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Cheilitis glandularis: a rare occurrence involving the upper lip

Article · December 2010

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Sowbhagya Malligere Basavaraju

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 Sowbhagya M. B.*
MDS CASE REPORT Hong Kong Dent J 2010;7:101-4
Maria Priscilla David *

MDS
Ramachandra Reddy Gowda
Venkatesha*
Cheilitis glandularis: a rare occurrence involving
Indira Annamallai
MDS
the upper lip
Ponnuswamy*
MDS

*
  M.R. Ambedkar Dental College and Hospital,
Bangalore, India (affiliated to Rajiv Gandhi
University of Heath Sciences)
ABSTRACT
A variety of pathologic conditions of diverse etiologies may involve the lips. Cheilitis
glandularis is a rare disorder of unknown etiology characterized by inflammation of
the minor salivary glands, predominantly those of the lower lip. With externalization
and chronic exposure, the delicate labial mucous membrane lining the minor salivary
glands are altered by environmental influences, leading to erosion, ulcerations, crusting
and, occasionally, infection. Importantly, susceptibility to actinic damage is increased.
Therefore, cheilitis glandularis can be considered a potential predisposing factor for the
development of actinic cheilitis and squamous cell carcinoma. This report is of a 12-year-
old boy with cheilitis glandularis in an uncommon site, the upper lip. An overview of
the etiology, clinical presentation, histopathologic features, and treatment are also
discussed.

Key words: Child; Cheilitis; Salivary glands

Introduction

Cheilitis glandularis (CG) is a clinical diagnosis that refers to an uncommon, poorly

understood, benign inflammatory disorder of the minor salivary glands in the lower lip.
Although some researchers have speculated that CG represents a hereditary autosomal
dominant condition, composite findings in most patients indicate that the condition
represents a clinical reaction to chronic irritation of the lip from diverse external causes,
such as actinic damage, factitial injury, atopy, infection, and tobacco irritation 1. Therefore,
CG can be considered a potential predisposing factor for the development of actinic
cheilitis and squamous cell carcinoma. This report is of an unusual occurrence of CG in an
uncommon site, the upper lip of a 12-year-old boy.

Case report

A 12-year-old boy presented to the M. R. Ambedkar Dental College and Hospital, Bangalore,
Correspondence to: India, in 2005 with diffuse swelling of the upper lip for the previous 1.5 years. The patient
Dr Sowbhagya M. B.
31/1, 1st Main, 2nd Cross, Azad Nagar,
was conscious of the lip swelling, although it was asymptomatic, and desired orthodontic
Bangalore, India 560018 treatment for his proclined upper front teeth. He had a habit of licking and biting the lip
Tel : (080-26) 745649
Fax : (082-74) 260156
for the previous 3 years. He also complained of excessive drying of the lip from mouth
email : sowbhagya143@gmail.com breathing and constant exposure to wind.

© Hong Kong Dental Association 101

M. B. et al

At examination, his lips were incompetent and the unknown etiology characterized by inflammation of the
swelling measured approximately 1.5 x 2.5 cm, and was oval- minor salivary glands, predominantly those of the lower
shaped and pale pink in color (Fig 1). There was associated lip. Von Volkmann 5 also described a clinically distinct,
crusting and scaling of the lip. On palpation, the swelling on deeply suppurative, chronic inflammatory condition of
the upper lip was soft in consistency, mobile, and fluctuant. the lower lip characterized by mucopurulent exudates
On application of pressure, a discharge of mucinous fluid
was seen. Intraoral examination revealed increased overjet
and deep bite with two supernumerary teeth present
between the upper central incisors, with no evidence of
dental caries. Based on these findings, a provisional clinical
diagnosis of CG was reached and differential diagnoses of
double lip, angioedema, cheilitis granulomatosa, orofacial
granulomatosis, chronic factitial injury, and actinic cheilitis
were considered 2.

The patient underwent routine blood and radiological

investigations. Based on the patient’s desire for orthodontic
treatment, an orthopantomograph was taken, which
revealed an impacted permanent left upper canine (23)
and a retained primary left upper canine (63). A lateral
cephalograph was taken to see the soft tissue profile where
the increase in the size of the upper lip was evident (Fig 2).
Routine blood investigation showed normal parameters for
all tests except for erythrocyte sedimentation rate, which
Figure 1 Clinical photograph showing swelling with crusting and
was slightly increased to 26 mm/h (reference range, 0-20
scaling of the upper lip
mm/h). A differential diagnosis of double lip was considered
as the condition could be associated with Ascher’s
syndrome 3, but this was ruled out by a complete physical
examination and the clinical opinion of a general medicine
consultant. The patient was referred to the Department of
Oral and Maxillofacial Surgery for cosmetic management,
and a transverse elliptical incision with debulking by surgical
cheiloplasty was done under local anesthesia to restore
normal lip architecture and function.

Hematoxylin and eosin–stained section revealed

parakeratinized stratified squamous epithelium with a few
areas of spongiosis (Fig 3). The underlying connective tissue
showed numerous dilated blood vessels with perivascular
inflammatory infiltrate and inflamed minor salivary acinar
units and ducts (Fig 4). The histology findings confirmed the
diagnosis of CG 4.

Discussion
Figure 2 Lateral cephalograph showing the soft tissue profile
Von Volkmann 5
introduced CG for a rare disorder of where the increase in the size of the upper lip was evident

102 Hong Kong Dent J Vol 7 No 2 December 2010


from the ductal orifices of the labial minor salivary glands.
Sutton 6 proposed that the characteristic lip swelling was
attributable to a congenital adenomatous enlargement
of the labial salivary glands. The incidence of CG is rare. In
many patients, dysplastic (premalignant) surface epithelial
change is evident histopathologically and frank carcinomas
have been reported in 18 to 35% of patients 7. The condition
often occurs in middle-aged or elderly men, but it also
occurs in children, teenagers, and young adults. The risk for
Figure 3 Photomicrograph showing stratified squamous
epithelium and underlying dense connective tissue with numerous
dilated blood vessels and dilated ducts (H&E, x 4)
Figure 4 Photomicrograph of the minor salivary gland with
features of chronic inflammatory response around the gland (H&E, x 40)
Cheilitis glandularis
dysplasia and carcinoma increases with age, especially in
fair-skinned individuals with sun-damaged skin, because
the characteristic eversion of the lower lip results in long-
term chronic exposure of the thin vulnerable labial mucosa
to actinic influence.
Patients typically present for diagnostic consultation
within 3 to 12 months of onset 8. The presentation varies
according to the degree of pain, enlargement, loss of
elasticity, and surface changes. Asymptomatic lip swelling
initially occurs with clear viscous secretion expressed from
dilated ductal openings on the mucosal surface. Some
patients report periods of relative quiescence interrupted
by transient or persistent painful episodes associated
with suppurative discharge 9. Cheilitis glandularis affects
the lower lip almost exclusively, and CG of the upper lip
is a rare oral anomaly 10. Cheilitis glandularis manifests as
progressive, often multinodular enlargement, eversion, and
induration. Salivary gland duct orifices may be dilated, with
spontaneous discharge of viscous clear secretions. In more
suppurative disease, application of gentle pressure can elicit
mucopurulent exudates. With advancing lip prominence
and mucosal eversion, the mucosal-vermilion junction is
obscured. Prolonged exposure to the external environment
results in desiccation and disruption of the labial mucous
membrane, predisposing the underlying minor salivary
glands to inflammation, infection, and actinic influences 11.
In this patient, CG could have been caused by a combination
of factors such as actinic exposure, mouth breathing, and
habitual lip biting.
Cheilitis glandularis has been subclassified into three
types: simple, superficial suppurative, and deep suppurative.
The superficial suppurative type has been termed Baelz’s
disease, and the deep suppurative type has been referred
to as myxadenitis labialis or cheilitis apostematosa 5,6. It
is thought that these subtypes represent a continuum
of disease wherein the simple type, if not treated, could
become secondarily infected and progress to become
Baelz’s disease and, eventually, the deep suppurative type.
Chronic abscess and fistula formation are commonly seen
in immunosuppressed patients. The latter two types of CG
have the highest association with dysplasia and carcinoma.
Lip enlargement is attributable to inflammation, hyperemia,
edema, and fibrosis. Surface keratosis, erosion, and crusting
develop consequent to long-standing actinic exposure,
Hong Kong Dent J Vol 7 No 2 December 2010 103
M. B. et al
unusual repeated manipulations that include self-biting or
other factitial trauma, excessive wetting from compulsive
licking, drying (sometimes associated with mouth breathing,
atopy, eczema, and asthma), and other repeated stimuli that
serve as a chronic aggravating factor 12. The histopathologic
findings for this patient were consistent with those of CG.
Differential diagnosis should include double lip,
angioedema, lymphangioma, chronic factitial injury, cheilitis
granulomatosa, and orofacial granulomatosis 2,3,13. Treatment
of CG is by vermilionectomy and antibiotic therapy for
suppurative disease. Medical management of CG includes
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