UNIVERSIDADE ESTADUAL PAULISTA
“JÚLIO DE MESQUITA FILHO”
Instituto de Ciência e Tecnologia
Campus de São José dos Campos
CASE REPORT DOI: https://doi.org/10.4322/bds.2023.e3998
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Systemic scleroderma: imaging findings of diagnosis and clinical
management of temporomandibular joint disorders
Esclerodermia sistêmica: diagnóstico por achados imaginológicos e manejo clínico das disfunções temporomandibulares
Letícia Cavassini TORQUATO1 , Clarissa Carvalho Martins MACIEL1 , Nátaly Domingues ALMEIDA1 , Wagner de OLIVEIRA2 ,
Maria Aparecida Neves JARDINI1 , Antonio Braulino de MELO FILHO3 , Sergio Lucio Pereira Castro LOPES4 ,
Andréa Carvalho DE MARCO1
1 - Universidade Estadual Paulista, Instituto de Ciência e Tecnologia, Departamento de Diagnóstico e Cirurgia, Divisão da Periodontia,
São José dos Campos, SP, Brasil
2 - Universidade Estadual Paulista, Instituto de Ciência e Tecnologia, Departamento de Odontologia Restauradora, Divisão da Oclusão e
Articulação Temporomandibular, São José dos Campos, SP, Brasil
3 - Universidade Estadual Paulista, Instituto de Ciência e Tecnologia, Departamento de Odontologia Social e Clínica Infantil, São José dos
Campos, SP, Brasil
4 - Universidade Estadual Paulista, Instituto de Ciência e Tecnologia, Departamento de Diagnóstico e Cirurgia, Divisão da Radiologia, São
José dos Campos, SP, Brasil
How to cite: Torquato LC, Maciel CCM, Almeida ND, Oliveira W, Jardini MAN, Melo AB Fo, et al. Systemic scleroderma: imaging
ndings of diagnosis and clinical management of temporomandibular joint disorders. Braz Dent Sci. 2023;26(4):e3998. https://doi.
org/10.4322/bds.2023.e3998
ABSTRACT
Scleroderma, an autoimmune disease, directly affects the production of collagen in the connective tissue. In its
systemic form, the disease causes oral manifestations such as: limited mouth opening, xerostomia, periodontal
disease, thickening of the periodontal ligament and bone resorption of the mandible. This case report aims to
draw attention to the difculties encountered in providing dental care to patients with scleroderma and also
to highlight the imaging ndings, with emphasis on the temporomandibular joints, which are of interest to
dentists about the disease. In the present case, the patient presented bilateral condylar erosion, in addition
to disc displacement without reduction. Due to the systemic condition of the patient, it was decided to make
an individualized occlusal splint. The limitation of mouth opening is a limiting factor for the manufacture of
prostheses and plates, which is why partial prostheses are indicated and are easily removed by the patient. The
decisions taken have a great impact on the health and quality of life of patients in these conditions, so there is a
need for multidisciplinary involvement in order to arrive at the best treatment plan. After ve years of using the
stabilizing plate overnight, the patient reports greater comfort and muscle relaxation upon waking up.
KEYWORDS
Case Reports; Diagnostic imaging; Systemic scleroderma; Temporomandibular joint; Temporomandibular joint disorders.
RESUMO
Esclerodermia, uma doença autoimune, afeta diretamente a produção de colágeno do tecido conjuntivo. Na forma
sistêmica, a doença causa manifestações bucais, como: limitação de abertura bucal, xerostomia, doença periodontal,
espessamento do ligamento periodontal e reabsorção da mandíbula. Este relato de caso tem por objetivo chamar
atenção para as diculdades encontradas ao promover atendimento odontológico para pacientes com esclerodermia
e também destacar os achados imaginológicos, com ênfase na articulação temporomandibular, que são da doença
e de interessa ao cirurgião-dentista. No presente caso, a paciente apresentava erosão condilar bilateral, com
deslocamento de disco sem redução. Devido à condição sistêmica da paciente, foi decidido confeccionar uma placa
oclusal individualizada. A limitação de abertura bucal é um fator limitante para confecção de próteses e placas,
por isso próteses parciais são indicadas, além de serem de fácil remoção pelo paciente. As decisões tomadas tem
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Torquato LC et al.
Systemic scleroderma: imaging findings of diagnosis and clinical management of temporomandibular joint disorders
Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
clinical management of temporomandibular joint disorders
INTRODUCTION
Scleroderma is an autoimmune connective
tissue disease of unknown etiology, which
causes vascular damage by excessive collagen
deposition. It can be classied as systemic when
it affects the internal organs and skin, localized
when the disease affects only the skin and
muscles. It has an incidence seven times higher
among women than men, usually occurring
between 40 and 50 years [1]. The prevalence
of systemic scleroderma (SS) varies between
four and 40 cases in every 100,000 people [2].
Its appearance is rare in children and the elder [2].
The Raynaud phenomenon is one of the rst
signs of SS, observed in 90-98% of patients, and
has characteristics of painful digital ischemia,
which results in reabsorption of the terminal
phalanges [3,4].
In its systemic form, in addition to
compromising the muscular structure, the
gastrointestinal, cardiopulmonary, and renal
systems are also affected. The orofacial region
in 80% of the cases presents most of the clinical
manifestations [5], which starts with the rigidity
of the tongue and hardening of the facial skin,
presenting perioral wrinkles, which results in a
classic appearance similar to a mask [3]. Some
secondary manifestations, such as periodontal
disease, may occur with the progression of SS [6].
Beyond the oral manifestations,
characteristics such as xerostomia, thickening of
the periodontal ligament and bone resorption in
the mandible, limitation of mouth opening due
to impairment of the temporomandibular joint,
and retraction of the lips appear [4,7].
Regarding imaging exams, patients with
SS have symmetrical erosions in the regions of
the coronoid process, mandibular angle, and
condyles [4]. Some authors report that ischemia
in bone tissue associated with SS patients [8]
may cause mandibular resorption.
This case report aims to raise awareness of the
imaging ndings of systemic scleroderma as well
as demonstrate the need for a multidisciplinary
team for adequate planning and management
oriented to the long-term quality of the patient’s
life.
CASE REPORT
The Research Ethics Committee (CEP)/
Plataforma Brasil has approved this study under
number: 2.002.426, the conception of the article
followed the Case Reporting (CARE) guidelines
for case reports [9].
In 2016, a 40-year-old female patient sought
dental care claiming difculty nding appropriate
treatment in the region. During the anamnesis,
the patient reported that was diagnosed with
Systemic Scleroderma in 2006 at 29 years
old; for dental treatment, the main complaint
was dentine sensitivity; she reported difculty
performing hygiene, complained of gastric reux,
and a “tearing the skin” [
sic
] sensation during
dental care.
The patient presented clinical characteristics
of SS, such as a pointed nose, thin and contracted
lips, perioral region of rmer consistency, and
limited mouth opening (Figure 1A); at rest,
the chin region had a wrinkled appearance,
demonstrating that the patient must force to
keep her lips closed. Thin and sensitive skin,
small and slim stature, small hands with a
slight curvature in all the ngers (Figure 1B)
which radiographically showed the process of
erosion of the distal phalanges and reduction of
the remaining joint spaces (Figure 1C), on cold
weather it was possible to notice the Raynaud
syndrome.
In the initial panoramic X-ray, in 2016, it
was possible to visualize some bone resorption
of the condyle and ramus of the mandible, where
bone resorption is generally more considerable
grande impacto na saúde e qualidade de vida de pacientes nessas condições, deste modo é necessário uma equipe
multidisciplinar envolvidas para chegar no melhor plano de tratamento. Após cinco anos fazendo uso da placa
estabilizadora durante a noite, a paciente relata maior conforto e relaxamento muscular ao acordar.
PALAVRAS-CHAVE
Articulação temporomandibular; Diagnóstico por imagem; Escleroderma sistêmico; Relatos de Casos; Transtornos
da articulação temporomandibular.
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Torquato LC et al.
Systemic scleroderma: imaging findings of diagnosis and clinical management of temporomandibular joint disorders
Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
clinical management of temporomandibular joint disorders
in cases of SS. It was also possible to visualize a
thickening of the periodontal ligament of some
teeth (Figure 2A). Despite the thickening of the
periodontal ligament, there were no clinical
signs of periodontitis or occlusal trauma. It was
observed gingival recession of many teeth,
that justifies the main complaint of dentine
sensitive of the patient, besides that, there
was gingival inflammation observed during
periodontal examination. The patient’s diagnosis
was plaque-induced gingivitis [10]. The oral
hygiene instruction, scaling, and planning
were conducted by sextants. The oral hygiene
instruction was conducted, considering the
patient’s motor and mouth opening limitations,
tooth extraction of the 36, and the application
of 5% uoride varnish on non-carious cervical
lesions was also conducted to treat dentine
hypersensitivity.
The patient remained in treatment and
follow-up to control the periodontal condition.
In 2018, the patient returned for care
complaining of muscle discomfort in the lower
third and temporomandibular joint (TMJ). A new
panoramic radiograph was requested to assess the
generalized oral condition when morphological
changes are observed in the condyles bilaterally,
hypoplastic images of the coronoid processes and
an elongation of the condylar neck, and resorption
of bilateral mandibular notch (Figure 2B); the
patient was referred for cone beam computed
tomography (CBCT) and magnetic resonance
imaging (MRI) scans.
On the CBCT images (Figure 3A), on the
left side, an advanced process of condylar
resorption/erosion is observed throughout its
extension, showing cortical irregularities, and
attening of the condylar slopes. Subchondral
involvement suggests medullary necrosis, in
addition, the temporal joint bone components
present with cortical irregularities and bone
sclerosis (Figure 3B). On the right side, there is
an erosion of the condylar and mandibular fossa
with discontinuity solution with consequent
communication with the middle fossa of the
skull. Additionally, a process of bone sclerosis
is observed in the joint bone components
(Figure 3C).
Figure 1 - (A) Frontal view of the patient, observe thin lips and contraction of the chin in resting position and narrowing of the nose; (B) Top
view of the patient’s left hand; (C) reabsorption in the distal phalanges.
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Torquato LC et al.
Systemic scleroderma: imaging findings of diagnosis and clinical management of temporomandibular joint disorders
Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
clinical management of temporomandibular joint disorders
Systemic scleroderma – case report: imaging findings of
diagnosis and clinical management of temporomandibular joint
disorders
Figure 2 - A: Panoramic radiograph of the patient at the start of treatment, in 2016. B: New panoramic radiograph requested in 2018.
Figure 3 - A: Computed tomography showing the condition of the condyles bilaterally. B: Erosion of the right condyle with solution of
discontinuity of the cortical bone, with probable dural communication. C: Computed tomography showing bone resorption in the left condyle.
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Torquato LC et al.
Systemic scleroderma: imaging findings of diagnosis and clinical management of temporomandibular joint disorders
Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
clinical management of temporomandibular joint disorders
Systemic scleroderma – case report: imaging findings of
diagnosis and clinical management of temporomandibular joint
disorders
At MRI, bilaterally, anterior displacement
of the disc is observed with complete
morphological, functional, and signal alteration
(Figure 4A and 4B), as well as reduction of the
joint space, fossa irregularity, and subchondral
sclerosis. Also, on the left side, there is an
area of hyposignal compatible with necrosis/
sclerosis, suggesting a degenerative process in
the condyle. Condylar morphological aspects
confirm the computed tomography findings.
The aspects observed in the images corroborate
joint degenerative aspects.
The treatment plan for this case was based on
making a stabilizing plate to relieve the internal
pressure of the joints and referring the patient to
an oral and maxillofacial surgeon, who, in turn,
recommended the use of the plate and follow-up
of the case. Because of the systemic involvement
and delicate prognosis, a conservative approach
to the case was preferable to performing invasive
surgery.
Due to the limitation of the mouth opening,
individual partial trays were made using
chemically activated acrylic resin (Figure 5A) for
the molding step (Figure 5B), based on the partial
molds (Figure 5C) partial models were made and
from them, the posterior parts of the plate were
made, reduced and adapted, in the mouth; the
two posterior parts were previously united with
chemically activated acrylic resin, then the plate
underwent a new reduction and adaptation so
that it could be easily installed and removed by
the patient herself (Figure 5D and 5E). After
ve years of using the stabilizing plate at night,
the patient reports greater comfort and muscle
relaxation upon waking up.
DISCUSSION
Systemic scleroderma is a rare chronic
condition that affects the connective tissue,
characterized by brosis of the skin, blood vessels,
and other body systems [11]. The oral cavity is
the region most involved in the pathogenesis of
scleroderma, the systematic review by Benz et al.
(2021) [12], demonstrated that the lips are the
most affected (57.6%), followed by the oral
mucosa (35.5%) and salivary glands (25.4%) [12].
Radiographically, it is possible to visualize
the thickening of the periodontal ligament,
more evident in the posterior teeth and present
in more than one quadrant, resorption of the
ramus and angle of the mandible, of the coronoid
process and condyles, which occurs in at least
20% of the cases [4,11,13 -16]. Matarese et al.
(2016) [17] observed that the frequency of bone
alterations was signicantly correlated with the
mean duration of the disease [17], in the present
case report, the patient showed signs of condylar
degeneration twelve years after the SS diagnosis.
Figure 4 - T1W MRI images showing: (A) Right TMJ with joint space reduction, fossa irregularity, subchondral sclerosis, in addition to total
anterior disc displacement without reduction on the right side. (B) Left TMJ with reduced joint space, fossa irregularity, subchondral sclerosis,
in addition to total anterior disc reduction. Green arrows: articular disc; Red arrows: condyle.
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Torquato LC et al.
Systemic scleroderma: imaging findings of diagnosis and clinical management of temporomandibular joint disorders
Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
clinical management of temporomandibular joint disorders
Systemic scleroderma – case report: imaging findings of
diagnosis and clinical management of temporomandibular joint
disorders
However, there is no explicit correlation between
the incidence of mandibular resorption and the
severity, progression, and duration of SS [18].
Regarding oral health, patients with SS
have higher rates of plaque, bleeding on probing,
caries, and periodontal disease. The thickening of
the periodontal ligament occurs in at least two-
thirds of cases of scleroderma and is considered
one of the rst radiographic signs that can help
in the diagnosis of the disease, being associated
with the increase in collagen production
that occurs throughout the body [19,20].
Iordache et al. (2019) [19] veried, using CBCT,
that periodontal ligament thickening is present
in at least one tooth in each patient diagnosed
with scleroderma, with the posterior teeth being
the most affected [19]. In periapical radiographs,
this thickening was 0.16 mm, twice the thickness
considered normal [19].
Chebbi et al. (2016) [21], in a cohort
study, observed that 30% of patients had
TMJ pain, however, 90% exhibited normal
radiological aspects, whereas only 10%
had condylar erosion [21], therefore, TMJ
pain does not necessarily indicate condylar
resorption [22]. Two observational studies
demonstrated that patients diagnosed with
SS tend to present more signs and symptoms
of temporomandibular disorders than healthy
patients [17,23]. A relationship was observed
between the presence of displacement of discs
without reduction and bone alterations in
patients with SS, possibly, this bone alteration
comes from the regular absence of the disc
between the condyle and temporal eminence
during mandibular movement, which over
time may determine alterations in the patient’s
sinus cavity, but this nding is not exclusive to
SS [17]. Moreover, the condyle reabsorption
seems to be affected by microvasculopathy and
pressure ischaemia, due to skin and musculature
atrophy [23].
The application of non-surgical treatments
for temporomandibular disorders remains the
most effective method of choice in the treatment
of the patient, the treatments described in the
literature include physiotherapy, application
of botulinum toxin, behavioral therapy, patient
re-education and awareness, and therapy with
occlusal device [24]. Surgical treatment is
Figure 5 - A) Individual tray made of chemically activated acrylic resin for the left and right hemi-arch; B) Partial impression taken with
individual acrylic resin trays; C) Partial impression of the left hemi-arch; D) Stabilizing plate complete; E) Stabilizer plate in position.
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Braz Dent Sci 2023 Oct/Dec.;26 (4): e3998
Torquato LC et al.
Systemic scleroderma: imaging findings of diagnosis and clinical management of temporomandibular joint disorders
Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
clinical management of temporomandibular joint disorders
Systemic scleroderma – case report: imaging findings of
diagnosis and clinical management of temporomandibular joint
disorders
chosen as the last option, in the present case,
considering the systemic status of the patient,
non-surgical treatment was chosen, for which
therapy with an occlusal splint was performed.
The main idea, when using occlusal splints, is
to reduce the maximum load on the TMJ, in
addition to avoiding the maximum contraction of
the masticatory muscles, which will promote pain
reduction. In the presence of a diagnosis of SS, the
limitation of mouth opening found in patients is
a limiting factor for the prostheses manufacture
and plates for bruxism. Therefore, it is indicated
the manufacture of partial prostheses for easy
removal by the patient [20].
Like Matarese et al. (2016) [17], the present
case report demonstrates the need to apply
imaging tests of different modalities to study
the involvement of the temporomandibular
joints in patients diagnosed with SS. Although
studies report that the present syndrome affects
the TMJ, no studies were found in the literature
that used different imaging modalities to describe
these alterations in the TMJs. Ideally, the dental
treatment of patients diagnosed with scleroderma
should be conducted individually and immediately
after diagnosis. As suggestions for future work,
texture analyses of the temporomandibular
region could provide a more descriptive our
early data about TMJ involvement in SS, since
it was able to determine degenerative changes
in the temporomandibular region in patients
with migraine, once changes were related to the
masticatory muscles and internal derangements
of the articular disc [25].
CONCLUSION
The present case report indicates the
importance of application of different modalities
of imaging to assess TMJ degeneration and
achieve an accurate diagnosis, decisions taken
have a significant impact on the health and
quality of life of patients in these conditions, so
there is a need for multidisciplinary involvement
to arrive at the best treatment plan. After ve
years of using the stabilizing plate at night,
the patient reports greater comfort and muscle
relaxation upon waking up.
Author’s Contributions
LCT, ACM: Conceptualization. LCT:
Methodology. CCMM, NDA, WO, ABMF, SLPCL:
Methodology. CCMM, NDA, WO: Writing –
Original Draft Preparation. MANJ, ABMF,
SLPCL, ACM: Writing – Review & Editing. ACM:
Visualization. ACM: Supervision. ACM: Project
Administration.
Conict of Interest
The authors declare that they have no
conicts of interest.
Funding
The authors did not receive support from
any organization for this work.
Regulatory Statement
This study was submitted and approved by
the Research Ethics Committee (CEP)/Plataforma
Brasil has approved this study under number:
2.002.426.
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Torquato LC et al. Systemic scleroderma: imaging findings of diagnosis and
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Andréa Carvalho De Marco
(Cooresponding address)
Universidade Estadual Paulista, Instituto de Ciência e Tecnologia, Departamento de
Diagnóstico e Cirurgia, Divisão da Periodontia, São José dos Campos, SP, Brasil.
Email: andrea.marco@unesp.br
Date submitted: 2023 Aug 14
Accept submission: 2023 Sept 27
