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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 2  |  Issue : 1  |  Page : 35-38

An appraisal of congenital maxillary double lip with a case report


1 Department of Oral and Maxillofacial Pathology, Yamuna Institute of Dental Sciences and Research, Rohtak, India
2 Oral Surgeon, Goverment Dental College, Rohtak, India
3 Department of Periodontics, Yamuna Institute of Dental Sciences and Research, Yamuna Nagar, Haryana, India
4 Oral Pathologist, Yamuna Institute of Dental Sciences and Research, Yamuna Nagar, Haryana, India

Date of Web Publication2-Feb-2015

Correspondence Address:
Preeti Sethi Bakshi
2A, Family Dental Clinic, Professor Colony, Yamuna Nagar, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1658-6816.150594

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  Abstract 

Double lip is a rare anomaly that may be congenital or acquired and affects upper lip more commonly than the lower lip. This deformity consists of an accessory fold of redundant mucous membrane inside the vermilion border. It affects esthetics and may also interfere with speech and mastication. Simple surgical excision produces good functional and cosmetic results. We describe a case of congenital maxillary double lip along with a brief review of the literature and discuss the differential diagnosis. This anomaly is of more relevance in dentistry as a general dental practitioner is usually, the first professional to recognize and establish the diagnosis.

Keywords: Ascher′s syndrome, congential disorders, double lip


How to cite this article:
Bakshi PS, Jindal N, Kaushik A, Leekha S. An appraisal of congenital maxillary double lip with a case report. Saudi J Oral Sci 2015;2:35-8

How to cite this URL:
Bakshi PS, Jindal N, Kaushik A, Leekha S. An appraisal of congenital maxillary double lip with a case report. Saudi J Oral Sci [serial online] 2015 [cited 2020 Sep 21];2:35-8. Available from: http://www.saudijos.org/text.asp?2015/2/1/35/150594


  Introduction Top


Double lip is a rare anomaly characterized by accessory fold of redundant mucous membrane, which is situated proximal to vermilion border and becomes more prominent on smiling. It may be congenital or acquired. This condition affects upper lip more frequently than the lower lip and has no racial predilection. [1],[2],[3],[4] There appears to be male gender predilection of 7:1 for this anomaly. [5] It may occur in isolation or as a part of Ascher's syndrome. [2],[6],[7] Treatment is accomplished surgically and is, usually, indicated for cosmetic and esthetic reasons or for functional impairment if it interferes with speech and mastication. Recurrences after surgery are extremely rare. [6]


  Case Report Top


A 32-year-old male reported to the outpatient department of Government Dental College Rohtak with the chief complaint of large and unsightly upper lip because of which patient felt embarrassed to smile and wanted to get it corrected. Although the anomaly was present since birth, he got conscious about this at the age of 18-20 years. There was no previous history of trauma, infection or surgery on lip. His family and medical history were noncontributory.

Clinical examination revealed extra fold of tissue bilaterally with midline constriction band on the inner aspect of upper lip. The anomaly was clearly visible when patient kept the lips apart [Figure 1] and became more prominent while smiling [Figure 2]. The overlying mucosal tissue appeared intact and smooth with no palpable masses or surface changes. This general blood picture was within normal limits. There were no associated congenital abnormalities like thyroid enlargement or blepharochalasis which refers to inflammation of the eyelid that is characterized by exacerbations and remissions of eyelid edema, resulting in a stretching and subsequent atrophy of the eyelid tissue as seen in Ascher's syndrome. A provisional diagnosis of congenital bilateral maxillary double lip was made, and surgical excision was planned under local anesthesia. Bilateral infraorbital blocks were administered. The accessory labial tissue was marked and excised by transverse elliptical incision from one commissure to another. The primary closure of the wound was carried out using 4-0 silk suture and a light pressure dressing were placed over upper lip for initial 24 h. The patient was reviewed after 1 week for suture removal [Figure 3] and after 3 months. No postoperative problem was there and highly satisfactory aesthetic results achieved [Figure 4] and [Figure 5].
Figure 1: Clinical photograph of maxillary double lip


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Figure 2: Anomaly became more prominent on smiling


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Figure 3: One week postoperative clinical photograph


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Figure 4: Postoperative photograph showing marked aesthetic improvement


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Figure 5: No anomaly visible even on smiling after surgery


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Histopathology of the specimen showed normal labial mucosa with acanthosis. The connective tissue revealed minor mucosal glands and bands of skeletal muscles [Figure 6].
Figure 6: Histopathology showing acanthosis in epithelium, minor mucus glands and skeletal muscles in connective tissue


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Patient was followed up for 2 years, and no surgical complications or recurrences were observed.


  Discussion Top


Double lip is one of the rarest forms of lip anomalies. [7] It is also referred to as macrocheilitis. [4] It, usually, occurs as a fold of hypertrophic tissue on the mucosal side of the lip. It occurs most often bilaterally on upper lip but may be unilateral and can affect both upper and lower lip. [8]

Males are affected more commonly with this anomaly in the ratio of 7:1. [5] The condition can be congenital or acquired. The congenital form is more common and is a hypothesis to arise during 2 or 3 month of gestation. It is during the development of the mucosa that the upper lip consists of two transverse zones, that is, an outer zone, which is smooth and similar to the skin called par glabrosa and the inner zone, which is villosa and similar to oral mucosa termed pars villosa. [9] The persistence of horizontal sulcus between pars glabrosa and pars villosa leads to the formation of double lip. [6],[10]

The acquired form of double lip may be secondary to trauma and oral habits and may develop in association with Ascher's syndrome. [8],[11] The acquired form could be due to "sucking in" of the tissues between the teeth or maloccluding dentures [12] or due to lip sucking habits. [13] Ascher syndrome consists of the triad of blepharochalasis, nontoxic thyroid enlargement and double lip. Ascher first noticed the association of double lip and blepharochalasis with thyroid enlargement. [8],[9],[14]

Double lip can also occur in association with other oral anomalies such as bifid uvula, [9] hemangioma, [12] cleft palate [14] and cheilities glandudaris. [12],[13],[15]

Differential diagnosis of double lip includes hemangioma, lymphangioma, angiodema, cheilitis glandularis, cheilitis granulomatosa mucus retention cyst, mucocele, minor salivary gland benign and malignant tumors, which have predilection for the upper lip for example pleomorphic adenoma, basal cell carcinoma, canalicular adenoma and lobular carcinoma., plasma cell cheilitis and inflammatory fibrous hyperplasia. [1],[2],[12],[15],[16] Congenital double lip can be differentiated from these conditions due to its "cupid bow" appearance or midline constriction. [17] The treatment of congenital double lip is indicated for aesthetic concern as in our case or when excess tissue interferes with mastication or speech or leads to habits as sucking or biting the redundant tissue.

The treatment includes several surgical techniques like W-plasty, [17] electrosurgical excision [18] and triangular excision. [4] Transverse elliptical excision is used in most cases and gives good results. [1],[9],[15] Bilateral infraorbital nerve blocks and mental nerve lock have been advocated by Dingman and Billman [1] to decrease the chance of tissue distortion when administering local anesthesia The surgery involves excision of excess mucosa and submucosa without involving underlying muscular layer. Although recurrence is rare, one case has been reported with recurrence.


  Conclusion Top


Double lip is a rare oral anomaly, and a general dental practitioner is often the first professional to detect and establish the diagnosis of this uncommon condition. [2] The dentist should rule out blepharochalasis and nontoxic thyroid enlargement, which are seen in Ascher's syndrome that also includes the occurrence of double lip. [11] Because of progressive nature or suspected Ascher's syndrome the patient should be advised follow-up for a long time. [19]

 
  References Top

1.
Kenny KF, Hreha JP, Dent CD. Bilateral redundant mucosal tissue of the upper lip. J Am Dent Assoc 1990;120:193-4.  Back to cited text no. 1
    
2.
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. 2
    
3.
Morgan AF, Stewart FW, Stein IJ. Double lip. Report of a case. Oral Surg Oral Med Oral Pathol 1960;13:236-9.  Back to cited text no. 3
    
4.
Lamster IB, Hackensack NJ. Mucosal reduction for correction of a maxillary double lip. Oral Surg 1983;55:457-8.  Back to cited text no. 4
    
5.
Palma MC, Taub DI. Recurrent double lip: Literature review and report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:e20-3.  Back to cited text no. 5
    
6.
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. 6
    
7.
Alkan A, Metin M. Maxillary double lip: Report of two cases. J Oral Sci 2001;43:69-72.  Back to cited text no. 7
    
8.
Barnett ML, Bosshardt LL, Morgan AF. Double lip and double lip with blepharochalasis (Ascher's syndrome). Oral Surg Oral Med Oral Pathol 1972;34:727-33.  Back to cited text no. 8
    
9.
Kara IG, Kara CO. Ascher syndrome. Otolaryngol Head Neck Surg 2001;124:236-7.  Back to cited text no. 9
    
10.
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.  Back to cited text no. 10
    
11.
Gorlin RJ, Pinolborg JJ, Lohen MM. Syndromes of Head and Neck. 2 nd ed. New York: McGraw Hill; 1964. p. 186-9.  Back to cited text no. 11
    
12.
Dhanpal R. Maxillary double lip and cheilitis glandularis: An unusual occurence. J Oral Maxillofac Pathol 2007;11:35-7.  Back to cited text no. 12
    
13.
Coher DM, Green JG, Dickmann SL. Concurrent anomalies: Cheilitis glandularis and double lip. Report of a case. Oral Surg 1988;66:397-9.  Back to cited text no. 13
    
14.
Converse JM. Reconstructive Plastic Surgery. Vol. 3. Philadelphia: Sanunders; 1977. p. 1534-4.  Back to cited text no. 14
    
15.
Rohit BR. Congenital double lip review of literature and a case report. Int J Contemp Dent 2012;3:46-8.  Back to cited text no. 15
    
16.
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. 16
    
17.
Guerrero-Santos J, Altamirano JT. The use of W-plasty for the correction of double lip deformity. Plast Reconstr Surg 1967;39:478-81.  Back to cited text no. 17
    
18.
Peterson A. Electrosurgical correction of maxillary double lip. Dent Dig 1972;78:182-8.  Back to cited text no. 18
    
19.
Daniels JS. Congenital double upper lip: A case report and review of the literature. Saudi Dent J 2010;22:101-6.  Back to cited text no. 19
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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