Correction of secondary cleft lip deformity: the whistle flap procedure.

Abstract

The Kapetansky flap was described to repair a whistle deformity or central vermilion defect following a primary bilateral cleft lip repair. The authors studied a modification of this technique, called the Whistle (wide-hinged island swing transposition labial enhancement) flap, to correct a wide array of secondary cleft lip deformities.
Patients with secondary cleft lip deformity who underwent the Whistle flap correction were studied (n = 21). Vertical lip height, lateral lip projections, and three-dimensional volumetric measurements were recorded along with physician and parent-patient satisfaction surveys.
From 1994 to 2006, 23 Whistle flap procedures were performed on 21 cleft lip patients (bilateral, n = 13; unilateral, n = 8). The average age at surgery was 15.4 years (range, 13 to 21 years). The average follow-up was 27.6 months. The vertical height of the cleft side Cupid's bow to vermilion bottom had a mean increase of 164 percent. Lateral projection of the upper lip showed a mean increase of 117 percent. Preoperatively, patients exhibited mean volumetric asymmetry of 29.4 percent, and this was reduced following treatment to approximately 3.1 percent. Physician satisfaction using a modified Whitaker classification (categories I through IV) demonstrated that 56 percent of patients were category I (no refinements necessary) and 44 percent were category II (minor revisions are advisable). Parent-patient satisfaction in the follow-up period was a mean of 3.5 as measured using a five-point scale ranging from 0 to 4. There were minimal perioperative complications.
The Whistle flap procedure provided a versatile and reliable option for the correction of vermilion defects from secondary cleft lip deformities.

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PEDIATRIC/CRANIOFACIAL
Correction of Secondary Cleft Lip Deformity:
The Whistle Flap Procedure
Navanjun S. Grewal, M.D.
Henry K. Kawamoto, M.D.,
D.D.S.
Anand R. Kumar, M.D.
Bryan Correa, M.D.
Arthur E. Desrosiers, III,
M.D.
James P. Bradley, M.D.
Los Angeles, Calif.
Background: The Kapetansky flap was described to repair a whistle deformity
or central vermilion defect following a primary bilateral cleft lip repair. The
authors studied a modification of this technique, called the Whistle (wide-
hinged island swing transposition labial enhancement) flap, to correct a wide
array of secondary cleft lip deformities.
Methods: PatientswithsecondarycleftlipdeformitywhounderwenttheWhistle
flap correction were studied (n ? 21). Vertical lip height, lateral lip projections,
and three-dimensional volumetric measurements were recorded along with
physician and parent-patient satisfaction surveys.
Results: From 1994 to 2006, 23 Whistle flap procedures were performed on 21
cleft lip patients (bilateral, n ? 13; unilateral, n ? 8). The average age at surgery
was 15.4 years (range, 13 to 21 years). The average follow-up was 27.6 months.
TheverticalheightofthecleftsideCupid’sbowtovermilionbottomhadamean
increase of 164 percent. Lateral projection of the upper lip showed a mean
increase of 117 percent. Preoperatively, patients exhibited mean volumetric
asymmetry of 29.4 percent, and this was reduced following treatment to ap-
proximately 3.1 percent. Physician satisfaction using a modified Whitaker clas-
sification (categories I through IV) demonstrated that 56 percent of patients
were category I (no refinements necessary) and 44 percent were category II
(minor revisions are advisable). Parent-patient satisfaction in the follow-up
period was a mean of 3.5 as measured using a five-point scale ranging from 0
to 4. There were minimal perioperative complications.
Conclusion: The Whistle flap procedure provided a versatile and reliable
option for the correction of vermilion defects from secondary cleft lip
deformities.(Plast. Reconstr. Surg. 124: 1590, 2009.)
R
tionofthecleftlipdeformity,secondarycentrallip
deformities occur in a significant number of pa-
tients. Secondary lip deformities include inade-
quate tissue volume of the upper lip, a large or
small prolabium segment with a poorly defined
Cupid’sbowandphiltrum,averticallyshortupper
lip, a shallow buccal sulcus, and/or a cleft-side
vermilionbottomscarcontracture.Thesepatients
eventually require a secondary procedure to re-
store lip symmetry and improve function, includ-
ing speech, lip competence, and mentalis muscle
evisionlipsurgeryfollowingprimarycleftlip
repair is frequently necessary. Despite sig-
nificant improvements in primary correc-
strain for lip seal. Cohen et al.1reported an aver-
age of 1.13 lip revisions on unilateral cleft lip
patients and 2.17 lip revisions on bilateral cleft lip
patients.
In the secondary cleft lip deformity, mismatch
ofwetanddryvermilionandlipprojectionmaybe
problematic. For significant deformities, rotation
advancement flap reoperation is often necessary.
For lesser deformities, many options exist, includ-
ing local flaps, Z-plasties, tongue grafts, and fillers
(e.g., hyaluronic acid, collagen, and fat grafts).
The uses of local axial-based flaps have also been
described. In addition, an Abbe flap2may be used
From the Department of Surgery, Division of Plastic and
Reconstructive Surgery, University of California, Los
Angeles.
Received for publication July 31, 2008; accepted December
11, 2008.
Copyright©2009bytheAmericanSocietyofPlasticSurgeons
DOI: 10.1097/PRS.0b013e3181b99e2e
Disclosure: The authors have no financial interest
to declare in relation to the content of this article. All
sources of funds supporting the completion of article
are under the auspices of the University of Califor-
nia, Los Angeles.
www.PRSJournal.com1590

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to recruit additional lip tissue and to correct the
cleft lip deformity. In 1971, Kapetansky described
an axial-based double pendulum flap for correc-
tion of central lip deformities in bilateral cleft lip
patients.3
We report a modification of Kapetansky’s ax-
ial-based pendulum flap and a new procedure for
correction of central lip deficiencies in both uni-
lateral and bilateral cleft lip patients called the
Whistle (wide-hinged island swing transposition
labial enhancement) flap procedure. Although
the Kapetansky technique entails preservation of
the labial artery and dissection of bilateral pen-
dulum flaps with subsequent medial transposition
for the correction of the central lip deficiency, the
Whistle flap modification transposes or “swings”
larger (superior and lateral) mucomuscular flaps
from lateral to medial (to fill the labial cleft de-
fect) without preservation of a labial artery pedi-
cle. We evaluated a series of patients with second-
ary cleft lip deformities who underwent the
Whistle flap procedure using physician and par-
ent-patient outcome surveys and comparative
measurements.
PATIENTS AND METHODS
Cleft lip and cleft lip and palate patients who
underwentcorrectionofsecondarylipdeformities
using the Whistle flap procedure from 1994 to
2006 at the University of California, Los Angeles
were included in this study (n ? 21). Excluded
from the study were patients with incomplete
records (examinations, photographs, or incom-
plete surveys), inadequate follow-up, or other si-
multaneous procedures that would affect the lip
(e.g., Le Fort I advancement or philtral column
augmentation). Each patient was assessed preop-
eratively by the multidisciplinary cleft lip and pal-
ate–craniofacial team. Sex, age, diagnosis of cleft
disorder, previous correctional procedures, and
indication for operation were recorded.
Operative Technique
The operation was carried out with the pa-
tients under general anesthesia. Patients were ad-
ministered Ancef, (Research Triangle Park, N.C.)
intravenously before incision. Before infiltration
of local anesthesia, Whistle flap markings were
made with Bonnie blue ink. A transverse, linear
markwasmadebetweenthewetanddryvermilion
along the cleft vermilion defect. Next, the Whistle
flap markings were made to use the excess lateral
lip element to fill the cleft vermilion defect. To
mark the flap, the length of the vermilion defect
was measured and used to mark the length of the
Whistle flap. Then, the width of the flap was es-
timated and marked as a long ellipse or W-plasty
incorporating equal portions of wet and dry ver-
milion (Fig. 1).
After local infiltration with 0.25% Marcaine
(AstraZenecaPharmaceuticals,Wilmington,Del.)
and 1:200,000 epinephrine, a linear incision was
madewithano.15scalpelbetweenthewetanddry
vermilion of the cleft lip defect and carried onto
the marked elliptical Whistle flap. (Alternatively,
a zigzag W-plasty incision may be used in the ver-
milion instead of a straight line.) Through the dry
mucosa incision, subcutaneous sharp dissection was
usedbetweentheorbicularismuscleandskin.Next,
through the wet vermilion incision, dissection was
performed between the orbicularis muscle and oral
mucosa glands. Dissection on either side of the flap
was performed approximately 2 cm deep (wide-
hinged). Whistle flap elevation was continued later-
ally by beveling the orbicularis muscle dissection
toward the central defect to facilitate movement of
the flap. This created an island of labial tissue to be
swung and transposed toward the central defect (is-
land swing transposition).
After adequate mobilization, the flap was then
transposed from the area of lateral fullness into
the central cleft deficiency toward the central tu-
bercle. [This equalized the distribution of the soft
tissues of the lip. With the mobilization and trans-
position of the Whistle flap, the lateral portion of
theupperlipwasreducedinbulkandtheareajust
underneath the lip scar was increased in bulk (la-
bial enhancement).] Finally, the wet portion of
Fig.1. IllustrationsoftheWhistleflapprocedure.(Bluehatching
indicatesthewide-hingedbaseofthelateralvermilionflap).(Left)
Intraoperativemarkingwithexcessivelaterallipelementinclud-
inghalfwetandhalfdryvermilion.Arrowdepictstranspositionor
swingofislandflap.(Right)Whistleflapinsettofillvermilionde-
fect below cleft scar.
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the mucosa was closed with interrupted 5-0 chromic
suture(Ethicon,Inc.,Somerville,N.J.).Thedrypor-
tion was closed with interrupted 5-0 plain catgut
suture (Ethicon). Bacitracin ointment was applied.
For bilateral cleft central deficiencies, a bilat-
eral Whistle flap may be used with similar mark-
ings, dissection, and transposition. With the bilat-
eral cleft, the Whistle flaps may be advanced
centrally to overlap each other.
Outcome Analysis
Secondary cleft lip deformity was character-
ized and included inadequate central lip volume,
lip contour asymmetries, vermilion bottom scar-
ring, or restriction of dynamic lip movement. The
whistling deformity was defined as insufficient tis-
sue in the lower border of a repaired cleft lip,
giving the appearance of whistling while in repose
and worsening on activation. The operative sur-
geondocumentedperioperativecourseproblems,
including wound breakdown, bleeding, infection,
hypertrophic scarring, functional lip problems, or
other complications. In addition, patient prob-
lems such as lip pain, tightness, smiling or speech
disturbances, or scabbing of malpositioned wet
vermilion were recorded.
Three independent examiners (a plastic sur-
gery resident, a geneticist/pediatrician, and a
craniofacial fellow) recorded upper lip measure-
ments of images in the preoperative and follow-up
periods (6 weeks and 1 year). Follow-up photo-
graphs were obtained before subsequent revision
operations (if any). Measurements were used to
calculate lip fullness and symmetry. The measure-
ments were performed in repose or a relaxed,
lips-separated position. Vertical upper lip mea-
surements included the vertical distance of the
white roll to the vermilion bottom at the tubercle,
Cupid’s bow points, and midlateral lip (five ver-
tical measurements per lip) (Fig. 2).4In addition,
lateral measurements of the upper lip were per-
formed. For this lateral projection measurement,
the right-angled distance from the N-ANS line
[radix (nasion) to the anterior nasal spine] to the
anterior lip projection point was used (Fig. 3).
Augmentation percentage was documented using
follow-up measurements compared with preoper-
ative measurements.
In addition, a digital three-dimensional pho-
togrammetry system (MU-4 imaging system;
3dMD, Atlanta, Ga.) was used to quantitate volu-
metric lip symmetry (Fig. 4). The 3dMD system is
a fully automated, digital, three-dimensional pho-
Fig.2. Comparativeupperlipverticalvermilionmeasurementsfromthewhiterollto
thevermilionbottom.(Left)Preoperativeimagedemonstrateshortvermilionheighton
thecleftside.(Right)Postoperativeimagedemonstratescorrectionofcleftsidevermil-
ion height. T, central tubercle; CB, Cupid’s bow points (right and left); MLL, midlateral lip
(rightandleft).
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togrammetric device capable of very fast (?1 sec-
ond), high-resolution, color surface captures of
craniofacialsurfacedata.5,6Three-dimensionalim-
aging has become increasingly recognized as an
acceptable tool for volumetric analysis.7,8The sys-
tem includes proprietary software that enables us-
ers to interactively locate landmarks directly on
captured objects and provides a set of measure-
ment tools for calculating linear distances and for
making volumetric grid calculations. During two
Fig.3. Comparativeupperliplateralprojectionmeasurements.Theright-angleddis-
tancefromanasion(N)totheanteriornasalspine(ANS)linetoapointatthelateralmost
projectionoftheupperlipwasrecorded.(Left)Preoperativeimagedemonstratingpoor
lateral lip projection on the cleft side. (Right) Postoperative image demonstrating im-
proved lateral lip projection on the cleft side.
Fig.4. Follow-uplipsymmetrywasassessedwithvolumetricandsurfaceareaanalysisusing
3dMDphotogrammetry.(Left)Frontalviewshowingvolumeandsurfaceareacorrectionwith
improved symmetry. (Right) Worm’s-eye view demonstrating intraoral view of volume and
surface correction.
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separate sessions, lip symmetry was calculated us-
ing a surface area analysis. The measurements
were obtained using two separate captures: (1)
mouth closed, lips parted, frontal view; and (2)
mouthopen,worm’seyeview.Astrictprotocolfor
the distance of the patient from the camera and
photographer has been consistent and standard-
ized throughout the duration of the study.
Lip asymmetry was calculated as the absolute
value of the difference in the percentage between
the volumetric measurement or surface area of
each hemilip according to the following formula:
asymmetry ? (right segment value – left segment
value) – 100/(right segment value ? left segment
value). This calculation used the sum of the right
and left values (which varied between patients).
Thus, perfect symmetry would result in a 0 value.
The absolute value was used, as some patients had
slight lip asymmetry to either the right or the left
side postoperatively. This measurement tech-
nique has been described previously.9
Aestheticoutcomedeterminedinfollow-upby
three reviewers was based on a modified Whitaker
classification as a way of determining the need for
further lip revisions based on lip symmetry and
fullness, as follows: category I, no refinements or
surgical revision is considered necessary; category
II, minor revisions are advisable; category III, ma-
jor revisions are advisable; and category IV, major
revision duplicating or exceeding the original pro-
cedureisnecessary(Table1).10Atfollow-up,parent-
patient satisfaction was recorded on a five-point
scale (0 ? dissatisfied or no improvement, 1 ?
onlypartiallysatisfiedor25percentimprovement,
2 ? half satisfied or 50 percent improvement, 3 ?
mostly satisfied or 75 percent improvement, and
4 ? totally satisfied or 100 percent improvement)
(Fig. 5).10This satisfaction survey was filled out by
the parents and patients and specifically ad-
dressed lip fullness, symmetry, pain, and overall
appearance. The mean and median scores of the
survey are given.
Statistical Analysis
An unpaired t test was used to determine sig-
nificance between preoperative and follow-up
measurements. Results between preoperative and
follow-up vertical height and lateral projection
measurements are given as mean ? SD. A value of
p ? 0.05 was considered statistically significant.
RESULTS
Between 1994 and 2006, 21 patients (10 women
and 11 men; 13 bilateral and eight unilateral) un-
derwent 23 Whistle flap procedures and had com-
pleterecordsandimagingnecessaryforcomparative
analyses. The mean age at the time of surgery was
15.4 years, and the age range was 13 to 21 years.
Fifty-eightpercentofpatientsunderwentminorcleft
lip revision after primary cleft lip repair before the
Whistle flap procedure. Study patients with second-
ary cleft lip deformity had inadequate central lip
volume (100 percent), lip contour asymmetries (86
percent), vermilion bottom scarring (86 percent),
or excess lateral lip volume (90 percent). In addi-
tion, they were documented as having worsening
whistle deformity on activation (81 percent), and
restriction of dynamic lip movement (71 percent).
In the perioperative course, minimal compli-
cations occurred: wound breakdown (0 percent),
bleeding (9 percent), infection (14 percent) that
resolved after a course of oral antibiotics, hyper-
trophic scarring (4 percent), functional lip prob-
lems during the first 6 weeks postoperatively (9
percent), or total number of patients with any
complications (21 percent). The average fol-
low-up was 27.6 months. Minimal long-term pa-
tient problems were noted: long-term lip pain (4
percent), tightness (9 percent), smiling difficul-
ties (9 percent), speech disturbances (0 percent),
or scabbing of malpositioned wet vermilion (4
percent). All patients demonstrated very good to
excellent functional outcomes without change in
the ability to smile. After the Whistle flap proce-
dure, 26 percent underwent future minor cleft lip
revisions (many at the time of other procedures)
and none underwent a major revision.
Comparative lip measurements showed im-
provement in lip symmetry and vermilion height
at the cleft side Cupid’s bow. When follow-up up-
per labial measurements after the Whistle flap
procedure were matched against preoperative up-
per labial measurements, a mean 164 ? 32 per-
centincreasewasseenincleftCupid’sbowvertical
height (p ? 0.05). Five vertical upper lip measure-
mentsincludedtheverticaldistanceofthewhiteroll
tothevermilionbottomatthetubercle,Cupid’sbow
Table 1. Aesthetic Outcome Scale
Category
I
Description
No refinement or surgical revision is considered
necessary
Minor revisions are advisable
Major revisions are advisable
Major revision duplicating or exceeding the
original procedure is necessary
*Based on modified Whitaker classification (Whitaker LA, Bartlett
SP, Schut L, Bruce D. Craniosynostosis: An analysis of the timing,
treatment and complications in 164 consecutive patients. Plast Re-
constr Surg. 1987;80:195–212).
II
III
IV
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