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Oral bullous lichen planus: Case report and review of management
Archana Patil, Shiva Prasad, L. Ashok, G. P. Sujatha
Abstract
A 34yearold female patient with the chief complaint of burning sensation in the oral cavity associated with generalized pruritis,
scalp and skin lesions diagnosed as Bullous lichen planus and treated with systemic prednisolone, levamisole, benzydamine
oral rinse. Patient is in follow up since 1year and free of lesions. Here we report the case and review current modalities in the
management of oral lichen planus.
Keywords: Bullous lichen planus, pruritis, burning sensation
Introduction
Oral lichen planus (OLP) is a common disorder that affects
stratified squamous epithelium virtually exclusively. It is seen
worldwide, mostly in the fifth to sixth decades of life, and
is twice as common in women than in men. Here we report
case of bullous lichen planus.
Case Report
A 34yearold Indian female patient reported to the
Department of Oral Medicine and Radiology with the chief
complaint of burning sensation of oral cavity since 4days.
Burning sensation was insidious in onset, preceded by
eruption of fluid filled vesicles that used to burst within few
seconds, continuous, severe intensity aggravated on having
spicy food substances and no relieving factors, episodes of
such occurrence of blisters 1015times per day. She was
also associated with generalized body itching and mild fever.
History of recurrent episodes of burning sensation since
8years but this time associated with severity. Her medical
history revealed occurrence of dermal and scalp lesions,
loss of hair, generalized pruritis for which she was taking
treatment by a dermatologist since 10years and presently was
Department of Oral Medicine and Radiology, Bapuji Dental
College and Hospital, Davangere, Karnataka, India
Correspondence: Dr.Archana Patil, Department of Oral
Medicine and Radiology, Bapuji Dental College and Hospital,
Davangere577004, Karnataka, India.
Email:[email protected]
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DOI:
10.4103/0976-237X.103634
Contemporary Clinical Dentistry | Oct-Dec 2012 | Vol 3 | Issue 4
on the following medications: pimecrolimus (pacroma cream
0.01%), halobresol propionate cream (halox cream 0.05%), and
ketoconazole shampoo (Keto Az shampoo). History of anal
hemorrhoids treated 4years back. Her past dental history
revealed that extraction of mobile teeth and gingival flap
surgery was done 8years back.
No significant family history. She was married since 13years
blessed with two children (girls) and stays in a joint family,
often her life was associated with stress and tension as
reported by the patient. Vegetarian by diet. No history of
any deleterious habits. She brushed once daily in horizontal
manner using brush and paste. General physical examination
revealed that she is moderately built and nourished, appeared
depressed and sad. Depression Anxiety and Stress Scale
[DASS] score indicated moderate depression, mild anxiety,
and moderate stress. Scalp had isolated central area of hair
loss, solitary ulcerative lesion measuring about 34cm,
margins were irregular, floor covered with yellow slough,
tender on palpation. Nails present with mild longitudinal
ridging. Black healed pigmented lesions of varying sizes were
evident on legs, hands, and back [Figure1].
On intraoral examination solitary mixed red and white
lesions, irregular in shape, measuring about 23cm
i.r.t right posterior buccal mucosa extending anteriorly
from mesial of 45 posteriorly distal to 48, superiorly
from occlusal plane of 14 inferiorly up to buccal vestibule
i.r.t45464748 regions, surface presented with bullae
filled with clear fluid measuring about 10.8cm,
margins are irregular with pseudo membrane slough in
the posterior buccal mucosal region, surrounding buccal
mucosa was normal [Figure2].
Left buccal mucosa presented with solitary red and
white lesion measuring about 33.5cm irregular in
shape, extending along the line of occlusion extending
superoinferiorly about 1cm on either side, has central
area of erythema surrounded surface by white striae
[Figure2].
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Patil, etal.:Oral lichen planus
Hard palate showed solitary white lesion measuring about
1.51cm irregular in shape, extending anteriorly from
midpalatal raphe posteriorly to line joining distal surface of
1323, white papules, interspersed with striae, surrounding
mucosa was normal.
On palpation inspectory findings were confirmed as tender,
smooth, and nonscrappable.
Gingiva was observed with generalized gingival inflammation,
erythematous, bleeding on probing, tender, lower right side
Figure1: Lesions on scalp and skin
Figure2: Lesions on right and left buccal mucosa
Figure3: Posttreatmenthealed lesions of scalp and buccal mucosa
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Contemporary Clinical Dentistry | Oct-Dec 2012 | Vol 3 | Issue 4
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Patil, etal.:Oral lichen planus
of posterior gingiva had typical white striae. Generalized
pockets were present. Hard tissue examination reveals teeth
15 14 13 12 11 21 22 23 3 24 25 27 48 47 46 45 44 43 42
41 31 32 33 34 35 38. 38 GradeIII mobility. Considering
history and clinical features Bullous lichen planus (LP) was
considered as provisional diagnosis with the differential
diagnosis phemphigus vulgaris, Bullous phemphigoid, mucus
membrane phemphigoid, Lichenoid contact stomatitis.
Pa t i e n t w a s s u b j e c t e d t o i n v e s t i g a t i o n s l i k e
orthopantomogram (OPG), complete hemogram, blood
sugar, cytosmear, and incisional biopsy from right buccal
mucosa. All laboratory findings were within normal
limits, and biopsy confirmed as LP, which showed typical
histopathological features.
She was treated with Tab levamisole (Vermisol) 150mg once
daily for 3days, Tab prednisolone (Wysolone) 10mg 4times
a day for 10days, benzydamine oral rinse 0.15% (Tantum
oral rinse), ranitidine 150mg (Histac EVT) once daily for
10days. On second visit after 10days, patient reported
with 50% reduction in burning sensation and also episodes
of occurrence of bullae was reduced from 1015times/day
to 78times/day. The dose of wysolone was tapered. On
third visit after 15days patient reported with complete
reduction of burning sensation and was enjoying the
taste of food. Episodes of bullae occurrence was 4times
in 15days. On fourth visit after 15days lesions on right
and left buccal mucosa had healed for remaining white
lesions, triamcinolone (caziq) was prescribed for topical
application. On fifth visit her lesions on right and left buccal
mucosa had healed, her scalp lesions too were healed and
erythematous gingiva was much reduced [Figure3]. Patient
was referred to Department of Periodontics for oral hygiene
prophylaxis. Patient is still under follow up since a year
and free of lesions.
Discussion
Oral Lichen planus (OLP) is a common chronic immunological
inflammatory mucocutaneous disorder that varies in
appearance from keratotic (reticular or plaque like) to
erythematous and ulcerative.[1] The history of LP dates back to
1869, when Erasmus Wilson[2] first delineated and named the
disease. In 1895, Thieberg identified the oral lesion.About
1-2% of world population suffer from LP. 1.5%of Indians suffer
from this disorder, age range of occurrence is 3070years,
with female predilection Male:Female1:1.4.[3]
The different etiological factors considered for LP are genetic
background, dental materials, drugs, infectious agent,
autoimmunity, immunodeficiency, food allergy, stress, habits,
trauma, diabetes, hypertension, malignant neoplasm, and
bowel diseases.[4]
Contemporary Clinical Dentistry | Oct-Dec 2012 | Vol 3 | Issue 4
The pathogenesis of LP is thought of from four mechanisms
Antigen specific cell mediated immune response (heat shock
proteins, CD4+ T helper cells, CD8+ cytotoxic T cells)
Nonspecific mechanism (epithelial basement membrane, mast
cells, chemokines, matrixmetaloproteinases) autoimmune
response, humoral immunity (circulating autoantibodies to
desmoglin 1 and 3).[4]
Extraoral manifestations
Patients with OLP frequently have concomitant disease in
one or more extraoral sites. Approximately 15% of patients
develop cutaneous lesions.[5] The classic appearance of skin
lesions described by the six ps: planar, plaque, pruritic, purple,
polygonal, and papular. Typically cutaneous lesions develop
after the appearance of oral lesions and severity of oral lesions
does not correlate with cutaneous lesions. Undoubtedly the
most frequent extraoral site in 20% of female patients with
OLP is genital mucosa. Association of LP of vulva, vagina, and
gingiva is recognized as vulvovaginalgingival syndrome.
When LP affects genital mucosa the erosive form of disease
is predominant type. The penogingival syndrome represents
male equivalent of vulvovaginal syndrome.[6]
Lichen planopilaris represents LP involvement of scalp and
hair follicles causing a scarring alopecia. Typically occurs in
three forms classic Lichen Planopilaris (LPP), frontal fibrosing
alopecia, GrahamLittle syndrome. The combination of
follicular LP with scarring alopecia of scalp and nonscarring
alopecia of axilla and pubis or other areas is known as
GrahamLittle syndrome.[7]
LP can also involve nails producing longitudinal ridging of nail
plate, onycholysis, subungual hyperkeratosis, trachyonychia,
pterygium formation. Other sites of involvement in LP are
esophagus, ocular, bladder, nasal, laryngeal, otic, gastric,
and anal.[8]
Oral manifestations
The red and white components of lesion can be part of
following textures.
Reticular
White fine striae/network can show annular/circular patterns.
The striae often present with peripheral erythemous zone,
which reflects sub epithelial inflammation. Most frequently
observed bilaterally in buccal mucosa and rarely on mucosal
side of lips.[9]
Papular
Present in initial phase of disease, clinically characterized
by small white dots which in most times intermingle with
reticular form.[9]
Plaque like
Homogenous well demarcated white plaque often but not
always surrounded by striae.[9]
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Patil, etal.:Oral lichen planus
Erythematous/atrophic
Characterized by homogenous red area, striae are frequently
seen at periphery. Some may exclusively display erythematous
OLP of attached gingiva, which represents desquamative
gingivitis.[9]
Erosive: Ulcerative, Bullous
Ulcerative and bullous are most disabling. Clinically present
with fibrin coated ulcers surrounded by erythematous zone
frequently displaying radiating white striae. Bullous lesions
vary from 4mm to 2cm and rupture leaving erythematous
zone.[9]
Diagnosis
The characteristic clinical aspects are sufficient to make a
correct diagnosis if classic lesions are present. An oral biopsy
with histopathologic study is recommended to confirm clinical
diagnosis and also to exclude dysplasia and malignancy.[10] The
value of direct immunofluorescence for confirmation of disease
is well accepted, especially with nondiagnostic histopathologic
features and for desquamative gingivitis.[7]
Management
To date no cure for OLP or dermal counterpart. The treatment
goal is 2fold, that is, alleviation of symptoms, monitoring of
dysplastic changes.[11]
Corticosteroids have shown to be predictable and effective
medications for controlling signs and symptoms. The
following topical medications have been tried in the
treatment of OLP:Fluocinonide 0.05% in an adhesive base
improved OLP with no adverse effects;[12] Betamethasone
showed effectiveness in symptomatic OLP in another
study;[13] Hydrocortisone hemisuccinate aqueous solutions
with little benefit in treating OLP;[14] Fluticasone propionate
spray and betamethasone sodium phosphate mouth rinse
have been used effectively in shortterm management of
symptomatic OLP;[15] Mometasone furoate microemulsion
resulted in significant reduction in pain in erosive ulcerative
OLP and significantly reduced surface area of erythema and
ulceration;[16] Clobetasol propionate 0.05% in various forms like
orabase, ointment or aqueous solution shown to be effective
for OLP in many studied subjects;[17,18] Application of clobetasol
17 proprionate orabase paste 0.05% plus 100,000IU/ml of
nystatin by means of tray appeared to be effacious treatment
for severe erosive gingival lesions and showed complete
response in 33cases over48week period.[19] Clobetasol
propionate 0.05% was found to be as useful as tacrolimus 0.1%
in treatment of OLP in another study.[20]
Triamcinolone acetonide 0.1% in orabase showed better
results than cyclosporin, [21] Pimecrolimus 1% cream, [22]
Betamethasone oral minipulse therapy, Fluocinolone
acetonide 0.1% orabase,[23] which have also been used for
treatment of OLP.
347
In lesions recalcitrant to topical therapy intralesional
corticosteroids can be effective, often triamcinolone
acetonide 5mg/ml combined with local anesthetic to
inject 0.1ml/cm3. Systemic steroids are indicated for brief
treatment of severe exacerbations of OLP. Prednisone
3060mg depending on severity of lesion is usually
administered.[24]
Retinoids are useful and are frequently used in combination
with topical steroids as adjuvant therapy. Topical
immunosuppressive drugs have been very much effective in
treating recalcitrant cases of OLP. Cyclosporin mouth rinse
(containing 100 mg of cyclosporine per milliliter) three
times daily. Despite of encouraging results in doubleblind
trials, its use is limited to due to hydrophobicity, high cost,
and poor taste.[25] In addition to concern over its role in
viral reproduction and malignant change have restricted its
use. 0.1% topical tacrolimus/pimecrolimus ointment used
effectively in treatment of erosive LP.[24]
Apart from these other treatment modalities are Dapsone
100 mg once daily for 3 months, PUVA therapy, Azathioprine:
150 mg/day, Levamisole: 150 mg/day for 3 consecutive days
in 1 week, Thalidomide: 200 mg/day or topical 1% paste,
giesofulvin have reported to be effective in treatment of
OLP in various case reports in literature,[25] unfortunately
randomized control studies are lacking in this aspect.
Lichen planus: A premalignant lesion
The possible premalignant character of LP is subject of
controversy and ongoing debate in literature, but the range
of malignant transformation is reported to be between 0.4%
and 5% over period of observation from 0.5to 20years.
Conclusion
The term OLP is a heterogeneous group of patients
afflicted with mucosal disease, identifying and eliminating
multifactorial agents associated with the disease is essential.
Relief can be achieved in most of patients with topical steroids
alone or in combination with other immunomodulatory
topical agents. Infrequently patients require prolonged
use of systemic medications. Patient should also be kept
under longterm follow up due to malignant tendency of LP.
All treatments are nonspecific and directed at eliminating
inflammation and therefore are partially successful.
Acknowledgment
Dr.Pramod B M, PG student Department of Orthopedics, JJMC,
Davangere.
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How to cite this article: Patil A, Prasad S, Ashok L, Sujatha GP. Oral
bullous lichen planus: Case report and review of management. Contemp
Clin Dent 2012;3:344-8.
Source of Support: Nil. Conflict of Interest: None declared.
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