|DENTAL SCIENCE - CASE REPORT
|Year : 2015 | Volume
| Issue : 6 | Page : 687-690
Management of severe sleep apnea secondary to juvenile arthritis with temporomandibular joint replacement and mandibular advancement
S Arun Paul1, S Sibu Simon1, Barney Issac2, Saurav Kumar1
1 Dental and Oral Surgery Unit-1, Christian Medical College and Hospital, Vellore, Tamil Nadu, India
2 General Surgery, Head and Neck, Christian Medical College and Hospital, Vellore, Tamil Nadu, India
|Date of Submission||28-Apr-2015|
|Date of Decision||28-Apr-2015|
|Date of Acceptance||22-May-2015|
|Date of Web Publication||1-Sep-2015|
S Arun Paul
Dental and Oral Surgery Unit-1, Christian Medical College and Hospital, Vellore, Tamil Nadu
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Variations affecting the growth centers can severely affect the normal formation and subsequent function of vital musculoskeletal structures. We report a case of bilateral condylar atrophy with a history of juvenile arthritis (JA) resulting in progressive obstructive sleep apnea (OSA) in adulthood. In addition to this, the case report emphasizes the role of temporomandibular joint replacement and advancement of the mandible to correct progressive OSA secondary to idiopathic JA. Computed tomography revealed micrognathia, condylar hypoplasia, and decreased pharyngeal airway space. The resultant increase in the retrolingual-pharyngeal airway space following the surgery, helped to completely resolve the presenting symptoms. It is hoped that the described technique could be used in similar cases with a predictable outcome.
Keywords: Condylar analogue, condylar atrophy, juvenile arthritis, mandibular advancement, obstructive sleep apnea, temporomandibular joint replacement
|How to cite this article:|
Paul S A, Simon S S, Issac B, Kumar S. Management of severe sleep apnea secondary to juvenile arthritis with temporomandibular joint replacement and mandibular advancement. J Pharm Bioall Sci 2015;7, Suppl S2:687-90
|How to cite this URL:|
Paul S A, Simon S S, Issac B, Kumar S. Management of severe sleep apnea secondary to juvenile arthritis with temporomandibular joint replacement and mandibular advancement. J Pharm Bioall Sci [serial online] 2015 [cited 2019 Aug 22];7, Suppl S2:687-90. Available from: http://www.jpbsonline.org/text.asp?2015/7/6/687/163479
Juvenile arthritis (JA) with sleep apnea in the adult group is a rarely reported phenomenon. Obstructive sleep apnea syndrome (OSAS) is a sleep disorder characterized by abnormal pauses in breathing (apnea) or instances of abnormally low breathing (hypopnea) during sleep, resulting in excessive daytime somnolence and lethargy on a short term basis.  On a long term, OSAS is associated with diabetes mellitus, hypertension, coronary artery disease, and cerebrovascular accidents.  The severity of sleep apnea is assessed using clinical questionnaires (e.g. Epworth sleepiness scale), radiographic analysis (e.g. Cephalogram) and polysomnography.  In this modern era, there are several methods employed for treating obstructive sleep apnea (OSA). The cause of sleep apnea determines the treatment method. OSAS can occur as a consequence of mandibular deformity and temporomandibular joint (TMJ) destruction occurring in JA.  JA is an autoimmune disease which is not fully understood till date. Rheumatoid factor is usually negative in this condition and this leads to the appellation "idiopathic JA". The notable clinical feature is persistent swelling of the affected joints, which commonly include the knee, ankle, wrist and small joints of the hands and feet. Swellings may be difficult to detect clinically, especially for joints such as those of the spine, sacroiliac joints, shoulder, hip, and jaw, where imaging techniques such as bone scan, ultrasound, and magnetic resonance imaging are very useful.  Our case report emphasizes one of such rare findings where bilateral condylar agenesis in a patient with JA resulted in progressive OSA in adulthood.
| Case Report|| |
A 29-year-old male reported to the Department of Dental Surgery, CMCH, Vellore, Tamil Nadu, India, with an inability to sleep for the since the past 1-year with a reduced mouth opening from childhood. He also complained about his bird-like facial appearance. A detailed history revealed that he was diagnosed with JA and was managed with methotrexate for a year, before 5 years. He had also been diagnosed with severe sleep apnea syndrome and had an Epworth sleepiness scale of 24/24. On examination, he was found to have a retrognathic mandible, proclined anteriors, decreased chin-neck angle (Andy Gump deformity), hypotonic/hypoplastic lower facial musculature, increased overjet and overbite, deranged occlusion with retruded chin, and a mouth opening of 16 mm [Figure 1] and [Figure 2]. Polysomnography was done to assess the degree of sleep apnea and a bone isotope scan was needed to evaluate residual TMJ arthritis. A computed tomography (CT) scan showed the extent of mandibular deformity which was resulting in the mechanical obstruction to breathing [Figure 3]. Bone isotope scan revealed the absence of arthritis in the TMJ and the rest of the skeleton, based on tracer uptake.
A three-dimensional CT revealed the absence of both the condyles. Condylar atrophy secondary to JA had resulted in a decrease in the mandibular ramal height as well as the length of the body [Figure 4]. On the basis of the presentation, a dual stage surgery was planned wherein a pair of titanium condylar analogues would be used to replace the missing condyles followed by a mandibular advancement at a later period. The Stage I (Condylar analogue) surgery was expected to improve the mouth opening as well as provide a posterior stop to the ramus (proximal segment) while advancing the distal segment of the mandible anteriorly in the second stage. Before surgical intervention, appropriate clearance was obtained from rheumatology, pulmonary medicine, and preanesthetic clinic.
|Figure 4: Preoperative three-dimensional view of right and left ramus of the mandible|
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| Treatment Phase 1|| |
A pair of titanium condylar analogue was used to reconstruct bilateral TMJ [Figure 5]. Following the surgery, the mouth opening improved from 16 mm to 35 mm. Postoperatively, a splint was fabricated to guide the mandible into the established occlusion. The patient reported a better sleep rhythm and had a fairly uninterrupted sleep. This was corroborated by his parents who observed improvement in his sleep pattern. After 4 weeks, Epworth sleepiness scale showed marginal improvement from 24/24 to 16/24, but was not satisfactory as the polysomnography revealed an apnea-hypopnea index (AHI) of 47.8/h, with a desaturation index (DSI) of 50.7/h indicating persistence of severe sleep apnea status.
| Treatment Phase 2|| |
In this phase, the body of the mandible was advanced by 10 mm. The body osteotomy was performed anterior to the mental foramen and fixed using titanium reconstruction plate with inter-positioning iliac graft bilaterally [Figure 6]. Postoperatively the patient was very comfortable with his sleep pattern and very much satisfied with his facial appearance because he was no longer a "chinless wonder" [Figure 7].
The postoperative Cephalogram confirmed the skeletal changes which were evident clinically. There was also an increase in the retrolingual-pharyngeal space (between the back of the tongue and posterior pharyngeal wall) from 2.7 mm to 10.2 mm, which is an index of the level of respiratory obstruction. Orthopantomogram showed intact plates and screws and condyles in position [Figure 8]. Epworth sleepiness scale had declined to normal limits and was 2/24. A polysomnography revealed marked improvement with an AHI of 7/h and DSI of 6/h, indicating only mild OSAS. A prosthodontic rehabilitation was done after 6 weeks. He had no complaints of sleep disturbance henceforth.
| Discussion|| |
Obstructive sleep apnea is a growing public health concern because it can cause hypertension, cardiac arrhythmias, stroke, and in rare circumstances, sudden death if untreated. Although nasal continuous positive airway pressure therapy is considered the first-line treatment of OSA, surgery has been shown to be a valid option for patients who are intolerant to positive pressure therapy.  Several surgical approaches have been recommended for the treatment of OSA.  Orthognathic surgery for repositioning the mandible and the genial tubercles for forward positioning of genioglossus has been recommended as a successful method of increasing the post glottic space and reducing the respiratory obstruction during sleep.  Other nonsurgical methods have included weight loss and mandibular repositioning devices. 
The use of orthognathic surgery to treat OSAS began toward the end of the 1970s when mandibular surgery was reported to have reversed the symptoms of sleep apnea.  Advancement of the mandible and maxilla increase the available tongue space and have a positive influence on the pharyngeal airway.  Kawamato seems to have pioneered the maxillomandibular advancement technique for treating OSAS.  Maxillomandibular advancement decreases the collapsibility of the velopharyngeal and suprahyoid musculature and improves lateral pharyngeal wall collapse, all of which have been shown to be significant components contributing to the upper airway obstruction in OSA.  The outcome of maxillomandibular advancement has been extensively reported, with success rates of 57% to 100%. 
The cephalometric radiograph, although rarely used by other specialties, is very valuable because it demonstrates both bony and soft tissue abnormalities related to the soft palate, pharyngeal airway, and maxillomandibular complex. If the radiographic and clinical examinations of the soft tissues of the pharynx reveal a narrow airway in conjunction with retrognathia of the maxilla and mandible, the patient should be considered a candidate for MMA surgery. Because an underlying skeletal abnormality is sometimes hard to detect clinically, it is reasonable for an oral and maxillofacial surgeon to assess these patients. The surgeon's goal is to optimize the advancement of the deficient structures while maintaining a normal facial balance for each patient. It is, therefore, beneficial to use a cephalometric analysis, which will clearly demonstrate all the maxillofacial abnormalities and provide a visual treatment objective.  Cephalometric analysis provides a reliable method of evaluating the changes in the tongue base before and after surgery. It is estimated that for each millimeter of maxillary and mandibular bone advancement there was a 0.76 mm increase in the retropalatal region and a 1.2 mm increase in the pharynx in the retrolingual region, respectively.  Bilateral TM joint reconstruction has been recommended for the replacement of the joint for the restoration of function and also for elongation of the shortened mandible in joint destructive diseases like Juvenile rheumatoid arthritis. The use of custom made two part total joint reconstruction of the cranial articular surface as well as the condylar head has been advocated. 
In this case, condylar analogue prosthesis was fixed to the residual stump of the vertical mandibular ramus. Consequently, the length of the vertical ramus could be increased by 6 mm, which could be maintained by a splint and intermaxillary traction. The mandible with the new condyles was made to function after a period of 1-month of traction. The patient was able to open his mouth more than before and the full range of mandibular movements was possible, proving that the condylar prostheses were functioning well.
Further increase in length of the mandible could be achieved (10 mm) through the second procedure for a single stage mandibular advancement with bone grafting and rigid plating. The forward repositioning of the chin enabled the posterior pharyngeal space to be enlarged in the anteroposterior direction by 7.5 mm, as measured cephalometrically. This resulted not only in a cosmetically pleasing facial appearance but a significant relief from the OSA. Review of the patient over a period of 10 months has shown that the mandibular advancement procedure had a stable outcome. Through the management of the described case it's our observation that OSA secondary to JA could be effectively managed with condylar replacement and mandibular advancement to produce a predictable outcome. However, early identification, diagnosis, and treatment planning could alleviate the agony manifested by these conditions.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Paiva T, Attarian H. Obstructive sleep apnea and other sleep-related syndromes. Handb Clin Neurol 2014;119:251-71.
Rudra A, Ray M, Sengupta S, Iqbal A, Maitra G, Chatterjee S. Obstructive sleep apnoea syndrome in children and anaesthesia. Indian J Anaesth 2010;54:18-23.
Silva GE, Vana KD, Goodwin JL, Sherrill DL, Quan SF. Identification of patients with sleep disordered breathing: Comparing the four-variable screening tool, STOP, STOP-Bang, and Epworth Sleepiness Scales. J Clin Sleep Med 2011;7:467-72.
Susarla SM, Abramson ZR, Dodson TB, Kaban LB. Upper airway length decreases after maxillomandibular advancement in patients with obstructive sleep apnea. J Oral Maxillofac Surg 2011;69:2872-8.
Munir S, Patil K, Miller E, Uleryk E, Twilt M, Spiegel L, et al
. Juvenile idiopathic arthritis of the axial joints: A systematic review of the diagnostic accuracy and predictive value of conventional MRI. AJR Am J Roentgenol 2014;202:199-210.
Hu ST, Yu CC, Lee PS, Tsao LI. Life experiences among obstructive sleep apnoea patients receiving continuous positive airway pressure therapy. J Clin Nurs 2014;23:268-78.
Laganà F, Sacco R, Giannì AB. Maxillomandibular advancement in obstructive sleep apnea syndrome. Eur J Intern Med 2013;24:e94-5.
Dort L, Remmers J. A combination appliance for obstructive sleep apnea: The effectiveness of mandibular advancement and tongue retention. J Clin Sleep Med 2012;8:265-9.
Barewal RM, Hagen CC. Management of snoring and obstructive sleep apnea with mandibular repositioning appliances: A prosthodontic approach. Dent Clin North Am 2014;58:159-80.
James D, Ma L. Mandibular reconstruction in children with obstructive sleep apnea due to micrognathia. Plast Reconstr Surg 1997;100:1131-7.
Goodday R. Diagnosis, treatment planning, and surgical correction of obstructive sleep apnea. J Oral Maxillofac Surg 2009;67:2183-96.
Andrews BT, Lakin GE, Bradley JP, Kawamoto HK Jr. Orthognathic surgery for obstructive sleep apnea: Applying the principles to new horizons in craniofacial surgery. J Craniofac Surg 2012;23:2028-41.
Li KK. Maxillomandibular advancement for obstructive sleep apnea. J Oral Maxillofac Surg 2011;69:687-94.
Faria AC, Xavier SP, Silva SN Jr, Trawitzki LV, de Mello-Filho FV. Cephalometric analysis of modifications of the pharynx due to maxillo-mandibular advancement surgery in patients with obstructive sleep apnea. Int J Oral Maxillofac Surg 2013;42:579-84.
Wolford LM, Mehra P. Custom-made total joint prostheses for temporomandibular joint reconstruction. Baylor University Medical Center Proceedings 2000;13:135-8.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]