TMJ ANKYLOSIS
886 J Oral Maxillofac 46:666-670,
FOLLOWING
MASTOIDITIS
Surg
1990
Temporomandibular Joint Ankylosis Following Mastoiditis: Report of a Case THOMAS
H. FAERBER, MD, DDS,* ROBERT L. ENNIS, DDS,t AND GREGORY A. ALLEN, DDSt
The etiology and treatment of temporomandibular joint (TMJ) ankylosis have been well documented in the literature,‘,’ with trauma and infection identified as the two leading causes3 Recent reports have implicated trauma as the major cause, with infection being a distant second.4‘6 The purpose of this report is to present a case of ankylosis following otitis media and mastoiditis and to summarize previously reported cases of TMJ ankylosis. Report of a Case A 15month-old white girl was admitted to the hospital by her pediatrician on March 15, 1983, with a temperature of 103.7”F. The child was lethargic, inattentive, and appeared to be in a toxic condition. There was postauricular and right-sided facial swelling, rhinorrhea, and erythematous tympanic membranes with obvious effusions bilaterally. The rest of the physical examination was normal. Cultures of blood, cerebrospinal fluid (CSF), the nose, and the pharynx were obtained. Initial laboratory tests showed a hemoglobin level of 10.4 g/dL, hematocrit of 33.3%, white blood cell (WBC) count of 11, 400/mm3, and a differential count with 40 bands and 31 segmented neutrophils. The CSF was grossly clear. Initial management entailed intravenous (IV) fluids and antibiotic therapy. Intravenous ampicillin and chloramphenicol were the empiric choices. An otolaryngologist was immediately consulted, who performed bilateral myringotomies. The ear drainage was cultured and the patient was continued on the initial antibiotic regimen. The CSF, nasal, and pharyngeal cultures were negative. Staphylococcus epidermidis was grown from the ear culture. * Resident, Oral and Maxillofacial Surgery, Graduate Training Program, Truman Medical Center and University of MissouriKansas Citv. Schools of Dentistry and Medicine, Kansas City, MO. t In
-’
0 1990 geons
American
private practice, Independence and Kansas City, MO. Address correspondence and reprint requests to Dr Faerber: Department of Oral and Maxillofacial Surgery, Truman Medical Center, 2301 Holmes St, Kansas City, MO 64108. Association
0278-2391/90/4808-0017$3.00/0
of Oral
and Maxillofacial
Sur-
The patient’s condition did not improve. The WBC count increased to 21,500/mm3 with 21 bands and 31 segmented neutrophils. The right ear developed a profuse mucopurulent discharge and there was persistent postauricular swelling. On approximately the fourth hospital day the antibiotic regimen was changed to IV nafcillin, with obvious improvement within 48 hours. A computed tomography (CT) scan, mastoid radiographs, and radioisotope scan were obtained, which showed a right-sided mastoiditis. A simple right mastoidectomy was performed. During this procedure a large subperiosteal abscess was found, which was drained and cultured. Anaerobic cultures were negative, but aerobic cultures were again positive for S epidermidis. Serum immunoglobulin electrophoresis was normal. The patient was continued on IV nafcillin for approximately 2 weeks. Her hemoglobin level dropped to 8.1 gldL and she received a packed red blood cell transfusion. The patient improved and was sent home with a prescription for cephalexin and a return appointment. At the age of 45 months, the patient was referred by her pediatrician for oral and maxillofacial surgery consultation because of limited mandibular movement. Initial examination revealed a maximum opening of 12 mm at the central incisors, with deviation to the right upon opening and limited left lateral excursion. A panoramic radiograph revealed a greatly enlarged right mandibular condyle and a moth-eaten appearance over the entire right TMJ. Following initial examination, physiotherapy was prescribed using jaw opening exercises for 10 minutes twice a day. The parents were informed that nonsurgical treatment would be used initially and that surgical treatment would be considered if complete ankylosis, asymmetry, or increased dysfunction developed. The patient was followed for approximately 3 years. The maximum mandibular opening achieved was 14 mm. It was also noted during later clinic visits that some mandibular asymmetry was developing. Follow-up panoramic radiographs showed continued abnormal right TMJ morphology and increased antegonial notching (Fig 1). Considering the lack of response to physiotherapy and the developing asymmetry, a surgical correction was planned. On May 9, 1988, a CT scan showed a grossly enlarged right mandibular condyle with obliteration of the glenoid fossa (Fig 2). A thin plate of bone remained between the middle cranial fossa and the glenoid fossa. A surgical treatment plan was discussed, with the understanding that additional surgery might be necessary in the future. Surgical correction was undertaken on August 23,
FAERBER,
867
ENNIS, AND ALLEN
FIGURE 1. Panoramic radiograph of patient at 52 months of age. The condyle-fossa relationship is grossly abnormal, as well as the bony anatomy. The right ramus shows a decreased vertical
dimension. Generalized radiopacity is present.
1988, when the patient was 6 years 8 months old. Fiberoptic oral endotracheal intubation was used due to the limited mandibular opening. A submandibular approach to the ramus and TMJ revealed a solid bony ankylosis. Ostectomy and osteoplasty with a tapered fissure bur were performed to free the ankylosis and provide room for a costochondral rib graft (Fig 3). A right coronoidectomy was also performed. After freeing the mandible, the oral endotracheal tube was exchanged for a nasal tube. Erich arch bars were placed, and maxillomandibular fixation obtained. A pediatric surgeon performed a harvest of a left rib, including the costochondral junction. The cartilaginous portion of the graft was placed in the prepared fossa, and the graft was secured with three 26gauge wires (Fig 4). A convenient wiring technique that provided three-dimensional stabilization to the thin pediatric rib was used. The rib was first grooved to accommodate the upper and lower 26-gauge wires. The middle wire was passed through two holes drilled in the rib and a hole in the ramus. The wires above and below ran circumferentially around the rib and through holes in the ramus. Together these wires provided three-dimensional fixation (Fig 5). The surgical site was closed in layers. The maxillomandibular fixation was released at 2 weeks, and training elastics were applied. On return appointments, the patient initially showed good opening and
FIGURE 3. ment.
FIGURE 2. CT scan of patient at 6 years, 5 months of age showing osteoblastic process of the right TMJ. The condyle is grossly distorted, especially in the medial-lateral dimension.
FIGURE 4. Fixation of costochondral graft to mandibular ramus, with placement of cartilaginous segment in surgically prepared glenoid fossa.
Osteotomy
through the ankylosed condylar seg-
compliance with physiotherapy. Later visits revealed reduced opening and incomplete compliance with physiotherapy. Because the ankylosis had recurred, she was returned to surgery on March 20, 1989. Reexploration of the right TMJ showed a fibrous ankylosis between the newly formed condyle and the glenoid fossa, and a bony ankylosis involving the entire sigmoid notch and newly reformed coronoid process bonding the latter regions to the articular eminence and adjacent temporal bone. Since
868
TMJ ANKYLOSIS FOLLOWING MASTOIDITIS
\B :,
.’
.‘,’
&
.I:.
FIGURE 5. Illustration depicting the wiring technique used. The rib is grooved to accommodate the 26-gauge wires. The middle wire enters the rib in a horizontal fashion; the wires above and below run circumferentially around the rib and to the ramus. Together these wires provide three-dimensional stabilization to the thin pediatric rib.
this was a recurrent ankylosis with abundant new bone formation, it was felt that an aggressive ostectomy was necessary to prevent ankylosis from recurring. Two cuts were made at 90” to each other with a tapered bur. The vertical cut, starting at the posterior slope of the sigmoid notch and extending inferiorly approximately 2 cm, was connected to a horizontal osteotomy below the reformed coronoid process. The horizontal osteotomy extended through the anterior border of the ramus. The bone was freed superiorly with a periosteal elevator and tapered bur, where necessary. Upon completion of the osteotomies and dissection, a 2 x 1.5 cm mass of bone consisting of the reformed coronoid process, mandibular ramus, sigmoid notch, and ankylotic bone was removed. The condyle was freed from its fibrous attachments to the glenoid fossa with a periosteal elevator. Following this, the mandible was mobilized and taken through a range of opening and excursive movements. It was noted that there was deviation to the left side on opening as well as a clinical impression of resistance to excursive movements from the left coronoid and maxillary tuberosity region. A left coronoidectomy was then done in an attempt to further improve mandibular opening and reduce deviation to the left side. A slight reduction in deviation on opening was noted following the coronoidectomy, but no change in vertical opening at the incisors. Follow-up visits after the second surgery were unremarkable. To date, the patient is opening 25 mm at the incisors and has apparent symmetrical mandibular growth. The well-developed condyle is visualized on the panoramic radiograph (Fig 6).
Discussion A classification of ankylosis was proposed by Kazanjian in 1938that divided the disorder into true
FIGURE 6. Panoramic radiograph following second surgery. Note the integration of the rib graft as well as the new condylar morphology (arrowhead). The left coronoidectomy is also visible.
and false varieties.7 True ankylosis was considered as any condition that produced fibrous or bony adhesions between the articular surfaces of the TMJ. False ankylosis resulted from pathologic conditions outside of the joint that resulted in limited mandibular mobility. Most authors agree that the onset of most cases of TMJ ankylosis is before 10 years of age, but surgical correction usually occurs after 10 years. ’ ,* The standard for surgical correction today is to operate when the ankylosis is recognized.‘P2,8 The theory that early surgical treatment inhibits mandibular growth is no longer supported.’ On the contrary, most agree that surgically correcting the ankylotic condyle with autogenous costochondral grafting as early as possible will give the best potential for subsequent growth.2*8 Infection and inflammation of the TMJ may cause true ankylosis. Straith and Lewis3 classified inflammatory lesions of the TMJ as 1) primary inflammation of the joint, 2) secondary inflammation due to a nearby local process, and 3) secondary inflammation due to a generalized bloodstream infection. Local infection of surrounding structures (eg, mastoiditis and otitis media) has been implicated in several large series’,3*‘9*20investigating TMJ ankylosis. Otitis media is a common complication of measles in children. It is reportedly more likely to affect the TMJ if there is an obstruction to the exit of pus, such as aural polyps, cholesteatoma, or impacted cerumen of keratosis obturans.’ Mastoiditis and osteomyelitis of the temporal bone or mandible are also in the category of localized infections. Local infections are on the decline due to improved antimicrobial therapy. Septicemia causing TMJ ankylosis is a rare occurrence. Children can develop septic arthritis following a variety of local or systemic infectious diseases. Typhoid and scarlet fever are good examples of systemic infections associated with septic arthritis, although rarely seen today. The former can involve the TMJ through the bloodstream or cause otitis and mastoiditis which spreads to the joint by direct extension.’ Metastatic septic arthritis may follow pneumonia, gonorrhea,
FAERBER,
869
ENNIS, AND ALLEN
meningitis, bacterial endocarditis, septic pharyngitis, osteomyelitis, measles, infected wounds, peritonsillar abscess, infected varicose ulcers, furuncles, infected burns, pyelitis, infections of the umbilicus, and sinusitis. lo Inflammation of the joint due to the direct extension of infection from other sites includes otitis media, mastoiditis, osteomyelitis of the temporal bone or condyle, and soft tissue abscesses. Otitis associated with measles is often caused by secondary infection with hemolytic streptococci. Secondary infection is also the probable cause of TMJ ankylosis in cases following measles, mumps, and smallpox. Zygomatic, temporal, parotid, and dentoalveolar abscesses all have been associated with TMJ ankylosis involving the joint by direct or hematogenous spread. ’ Topazian’ lists three mechanisms in which otitis media is implicated in causing ankylosis: I) primary otitis media causing secondary joint involvement, 2) suppurative arthritis of the TMJ involving the middle ear secondarily, and 3) suppurative arthritis of the joint draining into the cartilaginous canal. Murphy” suggests that pus in the middle ear burrows forward and ruptures into the glenoid cavity. A thin plate of bone separates the middle ear from the glenoid fossa. Barriers to drainage can force infection into the joint cavity. Bellinger12 states that a false diagnosis of otitis media can occur when suppurative arthritis of the joint drains into the external auditory meatus. Infections can spread from mastoiditis in three different ways: direct extension, thrombophlebitis, and hematogenous dissemination. Bone-destructive lesions, whether in the mastoid or petrous region, occur by direct extension, as do all of the aural complications. The bony walls of the pneumatized spaces of the mastoid are important barriers to the spread of infection. In the child, dense barriers of bone may not be developed to prevent the spread of infection. l3 Keith states that growth and maturation changes are not completed in the TMJ until the second decade of life. The articular eminence does not complete its mature S-shape morphology until 6 to 7 years of age.14 Wright and Moffet have described the postnatal development of the TMJ. They state that between 6 months and 2% years the articular tubercle and glenoid fossa take on a mature appearance. In their dissections they found that the tympanosquamosal fissure remains open medially and is divided into petrosquamosal and petrotympanic fissures by the presence of the tegmen tympani.15 The presence of these open sutures in the glenoid fossa could explain the spread of middle ear or mastoid infections to the TMJ in this young age group.
Delayed ossification of the tympanic plate could also provide a pathway for the direct extension of infection from the middle ear. Moffett states that the tympanic plate does not complete ossification until approximately 5 years of age. Dried skulls of those less than 10 years of age consistently show a tiny opening or perforation in the central part of the tympanic plate, reflecting incomplete ossification. In approximately 20% of adult skulls this opening can still be seen as the result of incomplete ossification.‘6 Ingervall et all7 describe the absence of ossification in the temporal component of the TMJ and demonstrated this in microradiographs of a TMJ from an 18-month-old child. The illustration in Fig 7 depicts the possible routes by which infection could spread to the TMJ based on the previously mentioned anatomical studies. Such infection could spread to the TMJ via open sutures, incomplete ossification, or delayed ossification. Therefore, it is understandable how a child with otitis or mastoiditis could have TMJ involvement. Review of some large series of TMJ ankylosis reveals that otitis media and mastoiditis have been associated with ankylosis.1*3*‘9~20 The degree to which these conditions occur, however, vary from study to study. Earlier reports in the literature had a larger number of cases associated with infectious causes; recent reports show a decline in ankylosis associated with infection.4-6 Improved antimicrobial therapy, without a doubt, has reduced the severity or progression of infectious causes such as otitis and mastoiditis. Orlow, in 1902, reported on 104 cases of TMJ ankylosis of which 29% were associated with trauma.‘* Blair, in 1914, in a series of 212 cases of ankylosis, stated that the vast majority were associated with trauma. Scarlet fever with joint suppuration was second in frequency, and otitis media and/or local joint suppuration was third.” Tympanlc
a’r cells PetrotyrApanic fissure
FIGURE 7. Illustration depicting two possible anatomical routes through which infection could spread directly from middle ear structures to the TMJ.
870
TMJ ANKYLOSIS
Dufourmental and Darcissac, in 1935, reported 100 cases of ankylosis in which 8 were associated with “osteomyelitis of auricular origin”.*’ Of note in this report is the number of ankylosis cases associated with other infections, (ie, 10 with gonococcal arthritis and 9 with osteomyelitis of dental origin). Straith and Lewis, in 1948, reported 6 cases out of a total of 16 that were associated with otitis media (4) and mastoiditis (2). Four of these infections followed episodes of scarlet fever.3 Topazian, in 1964, in discussing the etiology of TMJ ankylosis, reported 44 of his own cases and 185 cases from the literature. In his review, he described the incidence of the two major etiologic factors, trauma and infection. Trauma as an etiologic factor ranged from 26% to 75%, whereas infection ranged from 44% to 68%, depending on the series of cases reviewed. Of the 229 cases, otitis media was reported as the cause in 16 cases (6.9%), and mastoiditis in 4 cases (1.7%). In his series, infections were placed under an inflammation category, which included osteomyelitis, actinomycosis, rheumatoid arthritis, diphtheria, typhoid, tonsillitis, mastoiditis, suppurative arthritis, measles, abscesses around the TMJ, scarlet fever, gonococcal arthritis, otitis media, dental infection, and other infections.’ Chandra and Dave, in 1985, reported 258 cases and found 175 (67.8%) and 44 (17%) associated with trauma and infection, respectively. This study described the high frequency of TMJ ankylosis (1 in 500 hospital cases) in India.4 Dhawan, in 1969, in a smaller series from India, reported 16 cases: 10 (62.5%) associated with trauma, 3 (18.7%) with local infection, and 3 (18.7%) with systemic infection.6 Haidar, in 1986, reported 8 cases all of which were associated with trauma.5 Summary A case of TMJ ankylosis following otitis media and mastoiditis is described and its treatment presented. The infectious etiology of ankylosis is reviewed, with emphasis on mastoid infections. Theories are presented as to the possible mechanisms by
which such infections fossa.
FOLLOWING
MASTOIDITIS
can spread into the glenoid
Acknowledgment The authors would like to acknowledge that Norman Burk, DDS, and Loren J. Wendelburg, DDS, contributed their expertise in the surgical treatment of this case, postoperative care, and preparation of this manuscript.
References 1. Topazian RG: Etiology of ankylosis of the temporomandibular ioint: Analvsis of 44 cases. J Oral Sure: 22:227. 1964 2. Miller GA, Page HL, Griffith CR: Temporom&dibul& joint ankylosis: Review of the literature and report of two cases of bilateral involvement. J Oral Surg 33:792, 1975 3. Straith CL, Lewis JR: Ankylosis of the temporomandibular joint. Plast Reconstr Surg 3:464, 1948 4. Chandra P, Dave PK: Temporomandibular joint ankylosis. Prog Clin Biol Res p 449, 1985 5. Haidar Z: Ankylosis of the temporo-mandibular joint: Causes and management. J Oral Med 41:246, 1986 6. Dhawan IK: Ankylosis of the temporo-mandibular joint. Panminerva Med 11:71, 1969 7. Kazanjian VH: Ankylosis of the temporomandibular joint. Am J Orthod 24:1181, 1938 8. Kennett S: Temporomandibular joint ankylosis: The rationale for grafting in the young patient. J Oral Surg 31:744, 1973 9. Sleewaegen N, et al: Five cases of temporomandibular joint ankylosis in children. Acta Stomatol Belg 68:95, 1971 10. Balboni VG: Other forms of infectious arthritis, in Hollander JL (ed): Arthritis and Allied Conditions (ed 7). Philadelphia, PA, Lea & Febiger, 1966, p 1037 11. Murphy JB: Arthroplasty for intra-articular bony and fibrous ankylosis of the temporomandibular articulation: Report of nine cases. JAMA 62:1783, 1914 12. Bellinger DH: Temporomandibular ankylosis and its surgical corrections. J Am Dent Assoc 27:1563, 1940 13. Neely: cited in Otolaryngology-Head and Neck Surgery,vol 4. Schullerr DE, et al: St Louis, MO, Mosby, 1986, pp 2992-2996 14. Keith DA: Development of the human temporomandibular joint. Br J Oral .&rg 20:217, 1982 15. Wrieht DM. Moffett BC: The nostnatal develooment of the himan temporomandibularjbint. Am J Anat i41:23S1 1974 16. Moffett B: The morphogenesis of the temporoman&bular joint. Am J Orthod 52:401, 1986 17. Ingervall B, Carlsson GE, Thilander B: Postnatal development of the human temporomandibular joint. II. A microradiographic study. Acia Odontol Stand 34: 133, 1976 18. Orlow LW: Ankvlosis Mandibulae verae. Dtsch Z Chir 16:399, 1902 . 19. Blair VP: Operative treatment of ankylosis of the mandible. Surg Gynecol Obstet 19:436, 1914 20. Dufourmente L, Darcissac M: Bull et mem Sot d chirugiens de Paris. 27:148, 1935, cited by Padgett EC, Robinson DW, Stephenson KL: Ankylosis of the temporomandibular joint. Surgery 24:426, 1948
886 J Oral Maxillofac 46:666-670,
FOLLOWING
MASTOIDITIS
Surg
1990
Temporomandibular Joint Ankylosis Following Mastoiditis: Report of a Case THOMAS
H. FAERBER, MD, DDS,* ROBERT L. ENNIS, DDS,t AND GREGORY A. ALLEN, DDSt
The etiology and treatment of temporomandibular joint (TMJ) ankylosis have been well documented in the literature,‘,’ with trauma and infection identified as the two leading causes3 Recent reports have implicated trauma as the major cause, with infection being a distant second.4‘6 The purpose of this report is to present a case of ankylosis following otitis media and mastoiditis and to summarize previously reported cases of TMJ ankylosis. Report of a Case A 15month-old white girl was admitted to the hospital by her pediatrician on March 15, 1983, with a temperature of 103.7”F. The child was lethargic, inattentive, and appeared to be in a toxic condition. There was postauricular and right-sided facial swelling, rhinorrhea, and erythematous tympanic membranes with obvious effusions bilaterally. The rest of the physical examination was normal. Cultures of blood, cerebrospinal fluid (CSF), the nose, and the pharynx were obtained. Initial laboratory tests showed a hemoglobin level of 10.4 g/dL, hematocrit of 33.3%, white blood cell (WBC) count of 11, 400/mm3, and a differential count with 40 bands and 31 segmented neutrophils. The CSF was grossly clear. Initial management entailed intravenous (IV) fluids and antibiotic therapy. Intravenous ampicillin and chloramphenicol were the empiric choices. An otolaryngologist was immediately consulted, who performed bilateral myringotomies. The ear drainage was cultured and the patient was continued on the initial antibiotic regimen. The CSF, nasal, and pharyngeal cultures were negative. Staphylococcus epidermidis was grown from the ear culture. * Resident, Oral and Maxillofacial Surgery, Graduate Training Program, Truman Medical Center and University of MissouriKansas Citv. Schools of Dentistry and Medicine, Kansas City, MO. t In
-’
0 1990 geons
American
private practice, Independence and Kansas City, MO. Address correspondence and reprint requests to Dr Faerber: Department of Oral and Maxillofacial Surgery, Truman Medical Center, 2301 Holmes St, Kansas City, MO 64108. Association
0278-2391/90/4808-0017$3.00/0
of Oral
and Maxillofacial
Sur-
The patient’s condition did not improve. The WBC count increased to 21,500/mm3 with 21 bands and 31 segmented neutrophils. The right ear developed a profuse mucopurulent discharge and there was persistent postauricular swelling. On approximately the fourth hospital day the antibiotic regimen was changed to IV nafcillin, with obvious improvement within 48 hours. A computed tomography (CT) scan, mastoid radiographs, and radioisotope scan were obtained, which showed a right-sided mastoiditis. A simple right mastoidectomy was performed. During this procedure a large subperiosteal abscess was found, which was drained and cultured. Anaerobic cultures were negative, but aerobic cultures were again positive for S epidermidis. Serum immunoglobulin electrophoresis was normal. The patient was continued on IV nafcillin for approximately 2 weeks. Her hemoglobin level dropped to 8.1 gldL and she received a packed red blood cell transfusion. The patient improved and was sent home with a prescription for cephalexin and a return appointment. At the age of 45 months, the patient was referred by her pediatrician for oral and maxillofacial surgery consultation because of limited mandibular movement. Initial examination revealed a maximum opening of 12 mm at the central incisors, with deviation to the right upon opening and limited left lateral excursion. A panoramic radiograph revealed a greatly enlarged right mandibular condyle and a moth-eaten appearance over the entire right TMJ. Following initial examination, physiotherapy was prescribed using jaw opening exercises for 10 minutes twice a day. The parents were informed that nonsurgical treatment would be used initially and that surgical treatment would be considered if complete ankylosis, asymmetry, or increased dysfunction developed. The patient was followed for approximately 3 years. The maximum mandibular opening achieved was 14 mm. It was also noted during later clinic visits that some mandibular asymmetry was developing. Follow-up panoramic radiographs showed continued abnormal right TMJ morphology and increased antegonial notching (Fig 1). Considering the lack of response to physiotherapy and the developing asymmetry, a surgical correction was planned. On May 9, 1988, a CT scan showed a grossly enlarged right mandibular condyle with obliteration of the glenoid fossa (Fig 2). A thin plate of bone remained between the middle cranial fossa and the glenoid fossa. A surgical treatment plan was discussed, with the understanding that additional surgery might be necessary in the future. Surgical correction was undertaken on August 23,
FAERBER,
867
ENNIS, AND ALLEN
FIGURE 1. Panoramic radiograph of patient at 52 months of age. The condyle-fossa relationship is grossly abnormal, as well as the bony anatomy. The right ramus shows a decreased vertical
dimension. Generalized radiopacity is present.
1988, when the patient was 6 years 8 months old. Fiberoptic oral endotracheal intubation was used due to the limited mandibular opening. A submandibular approach to the ramus and TMJ revealed a solid bony ankylosis. Ostectomy and osteoplasty with a tapered fissure bur were performed to free the ankylosis and provide room for a costochondral rib graft (Fig 3). A right coronoidectomy was also performed. After freeing the mandible, the oral endotracheal tube was exchanged for a nasal tube. Erich arch bars were placed, and maxillomandibular fixation obtained. A pediatric surgeon performed a harvest of a left rib, including the costochondral junction. The cartilaginous portion of the graft was placed in the prepared fossa, and the graft was secured with three 26gauge wires (Fig 4). A convenient wiring technique that provided three-dimensional stabilization to the thin pediatric rib was used. The rib was first grooved to accommodate the upper and lower 26-gauge wires. The middle wire was passed through two holes drilled in the rib and a hole in the ramus. The wires above and below ran circumferentially around the rib and through holes in the ramus. Together these wires provided three-dimensional fixation (Fig 5). The surgical site was closed in layers. The maxillomandibular fixation was released at 2 weeks, and training elastics were applied. On return appointments, the patient initially showed good opening and
FIGURE 3. ment.
FIGURE 2. CT scan of patient at 6 years, 5 months of age showing osteoblastic process of the right TMJ. The condyle is grossly distorted, especially in the medial-lateral dimension.
FIGURE 4. Fixation of costochondral graft to mandibular ramus, with placement of cartilaginous segment in surgically prepared glenoid fossa.
Osteotomy
through the ankylosed condylar seg-
compliance with physiotherapy. Later visits revealed reduced opening and incomplete compliance with physiotherapy. Because the ankylosis had recurred, she was returned to surgery on March 20, 1989. Reexploration of the right TMJ showed a fibrous ankylosis between the newly formed condyle and the glenoid fossa, and a bony ankylosis involving the entire sigmoid notch and newly reformed coronoid process bonding the latter regions to the articular eminence and adjacent temporal bone. Since
868
TMJ ANKYLOSIS FOLLOWING MASTOIDITIS
\B :,
.’
.‘,’
&
.I:.
FIGURE 5. Illustration depicting the wiring technique used. The rib is grooved to accommodate the 26-gauge wires. The middle wire enters the rib in a horizontal fashion; the wires above and below run circumferentially around the rib and to the ramus. Together these wires provide three-dimensional stabilization to the thin pediatric rib.
this was a recurrent ankylosis with abundant new bone formation, it was felt that an aggressive ostectomy was necessary to prevent ankylosis from recurring. Two cuts were made at 90” to each other with a tapered bur. The vertical cut, starting at the posterior slope of the sigmoid notch and extending inferiorly approximately 2 cm, was connected to a horizontal osteotomy below the reformed coronoid process. The horizontal osteotomy extended through the anterior border of the ramus. The bone was freed superiorly with a periosteal elevator and tapered bur, where necessary. Upon completion of the osteotomies and dissection, a 2 x 1.5 cm mass of bone consisting of the reformed coronoid process, mandibular ramus, sigmoid notch, and ankylotic bone was removed. The condyle was freed from its fibrous attachments to the glenoid fossa with a periosteal elevator. Following this, the mandible was mobilized and taken through a range of opening and excursive movements. It was noted that there was deviation to the left side on opening as well as a clinical impression of resistance to excursive movements from the left coronoid and maxillary tuberosity region. A left coronoidectomy was then done in an attempt to further improve mandibular opening and reduce deviation to the left side. A slight reduction in deviation on opening was noted following the coronoidectomy, but no change in vertical opening at the incisors. Follow-up visits after the second surgery were unremarkable. To date, the patient is opening 25 mm at the incisors and has apparent symmetrical mandibular growth. The well-developed condyle is visualized on the panoramic radiograph (Fig 6).
Discussion A classification of ankylosis was proposed by Kazanjian in 1938that divided the disorder into true
FIGURE 6. Panoramic radiograph following second surgery. Note the integration of the rib graft as well as the new condylar morphology (arrowhead). The left coronoidectomy is also visible.
and false varieties.7 True ankylosis was considered as any condition that produced fibrous or bony adhesions between the articular surfaces of the TMJ. False ankylosis resulted from pathologic conditions outside of the joint that resulted in limited mandibular mobility. Most authors agree that the onset of most cases of TMJ ankylosis is before 10 years of age, but surgical correction usually occurs after 10 years. ’ ,* The standard for surgical correction today is to operate when the ankylosis is recognized.‘P2,8 The theory that early surgical treatment inhibits mandibular growth is no longer supported.’ On the contrary, most agree that surgically correcting the ankylotic condyle with autogenous costochondral grafting as early as possible will give the best potential for subsequent growth.2*8 Infection and inflammation of the TMJ may cause true ankylosis. Straith and Lewis3 classified inflammatory lesions of the TMJ as 1) primary inflammation of the joint, 2) secondary inflammation due to a nearby local process, and 3) secondary inflammation due to a generalized bloodstream infection. Local infection of surrounding structures (eg, mastoiditis and otitis media) has been implicated in several large series’,3*‘9*20investigating TMJ ankylosis. Otitis media is a common complication of measles in children. It is reportedly more likely to affect the TMJ if there is an obstruction to the exit of pus, such as aural polyps, cholesteatoma, or impacted cerumen of keratosis obturans.’ Mastoiditis and osteomyelitis of the temporal bone or mandible are also in the category of localized infections. Local infections are on the decline due to improved antimicrobial therapy. Septicemia causing TMJ ankylosis is a rare occurrence. Children can develop septic arthritis following a variety of local or systemic infectious diseases. Typhoid and scarlet fever are good examples of systemic infections associated with septic arthritis, although rarely seen today. The former can involve the TMJ through the bloodstream or cause otitis and mastoiditis which spreads to the joint by direct extension.’ Metastatic septic arthritis may follow pneumonia, gonorrhea,
FAERBER,
869
ENNIS, AND ALLEN
meningitis, bacterial endocarditis, septic pharyngitis, osteomyelitis, measles, infected wounds, peritonsillar abscess, infected varicose ulcers, furuncles, infected burns, pyelitis, infections of the umbilicus, and sinusitis. lo Inflammation of the joint due to the direct extension of infection from other sites includes otitis media, mastoiditis, osteomyelitis of the temporal bone or condyle, and soft tissue abscesses. Otitis associated with measles is often caused by secondary infection with hemolytic streptococci. Secondary infection is also the probable cause of TMJ ankylosis in cases following measles, mumps, and smallpox. Zygomatic, temporal, parotid, and dentoalveolar abscesses all have been associated with TMJ ankylosis involving the joint by direct or hematogenous spread. ’ Topazian’ lists three mechanisms in which otitis media is implicated in causing ankylosis: I) primary otitis media causing secondary joint involvement, 2) suppurative arthritis of the TMJ involving the middle ear secondarily, and 3) suppurative arthritis of the joint draining into the cartilaginous canal. Murphy” suggests that pus in the middle ear burrows forward and ruptures into the glenoid cavity. A thin plate of bone separates the middle ear from the glenoid fossa. Barriers to drainage can force infection into the joint cavity. Bellinger12 states that a false diagnosis of otitis media can occur when suppurative arthritis of the joint drains into the external auditory meatus. Infections can spread from mastoiditis in three different ways: direct extension, thrombophlebitis, and hematogenous dissemination. Bone-destructive lesions, whether in the mastoid or petrous region, occur by direct extension, as do all of the aural complications. The bony walls of the pneumatized spaces of the mastoid are important barriers to the spread of infection. In the child, dense barriers of bone may not be developed to prevent the spread of infection. l3 Keith states that growth and maturation changes are not completed in the TMJ until the second decade of life. The articular eminence does not complete its mature S-shape morphology until 6 to 7 years of age.14 Wright and Moffet have described the postnatal development of the TMJ. They state that between 6 months and 2% years the articular tubercle and glenoid fossa take on a mature appearance. In their dissections they found that the tympanosquamosal fissure remains open medially and is divided into petrosquamosal and petrotympanic fissures by the presence of the tegmen tympani.15 The presence of these open sutures in the glenoid fossa could explain the spread of middle ear or mastoid infections to the TMJ in this young age group.
Delayed ossification of the tympanic plate could also provide a pathway for the direct extension of infection from the middle ear. Moffett states that the tympanic plate does not complete ossification until approximately 5 years of age. Dried skulls of those less than 10 years of age consistently show a tiny opening or perforation in the central part of the tympanic plate, reflecting incomplete ossification. In approximately 20% of adult skulls this opening can still be seen as the result of incomplete ossification.‘6 Ingervall et all7 describe the absence of ossification in the temporal component of the TMJ and demonstrated this in microradiographs of a TMJ from an 18-month-old child. The illustration in Fig 7 depicts the possible routes by which infection could spread to the TMJ based on the previously mentioned anatomical studies. Such infection could spread to the TMJ via open sutures, incomplete ossification, or delayed ossification. Therefore, it is understandable how a child with otitis or mastoiditis could have TMJ involvement. Review of some large series of TMJ ankylosis reveals that otitis media and mastoiditis have been associated with ankylosis.1*3*‘9~20 The degree to which these conditions occur, however, vary from study to study. Earlier reports in the literature had a larger number of cases associated with infectious causes; recent reports show a decline in ankylosis associated with infection.4-6 Improved antimicrobial therapy, without a doubt, has reduced the severity or progression of infectious causes such as otitis and mastoiditis. Orlow, in 1902, reported on 104 cases of TMJ ankylosis of which 29% were associated with trauma.‘* Blair, in 1914, in a series of 212 cases of ankylosis, stated that the vast majority were associated with trauma. Scarlet fever with joint suppuration was second in frequency, and otitis media and/or local joint suppuration was third.” Tympanlc
a’r cells PetrotyrApanic fissure
FIGURE 7. Illustration depicting two possible anatomical routes through which infection could spread directly from middle ear structures to the TMJ.
870
TMJ ANKYLOSIS
Dufourmental and Darcissac, in 1935, reported 100 cases of ankylosis in which 8 were associated with “osteomyelitis of auricular origin”.*’ Of note in this report is the number of ankylosis cases associated with other infections, (ie, 10 with gonococcal arthritis and 9 with osteomyelitis of dental origin). Straith and Lewis, in 1948, reported 6 cases out of a total of 16 that were associated with otitis media (4) and mastoiditis (2). Four of these infections followed episodes of scarlet fever.3 Topazian, in 1964, in discussing the etiology of TMJ ankylosis, reported 44 of his own cases and 185 cases from the literature. In his review, he described the incidence of the two major etiologic factors, trauma and infection. Trauma as an etiologic factor ranged from 26% to 75%, whereas infection ranged from 44% to 68%, depending on the series of cases reviewed. Of the 229 cases, otitis media was reported as the cause in 16 cases (6.9%), and mastoiditis in 4 cases (1.7%). In his series, infections were placed under an inflammation category, which included osteomyelitis, actinomycosis, rheumatoid arthritis, diphtheria, typhoid, tonsillitis, mastoiditis, suppurative arthritis, measles, abscesses around the TMJ, scarlet fever, gonococcal arthritis, otitis media, dental infection, and other infections.’ Chandra and Dave, in 1985, reported 258 cases and found 175 (67.8%) and 44 (17%) associated with trauma and infection, respectively. This study described the high frequency of TMJ ankylosis (1 in 500 hospital cases) in India.4 Dhawan, in 1969, in a smaller series from India, reported 16 cases: 10 (62.5%) associated with trauma, 3 (18.7%) with local infection, and 3 (18.7%) with systemic infection.6 Haidar, in 1986, reported 8 cases all of which were associated with trauma.5 Summary A case of TMJ ankylosis following otitis media and mastoiditis is described and its treatment presented. The infectious etiology of ankylosis is reviewed, with emphasis on mastoid infections. Theories are presented as to the possible mechanisms by
which such infections fossa.
FOLLOWING
MASTOIDITIS
can spread into the glenoid
Acknowledgment The authors would like to acknowledge that Norman Burk, DDS, and Loren J. Wendelburg, DDS, contributed their expertise in the surgical treatment of this case, postoperative care, and preparation of this manuscript.
References 1. Topazian RG: Etiology of ankylosis of the temporomandibular ioint: Analvsis of 44 cases. J Oral Sure: 22:227. 1964 2. Miller GA, Page HL, Griffith CR: Temporom&dibul& joint ankylosis: Review of the literature and report of two cases of bilateral involvement. J Oral Surg 33:792, 1975 3. Straith CL, Lewis JR: Ankylosis of the temporomandibular joint. Plast Reconstr Surg 3:464, 1948 4. Chandra P, Dave PK: Temporomandibular joint ankylosis. Prog Clin Biol Res p 449, 1985 5. Haidar Z: Ankylosis of the temporo-mandibular joint: Causes and management. J Oral Med 41:246, 1986 6. Dhawan IK: Ankylosis of the temporo-mandibular joint. Panminerva Med 11:71, 1969 7. Kazanjian VH: Ankylosis of the temporomandibular joint. Am J Orthod 24:1181, 1938 8. Kennett S: Temporomandibular joint ankylosis: The rationale for grafting in the young patient. J Oral Surg 31:744, 1973 9. Sleewaegen N, et al: Five cases of temporomandibular joint ankylosis in children. Acta Stomatol Belg 68:95, 1971 10. Balboni VG: Other forms of infectious arthritis, in Hollander JL (ed): Arthritis and Allied Conditions (ed 7). Philadelphia, PA, Lea & Febiger, 1966, p 1037 11. Murphy JB: Arthroplasty for intra-articular bony and fibrous ankylosis of the temporomandibular articulation: Report of nine cases. JAMA 62:1783, 1914 12. Bellinger DH: Temporomandibular ankylosis and its surgical corrections. J Am Dent Assoc 27:1563, 1940 13. Neely: cited in Otolaryngology-Head and Neck Surgery,vol 4. Schullerr DE, et al: St Louis, MO, Mosby, 1986, pp 2992-2996 14. Keith DA: Development of the human temporomandibular joint. Br J Oral .&rg 20:217, 1982 15. Wrieht DM. Moffett BC: The nostnatal develooment of the himan temporomandibularjbint. Am J Anat i41:23S1 1974 16. Moffett B: The morphogenesis of the temporoman&bular joint. Am J Orthod 52:401, 1986 17. Ingervall B, Carlsson GE, Thilander B: Postnatal development of the human temporomandibular joint. II. A microradiographic study. Acia Odontol Stand 34: 133, 1976 18. Orlow LW: Ankvlosis Mandibulae verae. Dtsch Z Chir 16:399, 1902 . 19. Blair VP: Operative treatment of ankylosis of the mandible. Surg Gynecol Obstet 19:436, 1914 20. Dufourmente L, Darcissac M: Bull et mem Sot d chirugiens de Paris. 27:148, 1935, cited by Padgett EC, Robinson DW, Stephenson KL: Ankylosis of the temporomandibular joint. Surgery 24:426, 1948
