IDEAS AND INNOVATIONS

Bilateral Cleft Lip and Whistling Deformity: The X Flap Procedure for Its Correction
Antonio G. A. Assuncao, M.D. Ldia M. Ferreira, M.D., Ph.D. Ricardo L. Mondelli, M.D.
Sao Paulo, Brazil

he whistling lip deformity, a lack of tissue at the central tubercle of the cleft lip, is seen more often in bilateral cleft lips, frequently as a postoperative sequela. Its causes include (1) failure to fill the central tubercle with lateral vermilion flaps during primary lip repair and/or severe scar contraction as a result of crossing scars at the middle line of the central tubercle1 6; (2) use of a technique that does not primarily fill the central tubercle7; (3) diastasis of the orbicularis oris muscle at the base of the nose, which pulls up the central tubercle8,9; and (4) a combination of one or more of items 1 through 3. Many techniques have been described to correct the deformity in question; some10 12 rotated a lower lip flap to the upper lip to substitute for or to add a prolabium to it, thus correcting a lack of tissue at the central tubercle. Others suggested correction of the orbicularis oris muscle and elevation of the prolabial skin8 or added a revision of the lip scars to reduce a wide prolabium and insert two vertical scar tissue flaps to fill the central tubercle.9 Bilateral, medially based orbicularis oris muscle and vermilion,1316 or deepithelialized submucosal V-Y and triangular flaps,17 turned centrally, have also been used to fill the prolabium in cases where there was ample lateral lip mucosa and a complete orbicularis oris muscle continuity.18 Free composite submucosal-muscle flaps, from the inner substance on each side of the lip,19 from the palatal mucosa,20 or free dermis fat grafts21 have also been used to fill the central upper lip defect. The posteroanterior V-Y adFrom the Hospital of Rehabilitation of Craniofacial Malformations, University of Sao Paulo, Bauru, and Department of Surgery, Universidade Federal de Sao Paulo. Received for publication December 15, 2004; revised April 27, 2005. Copyright 2006 by the American Society of Plastic Surgeons DOI: 10.1097/01.prs.0000218328.24577.81

vancement was applied alone22 (for small lip defects) or together with bilateral lip advancement at the midline to create a new buccal sulcus.23 The Spina technique,2 which uses two lateral orbicularis oris muscle flaps sutured in a horizontal position parallel to each other to fill the central tubercle, is applied primarily for bilateral cleft lip repair. This technique, still used by many Brazilian plastic surgeons, sutures the orbicularis oris muscle to each side of the prolabium, leaving a diastasis of this muscle. In this article, we present a modification of the Spina technique2,24 to be used mainly for repairing whistling deformities as a secondary procedure and for correcting the diastasis of the orbicularis oris muscle and irregularities at the vermilion border through the same surgical incision in bilateral cleft lip patients.

PATIENTS AND METHODS

Nine patientssix male patients and three female patientsranging in age from 10 to 35 years were admitted to our hospital with bilateral cleft lips that had been operated on, with evident whistling deformity and various degrees of diastasis of the orbicularis oris muscle. All of them were primarily operated on in infancy or early childhood using the Spina technique.2,24 Six patients had undergone previous attempts to correct the whistling deformity elsewhere that failed partially or completely to solve the problem. They were then submitted to a secondary operation, using what we call the X flap procedure in an attempt to correct the whistling deformity. The operations were carried out during the period 1990 to 1993 by one surgeon (A.G.A.A.), by whom the results were assessed at least 1 year after the operation. Surgical Technique Under general anesthesia, vertical coneshaped flaps are drawn over both previous bilateral lip scars, with their tips pointing toward the

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Fig. 1. Diagrammatic representation of the technique used. (Above, left) Two vertical cone-shaped aps are drawn over both previous lip scars, with their tips pointing toward the base of the nose.(Above, center) The depth of incision is limited to the inner lip mucosa, and the aps are left laterally pediculate on each side. (Above, right and Center, left) A tunnel is dissected in both lateral lips and at the central tubercle. (Center, center) This drawing depicts the positioning of the stitches to pull down and lateralize the aps. (Center, right) The aps are overlapped at the central tubercle and xed on each side with a bolster suture. (Below) A vertical V-Y advancement may be necessary in the inner aspect of the lip to accommodate the X aps. Attention must be paid to avoid meeting the vermilion incision.

base of the nose (Fig. 1, above, left). The width and length of the flaps can vary according to the amount of tissue necessary to fill the whistling defect, subjectively calculated by the surgeon at

the time of the operation. Care is taken to remove the previous scars. The depth of the incision is limited to the inner lip mucosa, without opening it. The flaps,

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Fig. 2. (Above, left) A bilateral cleft patient showing whistling deformity. (Above, right) The aps elevated from both donor sites. (Center, left) The aps are crossed and overlapped at the midline of the central tubercle, held by a 4-0 nylon suture. (Center, right) The aps are xed on each side of the lip by a bolster suture and the skin closed by a 6-0 nylon suture. Note the 4-0 catgut suture in the vermilion midline, resulting from the V-Y ap advancement from the inner aspect of the lip. (Below) One-year postoperative result.

consisting of fibers of orbicularis oris muscle and fibrofat tissue from previous bilateral scars, are left laterally pediculate on each side (Fig. 1, above, center). A tunnel is dissected in both lateral lips ( 0.5 cm) and at the central tubercle (Fig. 1, above, right and center, left) to accommodate the once deepithelialized flaps, which are crossed at the midline and fixed on each contralateral side as an X (fill-

ing up the median defect), by a bolster suture using 4-0 mononylon (Figs. 1, center, center and right, and 2, center, left and right). The stitches are then removed on the fifth postoperative day. The donor site is closed by means of an interrupted 4-0 mononylon suture, and at this time any existing diastasis of the orbicularis oris muscle is corrected. The skin is closed by an interrupted 6-0 nylon suture. The dog-ear that usually results at

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Fig. 3. Full face before (left) and after (right) the operation.

the vermilion is excised as required, and any irregularity at the vermilion border is adjusted as required. In some cases, the vermilion at the central tubercle is too tight to accommodate the X flaps. Scar contraction crossing the midline of the central tubercle is mostly responsible for this condition. To solve this problem, a vertical V-Y19 flap advancement in the inner aspect of the lip can be added to the X flap procedure. Attention must be paid to avoid the legs of the V incision meeting the prolongation of the vermilion incision in the correction of dog-ears to avoid new scar contraction (Figs. 1, below and 2, center, right). The patients were assessed by visual inspection at a follow-up visit within 1 to 2 years. This inspection gives consideration to the filling of the central tubercle, which should be sufficient to allow the patient to close the mouth normally and the correct positioning of the orbicularis oris muscle.

one case, it was attributed to the recurrence of the diastasis of the orbicularis oris muscle that had undergone reoperation, only to reposition the muscle back in place; in the other case, there was an inadequate amount of tissue to fill the central defect. The X flap procedure was repeated successfully.

DISCUSSION
Many techniques described to correct whistling deformity create too many new scars at the vermilion, which can lead to an unpleasant appearance of the lip, and do not correct possible diastasis of the orbicularis oris muscle or irregularities at the vermilion border using the same surgical incision.9,1317,23,25 Rodgers and Mulliken18 used an accomplished technique to correct the whistling deformity, effectively hiding the scars. Nevertheless, it seems that its use is limited to small defects and is only applied to those cases where there is ample lateral lip mucosa availability and a complete orbicularis oris muscle continuity. The technique does not allow for any repositioning of the orbicularis oris muscle that may be necessary or for the correction of any irregularity at the vermilion border through the same incision used to correct the central tubercle defect. The techniques that used submucosal muscle grafting19 and grafting of the palatal mucosa20 to fill the central tubercle seem also to have their use

RESULTS
All patients but two showed satisfactory results (Figs. 2, below ; 3, right; and 4, below), with the whistling deformity and the diastasis of the orbicularis oris muscle satisfactorily corrected. All of the irregularities (100 percent) at the vermilion border were corrected. In two patients, the whistling deformity had recurred when checked at the 1-year follow-up. In

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more tissue to the flaps by giving direct access to the orbicularis oris muscle and thus facilitate repair of this structure, which is often a major component of the deformity where the correction of bigger defects is sought. The Spina technique2,24 is very ingenious in filling the central tubercle in most cases, but it fails with regard to putting together the orbicularis oris muscle, leaving a diastasis under the prolabium. In overlapping the flaps at the midline, it seems that the X flap procedure increases the projection of the central tubercle, making us suppose that it can be used to correct all sizes of whistling deformity. Moreover, it has the advantage of correcting any preexisting orbicularis oris muscle diastasis by using the same surgical approach; thus, no further scarring is added to the lip. The lack of accuracy in estimating the exact amount of tissue missing at the central tubercle is the main difficulty found in using the X flap technique. Nevertheless, this can be more easily overcome by an experienced cleft surgeon. Another possible reason for the recurrence of the defect is that, as the flaps are long and are not axial flaps, some parts of them can behave as grafts and suffer resorption. This was possibly the reason for the one failure of the two where the deformity recurred. In the other one, the recurrence of the diastasis of the orbicularis oris muscle was evident.

Fig. 4. Clinical examples. (Above) Bilateral cleft lip patient with a whistling deformity and irregularity of the vermilion border at the right side. (Right) Patient at 1-year follow-up, showing that the whistling deformity and irregularity of the right vermilion border have now been corrected.

CONCLUSIONS
limited to small defects, and the failure of the graft to take has to be considered as a disadvantage of this procedure. The raw area at the inner aspect of the lip, left to heal spontaneously,26 carries the risk of scar retraction and may induce a recurrence of the defect. The Aiache technique23 follows the same principles as the Barsky technique27 for primary repair of bilateral cleft lips and may reproduce the same complications as the Barsky technique,27 namely, an upper lip that is too long and too tight.28 The diastasis of the orbicularis oris muscle can create a pseudo-whistling deformity. The separation of this muscle at the base of the nose pulls up the prolabium vermilion.23 The restoration of the continuity of the orbicularis oris muscle, which has usually not been repaired during the primary operation, pulls down the prolabium, automatically correcting the central deformity.8,9 Kai and Ohishi9 added two vertical scar flaps, but they did not include orbicularis oris muscle fibers in their flaps; this differs from the X flaps in that it may add The X flap procedure was demonstrated to be effective for the correction of whistling deformities in patients primarily operated on using the Spina technique, presenting the advantage of correcting, at the same surgical procedure, coexisting diastasis of the orbicularis oris muscle and irregularities at the vermilion border in bilateral cleft lip patients without adding new scars to the preexisting ones. The whistling lip deformity, a lack of tissue at the central tubercle of the cleft lip, is more often seen in bilateral cleft lips, as a postoperative sequela. A modification of the Spina technique2,24 (for primarily bilateral cleft lip repair) is proposed here for the correction of the deformity in secondary bilateral cleft lips. It consists of two flaps composed of fibers of orbicularis oris muscle and fibrofat tissue from the scars of the upper lip bilaterally, which are overlapped and fixed in an X shape at the central tubercle. It is called the X flap procedure. This procedure has the advantage, apart from correcting the whistling deformity, of also correct-

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ing preexisting (1) orbicularis oris muscle diastasis and (2) irregularities at the vermilion border, using the same surgical approach without adding new scars to the lip. The patients (n 9) operated on between 1990 and 1993 were assessed by visual observation by one of the authors (A.G.A.A.) at the 1-year follow-up. The observations checked whether the central tubercle remained filled, allowing the patient to close his or her mouth completely; the continuity of the orbicularis oris muscle; and the regularity of the vermilion border. All patients but two had the whistling deformity and the diastasis of the orbicularis oris muscle satisfactorily corrected. The X flap procedure was demonstrated to be effective for the correction of whistling deformities, presenting the advantage of correcting coexisting diastasis of the orbicularis oris muscle and irregularities at the vermilion border in bilateral cleft lip patients without adding scars to the preexisting ones.
Antonio G. A. Assuncao, M.D. Rua Sao Goncalo, 673 apt. 114 Bauru, Sao Paulo 17012-170, Brazil dr.antonioassuncao@uol.com.br
9. Kai, S., and Ohishi, M. Secondary correction of the cleft lip and nose deformity: A new technique for revision of whistling deformity. Cleft Palate J. 22: 290, 1985. 10. Abbe, R. A new plastic operation for the relief of deformity due to double hare lip. Plast. Reconstr. Surg. 42: 481, 1968. 11. Bagatin, M., and Most, S. P. The Abbe flap in secondary cleft lip repair. Arch. Facial Plast. Surg. 4: 194, 2002. 12. Wagner, J. D., and Newman, M. H. Bipedicle axial cross-lip flap for correction of major vermilion deficiency after cleft lip repair. Cleft Palate Craniofac. J. 31: 148, 1994. 13. Kapetansky, D. I. Double pendulum flaps for whistling deformities in bilateral cleft lips. Plast. Reconstr. Surg. 47: 321, 1971. 14. Arons, M. S. Another method for secondary correction of whistling deformities in double cleft lips. Plast. Reconstr. Surg. 47: 389, 1971. 15. Juri, J., Juri, C., and Antuero, J. A modification of the Kapetansky technique for repair of whistling deformities of the upper lip. Plast. Reconstr. Surg. 57: 70, 1976. 16. Yoshimura, Y., Nakajima, T., and Yoneda, K. Propeller flap for reconstruction of the tubercle of the upper lip. Br. J. Plast. Surg. 44: 113, 1991. 17. Goumain, A. J., and Guimberteau, J. C. A procedure for correcting the whistling deformity. Ann. Chir. Plast. 23: 108, 1978. 18. Rodgers, C. M., and Mulliken, J. B. Deepithelialized of mucosal-submucosal flaps to correct the whistling lip deformity. Cleft Palate J. 26: 136, 1989. 19. Chong, J. K., and Winslow, R. B. Simple technique for correction of whistling deformity in repaired cleft lips. Plast. Reconstr. Surg. 48: 84, 1971. 20. Vecchione, T. R. Correction of whistling tip deformity using a palatal mucosa graft. Plast. Reconstr. Surg. 69: 344, 1982. 21. Patel, I. A., and Hall, P. N. Free dermis-fat graft to correct the whistle deformity in patients with cleft lip. Br. J. Plast. Surg. 57: 160, 2004. 22. Millard, D. R., Jr. Cleft Craft: The Evolution of its Surgery. Bilateral and Rare Deformities. Boston: Little, Brown, 1977. 23. Aiache, A. E. Whistling deformities following bilateral cleft lip repairs: a method of correction. Br. J. Plast. Surg. 30: 123, 1977. 24. Spina, V., Kamakura, L., and Lapa, F. Surgical management of bilateral cleft lip. Ann. Plast. Surg. 1: 497, 1978. 25. Robinson, D. W., Ketchum, L. D., and Masters, F. W. Double V-Y procedure for whistling deformity in repaired cleft lips. Plast. Reconstr. Surg. 46: 241, 1970. 26. Johnson, H. A. A simple method for the repair of minor postoperative cleft lip whistling deformity. Br. J. Plast. Surg. 25: 152, 1972. 27. Barsky, A. J. Principles and Practices of Plastic Surgery. Baltimore: Williams & Wilkins, 1950. 28. Cronin, T. D., Cronin, E. D., Roper, P., Jr., Millard, D. R., and McComb, H. Bilateral clefts. In J. G. McCarthy, (Ed.), Plastic Surgery. Philadelphia: Saunders, 1990.

ACKNOWLEDGMENTS

The authors thank Ana A. G. Grigolli and Sandra P. B. Pinheiro for their kind and competent assistance with the manuscript.
REFERENCES
1. Veau, V. Division Palatine: Anatomie, Chirurgie, Phonetique. Paris: Masson et Cie, 1931. 2. Spina, V. Cirurgia do labio leporino bilateral: novo conceito. Rev. Paul. Med. 65: 248, 1964. 3. Manchester, W. M. The repair of bilateral cleft lip and plate. Br. J. Surg. 52: 879, 1965. 4. Millard, D. R., Jr. Closure of bilateral cleft lip and elongation of columella by two operations in infancy. Plast. Reconstr. Surg. 47: 324, 1971. 5. Mulliken, J. B. Principles and techniques of bilateral cleft lip repair. Plast. Reconstr. Surg. 75: 477, 1985. 6. Noordhoff, M. S. Bilateral cleft lip reconstruction. Plast. Reconstr. Surg. 78: 45, 1986. 7. Skoog, T. The management of the bilateral cleft of the primary palate (lip and alveolus): I. General considerations and soft tissue repair. Plast. Reconstr. Surg. 35: 34, 1965. 8. Meijer, R. Secondary repair of the bilateral cleft lip deformity. Cleft Palate J. 21: 86, 1984.

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