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Single-stage repair of secondary unilateral cleft lip-nose deformity in adults
Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Single-stage repair of secondary unilateral cleft lip-nose deformity in adults Fan Zhou a, 1, Wen Lin a, 1, Yifei Du a, Sheng Li a, Hongbing Jiang a, Linzhong Wan a, **, Hua Yuan a, b, * a b
Department of Oral and Maxillofacial Surgery, Affiliated Hospital of Stomatology, Nanjing Medical University, Nanjing, China Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, China
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 25 May 2019 Accepted 2 December 2019 Available online xxx
Correction of cleft lip-nose deformity in adult patients is different from that in children. One-stage correction has proved to be a suitable technique for patients with cleft-lip nose deformity. This study aimed to explore a particular single-stage method and evaluate the effect of simultaneous reparation of secondary unilateral cleft lip-nose deformities. Cleft lip patients who had previously undergone nasolabial surgery with residual poor nasal/lip appearance were included. The alveolar bone defect was repaired with granular costal cortical bone. Lip revision and rhinoplasty were performed using diced costal cartilage. The lip, nose, and alveolar deformities were corrected in one stage. From 2011 to 2017, 53 cases were treated. The vermilion discrepancy was corrected in all cases. Fiftyone patients were successfully treated, with primary healing in the bony recipient area. Cancellous bone exposure occurred in two cases. The wounds were healed after debridement and drainage. Appearances were improved in all patients. The mean change in columellaelabial angle ranged from 82.50 to 92.78 (p < 0.001). This one-stage correction appears to have led to a distinct improvement in the nasal tip projection and lip. The method is considered to be effective and reliable in patients with secondary unilateral cleft lip-nose deformities. © 2019 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Unilateral cleft lip Rhinoplasty Deformity Diced cartilage
1. Introduction Cleft lip and palate is one of the most common congenital craniofacial deformities (Tanaka et al., 2012; Chepla et al., 2013). Although a satisfactory lip shape can be restored after primary cleft lip repair, secondary deformities of the cleft lip and nose can occur with development (Jeong et al., 2012; Pawar. et al., 2014). There are several reasons for this, which include poor adherence by children to instructions, flawed surgical procedures, congenital nasal cartilage abnormalities, muscular dystrophy, and
* Corresponding author. Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, 140 Hanzhong Rd., Nanjing, China. Fax: þ86 25 85031914. ** Corresponding author. Department of Oral and Maxillofacial Surgery, Affiliated Hospital of Stomatology, Nanjing Medical University, Nanjing, China. Fax: þ86 25 85031942. E-mail addresses: [email protected] (L. Wan), [email protected] (H. Yuan). 1 Contributed equally to this work.
abnormal development of the jaw bone (Haug, 2004). The incidence of secondary deformities is very high, ranging from 35% to 85% according to previous reports (Henkel et al., 1998). For the treatment of alveolar cleft, cleft lip, and nasal deformities, traditional methods have involved several operations at different stages. In some cases, available tissues are overlooked during the operation and deformities can be aggravated by delayed surgery, affecting the treatment outcome. Recently, onestage correction has become accepted as a treatment for secondary lip-nose deformity. This study describes the authors’ experiences in repairing secondary unilateral cleft lip-nose deformities.
2. Material and methods This study was reviewed by the Institutional Review Board of Nanjing Medical University. The clinical data of 53 patients with
https://doi.org/10.1016/j.jcms.2019.12.004 1010-5182/© 2019 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
2
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
secondary unilateral cleft lip-nose deformities who underwent surgery at the Affiliated Stomatological Hospital of Nanjing Medical University from January 2011 to August 2017 were retrospectively reviewed. Most of the patients were from poor families, and they wanted to improve their appearances without undergoing orthodontic or orthognathic treatment.
cut to support the columella and nasal dorsum. The remaining cartilage was cut into pieces of about 1 mm diameter using a No. 11 blade, and soaked in an antibiotic solution (1 g of kanamycin sulfate diluted in 250e500 ml 0.9% sodium chloride (Fig. 1).
2.1. Rib harvest
The diced cortical bone and cartilage graft were used to close the nasolabial fistula and elevate the alar base. The edges of the mucosa were incised, fully exposing the alveolar bone surface, pyriform aperture margin, and the surrounding bone surface. This was followed by closure of the mucous membrane of the nose floor and palatal side with sutures, forming the base of the bone graft zone. Multiple holes were drilled in the lateral wall of the two alveolar ridges, so that the bone marrow mesenchymal stem cells in the alveolar bone could enter the bone graft area. The diced cortical bone was packed tightly into the prepared bone defect zone, and the bone height of the graft zone was made slightly higher than normal d by 1e2 mm d in order to close the nasolabial fistula and elevate the alar base. The soft tissue incision was closed after the bone graft was completed.
Cartilage and cortical bone were harvested from the rib (Bergeron et al., 2009). The latter was prepared for alveolar bone graft. Firstly, the intersected costal cartilage was cut into two small pieces, 25 mm long and 8 mm wide. The two cartilages were then
2.2. Alveolar bone graft
2.3. Lip revision The planned incision for lip revision was drawn along the original scar using methylene blue (Fig. 2). Sometimes the scar tissue could also be used for repairing the nasal vestibular area in soft tissue defects or to extend the affected nasal columella. We dissected and reset the orbicularis oris muscle, along the labial bow
Fig. 1. (A) The harvested costochondral graft. (B) Two pieces of cartilage were cut to prepare for columella and nasal dorsum support. (C) The rest of the cartilage was cut into pieces of less than 1 mm.
Fig. 2. (A) Alar base deformity. (B) Lip splitting and muscle repositioning to correct the alar base.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
and the wet-dry border of the lip. The muscles were then reduced and the position of the base of the nasal alar was corrected. 2.4. Rhinoplasty Finally, reconstruction of the nasal shape was performed using the costal cartilage graft. A horizontal incision was made along the lower part of the nasal column to expose the nasal alar cartilage, forming a subperiosteal bag and creating a tunnel for the columella support insert. A piece of cartilage (2.5 cm) was then inserted into the columella and anterior nasal spine, and another into the nasal dorsum (Fig. 3a). The cartilage was carved to taper from front to back, with a groove carved into the front face, and was connected to the top of the supporting cartilage of the nasal columella. A
3
truncated 1 ml syringe was then used to inject the cartilage fragments into the nasal dorsum in order to allow correction of nose length and projection of the columella (Fig. 3b). This was followed by suturing of the columella and lower lateral cartilage. The results of the correction are shown in Fig. 4. The graft could then be externally molded to straighten and lengthen the nose. A standard protective mould was then placed over the nasal dorsum. Stitches were removed on day 7 postoperatively. Patients were instructed to massage the nose for 3e5 min, six times a day, for 1e2 months to reduce swelling and adjust the nasal shape if necessary. 2.5. Comparative measurements Standard facial photographs were taken during the preoperative and follow-up periods. These images were used to measure the alar baseenasal tipecolumellar base (ATC) angle, with the apex at the nasal tip (Fig. 5A, B), and the columellaelabial angle d the curved junction of the columella with the upper lip (Fig. 5C, D). These measurements were performed by three independent reviewers and were compared as mean values. 2.6. Investigation of postoperative patient satisfaction There is no international standard regarding classification of the severity of secondary nasal deformity after cleft lip surgery, or a unified standard of satisfaction scores for patients with nasal deformity. Based on the latest pre- and postoperative clinical images, two observers evaluated the degree of patient satisfaction with their surgical outcome according to the five-point global aesthetic
Fig. 3. (A) Placement of one carved cartilage in the columella and another in the nasal dorsum. (B, C) Placement of cartilage fragments into the nasal dorsum.
Fig. 4. (A, B) Results of correction.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
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F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Fig. 5. Patient preoperatively (A) and postoperatively (B), showing the alar baseenasal tipecolumellar base angle. Lateral view of patient preoperatively (C) and postoperatively (D) to show the columellaelabial angle.
improvement scale (GAIS) (Haug, 2004). For each postoperative outcome, the parents (or guardians) gave scores of 5 points for ‘very much improved’, 4 points for ‘much improved’, 3 points for ‘improved’, 2 points for ‘not changed’, and 1 point for ‘worsened’. Four assessors, including parents of the children, a lay person, and a senior cleft surgeon, assessed the repairs separately by indicating their level of satisfaction on a 10-point visual analog scale (VAS), with 0 indicating totally unsatisfied or ‘highly unattractive’ and 10 indicating totally satisfied or ‘highly attractive’ (Gatti et al., 2017). 3. Results Over a 7-year period (2011e2017), 53 patients were selected according to the eligibility criteria. Of these, 31 were males and 22 were females, with an age range of 18e37 years (mean 23 ± 1.5 years). The mean follow-up period was 3.92 ± 2.18 years (range 6 months to 6 years). The structure and shape of the cartilaginous framework in all cases were stable and aesthetically favorable. The incisions in 51 of these 53 patients were treated by one-stage healing. A small amount of diced bone was exposed in two cases. This occurred at the top of the alveolar ridge because of excessive tissue tensions. The wound healed a few days later after a prescribed medical treatment. No other complications were noted in the
patients. The height of the red lips appeared symmetrical, and the wet-dry lip lines were continuous. The height of the alar base was restored, with a symmetrical appearance, although the affected nostrils were slightly smaller than on the healthier side. The preoperative appearance and postoperative results for one patient are shown in Fig. 6. The changes in the parameters assessed preoperatively and postoperatively are shown in Table 1 (mean ± standard deviation). The preoperative measurements of patients showed a smaller ATC angle and larger columellaelabial angle when compared with the follow-up measurements. All values were reported as mean measurements with standard deviations. The mean values for the ATC angle were 55.35 preoperatively and 44.95 at follow-up. This gave a mean decrease in ATC angle of 10.40 , or 18.8 per cent (p < 0.001). There was a considerable increase in the columellaelabial angle (10.28 ± 8.11 ; p < 0.001). The average father satisfaction scores on the GAIS (Table 2) were 4.5 (nasal dorsum), 4.7 (nasal tip), 4.8 (nasal alar), 4.6 (nostril and nasal columella), and 4.7 (nasal base). The average mother satisfaction scores as assessed by GAIS were 4.4 (nasal dorsum), 4.6 (nasal tip), 4.5 (nasal alar), 4.5 (nostril and nasal columella), and 4.4 (nasal base). The average levels of aesthetic satisfaction as assessed by VAS (Table 3) were 8.9 (father), 8.7 (mother), 9.1 (lay person), and 8.5 (senior cleft surgeon).
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Fig. 6. Preoperative views (A, C, E) and 1-year postoperative views (B, D, F).
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
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F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Table 1 Preoperative and postoperative measurements, and the differences in values; mean ± standard deviationa. Parameters
Preoperative
Postoperative
Change
ATC angle ( ) Columellaelabial angle ( )
55.35 ± 6.27 82.50 ± 15.51
44.95 ± 5.09 92.78 ± 10.76
10.40 ± 6.29b 10.28 ± 8.11b
ATC, alar baseenasal tipecolumellar base. a n ¼ 53 patients. b p < 0.001.
Table 2 Satisfaction scores using the GAIS, and the differences in values; mean ± standard deviation. Assessor
Nasal dorsum
Nasal tip
Nasal alar
Nostril and nasal columella
Nasal base
Father (or 4.59 ± 0.23 4.73 ± 0.36 4.81 ± 0.43 4.62 ± 0.37 4.70 ± 0.42 male guardian) 4.43 ± 0.33 4.60 ± 0.21 4.52 ± 0.27 4.50 ± 0.46 4.45 ± 0.39 Mother (or female guardian)
Table 3 Aesthetic satisfaction scores assessed using VAS, and the differences in values; mean ± standard deviation. Assessor
Average score of satisfaction
Father (or male guardian) Mother (or female guardian) Lay person Senior cleft surgeon
8.93 8.72 9.10 8.56
± ± ± ±
1.02 0.56 0.94 0.45
4. Discussion Successful cleft lip and nasal repairs require comprehensive and sequential therapy. Unfortunately, several patients in this study had been unable to receive appropriate sequential treatment because of developmental imbalances across different regions and poor socioeconomic conditions. The study included mainly adult patients, most of whom were from poor families, and so it was difficult for them to opt for orthodontic treatment. Most of the patients included had not undergone surgery for the repair of alveolar cleft or orthodontic treatment at the appropriate time. The key to a successful outcome is the communication between the surgeons and patients in selecting an appropriate treatment (Akgüner et al., 1998; Daniel et al., 2004; Guyuron, 2008; Guo et al., 2010). The secondary unilateral cleft lip-nose deformities in these cases were repaired in one procedure. The appearances of almost all the patients were significantly improved, and all the patients and their families were satisfied with the outcome of the operation. Due to lack of bone support in the alveolar ridge and pyriform aperture of cleft lip-nose cases, the alar cartilage does not develop in the normal position. The upper lip of the affected side and the alar base appear collapsed, resulting in asymmetry (Bohluli et al., 2017). When these deformities are corrected, surgical outcome often remains unsatisfactory. Nowadays patients are increasingly willing to have an alveolar bone graft and secondary cleft lip operation simultaneously. This treatment corrects the three kinds of deformity in one procedure, and has many benefits. Firstly, the surgeons can take full advantage of autogenous tissue, and even the abandoned tissue, to achieve better results. For example, not only the cancellous bone, but also the cartilage and cortical bone can be harvested from the ribs (Kawamoto et al., 2008;
Unlu et al., 2009), while the scar tissue that is excised during the upper lip trimming is available for use in extending the ipsilaterally short columella or to make up for other deficiencies. Secondly, the correction of facial deformities reduces psychological disorders in patients. Thirdly, one-stage correction is considered to be economical for patients, as it minimizes the frequency of patient visits. Bone grafting in the alveolar ridge not only restores the continuity of the alveolar arch, but also increases the height of the alar base and the maxillary bone mass in the pyriform aperture area. If a bone graft comprises only cortical bone particles it may undergo more absorption. Therefore, we added extra cartilage particles to the cortical bone, hoping to reduce the impact of possible absorption on the height of the nose base, and thus maintain a stable shape for longer. The cartilage graft is one of the important steps in this therapy, as it is directly associated with the treatment result. Cartilage grafts are frequently used in cosmetic and reconstructive nasal surgeries. Autogenous rib cartilage can be easily obtained in large quantities, and can be carved into the desired shape. The strength and toughness of this tissue contribute to its supporting role. It has less absorptive capacity, is unlikely to be rejected, and can maintain a stable shape for a long time. Techniques for cartilage grafting have improved over time, but problems associated with this method remain, such as warping, visibility, migration, extrusion, and infection, while the long-term integrity of the graft is not guaranteed. In recent years, there has been increasing interest in using diced cartilage for rhinoplasty procedures. The use of diced cartilage rather than a solid piece of cartilage graft is an attractive concept due to its greater flexibility (Bracaglia et al., 2012), as well as minimal risk of warping, obviating the need for a long and straight cartilage graft donor site (Erol, 2012). The diced cartilage is usually wrapped in fascia before being inserted in the appropriate location (Daniel et al., 2004; Unlu et al., 2009; Erol, 2017). This wrapping is used to avoid visible irregularities associated with the angular cartilage cubes once the swelling has subsided. Our hospital has refined and simplified the diced cartilage technique in recent years d the cartilage is diced very finely and no fascia sleeve is needed. The other advantage of the diced cartilage method is that the nasal shape can be adjusted over 1e2 months postoperatively. Anatomical restoration of the orbicularis oris during upper lip trimming is conducive to the recovery of lip function and correction of nasal deformities. What is more, it can reduce the postoperative scar. Continuity of the maxillary arch is restored and maxillary stability is increased. There are some disadvantages associated with one-stage correction. It is not suitable for all cases, for example in malocclusion patients (Baumann, 2010; Gatti et al., 2017). In our study, most of our patients could not be treated with orthodontics because of economic problems, which will greatly reduce the effectiveness of this surgery. The assessment of esthetics by the different cohorts of people was performed in a basic way, so a more definitive evaluation technique is perhaps required. In conclusion, secondary unilateral cleft lip-nose deformity can be corrected in one stage, which is considered to be a safe, reliable, and effective option. Early and later outcomes are satisfactory, with no serious complications. Patient consent Not required. Ethical approval This study was approved by the Nanjing Medical University ethics board.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Funding This work was supported in part by the National Natural Science Foundation of China (81672678), Priority Academic Program Development of Jiangsu Higher Education Institutions (PAPD, 201887), and the Project of Invigorating Health Care through Science, Technology and Education (Jiangsu Provincial Medical Youth Talent QNRC2016852). It was also sponsored by the Qing Lan Project. Declaration of Competing Interest The authors declare that they have no conflicts of interest. References Akgüner M, Barutçu A, Karaca C: Adolescent growth patterns of the bony and cartilaginous framework of the nose: a cephalometric study. Annals of Plastic Surgery 41: 66e69, 1998 Baumann L: Dermal fillers. Journal of Cosmetic Dermatology 3: 249e250, 2010 Bergeron L, Chen P: Asian rhinoplasty techniques. Seminars in Plastic Surgery 23, 2009 016e021 Bohluli B, Varedi P, Sezavar M, Pakzad S, Bagheri SC: Component columella augmentation in cleft nose rhinoplasty: a preliminary study. International Journal of Oral and Maxillofacial Surgery 46: 548e553, 2017 Bracaglia R, Tambasco D, DʼEttorre M, Gentileschi S: ‘Nougat graft’: diced cartilage graft plus human fibrin glue for contouring and shaping of the nasal dorsum. Plastic and Reconstructive Surgery 130: 741e, 2012
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Chepla KJ, Gosain AK: Evidence-based medicine: cleft palate. Plastic and Reconstructive Surgery 132: 1644e1648, 2013 Daniel RK, Calvert JW: Diced cartilage grafts in rhinoplasty surgery. Plastic and Reconstructive Surgery 113: 2156e2171, 2004 Erol OO: Injection of compressed diced cartilage in the correction of secondary and primary rhinoplasty. Plastic and Reconstructive Surgery 140: 673ee685e, 2017 € Erol OO: Tip rhinoplasty in broad noses in a Turkish population. Plastic and Reconstructive Surgery 130: 185e197, 2012 Gatti GL, Freda N, Giacomina A, Montemagni M, Sisti A: Cleft lip and palate repair. The Journal of Craniofacial Surgery 28: 1918e1924, 2017 Guo JY, Wang YX: Study of the influencing factors of the secondary unilateral cleft nasal deformity. China Journal of Oral and Maxillofacial Surgery 8: 415e420, 2010 Guyuron B: MOC-PS(SM) CME article: late cleft lip nasal deformity. Plastic and Reconstructive Surgery 121: 1e11, 2008 Haug RH: Correction of secondary cleft lip and nasal deformities. Indonesian Journal of Oral and Maxillofacial Surgery 7(3), 2004 Henkel KO, Gundlach K, Saka B: Incidence of secondary lip surgeries as a function of cleft type and severity: one center's experience. The Cleft Palate-Craniofacial Journal 35: 310, 1998 Jeong H, Lee H, Shin K: Correction of unilateral secondary cleft lip nose deformity by a modified Tajima's method and several adjunctive procedures based on severity. Aesthetic Plastic Surgery 36: 406e413, 2012 Kawamoto HK, Jarrahy R, Sedrak MF, Ashley RK, Bradley JP: ‘Stuffy nose’ rhinoplasty: diced cartilage grafts for correction of cleft nasal tip deformities. Plastic and Reconstructive Surgery 122: 1138, 2008 Pawar S, Wang T: Secondary cleft rhinoplasty. JAMA Facial Plastic Surgery 16(58), 2014 Unlu RE, Altun S, Inozu E, Koc MN: Diced cartilage grafts in rhinoplasty surgery: current techniques and applications. Plastic and Reconstructive Surgery 124: 666, 2009 Tanaka SA, Mahabir RC, Jupiter DC, Menezes JM: Updating the epidemiology of cleft lip with or without cleft palate. Plastic and Reconstructive Surgery 129: 511e, 2012
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Single-stage repair of secondary unilateral cleft lip-nose deformity in adults Fan Zhou a, 1, Wen Lin a, 1, Yifei Du a, Sheng Li a, Hongbing Jiang a, Linzhong Wan a, **, Hua Yuan a, b, * a b
Department of Oral and Maxillofacial Surgery, Affiliated Hospital of Stomatology, Nanjing Medical University, Nanjing, China Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, Nanjing, China
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 25 May 2019 Accepted 2 December 2019 Available online xxx
Correction of cleft lip-nose deformity in adult patients is different from that in children. One-stage correction has proved to be a suitable technique for patients with cleft-lip nose deformity. This study aimed to explore a particular single-stage method and evaluate the effect of simultaneous reparation of secondary unilateral cleft lip-nose deformities. Cleft lip patients who had previously undergone nasolabial surgery with residual poor nasal/lip appearance were included. The alveolar bone defect was repaired with granular costal cortical bone. Lip revision and rhinoplasty were performed using diced costal cartilage. The lip, nose, and alveolar deformities were corrected in one stage. From 2011 to 2017, 53 cases were treated. The vermilion discrepancy was corrected in all cases. Fiftyone patients were successfully treated, with primary healing in the bony recipient area. Cancellous bone exposure occurred in two cases. The wounds were healed after debridement and drainage. Appearances were improved in all patients. The mean change in columellaelabial angle ranged from 82.50 to 92.78 (p < 0.001). This one-stage correction appears to have led to a distinct improvement in the nasal tip projection and lip. The method is considered to be effective and reliable in patients with secondary unilateral cleft lip-nose deformities. © 2019 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Unilateral cleft lip Rhinoplasty Deformity Diced cartilage
1. Introduction Cleft lip and palate is one of the most common congenital craniofacial deformities (Tanaka et al., 2012; Chepla et al., 2013). Although a satisfactory lip shape can be restored after primary cleft lip repair, secondary deformities of the cleft lip and nose can occur with development (Jeong et al., 2012; Pawar. et al., 2014). There are several reasons for this, which include poor adherence by children to instructions, flawed surgical procedures, congenital nasal cartilage abnormalities, muscular dystrophy, and
* Corresponding author. Jiangsu Key Laboratory of Oral Diseases, Nanjing Medical University, 140 Hanzhong Rd., Nanjing, China. Fax: þ86 25 85031914. ** Corresponding author. Department of Oral and Maxillofacial Surgery, Affiliated Hospital of Stomatology, Nanjing Medical University, Nanjing, China. Fax: þ86 25 85031942. E-mail addresses: [email protected] (L. Wan), [email protected] (H. Yuan). 1 Contributed equally to this work.
abnormal development of the jaw bone (Haug, 2004). The incidence of secondary deformities is very high, ranging from 35% to 85% according to previous reports (Henkel et al., 1998). For the treatment of alveolar cleft, cleft lip, and nasal deformities, traditional methods have involved several operations at different stages. In some cases, available tissues are overlooked during the operation and deformities can be aggravated by delayed surgery, affecting the treatment outcome. Recently, onestage correction has become accepted as a treatment for secondary lip-nose deformity. This study describes the authors’ experiences in repairing secondary unilateral cleft lip-nose deformities.
2. Material and methods This study was reviewed by the Institutional Review Board of Nanjing Medical University. The clinical data of 53 patients with
https://doi.org/10.1016/j.jcms.2019.12.004 1010-5182/© 2019 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
2
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
secondary unilateral cleft lip-nose deformities who underwent surgery at the Affiliated Stomatological Hospital of Nanjing Medical University from January 2011 to August 2017 were retrospectively reviewed. Most of the patients were from poor families, and they wanted to improve their appearances without undergoing orthodontic or orthognathic treatment.
cut to support the columella and nasal dorsum. The remaining cartilage was cut into pieces of about 1 mm diameter using a No. 11 blade, and soaked in an antibiotic solution (1 g of kanamycin sulfate diluted in 250e500 ml 0.9% sodium chloride (Fig. 1).
2.1. Rib harvest
The diced cortical bone and cartilage graft were used to close the nasolabial fistula and elevate the alar base. The edges of the mucosa were incised, fully exposing the alveolar bone surface, pyriform aperture margin, and the surrounding bone surface. This was followed by closure of the mucous membrane of the nose floor and palatal side with sutures, forming the base of the bone graft zone. Multiple holes were drilled in the lateral wall of the two alveolar ridges, so that the bone marrow mesenchymal stem cells in the alveolar bone could enter the bone graft area. The diced cortical bone was packed tightly into the prepared bone defect zone, and the bone height of the graft zone was made slightly higher than normal d by 1e2 mm d in order to close the nasolabial fistula and elevate the alar base. The soft tissue incision was closed after the bone graft was completed.
Cartilage and cortical bone were harvested from the rib (Bergeron et al., 2009). The latter was prepared for alveolar bone graft. Firstly, the intersected costal cartilage was cut into two small pieces, 25 mm long and 8 mm wide. The two cartilages were then
2.2. Alveolar bone graft
2.3. Lip revision The planned incision for lip revision was drawn along the original scar using methylene blue (Fig. 2). Sometimes the scar tissue could also be used for repairing the nasal vestibular area in soft tissue defects or to extend the affected nasal columella. We dissected and reset the orbicularis oris muscle, along the labial bow
Fig. 1. (A) The harvested costochondral graft. (B) Two pieces of cartilage were cut to prepare for columella and nasal dorsum support. (C) The rest of the cartilage was cut into pieces of less than 1 mm.
Fig. 2. (A) Alar base deformity. (B) Lip splitting and muscle repositioning to correct the alar base.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
and the wet-dry border of the lip. The muscles were then reduced and the position of the base of the nasal alar was corrected. 2.4. Rhinoplasty Finally, reconstruction of the nasal shape was performed using the costal cartilage graft. A horizontal incision was made along the lower part of the nasal column to expose the nasal alar cartilage, forming a subperiosteal bag and creating a tunnel for the columella support insert. A piece of cartilage (2.5 cm) was then inserted into the columella and anterior nasal spine, and another into the nasal dorsum (Fig. 3a). The cartilage was carved to taper from front to back, with a groove carved into the front face, and was connected to the top of the supporting cartilage of the nasal columella. A
3
truncated 1 ml syringe was then used to inject the cartilage fragments into the nasal dorsum in order to allow correction of nose length and projection of the columella (Fig. 3b). This was followed by suturing of the columella and lower lateral cartilage. The results of the correction are shown in Fig. 4. The graft could then be externally molded to straighten and lengthen the nose. A standard protective mould was then placed over the nasal dorsum. Stitches were removed on day 7 postoperatively. Patients were instructed to massage the nose for 3e5 min, six times a day, for 1e2 months to reduce swelling and adjust the nasal shape if necessary. 2.5. Comparative measurements Standard facial photographs were taken during the preoperative and follow-up periods. These images were used to measure the alar baseenasal tipecolumellar base (ATC) angle, with the apex at the nasal tip (Fig. 5A, B), and the columellaelabial angle d the curved junction of the columella with the upper lip (Fig. 5C, D). These measurements were performed by three independent reviewers and were compared as mean values. 2.6. Investigation of postoperative patient satisfaction There is no international standard regarding classification of the severity of secondary nasal deformity after cleft lip surgery, or a unified standard of satisfaction scores for patients with nasal deformity. Based on the latest pre- and postoperative clinical images, two observers evaluated the degree of patient satisfaction with their surgical outcome according to the five-point global aesthetic
Fig. 3. (A) Placement of one carved cartilage in the columella and another in the nasal dorsum. (B, C) Placement of cartilage fragments into the nasal dorsum.
Fig. 4. (A, B) Results of correction.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
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Fig. 5. Patient preoperatively (A) and postoperatively (B), showing the alar baseenasal tipecolumellar base angle. Lateral view of patient preoperatively (C) and postoperatively (D) to show the columellaelabial angle.
improvement scale (GAIS) (Haug, 2004). For each postoperative outcome, the parents (or guardians) gave scores of 5 points for ‘very much improved’, 4 points for ‘much improved’, 3 points for ‘improved’, 2 points for ‘not changed’, and 1 point for ‘worsened’. Four assessors, including parents of the children, a lay person, and a senior cleft surgeon, assessed the repairs separately by indicating their level of satisfaction on a 10-point visual analog scale (VAS), with 0 indicating totally unsatisfied or ‘highly unattractive’ and 10 indicating totally satisfied or ‘highly attractive’ (Gatti et al., 2017). 3. Results Over a 7-year period (2011e2017), 53 patients were selected according to the eligibility criteria. Of these, 31 were males and 22 were females, with an age range of 18e37 years (mean 23 ± 1.5 years). The mean follow-up period was 3.92 ± 2.18 years (range 6 months to 6 years). The structure and shape of the cartilaginous framework in all cases were stable and aesthetically favorable. The incisions in 51 of these 53 patients were treated by one-stage healing. A small amount of diced bone was exposed in two cases. This occurred at the top of the alveolar ridge because of excessive tissue tensions. The wound healed a few days later after a prescribed medical treatment. No other complications were noted in the
patients. The height of the red lips appeared symmetrical, and the wet-dry lip lines were continuous. The height of the alar base was restored, with a symmetrical appearance, although the affected nostrils were slightly smaller than on the healthier side. The preoperative appearance and postoperative results for one patient are shown in Fig. 6. The changes in the parameters assessed preoperatively and postoperatively are shown in Table 1 (mean ± standard deviation). The preoperative measurements of patients showed a smaller ATC angle and larger columellaelabial angle when compared with the follow-up measurements. All values were reported as mean measurements with standard deviations. The mean values for the ATC angle were 55.35 preoperatively and 44.95 at follow-up. This gave a mean decrease in ATC angle of 10.40 , or 18.8 per cent (p < 0.001). There was a considerable increase in the columellaelabial angle (10.28 ± 8.11 ; p < 0.001). The average father satisfaction scores on the GAIS (Table 2) were 4.5 (nasal dorsum), 4.7 (nasal tip), 4.8 (nasal alar), 4.6 (nostril and nasal columella), and 4.7 (nasal base). The average mother satisfaction scores as assessed by GAIS were 4.4 (nasal dorsum), 4.6 (nasal tip), 4.5 (nasal alar), 4.5 (nostril and nasal columella), and 4.4 (nasal base). The average levels of aesthetic satisfaction as assessed by VAS (Table 3) were 8.9 (father), 8.7 (mother), 9.1 (lay person), and 8.5 (senior cleft surgeon).
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Fig. 6. Preoperative views (A, C, E) and 1-year postoperative views (B, D, F).
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
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F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Table 1 Preoperative and postoperative measurements, and the differences in values; mean ± standard deviationa. Parameters
Preoperative
Postoperative
Change
ATC angle ( ) Columellaelabial angle ( )
55.35 ± 6.27 82.50 ± 15.51
44.95 ± 5.09 92.78 ± 10.76
10.40 ± 6.29b 10.28 ± 8.11b
ATC, alar baseenasal tipecolumellar base. a n ¼ 53 patients. b p < 0.001.
Table 2 Satisfaction scores using the GAIS, and the differences in values; mean ± standard deviation. Assessor
Nasal dorsum
Nasal tip
Nasal alar
Nostril and nasal columella
Nasal base
Father (or 4.59 ± 0.23 4.73 ± 0.36 4.81 ± 0.43 4.62 ± 0.37 4.70 ± 0.42 male guardian) 4.43 ± 0.33 4.60 ± 0.21 4.52 ± 0.27 4.50 ± 0.46 4.45 ± 0.39 Mother (or female guardian)
Table 3 Aesthetic satisfaction scores assessed using VAS, and the differences in values; mean ± standard deviation. Assessor
Average score of satisfaction
Father (or male guardian) Mother (or female guardian) Lay person Senior cleft surgeon
8.93 8.72 9.10 8.56
± ± ± ±
1.02 0.56 0.94 0.45
4. Discussion Successful cleft lip and nasal repairs require comprehensive and sequential therapy. Unfortunately, several patients in this study had been unable to receive appropriate sequential treatment because of developmental imbalances across different regions and poor socioeconomic conditions. The study included mainly adult patients, most of whom were from poor families, and so it was difficult for them to opt for orthodontic treatment. Most of the patients included had not undergone surgery for the repair of alveolar cleft or orthodontic treatment at the appropriate time. The key to a successful outcome is the communication between the surgeons and patients in selecting an appropriate treatment (Akgüner et al., 1998; Daniel et al., 2004; Guyuron, 2008; Guo et al., 2010). The secondary unilateral cleft lip-nose deformities in these cases were repaired in one procedure. The appearances of almost all the patients were significantly improved, and all the patients and their families were satisfied with the outcome of the operation. Due to lack of bone support in the alveolar ridge and pyriform aperture of cleft lip-nose cases, the alar cartilage does not develop in the normal position. The upper lip of the affected side and the alar base appear collapsed, resulting in asymmetry (Bohluli et al., 2017). When these deformities are corrected, surgical outcome often remains unsatisfactory. Nowadays patients are increasingly willing to have an alveolar bone graft and secondary cleft lip operation simultaneously. This treatment corrects the three kinds of deformity in one procedure, and has many benefits. Firstly, the surgeons can take full advantage of autogenous tissue, and even the abandoned tissue, to achieve better results. For example, not only the cancellous bone, but also the cartilage and cortical bone can be harvested from the ribs (Kawamoto et al., 2008;
Unlu et al., 2009), while the scar tissue that is excised during the upper lip trimming is available for use in extending the ipsilaterally short columella or to make up for other deficiencies. Secondly, the correction of facial deformities reduces psychological disorders in patients. Thirdly, one-stage correction is considered to be economical for patients, as it minimizes the frequency of patient visits. Bone grafting in the alveolar ridge not only restores the continuity of the alveolar arch, but also increases the height of the alar base and the maxillary bone mass in the pyriform aperture area. If a bone graft comprises only cortical bone particles it may undergo more absorption. Therefore, we added extra cartilage particles to the cortical bone, hoping to reduce the impact of possible absorption on the height of the nose base, and thus maintain a stable shape for longer. The cartilage graft is one of the important steps in this therapy, as it is directly associated with the treatment result. Cartilage grafts are frequently used in cosmetic and reconstructive nasal surgeries. Autogenous rib cartilage can be easily obtained in large quantities, and can be carved into the desired shape. The strength and toughness of this tissue contribute to its supporting role. It has less absorptive capacity, is unlikely to be rejected, and can maintain a stable shape for a long time. Techniques for cartilage grafting have improved over time, but problems associated with this method remain, such as warping, visibility, migration, extrusion, and infection, while the long-term integrity of the graft is not guaranteed. In recent years, there has been increasing interest in using diced cartilage for rhinoplasty procedures. The use of diced cartilage rather than a solid piece of cartilage graft is an attractive concept due to its greater flexibility (Bracaglia et al., 2012), as well as minimal risk of warping, obviating the need for a long and straight cartilage graft donor site (Erol, 2012). The diced cartilage is usually wrapped in fascia before being inserted in the appropriate location (Daniel et al., 2004; Unlu et al., 2009; Erol, 2017). This wrapping is used to avoid visible irregularities associated with the angular cartilage cubes once the swelling has subsided. Our hospital has refined and simplified the diced cartilage technique in recent years d the cartilage is diced very finely and no fascia sleeve is needed. The other advantage of the diced cartilage method is that the nasal shape can be adjusted over 1e2 months postoperatively. Anatomical restoration of the orbicularis oris during upper lip trimming is conducive to the recovery of lip function and correction of nasal deformities. What is more, it can reduce the postoperative scar. Continuity of the maxillary arch is restored and maxillary stability is increased. There are some disadvantages associated with one-stage correction. It is not suitable for all cases, for example in malocclusion patients (Baumann, 2010; Gatti et al., 2017). In our study, most of our patients could not be treated with orthodontics because of economic problems, which will greatly reduce the effectiveness of this surgery. The assessment of esthetics by the different cohorts of people was performed in a basic way, so a more definitive evaluation technique is perhaps required. In conclusion, secondary unilateral cleft lip-nose deformity can be corrected in one stage, which is considered to be a safe, reliable, and effective option. Early and later outcomes are satisfactory, with no serious complications. Patient consent Not required. Ethical approval This study was approved by the Nanjing Medical University ethics board.
Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004
F. Zhou et al. / Journal of Cranio-Maxillo-Facial Surgery xxx (xxxx) xxx
Funding This work was supported in part by the National Natural Science Foundation of China (81672678), Priority Academic Program Development of Jiangsu Higher Education Institutions (PAPD, 201887), and the Project of Invigorating Health Care through Science, Technology and Education (Jiangsu Provincial Medical Youth Talent QNRC2016852). It was also sponsored by the Qing Lan Project. Declaration of Competing Interest The authors declare that they have no conflicts of interest. References Akgüner M, Barutçu A, Karaca C: Adolescent growth patterns of the bony and cartilaginous framework of the nose: a cephalometric study. Annals of Plastic Surgery 41: 66e69, 1998 Baumann L: Dermal fillers. Journal of Cosmetic Dermatology 3: 249e250, 2010 Bergeron L, Chen P: Asian rhinoplasty techniques. Seminars in Plastic Surgery 23, 2009 016e021 Bohluli B, Varedi P, Sezavar M, Pakzad S, Bagheri SC: Component columella augmentation in cleft nose rhinoplasty: a preliminary study. International Journal of Oral and Maxillofacial Surgery 46: 548e553, 2017 Bracaglia R, Tambasco D, DʼEttorre M, Gentileschi S: ‘Nougat graft’: diced cartilage graft plus human fibrin glue for contouring and shaping of the nasal dorsum. Plastic and Reconstructive Surgery 130: 741e, 2012
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Please cite this article as: Zhou F et al., Single-stage repair of secondary unilateral cleft lip-nose deformity in adults, Journal of Cranio-MaxilloFacial Surgery, https://doi.org/10.1016/j.jcms.2019.12.004