97

Journal of Oral Science, Vol. 43, No. 2, 97-101, 2001

Ankylosis of the temporomandibular joint caused by

rheumatoid arthritis: a pathological study and review

Rie Kobayashi, Tadahiko Utsunomiya•˜, Hirotsugu Yamamoto•˜ and Hideaki Nagura

Departments of Oral Surgery and •˜Pathology, Nihon University School of Dentistry at Matsudo, Chiba 271-8587

(Received 9 September 2000 and accepted 12 March 2001)

Abstract: Rheumatoid arthritis (RA) of the ankylosis of the temporomandibular joint (TMJ) due to RA. temporomandibular joint (TMJ) in a 59-year-old To understand the characteristics of the present case, we Japanese woman is reported, including details of studied and discussed its clinical, radiological, and clinical, histopathological and radiological findings. pathological features including the review of the literatures. The patient had been diagnosed as having RA of the right knee joint 41 years previously, and suffered from Case report arthralgia of the right TMJ. Radiological examination A 59-year-old woman was referred to Nihon University showed a radiopaque lesion of the mandibular head and Dental Hospital at Matsudo on April 13, 1988, with a mandibular fossa in the right TMJ and ankylosis of the chief complaint of restricted mandibular opening. The right TMJ was diagnosed on the basis of the clinical maximal inter-incisal mouth opening distance was 22 mm and radiological findings. Condylectomy was at her first visit. Laboratory examination showed a slightly performed. Pathological examination of material from increased erythrocyte sedimentation ratio in the peripheral the joint region revealed a marked increase of collagen blood, and rheumatoid arthritis factor was positive upon fibers associated with slight capillary dilatation and serological examination. There was no contributory family hemorrhage. The final diagnosis was ankylosis of the history, but the patient had been diagnosed as having right TMJ due to RA. The literature on TMJ ankylosis rheumatoid arthritis of the right knee joint in 1947, and secondary to RA is reviewed and discussed. (J. Oral Sci. had been physically treated for the disease. Three years 43, 97-101, 2001) later, she had noticed restricted mouth opening. Since she had little pain in the TMJs, she had not sought any therapy. Key words: rheumatoid arthritis; temporomandibular In 1987, she had developed severe arthralgia in the right joint; ankylosis; radiology; pathology. TMJ. She was admitted to our hospital to undergo condylectomy of the right TMJ, after a clinical and radiological diagnosis of ankylosis on February 19, 1988. Introduction However, as an electrocardiographic examination had Rheumatoid arthritis (RA) is a systemic disease of revealed severe extrasystole, the operation was not carried unknown etiology and is classified as one of the so-called out. The patient was discharged from our hospital on collagen diseases (1). It is characterized by chronic and February 22, and readmitted for surgical treatment on progressive inflammation of the joints with fibrinoid March 28, 1988. Rotational panoramic radiography and degeneration. Temporomandibular arthritis, however, is posteroanterior projection showed a diffuse radiopaque not particularly common despite the fact that it is a lesion of the mandibular head and mandibular fossa of the polyarticular disease. We encountered a rare case of right TMJ. In contrast, the left TMJ revealed no remarkable change. The axial and transorbital images revealed the

Correspondence to Dr. Rie Kobayashi, Department of Oral radiopaque lesion more clearly than rotational panoramic Surgery, Nihon University School of Dentistry at Matsudo, 2- radiography and posteroanterior projection (Fig. 1a-c), 870-1, Sakaecho-Nishi, Matsudo, Chiba 271-8587, Japan and suggested that the joint space had partly disappeared. Tel: +81 473609397 Fax: +81 473609398 Bone destruction and deformity of the right TMJ were 98

therefore suspected (Fig. 2), and a tentative diagnosis of 31, 1988, with a resulting improvement of the mandibular ankylosis of the right TMJ was made. Under general opening to 35 mm. Mouth opening training with a mouth anesthesia, right condylectomy was performed, followed gag was started 3 weeks after the operation, and this by placement of freeze-dried cranio dura mater on March increased the opening to 39 mm. The patient was discharged from the hospital on 33rd day from the operation, and has been followed in our oral surgery clinic at routine intervals.

a) Rotational panoramic radiograph

Fig. 2 Frontal tomographic image showing the joint destruction and bony deformity, and the joint space partly disappeared.

Results

Pathological findings

The extirpated specimen was fixed in 10% neutral

formalin, and decalcified with 10% formic acid formalin.

The tissue was then embedded in paraffin, and sectioned

at a thickness of 4ƒÊm. The serial sections were stained b) Axial image with hematoxylin-eosin (H-E) and azan-Mallory. Macroscopically, the several pieces that comprised the

specimen were small bean to large bean-sized, and

contained grayish-yellow connective tissue with a bony hard

mass.

Microscopically, marked fibrosis and degenerative

cartilage were observed. The fibrosis showed slight

capillary growth and dilatation, and the cartilage was

connected to existing bone tissue. The bone marrow was

composed mainly of fatty marrow with partial fibrous

change (Fig. 3). Marked fibrosis stained with aniline blue

was seen in the mandibular head (Fig. 4). c) Transorbital image

Fig. 1 Rotational panoramic radiograph, axial image and Discussion transorbital images showing a radiopaque lesion of Rheumatoid arthritis is a systemic inflammatory the mandibular head and mandibular fossa in the right connective tissue disease. The etiology of RA has been TMJ. considered to be multifactorial, involving genetic factors,

abnormal immunoreaction, the endocrine system (sex 99

small peripheral joints of the hands and feet are affected first, but any joint of the body may be involved in the early stage. In the late stage, secondary osteoarthrosis and ankylosis may develop (3). The symptoms of arthritis occur frequently. The hand and phalangeal joints, such as the proximal interphalangeal and metacarpophalangeal joints, are affected most often, followed by the elbow, metatarsophalangeal and knee joints (3). The TMJ is affected relatively rarely in contrast with the other joints. However, it is said that the appearance of TMJ symptoms is recognized more than ten years after initial disease onset (4,5). Clinical and radiological examinations show involvement of the TMJ in about 70 % of RA patients (1, Fig. 3 Low power view of the TMJ region. Marked fibrosis 6-8). According to Ogus et al., when the duration of and degenerative fibrous cartilage connecting with the general disease was more than 5 years, TMJ symptoms were exiting bone tissue. (Hematoxylin-eosin stain, original evident in 86 percent of patients with RA (9). In the magnification;•~40) present case, the etiology was not clear, but previous microbiological infection was denied. The feature of the hormone), and environmental factors (infectious agents such disease in this patient conformed to those of RA described above. The progress of RA had occurred over a long period, and the symptoms of the right knee joint had been chronic. She had noticed restriction of mouth opening 3 years after initial diagnosis of RA, but had not sought treatment for more than 30 years because there had been no arthralgia of the TMJ. Radiological examination is a useful and important tool for diagnosis and assessment of TMJ disease. The radiographic features of joint changes are significantly related to the disease duration. The mandibular head may reveal flattening, erosion and obliteration, and gradually the joint space may become narrow due to granulation. The diagnostic imaging techniques used for RA include extraoral radiography (contrast radiography of the joint), Fig. 4 Photomicrograph of strong fibrosis of the articular CT, MRI, arthrography and arthrotomography (10). In the head. (Azan-Mallory stain, original magnification; present case, we used only plain radiography, and this ×100) clearly revealed articular destructive change. The severity of the radiographic changes is graded according to the as bacteria and viruses) (2). The general features of RA Larsen's classification into six grades (0-V): 0: Normal, include 1) a female predominance, 2) a peak in patients I: Slight abnormality, joint space is slightly narrow, II: Early 30-40 years old, 3) polyarthritis, 4) progressive change, 5) abnormality, joint space is narrow, with erosion (+), III: fatigue and subcutaneous nodules, 6) positivity for RA Moderate destruction, joint space is narrow and eroded (+), factor in the serum and 7) bone destruction demonstrated IV: Severe destruction, joint space is narrow, with erosion by radiography. Clinically, the onset of RA is usually (+) and bone deformity (+), V: Mutilating abnormality, insidious, with gradual progressive development of joint disappearance joint space, with erosion (destruction) and pain and swelling with pronounced morning stiffness. bone deformity (++) (7,11). The present case corresponded The symptoms become gradually worse, and extra-articular to grade V, since there was partial disappearance of the joint features also appear. RA has a variety of extra-articular space, joint destruction and bone deformity. features such as vasculitis, anemia, and nodules without Generally, the pathological findings in the TMJ affected articular features (3). However, RA is mainly represented by RA are initially slight, and progress chronically. In the by the articular features. Affected joints are tender on early stage, synovial hyperemia, lymphocyte infiltration, palpation, painful on movement, and swollen (3). Usually, fibrinoid degeneration, and pannus formation are seen 100

(12). The articular cartilage may be destroyed, and reactive Table 1 Reported cases of TMJ ankylosis secondary to RA and regenerative granulation tissue can be seen in the articular cavity. There may be fibrosis and cicatrization, and fibrous adhesion may be present. In the late stage, fibrous ankylosis is observed (3). In the present case, pathological examination revealed thickened fibrous tissue ankylosis, corresponding to the late stage of RA. In general, ankylosis of the joint can be intra-articular or extra-articular. It can also be complete or partial, and fibrous or bony from pathological view points (13). The present case corresponded to intra-articular, partial and fibrous ankylosis. TMJ ankylosis secondary to RA is generally found in the late stage of the disease, but it is a rare finding and has not been well documented (14-19) (Table 1). Previous cases, listed in table 1, included two cases of juvenile rheumatoid arthritis, but the other patients were in their Orlando, 196-214 sixth to seventh decades. The average age of patients 3. Sturrock, R.D. (1987) Rheumatoid arthritis. In with TMJ ankylosis secondary RA was higher than the Clinical rheumatology illustrated. Hart, F.D. ed., average age at which RA is diagnosed. Previous studies Williams and Wilkins and Associates, Sydney, 27- have suggested that ankylosis of the TMJ might occur when 43 the TMJ is affected by RA during childhood (16), and that 4. Miyamoto, H., Sakashita, H., Miyata, M., Miyaji, TMJ ankylosis is unusual in cases of adult RA (5). The Y. and Kurita, K. (1994) Primary symptoms of present patient had no medical history of RA in childhood. rheumatoid arthrosis at temporomandibular joint. The etiology of ankylosis is said to include trauma, birth Nihon Koku Kagakkai Zasshi 43, 645-648 (in injury, infection or RA (8). It is widely known that RA Japanese) patients suffer TMJ symptoms, although it is rare for such 5. Mizutani, H., Shinozuka, J., Mera, K., Oka, T. and symptoms to include TMJ ankylosis (8). According to Iwata, H. (1985) Rheumatoid arthritis and Ogus, TMJ ankylosis due to RA often occurs in the late temporomandibular joint -its clinical course and stage, because RA is often diagnosed and treated at an early radiographic changes-. Nippon Koku Geka Gakkai stage (9). In the present case, the patient sought treatment Zasshi 31, 2421-2431 (in Japanese) for restriction of mouth opening 41 years after being 6. Kopp, S. (1994) Rheumatoid arthritis. In diagnosed as having RA in the right knee joint. It is Temporomandibular joint and masticatory muscle considered that the condyle showed fibrous adhesion to disorders. 2nd ed., Zarb, G.A., Carlsson, G.E., the mandibular fossa because the TMJ had had little Sessle, B.J. and Mohl, N.D. eds., Munksgaard, movement with only weak jaw opening for a long period. Copenhagen, 346-366 Many RA patients with TMJ involvement can be treated 7.Akerman, S., Jonsson, K., Kopp, S., Petersson, A. effectively by medication, heat, rest, and physiotherapy (15). and Rohlin, M. (1991) Radiologic changes in In general, for treatment of TMJ ankylosis, mobilization temporomandibular, hand, and foot joints of patients of the TMJ is performed. Surgical treatment for ankylosis with rheumatoid arthritis. Oral Surg. Oral Med. can be done by condylectomy, osteoarthrotomy or Oral Pathol. 72, 245-250 arthroplasty (14). In this case, we selected condylectomy, 8. Izumi, Y., Kino, K., Ohmura, Y., Wake, H., Shibuya, and this restored satisfactory masticatory function. T. and Amagasa, T. (1994) Clinico-statistical study of temporomandibular joint ankylosis. Aletiology and References onset age-. Nippon Gakkansetsu Gakkai Zasshi 6, 1. Pindborg, J.J. and Hjorting-Hansen, E. (1974) Atlas 346-359 (in Japanese) of diseases of the jaws. W. B. Saunders Company, 9. Ogus, H. (1975) Rheumatoid arthritis of the Philadelphia, 178-179 temporomandibular joint. Br. J. Oral Surg. 12, 275- 2. AlarcOn, G.S. (1986) Rheumatoid arthritis. In 284 Rheumatology and immunology. 2nd ed., Cohen, 10. Miles, D. (1991) Rheumatoid arthritis. In Oral & A.S. and Bennett, J.C. eds., Grune & Stratton, maxillofacial radiology. Radiologic/pathologic 101

correlations. Miles, D.A., Kaugars,G.E., van Dis, M. 15. Lurie, R., Fisher, J.T. and Lownie, J.F. (1988) and Lovas, J.G.L. eds., W. B. Saunders Company, Temporomandibular joint ankylosis in rheumatoid Philadelphia, 275-277 arthritis. A case report. S. Afr. Med. J. 73, 57-58 11. Larsen, A., Dale, K. and Eek, M. (1977) 16. Seymour, R.L., Crouse, V.L. and Irby, W.B. (1975) Radiographic evaluation of rheumatoid arthritis and Temporomandibular ankylosis secondary to related conditions by standard reference films. Acta rheumatoid arthritis. Report of a case. Oral Surg. Oral Radiol. Diagn. 18, 481-491 Med. Oral Pathol. 40, 584-589 12. Cawson, R.A. and Eveson, J.W. (1987) Oral 17. Sanders, B.and Halliday, R. (1979) Psoriasis and pathology and diagnosis. Colour atlas with integrated rheumatoid arthritis: their relationship in TMJ text. William Heinemann Medical Books, London, ankylosis. J. Oral Med. 34, 4-7 8.16-8.18 18. Smith, H.J., Larheim, T.A. and Aspestrand, F. (1992) 13. Hansson, T.L. (1992) Rheumatoid. In The Rheumatic and nonrheumatic disease in the temporomandibular joint: a biological basis for temporomandibular joint: gadolinium-enhanced clinical practice. 4th ed., Sarnat, B.G. and Laskin, MR imaging. Radiology 185, 229-234 D.M. eds., W. B. Saunders Company, Philadelphia, 19. Larheim, T.A., Bjornland, T., Smith, H.J., 166-167 Aspestrand, F. and Kolbenstvedt, A. (1992) Imaging 14. Imamura, H., Kubota, E., Tanaka, K., Goto, M. and temporomandibular joint abnormalities in patients Katsuki, T. (1993) A case of rheumatoid arthritis with rheumatic disease. Comparison with surgical complicated with TMJ ankylosis. Nippon observations. Oral Surg. Oral Med. Oral Pathol. Gakkansetsu Gakkai Zasshi 5, 465-472 (in Japanese) 73, 494-501