Clinical cases

Oral lesions in adult- and juvenile-onset systemic lupus erythematosus patients:

A case series report
Firstine Kelsi Hartanto1,2,A–F, Irna Sufiawati3,C,E,F
1
Oral Medicine Residency Program, Faculty of Dentistry, Universitas Padjajaran, Bandung, Indonesia
2
Oral Medicine Department, Faculty of Dentistry, Universitas Trisakti, Jakarta, Indonesia
3
Oral Medicine Department, Faculty of Dentistry, Universitas Padjajaran, Bandung, Indonesia

A – research concept and design; B – collection and/or assembly of data; C – data analysis and interpretation;
D – writing the article; E – critical revision of the article; F – final approval of the article

Dental and Medical Problems, ISSN 1644-387X (print), ISSN 2300-9020 (online) Dent Med Probl. 2024;61(1):145–152

Address for correspondence

Firstine Kelsi Hartanto
Abstract
E-mail: [email protected] Systemic lupus erythematosus (SLE) is an autoimmune disease with various oral manifestations, including
ulceration, white keratotic plaques, oral discoid lupus erythematosus, oral lichen planus (OLP)-like lesions,
Funding sources
None declared
non-specific erythema, purpura, petechiae, and cheilitis, which resemble lesions of other systemic
diseases. Recognizing the oral manifestation of SLE is essential for comprehensive patient management.
Conflict of interest This study reports 4 cases of SLE with various oral lesions, underlying conditions and diagnostic methods.
None declared
In September 2019, 2 adult SLE patients and 2 juvenile SLE patients were consulted at the Oral Medicine
Acknowledgements Clinic. The assessment of systemic diseases was conducted by the Internal Medicine and Pediatrics
None declared resident, whereas the Oral Medicine resident performed the intraoral examinations. The medical history,
clinical findings and laboratory results were analyzed to establish the diagnosis.
Received on September 21, 2020 The first patient was a 38-year-old female presenting with multiple white keratotic plaques throughout
Reviewed on November 26, 2020
Accepted on January 7, 2021
the mucosa, an OLP-like lesion on the right buccal mucosa, petechiae on the hard palate, and petechiae
and purpura on the upper and lower extremities. The second case was a 24-year-old female with a malar
rash and multiple ulcerations on the vermilion zone, an OLP-like lesion on the left buccal mucosa, and
Published online on February 29, 2024
a palatal ulcer. The third and fourth cases were 16-year-old females with a prominent butterfly rash.
The patients presented with acute pseudomembranous candidiasis, an aphthous-like ulcer and keratotic
plaques. They received antimicrobial therapy for the intraoral lesions and showed promising results.
The oral lesions in adult- and juvenile-onset SLE patients varied depending on the disease severity and
treatment received.
Keywords: systemic lupus erythematosus, oral lesions, adult SLE, juvenile SLE, oral ulcer

Cite as
Hartanto FK, Sufiawati I. Oral lesions in adult- and
juvenile-onset systemic lupus erythematosus patients:
A case series report. Dent Med Probl. 2024;61(1):145–152.
doi:10.17219/dmp/132242

DOI
10.17219/dmp/132242

Copyright
Copyright by Author(s)
This is an article distributed under the terms of the
Creative Commons Attribution 3.0 Unported License (CC BY 3.0)
(https://creativecommons.org/licenses/by/3.0/).
146 F.K. Hartanto, I. Sufiawati. Oral lesions in adult and juvenile SLE

Introduction disorders, photosensitivity, butterfly rash, and mucosal

ulceration are more common in children than in adults,
Lupus erythematosus (LE), commonly referred to as whereas neurological symptoms and polyarthritis occur
systemic lupus erythematosus (SLE), is an autoimmune more frequently in adults.14 The present article describes
disease that can affect multiple organs and present with 4 clinical cases of various oral lesions identified in patients
various clinical manifestations.1,2 The incidence of SLE with SLE, along with their diagnosis and management.
has increased in recent years, with reported rates of 2–8
cases per 100,000 individuals in Europe, South America
and North America, 51 cases per 100,000 individuals in Material and methods
the USA,1 and 30–50 cases per 100,000 individuals in
Asia, where the incidence varied from 0.9/100,000 to This article is a case series study with a prospective de-
3.1% per year. The epidemiological data on SLE differs sign conducted in a single center using consecutive sam-
between Asian countries and is difficult to generalize. pling. The patients were admitted to Dr. Hasan Sadikin
However, there are similarities in the clinical presenta- Central General Hospital (Bandung, Indonesia), a gov-
tion of the disease.3 In 2010, 291 SLE patients were reg- ernment-run academic and community hospital. An In-
istered at the Rheumatology Clinic of Dr. Hasan Sadikin ternal Medicine and Pediatrics resident, supervised by
Central General Hospital (Bandung, Indonesia), ac- their consultant, performed the examinations of systemic
counting for 10.5% of all patients registered at the Rheu- conditions and laboratory assessments needed to support
matology Clinic.1 the diagnosis. At the same time, an Oral Medicine resi-
In 1997, the American College of Rheumatology (ACR) dent conducted intraoral examinations under the super-
issued a set of diagnostic criteria for SLE, which were vision of their consultant. The final diagnosis was based
revised in 2012 by the Systemic Lupus Erythematosus on patient complaints, clinical (extraoral and intraoral)
International Collaborating Clinics (SLICC) to be more observations and laboratory findings. The clinicians pro-
sensitive but less specific.4 These criteria are used to diag- vided medication for systemic diseases, whereas intraoral
nose SLE in children and adults. Several studies reported lesions were treated by oral medicine specialists. Patient
that oral lesions in SLE varied from 9% to 45% and from improvement was followed up for 3–4 weeks and any
3% to 20% in localized cutaneous disease.5–7 In 2019, the changes in medication type or dosage were reported. The
European League Against Rheumatism (EULAR) and the results of the examinations, diagnosis, treatment, and oral
ACR approved new criteria for SLE, which require a posi- lesion progress were documented in the patient’s medical
tive antinuclear antibody (ANA) test. The new criteria records. The patients provided consent to document the
have 96.1% sensitivity and 93.4% specificity, compared intraoral lesions for further evaluation.
with 82.8% sensitivity and 93.4% specificity of the ACR
1997 criteria and 96.7% sensitivity and 83.7% specificity
of the SLICC 2012 criteria.8 Results
Many terms have been used to describe the LE oral
lesion, including oral discoid lesion, chronic plaque, lu- The case series involved 4 female patients admitted
pus cheilitis, acute ulcer, oral ulcer, red ulcer, ulcerative to Dr. Hasan Sadikin Central General Hospital between
plaques, pebbly red area, honeycomb lesion, keratotic le- September 2019 and December 2019. All patients were
sion, white keratotic plaques, purpuric lesion, and diffuse of reproductive age. Table 1 describes the clinical char-
palatal petechial erythema.9–13 A study in the Hungarian acteristics of each patient, with a butterfly (malar) rash
population reported that lupus nephritis, hematological and extraoral pale conjunctiva being the most common.

Table 1. Clinical features present in patients with systemic lupus erythematosus (SLE)

Variable Case 1 Case 2 Case 3 Case 4

Age
38 24 16 16
[years]
Gender female female female female
• butterfly rash
• pale conjunctiva • butterfly rash
Extraoral • pale conjunctiva
• petechiae and purpura on the • pale conjunctiva • butterfly rash
findings • alopecia
upper and lower extremities • swelling and ulceration of the lips
• exfoliation of the vermilion zone
• keratotic plaques • multiple ulcerations • acute pseudomembranous
Intraoral • non-specific petechiae • acute pseudomembranous candidiasis • multiple ulcerations
findings • unpainful ulceration surrounded candidiasis • unpainful ulceration surrounded • keratotic plaques
by whitish striae (OLP-like lesion) • central palatal erythema by whitish striae (OLP-like lesion)

OLP – oral lichen planus.

Dent Med Probl. 2024;61(1):145–152 147

Intraoral manifestations ranged from aphthous-like ul-

cers, keratotic plaques and oral lichen planus (OLP)-like
lesions to central palatal erythema and acute pseudo-
membranous candidiasis. All patients were diagnosed
with SLE according to the SLICC criteria, which are sum-
marized in Table 2.
The first patient presented with secondary Evans syn-
drome, and the diagnosis of SLE was confirmed following
intraoral examination and ANA testing, which indicates
that intraoral findings may play a role in establishing a de-
finitive diagnosis. Extraoral findings included petechiae
and purpura on the upper and lower extremities, and pale
conjunctiva (Fig. 1A,B). Intraoral lesions included kera-
totic plaques on the upper and lower labial mucosa and
the left and right lateral border of the tongue that could
not be scraped (Fig. 1C–F), unpainful lesions on the right
buccal mucosa surrounded by whitish reticular plaque
(OLP-like lesions) (Fig. 1G), whereas the left buccal mu-
cosa showed no striae (Fig. 1H). Multiple petechiae were
also observed on the hard palate. The patient received the
treatment listed in Table 3, was discharged 5 days later,
and continued as an outpatient at a hospital closer to her
hometown.
The second patient was diagnosed with SLE in April
2009 at another hospital, and was undergoing SLE treatment.

Table 2. Systemic Lupus Erythematosus International Collaborating Clinics

(SLICC) criteria used for the patients’ diagnosis

SLICC criteria Case 1 Case 2 Case 3 Case 4

acute cutaneous
yes yes yes yes
lupus
chronic cutaneous
no no no no
lupus
oral or nasal ulcers yes yes yes yes
non-scarring Fig. 1. A,B. Petechiae and purpura on the upper and lower extremities;
no no yes no C,D. Non-scrapable whitish plaques on the upper and lower labial mucosa;
alopecia
Clinical E,F. Non-scrapable whitish plaques on the left and right lateral border
arthritis no no yes yes
criteria of the tongue; G. Unpainful ulceration on the right buccal mucosa
serositis no no no no surounded by whitish striae (oral lichen planus (OLP)-like lesion);
H. Unpainful ulcer with whitish keratotic plaques
renal no no no no
neurologic no no no no
hemolytic anemia yes yes yes yes She complained of swelling and ulceration of the lips.
leukopenia yes yes yes yes A few months earlier, she had similar symptoms after
thrombocytopenia yes yes yes yes taking an antibiotic, which was confirmed to be an al-
ANA reactive reactive reactive reactive lergic reaction. The patient stated that the current swell-
not not not not
ing was not related to any medication. Extraoral findings
anti-DNA revealed pale conjunctiva, a butterfly rash (Fig. 2A) and
done done done reactive
not not not not swelling of the lips associated with multiple minor ul­
anti-Sm
done done done done cerations on the lower labial mucosa (Fig. 2B). Intraoral
Immunologic
criteria antiphospholipid not not not not examination showed multiple ulcerations on the upper
antibody done done done done and labial mucosa as well as the left and right buccal
low complement not not not not mucosa. Additionally, acute pseudomembranous can­
(C3, C4, CH50) done done done done didiasis (Fig. 2C) and a central erythematous lesion
direct Coombs not not not not on the hard palate intermixed with whitish pseudo­
test done done done done membranous plaques (Fig. 2D) were observed. The differen­
ANA – antinuclear antibody; anti-Sm – anti-Smith. tial diagnosis consisted of herpes-associated erythema
148 F.K. Hartanto, I. Sufiawati. Oral lesions in adult and juvenile SLE

multiforme and drug-induced erythema multiforme.

Herpes simplex virus-1 (HSV-1) and immunoglobulin E
(IgE) serology were performed to rule out any possibility
of hypersensitivity reaction and HSV involvement. The
results for IgE were within the normal range and serol-
ogy for HSV-1 was non-reactive. Therefore, intraoral
HSV infection was excluded. Table 3 summarizes the
patient’s medications.
One week later, the lip swelling, ulceration and oral can-
didiasis (Fig. 2E,F) subsided. However, an unpainful cen-
tral erythematous lesion on the hard palate (Fig. 2G) was
revealed. Additionally, whitish radiant striae with central
ulceration resembling OLP were observed on the left buc-
cal mucosa (Fig. 2H). The patient was instructed to con-
tinue using the antimicrobial gel for the lesion on the pal-
ate and left buccal mucosa and petroleum jelly to reduce
lip dryness. Selective grinding was performed on teeth 27
and 36 to minimize traumatic contact with the ulcer on
the left buccal mucosa.
The third patient reported difficulty eating due to mul-
tiple ulcerations in the mouth. She had been treated for
pulmonary tuberculosis for the past 2 months. However,
a thoracic X-ray examination revealed that the pulmonary
tuberculosis was not active and there was no cardiomegaly.
Upon examination, we observed a prominent butterfly
rash (Fig. 3A), pale conjunctiva, alopecia, dryness, and
exfoliation of the vermilion. Intraoral findings revealed
multiple scrapable white patches, leaving an erythema-
tous base on the lower labial mucosa, hard palate and left
buccal mucosa (Fig. 3B–D). A thick coating was present
on the dorsum of the tongue (Fig. 3E). Laboratory find-
ings showed hemolytic anemia, elevated blood glucose,
serum glutamic-oxaloacetic transaminase (SGOT), urea,
and creatinine levels, and a reactive ANA test. Table 3 lists
the patient’s medications.
Two weeks after treatment, the patient’s condition im-
proved. The coated tongue showed marked improvement
(Fig. 3F), the dryness of the vermilion decreased (Fig. 3G),
and the oral candidiasis was reduced and almost com-
pletely resolved (Fig. 3H–J). The patient was discharged
and scheduled for regular control at the Pediatric Depart-
Fig. 2. A. Butterfly rash; B. Swelling on the lower labial mucosa associated ment, having followed the instructions for maintaining
with multiple ulcerations; C,D. Acute pseudomembranous candidiasis on
the dorsum of the tongue and palate; E. Lip swelling subsided; F. Whitish oral hygiene.
plaques disappeared; G. Central erythematous lesion on the hard palate; H. The fourth patient reported experiencing ulcerations in
Whitish radiant striae with central ulceration resembling oral lichen planus the mouth, pain while swallowing and pain inside the ear
on the left buccal mucosa

Table 3. Treatment of patients with systemic lupus erythematosus (SLE)

Case 1 Case 2 Case 3 Case 4

• systemic corticosteroid • antifungal oral suspension • intravenous corticosteroid
• calcium carbonate • antimicrobial oral gel • antibiotics
• intravenous corticosteroid
• pulse dose intravenous • petroleum jelly • calcium carbonate
• calcium carbonate
dexamethasone • calcium carbonate • vitamin D3
• antifungal oral suspension
• omeprazole • vitamin D3 • skin topical corticosteroid
• oral hygiene instructions
• oral topical corticosteroid • selective grinding • SPF 45 sunblock
• antimicrobial mouthwash (teeth 27 and 36) • antimicrobial mouthwash

SPF – sun protection factor.

Dent Med Probl. 2024;61(1):145–152 149

for 2 days before admission. The patient has been receiv- rash was observed on her face (Fig. 4A). Intraorally, mi-
ing treatment for SLE with skin and musculoskeletal in- nor ulcerations surrounded by an erythematous area and
volvement since March 2019. On examination, a butterfly a regular border were found on the lower labial mucosa
opposite tooth 32 (Fig. 4B). Multiple faint, non-scrapable
whitish plaques were observed at the anterior part of the
left and posterior part of the right buccal mucosa (oppo-
site teeth 44 and 46), and on the right and left lateral bor-
ders of the tongue (Fig. 4C–E). The patient’s treatment is
described in Table 3. Five days later, the ulceration on the
lower labial mucosa disappeared, but the white keratotic
plaques on the tongue and the faint whitish plaques on
the right buccal mucosa persisted.

Fig. 3. A. Butterfly rash; B. Multiple scrapable white patches that leave

an erythematous base on the lower labial mucosa; C,D. Multiple scrapable Fig. 4. A. Butterfly rash; B. Minor ulcer on the lower labial mucosa; C,D.
white patches that leave an erythematous base on the hard palate and Multiple faint non-scrapable whitish plaques on the right and left lateral
left buccal mucosa; E. Thickly coated tongue; F. Coated tongue improved; borders of the tongue; E. Multiple faint non-scrapable whitish plaques
G. Dryness of the vermilion decreased; H. Whitish plaques decreased; I,J. (marked with a circle) and a minor ulcer (marked with an arrow) on the
Whitish plaques almost completely resolved right buccal mucosa
150 F.K. Hartanto, I. Sufiawati. Oral lesions in adult and juvenile SLE

Discussion oral lichenoid reactions, which are usually associated

with an adjacent metallic dental restoration. A biopsy
Oral ulceration can be an indicator of various system- of the lesion is required when the clinical presentation
ic diseases, such as LP, LE, benign mucous membrane alone cannot establish a definitive diagnosis.22 Howev-
pemphigoid, pemphigus vulgaris, Crohn’s disease, and er, in our study, a biopsy could not be performed in the
Behçet’s syndrome. The ulcerations may present in vari- first patient due to hemolytic anemia and thrombocyto-
ous ways but maintain their characteristic features. In penia, while the second patient refused to undergo the
LE, oral lesions may present as oral discoid lesions, ery- procedure. Moreover, the OLP-like lesions observed in
thema, irregularly shaped ulcers, honeycomb plaques, our SLE patients were painless, unlike the erosive OLP,
raised keratotic plaques, purpura, petechiae, and chei- which may cause symptoms and interfere with patient
litis.15 These lesions may also be accompanied by other activities.
organ involvement or present as a solitary lesion. There- Lip swelling may resemble angioedema, but it does not
fore, it is crucial for clinicians to recognize the charac- need to be accompanied by multiple ulcerations as ob-
teristic presentation of the disease to achieve an accu- served in the second case. Erythema multiforme associ-
rate diagnosis. ated with HSV or induced by some drugs was also consid-
The first classifications of mucocutaneous SLE in the ered as a differential diagnosis due to the patient’s recent
1970s were divided into lupus-specific skin lesions (LE- history of similar lip swelling associated with antibiotic
specific) and non-specific skin lesions (LE-non-specific) treatment. However, we excluded the possibility of a hyper­
and are used in both adult- (adult SLE) and juvenile-onset sensitivity reaction due to the normal range of the se­
SLE (JSLE).16 The majority of the manifestations appear rology test and the absence of causative antibiotic or drug
similarly in both groups. A study reported that the LE- consumption. Fortunately, the swelling subsided within
specific butterfly rash and the generalized lupus rash were a few days and the ulcerations, which may have been non-
more frequent in JSLE than in adult SLE.17 In contrast, specific, healed.
adult SLE patients were more likely than JSLE patients The third patient did not have any SLE oral lesions, ex-
to present with subacute cutaneous lesions, discoid rash, cept for oral candidiasis. Multiple risk factors have been
generalized discoid LE (DLE), and LE panniculitis (pro- identified for oral candidiasis in SLE patients. A study rec-
fundus). In LE-non-specific cases, cutaneous vasculitis, ommends examining for oral candidiasis in those with ac-
oral and nasal ulcers, and bullous SLE were more common tive disease, proteinuria, high white blood cell count, and
in JSLE patients, whereas photosensitivity, non-scaring those taking prednisone, immunosuppressive agents or
alopecia, livedo reticularis, and Raynaud’s phenomenon antibiotics.23 In our study, the third patient had protein-
were more frequent in adult SLE.17 uria, was taking prolonged methylprednisolone and anti-
The most common LE-specific lesion in JSLE and biotics for SLE, and had pulmonary tuberculosis. There-
adult SLE is the butterfly rash. It presents as a symmetri- fore, he was at increased risk of opportunistic infection,
cal erythematous and edematous non-pruritic rash over such as oral or oropharyngeal candidiasis.
the nasal bridge, typically sparing the nasolabial folds. The fourth case demonstrated LE-non-specific aph-
This rash represents acute cutaneous lupus erythemato- thous oral ulcers. The white keratotic plaque observed at
sus (ACLE).18–20 In the present report, the butterfly rash the lateral border of the tongue may indicate initial ver-
was present in 3 out of 4 cases, with 2 patients reporting rucous LE. This white plaque-like lesion may resemble
a recent diagnosis of SLE within the previous 2 weeks or homogeneous leukoplakia or reticular OLP.24 In 2005,
6 months. the World Health Organization (WHO) defined leuko-
Diagnosing SLE in the first patient was more challeng- plakia as “a white plaque of questionable risk having ex-
ing due to the absence of a butterfly rash. The patient cluded (other) known diseases or disorders that carry no
presented with rashes on the upper and lower extremi- increased risk for cancer”.25 In contrast, there were no
ties, which are not specific to cutaneous LE. However, the changes in the tissue’s physical consistency, which is typi-
presence of oral ulcers and laboratory findings of hemo- cally identified in leukoplakia.
lytic anemia, leukopenia, thrombocytopenia, and a re- In the present report, the diagnosis of SLE adhered to
active ANA test helped establish the correct diagnosis. diagnostic criteria established by the SLICC. The disease
As stated in the literature, secondary Evans syndrome activity was measured using the MEX-SLEDAI (Mexican
is linked to suspected underlying autoimmune diseases Version of Systemic Lupus Erythematosus Disease
such as SLE, which causes hemolytic anemia, leukopenia Activity Index). The third and fourth cases had index
and thrombocytopenia.21 scores of 17 and 2, respectively. A score greater than 5 in-
In cases of adult SLE, oral ulcers may resemble reticu- dicates an active or flare condition, a score of 2–5 suggests
lar and erosive OLP, which typically present as a white the possibility of a flare, and a score of less than 2 rep-
lacy patch or Wickham’s striae and erythematous, ulcer- resents an inactive condition or remission.26 Regrettably,
ated or erosive mucosa, frequently on the bilateral buccal the disease activity was not evaluated in the first and sec-
mucosa and rarely on the palate. They may also resemble ond cases.
Dent Med Probl. 2024;61(1):145–152 151

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