Vol. 117 No.

5 May 2014

Treatment management of children with juvenile idiopathic

arthritis with temporomandibular joint involvement: a systematic
review
Emma C. te Veldhuis, DDS, Alwine H. te Veldhuis, DDS, and Maarten J. Koudstaal, MD, DDS, PhD
Erasmus University Medical Centre Rotterdam, Rotterdam, The Netherlands

Objective. This study aimed to systematically review the available literature on the treatment of patients with juvenile
idiopathic arthritis (JIA) with temporomandibular joint (TMJ) involvement.
Study Design. According to the PRISMA statement (Preferred Reporting Items for Systematic Reviews and Meta-Analyses)
guidelines, studies were included until August 2012.
Results. A total of 40 articles were identified. TMJ involvement in patients with JIA varies between 17% and 87%. The mean
age at diagnosis of JIA is 7.2 years. TMJ treatment can be divided into 2 main groups, an arthritis group and a dentofacial
deformity group. The main treatment modalities are counseling, pharmaceutical interventions, physiotherapy, orthodontic
treatment, surgery, or a combination of the aforementioned therapies.
Conclusions. TMJ involvement in patients with JIA has a high incidence. There is no consensus on the treatment of TMJ
pathology and dentofacial deformities in patients with JIA, and treatment varies from counseling to surgery. Treatment to
improve aesthetics and function and to obtain pain reduction can be effective. However, the articles are heterogeneous,
and the level of evidence is low (level IV). (Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:581-589)

Juvenile idiopathic arthritis (JIA), also known as juvenile
chronic arthritis and juvenile rheumatoid arthritis, is a
common chronic rheumatic disease in childhood that is
present for longer than 6 weeks and has an onset before
the age of 16.1,2 JIA is an autoimmune disorder that can
be divided into 7 subtypes based on clinical symptoms
during the first 6 months of the disease.3 These different
subtypes are systematic arthritis, oligoarticular arthritis
(persistent and extended), polyarticular rheumatoid fac-
tor (RF)-positive arthritis, polyarticular RF-negative
arthritis, enthesitis-related arthritis, psoriatic arthritis, and
undifferentiated arthritis.3
In JIA, different joints can be involved, including the
temporomandibular joint (TMJ). The TMJ can be the
first and/or the only affected joint,4 and it can be uni-
lateral or bilateral. TMJ involvement was first described
by Still in 1896.5 The reported prevalence of TMJ
involvement in patients with JIA varies from 17% to
87% depending on the population studied, the subtypes
of JIA represented, and the method by which involve-
ment is diagnosed.6-8
The etiology of JIA is not completely defined,3 and the
pathogenesis of the disease is not well understood. Auto-
immune, genetic, and environmental factors are suggested
to play an important role in the inflammatory cascade.1
Several suggested pathophysiologic factors for bone
destruction and erosion are stimulation of osteoclasts by
Department of Oral and Maxillofacial Surgery, Erasmus University
Medical Centre Rotterdam.
Received for publication Jul 17, 2013; returned for revision Dec 5,
2013; accepted for publication Jan 24, 2014.
Ó 2014 Elsevier Inc. All rights reserved.
2212-4403/$ - see front matter
http://dx.doi.org/10.1016/j.oooo.2014.01.226
interleukins, tumor necrosis factor a, macrophage
colony-stimulating factor, receptor activator of NFkB
ligand (RANKL),9,10 and inhibition of the functional
capacity of osteoblasts in patients with rheumatoid
arthritis.10
This article focuses only on TMJ involvement caused
by TMJ arthritis due to JIA. TMJ involvement is diag-
nosed on clinical examination (including a subjective
and objective assessment) and on imaging techniques,
including panoramic radiograph, computed tomography
(CT), magnetic resonance imaging (MRI), and ultra-
sonography (US). The gold standard to verify TMJ
inflammation is a contrast-enhanced MRI.11 Clinical
features of TMJ involvement in patients with JIA are,
among others, micrognathia, retrognathia, asymmetry of
the mandible, malocclusion, pain, limitation of maximal
mouth opening, trismus, deflection at maximal mouth
opening, swelling of the TMJ, limitation of laterotrusive
movements, joint noises, and local morning stiff-
ness.1,12,13 Unfortunately, clinical features and symp-
toms are not reliable in detection of TMJ involvement in
patients with JIA, because both swelling and pain are
rarely present in most cases of TMJ involvement.6 This
results in late diagnosis of TMJ involvement, in which
Statement of Clinical Relevance
This article describes a systematic review concern-
ing short-term and long-term treatment options for
temporomandibular joint involvement in patients
with juvenile idiopathic arthritis to improve aes-
thetics and function and to reduce pain.

581
ORAL MEDICINE
582 te Veldhuis, te Veldhuis and Koudstaal
joint damage and severe growth disturbances are
evident. Differential diagnosis of JIA in the TMJ in-
cludes infection, injuries, malignancy, and connective
tissue diseases.1,14
Management of TMJ pathology in patients with JIA
is based on a combination of interventions, such as
counseling, pharmacologic therapies, physiotherapy,
occlusal appliances, orthodontics, and surgery.1,12,13
Despite the frequency with which the TMJ is involved
in patients with JIA, consensus on treatment is lacking.
Therefore, the aim of this study is to systematically
review the available literature for those publications
that have addressed treatment in patients with JIA with
TMJ involvement, and to create a concise review.
METHODS
Search strategy
The PRISMA statement15 (Preferred Reporting Items
for Systematic Reviews and Meta-Analyses) was used
as a guideline. An electronic search was conducted in 6
databases: Embase, Medline OvidSP, Cochrane Library
CENTRAL database, CINAHL, Web of Science, and
PubMed, with defined combination in keywords spec-
ified for each database (Appendix I; available at doi: 10.
1016/j.oooo.2014.01.226). The reference lists of all
articles were screened for additional relevant sources.
Data collection and analysis
Two review authors (E.t.V. and A.t.V.) read the titles
and abstracts (when available) of all reports indepen-
dently. From all studies that appeared to meet the in-
clusion criteria, or when there was insufficient data in
the title, abstract, or both, the full-text version was ob-
tained to make a definitive decision. Both authors read
the full-text articles, and each author made an inde-
pendent decision whether the studies met the inclusion
criteria. Any disagreement was resolved by discussion,
and when no agreement could be reached, an additional
researcher was involved (M.K.), until consensus was
reached. Included studies had to describe treatment of
humans and be published in English. Included studies
were scored on quality of evidence using the Univer-
sity of Oxford Center for Evidence-Based Medicine
(CEBM) criteria,16 sample size, reasons for treatment,
mean age of diagnosis of JIA, and mean age of treat-
ment of TMJ involvement, type of intervention, treat-
ment outcome, side effects, and length of follow-up
(Table I).
RESULTS
The literature search yielded 969 citations; after correc-
tion for duplicates, 553 citations remained (EMBASE,
369; Medline OvidSP, 30; Cochrane Library CENTRAL
database, 0; CINAHL, 6; Web of Science, 101; and
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May 2014
PubMed, 47). Two articles were identified through
reference list search.
The 555 articles were screened according to title
and abstract. In case abstracts were missing or it was
questionable whether the abstract met the aforemen-
tioned criteria, full-text articles were retrieved as to
avoid excluding articles of possible relevance.
A total of 428 articles were excluded for various
reasons, including (1) articles focused on topics other
than treatment, on other conditions not specified as JIA
and involvement of the TMJ, or on comorbidity; (2)
review articles; (3) articles without patient studies; (4)
articles describing the same patients as another included
article; (5) non-English-language articles; (6) abstracts
of meetings; and (7) poster presentations.
The remaining 127 articles were screened on reading
the full-text. Another 87 articles were excluded. Two
articles could not be traced down by the institutional
library. This resulted in a total of 40 articles (Figure 1).
The included articles preferably would be divided
into 2 main groups without overlap. The first group,
focused on TMJ inflammation, would be called the
arthritis group. The second group, focused on growth
disturbances (and including treatment modalities to
improve mandibular growth; to resolve micrognathia,
retrognathia, malocclusion, facial asymmetries, anky-
losis, and obstructive sleep apnea [OSA]; and to
improve aesthetics), would be called the dentofacial
deformity group. Unfortunately, however, many articles
describe patients who were treated for both TMJ arthritis
and a dentofacial deformity. Therefore, although the 2
aforementioned group names have been used, there is
an overlap of articles included in the first and second
group. An overview of the scored items of all included
studies is given in Table I.
Arthritis group
The treatment goal in the arthritis group is to control the
disease and prevent further progression of JIA in the
TMJ. The following treatment modalities were found:
pharmacologic interventions (local or systemic), phys-
ical therapy, oral appliance, functional appliance, sur-
gery, or a combination of therapies.
The use of nonsteroidal anti-inflammatory drugs
(NSAIDs) is mentioned as the first treatment option to
reduce pain.17 However, if the use of NSAIDs is insuf-
ficiently effective, more rigorous treatment modalities
can be suggested.4
Corticosteroids, such as 0.5-mL or 1-mL triamcino-
lone acetonide (40 mg/mL)18-20 or triamcinolone
hexacetonide (5-20 mg/mL),18,20-25 are used for intra-
articular injections in the TMJ, to reduce the inflam-
mation. The volume of the injected corticosteroid fluid
was chosen based on the size of each joint space and
determined (1) by the amount of resistance encountered
Table I. Studies meeting criteria for inclusion in current review

Volume 117, Number 5

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CEBM level Sample Reason for Mean age at Mean age at
Author Year of evidence size (N) treatment diagnosis (y) treatment (y) Intervention Outcome Side effects Follow-up
Habibi19 2012 IV 38 Arth, P, LMM 8 12.25 IACIs Impr Scar injection site 8 wk
Stoll20 2012 IV 63 Arth 8.5 10 IACIs Impr Hypopigmentation NS
Farronato36 2011 IV 1 Asym NS NS O Impr NS 5y
Neto31 2011 IV 1 Arth, Cl, LMM, P NS 22 S, PT, BSSO, O Impr NS 6 mo
Mina32 2011 IV 28 LMM, P 11 13 DIP Impr Erythema, Skin blister NS
Metallic taste
Stoustrup34 2011 IV 22 Asym NS 7.5 S Impr NS NS
Parra21 2010 83 Arth NS 12 IACIs Impr Skin atrophy Swelling Pain 6 mo
Farronato37 2009 IV 72 Cl 4-16 NS O Impr NS 4y
Georgios43 2009 IV 1 Mho, SA, R 2 18 O, BSSO, GP Impr NS 1y
Hugle22 2009 IV 1 Arth 6 8 IACIs NS Lipoatrophy 1.5 y
Ringold16 2008 22 Arth 8 10 IACIs Impr Skin atrophy Intra-articular NS
calcifications
Synodinos40 2008 IV 1 Cl 2 20 O, HG Impr None NS
Cahill17 2007 IV 15 Arth 8.3 NS IACIs Impr None 9 mo
Stringer42 2007 IV 5 Cl, Mho, OB 3-6 14-18 CCG, OS, O Impr Chin wound Loss of graft 4-14 d
Leshem44 2006 IV 8 Cl NS 18 O, BSSO, LF, GP Impr None 8 mo
Mackool30 2006 IV 1 M, SA 4 25 O, VO, ED, PT Impr NS NS
Martinez-Toledo33 2006 IV 2 Arth 6.5 10.5 SE Impr NS 6 mo
Singer45 2006 IV 1 E, Cl, Mho NS 18 BSSO, LF, GP, O Impr NS NS
Arabshahi18 2005 IV 23 Arth 5.5 9 IACIs Impr Swelling 6-12 mo
Gogalniceanu53 2005 IV 1 Ank 9 24 AP, DG Impr None 10 mo
Scolozzi4 2005 IV 1 LMM, P 5 5 Ar, PT Impr NS 6 mo
Falcini15 2003 IV 1 Arth 9 9 N, O Impr NS 7 mo
Oye46 2003 IV 16 E NS 24 O, BSSO, GP Impr Scar Infection Endo-treatment 5y
Gingival retraction

te Veldhuis, te Veldhuis and Koudstaal 583

Reoperation Extraoral
changes
Kitai38 2002 IV 1 Asym, R 6 17 O Impr NS NS
Martini23 2001 IV 1 Arth 15 15 IACIs, SE Impr NS 2y
Ince24 2000 IV 45 Arth 14.1 6.7 MTX Impr NS NS
Svensson25 1998 IV 12 Arth, Asym, OB, R NS 13.7 CCG, DR, O, PT, S Impr Pain Mandibular overgrowth 4-7 y
Asym
Cohen50 1998 IV 1 R, SA NS 16 LF, ME, MA, GP, UPP Impr NS 6 mo
Maggioncalda39 1997 IV 1 Cl NS 11 O Impr None 2y
Pedersen35 1995 IV 1 R, Cl 0.75 9.1 O, S Impr None NS
Wolford51

REVIEW ARTICLE
1994 IV 1 Arth, Cl, M, OB, R NS NS O, multiple ost, JP Impr Temp. loss n. facialis 3y
Removal screws
Svensson29 1993 IV 7 E, RFH 4.5 12.5 CH, CCG, O, PT Impr NS 1-2 y
Bowler41 1991 IV 2 M, OB NS 14 CCG, O Impr NS 0.5 y
Kreiborg47 1990 IV 1 R 9.2 15 OS Impr None 1.5 y
(continued on next page)
 ORAL MEDICINE OOOO
584 te Veldhuis, te Veldhuis and Koudstaal May 2014

General abbreviations: CEBM, Centre for Evidence-Based Medicine (University of Oxford); NS, not stated; Impr, improvement in mandibular movements or decrease in pain, sleep apnea, or arthritis; wk,
with injection by the physician or (2) by the weight
Follow-up

Reason for treatment: Ank, ankylosis in the TMJ; Arth, arthritis in the TMJ; Asym, asymmetry of the face; Cl, class II malocclusion; E, poor esthetics; LMM, limitation in mandibular movements; M,

Treatment: AP, arthroplasty; Ar, arthrocentesis; BSSO, bilateral sagittal split osteotomy; CCG, costochondral graft; CH, condylar head resection; DG, Dacron graft; DIP, dexamethasone iontophoresis; DR,
disk resection; Distr, distraction; ED, external distraction; GP, genioplasty; HP, high-pull headgear; IACIs, intra-articular corticosteroid injections; JP, joint prosthesis; LF, LeFort I osteotomy; MA,
mandibular advancement; ME, maxillary expansion; MTX, methotrexate; N, nonsteroidal anti-inflammatory drug; O, orthodontic treatment; OS, orthognathic surgery; PT, physical therapy; S, splint; SB,
of the patient.18,20-24 The number of injections in the

5.5 y
5y TMJs varied from 1 up to 6.18-25 To ensure proper

1y

1y
NS
NS
needle placement in the TMJ, radiologic ultrasonogra-
phy,21,23,24 CT,19,20,24 and MRI20 guidance can be used.
Disadvantages of corticosteroid injections are transient
postoperative pain,23 subcutaneous atrophy at the in-
Side effects

jection site,18 lipoatrophy,24 facial swelling lasting from

1 day up to 2 weeks postoperatively,20,22,23 fever,22 skin
Reoperation

hypopigmentation at the site of the injection,22 and scar

micrognathia; MHo, mandibular hypoplasia; MHy, maxillary hyperplasia; OB, open bite; P, pain; R, retrognathia; RFH, restore facial height; SA, sleep apnea; T, trismus. formation at the injection site.21
NS

NS
NS
NS
NS

Methotrexate (MTX) is a disease-modifying anti-

rheumatic drug (DMARD). It has been found to be
Unchanged

effective in minimizing TMJ destruction and craniofa-

Outcome

cial dysmorphology in patients with JIA.26

Impr
Impr

Impr
Impr

Physical therapy is combined with oral appliance

NS

therapy, orthodontic treatment, surgical treatment, or a

combination of those.4,27-33 All except for 2 articles30,33
LF, CCG, BSSO, GP

described exercises that were based on increasing the

Intervention

maximal mouth opening. Three articles described the

silicone block; SE, synovectomy; UDO, unilateral distraction osteogenesis; UPP, uvulopalatoplasty; VO, vertical ramus osteotomy.

use of an exercising tool, such as a rubber balloon27,31

AP, SB, PT
BSSO, O

or an exercise appliance.28
O, VO
PT, O

Combined physical therapy with pharmacologic

O, S

intervention is described in only 2 case reports. Dexa-

methasone iontophoresis (IP) is a noninvasive physio-
treatment (y)
Mean age at

therapy modality that allows transdermal delivery of

9-23

11.8

6 mg dexamethasone per TMJ per session. Low-grade

35

13
14

16

electric currents are suggested to lead to the dissociation

of hydrophilic medications into ions that move to
penetrate deeper anatomic structures.34 Side effects of
diagnosis (y)
Mean age at

dexamethasone IP are transient nonpainful site ery-

1-11
2.5

9.2
13
9

thema, a metallic taste during dexamethasone IP, and a

small skin blister.34
A positive outcome is demonstrated in a case of a
M, LMM, MHy, T

Arth, Cl, LMM, P

15-year-old girl who received therapeutic exercises

Reason for
treatment

(to increase maximal mouth opening) and gold injection

Ank, P, SA

(to control the arthritis).29

Splint therapy as initial management for stabilization
OB, R

Cl, M
Ank

of the resorption process in both condyles, in combi-

nation with physical therapy, is suggested.33
size (N)
Sample

Arthrocentesis and lavage of the TMJs with 100 mL

1
7
1
1
1
1

of NaCl 0.9%, combined with physical therapy, im-

proved maximal mouth opening.4 Also, an arthroscopic
CEBM level

synovectomy followed by a corticosteroid injection

of evidence

with triamcinolone hexacetonide in the TMJ gave an

IV
IV
IV
IV
IV
IV

increased maximal mouth opening and a reduction in

pain.25,35
weeks; mo, months; y, years.
1988
1988
1986
1985
1978
1975
Year
Table I. Continued

Dentofacial deformity group

The treatment goal in the dentofacial deformity group
is to influence and restore function in craniofacial
deformities caused by involvement of the JIA in the
Author

Seymour26
52

Littman27

TMJ. Multiple treatment modalities were docu-

Guyuron

Turpin48
Ganik28
Myall49

mented, varying from a distraction splint to a total

joint prosthesis.
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Volume 117, Number 5
Fig. 1. Data extraction flowchart according to the PRISMA
statement (Preferred Reporting Items for Systematic Reviews
and Meta-Analyses).15
Distraction splints can be used in the treatment of JIA
involvement of the TMJ.36,37 In patients with unilateral
TMJ arthritis, it is suggested to reduce the asymmetry,36
and in a patient with retrognathia of the mandible, the
distraction splint is used in an initial phase of unloading
the TMJ by increasing the joint space and stretching
the capsule and the muscles, followed by a second
phase using an activator to generate a protrusion of the
mandible.37
Orthodontic treatment to improve mandibular ramus
growth, with a subsequent enhancement in occlusion,
masticatory function, and profile, is outlined in 6 arti-
cles.17,38-42 The use of an activator is demonstrated in 3
articles38-40 and high-pull headgear in 2 articles.41,42
Unfortunately, one article does not describe a detailed
orthodontic treatment.17 Two patients had a unilateral
involvement of the TMJ,17,38 and multiple patients had
bilateral involvement.39-42 Treatment with only ortho-
dontics is performed in patients aged 4 to 16 years old; in
these patients growth is expected.38 The disadvantages of
the orthodontic treatment are the long treatment time and
lack of patient adherence.39,41 One article proposed the
use of an activator after growth had ceased.40
Fifteen articles dealt with combined orthodontic and
surgical approaches in 64 patients aged 14 to 53 years.
Some of these articles described preoperative or post-
operative orthodontic treatment (or both).27,31-33,43-53
Indications for treatment included class II malocclusion,
anterior open bite, decreased posterior facial height,
REVIEW ARTICLE
te Veldhuis, te Veldhuis and Koudstaal 585
anterior maxillary vertical excess, hypoplastic mandible
with posterior rotation, decreased lower facial height,
OSA, and pain.27,31-33,43-53 Surgical treatment was
not homogeneous. The following surgical interventions
were mentioned: unilateral distraction, bilateral sagittal
split osteotomy distraction,45,46,48,49,51,52 vertical
distraction32,33,47,50 with45,48,49 or without a combined
genioplasty, condylectomy with insertion of costo-
chondral graft,27,31,43,44 and bimaxillary sur-
gery.44,46,47,49,51,52 Aesthetics were improved either by
a genioplasty44,46-48,51,52 or by augmentation of mandib-
ular symphysis with a Proplast implant50 or a porous
block hydroxylapatite.53 According to Cohen et al.
(1998),52 OSA can be reduced by a tongue reduction,
uvulopalatoplasty, and tongue hyoid suspension.
TMJ reconstruction and mandibular advancement
with a Techmedica custom-made total joint prosthesis
was performed in one patient.53
Outcomes measures are various and can include
resolution of OSA, improved aesthetics, reduction of
facial asymmetry, improved function, and improved
occlusion; all patients were more satisfied after surgery
than before surgery,27,31-33,43-45,47-53 except for one
patient who was not satisfied with her occlusion and
aesthetics.46
A combination of medication, orthodontic treatment,
and surgical intervention was suggested to resolve
ankylosis of the TMJ.28,54,55 The TMJ involvement was
either unilateral or bilateral. The use of an articular
interpositioning implant (a silicone rubber implant28
and a Dacron texture55) in the TMJ was demonstrated
in 2 cases. One article described a patient who under-
went a replacement of the TMJ with rib cartilage and
bone grafts.54 All patients had an increased maximal
mouth opening.
DISCUSSION
This review provides an overview of the scientific ev-
idence on treatment of TMJ involvement in children
with JIA. Unfortunately, most articles are retrospective
case series, and no randomized clinical trials were
found on the treatment management of children with
JIA with TMJ involvement. The articles found scored
low on methodologic quality, and owing to the het-
erogeneity in the articles with subsequent results, a
meta-analysis of the data was impossible. Many articles
do not report on the degree of TMJ involvement or the
type of JIA, which makes it almost impossible to
compare these studies. Although preferably the articles
would be divided into 2 main groups, this was rather
difficult owing to the poor quality of most articles and
the often reported combined treatment for arthritis and
craniofacial deformities.
NSAIDs, such as naproxen, ibuprofen, and indome-
tacin, are the most commonly used drugs in children
ORAL MEDICINE
586 te Veldhuis, te Veldhuis and Koudstaal
with JIA; generally, they are well tolerated and have
few side effects.1,14,56 In the results of this article,
NSAIDs were used as maintenance medication and
were not beneficial for reducing TMJ complaints, so
more aggressive interventions were necessary.4
A more invasive intervention is the use of corti-
costeroids, which can be used either locally or sys-
temically. Triamcinolone acetonide and triamcinolone
hexacetonide intra-articular corticosteroid injections
(IACIs) are increasingly used to reduce pain, to im-
prove jaw mobility, and to stop further TMJ inflam-
mation in patients with JIA.
Small differences in doses, in the amount of injected
volume, and in the frequency of injection are reported.
The additional benefit of multiple injections in the same
TMJ is unknown and not studied in humans. IACIs
seem to be safe and efficacious, although side effects
like transient postoperative pain,23 subcutaneous atro-
phy at the injection site,18 lipoatrophy,24 facial swelling
lasting from 1 day up to 2 weeks postoperatively,20,22,23
fever,22 skin hypopigmentation at the site of the injec-
tion,22 and scarring at the injection site21 are mentioned.
A standardized method for IACIs is lacking. Long-term
side effects are unknown for humans, as is whether
growth disturbances are prevented by the use of IACIs.
There are some concerns about the use of IACIs for
TMJ arthritis, owing to damage to the condyle, anky-
losis,57-59 and reduced mandibular growth in experi-
mental TMJ arthritis in rabbits.
IP is described as an effective, painless, and safe
treatment in patients with various musculoskeletal in-
flammatory conditions.60 The quantity and distribution
of radiolabeled dexamethasone in monkeys is demon-
strated in all tissues underlying the electrode down in
the deeper structures (beyond 1.5 cm).61 In the litera-
ture, 3 to 6 IP treatments are recommended.62 Although
one study reported a standard of 8 to 10 sessions,34 the
benefit of more than 3 to 6 sessions is unknown and has
not been studied for humans with JIA. A standardized
method for dexamethasone IP is lacking. Long-term
side effects of IP are unknown for humans.
This literature search yielded no articles using sys-
temic corticosteroids for the treatment of TMJ arthritis.
Side effects of systemic use of corticosteroids are, among
others, osteoporosis, growth arrest, and retardation.1,14,20
Systemic corticosteroids are suggested to have a negative
effect on mandibular growth, resulting in a more severe
class II pattern.63 The evidence of systemic corticoste-
roid use in patients with TMJ arthritis is lacking.
DMARDs appear to slow down or prevent further
joint damage.14 MTX is the most popular DMARD in
the treatment of JIA1,14 and is found to be effective in
minimizing TMJ destruction and craniofacial dysmor-
phology in patients with JIA.26 (Dose and dosage form
of the MTX are not mentioned in this article.26)
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May 2014
However, the use of MTX is not without side effects: it
may result in ulcerative stomatitis or interact with an-
tibiotics and NSAIDs.64
Tumor necrosis factor a inhibitors are mentioned to
reduce TMJ pain and improve oral function in the
literature for TMJ arthritis in adult patients65,66; no
articles were found for patients with JIA. Although
the use of salicylates, intramuscular gold salts, and the
antimalarial agents sulfasalazine and cyclosporine are
mentioned in the literature on treating patients with
JIA,64,67 there were no studies found addressing spe-
cific use of these medications for TMJ arthritis.
Sodium hyaluronate injections have been used in
adults with TMJ osteoarthritis,13 but no data were found
in this literature search for their use in patients with JIA.
Exercising is an important part of treating JIA; it
improves physical ability and improves the quality of
life.68 The positive effects of physical training on TMJ
disorders in adult patients with rheumatoid arthritis
and ankylosing spondylitis are described.69 Although
physical therapy is mentioned in combination with
other treatment modalities, only one article described in
detail the type of exercise and treatment time.31 A
standardized physical therapy training program for
children with JIA and TMJ involvement is lacking.
The occlusal splint therapy is said to have a positive
effect on patients with TMJ osteoarthritis.70 Unfortu-
nately, this positive effect is not described in articles
discussing patients with JIA with TMJ involvement,
although one can theorize that oral splint therapy can
improve function and change the load in the TMJ
during an active period of arthritis.
The hypothesized aim of the distraction splint is to
initiate an anterior rotation of the mandible and unload
the TMJ. A distraction splint is an active appliance,
which alters the mandibular position by gradual thick-
ening of the splint in the posterior area. In case the
patient has a remission of the disease, in the initial
phase, the use of a distraction splint is followed by a
second phase of treatment. The second phase is started
with an activator to gradually protrude the mandible
and to optimize dentoalveolar development on the
affected side, to the extent that occlusal collapse can be
avoided in case of unilateral TMJ involvement.36,37
Evidence of the use of a distraction splint is scarce, and
more research is necessary.
An apnea-hypopnea index score >1 was found in
patients with JIA, which is indicative of mild sleep-
disordered breathing71; because sleep-disordered
breathing is multifactorial, several reasons for this higher
index score are suggested: disease type, severity of the
disease, medication use, brain stem compression, upper
airway obstruction, increased body mass index as a result
of reduced mobility, impaired pharyngeal muscular
physiologic function, or craniofacial deformities.45,72
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Volume 117, Number 5
Few articles describe an orthodontic preparatory phase
of 1 to 2 years, and postoperative orthodontic treatment
is described in 7 articles27,31,32,47,50,51,53; unfortunately,
many articles did not specify the orthodontic treatment
time,33,43-45,48,49,52,53 used appliances,33,43-45,48,49,52,53 or
lacked a control group.27,31-33,43-49,51-53
Arthrocentesis of the TMJ is lavage of the upper joint
space, with hydraulic pressure and manipulation to
release adhesions and improve motion in patient with
degenerative joint diseases. Arthrocentesis is seen as a
minimally invasive treatment. Although arthrocentesis
seems promising, more research is necessary.73 In our
study, only one article suggested arthrocentesis of the
TMJ in a patient with JIA.4
Arthroscopy for temporomandibular disorders
(TMDs) is accomplished with a rigid optic fiber with a
diameter between 1.7 and 2.7 mm to obtain visualiza-
tion of the joint tissues, perform diagnosis, perform
irrigation, take biopsies, remove adhesions, correct
traumas located in the lateral capsules, and perform
imaging. There is no consensus for the use of arthros-
copy in patients with TMD,74 including those with JIA
involvement.
Growth disturbances due to JIA in the TMJ are
challenging for the orthodontist and maxillofacial sur-
geon. A distinction in treatment can be made between
discrepancies in jaw relation, malocclusion, and aes-
thetic improvement treatment in patients with JIA with
normal growth or abnormal growth. Clinical features
can be diverse, including micrognathia, Angle’s Class
II malocclusion (unilateral or bilateral), retrognathia,
‘bird face,’ anterior open bits, decreased ramus height,
and length.14 Many surgical techniques are discussed in
the different case reports and case series found in the
literature search. All the techniques are complex and
have in common the long treatment times to reach an
acceptable and functional outcome.
Although rare, complications of orthognathic sur-
gery are, among others, unfavorable osteotomy, hem-
orrhage, (transient) nerve damage, malocclusion,
infection, condylar resorption, ophthalmic impairment,
failure of osteosynthesis material, hearing problems,
dysphagia, and neuropsychiatric problems,75 as well
as mandibular overgrowth and asymmetry after early
surgical correction.27
CONCLUSION
Systemic treatment to control the TMJ involvement in
children with JIA is suggested; however, evidence is
lacking. Treatment goals to improve aesthetics and
function and to reduce pain showed acceptable results.
However, because the articles are heterogeneous, no
definitive conclusions can be drawn, and more research
is necessary. The general quality of the literature is
suboptimal, and there is a need for higher-quality
REVIEW ARTICLE
te Veldhuis, te Veldhuis and Koudstaal 587
studies, with well-defined patient populations and ex-
amination techniques. Ideally, randomized controlled
trials are needed to compare different treatment mo-
dalities. This is probably best provided by multicenter
studies of institutions treating patients with JIA with
TMJ involvement and would improve the care for all
our patients.
The authors acknowledge W. Bramer, biomedical information
specialist of the Erasmus University Medical Centre, for his
assistance with the literature search.
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Reprint requests:
Emma C. te Veldhuis, DDS
Department of Oral and Maxillofacial Surgery
Erasmus University Medical Centre
Room D-240, Dr Molewaterplein 40
3000 CA Rotterdam
The Netherlands
e.teveldhuis@erasmusmc.nl; ecteveldhuis@hotmail.com
ORAL MEDICINE
589.e1 te Veldhuis, te Veldhuis and Koudstaal
APPENDIX I SEARCH STRATEGIES
Embase
(‘juvenile rheumatoid arthritis’/de OR (((juvenile*)
NEAR/3 (arthrit* OR arthropath* OR polyarthr*)) OR
((stiel OR still OR chauffard) NEAR/3 (disease* OR
syndrome*))):ab,ti) AND ((’temporomandibular joint’/
de OR ‘temporomandibular joint disorder ’/de OR
(((temporomandibular* OR craniomandibul* OR man-
dibul* OR jaw* OR mandibulotempor*) NEAR/3
(joint* OR articulation OR disc* OR disk*)) OR
TMJ):ab,ti) OR (‘face pain’/de OR (((face OR facial OR
orofac*) NEAR/3 (pain*))):ab,ti) OR (‘face malforma-
tion’/exp OR ‘face asymmetry’/de OR ‘jaw malforma-
tion’/exp OR (((face OR facial OR orofac* OR
maxillofac* OR dentofac* OR jaw* OR chin OR man-
dib*) NEAR/3 (malformat* OR anomal* OR deformit*
OR disformit* OR defect* OR dysostos* OR abnormal*
OR underdevelop* OR receding OR asymmetr* OR
ankylos*)) OR micrognat* OR microgenia* OR retro-
gnat*):ab,ti) OR ‘temporomandibular ankylosis’/de)
Medline (ovidSP)
(Arthritis, Juvenile Rheumatoid/ OR (((juvenile*)
ADJ3 (arthrit* OR arthropath* OR polyarthr*)) OR
((stiel OR still OR chauffard) ADJ3 (disease* OR
syndrome*))).ab,ti.) AND ((exp Temporomandibular
Joint/ OR exp Temporomandibular Joint Disorders/ OR
(((temporomandibular* OR craniomandibul* OR man-
dibul* OR jaw* OR mandibulotempor*) ADJ3 (joint*
OR articulation OR disc* OR disk*)) OR TMJ).ab,ti.)
OR (facial pain/ OR (((face OR facial OR orofac*)
ADJ3 (pain*))).ab,ti.) OR (Facial Asymmetry/ OR exp
Jaw Abnormalities/ OR (((face OR facial OR orofac*
OR maxillofac* OR dentofac* OR jaw* OR chin OR
mandib*) ADJ3 (malformat* OR anomal* OR defor-
mit* OR disformit* OR defect* OR dysostos* OR
abnormal* OR underdevelop* OR receding OR asym-
metr* OR ankylos*)) OR micrognat* OR microgenia*
OR retrognat*).ab,ti.))
Cochrane
(“Arthritis, Juvenile Rheumatoid”/ OR (((juvenile*)
NEAR/3 (arthrit* OR arthropath* OR polyarthr*))
OR ((stiel OR still OR chauffard) NEAR/3 (disease*
OR syndrome*))):ab,ti) AND ((exp Temporoman-
dibular Joint/ OR exp Temporomandibular Joint
Disorders/ OR (((temporomandibular* OR cranio-
mandibul* OR mandibul* OR jaw* OR man-
dibulotempor*) NEAR/3 (joint* OR articulation OR
disc* OR disk*)) OR TMJ):ab,ti) OR (facial pain/ OR
(((face OR facial OR orofac*) NEAR/3 (pai-
n*))):ab,ti) OR (Facial Asymmetry/ OR exp Jaw
Abnormalities/ OR (((face OR facial OR orofac* OR
OOOO
May 2014
maxillofac* OR dentofac* OR jaw* OR chin OR
mandib*) NEAR/3 (malformat* OR anomal* OR
deformit* OR disformit* OR defect* OR dysostos*
OR abnormal* OR underdevelop* OR receding OR
asymmetr* OR ankylos*)) OR micrognat* OR
microgenia* OR retrognat*):ab,ti))
CINAHL
(MH Arthritis, Juvenile Rheumatoidþ OR TX (((juve-
nile*) N3 (arthrit* OR arthropath* OR polyarthr*)) OR
((stiel OR still OR chauffard) N3 (disease* OR syn-
drome*)))) AND ((MH Temporomandibular Jointþ OR
MH Temporomandibular Joint Disordersþ OR TX
(((temporomandibular* OR craniomandibul* OR man-
dibul* OR jaw* OR mandibulotempor*) N3 (joint* OR
articulation OR disc* OR disk*)) OR TMJ)) OR (MH
facial painþ OR TX (((face OR facial OR orofac*) N3
(pain*)))) OR (MH Facial Asymmetryþ OR MH Jaw
Abnormalitiesþ OR TX (((face OR facial OR orofac*
OR maxillofac* OR dentofac* OR jaw* OR chin OR
mandib*) N3 (malformat* OR anomal* OR deformit*
OR disformit* OR defect* OR dysostos* OR
abnormal* OR underdevelop* OR receding OR asym-
metr* OR ankylos*)) OR micrognat* OR microgenia*
OR retrognat*)))
Web of Science
(TS¼(((juvenile*) NEAR/3 (arthrit* OR arthropath*
OR polyarthr*)) OR ((stiel OR still OR chauffard)
NEAR/3 (disease* OR syndrome*)))) AND
((TS¼(((temporomandibular* OR craniomandibul*
OR mandibul* OR jaw* OR mandibulotempor*)
NEAR/3 (joint* OR articulation OR disc* OR disk*))
OR TMJ)) OR (TS¼(((face OR facial OR orofac*)
NEAR/3 (pain*)))) OR (TS¼(((face OR facial OR
orofac* OR maxillofac* OR dentofac* OR jaw* OR
chin OR mandib*) NEAR/3 (malformat* OR
anomal* OR deformit* OR disformit* OR defect*
OR dysostos* OR abnormal* OR underdevelop* OR
receding OR asymmetr* OR ankylos*)) OR micro-
gnat* OR microgenia* OR retrognat*)))
PubMed
((((juvenile*[tiab]) AND (arthrit*[tiab] OR arthropath*
[tiab] OR polyarthr*[tiab])) OR ((stiel[tiab] OR still
[tiab] OR chauffard[tiab]) AND (disease*[tiab] OR
syndrome*[tiab])))) AND (((((temporomandibular*
[tiab] OR craniomandibul*[tiab] OR mandibul*[tiab]
OR jaw*[tiab] OR mandibulotempor*[tiab]) AND
(joint*[tiab] OR articulation OR disc*[tiab] OR disk*
[tiab])) OR TMJ[tiab])) OR ((((face[tiab] OR facial
[tiab] OR orofac*[tiab]) AND (pain*[tiab])))) OR
((((face[tiab] OR facial[tiab] OR orofac*[tiab] OR
OOOO REVIEW ARTICLE
Volume 117, Number 5 te Veldhuis, te Veldhuis and Koudstaal 589.e2

maxillofac*[tiab] OR dentofac*[tiab] OR jaw*[tiab] abnormal*[tiab] OR underdevelop*[tiab] OR receding

OR chin[tiab] OR mandib*[tiab]) AND (malformat* [tiab] OR asymmetr*[tiab] OR ankylos*[tiab])) OR
[tiab] OR anomal*[tiab] OR deformit*[tiab] OR dis- micrognat*[tiab] OR microgenia*[tiab] OR retrognat*
formit*[tiab] OR defect*[tiab] OR dysostos*[tiab] OR [tiab]))) NOT medline[sb]