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Exfoliative Cheilitis: Case Reports

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51 views4 pages

Exfoliative Cheilitis: Case Reports

jurnal

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Asep J Permana
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© © All Rights Reserved
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J Oral Pathol Med 1995: 24: 177-9

Printed in Denmark . All rights reserved

Copyright

Munksgaard

1995

JOURNAL OF

Oral PathciogysMsdidne
ISSN 0904-2512

Case reports

Exfoliative cheilitis

Tom D. Daley^ and Adytia K. Gupta^


^Department of Pathology, University of
Western Ontario, London, ^Division of
Dermatology, 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 of
the upper, lower or both lips. It is characterized by the continuous production
and desquamation of unsightly, thick scales of keratin; when removed, these leave
a normal appearing lip beneath. The etiology is unknown, although some cases
may be factitious. Attempts at treatment by a wide variety of agents and techniques have been unsuccessful. Three patients with this disease are reported and
its relationship to factitious cheilitis and candidal cheiUtis is discussed.

Exfoliative cheilitis is a chronic condition which affects the vermilion zone of


the upper, lower or, more commonly,
both lips by the more or less continuous, excessive production and subsequent desquamation of thick keratin
scales. The disorder is restricted to
those cases not involving photosensitivity or allergic reactions (1). A review of
the world literature by READE & SIM (2)
in 1986 disclosed only 179 cases, the
great majority of which were reported
in the Russian and European literature.
There is a female gender predilection
and the onset of most cases is before the
age of 30 years (2). The characteristic
presence of desquamating flakes of keratin (3) is sometimes reported to be associated with ulceration, fissuring, and
bleeding (2, 4). Although some cases resolve, at least temporarily (3, 4), others
persist for years (4, 5). There is no apparent association with other dermatologic or systemic diseases. This paper
describes three new cases of this uncommon disease.
Case 1

A healthy 17-year-old man of Arab descent complained of a four-month history of scahng and flaking of the entire
lower lip and the mid-portion of the upper lip. Desquamation was followed immediately by the formation of new
scales which became thick within days.
Smaller scales desquamated asynchro-

nously from the vermilion, but occasionally a large scale involving most of
the lower lip would form a slough.
There was some variation in the severity
of the condition from time to time. Initially, there had been a tingling sensation but pain, ulceration, fissuring and
bleeding were denied. He denied excessive licking or biting of the lips, and he
denied skin, conjunctival and genital
lesions.
Examination revealed large, thick,
tan-coloured scales covering most of
the vermilion zone of the lower lip and
parts of the upper lip (Fig. la). These
could be detached easily and painlessly
in most places, leaving normal appearing underlying vermilion without associated erythema, ulceration, serous
crusting or significant fissuring. The adjacent skin and labial mucosa were not
affected.
Allergy testing with 30 common antigens was negative and the patient could
not identify a specific initiating cause,
although he thought it was worse if he
smoked cigarettes. Microscopic examination of the scales revealed thick membranes of parakeratin (Fig. Ib) associated focally with numerous fungal
spores interpreted to represent Candida,
mixed bacteria, and foreign material.
The patient was advised to change
his toothpaste and any other oral hygiene products to rule out a possible allergic reaction. There was no change in
the disorder. Ketoconazole cream was

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.

prescribed for treatment of the fungal


contaminant, but no change in the epithelial derangement was noted. Topical
and systemic corticosteroids were unsuccessful, as was the application of
Fucidin cream topically. Eventually, all
forms of treatment were discontinued.
The patient continued to remove the
scales as they became loose, for cosmetic reasons.
The disease process was the same
nine months after its onset. However,
during the 10th month, the patient reported that he had to "peel" his lips less
frequently, and by the 15th month the
lips were considered normal. He has not
had a relapse in three months.
Case 2

A 45-year-old Arab woman complained


of a 12-year history of continuous scaling and flaking of the vermilion of both
the upper and lower lips. There was no
associated ulceration, bleeding or deep
fissuring. Pain was absent, although the
patient complained of mild soreness or
an itchy feeling immediately following
episodes of massive desquamation of
large scales involving most of the vermihon of either or both lips. She did
not wear lipstick. She was advised to
change toothpaste but the condition remained unaltered. No other specific allergen could be identified. She had a
maxillary denture, but the desquamative disorder started before it was made.

178

DALEY & GUPTA

Fig. 2). Case 2. Spores and hyphae (arrows)


of Candida were present focally within the
parakeratin strips (PAS, XlOO).

normal tissue beneath (Fig. 3b). Microscopic examination of the scales showed
membranous strips of parakeratin without fungal contamination.
Unsuccessful treatments included the
use of topical corticosteroids, topical
antibiotics, topical antifungal agents,
petrolatum gels, and sunscreens. Cryosurgery of the vermilion by liquid nitrogen spray induced swelling and massive
desquamation. Upon healing, the disorder returned. All treatment was
stopped and the patient was instructed
to remove the scales as they became
loose, for cosmetic reasons. The condition persists, although somewhat variably, ten months after onset. Candidal
spores were found on microscopic examination of scales removed at nine
months.
Discussion

Fig. la). Case 1. Large keratin scales on the


vermilion zone ofthe lips; Ib) microscopically, the scales consisted of strips of parakeratin (H & E, XlOO).

She denied licking and biting of the Ups


prior to or since the onset of the disorder. She denied skin, conjunctival or
genital lesions and was otherwise weU.
Examination revealed dry, tan to yellow, thick scales some of which exhibited partial separation from the underlying tissue, involving most of the
upper lip vermilion and patches of the
lower lip vermilion. The skin and intraoral labial mucosa were unaffected. The
scales could be easily and painlessly detached, leaving clinically normal vermilion beneath. There was no associated erythema, ulceration, deep fissuring or bleeding. Intra-oral examination
revealed a Candida infection beneath
the maxillary denture. Microscopic examination of the scales showed they
were identical to those of Case 1, but
included the presence of hyphae of Candida focally (Fig. 2). Nystatin cream
was prescribed for the intraoral and the
labial candidiasis. The exfoliative lip
lesions did not respond to the antifungal therapy.
Other unsuccessful treatments included the use of keratinolytic agents
(lactic acid 2%, salicyclic acid 3%, glycolic acid 8%), topical corticosteroids,
antibiotic creams, and petrolatum gels.
Six months later the disorder was unchanged, although there was a recurrence of the candidal infection.

Fig. 3a). Case 3. A large scale involves most


of the lower lip vermilion; 3b) peeling of the
scales left a clinically normal vermilion zone
beneath.

Case 3

A healthy 20-year-old white man complained of a 7-month history of continuous, painless, patchy scaling and
flaking of the upper and lower lip vermilion zones. There was no history of
ulceration, bleeding or deep fissuring.
Prior to the onset of the condition, the
patient admitted to excessive licking
and gentle biting of the lips, often resulting in recurrent chapping. These
habits have been discontinued. He denied skin, conjunctival and genital
lesions. No allergens could be identified.
Examination revealed partially desquamated tan scales involving most of
the vermilion (Fig. 3a). These could be
removed easily and painlessly leaving

Exfoliative cheilitis is a benign but often


cosmetically unsightly condition. The
three patients presented herein, the second patient reported by READE & SIM
(2), the patients reported by POSTLEWAiTE & HENDRICKSE (3), by BROOKE (4)
and by TYLDESLEY (5) all appear to
show cheilitis associated predominantly
with keratin scales, usually in the absence of ulceration. The etiology is unknown, although some cases, such as
our third case, may be initiated but not
necessarily perpetuated by lip licking or
biting. Persistent crusting lip lesions
whether serous scabs or keratin scales,
that are associated with self-inflicted injury have been termed "factitious cheilitis". THOMAS et al (6), reported a variety of "crusting" lip lesions in six patients with psychiatric or emotional
disorders, while CROTTY & DICKEN (7)
reported 4 similar patients with "abnormal personality profiles". Although the
labial crusts found in some of these patients are dominated by keratin scales
and could be diagnosed as exfoliative
cheilitis, others appear to be dominated
by ulcerative lesions inconsistent with
this diagnosis. The confusion in terminology stems from the fact that "exfoliative cheilitis" is descriptive of the
disease process while "factitious cheilitis" is descriptive of a presumed etiology. Some cases of exfoliative cheilitis
appear to be related to factitious injury
and therefore could equally well be accurately diagnosed as factitious cheilitis. Other cases do not appar to be related to self-induced injury, and therefore a diagnosis of factitious cheiUtis
would be inappropriate.

Exfoliative cheilitis 179


Candidal infection of the vermilion,
apart from angular cheilitis, usually
presents as a hemorrhagic, ulcerative,
or crusting lesion of the lower lip that
responds to antifungal therapy (8). Keratin scaling is not a characteristic feature. A secondary candidal infection occurred in all three of our patients,
which suggests that the keratin scales
present a suitable environment for the
spores and sometimes the hyphae of the
fungus. Treatment with topical antifungal agents characteristically had no impact on the exfoliative process.
Actinic cheilitus (9) and cheilitis
glandularis are not characterized by recurrent episodes of desquamating,
thick, hyperkeratotic scales (10). Some
cases of cheilitis granulomatosa may be
associated with scaling of the vermilion
but the lips also exhibit the characteristic diffuse swelling typically seen to contain granulomaotus inflammation microscopically (10).
Many attempts at treatment of exfoliative cheilitis have failed. Topical and
systemic corticosteroids were unsuccessful (2, 3, 5), as was intralesional injection of triamcinolone (4). Antifungal
agents work against secondary fungal
infection but do not prevent the formation of keratin scales (2, 3, 5). Topical
and systemic antibiotics have failed to
alter the disease (3, 5), as have the application of several different types of
keratolytic agents (3). Petrolatum gels,
sunscreens, moisturizing preparations
and vitamin supplementation have been

equally ineffective (2-5). Radiation


therapy was unsuccessful in cases reported by Ti'LDESLEY (5) and by THOMAS et al. (case 4) (6). Cryotherapy was
unsuccessful in our cases 3. THOMAS et
al. (6) reported a cure (their case 1)
using 1% hydrocortisone cream; however, the diagnosis of this case as exfoliative cheilitis is doubtful, as it is for
their cases 2, 3 and 5, although their diagnosis of factitious cheilitis may be accurate. A similar argument applied to
cases 1 and 2 reported by CROTTY fe
DiciCEN (7). BROOKE (4) reported a cure
following measures to improve oral hygiene but the follow-up period was only
4 weeks. POSTLEWAITE & HENDRICKSE'S
(3) case spontaneously resolved after 6
months, without treatment, but recurred again an unspecified time later.
Case 3 reported by CROTTY & DICKEN
(7) also resolved, apparently with the
use of amitriptyline and psychotherapy,
but lesions of the lower Up had recurred
by one year. The lip lesions of our case
1 resolved spontaneously in the absence
of therapy and the lips have remained
normal for three months.
Exfoliative cheilitis presents a significant cosmetic problem. It often affects
young individuals who find themselves
socially handicapped. This can lead to
depression varying from mild to severe.
The interpretation by some authors
that the disease is a result of a psychological disorder relating to factitious
habits (1, 2, 6, 7) may be true for some
cases. However, the concept of a re-

active psychological disorder occurring


as a result of this disfiguring condition
must also be considered, especially in
those cases where no factitious habit
can be identified by the patient or by
the clinician.

References
1. CHAMPION RH,

BURTON J L , EBLING

FJG. In: Rook Wilkinson Ebling. Textbook of dermatology, 5th ed. Oxford:
Blackwell Scientific, 1992: 2769.
2. READE PC, SIM R . Exfoliative cheiliitis: a
factitious disorder? Int J Oral Maxillofac
Surg 1986; 15: 313-7.
3. POSTLEWAITE KR, HENDRICKSE N M . A

case of exfoliative cheilitis. Br Dent J


1988; 165: 23.
4. BROOKE R I . Exfoliative cheilitis. Oral
Surg Oral Med Oral Pathol 1978; 45: 525.
5. TVLDESLEY WR. Exfoliative cheilitis. Br
J Oral Surg 1973; 10: 357-9.
6. THOMAS JR, GREENE S L , DICKEN CH.

Facititous cheilitis. / Am Acad Dermatol


1983; 8: 368-72.
7. CROTTY CP, DICKEN CH. Factitious lip
crusting. Arch Dermatol 1981; 117: 33840.
8. READE PC, RICH AM, HAY KD, RADDEN

BG. Cheilo-candidosis - a possible clinical entity. Report of 5 cases. Br Dent J


1982; 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 . A

textbook of oral pathology. Philadelphia: Saunders, 1983: 17-8.

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