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Ankylosis of the temporomandibular joint as a sequela of septic arthritis and neonatal sepsis

Regev, Eran D.M.D., M.D.; Koplewitz, Benjamin Z. M.D.; Nitzan, Dorit W. D.M.D.; Bar-Ziv, Jacob M.D.

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The Pediatric Infectious Disease Journal: January 2003 - Volume 22 - Issue 1 - p 99-101
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Ankylosis of the temporomandibular joint (TMJ) can be a result of trauma, degenerative changes and infection or of a space-occupying lesion and, when occurring during early childhood, can result in severe functional disability and facial distortion. Septic arthritis of the TMJ is extremely rare in young infants. We report two children with TMJ ankylosis that is believed to have been a consequence of undiagnosed septic arthritis of the TMJ in the neonatal period.

Ankylosis of the temporomandibular joint (TMJ) is a gradually developing condition manifested by decrease in the range of motion of the mandible. It can result from trauma, degenerative changes, infection or a space-occupying lesion. 1 When it occurs during childhood, ankylosis of the TMJ can cause severe functional disability, can result in underdevelopment of one or both sides of the mandible and interfere with the harmony of facial growth.

Septic arthritis in the neonate is one of the consequences of neonatal sepsis and is usually due to local or hematogenous spread. The diagnosis of joint damage in young infants is difficult and is usually made late. 2 Septic arthritis of the TMJ is extremely rare in young infants and has been reported in one case previously. 3

We report two children with TMJ ankylosis that we believe to have been a consequence of an undiagnosed septic arthritis of the TMJ in the neonatal period.

Case 1.

A 6-year-old girl was referred to our clinic because of inability to open her mouth. The girl was supposed to have an operation for what was considered developmental dysplasia of the hip. Limited mouth opening found in the preoperative examination would interfere with intubation and general anesthesia, so she was referred to the Oral and Maxillofacial Surgery clinic for further evaluation. Her parents did not recall any previous facial trauma and considered the limited mouth opening to be a congenital condition.

The girl had been born in a normal delivery at the 37th week of a normal pregnancy after prolonged rupture of the membranes and weighed 1380 g (severe intrauterine growth retardation). There was no history of perinatal trauma. On the 9th day of life she developed sepsis with multifocal abscesses and septic arthritis of the right lower leg, right knee and left elbow. Escherichia coli was grown from the abscesses in the right leg, and Staphylococcus aureus was grown in blood culture. She was treated with intravenous antibiotics and was discharged at the age of 2 months, at which time she weighed 2460 g and appeared well.

On physical examination at the age of 6 years she was small for her age. Her left leg was shorter than the right leg, causing limp. She had facial asymmetry with chin deviation to the right and mandibular retrognathia (Fig. 1). The maximal mouth opening was 1 to 2 mm with posterior crossbite of the right side (Fig. 2).

Fig. 1
Fig. 1:
Frontal and lateral views of the face. Notice the facial asymmetry with chin deviation to the affected (right) side, on the frontal view, and the marked mandibular retrognathia, on the lateral view.
Fig. 2
Fig. 2:
Intraoral view shows the limited mouth opening, the missing upper incisors and posterior crossbite.

Computed tomographic (CT) scan of the TMJ in the axial plane was performed, and coronal, sagittal and three-dimensional reconstructions were obtained. The left TMJ was normal. On the right side the TMJ showed marked deformity of the condylar head, which was larger than on the left side, with flattening and irregularity of the articular surface. The borders of the condylar fossa on the right were irregular and sclerotic. The joint space was narrowed and small bony bars, crossing from the condyle to the condylar fossa, could be identified. The neck of the condyle was shortened, and the coronoid process was hypertrophied and irregular. Radiographs of the pelvis revealed coxa plana magna of the left femoral head, which was in a subluxed superolateral position. The greater trochanter was in a high position. The left femoral epiphysis was irregular, with a small dip in the articular surface.

The diagnosis was right TMJ bony ankylosis for which the patient underwent right TMJ condylectomy, bilateral coronoidectomy and reconstruction of the joint with autogenous costochondral graft.

Case 2.

An 1.5-year-old boy was referred to the Oral and Maxillofacial Surgery clinic by a pediatric surgeon because of limited mouth opening and micrognathia, which were noticed when the boy was examined before surgery for hydrocele. According to his parents he was born “like this” and had not had facial trauma. The neonatology intensive care unit medical records revealed that he was the first of twins, born in the 36th week of gestation, delivered by cesarean section because of twin pregnancy and previous cesarean sections. He weighed 1840 g and Twin B weighed 2800 g. On physical examination after birth, he was in good general condition.

Because of a hematocrit of 75% and hypoglycemia of 38 mg/dl, he was admitted to the neonatology intensive care unit. A sepsis work up was negative. On the sixth day of life, he developed fever of 37.9°C, vomiting and thrombocytopenia of 43 000/mm3. Empiric antibiotic treatment with amikacin and cefotaxime was initiated and changed to vancomycin after blood culture grew S. aureus. Platelet transfusion elevated the count to 300 000/mm3, and partial blood exchange was performed. Follow-up physical examinations were otherwise normal, and the patient recovered well. The child was discharged home at the age of 17 days in good condition weighing 2065 g.

On examination in the Oral and Maxillofacial Surgery clinic at the age of 19 months, the boy was much smaller than his twin brother and was 8 cm shorter. He had mandibular micrognathia and retrognathia. The maximal mouth opening was 15 mm; physical examination was otherwise normal.

The parents reported that the child snored and did not sleep well at night. Polysomnography revealed mild to moderate sleep apnea.

A CT scan of the TMJ disclosed deformed joints, bilaterally, with fibroankylosis. Both mandibular condylar heads were thickened, coarse and irregular, and the condylar fossa was flattened and sclerotic. The joint space was narrowed, and there was ankylosis along the posteromedial surface of the condylar head. With the diagnosis of sleep apnea secondary to micrognathia, he was scheduled for mandibular elongation by means of distraction osteogenesis. One week before the surgery, the child was examined in the clinic after he had fallen and broken his left clavicle while playing. A radiograph showed a left clavicular fracture and deformity of the head of the left humerus. The humeral head was underdeveloped and the humeral diaphysis was frayed and irregular.

Discussion.

Ankylosis of the TMJ as a result of infection is a well-known phenomenon. In his review of the literature, Topazian 4 found that infection was the etiology of TMJ ankylosis in as many as 68% of cases. Septic arthritis of the TMJ may result from direct spread from adjacent infections as in otitis media or externa, mastoiditis or parotitis. It can also develop after hematogenous spread.

We found only one reported case in the literature with hypomobility of the mandible as a consequence of neonatal sepsis, without, however, radiographic evidence of ankylosis. 3 In that case the infection could have spread directly from the parotid gland that was infected as well.

In our cases there was no history of perinatal or childhood trauma, but this could not be completely excluded. However, the radiographic appearance of TMJ ankylosis as a result of trauma is typical. The condylar head is pulled medially, anteriorly and inferiorly and remains in the new location beside the stump, which fuses to the base of the skull. The presence of an anteromedially dislocated condylar head is pathognomonic for ankylosis after trauma. 5 The CT scan findings in our cases, however, disclosed a different picture. The condyle and the condylar fossa showed marked irregularity and sclerosis, and there was joint space narrowing of the TMJ, with shortening and thickening of the condylar neck. These findings are not characteristic of trauma. In the first case the pelvic radiograph revealed findings with striking similarity; the femoral head was also irregular, shortened and thickened, creating a deformity that has previously been termed “the double headed femur.”6 This deformity is commonly a sequela of suppurative hip arthritis. In the second case the left humeral head showed marked underdevelopment with diaphysial irregularity and sclerosis. In both cases there was marked similarity between the CT findings of the condylar deformity and the radiographic findings of the other large joint deformities. The coexistence of these deformities, in conjunction with the history of neonatal sepsis (and no known previous trauma), has thus lead to the assumption that both deformities could be the result of the same infectious process.

The delayed diagnosis in these cases can be attributed to several reasons. The limited need for function of the joint in infants and young children makes the initial insult pass unnoticed. The child grows with a disability that does not significantly interfere with sucking and swallowing liquids. Because the ankylosis develops gradually over months or years, the facial deformity increases slowly until it is noticed only later in childhood.

In a manner similar to that of the hip, shoulder, knee and elbow, the TMJ can also be affected by neonatal sepsis and septic arthritis. In cases of neonatal sepsis, attention should be paid to the function of the TMJ, as well as the other joints. The crucial role of the TMJ in facial growth raises the need for specific attention of the pediatrician and the parents to the jaw range of motion in the growing child. Early diagnosis and intervention might prevent long term sequelae and decrease morbidity.

1. Kaban LB. Acquired abnormalities of the temporomandibular joint. In: Kaban LB, ed. Pediatric oral and maxillofacial surgery. Philadelphia: Saunders, 1990: 307–41.
2. Paterson DC. Acute suppurative arthritis in infancy and childhood. J Bone Joint Surg 1970; 52B: 474–82.
3. Kasznia-Kocot J, Cichos B, Sada-Cieslar M, Buszman Z. Staphylococcal septicemia in a newborn infant with multiple organ involvement (in Polish). Wiad-Lek 1990; 43: 301–4.
4. Topazian RG. Infectious arthritis of the temporomandibular joint. In: Topazian RG, Goldberg NH, eds. Oral and maxillofacial infections. Philadelphia: Saunders, 1987: 336–7.
5. Nitzan WD, Bar-Ziv J, Shteyer A. Surgical management of temporomandibular joint ankylosis type III by retaining the displaced condyle and disc. J Oral Maxillofac Surg 1998; 56: 1133–8.
6. Torok G, Bar-Ziv J. The double headed femur. Orthop Rev 1980; 9: 41–9.
Keywords:

Temporomandibular joint; ankylosis; neonatal sepsis; septic arthritis

© 2003 Lippincott Williams & Wilkins, Inc.