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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.
1
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.
2
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).
3
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].
5
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.
6
Conflicts of interest: The authors declare that they haveno conflicts of interest in relation to this article.
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11. De Argila D, Gonzalo A, Pimentel J, et al. Isolated Lichen planus ofthe lip successfully treated with chloroquine phosphate.Dermatology 1997;195:284–285.
12. Chiang CT, Chan HL. Superficial mycosis superimposing onisolated lichen planus of the lip: a case report and review of theliterature. Cutis 2002;69:305–308.
13. Whittle CH. Case for diagnosis? Lichen planus of lip. Proc R SocMed 1939;32:1402.
14. Dillenburg CS, Martins MA, Munerato MC, et al. Efficacy of laserphototherapy in comparison to topical clobetasol for thetreatment of oral lichen planus: a randomized controlled trial.J Biomed Opt 2014;19:68002.
15. Gencoglan G, Inanir I, Sahin O, et al. Imiquimod 5% cream forisolated lichen planus of the lip? J Dermatol Treat 2011;22:55–59.
16. Yu FY, Xu N, Zhao B, et al. Successful treatment of isolatedoral lichen planus on lower lip with traditional Chinese medicineand topical wet dressing: A case report. Medicine 2018;97:50.
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