sjdv-2013-0015

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177copy 2009 Te Serbian Association of Dermatovenereologists

the lower lip with clinical manifestations of nodularenlargement reduced mobility and lip inversion (5)It is most commonly seen in adult males (6) but it hasalso been described in females (7 8) and children (9)Cases involving the upper (10) or both lips (11) havealso been reported

Te etiological factors are sometimes hardto determine Some causes or predisposing factors

include bacterial infections (mostly Staphylococcusaureus ) syphilis actinic radiation smoking poororal hygiene compromised immune system butalso genetic transmission (autosomal dominanttransmission is suggested) (11 12 13) Leao (14)described a case of GC in a HIV-infected patient withan explanation that it was probably a coincidence

Based on clinical presentation glandular cheilitiscan be classi1047297ed into subtypes simplex (described by

Cheilitis is an in1047298ammatory condition of thevermilion border of the lips which is the

junction between the skin and the mucosa Cheilitismay arise as a primary disorder of the vermilion zonethe in1047298ammation may extend from the nearby skinor less often from the oral mucosa (1 2) Cheilitismay represent a focal in1047298ammatory process or amanifestation related with diseases of other systems

or organs (3) Te primary cheilitis lesions are eithersuper1047297cial or deep (4) Super1047297cial cheilitis can beclassi1047297ed into exfoliative (factitious) postmenopausalactinic (solar) allergic (contact) eczematous angularand abrasive precancerous (Manganottirsquos) Deep typesof cheilitis include glandular and granulomatous C

Glandular cheilitis (GC) is a rare conditioncharacterized by in1047298ammatory changes and swellingof salivary glands in the lips It commonly affects

CASE REPORS Serbian Journal of Dermatology and Venereology 2013 5 (4) 177-182

Abstract

Cheilitis is an inflammatory condition of the vermilion border of the lips which is the junction between the skin and the

mucosa Cheilitis may arise as a primary disorder of the vermilion zone the inflammation may extend from the nearby

skin or less often from the oral mucosa Primary cheilitis lesions are either superficial or deep Deep types includecheilitis glandularis (inflammatory changes and lip gland swelling) and granulomatous cheilitis (chronic swelling of the

lip due to granulomatous inflammation mostly of unknown origin) Cheilitis glandularis is a rare condition that mostly

affects the lower lip and it is characterized by nodular enlargement reduced mobility and lip erosion Based on clinical

presentation cheilitis glandularis may be classified into three subtypes simplex (described as Puente and Acevedo)

superficial suppurative (described by Baelz-Unna) and the most severe type ndash deep suppurative also known as cheilitis

glandularis apostematosa (Volkmannrsquos cheilitis) characterized by deep-seated inflammation forming abscesses and

fistulous tracts

This is a case report of a female patient with a deep suppurative type of cheilitis affecting both lips Treatment with

systemic antibiotics (using antibiogram tests) corticosteroids and topical therapy resulted in significant improvement

Key words

Cheilitis + diagnosis + etiology + classification + therapy Disease Progression Prognosis Treatment Outcome

Cheilitis Glandularis Apostematosa in a Female Patient ndash a CaseReport

Mirjana PARAVINA

Faculty of Medicine University of Niš Serbia

Correspondence Mirjana Paravina E-mail mirjanaparavinagmailcom

UDC 616317-002-092-08

DOI 102478sjdv-2013-0015

OPEN

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178 copy 2009 Te Serbian Association of Dermatovenereologists

were not affected (Figure 2) the lips were of hard-elastic consistency to touch and granular in structureextreme sensitivity caused hemorrhagic or purulentdischarge the regional lymph nodes were notenlarged the tongue was unaffected while the teeth

were neglected and mostly missing

Internist examinationTe internist examination showed normal 1047297ndings

Laboratory tests

Te relevant hematological and biochemical parameters were within physiological levels the serologic test forsyphilis and enzyme-linked immunosorbent assay

Puente and Acevedo) (15) super1047297cial suppurative(described by Baelz-Unna) (16 17) and a more severedeep suppurative type also known as myxadenitislabialis or cheilitis glandularis apostematosa(Volkmannrsquos cheilitis) (18) characterized by deep-seated in1047298ammation forming abscesses and 1047297stuloustracts

Von Volkmann (18) was the 1047297rst to describecheilitis glandularis apostematosa in 1870 as achronic suppurative in1047298ammation of the lower lipcharacterized by swelling of the mucus glands and themucopurulent discharge through the dilated ductalopenings

We report a patient with deep suppurative typeof cheilitis of both lips Te treatment with systemicantibiotics (using antibiogram tests) corticosteroids

and topical therapy resulted in signi1047297cantimprovement

Case Report

A 61-year-old village housewife claimed that the 1047297rstchanges occurred on the right half of her lower lipat the age of 56 in the form of prominent rednessbumps and wetting During the next year the changesaffected the entire lower lip At the age of 60 theinitial wetting was followed by purulent discharge

with scales and squamous lesions She was treated

by a dermatologist a dentist and an EN (ear noseand throat) specialist Various drugs were appliedmostly topically antibiotics antimycotics interferonand acyclovir Te treatment provided only mildtemporary improvement

Clinical status at 1047297rst examination (the 1047297rst contact withthe patient)Both lips and the vermilion border were covered withthick adherent scales and squamous crusts purulenthemorrhagic discharge was seen under pressure (Figure

1) lesions were painful especially sensitive to touch while normal functions such as speaking eating andchewing were compromised

Clinical status after crust removal Both lips were enlarged extremely erythematousin1047297ltrated with erosions and super1047297cial shallowulcerations and 1047297ssures the erythema and in1047297ltrationspread along the vermilion the corners of the mouth

M Paravina Cheilitis glandularis apostematosa in a female patientSerbian Journal of Dermatology and Venereology 2013 5 (4) 177-182

Figure 1 Both lips and the vermilion border arecovered with thick adherent scales and squamous

crusts with purulent hemorrhagic lesions underneath

Figure 2 Both lips are enlarged extremelyerythematous in1047297ltrated with erosions and

super1047297cial shallow ulcerations and 1047297ssures erythemaand in1047297ltration spread along the vermilion corners

of the mouth not affected

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Figure 3 After treatment lips are less in1047297ltrated and erythematous without layers of crusts and squamouslesions with some erosions of the central lower lip

(ELISA) for human immunode1047297ciency virus (HIV)antibodies were negative bacteriological examinationof lesion specimens showed Staphylococcus alphahaemolyticus and Neiseria catharalis

Histopathological analysis Probatory excision was performed 5 years earlier at theEar Nose and Troat Clinic in Belgrade histological1047297ndings were consistent with in1047298ammatoryleukoplakia the affected area showed folliculitis andthere were erosions of the vermilion lip Repeat biopsy

was rejected by the patient

Treatment Te therapy included oral cipro1047298oxacin (500 mgtwice a day) according to antibiogram during 10 days

15 mg prednisone per day during 3 months boricacid and antiseptic solutions were used to removecrusts and squamous lesions which was followed byapplication of antibiotic ointments (garamycin andlater chloramphenicol)

Local status after therapy Te lips were less in1047297ltrated and erythematous

without layers of crusts and squamous lesions withsome erosions of the central lower lip (Figure 3)repeated antibiotic and corticosteroid therapy resulted

in signi1047297cant improvement (Figure 4)

Discussion

Te classi1047297cation of GC into three subtypes was doneregarding the severity of in1047298ammation presence ofbacterial infection and lip enlargement (5 7 19

20) Te simplex GC is characterized by multiplepainless lesions with central depression and dilatedcanals as well as mucous secretion which may occurspontaneously or under pressure Te super1047297cialsuppurative type of GC presents swelling of the lipinduration and areas of ulcerations and crusting withsecretion of clear or viscous exudates from the salivaryduct openings Deep suppurative type of glandularcheilitis or cheilitis glandularis apostematosa ischaracterized by formation of deep abscesses and1047297stula tract that eventually heal by scarring Episodesof suppurative discharge are spontaneous

Many believe these subtypes probably representa continuation of the same disease process i e ifthe simple type is not treated properly it becomessecondarily infected and progresses to the next typeand then to the next (3) It is possible that theexcessive salivary secretion from minor salivary glandsrepresents an unusual response to irritation of the lipcaused by other reasons for example actinic damageor repeated licking (2) Te disease progression inour patient has proven this assumption Te 1047297rstsymptoms were typical for GC simplex probably

caused by actinic irritation without data on hereditary


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patches of erythema with dense plasma cell in1047297ltrates)(29)

Te treatment of GC depends on the typeit may include systemic corticosteroids but alsoextensive surgical resections (3) Te reduction orelimination of predisposing factors (sun or windexposure) is the 1047297rst step in the treatment followedby photoprotection and use of emollients (30)

Apart from topical use corticosteroids may be used

as intralesional and systemic Te treatment mayalso include anticholinergics antihistamines andantibiotics (3 9 29 31 32 33) Radiation therapyand surgical procedures cryosurgery vermilionectomyandor labial mucosal stripping may be used as well(33)

After application of local antiseptic andantibiotic ointments our patient received systemiccorticosteroids and antibiotics (according to anantibiogram) which led to initial improvementDue to some deterioration the therapy was repeatedresulting in signi1047297cant improvement

Te prognosis for quo ad sanationam wasunfavorable Although cases of spontaneous remission(11) have been reported the treatment outcome isuncertain Te possibility of malignant alterationshould not be ignored Patients with GC especiallythose with deep suppurative type should be followed-up due to the risk of squamous cell carcinoma (SCC)(21 31) Nico et al evaluated 22 patients diagnosed

burden Te subsequent bacterial infection probablycaused by poor oral hygiene led to the developmentof GC apostematosa

Based on literature data there is a difference inthe de1047297nition of the disease Tere is a disagreementregarding the obligatory hyperplasia of local salivaryglands While Von Volkmann (14) described cheilitisas swelling of the mucous glands many authors (46 8 11) point to the hyperplasia of minor salivary

glands or dilated ductal canals and some others pointto in1047298ammation and swelling (3 13 20 21) Tisdisagreement is based on different histopathological1047297ndings some authors (6 7 11) found hyperplasiaof minor salivary glands whereas others did not (3 912 14 21 -29) Based on histopathological 1047297ndingsit prevails that hyperplasia of salivary glands in GCis not typical chronic sclerosing sialadenitis andscarring are predominant whereas ductal ectasia is adominant histopathological and clinical 1047297nding (3)In general histopathological 1047297ndings of dense chronicin1047298ammatory in1047297ltrate are found only in more severe

types of GC while genuine hyperplasia of salivaryglands orand ductectasia are rather rare (2)

Differential diagnosis includes angioedema (noswelling between attacks) exfoliative C (persistentscaling) granulomatous C (histological changes arenot always conspicuous or speci1047297c) elephantiasisnostras (3) irritant or contact cheilitis as well asplasma cell cheilitis (circumscribed 1047298at or elevated

Figure 4 Repeated antibiotic and corticosteroid therapy resulted in signi1047297cant improvement


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with CG and reported three cases of super1047297ciallyinvasive carcinoma on the lower lip out of whichtwo were albino Tis points to the adverse effectsof sun exposure on the development of CG and thepossibility of malignant alteration (19 22) especiallyin cases of deep suppurative type of CG (30 31) Insome series 18 ndash 35 of cases progressed to SCC(22) Te reason for this probably lies in the highersusceptibility of the inverted lip to all risk factors forthe development of SCC rather than in GC beinga premalignant condition sui generis Te majority ofreported cases had deep suppurative type of the diseaserequiring surgical intervention and regular follow-up(3)

Conclusion

Tis is a report of a female patient with a severetype of glandular cheilitis affecting both lips with aprogressive course and good response to combinedantibiotic and corticosteroid therapy

Abbreviations

C - cheilitisGC - glandular cheilitisHIV - human immunode1047297ciency virusEN ndash ear nose and throatElisa - enzyme-linked immunosorbent assay

SCC - squamous cell carcinoma

References1Stanojević M Bolesti usana jezika i usne duplje U Paravina M

Spalević Lj Stanojević M iodorović J Binić I Jovanović DDermatovenerologija drugo dopunjeno izdanje Medicinskifakultet Niš Niš Prosveta AD 2006 str 277-85

2 Stoopler E Carrasco L Stanton DC Pringle G Sollecito PCheilitisglandularis an unusual histopathologic presentation OralSurg Oral Med Oral Pathol Oral Radiol Endod 200395313-7

3 Orlov S Kojović D Mirković B Oralna medicina NišEuroprint 2001 str 27-37

4 Louren SV Gori LM Boggio P Nico MMS Cheilitis

glandularis in albinos a report of two cases and review ofhistopathological 1047297ndings after therapeutic vermilionectomy JEADV 2007211265-7

5 aneja P Singh N Cheilitis glandularis a clinical report IntChin J Dent 200222-4

6 Weir W Johnson WC Cheilitis glandularis Arch Dermatol1971103433-7

7 Hillen U Franckson Goos M Cheilitis glandularis a casereport Acta Derm Venereol 20048477-9

8 Yacobi R Brown DA Cheilitis glandularis a paediatric casereport J Am Dent Assoc 1989 118317-8

9 Matsumoto H Kurachi Y Nagumo M Cheilitis glandularisreport of a case affecting upper lip Showa Shigakkai Zasshi19899441-5

10 Yanagawa Yamaguchi A Harada H Yamagata K IshibashiN Noguchi M et al Cheilitis Glandularis two case reportsof Asian-Japanese men and literature rewiew of Japanese

cases ISRN Dentistry 2011 Article ID 457567 6 pages doilo54022011457567

11 Lederman DA Suppurative stomatitis glandularis Oral SurgOral Med Oral Pathol 1994 78319-22

12 Mirowski GW Parker ER Biology and pathology of the oralcavity In Wolf K Goldsmith LA Katz SI Gilchrest BA Paller AS Leffel DJ eds Fitzpatrikrsquos dermatology in general medicine7th ed New York McGraw Hill Medical 2008 p 641-53

13 Leao JC Ferreira AMC Martins S Jardim ML Barret WSculi C et al Cheilitis glandularis an unusual presentation ina patient with HIV infection Oral Surg Oral Med Oral PatholOral Radiol Endod 200395142-4

14 Von Volkmann R Einigefalle von Cheilitisglandularisapostematosa Arch Pathol Anal 187050142-4

15 Carrington PR Horn D Cheilitis glandularis a clinicalmarker for both malignancy andor severe in1047298ammatorydisease of the oral cavity J Am Acad Dermatol 200654336-7

16 Binić I Janković A Heilitisi etiologija i mogućnosti lečenjaU Karadaglić Đ Jovanović M ur Bolesti sluzokože usneduplje šta je novo Beograd Monogra1047297je naučnih skupova AMN SLD 20101(2)37-53

17 Nico MMS de Melo JN Lourenco SV Cheilitis glandularisa clinicopathological study in 22 patients J Am AcadDermatol 201062233-8

18 Butt FM Chindia ML Ashani A Cheilitis glandularisprogressing to squamous carcinoma in an hiv-infected patientcase report East Afr Med J 200784(12)595-8

19 Swerlick RA Cooper PH Cheilitis glandularis a reevaluation

J Am Acad Dermatol 198410 466-7220 Rada DC Koranda FC Katz FS Cheilitis glandularis a disorder

of ductal ectasia J Dermatol Surg Oncol 198511372-521 Neville B Damm D Alen C Bouquet J editors Oral and

maxillofacial pathology 2nd ed Philadelphia WB Saunders2002 p 389-435

22 Stuller CB Schaberg SJ Stokos J Pierce GL Cheilitisglandularis Oral Surg 198253 602-5

23 Winchester L Scully C Prime SS Eveson JW Cheilitisglandularis a case affecting the upper lip Oral Surg Oral MedOral Pathol 198662654-6

24 Williams HK Williams DM Persistent sialadenitis of theminor glands - stomatitis glandularis Br J Oral MaxillofacSurg 198927212-6

25 Bender MM Rubenstein M Rosen Cheilitis glandularisin an African-American woman reponse to antibiotic therapySkinme 20054(6)312-7

26 Michalowski R Cheilitis glandularis heterotopic salivary glands andsquamous cell carcinoma of the lip Br J Dermatol 196272445-9

27 Rogers RS Bekic M Diseases of the lips Semin Cutan MedSurg 199716328-36

28 Haldar B Cheilitis glandularis treated by injection ofintralesional triamcinolone Indian J Dermatol 19762153-4

29 Verma S Cheilitis glandularis a rare entity Br J Dermatol20031483


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Cheilitis glandularis apostematosa kod osobe ženskog pola ndashprikaz slučaja

SažetakHeilitis (Cheilitis ) in1047298amatorno je oboljenje rumenezone usana (vermiliona) koja se nalazi na prelazu kožeu sluzokožu Heilitisi koji nastaju kao samostalnaoboljenja mogu biti površni ili duboki Duboki suCheilitis glandularis i Cheilitis granulomatosa Cheilitis

glandularis (CG) retka je bolest koja najčešće zahvatadonju usnu i karakteriše je nodularno uvećanjeredukovani mobilitet i everzija usne Kliničke varijante

su CG simplex (Puente and Acevedo) CG suppurativasuper1047297ciallis (Baelz-Unna ) i CG suppurativa profundaseu CG apostematosa (Von Volkmann)Mi prikazujemo bolesnicu sa dubokom supurativnomformom heilitisa na obema usnama kod koje jesistemska primena antibiotika prema antibiogramui kortikosteroida uz lokalnu terapiju dovela doznatnog poboljšanja

Ključne re

či

Cheilitis + dijagnoza + etiologija + klasi1047297kacija + terapija ok bolesti Prognoza Ishod lečenja


Unauthenticated


8202019 sjdv-2013-0015



were not affected (Figure 2) the lips were of hard-elastic consistency to touch and granular in structureextreme sensitivity caused hemorrhagic or purulentdischarge the regional lymph nodes were notenlarged the tongue was unaffected while the teeth

were neglected and mostly missing

Internist examinationTe internist examination showed normal 1047297ndings

Laboratory tests

Te relevant hematological and biochemical parameters were within physiological levels the serologic test forsyphilis and enzyme-linked immunosorbent assay

Puente and Acevedo) (15) super1047297cial suppurative(described by Baelz-Unna) (16 17) and a more severedeep suppurative type also known as myxadenitislabialis or cheilitis glandularis apostematosa(Volkmannrsquos cheilitis) (18) characterized by deep-seated in1047298ammation forming abscesses and 1047297stuloustracts

Von Volkmann (18) was the 1047297rst to describecheilitis glandularis apostematosa in 1870 as achronic suppurative in1047298ammation of the lower lipcharacterized by swelling of the mucus glands and themucopurulent discharge through the dilated ductalopenings

We report a patient with deep suppurative typeof cheilitis of both lips Te treatment with systemicantibiotics (using antibiogram tests) corticosteroids

and topical therapy resulted in signi1047297cantimprovement

Case Report

A 61-year-old village housewife claimed that the 1047297rstchanges occurred on the right half of her lower lipat the age of 56 in the form of prominent rednessbumps and wetting During the next year the changesaffected the entire lower lip At the age of 60 theinitial wetting was followed by purulent discharge

with scales and squamous lesions She was treated

by a dermatologist a dentist and an EN (ear noseand throat) specialist Various drugs were appliedmostly topically antibiotics antimycotics interferonand acyclovir Te treatment provided only mildtemporary improvement

Clinical status at 1047297rst examination (the 1047297rst contact withthe patient)Both lips and the vermilion border were covered withthick adherent scales and squamous crusts purulenthemorrhagic discharge was seen under pressure (Figure

1) lesions were painful especially sensitive to touch while normal functions such as speaking eating andchewing were compromised

Clinical status after crust removal Both lips were enlarged extremely erythematousin1047297ltrated with erosions and super1047297cial shallowulcerations and 1047297ssures the erythema and in1047297ltrationspread along the vermilion the corners of the mouth


Figure 1 Both lips and the vermilion border arecovered with thick adherent scales and squamous

crusts with purulent hemorrhagic lesions underneath

Figure 2 Both lips are enlarged extremelyerythematous in1047297ltrated with erosions and

super1047297cial shallow ulcerations and 1047297ssures erythemaand in1047297ltration spread along the vermilion corners

of the mouth not affected

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Discussion






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Conclusion


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Conclusion


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Ključne re

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Conclusion


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Ključne re

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