ONE-STAGE PALATE REPAIR IMPROVES SPEECH OUTCOME AND EARLY MAXILLARY GROWTH IN PATIENTS WITH CLEFT LIP AND PALATE

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1 JOURNAL OF PHYSIOLOGY AND PHARMACOLOGY 2009, 60, Suppl 8, W. PRADEL 1, D. SENF 2, R. MAI 1, G. LUDICKE 3, U. ECKELT 1, G. LAUER 1 ONE-STAGE PALATE REPAIR IMPROVES SPEECH OUTCOME AND EARLY MAXILLARY GROWTH IN PATIENTS WITH CLEFT LIP AND PALATE 1 Department of Oral and Maxillofacial Surgery, University Hospital Carl Gustav Carus Dresden Technical University Dresden, Germany; 2 Department of Otorhinolaryngology, University Hospital Carl Gustav Carus, Technical University Dresden, Dresden, Germany; 3 Department of Orthodontics, University Hospital Carl Gustav Carus, Technical University Dresden, Dresden, Germany There are several types of palatal surgery; each cleft centre chooses its own technique based on experience and treatment philosophy. The aim of this study was to compare speech outcome and maxillary growth in children with cleft lip and palate deformity after palate repair with either a one-stage or a two- stage procedure and to identify the better treatment protocol. In 24 children, speech outcome was assessed regarding resonance, nasal escape, compensatory articulations, facial grimace, and spontaneous speech. In addition, plaster models of 15 children were compared. In 12 children, a twostage procedure was performed (group A): at the age of 9-12 months, an intravelar veloplasty for repair of the soft palate, and at the age of months a bipedicled flap closure of the hard palate. In 12 children, the same techniques were used in a one-stage procedure, at the age of 9-12 months (group B). The children of group B showed less altered resonance and less nasal emission at 4 years of age compared to the children of group A. At 6 years, the children of group A had improved their speech skills, but they did not equal the results of group B. In the study models of group A at age 6 years, the transverse dimension (anterior and posterior width of the dental arch) was smaller than in the models of group B. The one-stage repair of cleft palate at the age of 9-12 months seems to have a more positive influence on speech development and early maxillary growth than the two-stage procedure. Key words: cleft lip and palate, speech outcome, early maxillary growth, palatoplasty, articulation disorder INTRODUCTION Success in cleft lip and palate surgery cannot only be judged by the aesthetic outcome, e.g. after lip repair, but also by assessing functional parameters, such as speech development after palate closure. The aim of palate repair is to create a complete closure, having an intact hard and soft palate with a normal functioning velopharyngeal mechanism. If not, there are resonance disorders, with an altered voice, compensatory articulation, and chronic middle ear infections (1). The timing of surgery and the choice of the operation technique leads to a dilemma. On the one hand, there is speech development and hearing capability, on the other there is the development of the maxilla. Speech and language therapists prefer to have the speech mechanism intact as soon as possible. Hardin-Jones and Jones (2) recommend an optimal treatment regimen for speech that includes primary palate repair no later than 13 months of age. However, wide undermining and development of mucoperiosteal flaps had the potential for impairing facial skeletal growth (3). Orthodontists would like to have a minimal amount of surgical scarring in the palatal area until facial growth is complete; therefore, delayed closure of the hard palate was recommended (4-5). A balance between these problems can be difficult. Established articulation disorder is difficult to treat through speech therapy (6). A single crossbite usually is not difficult to treat orthodontically (7). In a study published in 2001 about the efficacy of the different cleft centres in Europe, Shaw et al. (8) stated that 201 cleft centres have 194 different treatment protocols. Palate repair can be performed as a one- or a two-stage procedure. There are different opinions about the technique and the ideal timing of surgery. The range is 6 weeks up to 14 years (9). The aim of our study was to compare the speech outcome and early maxillary growth of children with cleft lip and palate deformity after a one-stage or after a two-stage palate repair, and to draw conclusions in respect to an optimal treatment protocol. PATIENTS AND METHODS Twenty-four children with non-syndromic unilateral/bilateral cleft lip and palate or complete cleft palate operated on at our cleft centre were included in the retrospective study. Five children had a bilateral cleft lip and palate, 15 children had a unilateral cleft lip and palate, and 4 children had a complete cleft palate. Surgical procedure Lip repair in children with cleft lips deformity was performed at an age of 4-6 months using a wave line incision according to Pfeifer.

2 38 In 12 children (group A, operated on from ), a two-stage procedure was performed consisting of an intravelar veloplasty according to Kriens (10), at between 9 and 12 months (mean: 10.5 months) and the von Langenbeck/Veau/Axhausen technique (11) for closure of the hard palate, at the age of 24 to 36 months (mean: 28.3 months). In 12 children (group B, operated on from ), a one-stage procedure was performed using similar operation techniques, at age 9 to 12 months (mean: 12.9 months). The intravelar veloplasty involved freeing the levator muscle from the posterior edge of the hard palate and three-layered closure across the midline. For closure of the hard palate, vomer flaps were used to form the nostril floors and bipedicled flaps were moved to the midline and sutured for oral cover. Speech assessment An experienced speech therapist is part of our cleft team. Speech was assessed twice; the first time was at an age of 4 years and the second time at 6 years. Patients were assessed for articulation development of simple sounds, words and connected sentences. All of the children underwent articulation therapy to different extents, depending on local conditions in their hometowns. We used the Muhler (12) classification, which was developed to assess cleft patients. The criteria are resonance, nasal escape, compensatory articulation, facial grimace, and spontaneous speech. Table 1 shows an overview of the assessment scores for each criterion. The scores are ranging from 0 as normal value to 3 as the worst value. In compensatory articulation, dental sounds and consonants were produced by articulatory movement to the palate (backing) and palatal sounds were produced using teeth and the alveolar ridge. Study model analysis Plaster dental models of 15 patients (8 patients in group A, 7 patients in group B) were compared at three stages: T1 immediately after birth, T2 at an age of 1 year, T3 at an age of 6 years. Eight children were excluded because they had complete bilateral clefts or complete cleft palate; one child was lost to follow-up. The models were evaluated in random order by one investigator (WP), blinded for surgical procedure using a landmark positioning technique described by Seckel et al. (13) and Mommaerts et al. (14). The surface of each cast was digitized two-dimensionally using a scanner (Snapscan 1236 S, Agfa, Germany) (15), landmarks were identified on the computer display and analysed with FR-Win software (Fa Computer konkret AG, Falkenstein, Germany). All measurements were performed twice and the arithmetic mean of the two values was recorded. The reference points are described in Figs. 1 and 2. Statistical analysis Means, medians, and standard deviations were calculated for all parameters. Nonparametric Mann Whitney U test was used for analysis of speech scores and for analysis of dental casts. Significance was defined at p<0.05. Table 1. Criteria and scores for speech assessment according to Muhler (1983).!!!! " " # " $ % & '" Fig. 1. Reference points of the casts at T1 (after birth) and T2 (at an age of 1 year): I incisal point, on the crest of the ridge on the line drawn from the labial frenulum to the incisive papilla, C/C - canine point, the intersection of the groove at the lateral labial frenulum and the crest of the ridge (C- C : width of the anterior arch), T/T tuberosity point at the intersection of the crest of the ridge and the outline of the maxillary tuberosity (T-T : width of the posterior arch), X intersection of the perpendicular directed from point I to the centre of the line T-T (I- X: length of the dental arch), P/P cleft edge point (P-P : width of the alveolar cleft), t/t point of intersection between the margins of the palatal shelves and the line T-T (tt : width of the palatal cleft).

3 39 Table 2. Comparison of mean values ± standard deviation of each score in speech outcome. 4 years 6 years Group A Group B Group A Group B Resonance 2.1±0.9** 0.9±1.0** 1.7± ±0.8 Nasal escape 1.3±1.1* 0.4±0.8* 0.6± ±1.0 Compensatory articulation (+) 2.2± ± ± ±1.1 Compensatory articulation (-) 0.5± ± ± ±1.0 Facial grimace 0.2± ± ± ±0.0 Spontaneous speech 3.3± ± ± ±0.8 Statistically significant (*p 0.05, ** p 0.01). Table 3. Percentage and number of children with normal speech in group A and B. 4 years 6 years Group A Group B Group A Group B Normal resonance 0% (0/12) 42% (5/12) 17% (2/12) 42% (5/12) No nasal escape 25% (3/12) 75% (9/12) 50% (6/12) 67% (8/12) No compensatory articulation 0% (0/12) 0% (0/12) 33% (4/12) 17% (2/12) No facial grimace 83% (10/12) 100% (12/12) 67% (8/12) 100% (12/12) Fig. 2. Reference points of the casts at T3 (at an age of 6 years): I - incisal point, on the crest of the ridge on the line drawn from the labial frenulum to the incisive papilla, CB/CB - most buccal point of the canine crown (CB - CB : width of the anterior arch), E/E - centre point of the occlusal surface of the deciduous molar (E-E : width of the posterior arch). Speech outcome RESULTS The results of speech outcome are summarized in Table 2 and 3. The assessment of resonance at 4 years of age shows a clear difference between the groups. Five children in group B have a normal resonance, none of the children in group A achieved such a good result. The mean score for resonance in group A was significantly higher (2.1±0.9) than in group B (0.9±1.0). In addition, in group B there was significantly less nasal escape (mean: 0,4±0,8) in comparison to group A (mean: 1.3±1.1). There were 9 of 12 children (75%) without nasal escape in group B in contrast to only 3 of 12 children (25%) in group A. The criteria compensatory articulation, facial grimace, and spontaneous speech revealed no significant differences between the groups. At the age of 6 years, the children of group A had improved their speech, but they did not equal the results of group B. The mean score for resonance was 1.7±1.3 in group A; in group B the mean score was nearly unchanged (0.8±0.7) compared to the mean score of 0.9±1.0 at 4 years of age. In group A, 2 of 12 children had unaltered resonance; in contrast to group B with 5 of 12 children. The criterion nasal escape showed equal mean scores in both groups. In group A, the number of children with no nasal escape was increased up to 50%. In compensatory articulation, facial grimace, and spontaneous speech, at the age of 6 years an improvement was observed, caused by a general development of the children, but no significant differences between both groups could be found. In 5 patients of group A, speech improving surgery was necessary, in contrast to none of the children in group B. Oronasal fistulae occurred in 1 child in Group A and in 3 children in Group B. All of this additional surgery was performed before

4 40 Table 4. Two-dimensional analysis of study models at T1, T2, and T3 (C-C rsp. CB-CB : anterior arch width, T-T resp. E-E : posterior arch width, I-X: dental arch length, P-P : alveolar cleft width, t-t : palatal cleft width). T1 (after birth) T2 (1 year) T3 (6 years) Group A Group B Group A Group B Group A Group B C - C 31.81± ± ± ±3.52 T - T 34.07± ± ± ±4.12 CB - CB 28.45±2.41** 31.87±1.97** E - E 35.70±3.63** 40.77±1.82** I - X 20.05± ± ± ± ±1.75* 19.38±1.10* P - P 8.39±4.48** 6.99±5.25** 1.27±1.30** 3.05±3.05** t - t 11.70± ± ± ±3.12 Statistically significant (*p 0.05, ** p 0.01). school entrance of the children. In both groups, blood transfusion was not necessary during cleft surgery. Maxillary growth Evaluation of all parameters at T1 (immediate after birth) and T2 (at an age of 1 year) showed a statistical significant difference in the width of the alveolar cleft (P-P ) between group A and group B. This difference is to be considered as random, because there was no preselection of the patients. The mean anterior width of the dental arch (C-C ) was similar at T1 (group A: 31.81±3.37, group B: 31.45±4.03) and at T2 (group A: 30.98±1.79, group B 31.88±3.52). Analysis of the casts concerning the posterior arch width (T-T ) at T1 and T2 also revealed no significant differences. In group A (two-stage) the mean value was 34.07±2.33 resp ±2.33 and in group B (one-stage) the mean value was 35.27±3.78 resp ±4.12. On analysis of the models at 6 years of age it was found, that the maxilla of group A (the two-stage) was smaller in the transverse dimension than those of group B. There was a significantly smaller anterior width of the arch (CB-CB ) in group A (28.45±2.41 vs ±1.97, p=0.001). In addition, the posterior dental arch width (E-E ) was significantly smaller in the two-stage group (35.7±3.63 vs ±1.82, p=0.000). After two-stage surgery the dental arch (I X) was significantly shorter (18.10±1.75) than after one-stage procedure (19.38±1.10). The results of the study model analysis are summarized in Table 4. DISCUSSION The present study compares a one-stage closure of the hard and the soft palate with a two-stage procedure, using the same surgical techniques in both groups. In the literature, most of the studies deal with different operating techniques and timing of surgery, therefore these publications are not directly comparable. The result of the present study is that the one-stage repair of cleft palate at the age of 9-12 months seems to have a more positive influence on speech development than the two-stage procedure particularly in cases of delayed palatal closure beyond 2 years of age. The Schwenkendiek procedure offers delayed hard palate closure to avoid early subperiosteal dissection and palatal scarring (4). After early repair of the soft palate (before 1 year of age) and delayed repair of the hard palate (after 5-6 years of age), a majority of the 32 children failed to develop acceptable speech spontaneously and required paryngeal flaps (16). Witzel et al. (6) stated in their review, that data for a beneficial maxillofacial growth response with delayed repair were lacking and noted severe speech problems with delayed palate closure. If closure of the palate is delayed beyond 2 years and the mechanisms for speech have already developed, the chances for normal speech are significantly diminished (17). Therefore, many cleft centres have abandoned this approach and instead are adopting an earlier closure of the hard palate, resulting in good results (9). The two-stage procedure has been compared to palatal closure in one operation, and better speech outcome was found in different studies. At the University Hospital of Gent, 103 adults were treated with two different protocols and the long-term speech outcome was assessed (18). One-stage Wardill-Killner palatoplasty had a significantly better speech outcome than the two-stage Furlow palatoplasty, therefore this technique was abandoned. In the Eurocleft Project, it was found that 45% of the cleft centres in Europe favour a one-stage procedure for closure of the palatal cleft (8). Assessment of speech outcome after one- or two-stage cleft repair, using a modified Furlow technique in 90 patients showed no differences between the groups. Therefore the current practice of these authors is the one-stage repair at between 10 and 12 months of age (19). Advantages for the child in the one-stage procedure are only one period of hospitalisation, less scars in the palate region, no dissection in scarred tissue of the soft palate as seen in the second surgery when using the two-stage repair and a better speech development as seen in both our results, and those in the literature. However, disadvantages such as a larger wound site with the risk of increased blood loss and airway obstruction have to be considered. Also in analysis of early maxillary growth, differences were found between the groups. In the study models at 1 year of age, the posterior dental arch in group A was slight smaller (not statistically significant) than in group B. The soft palate closure may cause this result. At 6 years of age, a significant reduction in the maxillary growth, in the transverse dimension, was observed in group A (two-stage). This result is surprising, as it is in contrast to the literature. We expected a disturbance in maxillary growth in group B, because an early one-stage repair in patients with isolated cleft palate before an age of 1 year, places the child at greater risk of disturbance of both transverse and anteroposterior maxillary arch development (20). In contrast, a two-centre study on 40 patients, in which no statistically significant difference in analysis of lateral cephalograms (angles SNA, SNB, ANB, SNPg) between the groups, at an age of 6 years, was observed. The treatment protocols were, one-stage palate repair at an age of 1 year versus two-stage surgery with closure of the soft palate at an age of 1 year and palatoplasty at 2.5 years (21). The transverse dimension was not evaluated in this study. However, differences in maxillary growth seem to disappear over time. In a two-centre study, 43 patients were analysed using two different treatment concepts. Twenty one patients underwent early one-stage closure at an age of 2 years; 21 patients had a delayed closure of the hard

5 41 palate at 7 years of age. The follow-up was until 18 years of age. In the analysis of the dental models, the anterior arch width was smaller in the one-stage group at an age of 6-10 years, but this difference could not be longer verified at the age of years (22, 23). However, the small number of patients in this study is not sufficient to draw any general conclusions. For assessment of the early maxillary growth, a twodimensional measurement procedure was chosen, because this method is appropriate for a quick, reliable cast analysis in patients with cleft lip and palate deformity. In a study on three- or two-dimensional cast analysis, repetitive linear measurements using a two-dimensional procedure, a standard deviation of 0.07 mm was calculated. The differences between the data from twoand three-dimensional analysis in the anterior region of the maxilla were 0.33 mm on average, and the authors stated this statistically significant result as clinically irrelevant (15). Crucial in the analysis of the models is the positioning of landmarks. The correct place for the landmark is unknown, because an observer always interprets its position from factors seen on casts. Seckel et al. (13) stated that an error of 0.8 mm in landmark positioning appears to be acceptable. In investigation of the intraobserver precision of landmark positioning, the error was mm, for interobserver precision it was mm (24). Therefore, in this study only highly significant values (p<0.01) were stated as appreciable difference between the groups. CONCLUSION The subjective and objective data from this retrospective analysis show a clear relationship between the treatment protocol (time of surgery and technique of palatal repair), speech outcome and early maxillary growth. The one-stage repair at the age of 9-12 months has a positive influence on speech development and early maxillary growth in contrast to the two-stage procedure. Therefore, we use the one-stage palate repair in our cleft centre and we recommend this approach, based on our experience and results. Conflict of interests: None declared. REFERENCES 1. Grant HR, Quiney RE, Mercer DM, Lodge S. Cleft palate and glue ear. Arch Di Child 1988; 63: Hardin-Jones MA, Jones DL. Speech production of preschoolers with cleft palate. Cleft Palate Craniofac J 2005; 42: Proff P, Weingartner J, Koppe T, Fanghanel J, Mack F, Gedrange T. Morphofunctional changes of orofacial muscles in patients with unilateral or bilateral cleft lip, alveolus and palate. Ann Anat 2007; 189: Schwenckendiek W. Primary veloplasty: long-term results without maxillary deformity. A twenty-five year report. Cleft Palate J 1978; 15: Hotz MM, Gnoinsky WM, Nussbaumer H, Kistler E. Early maxillary orthopedics in CLP cases: guidelines for surgery. Cleft Palate J 1978; 15: Witzel MA, Salyer KE, Ross RB. Delayed hard palate closure: the philosophy revisited. Cleft Palate J 1984; 21: Hotz MM. 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Precision of landmark positioning on digitized models from patients with cleft lip and palate. Cleft Palate Craniofac J 2006; 342: R e c e i v e d: October 22, 2009 A c c e p t e d: December 18, 2009 Author s address: Dr. Winnie Pradel, Department of Oral and Maxillofacial Surgery, University Hospital Carl Gustav Carus, Fetscherstrasse 74, Dresden, D 01307, Germany; Phone: ; Fax: ; winnie.pradel@uniklinikum-dresden.de