Journal of Cranio-Maxillo-Facial Surgery xxx (2015) 1e6
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A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition Praveen Ganesh a, *, Jyotsna Murthy b, Navitha Ulaghanathan c, V.H. Savitha c a b c
Department of Cranio-Maxillofacial Surgery, Mazumdar Shaw Medical Center, Narayana Health City, Bommasandra, Bangalore, Karnataka, 560099, India Department of Plastic and Reconstructive Surgery, Sri Ramachandra University, Chennai, India Department of Speech, Language & Hearing Sciences, Sri Ramachandra University, Chennai, India
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 18 September 2014 Accepted 26 March 2015 Available online xxx
Objective: To study the growth and speech outcomes in children who were operated on for unilateral cleft lip and palate (UCLP) by a single surgeon using two different treatment protocols. Material and methods: A total of 200 consecutive patients with nonsyndromic UCLP were randomly allocated to two different treatment protocols. Of the 200 patients, 179 completed the protocol. However, only 85 patients presented for follow-up during the mixed dentition period (7e10 years of age). The following treatment protocol was followed. Protocol 1 consisted of the vomer flap (VF), whereby patients underwent primary lip nose repair and vomer flap for hard palate single-layer closure, followed by soft palate repair 6 months later; Protocol 2 consisted of the two-flap technique (TF), whereby the cleft palate (CP) was repaired by two-flap technique after primary lip and nose repair. GOSLON Yardstick scores for dental arch relation, and speech outcomes based on universal reporting parameters, were noted. Results: A total of 40 patients in the VF group and 45 in the TF group completed the treatment protocols. The GOSLON scores showed marginally better outcomes in the VF group compared to the TF group. Statistically significant differences were found only in two speech parameters, with better outcomes in the TF group. Conclusions: Our results showed marginally better growth outcome in the VF group compared to the TF group. However, the speech outcomes were better in the TF group. © 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Mixed dentition Growth Speech outcomes Vomer flap Two-flap technique
1. Introduction The approximate incidence of CLP is 1.3 per 1000 live births in India (Mossey and Little, 2009). There are numerous techniques described for management of patients with CLP. However, there is sparse information relating to specific management, intervention methods, and long-term outcomes in patients who have completed a strict treatment protocol. The confounding factors and inconclusive outcomes found in the available literature from around the world have led to the development of many protocols; for instance, there existed as many as 194 protocols in 205 European centers in 2001 (Shaw et al., 2001). Inter-center comparisons
* Corresponding author. E-mail address: [email protected] (P. Ganesh).
have limitations in that they cannot distinguish the relationship between protocols of different centers or the elements of a center's protocol on the outcome, and the influence of the personnel delivering that protocol (Shaw et al., 2005). One possible way to overcome the above limitations is by conducting a randomized controlled trial in subjects from a single center operated on by a single surgeon. One of the surgical protocols for patients with UCLP is using a vomer flap for the cleft of the hard palate while repairing the cleft lip. The advantages of using vomer flap include simplicity and ease of execution, without adding to surgical trauma or prolonging surgical time. This technique aids in providing an effective nasal lining in almost all types of clefts (Kobus, 1987). A debate still exists regarding the relationship between use of vomer flap as a single lining for the hard palate and defective mid-face growth (Agrawal
http://dx.doi.org/10.1016/j.jcms.2015.03.033 1010-5182/© 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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and Panda, 2006). There is a deficit of randomized control trials using the vomer flap as a single layer for the hard palate repair in comparison to other techniques such as the Veau-Wardill-Kilner, Von Langenbeck, and two-flap techniques, among others. This study was a single-center, prospective, randomized controlled study. A comparative assessment of treatment outcomes of nonsyndromic UCLP patients with two different surgical protocols, namely, the vomer flap (VF) versus the two-flap technique (TF), was carried out. Keeping in mind the large case load being operated on by a single surgeon with no prior commitment to either of the techniques, the randomized controlled trial was deemed appropriate. This design would also suggest how the techniques would compare in the hands of a single surgeon. Success in cleft lip and palate surgery cannot be judged only by the esthetic outcomes, but should also consider functional parameters such as speech and dental arch relationships (Pradel et al., 2009; Hathorn et al., 1996; Mars and Houston, 1990). The two techniques in this study were thus evaluated and compared based on the dental arch relationship, speech outcomes, and occurrence of fistula. It was hypothesized that there would be no significant differences between the two surgical protocols across these parameters. 2. Material and methods After obtaining ethical clearance from Ramachandra University institutional ethics committee (Reference number- IEC/NI/03/MAY/ 13/33), 200 consecutive patients with nonsyndromic UCLP were included in this randomized trial from 2003 to 2005. The parents received explanations about the proposed study in understandable language. The parents were also informed that the standard of care would not be compromised if they decided to opt out of the study. None of the parents declined to join the study. Randomization was done by allocation concealment, whereby 200 chits were put in a box (100 for each group) and the parent or guardian was asked to pick one chit 1 day before the surgery. The patient was allotted to the treatment protocol as indicated in the chit. Fifteen children who did not complete the two surgeries as suggested in the protocols and 6 children operated on by more than one surgeon were excluded. Of the 179 patients who completed treatment for lip and palate repair, operated on by a single surgeon, only 85 (40 patients in the VF group and 45 patients in the TF group) came for regular follow-up through the period of mixed dentition. These patients were in the age range of 7e9 years at the time of last follow-up. Details on the mean age of intervention and follow-up of the participants is summarized in Table 1. For various reasons, speech samples were obtained from only 34 patients in the VF group and 39 in the TF group. Two protocols were selected for randomization. In the VF group, the cleft lip was repaired using the Millard technique along with nose correction. The vomer flap was used as a single layer for hard palate closure. After 6 months, soft palate repair was carried out with sharp separation of the muscle fibers from the enveloping oral and the nasal mucosa and from the hard palatal shelves. The tensor
Table 1 Mean age of intervention and outcomes of study Protocol
Vomer flap (VF)
Two-flap (TF)
Mean age of lip repair Mean age of palate repair Mean age of follow-up for dental arch relationship evaluation
5.22 Months 12.3 Months 7.8 Years
6.3 Months 12.9 Months 8.1 Years
tendon was released just medial to the hamulus, followed by retro positioning and plication of muscle bundles along the midline. During soft palate repair, minor to major lateral releasing incision (as in the von-Langenbeck technique), either unilateral or bilateral, was needed to close the junction area in 36 of 91 patients. In the TF group, cleft lip was repaired by the Millard technique with nose correction, and anterior palate repair up to the incisor foramen. Six months later, the palate was repaired with two-flap palatoplasty. The surgeon had experience of more than 10 years in using the TF technique, whereas the VF technique was introduced in practice only 1 year before the commencement of the randomized trial. None of the children had either preoperative or postoperative orthopedic intervention during the mixed dentition. All patients underwent routine speech evaluation between 4 and 6 years of age. Based on the profile of articulation, they were provided with three to five sessions of speech therapy at the hospital, focusing on demonstrating correction of specific articulation errors to the parents. Home training programs were recommended for correction of articulation. None of them received long-term institution-based speech therapy for correction of speech errors. To study the maxillary growth outcomes, digital intraoral photographs were taken for all patients during mixed dentition. The set of intraoral photographs included the frontal view in occlusion, right and left buccal views, and right and left overjet views. History of palatal fistula was taken from the records. Speech samples of all patients were audio-recorded by a speech pathologist in a soundtreated room. Recognizing the need for a comprehensive speech sample (Sell, 2005; Kuehn and Moller, 2000), the recorded sample comprised a 2-min conversation, counting of numbers from 1 to 10, syllable repetition, repetition of phonetically loaded words, and sentences in the Tamil language. All children passed hearing screening (pure tone average of less than 20 dBHL) at the time when the speech samples were recorded. However, the status of the middle ear was not examined. Mars et al. (1987) published a simple method named the GOSLON (an acronym denoting “Great Ormond Street, London and Oslo”) Yardstick to score the outcome of treatment in patients with unilateral cleft lip and palate. The outcome of treatment is viewed on occlusion and scored by experienced raters. Patients are categorized into one of the following five groups: group 1: positive overjet with average inclined or retroclined incisors with no crossbite or open bite with excellent long-term outcome; group 2: positive overjet with average inclined or proclined incisors with unilateral cross-bite or cross-bite tendency with or without open bite tendency around the cleft site with good long-term outcome; group 3: edge-to-edge bite with average inclined or proclined incisors or reverse overjet with retroclined incisors with unilateral cross-bite with or without open bite tendency around the cleft site, with fair long-term outcome; group 4: reverse overjet with average inclined or proclined incisors, with or without bilateral cross-bite tendency with or without open bite tendency around the cleft site, with poor outcome; and group 5, reverse overjet with proclined incisors, bilateral cross-bite, and poor maxillary arch form and palatal vault anatomy with very poor outcome (Mars et al., 2006). The GOSLON Yardstick does not involve application of precise and detailed criteria, but relies on a simple method of judgment (Lilja et al., 2006). Two examiners, a surgeon and an orthodontist, who were not members of the cleft team, scored the intraoral digital photographs on two separate occasions 2 weeks apart (Fig. 1). Each examiner was calibrated previously in the use of the GOSLON Yardstick to reduce systematic bias. The examiners were given a reference image of GOSLON score 1 to 5 as a guide to categorize the photographs during the rating (Liao et al., 2009). No conferring between examiners was allowed, and an overall GOSLON final score was
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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Table 3 Speech outcome parameters with significant differences between vomer flap (VF) and two-flap (TF) groups. Parameter
Grade
VF group
TF group
p Value
Hypernasality
Normal Mild Moderate Absent Present Absent Present
11.8% 70.6% 17.6% 58.8% 41.2% 58.8% 41.2%
20.5% 76.9% 2.6% 84.6% 15.4% 84.6% 15.4%
0.05
Weak oral pressure words Weak oral pressure sentences
Fig. 1. Distribution of GOSLON Yardstick scores between the two groups: vomer flap (VF) and two-flap (TF).
allocated to each set of photographs. Examiners were blinded to the sequence of surgeries performed in the patients. Two speech language pathologists, who were experienced in the area of cleft lip and palate, independently evaluated the speech samples. Speech samples were blinded and randomized before perceptual evaluation. Universal parameters for reporting speech outcomes in individuals with cleft described by Henningsson et al. (2008) were used for perceptual assessment. The rating of hypernasality was performed only at the level of sentences, and not using isolated words. All other parameters (hyponasality, nasal air emission/nasal turbulence, consonant production errors, speech understandability, speech acceptability, and presence of voice disorder) were rated as specified by Henningsson et al. (2008). The scores obtained were tabulated and subjected to statistical analysis. The linear weighted kappa statistical test (computing a lower 95% confidence interval) was used to evaluate intra- and interrater reliability of the GOSLON scores. An independent sample Student ttest was used to compare the GOSLON scores between the two groups. The interrater reliability using kappa statistics to determine the consistency among two speech evaluators showed substantial to almost perfect agreement across parameters (Table 2). The ManneWhitney U test was used to compare the speech ratings between the two groups. 3. Results The VF group included 24 male patients and 16 female patients. There was an almost equal distribution in the TF group, with 23 male and 22 female patients. The distribution of GOSLON Yardstick scores in both the groups is depicted in Fig. 1. Both intra- and interexaminer agreement were high, as indicated by weighted kappa coefficients of 0.85 and 0.90 respectively. In the VF group, 70% of the patients demonstrated
Table 2 Inter- and intrarater reliability of speech analysis. Substantial agreement
Moderate agreement
Velar words (0.789) Weak oral pressures words (0.708) Nasalized voiced pressure consonant (0.660) Developmental articulation error words(0.677) Speech understandability (0.703) Speech acceptability (0.628) Voice disorder (0.671)
Hypernasality sentences (0.551) Mid dorsum palatal words (0.489) Other words and sentences (0.490) Almost perfect agreement Glottal words and sentences (0.916) Nasal fricative words (0.850) Fair agreement Nasal air emission words (0.366)
0.014 0.014
good growth (GOSLON scores 1 and 2) and 30% revealed adequate growth (GOSLON score 3). In the TF group, 54% of the patients displayed good growth, 37.7% had the adequate growth category, and 8.8% had poor growth (GOSLON score 4). None of the patients from either group were found to have very poor GOSLON scores (GOSLON score 5). The mean GOSLON scores were 2.15 (SD ¼ 0.662) and 2.49 (SD ¼ 0.757) for the VF and TF group respectively (t ¼ 2.183). This difference was statistically significant (p ¼ 0.032) based on the analysis using the Student t-test. One patient in the VF group had a palatal fistula, whereas no patient in the TF group had a palatal fistula. This fistula was repaired before the speech sample was taken for analysis. None of these patients had undergone any corrective surgeries for velopharyngeal dysfunction. The ManneWhitney U test revealed a significant difference between the VF and TF groups in speech outcomes on parameters including hypernasality (p ¼ 0.05) and weak oral pressure consonants at the level of words and sentences (p ¼ 0.014), with better outcomes in the TF group (Table 3). In the VF group, 11.8% of patients had normal resonance and 17.6% had moderate hypernasality. In the TF group, 20.5% of patients had normal resonance and 2.6% showed moderate hypernasality. Weak oral pressure consonants were perceived in 41% and 15% of the VF and TF group, respectively. The difference in various parameters of speech and their statistical significance is presented in Table 4. 4. Discussion The outcome of primary repair surgery for cleft lip and palate is often expressed in the subsequent quality of speech, hearing, facial appearance and the dentoskeletal relationships (Johnston et al., 2004). Comparison of two different surgical technique protocols in a single center with a single operating surgeon offers an objective method of evaluating the efficacy of a particular technique. In this study, a randomized controlled trial was initiated for the purpose of comparing two protocols for unilateral complete cleft lip and palate. To date, dental arch relationships for measuring growth outcomes and speech assessment have been considered as the two most useful benchmarks to assess outcomes in the management of children with CLP (Sandy et al., 2001; Sell, 2005). Intraoral photographs provide a valid and reliable method for rating dental arch relationships in patients with UCLP at the age of 9 years and provide viable alternative to the application of the GOSLON Yardstick on dental casts (Liao et al., 2009). A significant difference was observed in growth outcomes between the VF and TF groups, with better results in the VF. This difference is likely to increase with further maxillary growth (Liao et al., 2013). Landheer et al. (2010) reported that two-stage cleft palate repair had a fistula incidence of 27%; whereas one-stage cleft repair had a fistula occurrence of 14%. In the current study, one of the 40 patients in the VF group had a palatal fistula, whereas none had in the TF group had a palatal fistula. The fistula in the VF group occurred at the junction of the hard and soft palate. The incidence of fistula in
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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Table 4 Speech outcome analysis. Parameter
Vomer flap (mean rank)
Nonevomer flap (mean rank)
p Value
Hypernasality Hyponasality Nasal air emission words Nasal air emission sentences Pharyngeal words Pharyngeal sentences Glottal words Glottal sentences Mid dorsum palatal word Mid dorsum palatal sentences Velar words Velar sentences Uvular words Uvular sentences Nasal fricative words Nasal fricative sentences Nasal consonant for oral pressure consonant words Nasal consonant for oral pressure consonant sentences Nasalized voiced pressure consonant words Nasalised voiced pressure consonant sentences Weak oral pressure words Weak oral pressure sentences Developmental/Arti error words Developmental/Arti error sentences Other words Other sentences Speech understandability Speech acceptability Voice disorder
41 36.50 36.81 35.81 36.50 36.50 39.44 39.44 36.57 36.57 33.74 33.24 36.50 36.50 37.72 37.72 36.50 36.50 36.0 35.50 42.03 42.03 38.90 38.90 35.50 35.50 37.59 35.49 35.15
33.51 37.44 37.17 38.04 37.44 37.44 34.87 34.87 37.37 37.37 39.85 40.28 37.44 37.44 36.37 36.37 37.44 37.44 37.87 38.31 32.62 32.62 35.35 35.35 38.31 38.31 36.49 38.32 38.62
.05 .35 .93 .589 .35 .35 .09 .09 .64 .64 .145 .095 .35 .35 .536 .536 .35 .35 .184 .101 .014 .014 .410 .410 .101 .101 .803 .519 .198
the VF group in this study could probably be attributed to the skills of the surgeon because it was a newly added technique in comparison with the 10 years of experience with the two-flap technique. There have been no randomized controlled trials in the literature comparing the speech outcome between groups of patients treated with the vomer flap and two-flap technique. In the current study (VF, 34 subjects; TF, 39 subjects), significant differences between the two groups were observed only in the presence of hypernasality and weak oral pressure consonants, with better speech outcomes in the TF group. Patients in the VF group had a higher percentage of hypernasality of moderate degree (17.6%) compared to patient in the TF group (2.6%). This difference could probably be attributed to the relatively new technique for the surgeon in the VF group. It was observed that soft palates were becoming shorter due to scarring in the area. After completion of the randomized trial, the technique was modified to overcome this shortcoming. The Norwegian center at the Riks Hospital in Oslo, Norway, popularized the technique of lip and vomer flap, followed by closure of the rest of the palate 3 months later. The growth results in their study measured using the GOSLON Yardstick compare well with results from other centers in Europe. The results showed less than 10% of patients belonging to the poor and very poor category (Mars et al., 1987). In the current study, about 8% of patients in the TF group are likely to need an osteotomy for class III occlusion (grade 4 and grade 5). None of the patients in the VF group were in the poor or very poor growth category. However, these children have to be followed up until age 16 years to obtain the final results. Vomer flap surgery involves the suture line of the vomer bone, which is supposed to be an important site for midface growth. Over the years, argument has been raised about the possible interference of vomer flap surgery with midface growth in children, resulting in a drastic decline in its surgical use (Friede and Johanson, 1977). However, more recent evidence suggests that septal resection,
refashioning, and re-implantation procedures do not produce any significant facial growth disturbance (Siegel and Sadler, 1981; Walker et al., 1993). Use of the vomer flap as a single layer for hard palate repair obviates the need for extensive dissection during subsequent palatoplasty, resulting in less midfacial growth disturbances (Ferdous et al., 2010). Without the use of the vomerine flap for the anterior hard palate, there may be a greater need for mucoperiosteal mobilization to close both the hard and soft palates at a later stage (Hathorn et al., 1996). Studies have compared the growth outcome following vomer flap versus single-stage palatoplasty. Xue et al. (2015) in their study concluded that vomer flap repair inhibited both sagittal and vertical growth of the maxilla compared to delayed hard palate repair. However the authors emphasized delaying the final evaluation until the growth of the facial skeleton is complete. Nollet et al. (2005) in their study concluded that the treatment protocol used in Eurocleft centers did not have major influence on the dental arch relationship. However, these are not randomized trials involving one surgeon, but intercenter comparisons in which an important confounding factor was “the surgeon.” Caseload and skill of the surgeon were said to be more important factors in the quality of the outcomes. Poor GOSLON ratings have been attributed to unsuccessful surgical procedures and factors such as low-volume operators (Bearns et al., 2001). The confounding factor “the surgeon” was avoided in the present study. The dental arch relationships of patients in the current study were compared with data from six centers from the Eurocleft study and other previous studies (Fudalej et al., 2009) (Fig. 2). Comparison was also made with the data from the Americleft study (Hathway et al., 2011) (Fig. 3). There have been numerous studies about the incidence of malocclusion in individuals without cleft in the Indian population. Trehan et al. (2009) reported a 3.17% incidence of class III malocclusion in subjects in the age group 10e13 years. In another study by Sandhu et al. (2012), class III malocclusion was 3.4%. Of 85
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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maxillary growth evaluation after completion of growth is necessary. Zemann et al. (2007, 2011), in their intercenter study on sagittal growth of facial skeleton in individuals treated with two different protocols, noted a similar growth pattern in both groups when the individuals were evaluated at 6 years and 10 years age. The growth was not affected by different surgical protocols. The authors concluded that a re-evaluation is needed after the final growth spurt, which is appropriate for the present study. The range of timing of palate repair (9e18 months) is rather wide. No stratification was done based on the age at which the palate repair was carried out. Due to limited experience with the VF technique, the surgeon encountered a short palate with scarring at the junction area, making palate repair more challenging. The vomer flap technique was later modified to improve lengthening of palate. 5. Conclusion
Fig. 2. Comparison of the mean GOSLON Yardstick score distribution in this study and in previous reports.
patients in the current study group, 8.8% belonged to the GOSLON score categories of 4 and 5, and thus there was a corresponding number of children with cleft with class III malocclusion. Hence, it can be inferred that the percentage of incidence of class III malocclusion in a population with cleft is almost double that of the class III malocclusion occurring in a population without such a cleft. This study has several limitations. There was severe attrition in the sample size over the length of the study. Only 42.5% of the initial sample size completed the protocol and were available for evaluation. This could be attributed to the long duration and the associated family, social, and other factors that are beyond the control of the investigating team. However the possibility of attrition was anticipated at the beginning of the study, and hence a large sample size was selected. GOSLON Yardstick scoring as a method of growth assessment can overestimate horizontal jaw disharmony if a vertical growth restriction element of the maxilla is present (Mars et al., 1987). The age of evaluation (7e9 years) for both speech and growth, especially growth, means that the result is not the final outcome. The main sagittal growth of the maxilla occurs after second dentition through puberty, and hence further follow-up and
Fig. 3. Comparison of the GOSLON Yardstick score distribution in this study and the Americleft study.
Statistically significant difference was obtained between the VF and TF techniques in terms of GOSLON scores and speech assessment. Patients in the VF group showed slightly better growth potential than those in the TF, whereas the patients in the TF group showed better speech outcomes. Further research should be done considering complete growth development and also in larger population of patients. At present, the statistically found differences between the two groups are very small, and hence neither of the protocols can be considered superior over the other. We shall present our long-term outcome of these individuals and also speech outcomes with a new modified vomer flap technique in the future. Our findings also imply that there are more complex embryological processes resulting in clefts of different types, even though clinically they may appear to be similar. These complexities invariably play a role in the overall outcome of cleft lip and palate management. Author contributions 1 Dr. Praveen G: Analysing the facial growth outcome and collecting data 2 Ms. Navitha: Analysing speech and collecting speech outcome data 3 Ms. Savitha H: Analysing speech and collecting speech outcome data 4 Dr. Jyotsna Murthy: Surgeon leading this research and executed all surgeries References Agrawal K, Panda KN: Use of vomer flap in palatoplasty: revisited. Cleft Palate Craniofac J 43: 30e37, 2006 Bearns D, Midinhall S, Murphy T, Murray JJ, Sell D, Shaw WC, et al: Cleft lip and palate care in the United Kingdomdthe Clinical Standards Advisory Group (CSAG) Study. Part 4: outcome comparisons, training, and conclusions. Cleft Palate Craniofac J 1: 38e43, 2001 Ferdous KMN, Salek AJM, Das BK, Karim MS: Repair of cleft lip and simultaneous repair of cleft hard palate with vomer flap in unilateral complete cleft lip and palate: a comparative study. Pediatr Surg Int 26: 995e1000, 2010 Friede H, Johanson B: A follow-up study of cleft children treated with vomer flap as part of a three-stage soft tissue surgical procedure. Facial morphology and dental occlusion. Scand J Plast Reconstr Surg 11: 45e57, 1977 Fudalej P, Dzierzbicka HM, Dudkiewicz Z, Semb G: Dental arch relationship in children with complete unilateral lip and palate following Warsaw (one-stage repair) and Oslo protocols. Cleft Palate Craniofac J 46: 648e653, 2009 Hathorn I, Roberts-Harry D, Mars M: The GOSLON Yardstick applied to a consecutive series of patients with unilateral clefts of the lip and palate. Cleft Palate Craniofac J 33: 494e496, 1996 Hathway R, Daskalogiannakis J, Mercado A, Russel K, Long E Ross, Cohen M, et al: The Americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palatedpart 2. Dental arch relationships. Cleft Palate Craniofac J 48: 244e251, 2011
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Pradel W, Senf D, Mai R, Ludicke G, Eckelt U, Lauer G: One-stage palate repair improves speech outcome and early maxillary growth in patients with cleft lip and palate. J Physiol Pharmacol Suppl 8: 37e41, 2009 Sandhu SS, Bansal N, Sandhu N: Incidence of malocclusion in India. A review. J Oral Health Comm Dent 6: 21e24, 2012 Sandy JR, Williams AC, Bearn D, Mildinhall S, Murphy T, Sell D, et al: Cleft lip and palate care in the United Kingdomdthe Clinical Standards Advisory Group (CSAG) Study. Part 1: background and methodology. Cleft Palate Craniofac J 38: 20e30, 2001 Sell D: Issues in perceptual speech analysis in cleft palate and related disorders: a review. Int J Lang Comm Dis 40: 103e121, 2005 Shaw WC, Semb G, Nelson P, Brattstrom V, Molsted K, Prahl-Andersen B, et al: The eurocleft Project 1996-2000: overview. J Craniomaxillofac Surg 29: 131e142, 2001 Shaw WC, Brattstrom V, Molsted K, Prahl-Anderson B, Roberts CT, Semb G: The Eurocleft study: intercenter study of treatment outcome in patients with complete cleft lip and palate. Part 5: discussion and conclusions. Cleft Palate Craniofac J 42: 93e98, 2005 Siegel MI, Sadler D: Nasal septum resection and craniofacial growth in a chimpanzee animal model: implications for cleft palate surgery. Plast Reconstr Surg 68: 849e853, 1981 Trehan M, Chugh VK, Sharma S: Prevalence of malocclusion in Jaipur, India. Int J Clin Pediatr Dent 1: 23e25, 2009 Walker PJ, Crysdate WS, Farkas LG: External septoplasty in children: outcome and effect on growth of septal excision and reimplantation. Arch Otolaryngol Head Neck Surg 119: 984e989, 1993 Xue X, Kwon HJ, Shi B, Zheng Q, Yin H, Li C: Influence of different palate repair protocols on facial growth in unilateral complete cleft lip and palate. J Craniomaxillofac Surg 43: 43e47, 2015 Zemann W, Mossbock R, Karcher H, Kozelj V: Sagittal growth of the facial skeleton of 6-year-old children with a complete unilateral cleft of lip, alveolus and palate treated with two different protocols. J Craniomaxillofac Surg 35: 343e349, 2007 Zemann W, Karcher H, Drevensek M, Kozelj V: Sagittal maxillary growth in children with unilateral of the lip, alveolus and palate at the age of 10 years: an intercentre comparison. J Craniomaxillofac Surg 39: 469e474, 2011
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A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition Praveen Ganesh a, *, Jyotsna Murthy b, Navitha Ulaghanathan c, V.H. Savitha c a b c
Department of Cranio-Maxillofacial Surgery, Mazumdar Shaw Medical Center, Narayana Health City, Bommasandra, Bangalore, Karnataka, 560099, India Department of Plastic and Reconstructive Surgery, Sri Ramachandra University, Chennai, India Department of Speech, Language & Hearing Sciences, Sri Ramachandra University, Chennai, India
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 18 September 2014 Accepted 26 March 2015 Available online xxx
Objective: To study the growth and speech outcomes in children who were operated on for unilateral cleft lip and palate (UCLP) by a single surgeon using two different treatment protocols. Material and methods: A total of 200 consecutive patients with nonsyndromic UCLP were randomly allocated to two different treatment protocols. Of the 200 patients, 179 completed the protocol. However, only 85 patients presented for follow-up during the mixed dentition period (7e10 years of age). The following treatment protocol was followed. Protocol 1 consisted of the vomer flap (VF), whereby patients underwent primary lip nose repair and vomer flap for hard palate single-layer closure, followed by soft palate repair 6 months later; Protocol 2 consisted of the two-flap technique (TF), whereby the cleft palate (CP) was repaired by two-flap technique after primary lip and nose repair. GOSLON Yardstick scores for dental arch relation, and speech outcomes based on universal reporting parameters, were noted. Results: A total of 40 patients in the VF group and 45 in the TF group completed the treatment protocols. The GOSLON scores showed marginally better outcomes in the VF group compared to the TF group. Statistically significant differences were found only in two speech parameters, with better outcomes in the TF group. Conclusions: Our results showed marginally better growth outcome in the VF group compared to the TF group. However, the speech outcomes were better in the TF group. © 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Mixed dentition Growth Speech outcomes Vomer flap Two-flap technique
1. Introduction The approximate incidence of CLP is 1.3 per 1000 live births in India (Mossey and Little, 2009). There are numerous techniques described for management of patients with CLP. However, there is sparse information relating to specific management, intervention methods, and long-term outcomes in patients who have completed a strict treatment protocol. The confounding factors and inconclusive outcomes found in the available literature from around the world have led to the development of many protocols; for instance, there existed as many as 194 protocols in 205 European centers in 2001 (Shaw et al., 2001). Inter-center comparisons
* Corresponding author. E-mail address: [email protected] (P. Ganesh).
have limitations in that they cannot distinguish the relationship between protocols of different centers or the elements of a center's protocol on the outcome, and the influence of the personnel delivering that protocol (Shaw et al., 2005). One possible way to overcome the above limitations is by conducting a randomized controlled trial in subjects from a single center operated on by a single surgeon. One of the surgical protocols for patients with UCLP is using a vomer flap for the cleft of the hard palate while repairing the cleft lip. The advantages of using vomer flap include simplicity and ease of execution, without adding to surgical trauma or prolonging surgical time. This technique aids in providing an effective nasal lining in almost all types of clefts (Kobus, 1987). A debate still exists regarding the relationship between use of vomer flap as a single lining for the hard palate and defective mid-face growth (Agrawal
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Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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and Panda, 2006). There is a deficit of randomized control trials using the vomer flap as a single layer for the hard palate repair in comparison to other techniques such as the Veau-Wardill-Kilner, Von Langenbeck, and two-flap techniques, among others. This study was a single-center, prospective, randomized controlled study. A comparative assessment of treatment outcomes of nonsyndromic UCLP patients with two different surgical protocols, namely, the vomer flap (VF) versus the two-flap technique (TF), was carried out. Keeping in mind the large case load being operated on by a single surgeon with no prior commitment to either of the techniques, the randomized controlled trial was deemed appropriate. This design would also suggest how the techniques would compare in the hands of a single surgeon. Success in cleft lip and palate surgery cannot be judged only by the esthetic outcomes, but should also consider functional parameters such as speech and dental arch relationships (Pradel et al., 2009; Hathorn et al., 1996; Mars and Houston, 1990). The two techniques in this study were thus evaluated and compared based on the dental arch relationship, speech outcomes, and occurrence of fistula. It was hypothesized that there would be no significant differences between the two surgical protocols across these parameters. 2. Material and methods After obtaining ethical clearance from Ramachandra University institutional ethics committee (Reference number- IEC/NI/03/MAY/ 13/33), 200 consecutive patients with nonsyndromic UCLP were included in this randomized trial from 2003 to 2005. The parents received explanations about the proposed study in understandable language. The parents were also informed that the standard of care would not be compromised if they decided to opt out of the study. None of the parents declined to join the study. Randomization was done by allocation concealment, whereby 200 chits were put in a box (100 for each group) and the parent or guardian was asked to pick one chit 1 day before the surgery. The patient was allotted to the treatment protocol as indicated in the chit. Fifteen children who did not complete the two surgeries as suggested in the protocols and 6 children operated on by more than one surgeon were excluded. Of the 179 patients who completed treatment for lip and palate repair, operated on by a single surgeon, only 85 (40 patients in the VF group and 45 patients in the TF group) came for regular follow-up through the period of mixed dentition. These patients were in the age range of 7e9 years at the time of last follow-up. Details on the mean age of intervention and follow-up of the participants is summarized in Table 1. For various reasons, speech samples were obtained from only 34 patients in the VF group and 39 in the TF group. Two protocols were selected for randomization. In the VF group, the cleft lip was repaired using the Millard technique along with nose correction. The vomer flap was used as a single layer for hard palate closure. After 6 months, soft palate repair was carried out with sharp separation of the muscle fibers from the enveloping oral and the nasal mucosa and from the hard palatal shelves. The tensor
Table 1 Mean age of intervention and outcomes of study Protocol
Vomer flap (VF)
Two-flap (TF)
Mean age of lip repair Mean age of palate repair Mean age of follow-up for dental arch relationship evaluation
5.22 Months 12.3 Months 7.8 Years
6.3 Months 12.9 Months 8.1 Years
tendon was released just medial to the hamulus, followed by retro positioning and plication of muscle bundles along the midline. During soft palate repair, minor to major lateral releasing incision (as in the von-Langenbeck technique), either unilateral or bilateral, was needed to close the junction area in 36 of 91 patients. In the TF group, cleft lip was repaired by the Millard technique with nose correction, and anterior palate repair up to the incisor foramen. Six months later, the palate was repaired with two-flap palatoplasty. The surgeon had experience of more than 10 years in using the TF technique, whereas the VF technique was introduced in practice only 1 year before the commencement of the randomized trial. None of the children had either preoperative or postoperative orthopedic intervention during the mixed dentition. All patients underwent routine speech evaluation between 4 and 6 years of age. Based on the profile of articulation, they were provided with three to five sessions of speech therapy at the hospital, focusing on demonstrating correction of specific articulation errors to the parents. Home training programs were recommended for correction of articulation. None of them received long-term institution-based speech therapy for correction of speech errors. To study the maxillary growth outcomes, digital intraoral photographs were taken for all patients during mixed dentition. The set of intraoral photographs included the frontal view in occlusion, right and left buccal views, and right and left overjet views. History of palatal fistula was taken from the records. Speech samples of all patients were audio-recorded by a speech pathologist in a soundtreated room. Recognizing the need for a comprehensive speech sample (Sell, 2005; Kuehn and Moller, 2000), the recorded sample comprised a 2-min conversation, counting of numbers from 1 to 10, syllable repetition, repetition of phonetically loaded words, and sentences in the Tamil language. All children passed hearing screening (pure tone average of less than 20 dBHL) at the time when the speech samples were recorded. However, the status of the middle ear was not examined. Mars et al. (1987) published a simple method named the GOSLON (an acronym denoting “Great Ormond Street, London and Oslo”) Yardstick to score the outcome of treatment in patients with unilateral cleft lip and palate. The outcome of treatment is viewed on occlusion and scored by experienced raters. Patients are categorized into one of the following five groups: group 1: positive overjet with average inclined or retroclined incisors with no crossbite or open bite with excellent long-term outcome; group 2: positive overjet with average inclined or proclined incisors with unilateral cross-bite or cross-bite tendency with or without open bite tendency around the cleft site with good long-term outcome; group 3: edge-to-edge bite with average inclined or proclined incisors or reverse overjet with retroclined incisors with unilateral cross-bite with or without open bite tendency around the cleft site, with fair long-term outcome; group 4: reverse overjet with average inclined or proclined incisors, with or without bilateral cross-bite tendency with or without open bite tendency around the cleft site, with poor outcome; and group 5, reverse overjet with proclined incisors, bilateral cross-bite, and poor maxillary arch form and palatal vault anatomy with very poor outcome (Mars et al., 2006). The GOSLON Yardstick does not involve application of precise and detailed criteria, but relies on a simple method of judgment (Lilja et al., 2006). Two examiners, a surgeon and an orthodontist, who were not members of the cleft team, scored the intraoral digital photographs on two separate occasions 2 weeks apart (Fig. 1). Each examiner was calibrated previously in the use of the GOSLON Yardstick to reduce systematic bias. The examiners were given a reference image of GOSLON score 1 to 5 as a guide to categorize the photographs during the rating (Liao et al., 2009). No conferring between examiners was allowed, and an overall GOSLON final score was
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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Table 3 Speech outcome parameters with significant differences between vomer flap (VF) and two-flap (TF) groups. Parameter
Grade
VF group
TF group
p Value
Hypernasality
Normal Mild Moderate Absent Present Absent Present
11.8% 70.6% 17.6% 58.8% 41.2% 58.8% 41.2%
20.5% 76.9% 2.6% 84.6% 15.4% 84.6% 15.4%
0.05
Weak oral pressure words Weak oral pressure sentences
Fig. 1. Distribution of GOSLON Yardstick scores between the two groups: vomer flap (VF) and two-flap (TF).
allocated to each set of photographs. Examiners were blinded to the sequence of surgeries performed in the patients. Two speech language pathologists, who were experienced in the area of cleft lip and palate, independently evaluated the speech samples. Speech samples were blinded and randomized before perceptual evaluation. Universal parameters for reporting speech outcomes in individuals with cleft described by Henningsson et al. (2008) were used for perceptual assessment. The rating of hypernasality was performed only at the level of sentences, and not using isolated words. All other parameters (hyponasality, nasal air emission/nasal turbulence, consonant production errors, speech understandability, speech acceptability, and presence of voice disorder) were rated as specified by Henningsson et al. (2008). The scores obtained were tabulated and subjected to statistical analysis. The linear weighted kappa statistical test (computing a lower 95% confidence interval) was used to evaluate intra- and interrater reliability of the GOSLON scores. An independent sample Student ttest was used to compare the GOSLON scores between the two groups. The interrater reliability using kappa statistics to determine the consistency among two speech evaluators showed substantial to almost perfect agreement across parameters (Table 2). The ManneWhitney U test was used to compare the speech ratings between the two groups. 3. Results The VF group included 24 male patients and 16 female patients. There was an almost equal distribution in the TF group, with 23 male and 22 female patients. The distribution of GOSLON Yardstick scores in both the groups is depicted in Fig. 1. Both intra- and interexaminer agreement were high, as indicated by weighted kappa coefficients of 0.85 and 0.90 respectively. In the VF group, 70% of the patients demonstrated
Table 2 Inter- and intrarater reliability of speech analysis. Substantial agreement
Moderate agreement
Velar words (0.789) Weak oral pressures words (0.708) Nasalized voiced pressure consonant (0.660) Developmental articulation error words(0.677) Speech understandability (0.703) Speech acceptability (0.628) Voice disorder (0.671)
Hypernasality sentences (0.551) Mid dorsum palatal words (0.489) Other words and sentences (0.490) Almost perfect agreement Glottal words and sentences (0.916) Nasal fricative words (0.850) Fair agreement Nasal air emission words (0.366)
0.014 0.014
good growth (GOSLON scores 1 and 2) and 30% revealed adequate growth (GOSLON score 3). In the TF group, 54% of the patients displayed good growth, 37.7% had the adequate growth category, and 8.8% had poor growth (GOSLON score 4). None of the patients from either group were found to have very poor GOSLON scores (GOSLON score 5). The mean GOSLON scores were 2.15 (SD ¼ 0.662) and 2.49 (SD ¼ 0.757) for the VF and TF group respectively (t ¼ 2.183). This difference was statistically significant (p ¼ 0.032) based on the analysis using the Student t-test. One patient in the VF group had a palatal fistula, whereas no patient in the TF group had a palatal fistula. This fistula was repaired before the speech sample was taken for analysis. None of these patients had undergone any corrective surgeries for velopharyngeal dysfunction. The ManneWhitney U test revealed a significant difference between the VF and TF groups in speech outcomes on parameters including hypernasality (p ¼ 0.05) and weak oral pressure consonants at the level of words and sentences (p ¼ 0.014), with better outcomes in the TF group (Table 3). In the VF group, 11.8% of patients had normal resonance and 17.6% had moderate hypernasality. In the TF group, 20.5% of patients had normal resonance and 2.6% showed moderate hypernasality. Weak oral pressure consonants were perceived in 41% and 15% of the VF and TF group, respectively. The difference in various parameters of speech and their statistical significance is presented in Table 4. 4. Discussion The outcome of primary repair surgery for cleft lip and palate is often expressed in the subsequent quality of speech, hearing, facial appearance and the dentoskeletal relationships (Johnston et al., 2004). Comparison of two different surgical technique protocols in a single center with a single operating surgeon offers an objective method of evaluating the efficacy of a particular technique. In this study, a randomized controlled trial was initiated for the purpose of comparing two protocols for unilateral complete cleft lip and palate. To date, dental arch relationships for measuring growth outcomes and speech assessment have been considered as the two most useful benchmarks to assess outcomes in the management of children with CLP (Sandy et al., 2001; Sell, 2005). Intraoral photographs provide a valid and reliable method for rating dental arch relationships in patients with UCLP at the age of 9 years and provide viable alternative to the application of the GOSLON Yardstick on dental casts (Liao et al., 2009). A significant difference was observed in growth outcomes between the VF and TF groups, with better results in the VF. This difference is likely to increase with further maxillary growth (Liao et al., 2013). Landheer et al. (2010) reported that two-stage cleft palate repair had a fistula incidence of 27%; whereas one-stage cleft repair had a fistula occurrence of 14%. In the current study, one of the 40 patients in the VF group had a palatal fistula, whereas none had in the TF group had a palatal fistula. The fistula in the VF group occurred at the junction of the hard and soft palate. The incidence of fistula in
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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Table 4 Speech outcome analysis. Parameter
Vomer flap (mean rank)
Nonevomer flap (mean rank)
p Value
Hypernasality Hyponasality Nasal air emission words Nasal air emission sentences Pharyngeal words Pharyngeal sentences Glottal words Glottal sentences Mid dorsum palatal word Mid dorsum palatal sentences Velar words Velar sentences Uvular words Uvular sentences Nasal fricative words Nasal fricative sentences Nasal consonant for oral pressure consonant words Nasal consonant for oral pressure consonant sentences Nasalized voiced pressure consonant words Nasalised voiced pressure consonant sentences Weak oral pressure words Weak oral pressure sentences Developmental/Arti error words Developmental/Arti error sentences Other words Other sentences Speech understandability Speech acceptability Voice disorder
41 36.50 36.81 35.81 36.50 36.50 39.44 39.44 36.57 36.57 33.74 33.24 36.50 36.50 37.72 37.72 36.50 36.50 36.0 35.50 42.03 42.03 38.90 38.90 35.50 35.50 37.59 35.49 35.15
33.51 37.44 37.17 38.04 37.44 37.44 34.87 34.87 37.37 37.37 39.85 40.28 37.44 37.44 36.37 36.37 37.44 37.44 37.87 38.31 32.62 32.62 35.35 35.35 38.31 38.31 36.49 38.32 38.62
.05 .35 .93 .589 .35 .35 .09 .09 .64 .64 .145 .095 .35 .35 .536 .536 .35 .35 .184 .101 .014 .014 .410 .410 .101 .101 .803 .519 .198
the VF group in this study could probably be attributed to the skills of the surgeon because it was a newly added technique in comparison with the 10 years of experience with the two-flap technique. There have been no randomized controlled trials in the literature comparing the speech outcome between groups of patients treated with the vomer flap and two-flap technique. In the current study (VF, 34 subjects; TF, 39 subjects), significant differences between the two groups were observed only in the presence of hypernasality and weak oral pressure consonants, with better speech outcomes in the TF group. Patients in the VF group had a higher percentage of hypernasality of moderate degree (17.6%) compared to patient in the TF group (2.6%). This difference could probably be attributed to the relatively new technique for the surgeon in the VF group. It was observed that soft palates were becoming shorter due to scarring in the area. After completion of the randomized trial, the technique was modified to overcome this shortcoming. The Norwegian center at the Riks Hospital in Oslo, Norway, popularized the technique of lip and vomer flap, followed by closure of the rest of the palate 3 months later. The growth results in their study measured using the GOSLON Yardstick compare well with results from other centers in Europe. The results showed less than 10% of patients belonging to the poor and very poor category (Mars et al., 1987). In the current study, about 8% of patients in the TF group are likely to need an osteotomy for class III occlusion (grade 4 and grade 5). None of the patients in the VF group were in the poor or very poor growth category. However, these children have to be followed up until age 16 years to obtain the final results. Vomer flap surgery involves the suture line of the vomer bone, which is supposed to be an important site for midface growth. Over the years, argument has been raised about the possible interference of vomer flap surgery with midface growth in children, resulting in a drastic decline in its surgical use (Friede and Johanson, 1977). However, more recent evidence suggests that septal resection,
refashioning, and re-implantation procedures do not produce any significant facial growth disturbance (Siegel and Sadler, 1981; Walker et al., 1993). Use of the vomer flap as a single layer for hard palate repair obviates the need for extensive dissection during subsequent palatoplasty, resulting in less midfacial growth disturbances (Ferdous et al., 2010). Without the use of the vomerine flap for the anterior hard palate, there may be a greater need for mucoperiosteal mobilization to close both the hard and soft palates at a later stage (Hathorn et al., 1996). Studies have compared the growth outcome following vomer flap versus single-stage palatoplasty. Xue et al. (2015) in their study concluded that vomer flap repair inhibited both sagittal and vertical growth of the maxilla compared to delayed hard palate repair. However the authors emphasized delaying the final evaluation until the growth of the facial skeleton is complete. Nollet et al. (2005) in their study concluded that the treatment protocol used in Eurocleft centers did not have major influence on the dental arch relationship. However, these are not randomized trials involving one surgeon, but intercenter comparisons in which an important confounding factor was “the surgeon.” Caseload and skill of the surgeon were said to be more important factors in the quality of the outcomes. Poor GOSLON ratings have been attributed to unsuccessful surgical procedures and factors such as low-volume operators (Bearns et al., 2001). The confounding factor “the surgeon” was avoided in the present study. The dental arch relationships of patients in the current study were compared with data from six centers from the Eurocleft study and other previous studies (Fudalej et al., 2009) (Fig. 2). Comparison was also made with the data from the Americleft study (Hathway et al., 2011) (Fig. 3). There have been numerous studies about the incidence of malocclusion in individuals without cleft in the Indian population. Trehan et al. (2009) reported a 3.17% incidence of class III malocclusion in subjects in the age group 10e13 years. In another study by Sandhu et al. (2012), class III malocclusion was 3.4%. Of 85
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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maxillary growth evaluation after completion of growth is necessary. Zemann et al. (2007, 2011), in their intercenter study on sagittal growth of facial skeleton in individuals treated with two different protocols, noted a similar growth pattern in both groups when the individuals were evaluated at 6 years and 10 years age. The growth was not affected by different surgical protocols. The authors concluded that a re-evaluation is needed after the final growth spurt, which is appropriate for the present study. The range of timing of palate repair (9e18 months) is rather wide. No stratification was done based on the age at which the palate repair was carried out. Due to limited experience with the VF technique, the surgeon encountered a short palate with scarring at the junction area, making palate repair more challenging. The vomer flap technique was later modified to improve lengthening of palate. 5. Conclusion
Fig. 2. Comparison of the mean GOSLON Yardstick score distribution in this study and in previous reports.
patients in the current study group, 8.8% belonged to the GOSLON score categories of 4 and 5, and thus there was a corresponding number of children with cleft with class III malocclusion. Hence, it can be inferred that the percentage of incidence of class III malocclusion in a population with cleft is almost double that of the class III malocclusion occurring in a population without such a cleft. This study has several limitations. There was severe attrition in the sample size over the length of the study. Only 42.5% of the initial sample size completed the protocol and were available for evaluation. This could be attributed to the long duration and the associated family, social, and other factors that are beyond the control of the investigating team. However the possibility of attrition was anticipated at the beginning of the study, and hence a large sample size was selected. GOSLON Yardstick scoring as a method of growth assessment can overestimate horizontal jaw disharmony if a vertical growth restriction element of the maxilla is present (Mars et al., 1987). The age of evaluation (7e9 years) for both speech and growth, especially growth, means that the result is not the final outcome. The main sagittal growth of the maxilla occurs after second dentition through puberty, and hence further follow-up and
Fig. 3. Comparison of the GOSLON Yardstick score distribution in this study and the Americleft study.
Statistically significant difference was obtained between the VF and TF techniques in terms of GOSLON scores and speech assessment. Patients in the VF group showed slightly better growth potential than those in the TF, whereas the patients in the TF group showed better speech outcomes. Further research should be done considering complete growth development and also in larger population of patients. At present, the statistically found differences between the two groups are very small, and hence neither of the protocols can be considered superior over the other. We shall present our long-term outcome of these individuals and also speech outcomes with a new modified vomer flap technique in the future. Our findings also imply that there are more complex embryological processes resulting in clefts of different types, even though clinically they may appear to be similar. These complexities invariably play a role in the overall outcome of cleft lip and palate management. Author contributions 1 Dr. Praveen G: Analysing the facial growth outcome and collecting data 2 Ms. Navitha: Analysing speech and collecting speech outcome data 3 Ms. Savitha H: Analysing speech and collecting speech outcome data 4 Dr. Jyotsna Murthy: Surgeon leading this research and executed all surgeries References Agrawal K, Panda KN: Use of vomer flap in palatoplasty: revisited. Cleft Palate Craniofac J 43: 30e37, 2006 Bearns D, Midinhall S, Murphy T, Murray JJ, Sell D, Shaw WC, et al: Cleft lip and palate care in the United Kingdomdthe Clinical Standards Advisory Group (CSAG) Study. Part 4: outcome comparisons, training, and conclusions. Cleft Palate Craniofac J 1: 38e43, 2001 Ferdous KMN, Salek AJM, Das BK, Karim MS: Repair of cleft lip and simultaneous repair of cleft hard palate with vomer flap in unilateral complete cleft lip and palate: a comparative study. Pediatr Surg Int 26: 995e1000, 2010 Friede H, Johanson B: A follow-up study of cleft children treated with vomer flap as part of a three-stage soft tissue surgical procedure. Facial morphology and dental occlusion. Scand J Plast Reconstr Surg 11: 45e57, 1977 Fudalej P, Dzierzbicka HM, Dudkiewicz Z, Semb G: Dental arch relationship in children with complete unilateral lip and palate following Warsaw (one-stage repair) and Oslo protocols. Cleft Palate Craniofac J 46: 648e653, 2009 Hathorn I, Roberts-Harry D, Mars M: The GOSLON Yardstick applied to a consecutive series of patients with unilateral clefts of the lip and palate. Cleft Palate Craniofac J 33: 494e496, 1996 Hathway R, Daskalogiannakis J, Mercado A, Russel K, Long E Ross, Cohen M, et al: The Americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palatedpart 2. Dental arch relationships. Cleft Palate Craniofac J 48: 244e251, 2011
Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
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Please cite this article in press as: Ganesh P, et al., A randomized controlled trial comparing two techniques for unilateral cleft lip and palate: Growth and speech outcomes during mixed dentition, Journal of Cranio-Maxillo-Facial Surgery (2015), http://dx.doi.org/10.1016/ j.jcms.2015.03.033
