Computerized tomography and Trotter's syndrome in the diagnosis of maxillofacial pain

Computerized tomography and Trotter’s syndrome in the diagnosis of maxillofacial pain Thomas R. Flynn, D.M.D.,* UNIVERSITY

OF CONNECTICUT

and Ellen Eisenberg, SCHOOL

OF DENTAL

D.M.D.,**

Farmington,

Conn.

MEDICINE

Trotter’s syndrome is a clinical triad of unilateral deafness, neuralgia affecting branches of the trigeminal nerve, and defective mobility of the soft palate, which is caused by malignant tumors involving the lateral pharyngeal recess (Rosenmtiller’s fossa). It is an ominous presentation, which can masquerade as dental or masticatory pain. Computerized tomography (CT) can be used not only to explain the anatomic basis of Trotter’s syndrome but also to determine the extent and distribution of the malignant tumor involved. The advantages of CT over conventional radiography are illustrated by a case of adenoid cystic carcinoma that presented as Trotter’s syndrome. Perineural invasion by tumor is shown on the gross level for the first time with CT, and important diagnostic considerations, which may aid in the early diagnosis of future cases, are discussed. (ORAL SURG. ORAL MED. ORAL PATHOL. 61:440-447, 1986)

T rotter’s

syndrome, a clinical triad of unilateral deafness, neuralgia affecting branches of the trigeminal nerve, and defective mobility of the soft palate, is caused by malignant tumors involving the lateral recess of the nasopharynx. Failure to recognize the presenting signs and symptoms of this syndrome commonly results in late diagnosis of primary nasopharyngeal disease. The case that we are presenting serves to illustrate that any malignant lesion that occupies the anatomic intersection just deep to the lateral pharyngeal recess (fossa of Rosenmiiller), regardless of its site of origin, can cause Trotter’s syndrome. It further illustrates that a high incidence of suspicion of nasopharyngeal malignancy is warranted when a patient complains of otherwise unexplained pain in the region of the jaws. It also demonstrates the value of CT in determining the extent of maxillofacial tumors. CASE

REPORT

A 29-year-old black woman was seen in the Oral Surgery Clinic at Cook County Hospital with a chief complaint of dull, constant, boring pain in the right maxilla

*Assistant Surgery. **Associate 440

Professor,

Department

of Oral

Professor,

Department

of Oral

and

Maxillofacial

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overlying the antrum, which radiated into the right neck and ear. She had been aware of the pain for approximately 20 months and related the onset to a severe cold and sore throat which did not resolve. Other complaints, including ear pain, nasal congestion, and dysphagia, were constant but varied in severity. Soon after the onset of her symptoms, the patient consulted several dentists and otolaryngologists and made several emergency room visits. She had been hospitalized for a rheumatologic work-up because of suspected autoimmune thyroiditis. She had also consulted a chiropractor and had been referred to a psychiatrist for treatment of “psychosomatic pain.” Management of the condition to date included three dental extractions, several courses of antibiotics and decongestants for “ear infections,” chiropractic cervical manipulations, and prescribed analgesics. Wound healing after all three dental extractions was complicated by intermittent bleeding, pain, and lack of soft tissue healing for approximately 4 months each time. Past medical history was significant for sickle cell trait, childbirth (at age 26) with a normal vaginal delivery, and tubal ligation (at age 28). In addition, there was a six pack-year history of cigarette smoking. On physical examination, the patient was unable to perform right lateral gaze (Fig. 1). The right tympanic membrane appeared erythematous and distended. There was mild nasal congestion, without exudate, on the right side. Oral examination disclosed the absence of the right maxillary first premolar and all molars. The extraction sites were fleshy in consistency, without surrounding bony plates. No oroantral fistula could be found. There was

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1. Inability to abduct right eye. The patient was askedto gaze toward her right.

Fig.

asymmetryof the palate,with submucosal fullnessobliterating the anglebetweenthe palatine and alveolar processes.The overlying mucosawassmoothbut hypervascular, and several dilated superficial veins (Fig. 2). A round ulcer, 2 mm in diameter,with a slightly raisedborder was noted on the right soft palate. There was no cervical lymphadenopathy, and the remainder of the physical examinationfindingswere within normal limits. Periapicalradiographsrevealedthe absenceof alveolar bone posterior to the site of the maxillary right first premolar. The floor of the right maxillary sinuswas not visible in thesefilms (Fig. 3). Incisional biopsieswere performed.Replacingalveolar bone in the maxillary right molar region was a friable, purple, nodular soft tissue massthat extended into the maxillary sinus. Specimensfrom the alveolus and soft palatewere histologicallyidentical and werediagnosedas “adenoidcystic carcinoma,solid type” (Fig. 4). The patient was then referred to the Otolaryngology Department for definitive work-up and treatment, including computerizedtomographic(CT) studies.It wasdeterminedfrom the clinical examinationthat the right cavernoussinushad beeninvadedby the tumor; this wasevident becauseof paralysisof the abducensnerve aswell as the chronic pain in the distribution of the maxillary divisionof the trigeminalnerve. Confirmationwasprovidedby CT, as discussed below.Thus, in February 1982,combinedintracranial and extracranial surgicalexcisionof the masswas performed.This includedright maxillectomy plusremoval of the nasalseptum,the ethmoidcribriform plate, and the right ethmoidandsphenoidsinuses.Immediatereconstruction includedrepair of the dura, a skingraft for resurfacing the wound bed, and immediateconstruction of a palatal obturator appliance. Two years after the initial surgical procedure, the patient underwenta left maxillectomy becauseof residual tumor. She receivedradiation therapy, maxillofacial prosthetic reconstruction,and a feedinggastrostomy,and was seenin the Pain Clinic for managementof chronic cranial pain. In March 1985the patient died, at the ageof 32, of residualintracranial tumor.

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Fig. 2. Initial oral examination.Note the appearanceof a massin the right palate and the dilated overlying veins. There is a small ulcer in the soft palate as well.

3. Periapicalradiographof right maxillary premolar-molar region. Note the absenceof alveolar boneand lack of definition of the maxillary sinusfloor in the molar region. Fig.

DISCUSSION

In 1911 Wilfred Trotter’ described a series of eight patients who had complained of unilateral deafness, neuralgia in the regions of distribution of the trigeminal nerve, and defective mobility of the soft palate. He correlated the simultaneous occurrence of these signs and symptoms with the growth of malignant tumors in the nasopharyngeal wall. Trotter explained the mechanism of this symptom triad on an anatomic basis, noting that the lateral pharyngeal recess (the area of the nasopharynx surrounding the opening of the eustachian tube) lies over the intersection of several important anatomic structures (Fig. 5). Obstruction of the eustachian tube can cause deafness, a sense of fullness in the ear, and improper drainage of the middle ear cavity, which may result in infection. Both the tensor and levator veli palatini

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Fig. 4. A, Low-powerview of nestsof adenoidcystic carcinomaadjacentto bone.Note cribriform and solidpatterns.(Hematoxylin and eosinstain. Original magnification,X 100.)B, Peripheralnervewith tumor in perineuralspaces.(Hematoxylin and eosinstain. Original magnification,X450.)

muscles originate in part from the eustachian tube. Infiltration of these muscles by a malignant tumor may be manifested as a unilateral defect in palatal movement. Furthermore, a tumor mass growing in the lateral pharyngeal recess can physically distend the soft palate, causing an intraoral swelling. Finally, since the mandibular branch of the trigeminal nerve courses deep to the eustachian tube and palatal muscles after exiting from the base of the skull at the foramen ovale, sensory innervation of the mandible, lips, and tongue, as well as motor innervation of the muscles of mastication, may be compromised when this nerve is infiltrated by a neoplasm. Thus, recognition of Trotter’s syndrome should be followed by thorough examination of the lateral pharyngeal recess (Fig. 6).

Trotter’s syndrome, or the presence of dental or facial pain with no obvious clinical cause, must carry with it a high index of suspicion of malignant change involving the cranial nerves. For this reason, dental extractions should be deferred, contingent upon diagnostic confirmation of unequivocal evidence of inflammatory odontogenic disease. Persistent pain and poor or delayed healing of an extraction site may also be ominous signs indicating the presence of a malignant lesion involving the alveolar process. Among the most common malignant conditions to be ruled out in this region are squamous cell carcinoma and adenoid cystic carcinoma, either of which may arise in the palate, paranasal sinuses, or nasopharynx. Clinical examination of the deep structures of the

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Fig. 5. Anatomy of lateral pharyngeal recessin horizontal section through maxillary sinus. The nasopharyngealrecess(fossa of Rosenmiiller)surroundsthe opening of the eustachiantube into the nasopharynx.Seetext for an explanationof how tumorsinfiltrating this areacan causeTrotter’s syndrome. (Modified from DoubledayLC, Jing BS, and Wallace S: Computedtomographyof the infratemporal fossa. Radiology 138:619-624,1981.)

head and neck for purposes of identifying and delineating tumors has long been a difficult problem. The inaccessibility of much of the mucosa of the nasal cavity, paranasal sinuses, and nasopharynx to direct visual examination hinders early detection of neoplastic processes. Often, tumor extension into deep spaces of the neck and pharynx, paranasal sinuses, orbit, and cranial cavity can only be inferred from radiographic changes detected in adjacent bone. On the other hand, bone may not be present in anatomic juxtaposition to some tumors. Moreover, osseous invasion may occur only as a late development. Thus, conventional radiographs are of significantly limited value in determining the presence and extent of soft tissue masses (Fig. 7). Computerized tomography is especially advantageous in the detection of tumors of the maxillofacial regions because it can be used to analyze small increments of attenuation of radiation by soft tissue. Muscle and mucosa register in CT, whereas fat and fascia do not. This provides the contrast necessary to outline denser structures. Conventional tomographic techniques are able to focus on a selected anatomic plane by blurring the radiographic representation of structures deep and superficial to the tomographic cut. In contrast, through mathematical subtraction of images generated by tissues lying outside the selected anatomic plane, computerized tomography provides a true anatomic “slice” of the region being examined.* Tomographic slice thickness in CT can be reduced to 1.5 mm; further, CT examination is exquisitely sensitive to density changes. X-ray attenuation of 0.5% can be detected by CT, as compared to 5.0% attenuation in conventional radiology.3 When applied to the maxillofacial regions, CT can

Fig. 6. Axial CT at level of hard and soft palate. Note the presence of a tumor massoccupyingthe right maxillary sinus(arrow), destroyingthe pterygoid platesand infiltrating submucosallythe musclesof the soft palate. The eustachianorifice is alsoobstructedby the tumor.

be used to delineate the deep spaces of the neck, pharynx, tongue, and floor of the mouth; the mucosae of the sinuses, nasopharynx, and lateral pharyngeal recesses; the foramina of the base of the skull; and the intracranial viscera.4-‘o In addition, CT can be used to determine the submucosal spread and depth of extension of a lesion.” It can also differentiate malignant or aggressive inflammatory processes

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7. A, Conventionalradiograph(Water’s view) of patient demonstratingmucosalthickening in right maxillary sinus(arrow) with someerosionof medial sinuswall. B, Coronal tomogramof samepatient in region of maxillary secondpremolar. The mucosalthickening (small arrow) and erosionof the medial maxillary sinuswall (large arrow) are demonstratedmoreclearly. C, CoronalCT through sameregion.The presenceof a soft tissuemassthat involvesthe maxillary sinusfloor (openarrow), medialsinuswall, inferior turbinate (white arrowhead), nasal septum, and the nasal floor bilaterally (small black arrows) is demonstratedclearly by CT. Normal sinusmucosais not visible in CT. D, More posterior CT section through molar region of maxilla demonstratesmore extensiveinvolvementby tumor, which hasdestroyed maxillary sinusfloor and alveolar process(openarrow) and involved more extensively inferior turbinate (white arrow), nasalseptum,and nasalfloor bilaterally (small black arrows). Fig.

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Fig. 8. A, CoronalCT through posterioraspectof maxillary sinusat level of pyramidal process of palatine bone(A), which encloses greater palatinenerve. Note involvementof the lateral (arrow), inferior, andmedial walls of the maxillary sinusby tumor in this region. Streaked artifacts causedby dental restorationsare apparent.B, CT section5 mm posteriorto that shownin A. Note that the posteriorwall of the maxillary sinusis completely involved by tumor (B and C’) and especiallythat the foramen rotundum has been obliteratedby the tumor. Note the normalforamenrotundumon the uninvolvedside(arrow). Note alsothe sphenoidsinus(A) and the optic foramen (openarrow).

from those that are benign according to whether they perforate the pharyngobasilar fascia that surrounds the pharynx. I* Computed tomography has been found to be particularly useful in, among other things, the diagnosis of craniofacial injuries.‘3l’4 It has also been used to monitor patients with head and neck tumors following treatment.* That CT is extremely effective in the detection of paranasal sinus tumors has been well demonstrated. In fact, CT is preferable to conventional radiographic techniques for disclosing soft tissue tumor extension posteriorly and superiorly from the sinuses.‘s Conventional radiography frequently fails to reveal such extension.16 CT has been shown to be the preferred method with which to make definitive evaluations of the infratemporal fossa.” In the case of malignant tumors involving the nasopharyngeal recess, CT has elucidated the foramina through which aggressive tumors can enter the cranial cavity. Nasopharyngeal tumors most commonly invade the cranial cavity through the foramina lacerum and ovale.” The tumor presented here

entered the middle cranial fossa through the foramen rotundum (Fig. 8, B). In the case presented here, perineural invasion by adenoid cystic carcinoma’8 was demonstrated on a gross anatomic level for the first time through the use of CT (Fig. 8). The patient’s inability to perform a right lateral gaze, chronic pain in the distribution of the maxillary division of the trigeminal nerve, and perforation of the lateral wall of the sphenoid sinus just opposite the medial wall of the cavernous sinus (Fig. 9, A) suggested involvement of the cavernous sinus. The lateral part of the cavernous sinus is the most peripheral point at which the second division of the fifth cranial nerve and the sixth cranial nerve approximate one another (Fig. 9, B). That the right cavernous sinus was invaded by the tumor was concluded clinically and confirmed by CT findings, respectively. The long-term prognosis in cases of adenoid cystic carcinoma is poor, particularly for those patients in whom the histologic pattern is characterized as “solid.“19 Survival rates decline significantly with

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Fig. 9. A, Coronal CT 10 mm posteriorto that shownin Fig. 8, B. Note the partial destructionof the pterygoid plates(A), body of the sphenoidbone(E), and invasionof the sphenoidsinus(C). Note alsothe perforation of the right lateral wall of the sphenoidsinus(arrow). B, Diagram of normal relations of sphenoidsinusand cavernoussinus.Note that the abducensnerve and the maxillary nerve lie in close proximity in the lateral portion of the cavernoussinus. (Modified from HollinsheadWH: Anatomy for surgeons,ed. 2, New York, 1968, Harper & Row Publishers,vol. 1, p. 62.)

each increment of 5 years, but there is a possibility of long-term survival in those patients with clear surgical margins.‘*~ 2oEarly detection and prompt surgical excision are, therefore, essential to reducing mortality from this disease. CONCLUSIONS

1. Trotter’s syndrome should be suspected in cases of unexplained dental or facial pain, especially when all or part of its triad is present. That triad consists of dental or facial pain in the mandibular or maxillary divisions of the trigeminal nerve; a sensation of ear pressure, distension of the tympanic membrane, or repeated middle ear infection; and defective mobility of, or the presence of a mass in, the soft palate. 2. In the absence of another identifiable cause of dental pain, extractions should be deferred until involvement of the trigeminal nerve by a neoplastic or inflammatory process has been ruled out. 3. Delayed or abnormal heaiing of dental extraction sites is an ominous sign and should be investigated thoroughly to rule out infections or neoplastic processes. 4. CT is the examination method of choice for diagnostic and treatment-planning assessment of the

maxillofacial regions when there is suspicion of a space-occupying lesion involving the base of the skull, the nasopharynx, the paranasal sinuses, or the deep facial planes of the upper neck. The authors thank Drs. Josef Krespi, John Sisto, C. William Hoekstra, and Warren Smith for their contributionsto the diagnosisand treatmentof the patient presented in this article. Dr. Daniel J. Traub madethe drawings shownin Figs. 5 and 9, B. REFERENCES

1. Trotter W: On certain clinically obscure malignant tumours of the naso-pharyngeal wall. Br Med J t: 1057-1059, 1911. 2. Ter-Pogossian MM: Computerized cranial tomography: equipment and physics. Semin Roentgen01 IZ: 13-25, 1977. 3. Noyek AM, et al: Clinically-directed CT in occult disease of the skull base involving foramen ovale. Laryngoscope 92: 1021-1027, 1982. 4. Larsson SG, Mancuso A, Hanafee W: Computed tomography of the tongue and floor of the mouth. Head Neck Radio1 143: 493-500, 1982. 5. Hesselink JR, et al: Computed tomography of the paranasal sinuses and face. Part I. Normal anatomy. J Comput Assist Tomogr 2: 559-567, 1978. 6. Hesselink JR, et al: Computed tomography of the paranasal sinuses and face. Part II. Pathological anatomy. J Comput Assist Tomogr 2: 568-576, 1978. 7. Mancuso AA, et al: Computed tomography of the nasopharynx: normal and variants of normal. Radiology 137: 113-121, 1980. 8. Som PM: The role of CT in the diagnosis of carcinoma of the

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paranasal sinuses and nasopharynx. J Otolaryngol 11: 340348, 1982. Noyek AM, et al: CT in occult disease of the skull base and basilar foramina. J Otolaryngol 11: 419-427, 1982. Keller MA, Holgate RC, McClarty BM: CT and intracranial manifestations of otorhinologic disease. J Otolaryngol 11: 407-410, 1982. Mancuso AA, Hanafee WN: Elusive head and neck carcinomas beneath intact mucosa. Laryngoscope 93: 133-139, 1983. Hoover LA, Hanafee WN: Differential diagnosis of nasopharyngeal tumors by computed tomography scanning. Arch Otolaryngol 109: 43-47, 1983. Fujii N, Yamashiro M: Computed tomography for the diagnosis of facial fractures. J Oral Surg 39: 735-741, 1981. Claussen C, Singer R: Progress in the diagnosis of craniofacial injuries and tumours by computer tomography. J Maxillofac Surg 7: 210-217, 1979. Parsons C, Hodson N: Computed tomography of paranasal sinus tumors. Radiology 132: 641-645, 1979. Proops DW, Phelps PD: Computed tomography in E.N.T. practice: an evaluation of two years’ experience in a British sub-regional centre. J Laryngol Otol 96: 417-437, 1982.

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17. Doubleday LC, Jing BS, Wallace S: Computed tomography of the infratemporal fossa. Radiology 138: 619-624, 1981. 18. Batsakis JG, Regezi JA: The pathology of head and neck tumors: salivary glands. Part 4. Head Neck Surg 1: 340-349, 1979. 19. Perzin KH, Gullane P, Clairmont AC: Adenoid cystic carcinomas arising in salivary glands: a correlation of histologic features and clinical course. Cancer 42: 265-282, 1978. 20. Miller RH, Calcaterra TC: Adenoid cystic carcinoma of the nose, paranasal sinuses, and palate. Arch Otolaryngol 106: 424-426, 1980. Reprint requests to: Dr. Thomas R. Flynn, L7073 Department of Oral and Maxillofacial Surgery University of Connecticut School of Dental Medicine Farmington, CT 06032