J Oral Med Oral Surg 2020;26:14© The authors, 2020https://doi.org/10.1051/mbcb/2020001

https://www.jomos.org

Up-to Date Review And Case Report

Isolated lichen planus of the lips: cases reportsand literature reviewMaroua Garma1,*, Wafa Hasni1, Bechir Annabi2, Badreddine Sriha3, Souha Boudegga1,Abdellatif Boughzella1

1 Department of Oral Medicine and Oral Surgery, Dental Medicine Unit, Farhat Hached Hospital, University of Monastir, Tunisia2 Department of Conservative Dentistry, University Clinic of Dental Medicine, University of Monastir, Tunisia3 Department of Pathology, Farhat Hached Hospital, University of Monastir, Tunisia

(Received: 7 August 2019, accepted: 12 January 2020)

Keywords:lip / lichen planus /oral / therapeutics

* Correspondence: marwa.

This is an Open Access article dun

Abstract -- Introduction: Lichen planus is an inflammatory mucocutaneous dermatosis involving skin, appendagesand mucosa. Oral mucosa is the most commonly involved in all its sites, rarely the lips especially when isolated. Theaim was to conduct a literature review about isolated lichen planus of the lips and reporting two case reports of thislesion in order to highlight epidemiologic, clinical and histological features and therapeutic modalities of this lesion.Observations: Case report 1: a 34-year-old diabetic male patient consulted for an erosive, crusted and hemorrhagiccheilitis of the lower lip. Clinical and histological examination led to the diagnosis of isolated lichen planus of thelips. Case report 2: a 33-year-old female patient was referred from dermatology department for biopsy of chroniccheilitis of the lower lip. Clinical and histological examination confirmed the diagnosis of isolated lichen planus ofthe lips. Discussion: The review based on 34 case reports of isolated lichen planus of the lips, in addition to literaturedata confirmed that it is a benign rare lesion affecting mostly male patients having middle age with preponderance ofthe lower lip, its erosive form is the most frequent and it presents a favorable healing with topical treatmentparticularly corticosteroids.

Introduction

Oral lichen planus is a benign inflammatory dermatosiswhich may involve all sites of oral mucosa. It occurs mostly onthe buccal mucosa, tongue, gingiva and palate. Lip involve-ment, particularly if isolated, is unusual.

Isolated lichen planus of the lips is underreported in theliterature. In addition, its clinical features are usuallyconfusing leading to many wrong diagnoses. That’s motivatedthis article.

The aim was to conduct a literature review about isolatedlichen planus of the lips and reporting two case reports of thislesion, in order to investigate its epidemiologic, clinical andhistological features, besides to the therapeutic modalities.

Case report no. 1

A 34-year-old diabetic male patient presented with a 3-month history of erosive crusted and hemorrhagic cheilitis ofthe lower lip.

garma@yahoo.fr

istributed under the terms of the Creative Commons Arestricted use, distribution, and reproduction in any

The extra oral examination showed ulceration of 3 cm ofdiameter with marked crusted and bleeding areas. Perilesionalwhite keratotic striae were also revealed (Fig. 1).

The upper lip and oral mucosa were normal.Anamnesis and physical examination revealed no history

of previous skin disorder, local trauma, excessive sun exposureor recent drug intake. Therefore, actinic cheilitis, allergiccontact cheilitis, Stevens-Johnson syndrome were ruledout.

Biopsy and direct immunofluorescence were performed.Direct immunofluorescence was negative.

Histological examination showed hyperplasic epithelioma,a parakeratosis, liquefaction and degeneration of the basallayer which was irregular, in addition to a band-likeplasmocytes infiltrate in the dermal-epidermal interface(Fig. 2).

These features were consistent with oral lichen planus ofthe lower lip.

The patient was treated with topical corticosteroids:Clobetasol twice a day.

Within five months the lesion had entirely resolved(Fig. 3).

ttribution License (https://creativecommons.org/licenses/by/4.0), which permitsmedium, provided the original work is properly cited.

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Fig. 1. Crusted hemorrhagic ulceration along the lower lip with lacywhite streaks on the lateral border of the lesion.

Fig. 2. HE * 40: Hyperplasic epithelioma, parakeratosis, liquefactionand degeneration of the basal layer. A band-like plasmocytes infiltratein the dermal–epidermal interface.

Fig. 3. Complete resolution within five months, persistence ofatrophic and reticular aspect of the lower lip.

Fig. 4. Swelling and atrophic mucosa of the lower lip with erosion,fissures and crusted areas. Lesions are surrounded by lacy whitereticular streaks.

J Oral Med Oral Surg 2020;26:14 M. Garma et al.

Case report no. 2

A 33-year-old female patient had been referred fromdermatology department for biopsy of chronic cheilitis of thelower lip that had evaluated for 8 years.

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Previous biopsy and direct immunofluorescence of thelower lip were inconclusive.

Her familial and post medical history was non-contributary.At anamnesis, she reported recurrence of edema and

crusting of the lower lip with hemorrhagic fissures. She had nohistory of drug intake, local injury and had not experiencedexcessive sun exposure.

Exobuccal examination revealed swelling, atrophic mucosaand erosion of the lower lip with fissures and crustedhemorrhagic areas, in addition to lacy white streaks forminga reticular pattern in the lateral border of the lesion (Fig. 4),

Fig. 6. Entirely resolved within seven months.

Fig. 5. HE*40: Apoptotic bodies in the epithelium, parakeratosis anda dense band-like lymphocytic infiltrate in the connective tissue witha degeneration of the basal layer.

J Oral Med Oral Surg 2020;26:14 M. Garma et al.

therefore a provisional diagnosis of oral lichen planus wasmade.

The upper lip was uninvolved. The reminder of the mucosalsurfaces and the skin showed no abnormality. Directimmunofluorescence was negative. Biopsy from the lower lipshowed an epithelium full of apoptotic bodies with para-keratosis and a dense band-like lymphocytic infiltrate in theconnective tissue with a degeneration of the basal layer(Fig. 5).

Treatment was commenced with a topical corticosteroid:betamethasone once a day at night. The patient showed afavorable improvement, but after three months, edema andcrusting were revealed in the lower lip. So, she wasrecommended a sun screen and the application of topicalfluocinonide for two weeks twice a day. Within three monthsthe lesion was entirely resolved again (Fig. 6).

Discussion

A review of the literature was conducted on the databaseMedline via its interface PubMed using Mesh Keywords : “lip”,”lichen planus, oral”, “therapeutics” and combining thefollowing Boolean equations : “lip” and “lichen planus, oral” /“lip” and “lichen planus, oral” and “therapeutics”, in the periodfrom 1939 to 2019. This bibliographic research concluded to 32case reports about isolated lichen planus of the lips from 19articles.

The parameters extracted from these cases were summa-rized in Table I and they concerned: age, sex, clinical form,localization, skin involvement, systemic pathologies, date ofappearance, treatment and outcomes (Tab. I).

Lichen planus is an inflammatory benign condition of theskin and mucosa whose etiology is still unknown.

In the oral cavity, commonly involved sites are buccalmucosa and tongue. However, gingiva, floor of mouth, palateand lips are rarely affected [1,2].

In fact, the prevalence of lip involvement varies from 6.3 to29.4% [1].

Isolated lip lichen planus is less frequent, its prevalencereported in the literature varies from 0.51 to 8.9% [1].

Isolated lichen planus of the lips involves patients in themiddle age with a male preponderance [1]. This has beenconfirmed by our review. In fact, the results showed 24 males/10 females and 27 patients were aged between 40 and 74 years(Tab. I).

Due to their anatomic localization, lips are currentlysubject to many injuries: such as sun exposure, make upapplication, biting. Therefore, clinical features of isolated orallichen planus of the lips are not pathognomonic and usuallymisdiagnosed and may mimic many other types of cheilitis.

Isolated lichen planus of the lips appears as whitish,reddish or mixed surfaces with crusting, erosion and ulcerationthat may be associated to some blisters along the vermilion ofthe upper, lower or the two lips [1]. These features wereidentified in our two patients.

All clinical variants of oral lichen planus that have beendescribed in the literature may also be identified in the isolatedform of the lips which are: reticular, papular, erosive, bullousand atrophic form [1,3].

The reticular form is the most typical characterized byWickham’s striae [1]. However, the erosive one is the mostcommon according to the literature [1,4]. In our review, theerosive form was the frequent one (22 cases), then thehyperkeratosic or plaque-like form (5 cases), the reticular onewas presented in four cases and finally the less common was theannular form (2 cases) (Tab. I).

Lower lip involvement shows a clear predominancecompared to the upper lip. In fact, the lower/upper lipinvolvement ratio was 6:5 [1]. These data were also concludedfrom our review which revealed a lower lip involvement in 27cases, five cases of upper and lower lip involvement and onlyone case of upper lip involvement (Tab. I).

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TableI.

Clinical

casesaboutisolated

lichenplanus

ofthelip

sreported

intheliteraturefrom

1939

to2019.

No.of

case

Articleandyear

Age

Sex

Localization

Clinical

form

Date

ofappearance

Skin

involvem

ent

System

icpathologies

Treatm

ent

Evolutionwith

treatm

ent

1Whittle

CH,1939

1369

Male

Lower

lipPlaque

–Genitalmucosa

NoMercure,arsenic,

X-ray

Stable

2P.

H.IT1N,

1995

544

Male

Lower

lipErosive

3years

noNo

Acitretin:

neotigazon

®30

mg/day

prednisone

15mg/day,

sunscreen.

Completeremission

in10

weeks

3S.

ALA.MN,

1996

651

Male

Lower

lipReticular

9mon

ths

noNo

Betamethasone

valerate®

cream

0.1%

/2weeks

Completeremission

in3mon

ths

4DDe

Argila,1997

1151

Male

Lower

lipErosive

11years

noNo

Chloroquineph

osph

ate

25mg/day

Completeremission

ofsymptom

sin

6mon

ths

5Ro

bertoCecchi,2002

1043

Male

Lower

lipReticular

7mon

ths

noNo

Betamethasone

Dipropionate

0.5%

ointment

Completeremission

in1mon

ths,no

symptom

sin

4mon

ths

6Chiang

CT,2002

1236

Female

Lower

lipErosive

–no

NoPrednisolone

Remission

7Yu

Tc,2003

1744

Male

Lower

lipErosive

–no

Hypertension

Clobetasol

Remission

8Do

novanJC,2005

1851

Male

–Erosive

–no

HCV-Hepatitis

Tacrolimus

Stable

9Petruzzi

M,2007

452

Female

Lower

lipHyperkeratosic

6mon

ths

–No

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

10Petruzzi

M,2007

454

Male

Lower

lipErosive,

atroph

ic10

mon

ths

–No

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

11Petruzzi

M,2007

473

Male

Lower

lipErosive,

atroph

ic8mon

ths

–HCV-Hepatitis

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Partialremission

12Petruzzi

M,2007

449

Male

Lower

lipErosiveatroph

ic4mon

ths

–No

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

13Petruzzi

M,2007

452

Male

Lower

lipHyperkeratosic

2mon

ths

–No

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

14Petruzzi

M,2007

462

Male

Lower

lipAtroph

ic,erosive

8mon

ths

–Diabetes

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Partialremission

15Petruzzi

M,2007

474

Female

Upper

lip/Lower

lipAtroph

ic,erosive

10mon

ths

–HCV-Hepatitis

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

16Petruzzi

M,2007

460

Male

Lower

lipAtroph

ic,erosive

3mon

ths

–HCV-Hepatitis

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

17Petruzzi

M,2007

471

Male

Upper

lip/Lower

lipHyperkeratosic

4mon

ths

–HCV-Hepatitis

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

18Petruzzi

M,2007

480

Female

Upper

lip/Lower

lipAtroph

ic,erosive

6mon

ths

–HCV-Hepatitis

Clobetasol

proprion

ate0.05

%:

cream

2/day,Tocoph

erol

oil

Completeremission

19John

sonH,2008

1942

Female

Lower

lipErosive

-no

NoTacrolimus

Stable

20GencoglanG,

2011

1556

Male

Lower

lipErosive

2mon

ths

noNo

Imiquimod

cream

5%,2/day

Completeremission

,no

recurrence

in18

mon

ths

21GencoglanG,

2011

1561

Male

Lower

lipErosive

6years

noNo

Imiquimod

cream

5%,2/day

Amelioration

within

2weeks,recurrence

within6mon

ths

22GencoglanG,

2011

1565

Male

Lower

lipReticular

11years

noNo

Imiquimod

cream

5%,2/day

Remission

within

2weeks

23GencoglanG,

2011

1522

Male

Lower

lipReticular

4years

noNo

Imiquimod

cream

5%,2/day

Healin

gin

2weeks,

norecurrence

in5mon

ths

24Sarika

Holmukhe

DNB,

2012

840

Male

Lower

lipAn

nular

3mon

ths

noNo

Tacrolimus

0.03

cream

3/day

–

25Do

mingues

E,2012

2044

Male

Lower

lipErosive

–yes

NoClobetasol

Remission

26SugashimaY,

2012

2132

Female

Upper

lip/Lower

lipAn

nular

–no

Allergyto

zinc

Tacrolimus

Regression

ofthelesion

J Oral Med Oral Surg 2020;26:14 M. Garma et al.

4

TableI.

(con

tinu

ed).

No.of

case

Articleandyear

Age

Sex

Localization

Clinical

form

Date

ofappearance

Skin

involvem

ent

System

icpathologies

Treatm

ent

Evolutionwith

treatm

ent

27DillipKu

mar

Samal,

2015

352

Male

Lower

lipPlaque

1years

noNo

Biopsy

exerese

Noreccurence

28Nu

zzoloP,

2016

172

Male

Upper

lipErosive

–no

Liver

insufficiency

HCV-hepatitis

Surgical

excision

Completeremission

29Nu

zzoloP,

2016

174

Male

Upper

lip/

Lower

lipErosive

–no

NoCortison

eStable

30Ellie

Choi,2017

962

Female

Lower

lipErosive

1years

noNo

Tacrolimus

hydrocortisone

vaselin

e,sunscreen

Completeremission

,No

reccurence

in2years

31FeiYan

Yu,2018

1638

Female

Lower

lipErosive

10years

noNo

TCM

:“Qingw

enJiedu

Kouyankang

granules,”

glucosides,

antiinflam

matory

horm

ones/4

mon

ths

Amelioration

in1mon

th,no

recuurence

in5mon

ths

32Mathu

rM,2019

244

Male

Lower

lip–

4mon

ths

noNo

Betamethasone

Diproprio

nate

0,5%

Amelioration

in15

days

33Case

report

no.1

34Male

Lower

lipErosive

3mon

ths

noDiabetes

Clobetasol

Completeremission

in5mon

ths

34Case

report

no.2

33Female

Lower

lipErosive

8years

noNo

Betamethasone

Remission

in7mon

ths

J Oral Med Oral Surg 2020;26:14 M. Garma et al.

Usually lips lesions are symptomatic, mostly when itconsists on the erosive variant. Symptoms are dominated byburning, tenderness and tingle sensations with discomfort thatare aggravated with spicy and acidic foods.

Unsightly appearance of lip lesions leads to psychologicaldistress reported by some patients [4].

Concomitant cutaneous lesions are exceptional in thegenital region. In the review, one case of skin involvement wasrevealed in the genital lesion (Tab. I).

Histologically, this lesion showed the pathognomoniccharacters of oral lichen planus which are irregular acanthosis,orthokeratosis with liquefactive vacuolar degeneration of thebasal cell layer. In addition, we revealed hypergranulosis,edema and a dense band like lymphocytic infiltrate in thedermal–epidermal interface. Colloid bodies representingnecrotic keratinocytes known as Civatte bodies are alsoidentified [1,3,5,6].

Oral lichen planus is a benign dermatosis, nevertheless,some cases of transformation of lichen planus of the lips intosquamous cell carcinoma were documented [2–4].

In fact, malignant transformation is still discussed.According to the literature, the rate of this transformationvaried from 0.4 to 5.6% [7]. This variation is due to thediversity of clinical forms of oral lichen planus, thedifficulty of distinction between this lesion and lichenoidone and other pathologies, besides to the variety of riskfactors [7].

Many diagnoses should be ruled out in case of isolated liplichen planus. For the erosive form it must be differentiatedfrom caustic or traumatic cheilitis, autoimmune blistersdermatosis, erythema multiform, Stevens-Johnson syndrome,herpes or bacterial infection.

For the keratotic variety, the differentiation betweenleukokeratosis, lupus, graft versus host disease and isolatedlip lichen planus may be difficult. Also, actinic cheilitis, atopicdermatosis or some neoplasia must be eliminated[4,5,8,9,10].

The pathogenesis of oral lichen planus is still notcompletely understood. The auto immune mechanism is themost involved [7]. Some risk factors are reported, such assolar exposure, tobacco and alcohol consumption, mechan-ic trauma and cosmetic products application. This mayexplain the greater incidence in the lower lip involvement[4,8,9].

This lesion can be associated with some systemic diseaseslike hepatitis infection, diabetes, thyroid disorders, Goodsyndrome, thymoma, graft versus host disease, hypertension[1,7], therefore some laboratory tests are required: HCVserology, diabetes and thyroid function tests. In our review, tenpatients presented systemic pathologies: two patients haddiabetes, one patient had hypertension and seven had HCV-hepatitis (Tab. I).

Usually, isolated lip lichen planus shows a great remissionwith topical treatment, most commonly with topical cortico-steroids. Systemic and intralesional administration are rarelyused [2–4].

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J Oral Med Oral Surg 2020;26:14 M. Garma et al.

Topical steroids such as Clobetasol propionate, Fluticasonepropionate are the first line treatment [3]. Also, Betametha-sone valerate 0.1%, Betamethasone dipropionate 0.05%,Fluocinonide and Chloroquine phosphate are applicated[2,4,10,11].

Prednisolone is rarely used due to its galenic form thatcan’t be adapted to the labial application [12]. The surgicalexcision is described in the literature [1,3]. Immunomodula-tory agents in form of tacrolimus and cyclosporine are usedtopically in patients not responding to topical steroids [3], inaddition to retinoids alone or in association with corticoste-roids [4].

Some other therapeutics are described in the literature:Wittle [13] proposed the treatment with Mercure, Arsenic andX-rays.

Dillenbug described as treatment the laser [14].Gencoglan proposed the Imiquimod cream 5% [15] and

finally in 2018, Feiyan [16] proposed a traditional Chinesemedicine comprising “Qingwen Jiedu KouyarKang granules”,total Paeonia glucosides and a combination of hormones andanti-inflammatory agents.

The exploration of the data review confirmed the topicaltreatment efficacity. The most common treatment used wasthe Clobetasol which was used in 13 cases with completeremission in 11 patients, then the Tacrolimus (5 cases), theBetamethasone (4 cases), and finally the Imiquimod (4 cases)(Tab. I).

Conclusion

Through this literature review we can conclude that isolatedlichen planus of the lips affects preferentially males in themiddle age with a lower lip preponderance. The erosive formwas the frequent one. This lesion presents a great response tothe topical treatment specially corticosteroids.

The prevention by risk factor elimination and oral hygienemaintenance is required to rule out active recurrence. Also,perfect monitoring of eventual cutaneous lesions or other orallocalizations is quite necessary in their early diagnosis andtreatment and in early detection of possible malignanttransformation.

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Conflicts of interest: The authors declare that they haveno conflicts of interest in relation to this article.

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