Abstract
Double lip or macrocheilitis is a rare facial anomaly, mostly congenital in origin. It commonly involves the upper lip than the lower lip. It may occur in isolation or as part of the Ascher’s syndrome. It results due to deposition of excessive areolar tissue and non-inflammatory hyperplasia of labial mucosa gland of pars villosa. It may be acquired as a result of injury to the lips or lip-biting habit. The double lip becomes conspicuous when the lips are retracted during smiling resulting in the characteristic ‘cupid’s bow’ appearance. This disfigurement can pose aesthetic and functional problems and may result in psychological distress. A surgical intervention is must for restoration of functions and to address the aesthetic concerns. The present article reports a case of non-syndromic double upper lip with triple labial frena and its surgical management with laser on one side and with scalpel on the other side.
Keywords: dentistry and oral medicine, mouth
Background
Double lip (labium duplex) is an infrequent developmental lip anomaly that can affect the upper or lower lip but more commonly involves the upper lip.1
Double lip may be either congenital or acquired. In the fetal stage, the lip consists of two transverse zones, an outer cutaneous zone known as pars glabrosa and an inner mucosal zone called pars villosa. Congenital double lip may arise as a result of hypertrophy of pars villosa and due to persistence of an exaggerated horizontal sulcus between both these zones, which is known to disappear between the second and third month of intrauterine life. The acquired double lip may occur secondary to trauma1 2 or as a result of parafunctional habits such as lip suction between diastema or faulty prosthesis.1
There was no gender or race predilection initially, however, Palma and Taub suggested that double lip was more commonly found in males in the ratio of 7:1.3
Clinically, a double lip becomes more apparent when the patient smiles or talks. While smiling, bilateral double lip typically presents as a cupid’s bow appearance due to the retracted lip and the excessive mucosa being placed over the maxillary teeth.4
Treatment of double lip involves intraoral excision of the mucosa and submucosal tissue, without inclusion of the underlying muscle.5
Case presentation
A 25-year-old male patient reported with an unappealing upper lip, lodgement of food between teeth and upper lip, interference in speech and difficulty in brushing. There was no family history of double lip and trauma in relation to the upper lip region. No parafunctional habits were reported. Ascher’s syndrome was ruled out as there was no blepharochalasis and thyroid gland enlargement. On clinical examination, excessive tissue could be seen bulging out on the internal aspect of the upper lip covering cervical third of the maxillary anterior teeth when the patient smiled or talked. At rest, the excessive tissue was not clinically apparent(figure 1).
Figure 1.
(A) Double upper lip, (B) Triple labial frena, (C) Right side cheiloplasty with scalpel, (D) Left side cheiloplasty with laser, (E) Postoperative view showing good cosmetic result.
Investigations
Complete haemogram, HbA1c (Glycosylated Haemoglobin), FBS (Fasting Blood Sugar), HIV (Human Immunodeficiency Virus), HCV (Hepatitis C Virus) and HBsAg (Hepatitis B Surface Antigen), USG (Ultrasonography) neck and thyroid profile.
Differential diagnosis
The differential diagnoses include angioedema, vascular tumours, lymphangioma, cheilitis granulomatosis, mucocele, salivary gland tumours, Miescher syndrome, inflammatory fibrous hyperplasia, sarcoidosis and plasma cell cheilitis.6
Treatment
The patient was planned for surgical correction of the upper lip after oral prophylaxis was done. The patient was painted and draped under all aseptic conditions. Lignocaine with epinephrine (1:200000) was administered to achieve local anaesthesia. Bilateral infraorbital nerve blocks were administered to prevent distortion of the tissue. The excessive tissue including the mucosal and submucosal layers on the left side was excised using laser and on the right side with scalpel, sparing the vermilion border on the inner aspect of the upper lip. After excision of the bulging tissue, closure was done using 5–0 silk suture superficially and 5–0 Vicryl suture in the inner layers to avoid dead spaces on both the left and right side. A moist gauze pack was placed in the vestibule. The patient was prescribed an antibiotic course for a period of 5 days and was recalled after 7 days for suture removal.
Outcome and follow-up
The healing phase was uneventful on both the sides, but wound healing was delayed in case of laser as compared with scalpel. The patient felt less postoperative pain on laser side as opposed to scalpel side. There was complete functional restoration and a pleasing aesthetic outcome after 1 month.
Discussion
Congenital double lip is a non-inflammatory condition of the lip occurring as a result of glandular tissue hyperplasia or due to persistence of the horizontal sulcus between the two developing zones of the lip—pars glabrosa and pars villosa during second to third week of intra-uterine life.7 Although double lip might be present at birth, it becomes obvious only after eruption of teeth.1 Our case presents as congenital double upper lip with triple labial frena.
The double lip may occur in the form of an isolated lesion or as part of the Ascher’s syndrome presenting with blepharochalasis and non-toxic thyroid gland enlargement.8 In our case, it was an isolated lesion.
The deformity is not apparent when the patient is at rest and it becomes appreciable only during talking or smiling.3 In our case too, the typical cupid’s bow appearance of the double lip could only be appreciated when the patient smiled or when the lips were retracted. At rest, the excessive tissue was concealed under the upper lip.
Habitual pulling of the labial mucosa through a midline diastema or between ill-fitting dentures may result in double lip.9 Our patient did not give history of any parafunctional habit such as lip biting or lip suction.
Blunt trauma may cause a prolapse of the labial mucosa thereby producing a unilateral double lip.10 Our patient did not report any history of trauma or prior surgery in relation to the upper lip region.
The treatment of double lip is mainly indicated for aesthetic concern as it may cause psychological distress to the patient. The excessive tissue interferes with mastication, speech and may lead to habits such as sucking or biting the redundant tissue.11 Our patient also sought treatment for the same reasons. The unaesthetic appearance caused a lot of embarrassment especially during social gatherings leading to psychological distress. Apart from the cosmetic aspect, there were functional problems too such as inability to maintain good oral hygiene and interference with speech.
Several surgical techniques have been discussed for the repair of double lip such as W-plasty, electrosurgical excision, triangular excision, elliptical excision and laser.12 In our case, we have used both laser and scalpel for the excision of the hyperplastic tissue of bilateral double upper lip. Each method has its own pros and cons. Laser provided a nearly bloodless field for excision whereas scalpel made the procedure quick. Postoperative healing was good for both the techniques. The surgery yielded good cosmetic results and no recurrence was noted.
Patient's perspective.
I came to this hospital for the treatment of my unattractive upper lip appearance. My major concerns were the unaesthetic appearance which caused a lot of social embarrassment, interference in speech and difficulty in mastication due to accumulation of food debris. I used to feel uncomfortable while brushing teeth and was unable to maintain proper oral hygiene. The surgery was painless, comfortable and rendered excellent results and has boosted my confidence tremendously.
Learning points.
As the congenital double upper lip is associated with an unaesthetic cupid’s bow appearance, patients become conscious of their looks especially during smiling which causes social embarrassment.
There are other functional impairments such as inability to maintain good oral hygiene by not being able to brush teeth properly leading to plaque deposition, calculus, caries and halitosis.
There is difficulty to perform any dental treatment in the upper anterior tooth region as the excessive soft-tissue causes poor visibility and inadequate reflection of flaps during surgical treatment.
In patients undergoing orthodontic treatment, the soft-tissue may also get stuck in the orthodontic brackets and may cause discomfort to the patient.
There is difficulty in fabrication of dental prosthesis and patients may suffer from accidental lip biting occasionally.
Footnotes
Contributors: MG was the operating surgeon. MG handled study design and concept. MG and HV handled the manuscript preparation. MG and YK involved in manuscript editing. MG and AR incharge of manuscript review.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests: None declared.
Provenance and peer review: Not commissioned; externally peer reviewed.
Ethics statements
Patient consent for publication
Obtained.
References
- 1.Martins WD, Westphalen FH, Sandrin R, et al. Congenital maxillary double lip: review of the literature and report of a case. J Can Dent Assoc 2004;70:466–9. [PubMed] [Google Scholar]
- 2.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. 10.1016/j.tripleo.2006.08.027 [DOI] [PubMed] [Google Scholar]
- 3.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. 10.1016/j.tripleo.2008.11.016 [DOI] [PubMed] [Google Scholar]
- 4.Goyal S, Godhi S, Goyal S. Non-Syndromic congenital maxillary double lip: a rare case. J Oral Health Community Dent 2008;2:10–12. 10.5005/johcd-2-1-10 [DOI] [Google Scholar]
- 5.Epker BN, Wolford LM. Reduction cheiloplasty: its role in the correction of dentofacial deformities. J Maxillofac Surg 1977;5:134–41. 10.1016/S0301-0503(77)80091-X [DOI] [PubMed] [Google Scholar]
- 6.Hanemann JAC, Oliveira DT, Gomes MF, et al. Congenital double lip associated to hemangiomas: report of a case. Med Oral 2004;9:156–8. [PubMed] [Google Scholar]
- 7.Aggarwal T, Chawla K, Lamba AK, et al. Congenital double lip: a rare deformity treated surgically. World J Plast Surg 2016;5:303. [PMC free article] [PubMed] [Google Scholar]
- 8.Kalra N, Tyagi R, Khatri A, et al. Double lip-an atypical facial anomaly: two case reports. Int J Clin Pediatr Dent 2018;11:451. 10.5005/jp-journals-10005-1556 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 9.Barnett ML, Bosshardt LL, Morgan AF. Double lip and double lip with blepharochalasis (Ascher’s syndrome). Oral Surgery, Oral Medicine, Oral Pathology 1972;34:727–33. 10.1016/0030-4220(72)90290-3 [DOI] [PubMed] [Google Scholar]
- 10.Oldfield MC. Prolapse of the lip mucosa. Br J Surg 1959;47:58–60. 10.1002/bjs.18004720112 [DOI] [PubMed] [Google Scholar]
- 11.Srivastava A, Chaudhri S, Kumar A, et al. Management of double lip. J Clin Diagn Res 2017;11:ZJ07. 10.7860/JCDR/2017/28816.10492 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 12.Kanneboina LM, Hadge P, Desai H. Smile makeover with double lip correction-case report. International Journal of Oral Care and Research 2015;3:86–8. [Google Scholar]