Int. J. Oral Maxillofac. Surg.

2022; 51: 799–805

https://doi.org/10.1016/j.ijom.2021.11.005, available online at https://www.sciencedirect.com

Clinical Paper
Orthognathic Surgery and TMJ Disorders

Orthognathic surgery for M. Raffaini1, F. Arcuri2

1
Face Surgery Centre, Parma, Italy; 2Unit of
Maxillofacial Surgery, IRCCS ‘‘Policlinico San
Martino’’, Genoa, Italy

juvenile idiopathic arthritis of the

temporomandibular joint: a
critical reappraisal based on
surgical experience
M. Raffaini, F. Arcuri: Orthognathic surgery for juvenile idiopathic arthritis of the
temporomandibular joint: a critical reappraisal based on surgical experience. Int. J.
Oral Maxillofac. Surg. 2022; 51: 799–805. ã 2021 International Association of Oral
and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.

Abstract. Juvenile idiopathic arthritis (JIA) involving the temporomandibular joint

(TMJ) can result in significant dentofacial deformities that may require orthognathic
surgical correction. The aim of this study was to assess the functional and aesthetic
results relative to stability after bimaxillary surgery with counterclockwise rotation
of the occlusal plane in patients with JIA. A retrospective chart review was
conducted of all patients affected by JIA who underwent orthognathic surgery
between January 2000 and December 2019 at the Face Surgery Centre (Parma,
Italy). Patient records were evaluated for surgical indications, complications, and
outcomes. The final study sample included 13 patients (12 female, one male). The
mean age of the patients was 18.6 years (range 17–26 years) at the time of surgery;
12 patients had bilateral TMJ disease. At the 1-year follow-up, all patients except
one had a stable occlusion with a natural, well-balanced morphology of the face and
Key words: juvenile idiopathic arthritis; dento-
adequate dynamic excursion of the mandible. The 1-year postoperative cone beam
facial deformities; orthognathic surgery; man-
computed tomography (CBCT) scan revealed complete ossification at all osteotomy dibular advancement; mandibular osteotomy.
sites. Bilateral sagittal split osteotomy with mandibular advancement is an effective
procedure with a low rate of complications for patients with JIA with stable disease Accepted for publication 1 November 2021
confirmed by preoperative CBCT or magnetic resonance imaging. Available online 21 November 2021

Worldwide, the incidence of juvenile idi- involved in 10–80% of cases of JIA2. temic, pauciarticular, and polyarticular.
opathic arthritis (JIA) varies from 0.07 to The cause of JIA is unknown, and multi- The American College of Rheumatology
4.01 cases per 1000 children annually1. factorial causes have been suggested. JIA defines JIA by the age of onset (<16 years)
The temporomandibular joint (TMJ) is is divided into three subcategories: sys- and duration of the disease (>6 weeks)3.

0901-5027/060799 + 07 ã 2021 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.
800 Raffaini and Arcuri

The diagnosis of JIA is based on the before surgery; bimaxillary surgery with variables: mean, minimum, maximum,
physical finding of arthritis (at least one or without genioplasty augmentation; a and median for continuous variables,
joint) that has lasted for at least 6 weeks, in mandible-first protocol with CCWR of and absolute and relative frequencies (per-
a patient younger than 16 years of age, the occlusal plane; non-smoking patient; centages) for qualitative variables. The
with other causes excluded. Arthritis is third molars removed at least 6 months statistical analysis was performed using
defined as either intra-articular swelling before surgery; stable mandibular resorp- IBM SPSS Statistics for Windows version
or as a limitation of joint motion in asso- tion at the time of surgery; available pre- 22.0 (IBM Corp., Armonk, NY, USA) and
ciation with warmth, pain, and erythema operative clinical data; postoperative the analysis of variance (ANOVA) test. A
of the joint; the disease may be transient or follow-up period of at least 1 year; and P-value <0.05 was considered significant.
chronic4. preoperative and 1-year postoperative
Typical features of patients affected by cone beam computed tomography
Results
bilateral TMJ involvement include de- (CBCT) scans and/or MRI scans available.
creased ramus height and posterior facial The following exclusion criteria were Sixteen patients affected by JIA under-
height, changes in condylar shape, and applied: treatment with surgical approaches went a combined orthodontic–orthog-
class II malocclusion with anterior open and techniques other than bimaxillary sur- nathic surgical approach at the Face
bite due to clockwise rotation of the man- gery with CCWR; insufficient preoperative Surgery Centre (Parma, Italy) during the
dible. These patients complain of difficul- and postoperative clinical data; presence of study period, at skeletal maturity. Three
ty chewing and aesthetic concerns. a syndrome other than JIA; and absence of patients were excluded due to a lack of
Moreover, a reduction in the posterior preoperative or 1-year imaging. adequate records. The final study sample
airway space is associated with obstruc- The patient records were evaluated for included 13 patients (12 female, one
tive sleep apnoea (OSA) and related surgical indications, complications, and male). The mean age of the patients was
comorbidities. In imaging studies (mag- outcomes. The condition of the condyles 18.6 years (range 17–26 years) at the time
netic resonance imaging (MRI)/computed was analysed radiologically by superim- of surgery, with a mean age of 15.1 years
tomography (CT)), more than 90% of posing the CBCT/MRI obtained immedi- (range 11–19 years) at the time of diagno-
patients with JIA are diagnosed with ately before orthodontic therapy with the sis of JIA. Twelve patients had bilateral
TMJ changes5,6. CBCT/MRI obtained immediately before TMJ disease. Preoperatively, all patients
The surgical management of dentofa- surgery to confirm the remission of the were diagnosed radiographically with
cial deformity due to condylar resorption disease (absence of progression of condy- resorption of the condyle and reduced
remains controversial, ranging from cost- lar resorption after a mean interval of 15 ramus height with CBCT (13 patients)
ochondral grafts (CCG)/TMJ prosthesis months). Patients underwent surgery un- and MRI (seven patients) before orthodon-
substitution to orthognathic surgery. Indi- der general anaesthesia and nasotracheal tic therapy and another CBCT/MRI im-
cations for surgery include the correction intubation. Mandibular advancement was mediately before surgery.
of dentofacial deformities, improvement performed first (mandible-first protocol) All patients were administered anti-
of TMJ function, sleep apnoea treatment/ in accordance with the method reported TNF-a medication (etanercept) for at
prevention with airway expansion, and by Raffaini and Wolford11,12. least 1 year before surgery at a standard
correction of aesthetic concerns7–10. In the superimposition of the CBCT dosage of 25 mg or 50 mg subcutaneous
The aim of this study was to assess images (i-CAT Vision Q 1.8.0.5; Imaging injection per week. Other drugs were used
functional and aesthetic results with a Sciences International, Hatfield, PA, in different therapeutic schemes according
focus on stability after bimaxillary surgery USA), the pogonion point (Pog) obtained to the severity and characteristics of the
with counterclockwise rotation (CCWR) preoperatively (T0), at 12 months postop- disease: tetracyclines (doxycycline, 100
of the occlusal plane in JIA patients with eratively (T1), and at the last follow-up mg per day), omega-3 fatty acids, non-
severe condylar resorption. (T2) was used to evaluate the stability of steroidal anti-inflammatory drugs, metho-
mandibular advancement. The CBCT trexate, tocilizumab, antioxidants (vitamin
images were collected in DICOM format C, E, and D3), antidepressants such as
Materials and methods
(Digital Imaging and Communications in amitriptyline used at a low dosage to
A retrospective single-centre chart review Medicine) and processed with three-di- obtain a muscle relaxant effect (4–5 mg
was conducted of all patients affected by mensional orthognathic surgery planning per day before night sleeping), and calci-
JIA who underwent orthognathic surgery software (NemoFab; Nemotec, Madrid, um supplementation. Postoperatively, all
between January 2000 and December Spain). Variations in this parameter were patients were continued on treatment with
2019 at the Face Surgery Centre (Parma, determined at two different intervals: T0 medication depending on the rheumatolo-
Italy). All surgeries were performed by a to T1 (DT1, mandibular advancement) gy consultation.
senior surgeon (M.R.). and T1 to T2 (DT2, postoperative relapse). All procedures were performed when
Written informed consent was obtained Follow-up was performed at 1, 2, 4 and 6 the progressive condylar resorption had
from all patients for the publication of data weeks, 2 months, 6 months, and 1 year stabilized after preoperative medical ther-
and images. All procedures were per- postoperatively, and then annually. During apy and CBCT/MRI (sometimes the con-
formed in accordance with the ethical the preoperative and postoperative follow- dyle had disappeared completely and the
standards of the 1964 Declaration of Hel- up examinations, all patients underwent glenoid cavity was also totally uninhabit-
sinki and those of the study institution. panoramic radiography, a CBCT scan, ed, with the neojoint in front of the articu-
This study was exempted by the Regione and facial photograph documentation. lar tubercle). The mean preoperative oral
Lombardia Ethics Committee. opening was 37.1 mm (range 32–46 mm).
Patient inclusion criteria were as fol- All patients underwent advancement
Statistical analysis
lows: JIA affecting the TMJ; anti-tumour bilateral sagittal split osteotomy (BSSO)
necrosis factor alpha (anti-TNF-a) medi- A descriptive analysis was used for the plus Le Fort I osteotomy (posteriorly
cation (etanercept) for at least 1 year most relevant statistics for all analysed down-grafted); 11 of the patients
 Orthognathic surgery for JIA of the TMJ 801

(84.6%) had an advancement genioplasty ceived regular medical therapy). The 1- indicated when TMJ inflammation is sus-
procedure. All patients had CCWR of the year postoperative CBCT scan revealed pected15.
occlusal plane13, with a mean value of 7.1 full ossification of all osteotomy sites. No There are two main conceptual opera-
(range 6–11 ); the mean advancement at exacerbation of the disease was documen- tive techniques for the correction of den-
pogonion was 20.4 mm (range 16–26 ted postoperatively, either on CBCT or tofacial deformities caused by JIA: TMJ
mm). The postoperative stability of the MRI (absence of sclerosis of the remain- reconstruction (resection of the remaining
mandible was evaluated by comparing ing condylar stumps and absence of osteo- TMJ and reconstruction with an autolo-
the results obtained at T1 and T2. Regard- phytes). Examples of a patient case are gous CCG or alloplastic prosthesis) and
ing backward movement of pogonion, the given in Figs 1–4. TMJ preservation (orthognathic surgery
mean relapse was 0.33  1.51. and distraction osteogenesis). The scien-
Surgery was uneventful in all patients. tific literature in this respect is controver-
No infections occurred in any of the cases, Discussion sial, with no clear guidelines. The number
even though all patients were on an im- From a medical point of view, drugs pre- of treated patients is limited, and thus no
munosuppressive regimen. The mean du- scribed in patients with JIA reduce pain, evidence-based treatment strategies are
ration of the procedure was 109.6 min, stiffness, and swelling of the TMJ while available for these patients16–20.
ranging from 100 to 165 min. The mean preventing joint damage. Most patients Leshem et al.2 described successful
hospital stay was 1.1 days, ranging from 1 often require a targeted cocktail of drugs. orthognathic surgery in eight patients af-
to 2 days. Etanercept is one of the most frequently fected by JIA, with a mean relapse of 2.1
All patients underwent pre-surgical and adopted medications for acute and chronic mm and a mean mandibular advancement
postoperative orthodontic therapy. The JIA. It is a potent TNF-a inhibitor that of 9.6 mm at 1 year postoperatively. Kahn-
mean postoperative oral opening was requires long-term administration to pre- berg and Holmstrom17 described orthog-
39.3 mm (range 39–48 mm). The mean vent relapse; however, the most common nathic surgery with chin augmentation in
follow-up period was 5.1 years (range 2– side effects are demyelinating and haema- 19 of 37 patients (three with JIA). They
18 years). The major complication (two tological diseases14. reported a low rate of complications (two
cases) was a delay in bone consolidation at Tetracyclines are cellular inhibitors of postoperative dehiscence and one infec-
the mandibular osteotomy with some per- matrix metalloproteinases (MMPs), which tion of the implant).
sistent mobility; consolidation occurred break down connective tissue, resulting in Pagnoni et al.18 described the successful
on its own after 6 months (the application decreased osteoclast activity and reduced treatment of five cases of quiescent JIA
of orthodontic intermaxillary elastics was bone and cartilage resorption5. Omega-3 with Le Fort I osteotomy and genioplasty,
sufficient to maintain the occlusion). fatty acids found in fish oils provide pros- without mandibular osteotomy, to avoid
At the 1-year follow-up, all patients tanoids and leukotrienes, which exhibit later condylar resorption with pain and
except one had a stable occlusion with a anti-inflammatory properties. The phar- functional impairment of the TMJ.
natural, well-balanced morphology of the macology of non-steroidal anti-inflamma- According to the authors, the occlusion
face and adequate dynamic excursion of tory drugs is complex. Through multiple and skeletal movements were stable at 8
the mandible. In one case (7.7%), there biological effects, they protect bone and months postoperatively, with relief of
was a mild recurrence of the class II cartilage from breakdown and diminish TMJ pain, headache, and sleep respiratory
deformity due to bone resorption at the pain and TMJ effusion. Based on current distress. However, the average advance-
condylar level (the patient had not re- information, 20 mg of daily piroxicam is ment at point A in these patients was small

Fig. 1. Photographs of patient 1 on presentation: (A) frontal, (B) lateral, (C) occlusion.
802 Raffaini and Arcuri

Fig. 2. Preoperative imaging of patient 1: (A) panoramic radiograph, (B) teleradiography, (C) CT scan.

(4 mm), and many studies have indicated the potential for overgrowth and the with an increased risk of mechanical fail-
successful treatment of JIA with mandib- need for an additional orthognathic pro- ure requiring reoperation or replacement
ular osteotomies without postoperative cedure. Moreover, the CCG carries the of a prosthesis24–27.
complications. risk of graft resorption and bone anky- Mehra et al.25 described 15 successful
Oye et al.19 described orthognathic sur- losis, and it is associated with donor site TMJ reconstructions using custom-made
gery for 16 patients affected by JIA, with a morbidity such as pneumothorax and total joint prostheses with simultaneous
mean skeletal relapse of 2.3 mm and an scarring21–23. genioplasty and maxillary surgery; how-
average mandibular advancement of 5.3 Svensson and Adell22 recommended ever, follow-up was short (34.3 months)
mm. The authors concluded that orthog- surgical treatment using CCGs and a sec- and there was an inconsistent increase in
nathic surgery reduces pain and is the ond-stage orthognathic operation. They mouth opening and a postoperative de-
treatment of choice for JIA of the TMJ. followed 12 patients affected by JIA for crease in lateral excursion. Although the
Nørholt et al.8 followed a group of 23 a mean of 5.3 years after CCG and authors reported good results, several
patients with JIA after distraction osteo- reported asymmetric mandibular over- issues need to be addressed before taking
genesis, with or without a maxillary growth in eight cases. Guyuron23 de- this method into consideration. A survey
osteotomy. Although they found predict- scribed skeletal relapse of 2 mm at 1 of American Society of Temporomandib-
able correction of asymmetry at 1 year year after CCG reconstruction and BSSO ular Joint Surgeons members found that
postoperatively, postoperative condylar of the mandible and genioplasty. complications and failure rates ranged
translation was not significantly im- Some authors have advocated an allo- between 4.97% and 8.22%26.
proved when compared to the preopera- plastic prosthesis in patients with JIA, According to the orthopaedic literature,
tive condition; moreover, two patients reporting stable long-term results and ear- a study describing the long-term (up to 27
developed permanent dysesthesia of the ly mastication. However, the long-term years) outcomes of total knee arthroplas-
lower lip. outcome of alloplastic prostheses beyond ties in patients <60 years of age with
There is controversy in the scientific 15 years is unknown. Moreover, patients osteoarthritis, reported that the alloplastic
literature regarding the use of the CCG who have not reached skeletal maturity are prosthesis needs to be revised during fol-
for patients with JIA, primarily due to expected to have a long life-expectancy, low-up28. In our opinion, the indication for
 Orthognathic surgery for JIA of the TMJ 803

Fig. 3. Patient 1 at the 1-year follow-up after bimaxillary surgery: (A) frontal, (B) lateral, (C) panoramic radiograph.

TMJ reconstruction in patients with JIA is in 12 of the 13 patients, without evident Funding
severely restricted to TMJ ankylosis with skeletal relapse measured at pogonion.
The authors have nothing to disclose. No
limited mouth opening and failure of pre- Mean mouth opening increased from
funding was received for this article.
vious orthognathic surgery in adult 37.1 mm to 39.3 mm due to a consistent
patients with persistent pain and an unsta- elongation of the whole mandible, which
ble occlusion. expresses itself in an augmentation of
The cases of 13 patients with JIA who the radius of rotation. Competing interests
underwent bimaxillary surgery with rel- Orthognathic treatment of patients with
None.
evant CCWR of the occlusal plane and JIA needs to be performed once the dis-
with preoperative and postoperative ease is in remission and the condyles are in
fixed orthodontic appliances are reported a stable condition. Remission or control of
here. Eleven of these 13 patients under- the disease should be verified by preoper- Ethical approval
went an advancement genioplasty pro- ative CBCT/MRI, which in the cases pre-
This study was exempted by the Regione
cedure. None of the patients had sented here demonstrated preoperative
Lombardia Ethics Committee. This study
additional prosthetic reconstruction of stability of the condyle.
was conducted in accordance with the
the TMJ or mandibular ramus distrac- In conclusion, BSSO with mandibular
current revision of the Declaration of Hel-
tion. All patients were administered an- advancement is an effective procedure
sinki. Current standards of scientific re-
tirheumatic medications pre- and with a low rate of complications for
search ethics were applied in the
postoperatively. At the end of the fol- patients with JIA with stable disease con-
performance of this retrospective study.
low-up period, the occlusion was stable firmed by preoperative CBCT/MRI.
804 Raffaini and Arcuri

Fig. 4. Patient 1 at the 8-year follow-up: (A) frontal, (B) lateral, (C) occlusion, (D) panoramic radiograph.

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