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Styloid-Stylohyoid Syndrome: Literature Review and Case Report

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Styloid-Stylohyoid Syndrome: Literature Review and Case Report CASE REPORTS J Oral Maxillofac Surg 65:1346-1353, 2007 Styloid-Stylohyoid Syndrome: Literature Review and Case Report Felipe Ladeira Pereira, DDS,* Liogi Iwaki Filho, DDS, PhD,† Angelo José Pavan, DDS, PhD,‡ Gustavo Jacobucci Farah, DDS, PhD,§ Evelyn Almeida Lucas Gonçalves, DDS, PhD,ʈ Vanessa Cristina Veltrini, DDS, PhD,¶ and Edevaldo Tadeu Camarini, DDS, PhD# The first reports on enlargement of the styloid pro- although it has been divided into 4 syndromes in cess date from the 17th century, when anatomists recent literature.1 explored this region. These postmortem findings Differential diagnoses are innumerable because were only useful as a mere anatomic curiosity and had many of the symptoms detected in the enlargement of no clinical correlation. In 1937, W.W. Eagle reported the styloid process (orofacial pains and dysfunctions) various cases of a cervicopharyngeal symptomatology are also found in patients without presence of the and associated them with radiographic findings. It elongated process. Furthermore, its attachments may was believed that trauma in the cervicopharyngeal be susceptible to stretch and whiplash type injuries, region, especially after tonsillectomy, might stimulate in which an acute force exceeds the physiologic lim- a subsequent growth of the styloid process.1-3 its of the temporal bone attachment.9 Various names were proposed for the syndrome: The objective of this article is to present a com- styloid process neuralgia,4 styloid syndrome,5 stylohy- prehensive systematic review of literature and to oid syndrome,3 elongated styloid process syndrome,4 report a case of stylohyoid ligament ossification Eagle’s syndrome,6 and styloalgia.7,8 The most appro- with 2 pseudoarticulations. priate, however, is styloid-stylohyoid syndrome be- cause such anomalies may be of the styloid process, of the stylohyoid ligament, or a combination of both,3 Literature Review The styloid process is a slender projection con- *Oral and Maxillofacial Surgeon, Private Practice, São Paulo, Bra- nected to the inferior aspect of the petrous part of the zil. temporal bone just below the tympanic membrane †Professor of Oral and Maxillofacial Surgery, State University of and behind the tympanic plaque which shields its Maringá, Maringá, Paraná, Brazil. attachments. It lies behind the pharyngeal wall of the ‡Professor of Oral and Maxillofacial Surgery, State University of palatine fossa, between the internal and external ca- 9 Maringá, Maringá, Paraná, Brazil. rotid arteries. Innervation comprises the glossopha- §Professor of Oral and Maxillofacial Surgery, CESUMAR Denistry ryngeal nerve in the posterior lateral wall of the ton- School, Maringá, Paraná, Brazil. sillar fossa (medial to the process), and the facial ʈProfessor of Oral Pathology and Diagnosis, State University of nerve emerging from the stylomastoid foramen which Maringá, Maringá, Paraná, Brazil. is slightly posterolateral to the base of the styloid ¶Professor of Oral Pathology, Diagnosis, and Radiology, CE- process. The accessory nerve, the hypoglossal nerve, SUMAR Dentistry School Maringá, Paraná, Brazil. and the vagus nerve are placed medially to the pro- #Professor of Oral and Maxillofacial Surgery, State University of cess, together with the internal jugular vein and the 9,10 Maringá, Maringá, Paraná, Brazil. internal carotid artery with its sympathetic chain. Address correspondence and reprint requests to Dr Camarini: The normal size of the styloid process varies signif- Department of Dentistry, State University of Maringá, Av. Curitiba, icantly in the literature (Table 1). 1 486 Sala 701, CEP 87013-380, Maringá, Paraná, Brazil; e-mail: According to Camarda et al, the ceratohyal ele- [email protected] ment degenerates with time. Nonetheless, its fibrous © 2007 American Association of Oral and Maxillofacial Surgeons sheath persists as the stylohyoid ligament, containing 0278-2391/07/6507-0014$32.00/0 a cartilaginous and bone potential because the styloid doi:10.1016/j.joms.2006.07.020 process normally ossifies 5 to 8 years after birth. 1346 PEREIRA ET AL 1347 Table 1. NORMAL LENGTH OF THE STYLOID PROCESS ACCORDING TO THE LITERATURE Normal Length Each Side Gender Both Sides Left Right Male Female Frommer18 3-5 cm – – 3.26 cm 2.97 cm Eagle26 Ͻ2.5 cm – – – – Correl et al17 2.5 cm – – – – Langlais et al21 2.5 cm – – – – Montalbetti34 2.5 cm – – – – Gossman and Tarsitano3 2.5 cm – – – – Stafne and Hollinshead35 2-3 cm – – – – Lindeman36 2-3 cm – – – – Kaufman et al20 – 2.95 cm 2.99 cm – – Silva et al22 – 3.135 cm 3.372 cm – – Moffat et al7 1.52-4.77 cm – – – – Pereira et al. Styloid-Stylohyoid Syndrome. J Oral Maxillofac Surg 2007. Variations in the ossification and fusion of the 4 ele- would be justified by the theory of anatomic variation; ments of the second brachial arch (under appropriate and finally pseudostylohyoid syndrome, which is ex- stimulation) in youngsters may lead to a marked vari- plained by the theory of aging developmental anomaly.1 ation in the radiographic appearance of the whole The symptoms of classical Eagle syndrome and ca- stylohyoid chain. rotid artery syndrome are well described in litera- Steinmann5 proposed 3 theories to explain such ture.1,3,5,6,9,10,12-24 The symptoms of the other 2 are ossification. The theory of reactive hyperplasia im- the same as classical Eagle syndrome, but without plies that if the styloid process is adequately stimu- prior trauma history and involving a specific age lated, as in pharyngeal trauma, ossification would take group, usually above 40 years for pseudostylohyoid place in the terminal portion of the process at the syndrome but not necessarily for stylohyoid syn- expense of the stylohyoid ligament. The theory of drome. In classical Eagle syndrome, the chief com- reactive metaplasia also involves traumatic stimulus, plaint is continuous throat pain during the convales- which would induce some sections of the stylohyoid cent period in patients submitted to tonsillectomy apparatus to undergo metaplastic changes and and a sensation of foreign body lodged in the throat. thereby become intermittently ossified. The third the- Pain related to swallowing and speech is frequently ory, of anatomic variation, involves the stylohyoid referred to the ear on the side of the elongated styloid ligament and/or the styloid process as ossified struc- process.3 It is assumed that healing tonsillectomy scar tures that develop in the early formative years after tissue tightens the mucosa across the tip of the elon- birth. The theory may fit in radiographic findings of gated styloid process and that movements of this ossification in children and young adolescents and in mucosa during function across it is thought to cause the absence of antecedent cervicopharyngeal trauma the symptoms.9 Pharyngeal pains are theoretically (as an inductive stimulus).1 This could not be adjusted generated by stretching or fibrous compression of the to the classical Eagle syndrome because there is no V, VIII, IX, and X cranial nerve endings in the tonsillar prior trauma.11 fossa during the healing phase. When the stylohyoid Camarda et al1 added the theory of aging develop- ligament is ossified, contraction of the stylopharyn- mental anomaly, in which there is an increased inelas- geal muscle lifts the pharynx upward and laterally ticity of the soft tissue. This may lead to the develop- and, with the ossified ligament remaining fixed in this ment of tendinosis in the junction of the stylohyoid maneuver, the glossopharyngeal nerve is pulled ligament with the lesser cornu of the hyoid bone, across it during the swallowing act and may be stim- secondary to the increased ligament resistance to ulated mechanically to produce pain.3 Symptoms of joint movement (between ligament and bone) in dysphagia, pain referred to the ear, dysphonia, and some older patients. the sensation of a foreign body in the pharynx during As such, there are 4 syndromes: classical Eagle functional movements (eg, eating, yawning, turning syndrome and carotid artery syndrome, with the re- the head, and swallowing) are part of classical Eagle active hyperplasia and metaplasia theories of ossifica- syndrome.9 tion which explains the first and maybe the second An important feature of carotid artery syndrome is (once trauma is not necessarily the cause of the ca- that it is not dependent on a tonsillectomy. Because rotid artery syndrome); stylohyoid syndrome, which the styloid process lies between the internal and ex- 1348 STYLOID-STYLOHYOID SYNDROME ternal carotid, any deviation in the process or ossifi- patients over 40 years of age, while it was those over cation of the ligament may produce pressure on ei- 50 for Fini et al.19 ther of these structures and produce regional There are many differential diagnoses concerning carotidynia (pericarotid sympathetic plexus irrita- the oral and maxillofacial area and other specialities. tion). When pressure is exerted on the external ca- They are presented in Table 2. rotid artery, the pain is regionalized to the anatomic To palpate the elongated styloid process, the structures supplied by this artery. Clinically, the pa- gloved finger is inserted along the occlusal line pos- tient may complain of constant pain in the neck, pain teriorly to the region of the tonsillar fossa. If the on turning the head, regional carotidynia, or tender- process is palpable, it will be firm and pointed.3,14 ness of a cervical lymph node. Pressure on the inter- Pressure to the region exacerbates symptoms and nal carotid artery may produce symptoms as a result local sensitivity.3,13,14,23,24,30
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