Cleft lip patients have a variety of secondary deformities after primary cheiloplasty. Whistle deformity, one of these secondary deformities, is common and usually requires surgical correction. Henkel et al. [
6] reported the following secondary revision rate: the incidence of secondary surgery for cleft lip patients was about 35%. Among all of the surgical correction cases, the lip was corrected in about 58%, and the nostril and columella in about 30%. About 29% of the cases underwent revision for whistle deformity of the lip. The causative factors of whistle deformity include tissue deficiency in the central tubercle area and mal-alignment of the muscles. There are various methods for correcting the whistle deformity, which can be mainly divided into three categories depending on the types of tissue for the correction of deformitiesn [
3,
7,
8].
First, if the vermilion deficiency is mild, the upper lip itself can be used for reconstruction, such as Z-plasty, V-Y advancement, double pendulum flaps, bilateral lateral vermilion border transposition flaps, and bilobed mucosal flaps. Z-plasty and V-Y advancement are performed both the most frequently and easily. However, these methods are restricted to cases of small lesions. Kapetansky [
9] reported double pendulum flaps for augmentation of the central defect. Juri et al. [
10] developed the bilateral mucomuscular flaps to fill up the insufficient central tubercle in the Kapetansky technique. Matsuo et al. [
11] developed bilateral lateral vermilion border transposition flapsn. This method can reduce the tension of the upper lips and deepen the labiogingival sulcus. However, the use of this method can impair the continuity of the orbicularis oris muscle because this flap contains only a thin layer of orbicularis oris muscle. In addition, a bilobed mucosal flap has some disadvantages in that there is a slight difference in the color between the dry and wet mucosa and the technique leaves additional scars in the vermilion [
3].
Secondly, if a defect in the vermilion is moderate or severe, the adjacent tissue, including the tongue and lower lip, can be used. Guerrero-Santos [
12] used two types of tongue flap for the reconstruction of the upper lip. However, these methods cause the discomfort to the patient of spending approximately three weeks with the tongue flap retained between the lips, and the patients must undergo secondary surgery. Furthermore, a postoperative mismatch in the texture and color remained. The Abbe flap is one of the good methods for treating the upper lip when the defect is severe [
13,
14]. This technique also needs two stage surgery and leaves a donor site deformity on the lower lip as well as a mismatch in the texture and color on the upper lip.
Finally, autograft or allograft material for reconstruction can be used. Patel and Hall [
7] used dermal fat grafting to correct a whistle deformity although it has been conventionally used to correct a facial contour or nose deformity. Trussler et al. [
2] reported upper lip augmentation using the palmaris longus tendon. Niechajev [
15] also reported lip enhancement using various alternatives including implants, autologous fat graft, and dermofat graft. Dermofat and autologous fat graft methods are simple and easy techniques, but these were less reliable because it is difficult to predict the degree of absorption and patients are at increased risk of developing an infection [
16,
17]. Wakami et al. [
17] used the mucosal flap concomitantly with artificial dermis. As described here, there are various surgical methods depending on the size of defects and the clinical characteristics of patients, each of which has its own benefits and drawbacks.
The authors have speculated that the mucosa or other tissues, except muscle, are less reliable and not firm enough for reconstructing a defect. In whistle deformity in secondary unilateral cleft lip, the method of using orbicularis oris muscle flaps on the lateral portion and medial tubercle portion was planned. The characteristics of the author's cases in secondary unilateral cleft lip are volume deficiency in half of the central vermilion notching, and lateral bulging. For solving these problems, we modified a crossed-denuded flap that Guerreo-Santos et al. used for the primary surgery of cleft lip in order to prevent the vermilion notching [
1]. After excision of a previous scar on the vermilion notching area, we dissected half of the muscle on the medial and lateral vermilion. Dissected bulged orbicularis oris muscle on the affected side and medial orbicularis oris muscle were crossed and turned over for the reconstruction of the defective central tubercle. In cases that had vermilion notching with a lateral bulging segment on the affected side, we could lessen the lateral bulging segment on the affected side and augment the half area of the defective central tubercle and correct the lip notching deformity at the same time. After judging the direction of the vermilion notching, the orbicularis oris muscle flap was created according to the location of the notching. If the vermilion notching was situated at the antero-posterior portion, the authors produced both muscle flaps in the anteroposterior portion. This means that the position of the muscle flap was dependent on the location of the vermilion notching. This point is different from the method reported by Cho and Kim [
18], in which vertical interdigitation of the orbicularis oris muscle flap was used to reconstruct a depressed philtralcolumn. Additionally, in cases of philtral reconstruction, the present authors have been using a buried deepithelized scar to add projection to the philtral column in a roll-over muscle flap from the central vermilion [
19]. The surgical techniques of our cross-muscle flap can be easily performed and the correction can be achieved with a one-time procedure. The additional scars can be avoided by cutting through the previous vermilion scar incision. In addition, the orbicularis oris muscle plays a primary role in generating facial expressions [
20]. Our methods can realign the orbicularis oris muscle and obtain an adequate volume of tissue and thereby restore the continuity of the orbicularis oris muscle. This method eventually contributed to achieving muscle rearrangement as well as symmetry on the upper lip. Thus, satisfactory outcomes from both functional and aesthetic perspectives could be obtained.
In conclusion, the authors' cross-muscle flap can correct the whistle deformity, which isa defect of volume in the affected central tubercle, vermilion notching, and lateral bulging. It uses partial orbicularis oris muscle flaps from the lateral bulging segment and medial tubercle area in cases of secondary unilateral cleft lip. It is an easy and effective method and leaves minimal scars and fewer complications. Furthermore, this is one-time procedure and does not create additional scarring on theupper or lower lip. A cross-muscle flap is a reliable option worth considering for reconstruction of whistle deformity.