Elongated and mineralized stylohyoid ligament complex: A proposed classification and report of a case of Eagle's syndrome

Elongated and mineralized stylohyoid ligament complex: A proposed classification and report of a case of Eagle's syndrome

Elongated and mineralized stylohyoid ligament complex: A proposed classification and report of a case of Eagle’s syndrome Robert P. Langlais, D.D.S.. ...

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Elongated and mineralized stylohyoid ligament complex: A proposed classification and report of a case of Eagle’s syndrome Robert P. Langlais, D.D.S.. M.S.,* Dale A. Miles, D.D.S.,** and Margot L. Van Dis, D.D.S.,*** San Antonio, Texas, Halifax, Nova Scotia, Canada, and Columbus, Ohio

A classification of the radiographic appearance of elongated and mineralized stylohyoid ligament complexes based on three types of complexes-Type I, elongated; Type II, pseudoarticulated; and Type Ill, segmented-is proposed. These types are further described by a pattern of calcification: calcified outline, partially calcified, nodular, and completely calcified. The classification is illustrated in a case of Eagle’s syndrome in a 55year-old Mexican-American man with symptoms of chronic otalgia and cephalgia. The surgical management and follow-up of this patient are discussed. (ORAL SURC. ORAL MED. ORAL PATHOL. 61527432, 1986)

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ineralization or calcification of the stylohyoid complex is relatively common. Correll et al.’ found an incidence of 18.2% on panoramic radiographs in a review of 1771 cases. Fortunately, most authors agree that few of these atIlicted processes become symptomatic,1-4 with the reported prevalence from 1% to 5%’ When symptoms are present, the condition is known variously as Eagle’s syndrome, elongated styloid process syndrome, styloid processcarotid artery syndrome, stylohyoid syndrome, or styloid process neuralgia.5 Eagle6 first presented two cases of symptomatic elongated, calcified stylohyoid processes in 1937. In this report, he described complaints of a “nagging or aching sensation in the throat similar to chronic pharyngitis” and pains radiating to the ear or mastoid region. Other symptoms include difficulty in swallowing, a sensation of a foreign object lodged in the throat, or both. Subsequent authors have reported cases with mainly a

*Professor and Head of the Graduate Division, Department of Dental Diagnostic Science, Dental School, University of Texas Health Science Center at San Antonio. **Assistant Professor, Division of Oral Diagnosis and Radiology, Dalhousie University, Halifax, Nova Scotia, Canada. ***Assistant Professor, Department of Dental Diagnostic Sciences, The Ohio State University, Columbus, Ohio.

swallowing complaint.7ey In his original report, Eagle alluded to symptoms of pain referral and included in his descriptions a carotid artery syndrome. Both syndrome types (the classic and the carotid artery) have been well described recently by Glogoff et al.’ and by Gossman and Tarsitano.3 Of interest to us is the variety of radiographic descriptions of the stylohyoid complex, including “ram’s horn,“‘O elongated,3 segmented,’ jointlike,‘, I’ pseudoarticulated,‘2*‘3 jointed,14 crooked,3 and nodular.’ FrommeP demonstrated the extreme variability of the stylohyoid chain during his dissection of 241 processes. Variations were noted in length of the process, thickness of segments, angle and direction of deviation, degree of ossification, and relation of the bony sheath to the root of the styloid process. To simplify the description of the elongated structures, a radiographic classification of the mineralized stylohyoid ligament complex is proposed. In addition, a case is presented in which the elongated, mineralized stylohyoid ligament complex was symptomatic. CLASSIFICATION

In a study of 4200 cases, Gossman and Tarsitano3 found a radiographic incidence of mineralized complexes of 1.4%. They proposed a classification in which the variations were referred to as slightly 527

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Fig. 1. From left to right, Type I (elongated), Type II (pseudoarticulated),

and Type III (segmented).

Table I Classification Type Type Type

I-elong&.d II-pseudoarticulated I I l-segmented

Pattern

of calcification

Calcified outline Partially calcified Nodular Completely calcified

a specific description of the nature of the mineralizing process. Type I-Elongated

Fig. 2. Pattern of calcification. Upper left, calcified outline; upper right, partially calcified; lower left, nodular; lower right, completely calcified.

elongated (16 cases), crooked (4 cases), segmented (16 cases), and very elongated (22 cases). No mention was made of jointlike or pseudoarticulated variants as reported elsewhere.‘* ‘l-l3 Our classification includes three types of radiographic appearance and four patterns of calcification or mineralization (Figs. 1 and 2). As proposed by Correll and associates,’ we agree that the term mineralization of the stylohyoid complex may be more accurate, since it is impossible to determine the exact nature of the radiopaque material in standard radiographs. However, we prefer the term calcified, since it is the more traditional term used in the radiographic literature, and in our classification it is used in its most general or broad sense rather than as

The radiographic appearance of this type of mineralized complex is characterized by an uninterrupted integrity of the styloid image (Fig. 3). The “normal” reported length of the styloid process ranges from 25’-3~s~7to 32 mm.ls We have accepted the normal length of 25 mm for most radiographic projections. However, if panoramic films are studied, measurements to 28 mm may be considered within the normal range because of the inherent magnification in most panoramic projections. Type II - Pseudoarticulated

Fig. 4 illustrates the pseudoarticulated variant. The styloid process is apparently joined to the mineralized stylomandibular or stylohyoid ligament by a single pseudoarticulation, which is usually located superior to a level tangential to the inferior border of the mandible. This gives an overall picture of an apparently articulated elongated styloid process. It is our experience that this type of mineralized complex appears with some regularity, although much less frequently than Type I. This type may be seen in the radiographs of our patient (Figs. 7 to 10).

Volume Number

Elongated

61 5

Fig. 3. Patient shows bilateral partially calcified.

Type

5. Bilateral

stylohyoid

Type 1 processes with right side having calcified

Fig.

Fig.

and mineralized

Type III, completely

4. Bilateral

ligament complex

outline

529

and left side

Type II, calcified outline.

calcified.

Fig. 6. Right side, Type III, calcified outline. Type I, nodular.

Left side,

Ill-Segmented

Type III consists of either short or long noncontinuous portions of the styloid process or interrupted segments of mineralized ligament. In either instance, two or more segments are seen, with interruptions either above or below the level of the inferior border of the mandible, or both. The overall appearance is one of a segmented mineralized stylohyoid complex. Segmented versions of the stylohyoid complex had an incidence of about 27% in the study by Gossman and

Tarsitano.3 FrommerlS described fibrous or cartilaginous unions existing between mineralized segments (Fig. 5) in his dissections. PATTERN

OF CALCIFICATION

In addition to classification by type, it is necessary to ascribe a pattern of calcification to each styloid complex to adequately describe the appearance radiographically. A calcified outline (Figs. 2 to 4)

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7. Bilateral Type II, calcified outline. Note large nodularareasnear pseudoarticulations.

Fig.

describes a thin radiopaque border with a central radiolucency that constitutes the majority of the process. This pattern is reminiscent of the radiographic appearance of a long bone. Partially calcified (Figs. 2 and 3) describes a process that has a thicker radiopaque outline and almost complete opacification, but small, sometimes discontinuous radiolucent cores. A nodular complex (Figs. 2 and 6) has a knobby or scalloped outline. It may be partially or completely calcified with varying degrees of central radiolucency. A completely calcified process (Figs. 2 and 5) is totally radiopaque with no evidence of a radiolucent interior. (Table I summarizes the classification.) CASEREPORT Medical history

Fig. 8. Lateral skull film. Length of left complex on panoramicfilm is 96 mm from apparent point of origin from temporalboneto tip of terminal segment.Length on lateral film is 94 mm from samelandmarks.

A 55-year-old,edentulousMexican-American man was a patient in the Dental DiagnosticScienceReferral Clinic in the Dental School at the University of Texas Health ScienceCenter at San Antonio. He related a history of chronic otalgia and nonspecificear infectionsfor a period of 5 years. In addition, he reported that he had an increasingnumber of headachesduring this period. The patient had a tonsillectomywhenhe wasa child, but there wasno other pertinent medicalhistory.

calcified outline and a radiolucent center. The right process revealed a pseudoarticulatedportion midway betweenthe mastoidarea and the hyoid bone. The left processwas comparable except for an ovoid opacity approximately 0.5 cm in diameter, which appearedto be situatedbetweenthe distal and proximal segmentsof the elongatedprocess(Fig. 7). Lateral skull, lateral jaw, and posteroanteriorradiographsrevealedsmall calcified segmentsof the complexat the level of the lesserhornsof the hyoid bone.There wasmedialcurvature to both processes (Figs. 8 to 10).

Clinical

Treatment

findings

Several scarswere apparenton the forehead,midface, and chin region on the patient’s left side and were attributed to various fights. The patient had no palpable lymphadenopathy,muscletrismus, or other abnormality extraorally. He had no restrictionof movementof the head or jaws or any painful symptomsduring thesemovements. Intraorally he had sharp pain bilaterally on palpation of the tonsillar fossae;however,there was no pain radiation. The buccal mucosa,tongue, and other oral tissueswere normal. Radiographic

findings

The initial panoramic radiograph revealed bilateral Type II pseudoarticulatedstylohyoid processeswith a

The definitive diagnosisof Eagle’ssyndromeis made when cessationof symptomsoccurs following surgical intervention. The patient wasreferred to the Department of Surgery, Division of Otolaryngology at the Medical Center Hospital, San Antonio, Texas, for further evaluation and surgicalmanagement,In the past, treatmentsfor Eagle’s syndrome included a simple fracturing of the processand surgicalshorteningby intraoral approach.In this case, treatment consistedof the removal of distal segmentsof 2.5 cm (right side) and 3.0 cm (left side)of both stylohyoid complexes.The approachwas extraoral becauseof the large size of the mineralizedcomplexes.At this writing, 9 monthsafter surgery, the patient is symptom free.

Volume 6 1 Number 5

Fig. 9. Lateral jaw panoramic film is 57 tion to tip of terminal is 54 mm from same

Elongated and mineralized stylohyoid ligament complex

film. Length of right complex on mm from proximal pseudoarticulasegment. Length on lateral jaw film landmarks.

Fig.

53 I

skul1film. Note medial curva-

10. Posteroanterior

ture of complexes.

DISCUSSION

SUMMARY

The carotid artery syndrome reportedly produces symptoms by pressure of the deviated or enlarged styloid process on either the internal or external carotid arteries. A regional carotodynia may be produced or pain may be referred to any region supplied by these vessels. Hence, symptoms may include parietal, ophthalmic, or otalgic pain, vertigo, dysphagia, and syncope.4s’6 In our case, the patient complained mainly of headaches and ear pain. Although he had had a tonsillectomy, symptoms of dysphagia or a feeling of “something in the throat” (ascribed to the classic syndrome) were notably absent. The pain following tonsillectomy in the classic syndrome is reportedly generated by stretching or compressing the nerve endings of cranial nerves V, VIII, IX, or X in the tonsillar fossa either during healing or shortly thereafter.’ A similar type of pain may be associated with contact of the carotid artery and associated nerves against the process or mineralized ligament on turning the head. Thus, in patients with dysphagia and a history of tonsillectomy, referred pain must be present to distinguish between the classic and the carotid subtypes of Eagle’s syndrome. No determination of syndrome subtype is possible on the basis of radiographic appearance alone. However, the posteroanterior radiographs may demonstrate a medial deviation of the process. In the presence of symptoms, this finding may be suggestive of the carotid artery subtype of Eagle’s syndrome.

Elongation and mineralization of the stylohyoid complex is a relatively common occurrence that may produce a variety of clinical symptoms and radiographic appearances. The phenomenon is readily observed on panoramic radiographs. A classification is proposed in an attempt to standardize and simplify the description of the condition. A case of Eagle’s syndrome is reported to illustrate the classification. REFERENCES 1. Correll RW, Jensen JL, Taylor JB, Rhyne RR: Mineralization of the stylohyoid-stylomandibular ligament complex. ORAL SURGORAL MED ORAL PATHOL 48: 286-29 1, 1979. Glogoff MR, Baum SM, Cheifetz I: Diagnosis and treatment of Eagle’s syndrome. J Oral Surg 39: 94 l-944, 198 1. Gossman JR Jr, Tarsitano JJ: The stylo-stylohyoid syndrome. J Oral Surg 35: 555-560, 1977. Baddour HM, McAnear JT, Tilson HB: Eagle’s syndrome. ORAL

SURG

ORAL

MED

ORAL

PATHOL

46:

486-494.

1978.

Langland OE, Langlais RP, Morris CR: Principles and practice of panoramic radiology, Philadelphia, 1982, W. B. Saunders Co., p. 362. 6. Eagle WW: Elongated styloid process: Report of two cases. Arch Otolaryngol 25: 584-587, 1937. 7. Haidar Z, Kalamchi S: Painful dysphagia due to fracture of the styloid process. ORAL SURGORAL MED ORAL PATHOL 49: 5-6, 1980. 8. Reichart PA, Sooss W: Fracture of the styloid process of the temporal bone: An unusual complication of dental treatment. ORAL

SURG

ORAL

MED

ORAL

PATHOL

42:

150-154,

1976.

9. Ettinger RL, Hanson JG: The styloid or “Eagle” syndrome: An unexpected consequence. ORAL SURG ORAL MED ORAL PATHOL

40: 336-340,

1975.

10. Eagle WW: Elongated styloid process: Further observations and a new syndrome. Arch Otolaryngol 47: 630-640, 1958. 1I. Stafne EC, Hollinshead WH: Roentgenographic observations on the stylohyoid chain. ORAL SURG ORAL MED ORAL PATHOL 15:

1195-1200,

1962.

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12. Lavine MH, Stoopack JC, Jerrold TL: Calcification of the stylohyoid ligament. ORAL SURG ORAL MED ORAL PATHOL 25: 55-58, 1968. 13. Evans JT, Clairmont AA: The nonsurgical treatment of Eagle’s syndrome. Ear Nose Throat J 55: 44-45, 1976. 14. Lipshutz B: The clinical importance of the ossification of the stylohyoid ligament. JAMA 79: 1982-1984, 1922. 15. Frommer J: Anatomic variations in the stylohyoid chain and their possible clinical significance. ORAL SURG ORAL MED ORAL PATHOL 38: 659-666, 1974. 16. Correll RW, Wescott WB: Eagle’s syndrome diagnosed after

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history of headache, dysphagia, otalgia, and limited movement. J Am Dent Assoc 104: 491-492, 1982.

neck

Reprint requests to: Dr. Robert P. Langlais Department of Dental Diagnostic Science University of Texas Health Science Center at San Antonio 7703 Floyd Curl Dr. San Antonio, TX 78284