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 Table of Contents  
Year : 2017  |  Volume : 4  |  Issue : 3  |  Page : 216-218

Congenital double-double lip: A rare case report

1 Department of Oral Medicine and Radiology, Dentist at Private Practice, Jind, Haryana, India
2 Department of Prosthodontics, Dentist at Private Practice, Jind, Haryana, India

Date of Web Publication13-Jul-2017

Correspondence Address:
Swati Phore
Department of Oral Medicine and Radiology, Private Practice, Haryana
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/cjhr.cjhr_25_17

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A double lip is a rare anomaly characterized by a horizontal fold of redundant mucosal tissue that is situated proximal to the vermilion border. It may be either congenital or acquired and has no gender or race predilection. It occurs most often in the upper lip, although both upper and lower lips are occasionally involved. Surgical intervention (simple excision) produces good functional and cosmetic results. In this report, a case of a nonsyndromic congenital maxillary double upper lip and lower lip, both are described. Double lip is of special interest in dental profession as a dental surgeon is normally the first one to diagnose this rare and uncommon condition. Authors hereby discuss a very rare condition in which both upper and lower lips are involved.

Keywords: Congenital, double lip, vermilion border

How to cite this article:
Phore S, Panchal RS. Congenital double-double lip: A rare case report. CHRISMED J Health Res 2017;4:216-8

How to cite this URL:
Phore S, Panchal RS. Congenital double-double lip: A rare case report. CHRISMED J Health Res [serial online] 2017 [cited 2022 Jan 25];4:216-8. Available from: https://www.cjhr.org/text.asp?2017/4/3/216/210486

  Introduction Top

Double lip is an infrequent oral anomaly of congenital or acquired origin that is equally prevalent in both genders and also shows no racial predilection.[1] It is also referred to as macrocheilitis.[2] It usually affects the upper lip bilaterally, although it can also occur unilaterally in both the upper and lower lips. The condition consists of a fold of excess or redundant hypertrophic tissue on the mucosal side of the lip.[1]

It is caused by hyperplastic tissue of the labial mucosa that becomes more prominent with tension caused by smiling. During smiling, the lip is retracted and the mucosa is positioned over the maxillary teeth, resulting in a “cupid's bow” appearance whereas it is not evident when the mouth is closed.[3]

The current incidence of the double lip is unknown, although Calnan stated in 1952 that only a dozen are cases reported in the literature.[4]

These deformities, though not posing functional problems, may cause severe psychological distress to the affected person because of the disfiguring effect on smiling.[5]

  Case Report Top

A 32-year-old male patient [Figure 1] came to the dental clinic with a chief complaint of dirty teeth. The patient did not report any relevant medical/family history, with no history of allergy or drugs. On examination, a thick, extra fold of redundant tissue was present on the inner surface of both upper [Figure 2] and lower lip [Figure 3], with midline constriction band between two mucosal bulges which was even visible at rest. The swellings were of normal mucosal color and on palpation were soft in consistency, mobile and fluctuant. On further evaluation, there was a negative history of trauma, lip sucking, or lip biting. The hyperplastic tissue was present since birth with no change in appearance or size. There was no blepharochalasis and no thyroid gland enlargement.
Figure 1: Extraoral profile of the patient

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Figure 2: Upper lip

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Figure 3: Lower lip

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A provisional diagnosis of congenital double-double lip was made. As the patient had no problem with the hypertrophic tissues, so was left untreated.

  Discussion Top

Double lip is an accessory fold of redundant mucous membrane inside the vermilion border. Apart from a deformity that interferes with speech and mastication, the operation may be indicated for cosmetic reasons.[6] Acquired cases of the disease usually result from trauma, while congenital cases stem from a developmental anomaly. During development, the upper lip mucosa is made up of two transversal zones: an outer cutaneous zone (pars glabra) and an inner mucosal zone (pars villosa).[5],[7]

Although the enlargement of the lip may be present from birth, it can become more apparent after the eruption of the teeth. Moreover, it has been suggested that the original double lip may be enhanced by a reactive process after a “sucking-in” of the tissue between the teeth, or mal-occluding dentures.[8],[9]

Reported histological findings include prominent salivary glands and mixed inflammatory cell infiltration.[10]

Since the present case did not reveal any history of thyroid disturbance or blepharochalasis we consider it as nonsyndromic type of double-double lip. But because of progressive nature of disorder or suspected Ascher's syndrome, the patient should be followed up because blepharochalasis and nontoxic enlargement of thyroid gland can develop later.[1],[11]

The differential diagnosis should also include vascular tumors, lymphangioma, angioedema, cheilitis granulomatosis, Miescher syndrome, mucocele, salivary gland tumors, inflammatory fibrous hyperplasia, sarcoidosis, and plasma cell cheilitis. Such lesions are frequently associated with a uniformly enlarged lip without a midline constriction dividing the lip.[8]

Treatment is indicated when the condition interferes with speech and chewing, or for cosmetic reasons. Various surgical techniques to correct a double lip have been described, but simple excision through an elliptical incision is usually recommended.[12],[13]

  Conclusion Top

Double lip is a clinical diagnosis, and no laborious investigations are required. Surgical treatment is required if it pose cosmetic or functional problems, or may have a serious impact on psychological well-being. Lasers can be used for the cosmetic correction. As our patient was not esthetically worried about his appearance, he was only kept under observation.

This sporadic oral anomaly is of special interest for a reason that it is a very rare finding to involve both lips and only a few cases have been reported till date in the literature.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Chidzonga MM, Mahomva L. Congenital double lower lip: Report of a case. Int J Paediatr Dent 2006;16:448-9.  Back to cited text no. 1
Lamster IB. Mucosal reduction for correction of a maxillary double lip. Report of a case. Oral Surg Oral Med Oral Pathol 1983;55:457-8.  Back to cited text no. 2
Bhatia V. Unilateral maxillary double lip – A rare case report. J Adv Dent Sci Res 2014;2:89-92.  Back to cited text no. 3
Goyal S, Godhi S, Goyal S. Non-syndromic congenital maxillary double lip: A rare case. J Oral Health Community Dent 2008;2:10-2.  Back to cited text no. 4
Suliman MT, Alhassan M. Double lip: Report of five cases and review of the literature. Aesthet Surg J 2007;27:289-91.  Back to cited text no. 5
Mago V. Congenital double lip. Indian J Med Case Rep 2012;1:13-4.  Back to cited text no. 6
Ali K. Ascher syndrome: A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:e26-8.  Back to cited text no. 7
Hanemann JA, Oliveira DT, Gomes MF, dos Anjos MJ, Sant'ana E. Congenital double lip associated to hemangiomas: Report of a case. Med Oral 2004;9:156-8, 155-6.  Back to cited text no. 8
Alkan A, Metin M. Maxillary double lip: Report of two cases. J Oral Sci 2001;43:69-72.  Back to cited text no. 9
Gomez-Duaso AJ, Seoane J, Vazquez-Garcia J, Arjona C. Ascher syndrome: Report of two cases. J Oral Maxillofac Surg 1997;55:88-90.  Back to cited text no. 10
Martins WD, Westphalen FH, Sandrin R, Campagnoli E. Congenital maxillary double lip: Review of the literature and report of a case. J Can Dent Assoc 2004;70:466-8.  Back to cited text no. 11
Eski M, Nisanci M, Aktas A, Sengezer M. Congenital double lip: Review of 5 cases. Br J Oral Maxillofac Surg 2007;45:68-70.  Back to cited text no. 12
Santos PP, Alves PM, Freitas VS, Souza LB. Double lip surgical correction in Ascher's syndrome: Diagnosis and treatment of a rare condition. Clinics (Sao Paulo) 2008;63:709-12.  Back to cited text no. 13


  [Figure 1], [Figure 2], [Figure 3]


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