J Oral Pathol Med 1995: 24: 177-9Printed in Denmark . All rights reserved

Copyright © Munksgaard 1995

JOURNAL OF

Oral PathciogysMsdidneISSN 0904-2512

Case reports

Exfoliative cheilitis Tom D. Daley^ and Adytia K. Gupta^^Department of Pathology, University ofWestern Ontario, London, ^Division ofDermatology, Department of Medicine,University of Toronto, Ontario, Canada.

Daley TD, Gupta AK: Exfoliative cheilitis. J Oral Pathol Med 1995; 24: 177-9.© Munksgaard, 1995.

ExfoUative cheilitis is an uncommon condition affecting the vermihon zone ofthe upper, lower or both lips. It is characterized by the continuous productionand desquamation of unsightly, thick scales of keratin; when removed, these leavea normal appearing lip beneath. The etiology is unknown, although some casesmay be factitious. Attempts at treatment by a wide variety of agents and tech-niques have been unsuccessful. Three patients with this disease are reported andits relationship to factitious cheilitis and candidal cheiUtis is discussed.

Key words: cheilitis; exfoliative cheilitis

Tom Daley, Department of Pathology,University of Westem Ontario, London,Ontario, Canada N6A 5C1

Accepted for publication May 15, 1994.

Exfoliative cheilitis is a chronic condi-tion which affects the vermilion zone ofthe upper, lower or, more commonly,both lips by the more or less con-tinuous, excessive production and sub-sequent desquamation of thick keratinscales. The disorder is restricted tothose cases not involving photosensitiv-ity or allergic reactions (1). A review ofthe world literature by READE & SIM (2)in 1986 disclosed only 179 cases, thegreat majority of which were reportedin the Russian and European literature.There is a female gender predilectionand the onset of most cases is before theage of 30 years (2). The characteristicpresence of desquamating flakes of ker-atin (3) is sometimes reported to be as-sociated with ulceration, fissuring, andbleeding (2, 4). Although some cases re-solve, at least temporarily (3, 4), otherspersist for years (4, 5). There is no ap-parent association with other dermato-logic or systemic diseases. This paperdescribes three new cases of this un-common disease.

Case 1

A healthy 17-year-old man of Arab de-scent complained of a four-month his-tory of scahng and flaking of the entirelower lip and the mid-portion of the up-per lip. Desquamation was followed im-mediately by the formation of newscales which became thick within days.Smaller scales desquamated asynchro-

nously from the vermilion, but occa-sionally a large scale involving most ofthe lower lip would form a slough.There was some variation in the severityof the condition from time to time. Ini-tially, there had been a tingling sensa-tion but pain, ulceration, fissuring andbleeding were denied. He denied exces-sive licking or biting of the lips, and hedenied skin, conjunctival and genitallesions.

Examination revealed large, thick,tan-coloured scales covering most ofthe vermilion zone of the lower lip andparts of the upper lip (Fig. la). Thesecould be detached easily and painlesslyin most places, leaving normal appear-ing underlying vermilion without asso-ciated erythema, ulceration, serouscrusting or significant fissuring. The ad-jacent skin and labial mucosa were notaffected.

Allergy testing with 30 common anti-gens was negative and the patient couldnot identify a specific initiating cause,although he thought it was worse if hesmoked cigarettes. Microscopic exami-nation of the scales revealed thick mem-branes of parakeratin (Fig. Ib) asso-ciated focally with numerous fungalspores interpreted to represent Candida,mixed bacteria, and foreign material.

The patient was advised to changehis toothpaste and any other oral hy-giene products to rule out a possible al-lergic reaction. There was no change inthe disorder. Ketoconazole cream was

prescribed for treatment of the fungalcontaminant, but no change in the epi-thelial derangement was noted. Topicaland systemic corticosteroids were un-successful, as was the application ofFucidin cream topically. Eventually, allforms of treatment were discontinued.The patient continued to remove thescales as they became loose, for cosme-tic reasons.

The disease process was the samenine months after its onset. However,during the 10th month, the patient re-ported that he had to "peel" his lips lessfrequently, and by the 15th month thelips were considered normal. He has nothad a relapse in three months.

Case 2

A 45-year-old Arab woman complainedof a 12-year history of continuous scal-ing and flaking of the vermilion of boththe upper and lower lips. There was noassociated ulceration, bleeding or deepfissuring. Pain was absent, although thepatient complained of mild soreness oran itchy feeling immediately followingepisodes of massive desquamation oflarge scales involving most of the ver-mihon of either or both lips. She didnot wear lipstick. She was advised tochange toothpaste but the condition re-mained unaltered. No other specific al-lergen could be identified. She had amaxillary denture, but the desquama-tive disorder started before it was made.

178 DALEY & GUPTA

Fig. 2). Case 2. Spores and hyphae (arrows)of Candida were present focally within theparakeratin strips (PAS, XlOO).

Fig. la). Case 1. Large keratin scales on thevermilion zone ofthe lips; Ib) microscopical-ly, the scales consisted of strips of parakera-tin (H & E, XlOO).

She denied licking and biting of the Upsprior to or since the onset of the dis-order. She denied skin, conjunctival orgenital lesions and was otherwise weU.

Examination revealed dry, tan to yel-low, thick scales some of which exhib-ited partial separation from the un-derlying tissue, involving most of theupper lip vermilion and patches of thelower lip vermilion. The skin and intra-oral labial mucosa were unaffected. Thescales could be easily and painlessly de-tached, leaving clinically normal ver-milion beneath. There was no associ-ated erythema, ulceration, deep fissur-ing or bleeding. Intra-oral examinationrevealed a Candida infection beneaththe maxillary denture. Microscopic ex-amination of the scales showed theywere identical to those of Case 1, butincluded the presence of hyphae of Can-dida focally (Fig. 2). Nystatin creamwas prescribed for the intraoral and thelabial candidiasis. The exfoliative liplesions did not respond to the antifun-gal therapy.

Other unsuccessful treatments in-cluded the use of keratinolytic agents(lactic acid 2%, salicyclic acid 3%, gly-colic acid 8%), topical corticosteroids,antibiotic creams, and petrolatum gels.

Six months later the disorder was un-changed, although there was a recur-rence of the candidal infection.

Fig. 3a). Case 3. A large scale involves mostof the lower lip vermilion; 3b) peeling of thescales left a clinically normal vermilion zonebeneath.

Case 3

A healthy 20-year-old white man com-plained of a 7-month history of con-tinuous, painless, patchy scaling andflaking of the upper and lower lip ver-milion zones. There was no history ofulceration, bleeding or deep fissuring.Prior to the onset of the condition, thepatient admitted to excessive lickingand gentle biting of the lips, often re-sulting in recurrent chapping. Thesehabits have been discontinued. He de-nied skin, conjunctival and genitallesions. No allergens could be iden-tified.

Examination revealed partially des-quamated tan scales involving most ofthe vermilion (Fig. 3a). These could beremoved easily and painlessly leaving

normal tissue beneath (Fig. 3b). Micro-scopic examination of the scales showedmembranous strips of parakeratin with-out fungal contamination.

Unsuccessful treatments included theuse of topical corticosteroids, topicalantibiotics, topical antifungal agents,petrolatum gels, and sunscreens. Cryo-surgery of the vermilion by liquid nitro-gen spray induced swelling and massivedesquamation. Upon healing, the dis-order returned. All treatment wasstopped and the patient was instructedto remove the scales as they becameloose, for cosmetic reasons. The condi-tion persists, although somewhat varia-bly, ten months after onset. Candidalspores were found on microscopic ex-amination of scales removed at ninemonths.

Discussion

Exfoliative cheilitis is a benign but oftencosmetically unsightly condition. Thethree patients presented herein, the se-cond patient reported by READE & SIM

(2), the patients reported by POSTLEW-

AiTE & HENDRICKSE (3), by BROOKE (4)and by TYLDESLEY (5) all appear toshow cheilitis associated predominantlywith keratin scales, usually in the ab-sence of ulceration. The etiology is un-known, although some cases, such asour third case, may be initiated but notnecessarily perpetuated by lip licking orbiting. Persistent crusting lip lesionswhether serous scabs or keratin scales,that are associated with self-inflicted in-jury have been termed "factitious cheili-tis". THOMAS et al (6), reported a vari-ety of "crusting" lip lesions in six pa-tients with psychiatric or emotionaldisorders, while CROTTY & DICKEN (7)reported 4 similar patients with "abnor-mal personality profiles". Although thelabial crusts found in some of these pa-tients are dominated by keratin scalesand could be diagnosed as exfoliativecheilitis, others appear to be dominatedby ulcerative lesions inconsistent withthis diagnosis. The confusion in termi-nology stems from the fact that "exfoli-ative cheilitis" is descriptive of thedisease process while "factitious cheili-tis" is descriptive of a presumed etiolo-gy. Some cases of exfoliative cheilitisappear to be related to factitious injuryand therefore could equally well be ac-curately diagnosed as factitious cheili-tis. Other cases do not appar to be re-lated to self-induced injury, and there-fore a diagnosis of factitious cheiUtiswould be inappropriate.

Candidal infection of the vermilion,apart from angular cheilitis, usuallypresents as a hemorrhagic, ulcerative,or crusting lesion of the lower lip thatresponds to antifungal therapy (8). Ker-atin scaling is not a characteristic fea-ture. A secondary candidal infection oc-curred in all three of our patients,which suggests that the keratin scalespresent a suitable environment for thespores and sometimes the hyphae of thefungus. Treatment with topical antifun-gal agents characteristically had no im-pact on the exfoliative process.

Actinic cheilitus (9) and cheilitisglandularis are not characterized by re-current episodes of desquamating,thick, hyperkeratotic scales (10). Somecases of cheilitis granulomatosa may beassociated with scaling of the vermilionbut the lips also exhibit the characteris-tic diffuse swelling typically seen to con-tain granulomaotus inflammation mi-croscopically (10).

Many attempts at treatment of exfo-liative cheilitis have failed. Topical andsystemic corticosteroids were unsuccess-ful (2, 3, 5), as was intralesional injec-tion of triamcinolone (4). Antifungalagents work against secondary fungalinfection but do not prevent the forma-tion of keratin scales (2, 3, 5). Topicaland systemic antibiotics have failed toalter the disease (3, 5), as have the ap-plication of several different types ofkeratolytic agents (3). Petrolatum gels,sunscreens, moisturizing preparationsand vitamin supplementation have been

equally ineffective (2-5). Radiationtherapy was unsuccessful in cases re-ported by Ti'LDESLEY (5) and by THOM-

AS et al. (case 4) (6). Cryotherapy wasunsuccessful in our cases 3. THOMAS etal. (6) reported a cure (their case 1)using 1% hydrocortisone cream; how-ever, the diagnosis of this case as exfoli-ative cheilitis is doubtful, as it is fortheir cases 2, 3 and 5, although their di-agnosis of factitious cheilitis may be ac-curate. A similar argument applied tocases 1 and 2 reported by CROTTY «feDiciCEN (7). BROOKE (4) reported a curefollowing measures to improve oral hy-giene but the follow-up period was only4 weeks. POSTLEWAITE & HENDRICKSE'S

(3) case spontaneously resolved after 6months, without treatment, but re-curred again an unspecified time later.Case 3 reported by CROTTY & DICKEN

(7) also resolved, apparently with theuse of amitriptyline and psychotherapy,but lesions of the lower Up had recurredby one year. The lip lesions of our case1 resolved spontaneously in the absenceof therapy and the lips have remainednormal for three months.

Exfoliative cheilitis presents a signi-ficant cosmetic problem. It often affectsyoung individuals who find themselvessocially handicapped. This can lead todepression varying from mild to severe.The interpretation by some authorsthat the disease is a result of a psycho-logical disorder relating to factitioushabits (1, 2, 6, 7) may be true for somecases. However, the concept of a re-

Exfoliative cheilitis 179

active psychological disorder occurringas a result of this disfiguring conditionmust also be considered, especially inthose cases where no factitious habitcan be identified by the patient or bythe clinician.

References

1. CHAMPION RH, BURTON J L , EBLINGFJG. In: Rook Wilkinson Ebling. Text-book of dermatology, 5th ed. Oxford:Blackwell Scientific, 1992: 2769.

2. READE PC, SIM R. Exfoliative cheiliitis: afactitious disorder? Int J Oral MaxillofacSurg 1986; 15: 313-7.

3. POSTLEWAITE KR, HENDRICKSE N M . Acase of exfoliative cheilitis. Br Dent J1988; 165: 23.

4. BROOKE R I . Exfoliative cheilitis. OralSurg Oral Med Oral Pathol 1978; 45: 52-5.

5. TVLDESLEY WR. Exfoliative cheilitis. BrJ Oral Surg 1973; 10: 357-9.

6. THOMAS JR, GREENE SL, DICKEN CH.Facititous cheilitis. / Am Acad Dermatol1983; 8: 368-72.

7. CROTTY CP, DICKEN CH. Factitious lipcrusting. Arch Dermatol 1981; 117: 338-40.

8. READE PC, RICH AM, HAY KD, RADDENBG. Cheilo-candidosis - a possible clin-ical entity. Report of 5 cases. Br Dent J1982; 152: 305-8.

9. REGEZI JA, SCIUBBA JJ. Oral pathology:clinical-pathologic correlations, 2nd ed.Philadelphia: Saunders, 1993: 102^.

10. SHAFER W G , HINE M K , LEVY B M . Atextbook of oral pathology. Philadel-phia: Saunders, 1983: 17-8.