IDEAS AND INNOVATIONS

Bilateral Cleft Lip and Whistling Deformity:The X Flap Procedure for Its CorrectionAntonio G. A. Assuncao, M.D.Lıdia M. Ferreira, M.D., Ph.D.

Ricardo L. Mondelli, M.D.Sao Paulo, Brazil

The whistling lip deformity, a lack of tissueat the central tubercle of the cleft lip, isseen more often in bilateral cleft lips,

frequently as a postoperative sequela. Itscauses include (1) failure to fill the centraltubercle with lateral vermilion flaps duringprimary lip repair and/or severe scar contrac-tion as a result of crossing scars at the middleline of the central tubercle1–6; (2) use of a tech-nique that does not primarily fill the centraltubercle7; (3) diastasis of the orbicularis oris mus-cle at the base of the nose, which pulls up thecentral tubercle8,9; and (4) a combination of oneor more of items 1 through 3.

Many techniques have been described tocorrect the deformity in question; some10 –12

rotated a lower lip flap to the upper lip tosubstitute for or to add a prolabium to it, thuscorrecting a lack of tissue at the central tuber-cle. Others suggested correction of the orbic-ularis oris muscle and elevation of the prola-bial skin8 or added a revision of the lip scars toreduce a wide prolabium and insert two verti-cal scar tissue flaps to fill the central tubercle.9

Bilateral, medially based orbicularis oris mus-cle and vermilion,13–16 or deepithelialized sub-mucosal V-Y and triangular flaps,17 turned cen-trally, have also been used to fill the prolabiumin cases where there was ample lateral lip mu-cosa and a complete orbicularis oris musclecontinuity.18

Free composite submucosal-muscle flaps, fromthe inner substance on each side of the lip,19

from the palatal mucosa,20 or free dermis fatgrafts21 have also been used to fill the centralupper lip defect. The posteroanterior V-Y ad-

vancement was applied alone22 (for small lipdefects) or together with bilateral lip advance-ment at the midline to create a new buccalsulcus.23

The Spina technique,2 which uses two lateralorbicularis oris muscle flaps sutured in a hori-zontal position parallel to each other to fill thecentral tubercle, is applied primarily for bilateralcleft lip repair. This technique, still used bymany Brazilian plastic surgeons, sutures the or-bicularis oris muscle to each side of the pro-labium, leaving a diastasis of this muscle. In thisarticle, we present a modification of the Spinatechnique2,24 to be used mainly for repairingwhistling deformities as a secondary procedureand for correcting the diastasis of the orbicularisoris muscle and irregularities at the vermilionborder through the same surgical incision inbilateral cleft lip patients.

PATIENTS AND METHODSNine patients—six male patients and three

female patients—ranging in age from 10 to 35years were admitted to our hospital with bilat-eral cleft lips that had been operated on, withevident whistling deformity and various degreesof diastasis of the orbicularis oris muscle. All ofthem were primarily operated on in infancy orearly childhood using the Spina technique.2,24

Six patients had undergone previous attempts tocorrect the whistling deformity elsewhere thatfailed partially or completely to solve the prob-lem. They were then submitted to a secondaryoperation, using what we call the X flap proce-dure in an attempt to correct the whistling de-formity. The operations were carried out duringthe period 1990 to 1993 by one surgeon(A.G.A.A.), by whom the results were assessed atleast 1 year after the operation.

Surgical TechniqueUnder general anesthesia, vertical cone-

shaped flaps are drawn over both previous bilat-eral lip scars, with their tips pointing toward the

From the Hospital of Rehabilitation of Craniofacial Malfor-mations, University of Sao Paulo, Bauru, and Departmentof Surgery, Universidade Federal de Sao Paulo.Received for publication December 15, 2004; revised April27, 2005.Copyright ©2006 by the American Society of Plastic Surgeons

DOI: 10.1097/01.prs.0000218328.24577.81

www.plasreconsurg.org1986

base of the nose (Fig. 1, above, left). The width andlength of the flaps can vary according to theamount of tissue necessary to fill the whistlingdefect, subjectively calculated by the surgeon at

the time of the operation. Care is taken to removethe previous scars.

The depth of the incision is limited to theinner lip mucosa, without opening it. The flaps,

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

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consisting of fibers of orbicularis oris muscle andfibrofat tissue from previous bilateral scars, are leftlaterally pediculate on each side (Fig. 1, above,center).

A tunnel is dissected in both lateral lips (�0.5cm) and at the central tubercle (Fig. 1, above, rightand center, left) to accommodate the once deepi-thelialized flaps, which are crossed at the midlineand fixed on each contralateral side as an X (fill-

ing up the median defect), by a bolster sutureusing 4-0 mononylon (Figs. 1, center, center andright, and 2, center, left and right). The stitches arethen removed on the fifth postoperative day.

The donor site is closed by means of an inter-rupted 4-0 mononylon suture, and at this time anyexisting diastasis of the orbicularis oris muscle iscorrected. The skin is closed by an interrupted 6-0nylon suture. The dog-ear that usually results at

Fig. 2. (Above, left) A bilateral cleft patient showing whistling deformity. (Above, right) The flaps elevated from both donor sites.(Center, left) The flaps are crossed and overlapped at the midline of the central tubercle, held by a 4-0 nylon suture. (Center, right) Theflaps are fixed 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 thevermilion midline, resulting from the V-Y flap advancement from the inner aspect of the lip. (Below) One-year postoperative result.

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the vermilion is excised as required, and any ir-regularity at the vermilion border is adjusted asrequired.

In some cases, the vermilion at the centraltubercle is too tight to accommodate the X flaps.Scar contraction crossing the midline of the cen-tral tubercle is mostly responsible for this condi-tion. To solve this problem, a vertical V-Y19 flapadvancement in the inner aspect of the lip can beadded to the X flap procedure. Attention must bepaid to avoid the legs of the V incision meeting theprolongation of the vermilion incision in the cor-rection of dog-ears to avoid new scar contraction(Figs. 1, below and 2, center, right).

The patients were assessed by visual inspectionat a follow-up visit within 1 to 2 years. This inspec-tion gives consideration to the filling of the centraltubercle, which should be sufficient to allow thepatient to close the mouth normally and the cor-rect positioning of the orbicularis oris muscle.

RESULTSAll patients but two showed satisfactory results

(Figs. 2, below ; 3, right; and 4, below), with thewhistling deformity and the diastasis of the orbic-ularis oris muscle satisfactorily corrected. All ofthe irregularities (100 percent) at the vermilionborder were corrected.

In two patients, the whistling deformity hadrecurred when checked at the 1-year follow-up. In

one case, it was attributed to the recurrence of thediastasis of the orbicularis oris muscle that hadundergone reoperation, only to reposition themuscle back in place; in the other case, there wasan inadequate amount of tissue to fill the centraldefect. The X flap procedure was repeated suc-cessfully.

DISCUSSIONMany techniques described to correct whis-

tling deformity create too many new scars at thevermilion, which can lead to an unpleasant ap-pearance of the lip, and do not correct possiblediastasis of the orbicularis oris muscle or irregu-larities at the vermilion border using the samesurgical incision.9,13–17,23,25 Rodgers and Mulliken18

used an accomplished technique to correct thewhistling deformity, effectively hiding the scars.Nevertheless, it seems that its use is limited to smalldefects and is only applied to those cases wherethere is ample lateral lip mucosa availability and acomplete orbicularis oris muscle continuity. Thetechnique does not allow for any repositioning ofthe orbicularis oris muscle that may be necessaryor for the correction of any irregularity at thevermilion border through the same incision usedto correct the central tubercle defect.

The techniques that used submucosal musclegrafting19 and grafting of the palatal mucosa20 tofill the central tubercle seem also to have their use

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

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limited to small defects, and the failure of the graftto take has to be considered as a disadvantage ofthis procedure. The raw area at the inner aspectof the lip, left to heal spontaneously,26 carries therisk of scar retraction and may induce a recur-rence of the defect.

The Aiache technique23 follows the same prin-ciples as the Barsky technique27 for primary repairof bilateral cleft lips and may reproduce the samecomplications as the Barsky technique,27 namely,an upper lip that is too long and too tight.28

The diastasis of the orbicularis oris muscle cancreate a pseudo-whistling deformity. The separa-tion of this muscle at the base of the nose pulls upthe prolabium vermilion.23 The restoration of thecontinuity of the orbicularis oris muscle, which hasusually not been repaired during the primary op-eration, pulls down the prolabium, automaticallycorrecting the central deformity.8,9 Kai andOhishi9 added two vertical scar flaps, but they didnot include orbicularis oris muscle fibers in theirflaps; this differs from the X flaps in that it may add

more tissue to the flaps by giving direct access tothe orbicularis oris muscle and thus facilitate re-pair of this structure, which is often a major com-ponent of the deformity where the correction ofbigger defects is sought. The Spina technique2,24 isvery ingenious in filling the central tubercle inmost cases, but it fails with regard to putting to-gether the orbicularis oris muscle, leaving a dias-tasis under the prolabium.

In overlapping the flaps at the midline, itseems that the X flap procedure increases theprojection of the central tubercle, making us sup-pose that it can be used to correct all sizes ofwhistling deformity. Moreover, it has the advan-tage of correcting any preexisting orbicularis orismuscle diastasis by using the same surgical ap-proach; thus, no further scarring is added to thelip.

The lack of accuracy in estimating the exactamount of tissue missing at the central tubercle isthe main difficulty found in using the X flap tech-nique. Nevertheless, this can be more easily over-come by an experienced cleft surgeon. Anotherpossible reason for the recurrence of the defect isthat, as the flaps are long and are not axial flaps,some parts of them can behave as grafts and sufferresorption. This was possibly the reason for theone failure of the two where the deformity re-curred. In the other one, the recurrence of thediastasis of the orbicularis oris muscle was evident.

CONCLUSIONSThe X flap procedure was demonstrated to be

effective for the correction of whistling deformi-ties in patients primarily operated on using theSpina technique, presenting the advantage of cor-recting, at the same surgical procedure, coexistingdiastasis of the orbicularis oris muscle and irreg-ularities at the vermilion border in bilateral cleftlip patients without adding new scars to the pre-existing ones.

The whistling lip deformity, a lack of tissueat the central tubercle of the cleft lip, is moreoften seen in bilateral cleft lips, as a postoper-ative sequela. A modification of the Spinatechnique2,24 (for primarily bilateral cleft lip re-pair) is proposed here for the correction of thedeformity in secondary bilateral cleft lips. It con-sists of two flaps composed of fibers of orbicu-laris oris muscle and fibrofat tissue from thescars of the upper lip bilaterally, which are over-lapped and fixed in an X shape at the centraltubercle. It is called the X flap procedure.

This procedure has the advantage, apart fromcorrecting the whistling deformity, of also correct-

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

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ing preexisting (1) orbicularis oris muscle diastasisand (2) irregularities at the vermilion border, us-ing the same surgical approach without addingnew scars to the lip. The patients (n � 9) operatedon between 1990 and 1993 were assessed by visualobservation by one of the authors (A.G.A.A.) atthe 1-year follow-up. The observations checkedwhether the central tubercle remained filled, al-lowing the patient to close his or her mouth com-pletely; the continuity of the orbicularis oris mus-cle; and the regularity of the vermilion border.

All patients but two had the whistling defor-mity and the diastasis of the orbicularis oris musclesatisfactorily corrected. The X flap procedure wasdemonstrated to be effective for the correction ofwhistling deformities, presenting the advantage ofcorrecting coexisting diastasis of the orbicularisoris muscle and irregularities at the vermilion bor-der in bilateral cleft lip patients without addingscars to the preexisting ones.

Antonio G. A. Assuncao, M.D.Rua Sao Goncalo, 673 apt. 114

Bauru, Sao Paulo 17012-170, Brazildr.antonioassuncao@uol.com.br

ACKNOWLEDGMENTSThe authors thank Ana A. G. Grigolli and Sandra

P. B. Pinheiro for their kind and competent assistancewith the manuscript.

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Paris: Masson et Cie, 1931.2. Spina, V. Cirurgia do labio leporino bilateral: novo conceito.

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Br. J. Surg. 52: 879, 1965.4. Millard, D. R., Jr. Closure of bilateral cleft lip and elongation

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9. Kai, S., and Ohishi, M. Secondary correction of the cleft lipand nose deformity: A new technique for revision of whistlingdeformity. Cleft Palate J. 22: 290, 1985.

10. Abbe, R. A new plastic operation for the relief of deformitydue to double hare lip. Plast. Reconstr. Surg. 42: 481, 1968.

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12. Wagner, J. D., and Newman, M. H. Bipedicle axial cross-lipflap for correction of major vermilion deficiency after cleftlip repair. Cleft Palate Craniofac. J. 31: 148, 1994.

13. Kapetansky, D. I. Double pendulum flaps for whistling de-formities in bilateral cleft lips. Plast. Reconstr. Surg. 47: 321,1971.

14. Arons, M. S. Another method for secondary correction ofwhistling deformities in double cleft lips. Plast. Reconstr. Surg.47: 389, 1971.

15. Juri, J., Juri, C., and Antuero, J. A modification of the Ka-petansky technique for repair of whistling deformities of theupper lip. Plast. Reconstr. Surg. 57: 70, 1976.

16. Yoshimura, Y., Nakajima, T., and Yoneda, K. Propeller flapfor 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 forcorrecting the whistling deformity. Ann. Chir. Plast. 23: 108,1978.

18. Rodgers, C. M., and Mulliken, J. B. Deepithelialized of mu-cosal-submucosal flaps to correct the “whistling” lip defor-mity. Cleft Palate J. 26: 136, 1989.

19. Chong, J. K., and Winslow, R. B. Simple technique for cor-rection of “whistling” deformity in repaired cleft lips. Plast.Reconstr. Surg. 48: 84, 1971.

20. Vecchione, T. R. Correction of whistling tip deformity usinga palatal mucosa graft. Plast. Reconstr. Surg. 69: 344, 1982.

21. Patel, I. A., and Hall, P. N. Free dermis-fat graft to correct thewhistle 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. Bilateraland Rare Deformities. Boston: Little, Brown, 1977.

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24. Spina, V., Kamakura, L., and Lapa, F. Surgical managementof bilateral cleft lip. Ann. Plast. Surg. 1: 497, 1978.

25. Robinson, D. W., Ketchum, L. D., and Masters, F. W. DoubleV-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 minorpostoperative cleft lip “whistling deformity”. Br. J. Plast. Surg.25: 152, 1972.

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28. Cronin, T. D., Cronin, E. D., Roper, P., Jr., Millard, D. R., andMcComb, H. Bilateral clefts. In J. G. McCarthy, (Ed.), PlasticSurgery. Philadelphia: Saunders, 1990.

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