Oral lichen sclerosus et atrophicus
A case report
Waranun Buajeeb, BSc, DDS, MSc (Oral Medicine),a Petcharat Kraivaphan, BSc, DDS, MSc,b
Jirapa Punyasingh, BSc, DDS, MSc,c and Penpan Laohapand, BSc, DDS, MDS,a Bangkok,
Thailand
MAHIDOL UNIVERSITY
Lichen sclerosus et atrophicus affecting only the oral mucosa is extremely rare. We report here a case of oral lichen
sclerosus et atrophicus presenting as a white, flat lesion involving the right buccal and labial mucosa and vermillion border.
The diagnosis was based on histopathologic features. Treatment with intralesional corticosteroid was successful in reducing the
size of the lesion and the symptoms of the patient. A free gingival graft was also performed to restore the lost attached gingiva.
No recurrence of the lesion was found after a 1-year follow-up period, and no skin or genital lesions developed during the 3
years of treatment. (O ral Surg O ral M ed O ral Pathol O ral Radiol Endod 1999;88:702-6)
Lichen sclerosus et atrophicus (LSA) is a rare mucocutaneous disease of unknown etiology. It is characterized by ivory or porcelain-white, round, shiny macules
or papules that generally form plaques. Atrophy and
sclerosis of the skin and mucous membrane are
common. In addition, telangiectasia is frequently
found. Genital and perianal involvement are the most
prevalent areas of involvement1; however, the condition may be seen in any part of the body.
In 1887, Hallopeau2 first described the disease as
“lichen planus atrophicus.” Later, in 1892, Darier3
reported the histopathologic features of a similar lesion
called “lichen planus sclerosus.” Although LSA was
initially considered to be a variety of lichen planus,2,3
it has recently been regarded as a distinctive disease
with specific clinical and histopathologic features.4,5
LSA involving the oral mucosa is extremely rare. It
can affect the buccal and labial mucosa, gingiva, palate,
or vermillion border, with or without skin or genital
lesions. Only 19 cases have been reported in the literature.6-14 Of these, 5 cases were histologically proven
oral LSA without any skin or genital lesions after a
follow-up period (Table I). The purpose of this report is
to document a new case of LSA involving only the oral
mucosa and to discuss the treatment of this disorder.
CASE REPO RT
A 22-year-old Thai woman was referred by her dentist to the
Department of Oral Medicine, Faculty of Dentistry, Mahidol
aAssociate
Professor, Department of Oral Medicine, Faculty of
Dentistry.
bAssociate Professor, Department of Pharmacy, Faculty of Dentistry.
cAssociate Professor, Department of Oral Pathology, Faculty of
Dentistry.
Received for publication Mar 1, 1999; returned for revision Apr 8,
1999; accepted for publication May 25, 1999.
Copyright © 1999 by Mosby, Inc.
1079-2104/99/$8.00 + 0 7/14/100426
702
Fig 1. Clinical appearance shows white lesion on buccal
mucosa and mucobuccal fold.
University, Bangkok, Thailand, for examination and treatment
of a white lesion on her oral mucosa. Her chief complaints
were slight tightness on opening at the right buccal mucosa
and soreness of the mandibular right gingiva on tooth brushing.
Her medical history disclosed that she had had asthma since
childhood; the last attack was 6 years previously. The patient
was otherwise well, and she was taking no medication. She did
not drink alcohol, smoke tobacco, or chew areca nut. No
history of trauma or surgery in the area of the mandibular right
buccal mucosa and gingiva was disclosed.
Clinical examination revealed a white macular lesion on the
patient’s right buccal mucosa and mucobuccal fold from the
distal aspect of the mandibular right second molar to the labial
aspect of the mandibular right canine, extending anteriorly onto
the labial mucosa and vermillion border of the lower lip (Fig 1).
The lesion was approximately 2 × 7 cm in size, and on palpation it was firmer than the surrounding areas. The patient’s oral
hygiene was good; she had mild generalized gingivitis except
for the mandibular right quadrant. Gingival recession and
moderate inflammation were noted at the buccal and lingual
aspects of the mandibular right second molar to canine. No
Buajeeb et al 703
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
Volume 88, Number 6
Fig 2. Photomicrograph shows hyperparakeratosis, hydropic degeneration of basal cells, subepithelial hyalinization, and underlying inflammatory cell infiltrate (hematoxylin-eosin, original magnification ×100).
Table I. Histologically verified reported cases of lichen sclerosus et atrophicus involving oral mucosa only
Author(s)
Year of publication
Patient’s sex
Patient’s age (y)
Oral site(s)
Ravits7
Macleod and Soames12
Schulten et al13
1957
1991
1993
Brown et al14
1997
M
F
F
M
M
24
57
59
12
44
Buccal mucosa and gingiva
Tongue and palate
Labial mucosa, commissure and tongue
Labial mucosa
Soft palate midline
attached gingiva was found at the buccal side of these teeth; this
caused movement of the free gingiva during mucosal retraction.
There was no evidence of genital or skin lesions.
Radiographic examination showed normal findings except
for a thickening of the periodontal ligament space and lamina
dura at the mandibular right first molar. Laboratory investigations revealed a hemoglobin level of 12.8 g/dL (12-16 g/dL),
a white blood cell count of 7.6 × 109/L (5.0-10.0×109/L), and
a normal platelet count. Liver function was within normal
limits. The patient’s antinuclear antibody titers were negative,
with normal levels of IgG, IgA, and IgM.
H istopathologic findings
An incisional biopsy of the lesion was performed on the
labial mucosa with the patient under local anesthesia.
Examination of sections stained with hematoxylin and eosin
revealed a mucosal strip with minor salivary glands. The
epithelium showed atrophy and focal areas of hyperparakeratosis. Hydropic degeneration of the basal cells and intracellular edema of the spinous layers were noted occasionally.
The underlying connective tissue revealed hyalinization of a
band of the connective tissue with inflammatory cells and
vessels (Figs 2 and 3). Telangiectasia of the vessels was also
noted. Immunofluorescent staining of the section was negative for immunoglobulins G, M, and A as well as C3 and
fibrinogen. A section stained by Verhoeff’s method showed
scantiness of elastic fibers in the superficial connective tissue.
It was concluded that the histopathologic features were
consistent with a diagnosis of LSA.
Treatment
Scaling, root planing, and oral hygiene procedures were
performed regularly; the patient herself could not clean her
mandibular right teeth properly because of soreness during
tooth brushing. The patient asked for relief of tightening at the
right buccal mucosa. Topical triamcinolone acetonide 0.1%
(Kenalog in Orabase) was prescribed; it was applied 4 times
daily for approximately 2 months, without any evidence of
regression of the lesion. Therefore, methylprednisolone
acetate 40 mg/mL (Depo-Medrol 40) was used, 0.1 mL per 1
cm2 being injected on divided areas. The lesion was injected
in 3 or 4 areas each time; this made for a series of 5 treatments
704 Buajeeb et al
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
December 1999
Fig 4. After free soft tissue grafting, attached gingiva has been
gained at buccal aspect of teeth in right mandibular quadrant.
Fig 3. Photomicrograph shows hyperparakeratosis, intracellular edema of spinous cell layer, and hyalinization of
collagen in upper lamina propria (hematoxylin-eosin, original
magnification ×200).
with 1-month intervals. Subsequent examinations showed
regression in the size of the lesion on the buccal mucosa and
disappearance of the white lesion on the labial mucosa. On
palpation, the lesion was softer than it had been previously.
The patient felt less tightening at the right buccal mucosa;
however, soreness and inflammation of the mandibular right
gingiva still existed. Therefore, a free soft tissue graft (from
the palatal mucosa) was performed at the buccal aspect of the
mandibular right second molar to canine to restore the lost
attached gingiva. After grafting, a significant increase in
attached gingiva was achieved (Fig 4) and the patient could
brush her teeth properly without soreness of the area.
D ISCU SSIO N
According to the reports in the literature, the prevalence of LSA affecting the oral mucosa alone is extremely rare. Five cases with histologically verified oral LSA
have been reported.7,12-14 No skin or genital lesions developed after the follow-up period in any of the reported
cases or in our case. This suggests that oral LSA may
appear without accompanying skin or genital lesions.
Oral LSA has been described on labial and buccal
mucosa, lip, gingiva, palate, tongue, and anterior tonsillar
pillar. The lesions manifest as white macules or
plaques,6-9,13,14 with reticular striations6,10 and superficial ulceration.11,12 Clinically, it may be difficult to
distinguish oral LSA from other oral white lesions, especially lichen planus,6,9,10 leukoplakia,15 submucous
fibrosis,10 and localized scleroderma.15 Oral lichen
planus usually presents as white reticular striations. A
minority of patients with oral lichen planus develop
dense, white, homogeneous plaques.16 Oral leukoplakia
shows a variety of appearances, ranging from fine white
flecks to dense thick plaques.16 It may be seen in smokers
and users of smokeless tobacco. In the present case, the
firm white macule was difficult to differentiate clinically
from submucous fibrosis and localized scleroderma.
However, the patient had no history of chewing areca nut
and the lesion was localized; submucous fibrosis could
therefore be excluded clinically.
LSA is asymptomatic, an exception being the case
reported by Siar and Ng,10 in which the symptoms
probably resulted from atrophy of the lesion. Except
for slight tightening and soreness at the adjacent buccal
gingiva during tooth brushing, our patient was asymptomatic. Both of her symptoms could be explained by
sclerosis of the lesion. The lack of attached gingiva and
the inflamed marginal tissue also caused the soreness.
The histologic features of cutaneous LSA are characteristic, including follicular plugging, atrophy of the
epidermis with vacuolar degeneration of the basal
layer, edema, homogenization of the collagen and
scantiness or loss of elastic fibers in the upper dermis,
and an inflammatory infiltrate in the mid dermis.5 The
histologic features of oral LSA are quite similar to
those of LSA of the skin. De Araujo et al9 have
concluded that the characteristic features of oral LSA
are pronounced edema and homogenization of the
Buajeeb et al 705
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
Volume 88, Number 6
Table II. Comparison of histologic features of oral mucosal lesions with similar clinical appearances
Oral lesions
Epithelium
Basal cell
liquefaction
LSA
Lichen planus
Leukoplakia
Submucous fibrosis
Scleroderma
Hyperkeratosis to atrophy
Hyperkeratosis to atrophy
Hyperkeratosis to atrophic parakeratosis
Atrophy
Atrophy
+/–
+
–
–
–
collagen fibers within the lamina propria, together with
hydropic degeneration of the basal cells. The density of
the mononuclear infiltrate is greater in early lesions
than in longstanding lesions.17 In addition, the younger
the lesion, the more superficial the infiltrate.5
It is sometimes difficult to differentiate oral LSA
from oral lichen planus, oral submucous fibrosis, and
oral scleroderma by means of histologic features.
However, the distinct band of lymphocytes at the
epithelial-connective tissue junction that is usually
found in lichen planus is not evident in LSA.18
The distinction from oral submucous fibrosis is more
difficult. However, pyknotic changes in the nuclei of
the basal cell layer,19 epithelial atypia,20 and obliteration or narrowing of the blood vessels21 are not
observed in LSA.
Unlike scleroderma, in which the dominant pathologic process involves deep dermis, LSA involves only
the superficial dermis.17 Furthermore, scantiness or
loss of elastic fibers, characteristic of LSA, is not found
in scleroderma.22 Collagen biosynthesis is increased in
scleroderma but decreased in LSA.23
Although our case clinically resembled oral submucous fibrosis, the histopathologic features were used to
differentiate oral LSA from oral submucous fibrosis;
furthermore, telangiectasia and the scantiness of elastic
fibers localized in the upper lamina propria were used
to differentiate oral LSA from scleroderma. Therefore,
histologic findings were critical in making a diagnosis
of oral LSA. With respect to some histologic features,
oral LSA is compared with other oral white lesions in
Table II.
The etiology of LSA is unknown, but many investigators believe the condition to be autoimmune in nature. It
was found to be strongly correlated with autoimmune
diseases such as thyroiditis, insulin-dependent diabetes
mellitus, and vitiligo.24 Direct immunofluorescent
studies may reveal immunoglobulin G, C3, and
fibrinogen at the basement membrane level.25,26 In our
case, negative immunofluorescent staining was
reported. In addition, the patient had no autoimmune
diseases. The histopathologic findings were consistent
with the diagnosis of LSA. Other possible causes, such
Band of lymphocytes at Scantiness
epithelium connective
or loss
tissue junction
of elastic fibers
–
+
–
–
–
+
–
–
–
–
Obliteration
or narrowing
of blood vessels
–
–
–
+
+
as traumatic, genetic, and hormonal factors, were not
found to be related in this case.
Treatment of oral LSA is usually unnecessary because
of its asymptomatic nature. Topical application of corticosteroids has been reported, the outcome being variable.10,12 In our patient, the efficacy of intralesional corticosteroids was demonstrated. Gingival recession
resulting from oral LSA affecting the gingiva, which was
never before reported, is probably caused by sclerosis of
the adjacent buccal mucosa. We succeeded in grafting the
attached gingiva on the buccal aspect of the mandibular
right second molar to canine, which helped to restore the
gingival health because the patient could then clean her
teeth properly without soreness. There was no evidence
of recurrence of the lesion after 3 months of follow-up.
No skin or genital lesions developed during 3 years of
follow-up.
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ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
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Reprint requests:
Waranun Buajeeb, BSc, DDS, MSc
Department of Oral Medicine, Faculty of Dentistry,
Mahidol University
6 Yothi Street, Rachathavee, Payathai
Bangkok, Thailand 10400
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