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Case Report

Maxillary double lip: A case series with review

of literature
Vela D Desai, Sudakshina Das, Sunil MV Kumar1, Harsha Sadhwani
Departments of Oral Medicine and Radiology and 1Prosthodontics, Jaipur Dental College, Jaipur, Rajasthan, India

ABSTRACT

Double lip is a rare dental anomaly affecting either upper or lower lip or concurrently. It may be congenital or acquired.
This deformity can affect the facial esthetics as it gets exaggerated while speaking, smiling, or chewing food. When it
interferes with speech, double lip may cause potential functional problems. The purpose of this article is to review the
literature on double lip and present a case series of maxillary double lip.
Key words: Ascher’s syndrome, counseling, double lip, macrochelia, oral anomaly, pars glabrosa, pars villosa, surgery

Introduction Case Reports

D
ouble lip is a rare oral anomaly affecting the
lips and is characterized by an excess mass of
redundant mucosal tissue. The prevalence of
double lip or “macrochelia” affecting the upper lip
is greater than that of lower lip.[1,2] Current incidence
of double lip is unknown, although Calnan stated in
1952 that there were only a dozen cases reported in
literature.[3] English reported its rate of occurrence as
approximately 1 in 480 Chileans and 1 in 200 White
people in Utah.[4] Cases of double lip do occur in the
general population, but probably they are not well
documented in literature. Male predilection of 7:1 has
been reported.[5] Double lip may occur in isolation or
in association with Ascher’s syndrome, hemangioma,
bifid uvula, cleft palate, or cheilitis glandularis.[5-7] The
authors present a case series of maxillary double lip in
this article.
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Case 1
A 41-year-old healthy male patient reported to the
Department of Oral Medicine and Radiology with a
complaint of dirty teeth and bleeding gums for the last
3-4 months. The patient did not report any history of
trauma, allergy, and/or medical problems. Clinical
examination revealed an extra fold of tissue originating
from the inner aspect of the upper lip [Figure 1], which
was evident at rest but got accentuated when the patient
smiled. The fold of excess tissue was separated by a mild
midline constriction [Figure 2]. A provisional diagnosis
of congenital bilateral maxillary double lip and chronic
generalized periodontitis was made. As the patient
was unaware of this finding and denied any esthetic
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DOI: How to cite this article: Desai VD, Das S, Kumar SM, Sadhwani H.
10.4103/0972-1363.170173
Maxillary double lip: A case series with review of literature.
J Indian Acad Oral Med Radiol 2015;27:314-7.

Address for correspondence: Dr. Vela D Desai, B-4O6, Trimurthy Apartment, Opposite BSNL Telecom Colony, Malviya Nagar,
Jaipur - 302 017, Rajasthan, India. E-mail: veladesai@hotmail.com
Received: 27-01-2015  Accepted: 19-10-2015  Published: 21-11-2015

314 © 2015 Journal of Indian Academy of Oral Medicine and Radiology | Published by Wolters Kluwer - Medknow
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Desai VD et al.: Double lip
Figure 1: A fold of excess maxillary labial tissue
or functional complaints, no surgical correction was
instituted. He was treated for generalized periodontitis,
counselled, and followed up.
Case 2
A 21-year-old female patient visited the outpatient
department of Oral Medicine and Radiology with a
chief complaint of missing upper anterior tooth for
the last 6 months. Medical and family histories were
non-contributory. There was no history of lip sucking
habit. Examination revealed additional labial tissue
on the upper lip [Figure 3]. It appeared to involve the
entire upper lip like a uniform swelling rather than as
an exuberant tissue extending from the inner aspect
without any midline constriction. It was not visible at
rest, but became evident when the patient smiled. It
appeared more prominent on the right side than on the
left half of the lip because of the missing upper central
incisors. The patient was aware of this anomaly since
birth and no appreciable changes were noted since
then. There were no other congenital abnormalities. A
provisional diagnosis of bilateral maxillary double lip
was made. Hematological investigations revealed no
significant finding. Since the patient had no complaint
with double lip, no treatment was done, except for
replacement of missing upper teeth with implant-
supported prosthesis [Figure 4].
Case 3
A 21-year-old young female patient reported to the
department with the complaint of food lodgment in left
lower back teeth region for the last 1 year. There was no
relevant history of trauma, lip biting, or medical illness.
Clinical examination revealed bilateral maxillary double
lip which became pronounced as the patient smiled
[Figure 5]. The patient was unaware of this appearance.
The tissue did not appear to extend from the inner
surface of the lip, but rather appeared as a uniform
swelling without midline constriction.
Journal of Indian Academy of Oral Medicine & Radiology | Apr-Jun 2015 | Vol 27 | Issue 2
Figure 2: Labial tissue approaching the cervical third of maxillary incisors
Case 4
A 55-year-old healthy male patient came to the Oral
Medicine department with the complaint of missing teeth
in the upper arch for the last 1 year. Medical history was
non-contributory. The patient did not report any history
of trauma or drug allergy. Bilateral maxillary double lip
was not evident during rest, but was accentuated during
smiling [Figure 6]. The edentulous maxillary alveolar
ridge made the double lip appear prominent. History
of consanguinity was present in all the patients, but
none of the family members revealed similar findings.
Relevant blood investigations were carried out for all the
cases to rule out any thyroid involvement. Since none
of the patients had any functional or esthetic problem,
no surgical treatment for the double lip was done; their
families were counseled and were regularly followed up.
Discussion
Double lip is usually present at birth, but this congenital
dental anomaly becomes evident after the eruption of
teeth. Congenital double lip is thought to arise during
the second or third month of gestation owing to the
persistence of a sulcus between the pars glabrosa (outer
part) that is smooth and similar to skin and the pars
villosa (inner part) of the lip that is similar to the oral
mucosa. [8] Acquired deformity may be secondary to
trauma or habits such as sucking lips between diastema
or ill-fitting dentures. This is generally not evident at
rest, but becomes prominent when the labial tissue
gets tensed as during smiling or talking.[9] On smiling,
the lip assumes a Cupid’s bow shape that is attributed
to the midline constriction between pars glabrosa and
pars villosa.[1] During smiling, the orbicularis oris, one
of the muscles of facial expression, retracts leading to
repositioning of the mucosa over the maxillary teeth,
thus exaggerating the double lip, which was also evident
in two of the cases (cases 1 and 4) presented.[10] These
hamartomas may present either as unilateral or bilateral
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Desai VD et al.: Double lip
Figure 3: Bilateral asymmetrical maxillary double lip
Figure 5: Maxillary double lip, as the patient smiled
hyperplastic tissue. Also, sometimes, these bilateral
hyperplastic tissues are asymmetrical with one side
being bigger than the other, as in the second case.[5]
Double lip is a clinical diagnosis; laborious investigative
procedures are not indicated for the diagnosis. The
differential diagnosis of double lip must include
hemangioma, angioedema, and cheilitis glandularis.[6,7]
It is important to distinguish double lip from cheilitis
glandularis, as the latter is coupled with increased risk
of squamous cell carcinoma.[7] Ascher’s syndrome is an
important differential diagnosis characterized by a triad
of double lip, blepharochalasis, and a medical history of
non-toxic thyroid enlargement.[8] Parmar and Muranjan
reported a newly recognized syndrome with double
upper and lower lips, hypertelorism, eyelid ptosis,
blepharophimosis, and third finger clinodactyly.[11]
Surgical intervention is required for double lip if there is
esthetic or functional impairment, and involves excision
of excess mucosa and submucosa sparing the underlying
connective tissue.[2,8,9] Many surgical techniques have
316 Journal of Indian Academy of Oral Medicine & Radiology | Apr-Jun 2015 | Vol 27 | Issue 2
Figure 4: Maxillary double lip- as the patient smiled. Also, missing teeth
replaced by implant (Healing abutment, second stage)
Figure 6: Accentuated maxillary double lip in edentulous arch
been described for the correction of double lip: W-plasty,
electrosurgical excision, triangular excision, and transverse
elliptical excision.[10,12] Histopathological investigation of
the excised tissue reveals mucous glands, numerous blood
capillaries, areolar tissue, and perivascular infiltration
with plasma cells and lymphocytes.[13,14]
Conclusion
Double lip is an infrequently recorded and reported
anomaly of the lip. Esthetics and functional needs may
require surgical corrections which provide excellent
prognosis. [1,15] All the four cases in this report had
non-syndromic bilateral maxillary double lip. Two of the
presented cases (cases 2 and 3) had a varied presentation
of the excess mass of labial tissue that was seen as a
swelling involving whole of the upper lip, rather than
projecting from the inner aspect of the lip without any
midline constriction which contributes to the existing
literature. Double lip is of special interest in dental
profession, as dentists may normally be the first ones to
diagnose this rare and uncommon condition.
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Desai VD et al.: Double lip

Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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