INFRATEMPORAL SPACE PA TH S
MIMICKING TMJ DISORDERS
DAVID A. KEITH , B .D .S ., F.D.S. R.O.S., D .M .D .; M A R K L. G L Y M A N , D.D .S., M .D .
A B S T R A C T
S y m p to m s o f te m p o r o m a n d ib u la r j o i n t d is o r d e r s — fa c ia l p a in o r ja w m o v e m e n t lim ita tio n —ca n b e c a u se d b y o th e r p r o b le m s in th e in fr a te m p o r a l sp a c e .
© ith an estim ated 12 percent to 59 percent of the population aw are of some tem porom andibular joint dysfunction, a careful diagnosis should be made for patients with head or facial pain or jaw m ovem ent limitation.' We describe here four cases of pathology of the infratem poral space originally diagnosed as TMJ disorders. C A S E
1
A 51-year-old male m achine tool operator com plained of right facial pain and a progressive shift of his jaw to the left. The patient had psoriasis and had smoked two packs of cigarettes every day for 35 years. His dentist perform ed an occlusal equilibration with no im provem ent of symptoms. An otolaryngologist noted no abnormalities. An oral and maxillofacial surgeon initially thought that the patient had a chronic dislocation of the right condyle and had two unsuccessful condylar relocations. Tomograms showed the m andibular condyle in an abnormally anterior location. Reduction of the right condyle with the patient under general anesthesia was not successful. Maxillary and m andibular arch bars were applied and postoperatively elastic m axillom andibular fixation was placed with the hope of reducing the suspected dislocation slowly. Fixation, however, increased the pain and the elastics were removed. A neurologist saw the patient and concluded that he had a “com pression of the right facial nerve of undeterm ined etiology.” A CT scan was “norm al.” The patient was adm itted to another hospital for evaluation of right facial pain, swelling and progressive shift of his mandible to the left, inability to open his m outh fully and num bness of his lower right lip. A repeat CT scan dem onstrated right pterygoid muscle enlargem ent. An ENT consultant found no evidence of any lesions. A bone scan of the right infratem poral region indicated non-specific increased uptake on the right. Blood and urine tests were within norm al limits except for a slight increase in alkaline phosphatase (46 international units per liter-normal range 13-39IU/L). As the main finding was enlargem ent of the right lateral pterygoid muscle, a biopsy of the muscle was made through a right preauricular incision. The right side of the m andible could not be m anipulated into position, and the condyle was found anteriorly displaced. The lateral JADA, Vol. 122, November 1991
59
pterygoid muscle was exposed at the attachm ent to the right condyle and the tissue, which looked and felt like granulation tissue, was sent to pathology for evaluation. The biopsy specimens were re ported as “poorly differentiated car cinoma.” Rare poorly formed gland like structures suggested a “poorly differentiated adenocarcinoma.” The patient tolerated two cycles of chem otherapy well with pain reduction and tum or size dim inution. He received 64 Gy twice daily. He died 18 months after the tum or diagnosis and 22 m onths after the symptom onset. C A S E
2
A 62-year-old salesman had tris mus and right facial pain. He had idiopathic hypertrophic subaortic stenosis and depression for which he took amitryptyline hydrochlor ide (Amitryptyline HCL, Merck Sharp & Dohme). He took 26 units NPH insulin daily for diabetes. Nine m onths earlier he had a tightening of his right jaw and trism us which slowly progressed to 4 millimeters. He also had interm ittent right facial pain, which became worse at the end of the day and slowly diminishing hearing in his right ear. He reported interm ittent right facial swelling. When we saw the patient, there was a visible tender swelling over the right zygomatic arch anterior to the right ear; he could open his m outh to 4 m m with no lateral excursions. Hearing in the right ear was dim inished but all other neurological examinations were normal. Radiographs showed a soft tissue mass over the sphenoid sinus and poor aeration of the right mastoid air cells. A CT scan of the base of skull, infratemporal fossa and nasophar ynx showed a soft tissue abnor mality in the infratemporal fossa 60
JADA, Vol. 122, November 1991
obliteration of the soft tissue planes. We surgically explored the right TMJ region and did a tissue biopsy. A preauricular incision showed dense and fibrous skin and super ficial tissues with no usual fascial planes. We carried the incision down to the zygomatic arch. Just superior to the root of the zygoma, a cavity yielded about 15 milliliters of a milky fluid with numerous yellow clumps. Aerobic and anaerobic cultures were taken and analyzed. The area was thoroughly explored and normal temporal bone was found at the base of the cavity. We prescribed 20 million units of IV penicillin a day. In the first four postoperative days, the facial swelling was reduced, the temporal pain disappeared and the hearing in the right ear improved. Actinobacillus was identified. The patient recalled that a m andibular right third molar was extracted several weeks before the symptoms began. C A S E
3
A 31-year-old male had bruxism for three years, right TMJ fatigue and progressive m andibular hypomobility. The patient was initially treated by his dentist who noticed a right preauricular mass. He was then seen by an otolaryngologist who performed a right parotid exploration. No neoplasm was discovered and a biopsy identified normal m asseter muscle. The right TMJ was not explored at that time and radiographs of the maxilla, mandible and TMJs were normal. A second dentist fabricated a hydrodynamic splint to relieve symptoms. In three months, range of motion increased from 18 mm to 27 mm, and the mass size decreased slightly. After other examinations showed no significant findings, we explored the area and tried to increase the maximal opening
using a m outh prop. We found a hard, preauricular mass and severe m andibular hypomobility with a range of motion of 24 mm. Radiographs revealed symmetric condyles bu t no subsequent bony destruction on the tomograms. With this procedure, the patient could open his m outh to 27 mm. Within two months, the range of motion decreased to 18 mm with no other change of symptoms. In another three months, range of motion decreased to 14 mm. A large high-grade muco-epidermoid tum or was found wedged among the base of the skull, the mastoid process, the posterior border of the mandible and the 7th nerve. Be cause of the patient’s longstanding periodontal disease, we extracted many teeth. After radiation, the tum or recurred several times in spite of a right radical neck dissection and chemotherapy. C A S E
4
A 12-year-old healthy female had facial pain and left TMJ pro blems. She also Dr. Glyman is a resident in oral and complained of m axillofacial surgery» some interm it M assachusetts General Hospital, tent swelling Harvard School of and recently a Dental M edicine and constant numb Harvard M edical ness of the lower School, Boston. left lip and chin. The only injury the patient recalled was a fall from a recreational vehicle. But about a m onth before evaluation, she experienced sudden sharp pain in the left TMJ when she moved her mandible forward, leaving a constant low level pain. She said it was uncomfortable to chew. The left condyle and preaur icular area as well as the left medial pterygoid muscle were tender. The left and right lateral pterygoid muscles had mild but
equal discomfort. She opened more than 35 mm with minimal deviation to the left. There was no external swelling. Initially, we considered a stressrelated TMJ dysfunction with an acute sprain of the left TMJ. The finding of decreased sensation in the third division of the trigeminal nerve raised the question of a craniopharyngeal or nasopharyn geal lesion. Initial treatm ent was directed at sym ptomatic relief with diazepam (Valium, Roche), soft diet, heat and rest. After a week, the patient had progressive pain in the left ear and preauricular area. A CT scan of the cranial base and nasopharynx revealed a tum or involving the pterygoid space and infra-temporal fossa extending to, but not invading the base of the skull. A biopsy specim en resulted in a fibrosarcoma. Subsequently, the patient received weekly Vincristine Sulfate (Quad) therapy and radiation daily. D IS C U S S IO N
These cases illustrate the fact that pathosis of the infratem poral space can, in the early stages, mimic TMJ dysfunction through pain and m andibular limitation.14 The subsequent sensory loss, m andibular deviation and swelling led to a m ore thorough evaluation and the discovery of the underlying causes. Although “common things occur commonly,” practitioners should know th at occasionally a serious disease may cause seemingly m undane symptoms. We should make a detailed history and full examination, for any patient who does not respond as anticipated to appropriate treatm ent. Loss of sensation in the orofacial region not associated with traum a or surgery is always a sinister sign dem anding an
aggressive workup. In case 1, initial facial pain sym ptoms and m andibular deviation seemed to indicate a TMJ problem, b u t with 5th nerve paresthesia, this diagnosis became untenable. Despite extensive workups (radiographs, CT scans) and consultations, the tum or diagnosis was not confirmed until after an open biopsy of the infiltrated lateral pterygoid muscle. The prim ary site was not discovered until a later ENT evaluation. The two risk factors in this case were tobacco use and occupation.56 In case 2, a chronic infection apparently was introduced into the tissues w hen an infected tooth was extracted and caused symptoms—interpreted for nine months as TMJ related. Actinomycosis is a specific gran ulomatosis mycotic disease occur ring mostly in the cervicofacial re gion.79The disease is characterized by indurated swellings, which break down to form multiple fistulae. The reported anatomic location of head and neck actinomycosis includes most anatom ic sites: the retromaxillary space, the TMJ and the mastoid and tem poral bone. No infection, however, has been reported in the infratem poral space. The most common parotid tum or is a painless mass in the preor infra-auricular area.1012A small portion of the parotid gland resides deep to the facial nerve and is wedged betw een the ramus of the m andible and the mastoid process. This hourglass- or “dumbbell”- shaped tum or is considered relatively rare and can produce confusing symptoms. Facial paralysis is rarely associated with deep lobe tumors; however, when present, it is considered suspicious as it may indicate advanced malignancy. Progressive hypomobility of the
mandible has never previously been reported with clinical evidence of deep lobe neoplasm. Because of their location, deep lobe parotid tum ors are surgically less accessible and often result in a higher degree of morbidity. S U M M A R Y
The differential diagnosis of pain in the face and dysfunction of the m asticatory system is complex. Although m ost diagnoses are readily made, common symptoms can occasionally belie more serious diseases. Each patient was originally treated for TMJ dysfunc tions and subsequently was diag nosed as suffering from pathosis of the infratem poral space. ■ Additional case histories a re provided by Dr. J.B. Carter and Dr. J. D. Stone, Wang Ambulatory Care Center 230, Boston 02114.
Ü!
1. Heggie AA, Reade PC. A m alignant lesion of the infratem poral fossa causing m andibular displacement. Case report. Aust Dent J Dr. Keith is 1984;29:145-9. associate professor 2. Cohen SG, Quinn PD. Facial trism us and of oral and m axillo myofascial pain associated facial surgery, with infections and Harvard School of m alignant disease. Oral Surg Dental Medicine; Oral Med Oral Pathol visiting oral and 1988;65:538-44. 3. DelBalso AM, Sweeney maxillofacial AT, Kapur S. An unusual surgeon, M assa cause of facial trism us in a chusetts General child: report of case. JADA Hospital; Chief, Oral 1986;112:207-9. Surgery, Harvard 4. Grace EG, North AF. Tem porom andibular joint Community Health dysfunction and orofacial Plan, W ang Am bula pain caused by parotid gland tory C are C enter malignancy: report of case. 230, Boston 0 2 1 1 4 . JADA 1988;116:348-50. 5. Thawley SE, Panje WR. Address requests for In: Kornblut AD. reprints to Dr. Keith. Comprehensive m anagem ent of head and neck tumors: clinical evaluation of tum ors of the oral cavity. Philadelphia:Saunders;1987:460. 6. Huygen PLM, Fischer AJEM, Vanden BP. Nasopharyngeal cancer: a clinical study with special refer ence to age and occupation. Clin Otolaryngol 1980;5:37-47. 7. N orm an JEdeB. Cervicofacial actinomycosis. Oral Surg Oral Med Oral Pathol 1970;29:735-45. 8. Rankow RM, Abraham DM. Actinomycosis m asquerading in the head and neck. Ann Otol Rhinol Laryngol 1978;87:230-7. 9. Richtsm eier WJ, Johns ME. Actinomycosis of the head and neck. CRC Crit Rev Clin Lab Sci 1979;11:175-202. 10. H anna DC, Gaisford JC, Richardson GS, et al. Tum ors of the deep lobe of the parotid gland. Am J Surg 1968;116:524-7. 11. Morfit HM. Retrom andibular parotid tumors: their surgical treatm ent and m ode of origin. AMA Arch Surg 1955;70:906-13. 12. Patey DH, Thackray AC. The pathologic anatom y and treatm ent of parotid tum ors with retropharyngeal extension (dumbbell tum or) w ith a report of four cases. Br J Surg 1957;44:352. i m
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vH
JADA, Vol. 122, November 1991
61
MIMICKING TMJ DISORDERS
DAVID A. KEITH , B .D .S ., F.D.S. R.O.S., D .M .D .; M A R K L. G L Y M A N , D.D .S., M .D .
A B S T R A C T
S y m p to m s o f te m p o r o m a n d ib u la r j o i n t d is o r d e r s — fa c ia l p a in o r ja w m o v e m e n t lim ita tio n —ca n b e c a u se d b y o th e r p r o b le m s in th e in fr a te m p o r a l sp a c e .
© ith an estim ated 12 percent to 59 percent of the population aw are of some tem porom andibular joint dysfunction, a careful diagnosis should be made for patients with head or facial pain or jaw m ovem ent limitation.' We describe here four cases of pathology of the infratem poral space originally diagnosed as TMJ disorders. C A S E
1
A 51-year-old male m achine tool operator com plained of right facial pain and a progressive shift of his jaw to the left. The patient had psoriasis and had smoked two packs of cigarettes every day for 35 years. His dentist perform ed an occlusal equilibration with no im provem ent of symptoms. An otolaryngologist noted no abnormalities. An oral and maxillofacial surgeon initially thought that the patient had a chronic dislocation of the right condyle and had two unsuccessful condylar relocations. Tomograms showed the m andibular condyle in an abnormally anterior location. Reduction of the right condyle with the patient under general anesthesia was not successful. Maxillary and m andibular arch bars were applied and postoperatively elastic m axillom andibular fixation was placed with the hope of reducing the suspected dislocation slowly. Fixation, however, increased the pain and the elastics were removed. A neurologist saw the patient and concluded that he had a “com pression of the right facial nerve of undeterm ined etiology.” A CT scan was “norm al.” The patient was adm itted to another hospital for evaluation of right facial pain, swelling and progressive shift of his mandible to the left, inability to open his m outh fully and num bness of his lower right lip. A repeat CT scan dem onstrated right pterygoid muscle enlargem ent. An ENT consultant found no evidence of any lesions. A bone scan of the right infratem poral region indicated non-specific increased uptake on the right. Blood and urine tests were within norm al limits except for a slight increase in alkaline phosphatase (46 international units per liter-normal range 13-39IU/L). As the main finding was enlargem ent of the right lateral pterygoid muscle, a biopsy of the muscle was made through a right preauricular incision. The right side of the m andible could not be m anipulated into position, and the condyle was found anteriorly displaced. The lateral JADA, Vol. 122, November 1991
59
pterygoid muscle was exposed at the attachm ent to the right condyle and the tissue, which looked and felt like granulation tissue, was sent to pathology for evaluation. The biopsy specimens were re ported as “poorly differentiated car cinoma.” Rare poorly formed gland like structures suggested a “poorly differentiated adenocarcinoma.” The patient tolerated two cycles of chem otherapy well with pain reduction and tum or size dim inution. He received 64 Gy twice daily. He died 18 months after the tum or diagnosis and 22 m onths after the symptom onset. C A S E
2
A 62-year-old salesman had tris mus and right facial pain. He had idiopathic hypertrophic subaortic stenosis and depression for which he took amitryptyline hydrochlor ide (Amitryptyline HCL, Merck Sharp & Dohme). He took 26 units NPH insulin daily for diabetes. Nine m onths earlier he had a tightening of his right jaw and trism us which slowly progressed to 4 millimeters. He also had interm ittent right facial pain, which became worse at the end of the day and slowly diminishing hearing in his right ear. He reported interm ittent right facial swelling. When we saw the patient, there was a visible tender swelling over the right zygomatic arch anterior to the right ear; he could open his m outh to 4 m m with no lateral excursions. Hearing in the right ear was dim inished but all other neurological examinations were normal. Radiographs showed a soft tissue mass over the sphenoid sinus and poor aeration of the right mastoid air cells. A CT scan of the base of skull, infratemporal fossa and nasophar ynx showed a soft tissue abnor mality in the infratemporal fossa 60
JADA, Vol. 122, November 1991
obliteration of the soft tissue planes. We surgically explored the right TMJ region and did a tissue biopsy. A preauricular incision showed dense and fibrous skin and super ficial tissues with no usual fascial planes. We carried the incision down to the zygomatic arch. Just superior to the root of the zygoma, a cavity yielded about 15 milliliters of a milky fluid with numerous yellow clumps. Aerobic and anaerobic cultures were taken and analyzed. The area was thoroughly explored and normal temporal bone was found at the base of the cavity. We prescribed 20 million units of IV penicillin a day. In the first four postoperative days, the facial swelling was reduced, the temporal pain disappeared and the hearing in the right ear improved. Actinobacillus was identified. The patient recalled that a m andibular right third molar was extracted several weeks before the symptoms began. C A S E
3
A 31-year-old male had bruxism for three years, right TMJ fatigue and progressive m andibular hypomobility. The patient was initially treated by his dentist who noticed a right preauricular mass. He was then seen by an otolaryngologist who performed a right parotid exploration. No neoplasm was discovered and a biopsy identified normal m asseter muscle. The right TMJ was not explored at that time and radiographs of the maxilla, mandible and TMJs were normal. A second dentist fabricated a hydrodynamic splint to relieve symptoms. In three months, range of motion increased from 18 mm to 27 mm, and the mass size decreased slightly. After other examinations showed no significant findings, we explored the area and tried to increase the maximal opening
using a m outh prop. We found a hard, preauricular mass and severe m andibular hypomobility with a range of motion of 24 mm. Radiographs revealed symmetric condyles bu t no subsequent bony destruction on the tomograms. With this procedure, the patient could open his m outh to 27 mm. Within two months, the range of motion decreased to 18 mm with no other change of symptoms. In another three months, range of motion decreased to 14 mm. A large high-grade muco-epidermoid tum or was found wedged among the base of the skull, the mastoid process, the posterior border of the mandible and the 7th nerve. Be cause of the patient’s longstanding periodontal disease, we extracted many teeth. After radiation, the tum or recurred several times in spite of a right radical neck dissection and chemotherapy. C A S E
4
A 12-year-old healthy female had facial pain and left TMJ pro blems. She also Dr. Glyman is a resident in oral and complained of m axillofacial surgery» some interm it M assachusetts General Hospital, tent swelling Harvard School of and recently a Dental M edicine and constant numb Harvard M edical ness of the lower School, Boston. left lip and chin. The only injury the patient recalled was a fall from a recreational vehicle. But about a m onth before evaluation, she experienced sudden sharp pain in the left TMJ when she moved her mandible forward, leaving a constant low level pain. She said it was uncomfortable to chew. The left condyle and preaur icular area as well as the left medial pterygoid muscle were tender. The left and right lateral pterygoid muscles had mild but
equal discomfort. She opened more than 35 mm with minimal deviation to the left. There was no external swelling. Initially, we considered a stressrelated TMJ dysfunction with an acute sprain of the left TMJ. The finding of decreased sensation in the third division of the trigeminal nerve raised the question of a craniopharyngeal or nasopharyn geal lesion. Initial treatm ent was directed at sym ptomatic relief with diazepam (Valium, Roche), soft diet, heat and rest. After a week, the patient had progressive pain in the left ear and preauricular area. A CT scan of the cranial base and nasopharynx revealed a tum or involving the pterygoid space and infra-temporal fossa extending to, but not invading the base of the skull. A biopsy specim en resulted in a fibrosarcoma. Subsequently, the patient received weekly Vincristine Sulfate (Quad) therapy and radiation daily. D IS C U S S IO N
These cases illustrate the fact that pathosis of the infratem poral space can, in the early stages, mimic TMJ dysfunction through pain and m andibular limitation.14 The subsequent sensory loss, m andibular deviation and swelling led to a m ore thorough evaluation and the discovery of the underlying causes. Although “common things occur commonly,” practitioners should know th at occasionally a serious disease may cause seemingly m undane symptoms. We should make a detailed history and full examination, for any patient who does not respond as anticipated to appropriate treatm ent. Loss of sensation in the orofacial region not associated with traum a or surgery is always a sinister sign dem anding an
aggressive workup. In case 1, initial facial pain sym ptoms and m andibular deviation seemed to indicate a TMJ problem, b u t with 5th nerve paresthesia, this diagnosis became untenable. Despite extensive workups (radiographs, CT scans) and consultations, the tum or diagnosis was not confirmed until after an open biopsy of the infiltrated lateral pterygoid muscle. The prim ary site was not discovered until a later ENT evaluation. The two risk factors in this case were tobacco use and occupation.56 In case 2, a chronic infection apparently was introduced into the tissues w hen an infected tooth was extracted and caused symptoms—interpreted for nine months as TMJ related. Actinomycosis is a specific gran ulomatosis mycotic disease occur ring mostly in the cervicofacial re gion.79The disease is characterized by indurated swellings, which break down to form multiple fistulae. The reported anatomic location of head and neck actinomycosis includes most anatom ic sites: the retromaxillary space, the TMJ and the mastoid and tem poral bone. No infection, however, has been reported in the infratem poral space. The most common parotid tum or is a painless mass in the preor infra-auricular area.1012A small portion of the parotid gland resides deep to the facial nerve and is wedged betw een the ramus of the m andible and the mastoid process. This hourglass- or “dumbbell”- shaped tum or is considered relatively rare and can produce confusing symptoms. Facial paralysis is rarely associated with deep lobe tumors; however, when present, it is considered suspicious as it may indicate advanced malignancy. Progressive hypomobility of the
mandible has never previously been reported with clinical evidence of deep lobe neoplasm. Because of their location, deep lobe parotid tum ors are surgically less accessible and often result in a higher degree of morbidity. S U M M A R Y
The differential diagnosis of pain in the face and dysfunction of the m asticatory system is complex. Although m ost diagnoses are readily made, common symptoms can occasionally belie more serious diseases. Each patient was originally treated for TMJ dysfunc tions and subsequently was diag nosed as suffering from pathosis of the infratem poral space. ■ Additional case histories a re provided by Dr. J.B. Carter and Dr. J. D. Stone, Wang Ambulatory Care Center 230, Boston 02114.
Ü!
1. Heggie AA, Reade PC. A m alignant lesion of the infratem poral fossa causing m andibular displacement. Case report. Aust Dent J Dr. Keith is 1984;29:145-9. associate professor 2. Cohen SG, Quinn PD. Facial trism us and of oral and m axillo myofascial pain associated facial surgery, with infections and Harvard School of m alignant disease. Oral Surg Dental Medicine; Oral Med Oral Pathol visiting oral and 1988;65:538-44. 3. DelBalso AM, Sweeney maxillofacial AT, Kapur S. An unusual surgeon, M assa cause of facial trism us in a chusetts General child: report of case. JADA Hospital; Chief, Oral 1986;112:207-9. Surgery, Harvard 4. Grace EG, North AF. Tem porom andibular joint Community Health dysfunction and orofacial Plan, W ang Am bula pain caused by parotid gland tory C are C enter malignancy: report of case. 230, Boston 0 2 1 1 4 . JADA 1988;116:348-50. 5. Thawley SE, Panje WR. Address requests for In: Kornblut AD. reprints to Dr. Keith. Comprehensive m anagem ent of head and neck tumors: clinical evaluation of tum ors of the oral cavity. Philadelphia:Saunders;1987:460. 6. Huygen PLM, Fischer AJEM, Vanden BP. Nasopharyngeal cancer: a clinical study with special refer ence to age and occupation. Clin Otolaryngol 1980;5:37-47. 7. N orm an JEdeB. Cervicofacial actinomycosis. Oral Surg Oral Med Oral Pathol 1970;29:735-45. 8. Rankow RM, Abraham DM. Actinomycosis m asquerading in the head and neck. Ann Otol Rhinol Laryngol 1978;87:230-7. 9. Richtsm eier WJ, Johns ME. Actinomycosis of the head and neck. CRC Crit Rev Clin Lab Sci 1979;11:175-202. 10. H anna DC, Gaisford JC, Richardson GS, et al. Tum ors of the deep lobe of the parotid gland. Am J Surg 1968;116:524-7. 11. Morfit HM. Retrom andibular parotid tumors: their surgical treatm ent and m ode of origin. AMA Arch Surg 1955;70:906-13. 12. Patey DH, Thackray AC. The pathologic anatom y and treatm ent of parotid tum ors with retropharyngeal extension (dumbbell tum or) w ith a report of four cases. Br J Surg 1957;44:352. i m
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JADA, Vol. 122, November 1991
61
