The sequential management of recurrent temporomandibular joint ankylosis in a growing child: a case report
© The Author(s). 2016
Received: 21 June 2016
Accepted: 18 September 2016
Published: 5 October 2016
Temporomandibular joint (TMJ) ankylosis in children often leads to facial deformity, functional deficit, and negative influence of the psychosocial development, which worsens with growth. The treatment of TMJ ankylosis in the pediatric patient is much more challenging than in adults because of a high incidence of recurrence and unfavorable growth of the mandible.
This is a case report describing sequential management of the left TMJ ankylosis resulted from trauma in early childhood. The multiple surgeries including a costochondral graft and gap arthroplasty using interpositional silicone block were performed, but re-ankylosis of the TMJ occurred after surgery. Alloplastic TMJ prosthesis was conducted to prevent another ankylosis, and signs or symptoms of re-ankylosis were not found. Additional reconstruction surgery was performed to compensate mandibular growth after confirming growth completion. During the first 3 years of long-term follow-up, satisfactory functional and esthetic results were observed.
This is to review the sequential management for the recurrent TMJ ankylosis in a growing child. Even though proper healing was expected after reconstruction of the left TMJ with costal cartilage graft, additional surgical interventions, including interpositional arthroplasty, were performed due to re-ankylosis of the affected site. In this case, alloplastic prosthesis could be an option to prevent TMJ re-ankylosis for growing pediatric patients with TMJ ankylosis in the beginning.
Temporomandibular joint (TMJ) ankylosis can be defined as the union of mandibular condyle to the cranial base which is the articular surface through osseous or fibrous tissue, with partial or complete mandibular impediment . The etiological factors for TMJ ankylosis include trauma, rheumatoid arthritis, congenital anomalies, infection, and neoplastic processes. Trauma is well known as the most predominant factor in TMJ ankylosis particularly in children and is associated with inadvertent use of forceps during delivery, traffic accident, and falls [2–4]. When TMJ ankylosis occurs in children, the future growth and development of the jaws and teeth are affected negatively. Furthermore, psychosocial development of the children affected is profoundly influenced due to the obvious facial deformity, which worsens as they grow . Condyle reconstruction is carried out in order to restore TMJ function and facial deformity in adults, whereas high incidence of recurrence and the probable change in the unfavorable growth of the mandible are also needed to be considered in children [5, 6].
It is generally recommended that as soon as the condition is diagnosed, the surgery of TMJ ankylosis should be initiated. The main purpose of the surgery is the re-establishment of joint and harmonious jaw functions in children [5, 7]. This case report presents sequential management of recurrent TMJ ankylosis with a variety of methods in a growing child.
First procedure, 12 years old; costochondral graft
Second procedure, 13 years old; gap arthroplasty with interpositional silicone block
Third procedure, 15 years old; reconstruction with alloplastic condyle
Fourth procedure, 17 years old; reconstruction of mandibular ramus with iliac bone
This case report introduces sequential management of the left TMJ ankylosis resulted from trauma in early childhood. TMJ reconstruction was carried out using costal cartilage graft after removing ankylosed tissues of the left TMJ. The use of costochondral graft is a common practice for condyle reconstruction in children with ankylosis. The advantages of this procedure include biologic and anatomic similarity to the mandibular condyle, growth potential in pediatric patients, ease of harvesting and adapting the graft, and low morbidity of the donor site [7, 9]. Because of the similarities of its primary and secondary cartilages to those of the mandibular condyle , the costochondral graft will provide growth potential and keep pace with the growth of the unaffected side, maintaining mandibular symmetry throughout growth . However, long-term studies on mandibular growth in children with reconstructed TMJs using costochondral grafts show excessive growth on the treated side, occurring in 54 % of the 72 cases evaluated, and only 38 % of the cases presented equal growth with the opposite side, and ankylosis can be expected in rare instances from the recipient site [10–12]. It is recommended that early mobilization and aggressive physiotherapy should be done after releasing the intermaxillary fixation (IMF) and immediately postoperatively for patients reconstructed with the costochondral graft . In this case, there were radiographic and clinical evidences confirming re-ankylosis on the recipient site after 1 year postoperatively and mainly due to the IMF with elastic over 8 weeks after surgery and non-compliance with proper physiotherapy.
Even though proper healing was expected after reconstruction of the left TMJ with costal cartilage graft, additional surgical interventions, including interpositional arthroplasty, were performed due to re-ankylosis of the affected site. There is no consensus in the literature on a standard protocol for management of TMJ ankylosis, but three modalities are commonly used: (1) gap arthroplasty, (2) interpositional arthroplasty, and (3) excision and articular reconstruction . The first modality is performed without intervening grafts or materials and is based on resection of ankylosed bone. According to the literature, a minimum of 15-mm gap is recommended between the recontoured glenoid fossa and the mandible for preventing re-ankylosis [14, 15]. Gap arthroplasty offers an advantage of a simple procedure and requires a short surgical time. However, disadvantages include the following: (1) creation of a pseudoarticulation, (2) a short mandibular ramus with anterior open bite in bilateral cases and posterior open bite in unilateral cases, (3) failure of removal of pathologic bone tissue, and (4) high risk of recurrence [16, 17]. The interpositional arthroplasty is recommended after gap arthroplasty as a means to limit resection and recurrence. In this procedure, autogenous and alloplastic materials are placed in the osteotomized area. The important criteria in the choice of graft or interpositional material are cost, esthetic consequences after graft removal, long-term behavior, risk of infection, biocompatibility, tolerance, and prevention of recurrence . In a comparative study, satisfactory results were observed in 92 % of cases with skin graft  and 83 % of cases with temporal muscle flaps . Among the several alloplastic materials, gold foil, silastic sheet, acrylic, stainless steel, and silicone prostheses have been used [19–21].
Alloplastic temporomandibular joint replacement can provide a viable option for the multiple operated patients with distorted TMJ anatomy or severe anatomical discrepancies involving the TMJ with recurrent ankylosis [22, 23]. Orthopedic surgeons often prefer alloplastic prosthesis in the replacement of joint in similar situations involving other joints over the use of autogenous bone into the area where reactive or heterotropic bone is forming . In this case, alloplastic prosthesis could be a good selection to prevent recurrent TMJ ankylosis in a growing child.
It is proposed that alloplastic prosthesis could be performed to prevent TMJ re-ankylosis for growing pediatric patients with TMJ ankylosis in the beginning. And then there is an additional surgery to compensate mandibular growth after confirming growth completion.
This study received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
J-WC, J-HP, and J-WK are responsible for the data collection, drafting the article, and the critical revision of the article. S-JK is responsible for the conception and design of the study, the critical revision of the article, and the approval of the article. All authors read and approved the final manuscript.
The authors declare that they have no competing interests.
Consent for publication
Written informed consent was obtained from the patient for the publication of this report and any accompanying images.
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