extraoral approach could also leave the shortest residue of the styloid process. The main disadvantage of the extraoral approach is the postoperative cosmetic deformity resulting from scar formation. Other disadvantages include the long duration of surgery, extensive facial dissection, and uncomfortable paresthesia of cutaneous nerves.13,14 In contrast, the transoral approach for resecting the styloid process is easy to perform and causes no cosmetic deformity. The operation duration and recovery times are short. Apparently, the main risk of the transoral approach is damage to the major blood vessels. 10 Other disadvantages of the transoral approach include deep cervical infection and temporary edema that can cause respiratory obstruction. Furthermore, it is difficult to remove a huge styloid process using the transoral route. The treatment choice usually depends on the clinical practice and surgeon experience. For our patient, we chose to resect the styloid process using the extraoral approach. We had at least 3 reasons for the choice. First, the anatomic structures around the apex of the styloid process are complicated. The styloid process is a bony projection arising from the lower surface of the temporal bone. It originates from Reichert’s cartilage of the second branchial arch and persists as a structure running from the base of the skull to the lesser horn of the hyoid. The muscles and ligaments that attach the styloid process include the stylopharyngeal muscle, styloglossus muscle, stylohyoid muscle, stylohyoid ligament, and stylomandibular ligament. The styloid process is located between the internal and external carotid arteries. The facial nerve runs anterior and medial to the styloid process. The glossopharyngeal, accessory, vagus, and hypoglossal nerves were in close proximity to the styloid process. Thus, to obtain a clear surgical view to avoid neurovascular injury, the extraoral approach was the best choice. Second, the patient was experiencing paresthesia of the left side of his tongue (which has rarely been reported in published studies). The symptom was considered to have been caused by the elongated styloid process, which might be exerting pressure or impinging on the lingual and glossopharyngeal nerves. The extraoral approach was thus chosen to obtain the best result for nerve decompression. Finally, 3D-CTA showed that the left styloid process in this patient was huge. Generally, if the styloid process elongation is medium to severe, the transoral approach will not be advocated, in accor-

dance with the findings from Chase et al.8 We thought that the styloid process was too large to withdraw it using the transoral approach without inducing neurovascular injury. In conclusion, the present patient had a rare, hugely elongated styloid process that might be exerting pressure or impinging on the lingual and glossopharyngeal nerves simultaneously. This could have been causing the hemitongue paresthesia of which the patient complained. The symptoms have rarely been reported in published studies. We found that 3D-CTA is one of best radiographic tools for studying the surrounding tissues, especially the vessels near the styloid process. 3D-CTA was also useful for establishing the diagnosis and planning the treatment of Eagle’s syndrome in our patient. Finally, if the degree of elongation of the styloid process is medium to severe, it will be difficult to withdraw it using the transoral approach without causing neurovascular injury. Thus, the extraoral approach should be recommended for these patients.

References 1. Eagle WW: Elongated styloid process: Report of 2 cases. Arch Otolaryngol 25:584, 1937 2. Eagle WW: Symptomatic elongated styloid process. Arch Otolaryngol 49:490, 1949 3. Eagle WW: Elongated styloid process: Symptoms and treatment. Arch Otolaryngol 64:172, 1958 4. Gossman JR, Tarsitano JJ: The styloid-stylohyoid syndrome. J Oral Surg 35:555, 1977 5. Nakamaru Y, Fukuda S, Miyashita S, et al: Diagnosis of the elongated styloid process by three-dimensional computed tomography. Auris Nasus Larynx 29:55, 2002 6. Bafaqeeh SA: Eagle syndrome: Classic and carotid artery types. J Otolaryngol 29:88, 2000 7. Diamond LH, Cottrell DA, Hunter MJ, et al: Eagle’s syndrome: A report of 4 patients treated using a modified extraoral approach. J Oral Maxillofac Surg 59:1420, 2001 8. Chase DC, Zarmen A, Bigelow WC, et al: Eagle’s syndrome: A comparison of intraoral versus extraoral surgical approaches. Oral Surg Oral Med Oral Pathol 62:625, 1986 9. Buono U, Mangone GM, Michelotti A, et al: Surgical approach to the stylohyoid process in Eagle’s syndrome. Oral Surg Oral Med Oral Pathol 63:714, 2005 10. Beder E, Ozgursoy OB, Karatayli Ozgursoy S: Current diagnosis and transoral surgical treatment of Eagle’s syndrome. J Oral Maxillofac Surg 63:1742, 2005 11. Prasad KC, Kamath MP, Reddy KJ, et al: Elongated styloid process (Eagle’s syndrome): A clinical study. J Oral Maxillofac Surg 60:171, 2002 12. Fini G, Gasparini G, Filippini F, et al: The long styloid process syndrome or Eagle’s syndrome. J Craniomaxillofac Surg 28:123, 2000 13. Matsumoto F, Kase K, Kasai M, et al: Endoscopy-assisted transoral resection of the styloid process in Eagle’s syndrome: Case report. Head Face Med 30:21, 2012 14. Chrcanovic BR, Custodio AL, de Oliveira DR: An intraoral surgical approach to the styloid process in Eagle’s syndrome. Oral Maxillofac Surg 13:145, 2009

Eagle's syndrome associated with lingual nerve paresthesia: a case report.

Eagle's syndrome is characterized by a variety of symptoms, including throat pain, sensation of a foreign body in the pharynx, dysphagia, referred ota...
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