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Congenital double lip: Review of 5 cases
British Journal of Oral and Maxillofacial Surgery 45 (2007) 68–70
Short communication
Congenital double lip: Review of 5 cases Muhitdin Eski a,∗ , Mustafa Nisanci a , Alper Aktas b , Mustafa Sengezer a a
Department of Plastic and Reconstructive Surgery, Gulhane Military Medical Academy, 06010 Etlik, Ankara, Turkey b Mevki Hospital, Division of Plastic and Reconstructive Surgery, 06130 Diskapı, Ankara, Turkey Received 6 August 2004; accepted 18 April 2005 Available online 8 June 2005
Abstract Congenital double lip is rare and usually involves the upper lip. Apart from a deformity that interferes with speech and mastication, operation may be indicated for cosmetic reasons. We have operated on five patients with double lip deformities for cosmetic reasons. Although a midline constriction band between two mucosal bulges is thought to be a constant feature, four of our five cases did not have a midline constriction. We used an elliptical excision of the mucosal excess in the four patients. The one with central constriction had an elliptical excision on each side, combined with a vertical midline Z-plasty to release the central constriction. Satisfactory aesthetic results were achieved in all patients. © 2005 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Double lip; Mucosal excision; Z-plasty
Introduction Congenital double lip is rare and generally involves the upper lip.1–4 A double vermilion with a transverse furrow between the two borders appears when the orbicularis oris muscle contracts during a smile. The incidence of this anomaly is not known and it may be either isolated or in association with other congenital abnormalities.5 Treatment is by excision of the excess mucosa and submucosal tissue. We present our experiences in the treatment of five young men with double lips.
Case reports Five young men who complained of disfigured upper lips were referred to us for correction. They were all in their 20s. The deformities had been present since birth and became more prominent as they grew. Past medical and family histories were not helpful. The deformities of the upper lips
Fig. 1. The lips look normal even the mouth is open.
were almost identical in all patients, except the one with a central constricting band. No other congenital deformities were noted. The deformities
∗
Corresponding author. Tel.: +90 312 304 5401; fax: + 90 312 304 5412. E-mail address: [email protected] (M. Eski).
The lips looked normal whether the mouth was open or closed (Fig. 1). However, when they smiled a transverse mucosal
0266-4356/$ – see front matter © 2005 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.bjoms.2005.04.017
M. Eski et al. / British Journal of Oral and Maxillofacial Surgery 45 (2007) 68–70
69
bulge appeared behind the vermilion line, producing the appearance of a double-lip (Fig. 2). In one patient the upper lip was thick and there was a central constricting band that divided the buccal portion into two halves. This central band was a continuation of the frenulum over the vermilion of the lip. There was a chronic fissure on the band. When the mouth opened, the buccal portion of the lip became prominent, with a mucosal bulge on both sides of the central constriction band (Fig. 3). Smiling worsened the deformity and caused pain from the stretched fissure. Operation We operated under local anaesthesia (infraorbital block and ring block). The excess buccal mucosa was excised through a transverse elliptical incision in four patients. Only excess labial mucosa was removed, sparing the muscle fibres. Closure was by running 4/0 polyglactin 910 (Vycril) suture. In the man who had a constricting band, the deformity was corrected by two elliptical excisions, one on each half of the lip, and combined with a central Z-plasty to release the constricting band (Fig. 5).
Fig. 4. The upper lip 3 months after the operation.
Fig. 5. Diagram of the Z-plasty with two elliptical excisions.
Results Fig. 2. Double lip deformity appears when the man smiles, with no central constricting band.
Postoperative recovery was uneventful apart from swelling that resolved in about 10 days. Despite meticulous haemostasis, the man with a central constricting band developed a haematoma that subsided spontaneously. His postoperative swelling persisted longer than in the others. All the patients were happy with the cosmetic results. After two years there were no signs of relapse or late complications such as glandular hypertrophy or mucocele.
Discussion
Fig. 3. The upper lip when the mouth is open; note the short central constriction and mucosal bulging on both sides.
Double lip may be either a congenital abnormality or an acquired deformity. The congenital form usually involves the upper lip, but it may also affect the lower lip.6,8 It is usually present at birth but does not become apparent until the permanent teeth erupt.6 Acquired double lip deformity can occur after an injury, in association with Ascher syndrome, or as a result of habitual pulling of the mucosa through a diastema
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M. Eski et al. / British Journal of Oral and Maxillofacial Surgery 45 (2007) 68–70
or between ill-fitting dentures.1,2 The condition is not evident when the upper lip is at rest, but the redundant tissue projects beyond the vermilion border of the lip when the upper lip is tensed as when smiling. The mucosal bulge is composed of hypertrophied submucosal glands.1,6,8 According to Converse et al.6 the anatomy of the orbicularis oris muscle is normal and no muscle fibres are present in the double lip. In the fetus, the mucosa of the lip is divided into two zones: an outer zone (pars glabra) that is smooth and similar to skin, and an inner zone (pars villosa) that is villous and similar to the oral mucosa. A double lip (which is the result of hypertrophy of the pars villosa) is thought to result from an exaggerated horizontal sulcus between the pars glabra and the pars villosa during the development of the lips.8 Various operations to correct a double lip have been described. Guerrero-Santos and Altamirano described the use of a W-plasty,7 and Benmeir et al. also used this technique in 5 patients.3 Simple excision through an elliptical incision was advocated by Reddy and Kotewara.8 In patients with a short central constriction band, the use of two elliptical incisions combined with a vertical Z-plasty result in a pleasing
appearance of the upper lip with a natural-looking tubercle (Fig. 4).
References 1. Alkan A, Metin M. Maxillary double lip: report of two cases. J Oral Sci 2001;43:69–72. 2. 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. 3. Benmeir P, Weinberg A, Neuman, et al. Congenital double lip: report of five cases and a review of the literature. Ann Plast Surg 1992;28:180–2. 4. Cohen DM, Green JG, Diekmann SL. Concurrent anomalies: Cheilitis glandularis and double lip. Oral Surg Oral Med Oral Pathol 1988;66:397–9. 5. Calnan J. Congenital double lip: a report of a case with note on embryology. Br J Plast Surg 1952;5:197–200. 6. Converse JM, Wood-Smith D, Macomber WB, Wang MKH. Reconstructive plastic surgery. 2nd ed. Philadelphia: Saunders; 1977. p. 1543. 7. Guerrero-Santos J, Altamirano JT. The use of W-plasty for the correction of double-lip deformity. Plast Reconstr Surg 1967;39:478–81. 8. Reddy KA, Kotewara A. Congenital double lip: a review of seven cases. Plast Reconstr Surg 1989;84:420–3.
Short communication
Congenital double lip: Review of 5 cases Muhitdin Eski a,∗ , Mustafa Nisanci a , Alper Aktas b , Mustafa Sengezer a a
Department of Plastic and Reconstructive Surgery, Gulhane Military Medical Academy, 06010 Etlik, Ankara, Turkey b Mevki Hospital, Division of Plastic and Reconstructive Surgery, 06130 Diskapı, Ankara, Turkey Received 6 August 2004; accepted 18 April 2005 Available online 8 June 2005
Abstract Congenital double lip is rare and usually involves the upper lip. Apart from a deformity that interferes with speech and mastication, operation may be indicated for cosmetic reasons. We have operated on five patients with double lip deformities for cosmetic reasons. Although a midline constriction band between two mucosal bulges is thought to be a constant feature, four of our five cases did not have a midline constriction. We used an elliptical excision of the mucosal excess in the four patients. The one with central constriction had an elliptical excision on each side, combined with a vertical midline Z-plasty to release the central constriction. Satisfactory aesthetic results were achieved in all patients. © 2005 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Double lip; Mucosal excision; Z-plasty
Introduction Congenital double lip is rare and generally involves the upper lip.1–4 A double vermilion with a transverse furrow between the two borders appears when the orbicularis oris muscle contracts during a smile. The incidence of this anomaly is not known and it may be either isolated or in association with other congenital abnormalities.5 Treatment is by excision of the excess mucosa and submucosal tissue. We present our experiences in the treatment of five young men with double lips.
Case reports Five young men who complained of disfigured upper lips were referred to us for correction. They were all in their 20s. The deformities had been present since birth and became more prominent as they grew. Past medical and family histories were not helpful. The deformities of the upper lips
Fig. 1. The lips look normal even the mouth is open.
were almost identical in all patients, except the one with a central constricting band. No other congenital deformities were noted. The deformities
∗
Corresponding author. Tel.: +90 312 304 5401; fax: + 90 312 304 5412. E-mail address: [email protected] (M. Eski).
The lips looked normal whether the mouth was open or closed (Fig. 1). However, when they smiled a transverse mucosal
0266-4356/$ – see front matter © 2005 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.bjoms.2005.04.017
M. Eski et al. / British Journal of Oral and Maxillofacial Surgery 45 (2007) 68–70
69
bulge appeared behind the vermilion line, producing the appearance of a double-lip (Fig. 2). In one patient the upper lip was thick and there was a central constricting band that divided the buccal portion into two halves. This central band was a continuation of the frenulum over the vermilion of the lip. There was a chronic fissure on the band. When the mouth opened, the buccal portion of the lip became prominent, with a mucosal bulge on both sides of the central constriction band (Fig. 3). Smiling worsened the deformity and caused pain from the stretched fissure. Operation We operated under local anaesthesia (infraorbital block and ring block). The excess buccal mucosa was excised through a transverse elliptical incision in four patients. Only excess labial mucosa was removed, sparing the muscle fibres. Closure was by running 4/0 polyglactin 910 (Vycril) suture. In the man who had a constricting band, the deformity was corrected by two elliptical excisions, one on each half of the lip, and combined with a central Z-plasty to release the constricting band (Fig. 5).
Fig. 4. The upper lip 3 months after the operation.
Fig. 5. Diagram of the Z-plasty with two elliptical excisions.
Results Fig. 2. Double lip deformity appears when the man smiles, with no central constricting band.
Postoperative recovery was uneventful apart from swelling that resolved in about 10 days. Despite meticulous haemostasis, the man with a central constricting band developed a haematoma that subsided spontaneously. His postoperative swelling persisted longer than in the others. All the patients were happy with the cosmetic results. After two years there were no signs of relapse or late complications such as glandular hypertrophy or mucocele.
Discussion
Fig. 3. The upper lip when the mouth is open; note the short central constriction and mucosal bulging on both sides.
Double lip may be either a congenital abnormality or an acquired deformity. The congenital form usually involves the upper lip, but it may also affect the lower lip.6,8 It is usually present at birth but does not become apparent until the permanent teeth erupt.6 Acquired double lip deformity can occur after an injury, in association with Ascher syndrome, or as a result of habitual pulling of the mucosa through a diastema
70
M. Eski et al. / British Journal of Oral and Maxillofacial Surgery 45 (2007) 68–70
or between ill-fitting dentures.1,2 The condition is not evident when the upper lip is at rest, but the redundant tissue projects beyond the vermilion border of the lip when the upper lip is tensed as when smiling. The mucosal bulge is composed of hypertrophied submucosal glands.1,6,8 According to Converse et al.6 the anatomy of the orbicularis oris muscle is normal and no muscle fibres are present in the double lip. In the fetus, the mucosa of the lip is divided into two zones: an outer zone (pars glabra) that is smooth and similar to skin, and an inner zone (pars villosa) that is villous and similar to the oral mucosa. A double lip (which is the result of hypertrophy of the pars villosa) is thought to result from an exaggerated horizontal sulcus between the pars glabra and the pars villosa during the development of the lips.8 Various operations to correct a double lip have been described. Guerrero-Santos and Altamirano described the use of a W-plasty,7 and Benmeir et al. also used this technique in 5 patients.3 Simple excision through an elliptical incision was advocated by Reddy and Kotewara.8 In patients with a short central constriction band, the use of two elliptical incisions combined with a vertical Z-plasty result in a pleasing
appearance of the upper lip with a natural-looking tubercle (Fig. 4).
References 1. Alkan A, Metin M. Maxillary double lip: report of two cases. J Oral Sci 2001;43:69–72. 2. 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. 3. Benmeir P, Weinberg A, Neuman, et al. Congenital double lip: report of five cases and a review of the literature. Ann Plast Surg 1992;28:180–2. 4. Cohen DM, Green JG, Diekmann SL. Concurrent anomalies: Cheilitis glandularis and double lip. Oral Surg Oral Med Oral Pathol 1988;66:397–9. 5. Calnan J. Congenital double lip: a report of a case with note on embryology. Br J Plast Surg 1952;5:197–200. 6. Converse JM, Wood-Smith D, Macomber WB, Wang MKH. Reconstructive plastic surgery. 2nd ed. Philadelphia: Saunders; 1977. p. 1543. 7. Guerrero-Santos J, Altamirano JT. The use of W-plasty for the correction of double-lip deformity. Plast Reconstr Surg 1967;39:478–81. 8. Reddy KA, Kotewara A. Congenital double lip: a review of seven cases. Plast Reconstr Surg 1989;84:420–3.