Journal of Indian Academy of Oral Medicine and Radiology

CASE REPORT
Year
: 2015  |  Volume : 27  |  Issue : 2  |  Page : 314--317

Maxillary double lip: A case series with review of literature


Vela D Desai1, Sudakshina Das1, Sunil MV Kumar2, Harsha Sadhwani1,  
1 Department of Oral Medicine and Radiology, Jaipur Dental College, Jaipur, Rajasthan, India
2 Department of Prosthodontics, Jaipur Dental College, Jaipur, Rajasthan, India

Correspondence Address:
Vela D Desai
B-4O6, Trimurthy Apartment, Opposite BSNL Telecom Colony, Malviya Nagar, Jaipur - 302 017, 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.



How to cite this article:
Desai VD, Das S, Kumar SM, Sadhwani H. Maxillary double lip: A case series with review of literature.J Indian Acad Oral Med Radiol 2015;27:314-317


How to cite this URL:
Desai VD, Das S, Kumar SM, Sadhwani H. Maxillary double lip: A case series with review of literature. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2022 Nov 26 ];27:314-317
Available from: http://www.jiaomr.in/text.asp?2015/27/2/314/170173


Full Text

 Introduction



Double 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],[6],[7] The authors present a case series of maxillary double lip in this article.

 Case Reports



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 or functional complaints, no surgical correction was instituted. He was treated for generalized periodontitis, counselled, and followed up.{Figure 1}{Figure 2}

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].{Figure 3}{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.{Figure 5}

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.{Figure 6}

 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 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 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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1Kenny KF, Hreha JP, Dent CD. Bilateral redundant mucosal tissue of the upper lip. J Am Dent Assoc 1990;120;193-4.
2Peterson A. Electrosurgical correction of maxillary double lip. Den Dig 1972;78:182-8.
3Calnan J. Congenital double lip: Record of a case with a note on the embryology. Br J Plast Surg 1952;5:197-202.
4English GM. Embryology and anomalies of the mouth and throat. Chapter 5. In: English GM, editor. Otolaryngology. 18 th ed. Philadelphia: JB Lippincott Company; 1988. p. 1-31.
5Palma 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.
6Hanemann JA, Oliveira DT, Gomes MF, dos Anjos MJ, Sant'ana E. Congenital double lip associated to hemangiomas: Report of a case. Med Oral 2004;9:155-8.
7Cohen DM, Green JG, Diekmann SL. Concurrent anomalies: Cheilitis glandularis and double lip. Report of a case. Oral Surg Oral Med Oral Pathol 1988;66:397-9.
8Rintala AE. Congenital double lip and Ascher syndrome: II. Relationship to the lower lip sinus syndrome. Br J Plast Surg 1981;34:31-4.
9Lamster IB. Mucosal reduction for correction of a maxillary double lip. Report of a case. Oral Surg Oral Med Oral Pathol 1983;55:457-8.
10Martins WD, Westphalen FH, Sandrin R, Campagnoli E. Congenital maxillary double upper lip: Review of the literature and report of a case. J Can Dent Assoc 2004;70:466-8.
11Parmar RC, Muranjan MN. A newly recognized syndrome with double upper and lower lip, hyperthelorism, eyelid ptosis, blepharophimosis, and third finger clinodactyly. Am J Med Genet A 2004;124A:200-1.
12Converse JM. Reconstructive Plastic Surgery. Vol. 3. Philadelphia: Saunders; 1977. p. 1543-4.
13Gomez-Duaso AJ, Seoane J, Vazquez-Garcia J, Arjona C. Ascher syndrome: Report of two cases. J Oral Maxillofac Surg 1997;55:88-90.
14Oeak A, Cakur B. Double lip: A case report. J Dent App 2015;2:162-3.
15Gadre PK, Agarwal R, Kumar S, Singh D, Chaudhary M. Maxillary double lip - A case report and review literature. Int J Res Health Sci 2014;2:871-4.