PEDIATRIC/CRANIOFACIAL Unilateral Cleft Lip Repair Using the Anatomical Subunit Approximation: Modifications and Analysis of Early Results in 100 Consecutive Cases Raymond Tse, M.D. Samuel Lien, M.D. Seattle, Wash.
Background: The anatomical subunit approximation for unilateral cleft lip repair has gained acceptance; however, outcomes have not been reported since the original description. The purpose of this article is to report the experience using this technique. Methods: One hundred two consecutive patients underwent primary cleft lip repair performed by a single surgeon over a 3-year period. Objective analysis involved anthropometric measurements performed on preoperative and postoperative three-dimensional images. Subjective analysis involved Asher-McDade scores by blinded independent surgeons. Observational insights were gained by review of surgical records. Anthropometric measurements were expressed as ratios of the cleft side to the noncleft side. Differences in ratios were assessed by using the rank sum test. Differences in Asher-McDade scores were assessed using the Mann-Whitney test. Results: Demographic, cleft type, cleft extent, and cleft severity were consistent with our center’s norms. The mean age at surgery was 6 months and the mean inferior triangle used was 1.8 ± 0.9 mm. Anthropometric ratios were significantly improved postoperatively and approached 1, regardless of initial cleft severity. Ten subjects who underwent repair early in the experience were compared with 10 subjects who underwent repair late in the experience. There was no significant difference in postoperative anthropometric measures or Asher-McDade scores. Conclusions: The anatomical subunit approximation for unilateral cleft lip repair in a single-surgeon series can achieve improvements in anthropometric measures and early favorable postoperative form. The technique could be applied to all cleft types and there was little change in outcome with greater surgeon experience. Long-term follow-up is necessary. (Plast. Reconstr. Surg. 136: 119, 2015.) CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.
P
rinciples of any reconstruction include a detailed analysis of form, a surgical plan, wide release, and component reconstruction.1 Surgical markings for unilateral cleft lip repair are one component of repair and constitute the plan that helps to achieve balanced nasolabial form and a favorably positioned scar.2 The ideal technique should minimize variations From Seattle Children’s Hospital, University of Washington. Received for publication October 21, 2014; accepted J anuary 27, 2015. Presented at the 71st Annual Meeting of the American Cleft Palate-Craniofacial Association, in Indianapolis, Indiana, March 24 through 29, 2014. Copyright © 2015 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000001369
resulting from surgeon experience and ability, be applicable to a wide spectrum of severity, and Disclosure: The authors have no financial interest to declare in relation to the content of this article. No external funding was received. Supplemental digital content is available for this article. Direct URL citations appear in the text; simply type the URL address into any Web browser to access this content. Clickable links to the material are provided in the HTML text of this article on the Journal’s Web site (www. PRSJournal.com).
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Plastic and Reconstructive Surgery • July 2015 respect the principle of placing scars along anatomical subunits.3,4 Millard’s rotation advancement5 and its modifications6–8 involve a “cut-as-you-go” approach that can be prone to surgeon ability and experience and has been criticized for producing short lips when used for wide clefts.9–11 The Tennison- Randall repair is more geometric and is thought to be easier to learn12 and more suitable for wide clefts13; however, the resultant scar inevitably crosses natural contours.14 The Anatomical Subunit Approximation for unilateral cleft lip repair was described in 200515 and has been characterized as a “hybrid” technique.12 The design includes multiple landmarks with the intention of approximating incisions along the borders of anatomical subunits.15 Although the technique has gained popularity,16 the experience using this approach has yet to be described by other surgeons. The purpose of this study is to report the early experience, modifications, and results using the anatomical subunit approximation technique for unilateral cleft lip repair performed by a single surgeon.
PATIENTS AND METHODS Approval for this study was obtained from the institutional review board. Consecutive patients who underwent primary unilateral cleft lip repair performed by the senior author (R.T.) from 2010 to 2013 were included in this study. Cleft Lip Repair Design/Lip Markings Landmarks for cleft lip repair follow those described for the Anatomical Subunit Approximation technique.15 Most are standard anthropometric landmarks (Fig. 1, above, left) and are described here using anthropometric terminology rather than assigned numbers. Marks are placed starting at the midline, identifying the noncleft side, and then, using the known landmarks, to define the less distinct cleft side landmarks. The full stepwise sequence of landmarks is illustrated in the slide presentation. (See Figure, Supplemental Digital Content 1, which shows the sequence of landmarks for the anatomical subunit approximation for unilateral cleft lip repair, http://links.lww.com/ PRS/B332.) Lip points. The subnasale (sn) and the top of the philtral columns [crista philtri superioris, (cphs and cphs′)] are found along the nasolabial crease. The Cupid’s bow is identified along the vermilion
border and is defined by the two peaks [crista philtri inferioris (cphi and cphi′)] and its low point [labiale superioris (ls)]. Given that transitions in contours and concavities hide scars better,3 the philtral column points, cphi and chps, are identified along the medial border of the philtral ridge, where the convexity of the column transitions to the concavity of the philtral dimple (Fig. 1, above, left). The lateral point of lip closure, cphiʺ, is identified based on tissue adequacy according to Noordhoff and is defined as the point along the vermilion border at which the vermilion is the widest.17 Medial to this point, the white roll becomes less distinct and the vermilion becomes deficient. Once all of the landmarks along the vermilion have been identified, corresponding points above the white roll (black dots with white outline in Fig. 1, center, left) and along the mucosa-vermilion border (black dots with red outline in Fig. 1, center, left) can be identified (perpendicular to the lip). Nose points. The subalare (sbal), the lowest point along the ala, is identified along the nose-lip crease. The alar insertion point (ai) was not part of Fisher’s initial description and is found at the intersection of the alar rim with the nasal sill, along the noselip crease (Fig. 1, above, left). These landmarks are used to determine the nasal floor closure needed to correct the nasal deformity. Contrary to the lip, nose landmarks are identified with the deformity manually corrected so that the orientation of sbal-ai on both sides match (Fig. 1, above, right). On the noncleft side, an arbitrary point (x) is defined based on the intersection of the linear projections from sbal-ai and sn-cphs (i.e., sbal-aix and sn-cphs-x should both form straight lines). The corresponding arbitrary points (x′ and xʺ) are then defined on the cleft side using distances from sn-cphs′ and sbal′-ai′ that mirror the corresponding distances from the noncleft side. When x′ and xʺ are brought together (i.e., to mimic x), the nasal deformity should be corrected (Fig. 1, above, right). When the nasal floor is intact, appropriate nasal floor excision can be further verified by measuring nostril circumferences using urethral sounds (circumference = diameter × π). The distance between x′ and xʺ should not exceed the difference in circumferences to avoid producing a micronostril. Medial markings: connect the dots. The medial lip markings simply connect the landmarks along the nasal sill (x′-cphs′); along the philtral column (cphs′-cphi′); and across the white roll, vermilion border, and red line at cphi′
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Volume 136, Number 1 • Anatomical Subunit Approximation
Fig. 1. Lip markings. (Above, left) Standard anthropometric landmarks. Alar insertion point (ai) is the intersection of the nostril rim and the nasal sill along the alar crease. (Above, right) Nose landmarks need to be identified with the nasal deformity corrected. Point x is an arbitrary point that is collinear with both sbal-ai and sn-cphs. The points x ’ and x" are measured off of sn’-cphs’ and sbal’-ai’, respectively, by mirroring distances on the noncleft side. When x ’ and x" are brought together, they should be the equivalent of x and the nasal deformity should then be corrected. (Center, left) The medial lip markings connect the dots along the border of anatomical subunits with back-cuts above the white roll and along the mucovermilion border. The medial vermilion height is transposed to the vermilion at cphi" and the remaining vermilion is used to augment the deficient medial vermilion. The cphi" remains fixed and the remaining lateral lip markings fit the space from above the white roll to the lateral point of nasal closure, x". (Center, right) An equilateral triangle is used to augment medial lip height. The base width, C, is calculated based on the difference in philtral heights, minus 1 mm, given the RoseThomson effect (lengthening with diamond-shaped excision and closure). The limbs of the lateral lip markings need to match the medial lip and can articulate open or closed. The angle between limbs s and B should match the medial side as much as possible. (Below) Variations in lateral lip design based on lateral lip height.
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Plastic and Reconstructive Surgery • July 2015 (Fig. 1, center, left). This last incision is made perpendicular to the lip so that its incidence with cphs′-cphi′ forms an angle that will open following incision (Rose-Thompson effect). A small backcut is designed above the white roll and is oriented to provide maximal vertical release. The length of the backcut is based on the size of the calculated equilateral triangle from the lateral lip that will be needed to augment medial lip height. Another incision is designed along the red line so that the deficient vermilion height may be augmented with a lateral lip vermilion flap.17,18 Lateral markings: match the medial marks and fill the deficits. On the lateral lip, the deficient vermilion height at cphi′ is transposed to the vermilion height at cphiʺ so that the excess lateral vermilion height can be used as a triangle to augment medial height (black dots with red outline in Fig. 1, center, left). The incision across the white roll connects cphiʺ and its corresponding point above the white roll. The remaining lateral lip markings then need to fit between this last point and the lateral point of nasal closure, xʺ (Fig. 1, center, left). Limbs that match the medial lip incisions (s, along the nasal sill; B, the cleft side philtral height), with the addition of a small triangle to augment medial lip height, need to be drawn and fit into the fixed space (Fig. 1, center, right). The triangle base width, C, is the difference in philtral heights (cphi-cphs versus cphi′-cphs′) minus 1 mm because of the Rose-Thompson effect (i.e., C = A − B − 1 mm). The lateral lip components can articulate open or close to fit the lateral lip height. An arc with radius, C, can be drawn from above the white roll point at cphiʺ and an arc with radius, s, can be drawn from xʺ. The cleft side philtral height (B) is then placed to connect the two arcs (Fig. 1, center, right). In contrast to other techniques, the cphiiʺ is fixed and is not shifted lateral in the case of vertical lip deficiency.19 When the lateral lip is short (i.e., complete clefts), the components need to fit into a smaller space and angles between incisions are made more acute (Fig. 1, below, left). In the case of greater lateral lip height (i.e., incomplete clefts), the components can fit into a larger space and the angles between components can be made more obtuse (Fig. 1, below, center). In the case of vertical lip excess (i.e., minor incomplete clefts), angles between lateral lip components should not be made so obtuse that they no longer match the medial lip incisions. In this case, a vertical wedge excision20 along the alar crease should be used to
effectively reduce the vertical height (Fig. 1, below, right). Remeasure and Cut as You Go The medial lip is incised and dissected first to verify that the backcut above the white roll is needed. The triangular base width (C) can also be verified with Cupid’s bow manually leveled. The designed points of nasal floor closure can be verified by manually correcting the nasal deformity after release of the upper sulcus and piriform aperture. Markings rarely need to be modified. At final closure, the lateral triangle above the white roll can be altered if needed. Release and Component Reconstruction Release and component reconstruction are equally as important as the design and incisions and have been previously described in more detail.2 The orbicularis muscle is released from the base of the columella at the level of the alveolus to allow the medial lip to drop. The orbicularis on the lateral side is dissected as a single layer between mucosa and skin and is released from the alveolus and alar base. This results in an empty triangle of muscle along the nose-cheek junction so that when the muscle is advanced medially, the alar crease is accentuated. Whereas the lateral muscle is dissected widely, dissection on the medial side is conservative to preserve the philtral dimple. An upper buccal sulcus incision and supraperiosteal dissection over the maxilla allow mobilization of the lateral lip as needed. The nose is released along the piriform aperture and can involve a submucosal dissection or transmucosal release. The nasal floor is fully closed to the level of the incisive foramen and the nasal sidewall is reconstructed with a turbinate flap,8,15 L-flap,21 or lateral nasal wall advancement.2 These have been previously described in further detail2 and are summarized in Figure 2. Each of these flaps provide mucoperiosteum to fill the dead space left by mucoperiosteal release of the lateral nasal wall with anterior repositioning of the ala. These flaps are incorporated into the closure of the nasal floor. A primary perichondrial-sparing septoplasty is performed to reposition the caudal septum into the midline of the face.22–25 Sutures are used to ensure the new septal position remains stable. No nasal tip dissection is performed. The orbicularis muscle is brought across the nasal floor/sill and inserted into the base of the columella to maintain the lengthened medial lip. The muscle is carefully reapproximated and the J shape of the pars marginalis is preserved, as it contributes to the pout of the lip.
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Volume 136, Number 1 • Anatomical Subunit Approximation
Fig. 2. Lateral nasal wall and nasal floor reconstruction. Options for lateral nasal wall reconstruction were based on cleft type and available exposure. (Left) Turbinate flap. Mucoperiosteum based anteriorly is transposed 90 degrees to fill the defect following alar base release and anterior repositioning. This requires an open cleft to allow posterior incision/release. (Center) L-flap. Marginal tissue is based on alveolar mucoperiosteum and transposed 90 degrees to fill the defect. (Right) Lateral nasal wall flap. The lateral nasal wall is raised and a backcut is made posteriorly to allow the ala to advance anteriorly. The donor site is made along the bony wall. (Reproduced with permission from Tse R. Unilateral cleft lip: Principles and practice of surgical management. Semin Plast Surg. 2012;26:145–155.)
Final Closure Skin flaps are inset using deep dermal sutures. For the first half of this case series, final skin closure involved 7-0 polypropylene sutures in a simple interrupted manner requiring removal under general anesthetic 1 week postoperatively. For the second half of this case series, final skin closure was modified so that suture removal could be achieved in the clinic without the use of anesthetic
or sedation. This involved a 6-0 polypropylene running intracuticular suture along the vertical limb of the philtral column (B) and 8-0 polyglactin in a simple interrupted manner for everything else. Analysis Demographic and surgical details were obtained by chart review. Previously captured twoand three-dimensional images were collected from
Table 1. Cleft Lip Type, Relative Dimensions, and Triangular Flap Base Width Cleft Type Microform Incomplete Complete plus band† Complete All types
No. 3 47 12 40 102
A, Total Lip Height (cphi-cphs) (mm)*
B, Greater Lip Height (cphi′-cphs′) (mm)*
7.0 (0.8) 7.5 (1.5) 7.2 (1.3) 8.3 (1.3) 7.8 (1.4)
5.8 (0.5) 5.2 (1.5) 4.0 (0.8) 5.0 (0.9) 5.0 (1.3)
C, Lesser Lip Height (triangle base width) (mm)* 0 (0) 1.4 (0.8) 2.1 (0.6) 2.2 (0.7) 1.8 (0.9)
*Values are expressed as means (SD). †A band is any soft-tissue connection of lip or nasal sill in the presence of a complete cleft alveolus (previously known as Simonart band).
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Plastic and Reconstructive Surgery • July 2015 our center’s archives for analysis. Standardized two-dimensional photographs were captured by a professional imaging technologist and included frontal, worm’s-eye, and lateral profile views that were consistent with Americleft guidelines.26 Three-dimensional images were captured using the 3dMDCranial system (3dMD, Atlanta, Ga.) according to a previously described protocol.27 Anthropometric measurements were performed using 3dMD Vultus v.2.2.011 on threedimensional images that were captured at the immediate preoperative and postoperative clinic visits. The system of quantitative morphometry has been previously described28 and is consistent with other reports.29 We focused on measurements that have been found to be reliable on three-dimensional imaging.28 These included medial lip height (sn-chpi), lateral lip height (sa-chpi), lateral lip length (ch-cphi), columellar angle, and nostril width. The columellar angle is measured from the submental view and is defined as the angle made by the columella relative to the sagittal plane of the face.30 It has previously been found to be one of the most important objective measures of preoperative cleft lip nasal severity.30–32 To characterize nasolabial balance, lip and nose measurements were expressed as ratios of cleft to noncleft side measurements. Ratios approaching 1 would suggest greater symmetry. We obtained more global assessments of nasolabial form using two-dimensional images that were captured at the immediate postoperative clinic visit. We followed methods described by Asher-McDade et al.33 and used by Americleft.26 Two-dimensional photographs were cropped to exclude features outside of the nasolabial region.33 Two groups of 10 consecutive cases were analyzed so that changes in outcome over this experience could be identified. The first group was early in the surgeon’s experience (2010) and the second group was later in the surgeon’s experience (2013). Three reviewers (Joseph Gruss, craniofacial surgeon with 30 years of cleft experience; Clinton Morrison, craniofacial fellow; and Samuel Lien, resident), who were blind to the groups, rated outcomes using reference photographs.34 To provide clinical examples, families of the middle 20 consecutive cases (in which 1-year follow-up was available) were asked for permission to publish their child’s photographs and cleft characteristics. All of the families consented. Statistical Analysis Outcomes of the two groups of patients at the beginning and end of the experience were analyzed.
Anthropometric ratios were compared using a rank sum test after all subjects were ranked according to their distance from the goal ratio of 1. Asher-McDade scores were compared using a Mann-Whitney test.
RESULTS Demographics These cases were performed during the first few years of the primary surgeon’s practice as a full-time pediatric plastic surgeon. One hundred two consecutive patients were included, of which 53 were male patients and 49 were female patients. Cleft extent was as follows: cleft lip, n = 41 (40 percent); cleft lip and alveolus, n = 14 (14 percent); and cleft lip and palate, n = 47 (46 percent). The distribution of cleft lip type is detailed in Table 1. Five patients had associated anomalies/ syndromes, including midline facial deficiency, craniofacial microsomia, popliteal pterygium, congenital constriction band, and undetermined chromosome abnormality. Median age at surgery was 6 months (range, 3 to 45 months). Reasons for later age at surgery included microform cleft, late presentation, and international adoption. Lip Design Relative lip heights are detailed in Table 1. The mean triangle base width, C, was 1.8 mm. The triangle tended to be smaller for less severe clefts and larger for more severe clefts. Anthropometric Measurements The three-dimensional images for 89 of the 102 patients were available for full analysis (six missing preoperatively, five missing postoperatively, and two missing both). Linear lip and nose dimensions were corrected and approached 1 postoperatively (Tables 2 and 3). Although these ratios varied with cleft lip type, the postoperative ratios were similar regardless of the initial cleft. Mean columellar angle was corrected to within 10 degrees, regardless of the cleft type and the preoperative columellar angle. Asher-McDade Scores Interrater reliability assessed by Pearson coefficients ranged from 0.3 to 0.5 between the three raters. Ratings by the three raters for the two groups are displayed in Figure 3, below. Early versus Late Experience There were no significant differences in anthropometric measurements (not displayed) and AsherMcDade scores for the two groups (Fig. 3).
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Volume 136, Number 1 • Anatomical Subunit Approximation severity. Given that the surgical design is based on anatomical landmarks, rather than dimensions, the lip markings inherently adjust for different cleft types. We found little change in outcome over time with greater surgeon experience. Anthropometric measures and subjective ratings were similar early and late in this series. In contrast to the freehand, cut-as-you-go style of rotation-advancement, Fisher’s original description included 25 numbered landmarks and far more initial measurements than any other approach.15 Although these may appear cumbersome, they are based on standard anthropometric landmarks (Fig. 1, above, left) that experienced cleft surgeons will identify or at least recognize when analyzing a cleft deformity. Understanding the system of landmarks requires considerable effort; however, the time spent may help to reduce the steepness of the learning curve. A technique should match a surgeon’s style and personality. The senior surgeon feels that the extra time spent on design allows incisions to be made with little hesitation, giving this technique a “measure-twice, cut-once” flavor. This may be helpful for less experienced surgeons and resident training. More experienced surgeons who consistently produce favorable results may have no need to adopt a new technique; however, for
Example Cases Cleft severity, as indicated by columellar angle,30 for the middle 20 subjects, was similar in distribution to the entire case series (Fig. 4). Ten of these subjects, spanning the spectrum of severity, are displayed in Figure. 5, and the full set of 20 subjects is displayed in Figure, Supplemental Digital Content 2 (http://links.lww.com/ PRS/B333). Subjects are arranged by cleft type and increasing columellar angle. One patient with an incomplete cleft lip had a contralateral mini-microform cleft lip35 that was left unaddressed. As a result, he has a minor whistle notch deformity caused by prolabial deficiency. One patient with complete cleft lip underwent nasoalveolar molding, whereas the others did not. The final patient had a midline deficiency with absent premaxilla consistent with what has been described as median facial dysgenesis/ dysplasia/hypoplasia36–38 or binderoid cleft lip.39 The caudal septum was absent, thereby limiting changes produced by septal repositioning.
DISCUSSION We found the Anatomical Subunit Approximation to be applicable across the spectrum of cleft severity. Anthropometric measures of asymmetry were corrected regardless of cleft type and
Table 2. Preoperative and Postoperative Lip Measurements/Ratios Medial Lip Height Ratio* Cleft Type Microform Incomplete Complete plus band† Complete All types
Lateral Lip Height Ratio*
Lateral Lip Length Ratio*
Preop
Postop
Preop
Postop
Preop
Postop
0.94 (0.04) 0.62 (0.12)
1.08 (0.09) 1.09 (0.12)
0.84 (0.12) 0.84 (0.12)
0.73 (0.11) 0.93 (0.07)
0.93 (0.12) 0.88 (0.09)
0.90 (0.00) 0.92 (0.10)
0.62 (0.19) 0.53 (0.12) 0.59 (0.15)
1.21 (0.21) 1.05 (0.11) 1.10 (0.15)
0.74 (0.16) 0.71 (0.13) 0.77 (0.14)
0.89 (0.10) 0.90 (0.08) 0.91 (0.09)
0.88 (0.10) 0.88 (0.09) 0.88 (0.08)
0.93 (0.10) 0.93 (0.09) 0.93 (0.09)
Preop, preoperative; Postop, postoperative. *Values are expressed as ratios of cleft to noncleft side (SD). †A band is defined as any soft-tissue connection of lip or nasal sill in the presence of a complete cleft alveolus (previously known as Simonart band).
Table 3. Preoperative and Postoperative Nose Measurements/Ratios Columellar Angle* Cleft Type Microform Incomplete Complete plus band† Complete All types
Nostril Width Ratio*
Preop
Postop
Preop
Postop
15.29 (0.73) 17.47 (7.88) 22.27 (9.52) 37.02 (11.64) 26.22 (13.43)
9.19 (1.92) 8.52 (5.22) 7.51 (5.12) 8.98 (5.28) 8.58 (5.20)
1.41 (0.22) 1.42 (0.25) 1.66 (0.28) 2.30 (0.43) 1.82 (0.54)
1.15 (0.08) 1.10 (0.15) 1.02 (0.14) 1.17 (0.24) 1.12 (0.20)
Preop, preoperative; Postop, postoperative. *Values are expressed as ratios of cleft to noncleft side (SD). †A band is defined as any soft-tissue connection of lip or nasal sill in the presence of a complete cleft alveolus (previously known as Simonart band).
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Plastic and Reconstructive Surgery • July 2015
Fig. 3. Asher-McDade scores for 10 subjects early in the experience and 10 subjects late in the experience. (Above) Distribution of scores. (Below) Mean scores.
some, the approach may provide a foundation of measurements from which results can be refined. There are several concepts used in this technique that are important to highlight. First, it uses a diamond-shaped excision to lengthen the lip (Rose-Thomson effect) and to minimize the size of the triangle needed to level Cupid’s bow. This allows the scar to be hidden behind the convexity of the white roll. Second, it uses a fixed cphiʺ that is selected based on tissue adequacy rather than on measured distances from the commissures21 or needed lateral lip height.29,40 The former21 can incorporate deficient tissues
into the repair,41 and the latter29,40 can sacrifice lateral lip length.19 Although measured ch-cphi increases with time following rotation advancement,29,40 preservation of lateral lip length may be important to avoid the accordion effect of tissue advancement that reduces the natural fullness of the lip. Third, in contrast to rotationadvancement, the alar base is incorporated into the design of the lip repair. This avoids additional scar along the alar base. Although lip markings are important for surgical planning, wide surgical release and component reconstruction are equally important in
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Volume 136, Number 1 • Anatomical Subunit Approximation
Fig. 4. Severity of cleft lip nasal deformity as measured by columellar angle for the entire series and for the middle 20 subjects presented as clinical examples.
producing favorable outcomes. Given that the underlying bony deformity will not be corrected until later in life, emphasis was placed on release, repositioning, and retention of the nasal foundation in three-dimensional space. Both the caudal septum22–25 and alar base were aggressively repositioned. Focus was placed on reconstruction of the nasal floor and lateral nasal wall to retain the basal nose components in space.2 Through the course of this series, muscle dissection evolved to be more aggressive, emphasizing the creation of an empty triangle of muscle lateral to the alar base. This dissection accentuates the alar crease when the muscle is repaired and may add to soft-tissue stability. Although a difference was not apparent using the outcome measures used at early followup, the additional soft-tissue corrections may be more subtle than can be measured and may contribute to long-term retention of corrected form. Gillies once wrote: “Never do today what can honourably be put off till tomorrow.”1 Nasal tip dissection was not performed in this series to avoid unnecessary scarification. Final skin closure was modified midway through this series to eliminate the need for suture removal under general anesthesia. No change in scar quality was noted; however, formal specific long-term evaluation would help to confirm this. The timing of surgery in this series was 6 months rather than 3 months of age15 to reduce the risks of anesthesia (immediate and long-term42) and to take advantage of larger and more robust anatomy.
Several pearls were noted on review of this series. First, when designing the lateral lip markings, the angle between limbs s and B should approximate that of the medial lip markings. If too obtuse, the desired line of closure along anatomical subunits will be blunted. To avoid this, if there is excess lateral lip height, a triangle should be excised (Fig. 1, below). Second, the nose deformity should be corrected manually when identifying nasal landmarks (Fig. 1, above, right). Final alar base position and rotation rely on the surgical markings. Although the alar insertion point was not included in Fisher’s original description, the additional landmark is particularly helpful for orienting alar rotation. Third, remeasure and modify as you go. The medial lip can be overlengthened. After making the backcut above the white roll on the medial lip, manually level Cupid’s bow and remeasure the base width of the triangle needed to augment medial lip height. The lateral triangle can be reduced before incising the lateral lip. Interpreting the literature on cleft lip repair can be difficult given that outcomes are subjective and many reports display only the best or highly selected outcomes.5,6,43–48 Our study included all patients in a consecutive series. Similar to Mulliken, we used anthropometric analysis29,49 to provide objective measures of specific changes. We also used Asher-McDade scores to provide a more global assessment of outcome.33 Given that raters were blinded to the groups, we could use their scores to compare results early and later in the surgeon’s experience; however,
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Plastic and Reconstructive Surgery • July 2015
Fig. 5. Example clinical cases preoperatively and 1 year postoperatively. The middle 20 subjects who had 1-year follow-up available were selected as clinical examples. Subjects were arranged by cleft type and increasing severity as measured by columellar angle. Ten cases that span the spectrum of severity are displayed here. The complete set of 20 are shown in Supplemental Digital Content 1, http://links.lww.com/PRS/B332.
because the raters were from our institution, comparison of scores to those of other studies should be tempered. We analyzed three-dimensional and twodimensional images captured immediately preoperatively and postoperatively given that our focus was on a technique of surgical planning, the purpose of which is to produce nasolabial balance at the completion of a cleft lip repair. Although previous studies have demonstrated that immediate results are the best predictor of eventual
outcome50,51 and that lip dimensions remain stable over time,9,11,29,40,50–53 longer term follow-up would be useful, and we plan to reassess this series when 4-year follow-up is available for all subjects. In the absence of ideal outcome measures, we have presented example photographs. Previous reports have used 10 consecutive patients to avoid selection bias54–57; however, Power and Matic argue that more subjects are needed.58 As such, we used the middle 20 consecutive cases as representative examples. Our study is one of the most
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Volume 136, Number 1 • Anatomical Subunit Approximation comprehensive reports of early outcome following cleft lip repair.
CONCLUSIONS The anatomical subunit approximation for unilateral cleft lip repair can help to produce reliable and favorable results across all cleft types, with little change in outcome with experience. Although it requires many landmarks, most of these are standard anthropometric points that are used for analysis of the cleft deformity. Long-term outcomes need to be assessed. Raymond Tse, M.D. Seattle Children’s Hospital 4800 Sand Point Way NE M/S OB.9.527 Seattle, Wash. 98105 [email protected]
acknowledgments
Special thanks to Joseph Gruss, M.D., and Clinton Morrison, M.D., who provided independent, subjective ratings and to Babette Saltzman, M.D., for statistical support. Thanks also to David Fisher, M.D., for mentorship, guidance, and feedback. patient consent
Parents or guardians provided written consent for the use of patients’ images. references 1. Gillies HD, Millard DR. The Principles and Art of Plastic Surgery. Boston: Little, Brown; 1957. 2. Tse R. Unilateral cleft lip: Principles and practice of surgical management. Semin Plast Surg. 2012;26:145–155. 3. Burget GC, Menick FJ. The subunit principle in nasal reconstruction. Plast Reconstr Surg. 1985;76:239–247. 4. Gonzalez-Ulloa M, Castillo A, Stevens E, Alvarez Fuertes G, Leonelli F, Ubaldo F. Preliminary study of the total restoration of the facial skin. Plast Reconstr Surg (1946) 1954;13:151–161. 5. Millard DR Jr. A radical rotation in single harelip. Am J Surg. 1958;95:318–322. 6. Mohler LR. Unilateral cleft lip repair. Plast Reconstr Surg. 1987;80:511–517. 7. Stal S, Brown RH, Higuera S, et al. Fifty years of the Millard rotation-advancement: Looking back and moving forward. Plast Reconstr Surg. 2009;123:1364–1377. 8. Noordhoff MS. The Surgical Technique for the Unilateral Cleft LipNasal Deformity. Taipei: Noordhoff Craniofacial Foundation; 1997. 9. Holtmann B, Wray RC. A randomized comparison of triangular and rotation-advancement unilateral cleft lip repairs. Plast Reconstr Surg. 1983;71:172–179. 10. Lazarus DD, Hudson DA, van Zyl JE, Fleming AN, Fernandes D. Repair of unilateral cleft lip: A comparison of five techniques. Ann Plast Surg. 1998;41:587–594.
11. Joss G, Rouillard LM. A critical evaluation of the rotationadvancement (Millard) method for unilateral cleft lip repair. Br J Plast Surg. 1962;15:349–361. 12. Demke JC, Tatum SA. Analysis and evolution of rotation principles in unilateral cleft lip repair. J Plast Reconstr Aesthet Surg. 2011;64:313–318. 13. Meyer E, Seyfer A. Cleft lip repair: Technical refinements for the wide cleft. Craniomaxillofac Trauma Reconstr. 2010;3:81–86. 14. Onizuka T, Ichinose M, Hosaka Y, Usui Y, Jinnai T. The contour lines of the upper lip and a revised method of cleft lip repair. Ann Plast Surg. 1991;27:238–252. 15. Fisher DM. Unilateral cleft lip repair: An anatomical subunit approximation technique. Plast Reconstr Surg. 2005;116:61–71. 16. Sitzman TJ, Girotto JA, Marcus JR. Current surgical practices in cleft care: Unilateral cleft lip repair. Plast Reconstr Surg. 2008;121:261e–270e. 17. Noordhoff MS. Reconstruction of vermilion in unilateral and bilateral cleft lips. Plast Reconstr Surg. 1984;73:52–61. 18. Sitzman TJ, Fisher DM. Presurgical unilateral cleft lip anthropometrics: Incidence of vermilion height asymmetry. Plast Reconstr Surg. 2013;131:935e–937e. 19. Boorer CJ, Cho DC, Vijayasekaran VS, Fisher DM. Presurgical unilateral cleft lip anthropometrics: Implications for the choice of repair technique. Plast Reconstr Surg. 2011;127:774–780. 20. Vecchione TR. Design for unilateral cleft lip repair to prevent a long lip. Plast Reconstr Surg. 1978;62:604–605. 21. Millard DR. Cleft Craft: The Evolution of Its Surgery. Vol. 1: The Unilateral Deformity. Boston: Little, Brown; 1976. 22. Ridgway EB, Andrews BT, Labrie RA, Padwa BL, Mulliken JB. Positioning the caudal septum during primary repair of unilateral cleft lip. J Craniofac Surg. 2011;22:1219–1224. 23. Anderl H, Hussl H, Ninkovic M. Primary simultaneous lip and nose repair in the unilateral cleft lip and palate. Plast Reconstr Surg. 2008;121:959–970. 24. Smahel Z, Müllerová Z, Nejedlý A. Effect of primary repositioning of the nasal septum on facial growth in unilateral cleft lip and palate. Cleft Palate Craniofac J. 1999;36:310–313. 25. Tvrdek M, Hrivnáková J, Kuderová J, Smahel Z, Borský J. Influence of primary septal cartilage reposition on development of the nose in UCLP. Acta Chir Plast. 1997;39:113–116. 26. Mercado A, Russell K, Hathaway R, et al. The Americleft study: An inter-center study of treatment outcomes for patients with unilateral cleft lip and palate part 4. Nasolabial aesthetics. Cleft Palate Craniofac J. 2011;48:259–264. 27. Heike CL, Upson K, Stuhaug E, Weinberg SM. 3D digital stereophotogrammetry: A practical guide to facial image acquisition. Head Face Med. 2010;6:18. 28. Tse R, Booth L, Keys K, et al. Reliability of nasolabial anthropometric measures using three-dimensional stereophotogrammetry in infants with unrepaired unilateral cleft lip. Plast Reconstr Surg. 2014;133:530e–542e. 29. Mulliken JB, LaBrie RA. Fourth-dimensional changes in nasolabial dimensions following rotation-advancement repair of unilateral cleft lip. Plast Reconstr Surg. 2012;129:491–498. 30. Fisher DM, Tse R, Marcus JR. Objective measurements for grading the primary unilateral cleft lip nasal deformity. Plast Reconstr Surg. 2008;122:874–880. 31. He X, Li H, Shao Y, Shi B. Objective measurements for grading the nasal esthetics on basal view in individuals with secondary cleft nasal deformity. Cleft Palate Craniofac J. 2014;51:66–69. 32. Meltzer NE, Vaidya D, Capone RB. The cleft-columellar angle: A useful variable to describe the unilateral cleft
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Plastic and Reconstructive Surgery • July 2015 lip-associated nasal deformity. Cleft Palate Craniofac J. 2013;50:82–87. 33. Asher-McDade C, Roberts C, Shaw WC, Gallager C. Development of a method for rating nasolabial appearance in patients with clefts of the lip and palate. Cleft Palate Craniofac J. 1991;28:385–390; discussion 390. 34. Kuijpers-Jagtman AM, Nollet PJ, Semb G, Bronkhorst EM, Shaw WC, Katsaros C. Reference photographs for nasolabial appearance rating in unilateral cleft lip and palate. J Craniofac Surg. 2009;20(Suppl 2):1683–1686. 35. Yuzuriha S, Mulliken JB. Minor-form, microform, and mini-microform cleft lip: Anatomical features, operative techniques, and revisions. Plast Reconstr Surg. 2008;122:1485–1493. 36. Noordhoff MS, Cheng WS. Median facial dysgenesis in cleft lip and palate. Ann Plast Surg. 1982;8:83–92. 37. Liao YF, Numhom S, Lo LJ, Noordhoff MS. Craniofacial and dental dysmorphology in patients with median facial dysplasia: Long-term follow-up. Int J Oral Maxillofac Surg. 2011;40:672–678. 38. Noordhoff MS, Huang CS, Lo LJ. Median facial dysplasia in unilateral and bilateral cleft lip and palate: A subgroup of median cerebrofacial malformations. Plast Reconstr Surg. 1993;91:996–1005; discussion 1006. 39. Mulliken JB, Burvin R, Padwa BL. Binderoid complete cleft lip/palate. Plast Reconstr Surg. 2003;111:1000–1010. 40. Cutting CB, Dayan JH. Lip height and lip width after extended Mohler unilateral cleft lip repair. Plast Reconstr Surg. 2003;111:17–23; discussion 24. 41. Losee JE, Selber JC, Arkoulakis N, Serletti JM. The cleft lateral lip element: Do traditional markings result in secondary deformities? Ann Plast Surg. 2003;50:594–600. 42. Rappaport B, Mellon RD, Simone A, Woodcock J. Defining safe use of anesthesia in children. N Engl J Med. 2011;364:1387–1390. 43. LeMesurier AB. Hare-Lips and Their Treatment. Baltimore: Williams & Wilkins; 1962. 44. Rossell-Perry P, Gavino-Gutierrez AM. Upper double-rotation advancement method for unilateral cleft lip repair of severe forms. J Craniofac Surg. 2011;22:2036–2042. 45. Chait L, Kadwa A, Potgieter A, Christofides E. The ultimate straight line repair for unilateral cleft lips. J Plast Reconstr Aesthet Surg. 2009;62:50–55.
46. Nakajima T, Tamada I, Miyamoto J, Nagasao T, Hikosaka M. Straight line repair of unilateral cleft lip: New operative method based on 25 years experience. J Plast Reconstr Aesthet Surg. 2008;61:870–878. 47. Randall P. A triangular flap operation for the primary repair of unilateral clefts of the lip. Plast Reconstr Surg Transplant Bull. 1959;23:331–347. 48. Salyer KE, Xu H, Genecov ER. Unilateral cleft lip and nose repair; closed approach Dallas protocol completed patients. J Craniofac Surg. 2009;20(Suppl 2):1939–1955. 49. Mulliken JB, Martínez-Pérez D. The principle of rotation advancement for repair of unilateral complete cleft lip and nasal deformity: Technical variations and analysis of results. Plast Reconstr Surg. 1999;104:1247–1260. 50. Xing H, Bing S, Kamdar M, et al. Changes in lip 1 year after modified Millard repair. Int J Oral Maxillofac Surg. 2008;37:117–122. 51. Lee TJ. Upper lip measurements at the time of surgery and follow-up after modified rotation-advancement flap repair in unilateral cleft lip patients. Plast Reconstr Surg. 1999;104:911–915. 52. Saunders DE, Malek A, Karandy E. Growth of the cleft lip following a triangular flap repair. Plast Reconstr Surg. 1986;77:227–238. 53. Brusati R, Mannucci N, Biglioli F, Di Francesco A. Analysis on photographs of the growth of the cleft lip following a rotation-advancement flap repair: Preliminary report. J Craniomaxillofac Surg. 1996;24:140–144. 54. Grasseschi MF. Minimal scar repair of unilateral cleft lip. Plast Reconstr Surg. 2010;125:620–628. 55. McComb HK, Coghlan BA. Primary repair of the unilateral cleft lip nose: Completion of a longitudinal study. Cleft Palate Craniofac J. 1996;33:23–30; discussion 30. 56. Boo-Chai K. Primary repair of the unilateral cleft lip nose in the Oriental: A 20-year follow-up. Plast Reconstr Surg. 1987;80:185–194. 57. Tindlund RS, Holmefjord A, Eriksson JC, Johnson GE, Vindenes H. Interdisciplinary evaluation of consecutive patients with unilateral cleft lip and palate at age 6, 15, and 25 years. J Craniofac Surg. 2009;20(Suppl 2):1687–1698. 58. Power SM, Matic DB. Critical analysis of consecutive unilateral cleft lip repairs: Determining ideal sample size. Cleft Palate Craniofac J. 2013;50:144–149.
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Background: The anatomical subunit approximation for unilateral cleft lip repair has gained acceptance; however, outcomes have not been reported since the original description. The purpose of this article is to report the experience using this technique. Methods: One hundred two consecutive patients underwent primary cleft lip repair performed by a single surgeon over a 3-year period. Objective analysis involved anthropometric measurements performed on preoperative and postoperative three-dimensional images. Subjective analysis involved Asher-McDade scores by blinded independent surgeons. Observational insights were gained by review of surgical records. Anthropometric measurements were expressed as ratios of the cleft side to the noncleft side. Differences in ratios were assessed by using the rank sum test. Differences in Asher-McDade scores were assessed using the Mann-Whitney test. Results: Demographic, cleft type, cleft extent, and cleft severity were consistent with our center’s norms. The mean age at surgery was 6 months and the mean inferior triangle used was 1.8 ± 0.9 mm. Anthropometric ratios were significantly improved postoperatively and approached 1, regardless of initial cleft severity. Ten subjects who underwent repair early in the experience were compared with 10 subjects who underwent repair late in the experience. There was no significant difference in postoperative anthropometric measures or Asher-McDade scores. Conclusions: The anatomical subunit approximation for unilateral cleft lip repair in a single-surgeon series can achieve improvements in anthropometric measures and early favorable postoperative form. The technique could be applied to all cleft types and there was little change in outcome with greater surgeon experience. Long-term follow-up is necessary. (Plast. Reconstr. Surg. 136: 119, 2015.) CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.
P
rinciples of any reconstruction include a detailed analysis of form, a surgical plan, wide release, and component reconstruction.1 Surgical markings for unilateral cleft lip repair are one component of repair and constitute the plan that helps to achieve balanced nasolabial form and a favorably positioned scar.2 The ideal technique should minimize variations From Seattle Children’s Hospital, University of Washington. Received for publication October 21, 2014; accepted J anuary 27, 2015. Presented at the 71st Annual Meeting of the American Cleft Palate-Craniofacial Association, in Indianapolis, Indiana, March 24 through 29, 2014. Copyright © 2015 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000001369
resulting from surgeon experience and ability, be applicable to a wide spectrum of severity, and Disclosure: The authors have no financial interest to declare in relation to the content of this article. No external funding was received. Supplemental digital content is available for this article. Direct URL citations appear in the text; simply type the URL address into any Web browser to access this content. Clickable links to the material are provided in the HTML text of this article on the Journal’s Web site (www. PRSJournal.com).
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Plastic and Reconstructive Surgery • July 2015 respect the principle of placing scars along anatomical subunits.3,4 Millard’s rotation advancement5 and its modifications6–8 involve a “cut-as-you-go” approach that can be prone to surgeon ability and experience and has been criticized for producing short lips when used for wide clefts.9–11 The Tennison- Randall repair is more geometric and is thought to be easier to learn12 and more suitable for wide clefts13; however, the resultant scar inevitably crosses natural contours.14 The Anatomical Subunit Approximation for unilateral cleft lip repair was described in 200515 and has been characterized as a “hybrid” technique.12 The design includes multiple landmarks with the intention of approximating incisions along the borders of anatomical subunits.15 Although the technique has gained popularity,16 the experience using this approach has yet to be described by other surgeons. The purpose of this study is to report the early experience, modifications, and results using the anatomical subunit approximation technique for unilateral cleft lip repair performed by a single surgeon.
PATIENTS AND METHODS Approval for this study was obtained from the institutional review board. Consecutive patients who underwent primary unilateral cleft lip repair performed by the senior author (R.T.) from 2010 to 2013 were included in this study. Cleft Lip Repair Design/Lip Markings Landmarks for cleft lip repair follow those described for the Anatomical Subunit Approximation technique.15 Most are standard anthropometric landmarks (Fig. 1, above, left) and are described here using anthropometric terminology rather than assigned numbers. Marks are placed starting at the midline, identifying the noncleft side, and then, using the known landmarks, to define the less distinct cleft side landmarks. The full stepwise sequence of landmarks is illustrated in the slide presentation. (See Figure, Supplemental Digital Content 1, which shows the sequence of landmarks for the anatomical subunit approximation for unilateral cleft lip repair, http://links.lww.com/ PRS/B332.) Lip points. The subnasale (sn) and the top of the philtral columns [crista philtri superioris, (cphs and cphs′)] are found along the nasolabial crease. The Cupid’s bow is identified along the vermilion
border and is defined by the two peaks [crista philtri inferioris (cphi and cphi′)] and its low point [labiale superioris (ls)]. Given that transitions in contours and concavities hide scars better,3 the philtral column points, cphi and chps, are identified along the medial border of the philtral ridge, where the convexity of the column transitions to the concavity of the philtral dimple (Fig. 1, above, left). The lateral point of lip closure, cphiʺ, is identified based on tissue adequacy according to Noordhoff and is defined as the point along the vermilion border at which the vermilion is the widest.17 Medial to this point, the white roll becomes less distinct and the vermilion becomes deficient. Once all of the landmarks along the vermilion have been identified, corresponding points above the white roll (black dots with white outline in Fig. 1, center, left) and along the mucosa-vermilion border (black dots with red outline in Fig. 1, center, left) can be identified (perpendicular to the lip). Nose points. The subalare (sbal), the lowest point along the ala, is identified along the nose-lip crease. The alar insertion point (ai) was not part of Fisher’s initial description and is found at the intersection of the alar rim with the nasal sill, along the noselip crease (Fig. 1, above, left). These landmarks are used to determine the nasal floor closure needed to correct the nasal deformity. Contrary to the lip, nose landmarks are identified with the deformity manually corrected so that the orientation of sbal-ai on both sides match (Fig. 1, above, right). On the noncleft side, an arbitrary point (x) is defined based on the intersection of the linear projections from sbal-ai and sn-cphs (i.e., sbal-aix and sn-cphs-x should both form straight lines). The corresponding arbitrary points (x′ and xʺ) are then defined on the cleft side using distances from sn-cphs′ and sbal′-ai′ that mirror the corresponding distances from the noncleft side. When x′ and xʺ are brought together (i.e., to mimic x), the nasal deformity should be corrected (Fig. 1, above, right). When the nasal floor is intact, appropriate nasal floor excision can be further verified by measuring nostril circumferences using urethral sounds (circumference = diameter × π). The distance between x′ and xʺ should not exceed the difference in circumferences to avoid producing a micronostril. Medial markings: connect the dots. The medial lip markings simply connect the landmarks along the nasal sill (x′-cphs′); along the philtral column (cphs′-cphi′); and across the white roll, vermilion border, and red line at cphi′
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Volume 136, Number 1 • Anatomical Subunit Approximation
Fig. 1. Lip markings. (Above, left) Standard anthropometric landmarks. Alar insertion point (ai) is the intersection of the nostril rim and the nasal sill along the alar crease. (Above, right) Nose landmarks need to be identified with the nasal deformity corrected. Point x is an arbitrary point that is collinear with both sbal-ai and sn-cphs. The points x ’ and x" are measured off of sn’-cphs’ and sbal’-ai’, respectively, by mirroring distances on the noncleft side. When x ’ and x" are brought together, they should be the equivalent of x and the nasal deformity should then be corrected. (Center, left) The medial lip markings connect the dots along the border of anatomical subunits with back-cuts above the white roll and along the mucovermilion border. The medial vermilion height is transposed to the vermilion at cphi" and the remaining vermilion is used to augment the deficient medial vermilion. The cphi" remains fixed and the remaining lateral lip markings fit the space from above the white roll to the lateral point of nasal closure, x". (Center, right) An equilateral triangle is used to augment medial lip height. The base width, C, is calculated based on the difference in philtral heights, minus 1 mm, given the RoseThomson effect (lengthening with diamond-shaped excision and closure). The limbs of the lateral lip markings need to match the medial lip and can articulate open or closed. The angle between limbs s and B should match the medial side as much as possible. (Below) Variations in lateral lip design based on lateral lip height.
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Plastic and Reconstructive Surgery • July 2015 (Fig. 1, center, left). This last incision is made perpendicular to the lip so that its incidence with cphs′-cphi′ forms an angle that will open following incision (Rose-Thompson effect). A small backcut is designed above the white roll and is oriented to provide maximal vertical release. The length of the backcut is based on the size of the calculated equilateral triangle from the lateral lip that will be needed to augment medial lip height. Another incision is designed along the red line so that the deficient vermilion height may be augmented with a lateral lip vermilion flap.17,18 Lateral markings: match the medial marks and fill the deficits. On the lateral lip, the deficient vermilion height at cphi′ is transposed to the vermilion height at cphiʺ so that the excess lateral vermilion height can be used as a triangle to augment medial height (black dots with red outline in Fig. 1, center, left). The incision across the white roll connects cphiʺ and its corresponding point above the white roll. The remaining lateral lip markings then need to fit between this last point and the lateral point of nasal closure, xʺ (Fig. 1, center, left). Limbs that match the medial lip incisions (s, along the nasal sill; B, the cleft side philtral height), with the addition of a small triangle to augment medial lip height, need to be drawn and fit into the fixed space (Fig. 1, center, right). The triangle base width, C, is the difference in philtral heights (cphi-cphs versus cphi′-cphs′) minus 1 mm because of the Rose-Thompson effect (i.e., C = A − B − 1 mm). The lateral lip components can articulate open or close to fit the lateral lip height. An arc with radius, C, can be drawn from above the white roll point at cphiʺ and an arc with radius, s, can be drawn from xʺ. The cleft side philtral height (B) is then placed to connect the two arcs (Fig. 1, center, right). In contrast to other techniques, the cphiiʺ is fixed and is not shifted lateral in the case of vertical lip deficiency.19 When the lateral lip is short (i.e., complete clefts), the components need to fit into a smaller space and angles between incisions are made more acute (Fig. 1, below, left). In the case of greater lateral lip height (i.e., incomplete clefts), the components can fit into a larger space and the angles between components can be made more obtuse (Fig. 1, below, center). In the case of vertical lip excess (i.e., minor incomplete clefts), angles between lateral lip components should not be made so obtuse that they no longer match the medial lip incisions. In this case, a vertical wedge excision20 along the alar crease should be used to
effectively reduce the vertical height (Fig. 1, below, right). Remeasure and Cut as You Go The medial lip is incised and dissected first to verify that the backcut above the white roll is needed. The triangular base width (C) can also be verified with Cupid’s bow manually leveled. The designed points of nasal floor closure can be verified by manually correcting the nasal deformity after release of the upper sulcus and piriform aperture. Markings rarely need to be modified. At final closure, the lateral triangle above the white roll can be altered if needed. Release and Component Reconstruction Release and component reconstruction are equally as important as the design and incisions and have been previously described in more detail.2 The orbicularis muscle is released from the base of the columella at the level of the alveolus to allow the medial lip to drop. The orbicularis on the lateral side is dissected as a single layer between mucosa and skin and is released from the alveolus and alar base. This results in an empty triangle of muscle along the nose-cheek junction so that when the muscle is advanced medially, the alar crease is accentuated. Whereas the lateral muscle is dissected widely, dissection on the medial side is conservative to preserve the philtral dimple. An upper buccal sulcus incision and supraperiosteal dissection over the maxilla allow mobilization of the lateral lip as needed. The nose is released along the piriform aperture and can involve a submucosal dissection or transmucosal release. The nasal floor is fully closed to the level of the incisive foramen and the nasal sidewall is reconstructed with a turbinate flap,8,15 L-flap,21 or lateral nasal wall advancement.2 These have been previously described in further detail2 and are summarized in Figure 2. Each of these flaps provide mucoperiosteum to fill the dead space left by mucoperiosteal release of the lateral nasal wall with anterior repositioning of the ala. These flaps are incorporated into the closure of the nasal floor. A primary perichondrial-sparing septoplasty is performed to reposition the caudal septum into the midline of the face.22–25 Sutures are used to ensure the new septal position remains stable. No nasal tip dissection is performed. The orbicularis muscle is brought across the nasal floor/sill and inserted into the base of the columella to maintain the lengthened medial lip. The muscle is carefully reapproximated and the J shape of the pars marginalis is preserved, as it contributes to the pout of the lip.
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Volume 136, Number 1 • Anatomical Subunit Approximation
Fig. 2. Lateral nasal wall and nasal floor reconstruction. Options for lateral nasal wall reconstruction were based on cleft type and available exposure. (Left) Turbinate flap. Mucoperiosteum based anteriorly is transposed 90 degrees to fill the defect following alar base release and anterior repositioning. This requires an open cleft to allow posterior incision/release. (Center) L-flap. Marginal tissue is based on alveolar mucoperiosteum and transposed 90 degrees to fill the defect. (Right) Lateral nasal wall flap. The lateral nasal wall is raised and a backcut is made posteriorly to allow the ala to advance anteriorly. The donor site is made along the bony wall. (Reproduced with permission from Tse R. Unilateral cleft lip: Principles and practice of surgical management. Semin Plast Surg. 2012;26:145–155.)
Final Closure Skin flaps are inset using deep dermal sutures. For the first half of this case series, final skin closure involved 7-0 polypropylene sutures in a simple interrupted manner requiring removal under general anesthetic 1 week postoperatively. For the second half of this case series, final skin closure was modified so that suture removal could be achieved in the clinic without the use of anesthetic
or sedation. This involved a 6-0 polypropylene running intracuticular suture along the vertical limb of the philtral column (B) and 8-0 polyglactin in a simple interrupted manner for everything else. Analysis Demographic and surgical details were obtained by chart review. Previously captured twoand three-dimensional images were collected from
Table 1. Cleft Lip Type, Relative Dimensions, and Triangular Flap Base Width Cleft Type Microform Incomplete Complete plus band† Complete All types
No. 3 47 12 40 102
A, Total Lip Height (cphi-cphs) (mm)*
B, Greater Lip Height (cphi′-cphs′) (mm)*
7.0 (0.8) 7.5 (1.5) 7.2 (1.3) 8.3 (1.3) 7.8 (1.4)
5.8 (0.5) 5.2 (1.5) 4.0 (0.8) 5.0 (0.9) 5.0 (1.3)
C, Lesser Lip Height (triangle base width) (mm)* 0 (0) 1.4 (0.8) 2.1 (0.6) 2.2 (0.7) 1.8 (0.9)
*Values are expressed as means (SD). †A band is any soft-tissue connection of lip or nasal sill in the presence of a complete cleft alveolus (previously known as Simonart band).
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Plastic and Reconstructive Surgery • July 2015 our center’s archives for analysis. Standardized two-dimensional photographs were captured by a professional imaging technologist and included frontal, worm’s-eye, and lateral profile views that were consistent with Americleft guidelines.26 Three-dimensional images were captured using the 3dMDCranial system (3dMD, Atlanta, Ga.) according to a previously described protocol.27 Anthropometric measurements were performed using 3dMD Vultus v.2.2.011 on threedimensional images that were captured at the immediate preoperative and postoperative clinic visits. The system of quantitative morphometry has been previously described28 and is consistent with other reports.29 We focused on measurements that have been found to be reliable on three-dimensional imaging.28 These included medial lip height (sn-chpi), lateral lip height (sa-chpi), lateral lip length (ch-cphi), columellar angle, and nostril width. The columellar angle is measured from the submental view and is defined as the angle made by the columella relative to the sagittal plane of the face.30 It has previously been found to be one of the most important objective measures of preoperative cleft lip nasal severity.30–32 To characterize nasolabial balance, lip and nose measurements were expressed as ratios of cleft to noncleft side measurements. Ratios approaching 1 would suggest greater symmetry. We obtained more global assessments of nasolabial form using two-dimensional images that were captured at the immediate postoperative clinic visit. We followed methods described by Asher-McDade et al.33 and used by Americleft.26 Two-dimensional photographs were cropped to exclude features outside of the nasolabial region.33 Two groups of 10 consecutive cases were analyzed so that changes in outcome over this experience could be identified. The first group was early in the surgeon’s experience (2010) and the second group was later in the surgeon’s experience (2013). Three reviewers (Joseph Gruss, craniofacial surgeon with 30 years of cleft experience; Clinton Morrison, craniofacial fellow; and Samuel Lien, resident), who were blind to the groups, rated outcomes using reference photographs.34 To provide clinical examples, families of the middle 20 consecutive cases (in which 1-year follow-up was available) were asked for permission to publish their child’s photographs and cleft characteristics. All of the families consented. Statistical Analysis Outcomes of the two groups of patients at the beginning and end of the experience were analyzed.
Anthropometric ratios were compared using a rank sum test after all subjects were ranked according to their distance from the goal ratio of 1. Asher-McDade scores were compared using a Mann-Whitney test.
RESULTS Demographics These cases were performed during the first few years of the primary surgeon’s practice as a full-time pediatric plastic surgeon. One hundred two consecutive patients were included, of which 53 were male patients and 49 were female patients. Cleft extent was as follows: cleft lip, n = 41 (40 percent); cleft lip and alveolus, n = 14 (14 percent); and cleft lip and palate, n = 47 (46 percent). The distribution of cleft lip type is detailed in Table 1. Five patients had associated anomalies/ syndromes, including midline facial deficiency, craniofacial microsomia, popliteal pterygium, congenital constriction band, and undetermined chromosome abnormality. Median age at surgery was 6 months (range, 3 to 45 months). Reasons for later age at surgery included microform cleft, late presentation, and international adoption. Lip Design Relative lip heights are detailed in Table 1. The mean triangle base width, C, was 1.8 mm. The triangle tended to be smaller for less severe clefts and larger for more severe clefts. Anthropometric Measurements The three-dimensional images for 89 of the 102 patients were available for full analysis (six missing preoperatively, five missing postoperatively, and two missing both). Linear lip and nose dimensions were corrected and approached 1 postoperatively (Tables 2 and 3). Although these ratios varied with cleft lip type, the postoperative ratios were similar regardless of the initial cleft. Mean columellar angle was corrected to within 10 degrees, regardless of the cleft type and the preoperative columellar angle. Asher-McDade Scores Interrater reliability assessed by Pearson coefficients ranged from 0.3 to 0.5 between the three raters. Ratings by the three raters for the two groups are displayed in Figure 3, below. Early versus Late Experience There were no significant differences in anthropometric measurements (not displayed) and AsherMcDade scores for the two groups (Fig. 3).
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Volume 136, Number 1 • Anatomical Subunit Approximation severity. Given that the surgical design is based on anatomical landmarks, rather than dimensions, the lip markings inherently adjust for different cleft types. We found little change in outcome over time with greater surgeon experience. Anthropometric measures and subjective ratings were similar early and late in this series. In contrast to the freehand, cut-as-you-go style of rotation-advancement, Fisher’s original description included 25 numbered landmarks and far more initial measurements than any other approach.15 Although these may appear cumbersome, they are based on standard anthropometric landmarks (Fig. 1, above, left) that experienced cleft surgeons will identify or at least recognize when analyzing a cleft deformity. Understanding the system of landmarks requires considerable effort; however, the time spent may help to reduce the steepness of the learning curve. A technique should match a surgeon’s style and personality. The senior surgeon feels that the extra time spent on design allows incisions to be made with little hesitation, giving this technique a “measure-twice, cut-once” flavor. This may be helpful for less experienced surgeons and resident training. More experienced surgeons who consistently produce favorable results may have no need to adopt a new technique; however, for
Example Cases Cleft severity, as indicated by columellar angle,30 for the middle 20 subjects, was similar in distribution to the entire case series (Fig. 4). Ten of these subjects, spanning the spectrum of severity, are displayed in Figure. 5, and the full set of 20 subjects is displayed in Figure, Supplemental Digital Content 2 (http://links.lww.com/ PRS/B333). Subjects are arranged by cleft type and increasing columellar angle. One patient with an incomplete cleft lip had a contralateral mini-microform cleft lip35 that was left unaddressed. As a result, he has a minor whistle notch deformity caused by prolabial deficiency. One patient with complete cleft lip underwent nasoalveolar molding, whereas the others did not. The final patient had a midline deficiency with absent premaxilla consistent with what has been described as median facial dysgenesis/ dysplasia/hypoplasia36–38 or binderoid cleft lip.39 The caudal septum was absent, thereby limiting changes produced by septal repositioning.
DISCUSSION We found the Anatomical Subunit Approximation to be applicable across the spectrum of cleft severity. Anthropometric measures of asymmetry were corrected regardless of cleft type and
Table 2. Preoperative and Postoperative Lip Measurements/Ratios Medial Lip Height Ratio* Cleft Type Microform Incomplete Complete plus band† Complete All types
Lateral Lip Height Ratio*
Lateral Lip Length Ratio*
Preop
Postop
Preop
Postop
Preop
Postop
0.94 (0.04) 0.62 (0.12)
1.08 (0.09) 1.09 (0.12)
0.84 (0.12) 0.84 (0.12)
0.73 (0.11) 0.93 (0.07)
0.93 (0.12) 0.88 (0.09)
0.90 (0.00) 0.92 (0.10)
0.62 (0.19) 0.53 (0.12) 0.59 (0.15)
1.21 (0.21) 1.05 (0.11) 1.10 (0.15)
0.74 (0.16) 0.71 (0.13) 0.77 (0.14)
0.89 (0.10) 0.90 (0.08) 0.91 (0.09)
0.88 (0.10) 0.88 (0.09) 0.88 (0.08)
0.93 (0.10) 0.93 (0.09) 0.93 (0.09)
Preop, preoperative; Postop, postoperative. *Values are expressed as ratios of cleft to noncleft side (SD). †A band is defined as any soft-tissue connection of lip or nasal sill in the presence of a complete cleft alveolus (previously known as Simonart band).
Table 3. Preoperative and Postoperative Nose Measurements/Ratios Columellar Angle* Cleft Type Microform Incomplete Complete plus band† Complete All types
Nostril Width Ratio*
Preop
Postop
Preop
Postop
15.29 (0.73) 17.47 (7.88) 22.27 (9.52) 37.02 (11.64) 26.22 (13.43)
9.19 (1.92) 8.52 (5.22) 7.51 (5.12) 8.98 (5.28) 8.58 (5.20)
1.41 (0.22) 1.42 (0.25) 1.66 (0.28) 2.30 (0.43) 1.82 (0.54)
1.15 (0.08) 1.10 (0.15) 1.02 (0.14) 1.17 (0.24) 1.12 (0.20)
Preop, preoperative; Postop, postoperative. *Values are expressed as ratios of cleft to noncleft side (SD). †A band is defined as any soft-tissue connection of lip or nasal sill in the presence of a complete cleft alveolus (previously known as Simonart band).
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Plastic and Reconstructive Surgery • July 2015
Fig. 3. Asher-McDade scores for 10 subjects early in the experience and 10 subjects late in the experience. (Above) Distribution of scores. (Below) Mean scores.
some, the approach may provide a foundation of measurements from which results can be refined. There are several concepts used in this technique that are important to highlight. First, it uses a diamond-shaped excision to lengthen the lip (Rose-Thomson effect) and to minimize the size of the triangle needed to level Cupid’s bow. This allows the scar to be hidden behind the convexity of the white roll. Second, it uses a fixed cphiʺ that is selected based on tissue adequacy rather than on measured distances from the commissures21 or needed lateral lip height.29,40 The former21 can incorporate deficient tissues
into the repair,41 and the latter29,40 can sacrifice lateral lip length.19 Although measured ch-cphi increases with time following rotation advancement,29,40 preservation of lateral lip length may be important to avoid the accordion effect of tissue advancement that reduces the natural fullness of the lip. Third, in contrast to rotationadvancement, the alar base is incorporated into the design of the lip repair. This avoids additional scar along the alar base. Although lip markings are important for surgical planning, wide surgical release and component reconstruction are equally important in
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Volume 136, Number 1 • Anatomical Subunit Approximation
Fig. 4. Severity of cleft lip nasal deformity as measured by columellar angle for the entire series and for the middle 20 subjects presented as clinical examples.
producing favorable outcomes. Given that the underlying bony deformity will not be corrected until later in life, emphasis was placed on release, repositioning, and retention of the nasal foundation in three-dimensional space. Both the caudal septum22–25 and alar base were aggressively repositioned. Focus was placed on reconstruction of the nasal floor and lateral nasal wall to retain the basal nose components in space.2 Through the course of this series, muscle dissection evolved to be more aggressive, emphasizing the creation of an empty triangle of muscle lateral to the alar base. This dissection accentuates the alar crease when the muscle is repaired and may add to soft-tissue stability. Although a difference was not apparent using the outcome measures used at early followup, the additional soft-tissue corrections may be more subtle than can be measured and may contribute to long-term retention of corrected form. Gillies once wrote: “Never do today what can honourably be put off till tomorrow.”1 Nasal tip dissection was not performed in this series to avoid unnecessary scarification. Final skin closure was modified midway through this series to eliminate the need for suture removal under general anesthesia. No change in scar quality was noted; however, formal specific long-term evaluation would help to confirm this. The timing of surgery in this series was 6 months rather than 3 months of age15 to reduce the risks of anesthesia (immediate and long-term42) and to take advantage of larger and more robust anatomy.
Several pearls were noted on review of this series. First, when designing the lateral lip markings, the angle between limbs s and B should approximate that of the medial lip markings. If too obtuse, the desired line of closure along anatomical subunits will be blunted. To avoid this, if there is excess lateral lip height, a triangle should be excised (Fig. 1, below). Second, the nose deformity should be corrected manually when identifying nasal landmarks (Fig. 1, above, right). Final alar base position and rotation rely on the surgical markings. Although the alar insertion point was not included in Fisher’s original description, the additional landmark is particularly helpful for orienting alar rotation. Third, remeasure and modify as you go. The medial lip can be overlengthened. After making the backcut above the white roll on the medial lip, manually level Cupid’s bow and remeasure the base width of the triangle needed to augment medial lip height. The lateral triangle can be reduced before incising the lateral lip. Interpreting the literature on cleft lip repair can be difficult given that outcomes are subjective and many reports display only the best or highly selected outcomes.5,6,43–48 Our study included all patients in a consecutive series. Similar to Mulliken, we used anthropometric analysis29,49 to provide objective measures of specific changes. We also used Asher-McDade scores to provide a more global assessment of outcome.33 Given that raters were blinded to the groups, we could use their scores to compare results early and later in the surgeon’s experience; however,
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Plastic and Reconstructive Surgery • July 2015
Fig. 5. Example clinical cases preoperatively and 1 year postoperatively. The middle 20 subjects who had 1-year follow-up available were selected as clinical examples. Subjects were arranged by cleft type and increasing severity as measured by columellar angle. Ten cases that span the spectrum of severity are displayed here. The complete set of 20 are shown in Supplemental Digital Content 1, http://links.lww.com/PRS/B332.
because the raters were from our institution, comparison of scores to those of other studies should be tempered. We analyzed three-dimensional and twodimensional images captured immediately preoperatively and postoperatively given that our focus was on a technique of surgical planning, the purpose of which is to produce nasolabial balance at the completion of a cleft lip repair. Although previous studies have demonstrated that immediate results are the best predictor of eventual
outcome50,51 and that lip dimensions remain stable over time,9,11,29,40,50–53 longer term follow-up would be useful, and we plan to reassess this series when 4-year follow-up is available for all subjects. In the absence of ideal outcome measures, we have presented example photographs. Previous reports have used 10 consecutive patients to avoid selection bias54–57; however, Power and Matic argue that more subjects are needed.58 As such, we used the middle 20 consecutive cases as representative examples. Our study is one of the most
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Volume 136, Number 1 • Anatomical Subunit Approximation comprehensive reports of early outcome following cleft lip repair.
CONCLUSIONS The anatomical subunit approximation for unilateral cleft lip repair can help to produce reliable and favorable results across all cleft types, with little change in outcome with experience. Although it requires many landmarks, most of these are standard anthropometric points that are used for analysis of the cleft deformity. Long-term outcomes need to be assessed. Raymond Tse, M.D. Seattle Children’s Hospital 4800 Sand Point Way NE M/S OB.9.527 Seattle, Wash. 98105 [email protected]
acknowledgments
Special thanks to Joseph Gruss, M.D., and Clinton Morrison, M.D., who provided independent, subjective ratings and to Babette Saltzman, M.D., for statistical support. Thanks also to David Fisher, M.D., for mentorship, guidance, and feedback. patient consent
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