Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1224e1231
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One-stage (Warsaw) and two-stage (Oslo) repair of unilateral cleft lip and palate: Craniofacial outcomes Piotr Stanislaw Fudalej a, b, *, Ewa Wegrodzka c, Gunvor Semb d, Maria Hortis-Dzierzbicka e, f a
Department of Orthodontics and Dentofacial Orthopedics, University of Bern, (Head: Prof. Christos Katsaros), Freiburgstrasse 7, CH-3010 Bern, Switzerland Department of Orthodontics, Medical Faculty of Palacký University, (Head: Assoc. Prof. Milos Spidlen), Palackeho Str. 12, 772 00 Olomouc, Czech Republic Private Practice, Warsaw, Poland d School of Dentistry, University of Manchester, (Head: Prof. Paul Coulthard), Higher Cambridge Street, Manchester M15 6FH, United Kingdom e Laboratory of Speech Pathology and Upper Airway Endoscopy, Institute of Mother and Child, (Head: Assoc. Prof. Maria Hortis-Dzierzbicka), Kasprzaka Str. 17A, 01-211 Warsaw, Poland f Department of Otolaryngology and Maxillofacial Surgery, Universitary Clinical Hospital, (Head: Prof. Andrzej Kukwa), Warszawska Str. 30, 10-082 Olsztyn, Poland b c
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 11 November 2014 Accepted 30 April 2015 Available online 9 May 2015
The aim of this study was to compare facial development in subjects with complete unilateral cleft lip and palate (CUCLP) treated with two different surgical protocols. Lateral cephalometric radiographs of 61 patients (42 boys, 19 girls; mean age, 10.9 years; SD, 1) treated consecutively in Warsaw with one-stage repair and 61 age-matched and sex-matched patients treated in Oslo with two-stage surgery were selected to evaluate craniofacial morphology. On each radiograph 13 angular and two ratio variables were measured in order to describe hard and soft tissues of the facial region. The analysis showed that differences between the groups were limited to hard tissues e the maxillary prominence in subjects from the Warsaw group was decreased by almost 4 in comparison with the Oslo group (sella-nasion-Apoint (SNA) ¼ 75.3 and 79.1, respectively) and maxillo-mandibular morphology was less favorable in the Warsaw group than the Oslo group (ANB angle ¼ 0.8 and 2.8 , respectively). The soft tissue contour was comparable in both groups. In conclusion, inter-group differences suggest a more favorable outcome in the Oslo group. However, the distinctiveness of facial morphology in background populations (ie, in Poles and Norwegians) could have contributed to the observed results. © 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Cephalometrics Cleft lip Cleft palate Morphology Treatment
1. Introduction The treatment of cleft lip and palate (CLP) is far from uniform. Despite ongoing experimental and clinical research, numerous aspects of CLP care, such as the influence of non-surgical factors on maxillofacial morphology (Xu et al., 2014), the extension of primary surgery (Janiszewska-Olszowska, 2013), the role of various types of palatoplasties on facial growth (Xu et al., 2015) or who should judge the final outcome (Gkantidis et al., 2013), continue to be discussed. The results of two large multi-center comparative outcome studies: Eurocleft (Shaw et al., 2005) and Americleft (Russell et al.,
* Corresponding author. Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Freiburgstrasse 7, CH-3010 Bern, Switzerland. E-mail address: [email protected] (P.S. Fudalej).
2011) showed a diversity of outcomes e some participating centers produced more favorable results and some produced less favorable results. Due to the retrospective design of the first 9-year comparisons of Eurocleft (with prospective follow-up at 12 and 17 years) and Americleft, it was not possible to identify those elements of treatment which were responsible for the final outcome. Nevertheless, relatively good results obtained by centers which employ simpler and less burdensome protocols without the use of presurgical orthopedics or primary bone grafting, and with fewer operating surgeons, imply that demanding protocols may be unjustified. Treatment of complete unilateral cleft lip and palate (CUCLP) using a one-stage simultaneous repair of the entire cleft exemplifies a simple and economic therapeutical method. The concept of one-stage repair dates back to 1958, when Farina (1958) described the surgical technique employed during one-stage closure of
http://dx.doi.org/10.1016/j.jcms.2015.04.027 1010-5182/© 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
P.S. Fudalej et al. / Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1224e1231
CUCLP. He listed a decreased risk associated with general anesthesia, better healing of soft tissues, lower incidence of fistulae, and reduction of costs of hospitalization as the rationale for performing this type of closure of the cleft. Farina also mentioned that no postoperative complications had been encountered; however, the reliability of his findings is uncertain. Despite the long history, only 9 out of 201 European cleft teams have adopted this method (Shaw et al., 2000) and few studies have so far examined the long-term outcome (De Mey et al., 2009; Savaci et al., 2005; Fudalej et al., 2009a). Overall, the cleft teams from Brussels, Belgium (De Mey et al., 2009), Konya and Zonguldak, Turkey (Savaci et al., 2005) and Warsaw, Poland (Fudalej et al., 2010) found that their protocols employing one-stage closure of UCLP produced favorable morphological results. As in other studies, the authors observed a retrognathic maxillo-mandibular complex in cleft groups compared with non-cleft controls. However, no significant differences in cephalometric measurements between patients with CUCLP treated with the one-stage and alternative protocols were found. At the Warsaw Institute of Mother and Child (IMC) the one-stage approach of treatment of UCLP was developed in the early 1980s by Dudkiewicz (1991) in response to previously unsatisfactory results, as a large proportion of patients treated in the 1970s demonstrated poor facial development and had speech deficiencies. In 2009, the dental arch relationship in patients treated in Warsaw and Oslo was compared (Fudalej et al., 2009a). The Oslo Cleft Center was chosen as a reference center for Warsaw because Oslo had participated in numerous intercentre comparison studies, most of the time with € m et al., 2005; Meazzini et al., 2008, favorable results (Brattstro 2010; Del Guercio et al., 2010; Bartzela et al., 2012) and it has a long-established protocol, a large record collection of consecutively treated patients, and a willingness to participate in research. The WarsaweOslo comparison of dental arch relationships showed that the one-stage protocol practiced in Warsaw and two-stage protocol employed in Oslo were equally successful (Fudalej et al., 2009a). However, some patients from Warsaw and Oslo had been treated orthodontically before study models used in the comparison were prepared, but the duration and intensity of orthodontic treatment could not be confirmed in all Polish subjects. Although the GOSLON (Great Ormond Street London and Oslo, Norway) Yardstick, which was used for rating, has for experienced cleft orthodontists some capacity to compensate for effects of an initial phase of orthodontic therapy, a recent study (Southall et al., 2012) demonstrated that GOSLON scores could be improved with orthodontic treatment. In other words, the inclusion of patients who had received more extensive orthodontic treatment prior to the taking of study models being used for GOSLON Yardstick scoring, can result in a more favorable outcome. The orthodontic appliances used in the initial phase of orthodontic therapy in patients from Warsaw and Oslo were simple removable plates or fixed appliances to align incisors. They would have affected the alignment of the front teeth but probably had little or no effect on the antero-posterior position of the maxilla. The objective of this study was to compare facial morphology in a sample of patients with UCLP treated with the one-stage repair and a control group treated by the Oslo Cleft Team with the two-stage repair. The null hypothesis is that facial morphology in both groups is comparable.
1225
treated with two different protocols. The Warsaw group consisted of 61 subjects (42 male, 19 female) consecutively treated at the Warsaw Institute of Mother and Child, Poland. All subjects in the Polish sample were born between May 1992 and January 1996 and were operated on by the same experienced surgeon. The mean age when radiographic assessment was carried out was 10.9 years (SD ¼ 1.0). The Oslo group was taken from a consecutive series of patients with CUCLP born between 1975 and 1980 treated by the Oslo Cleft Team (and a part of the Eurocran Good Practice Archive) and matched with the Polish sample for age, sex, and soft tissue band. Three senior surgeons did the first operations (lip and hard palate closure) while two additional surgeons were involved in the closure of the soft palate. The mean age when radiographic assessment was carried out was 11 years (SD ¼ 1.9). 2.2. Surgical management 2.2.1. Warsaw sample No pre-surgical orthopedic treatment (PSOT) was carried out. The lip, and hard and soft palate were all closed in a single operation according to the following protocol: lip closure was undertaken using a triangular flap; for hard palate repair an extended vomer flap with tight closure of all surgical wounds on the anterior palate was performed. Relaxing incisions along the alveoli were small and limited to the premolar region. Soft palate closure was done by one-cut dissection of all abnormal muscle insertions from the posterior margin of the hard palate up to the pterygoid hamuli, which were always fractured. The mean age at surgery was 9.2 months (range: 6.0e15.8; SD ¼ 2.0). Alveolar bone grafting was performed between 9 and 12 years. 2.2.2. Oslo sample No PSOT was performed. In the first operation: the lip was closed using the Millard technique and simultaneous hard palate closure was done using a single layer vomer flap. The mean age for this operation was 3.3 months (range 2.1e4.3). The soft palate was closed at a mean age of 17.2 months (range 15.7e31.0 months) using a modified von Langenbeck technique. Alveolar bone grafting was performed at a mean age of 9.7 years (range 8.8e12.6). Details of the surgical protocol can be found in Åbyholm (1996). A summary of the Warsaw and Oslo protocols is shown in Table 1. 2.3. Methods Lateral cephalograms taken in centric occlusion were analyzed € m et al., 2005) and Americleft according to the Eurocleft (Brattstro (Daskalogiannakis et al., 2011) protocols. Cephalograms were scanned with the PowerLook III (UMAX, Taipei, Taiwan) scanner and subsequently analyzed with the NemoCeph NX 2005 software (Nemotec, Madrid, Spain). Eight hard and 10 soft tissue landmarks were digitized by one observer (Fig. 1). Thirteen angular and two ratio variables were calculated. To determine the method reliability, 30 cephalograms were selected at random and measured twice. The bias was assessed with BlandeAltman plots (Fig. 2). 2.4. Statistical analysis
2. Material and methods 2.1. Subjects In this retrospective comparative study, facial morphology was examined in two groups of children with non-syndromic CUCLP
Descriptive statistics (means and standard deviation) were computed for each group. Independent t-tests were used to assess differences in craniofacial morphology between the Warsaw and Oslo groups. Regression analyses were performed to investigate an association between the mean GOSLON score (dependent variable;
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P.S. Fudalej et al. / Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1224e1231
GOSLON scores derived from a study by Fudalej et al. (2009a)) and cephalometric parameters (independent variables e SNA: sellanasion-A-point; ANB: A-point-nasion-B-point; SNPog: sellanasion-pogonion; sANB: soft-tissue-A-point-nasion-B-point; and sANPog: soft-tissue-A-point-nasion-pogonion). All statistical analyses were done with SPSS software (version 20). 3. Results The morphology of the facial skeleton in patients treated with one-stage and two-stage protocols was different (Table 2). Maxillary prominence in subjects from the Warsaw group was decreased by almost 4 in comparison to the Oslo group (SNA ¼ 75.3 and 79.1, respectively), which likely led to the reduction of the ANB angle (0.8 and 2.8 in Warsaw and Oslo groups, respectively). In the Warsaw group, the maxillary plane was steeper and the anterior teeth were at higher inclination relative to the maxillary plane than in the Oslo group (p < 0.05). The interincisal angle, however, was comparable in both groups. Inter-group differences were limited to hard tissues e the soft tissue contour in the groups was comparable. The convexity of the subnasal region, chin, and nose did not differ between the groups (p > 0.05) (Table 3). The regression analysis with the GOSLON rating as a dependent variable and SNA, ANB, SNPog, sANB, and sANPog as independent variables showed that only ANB, sANB and sANPog had an effect on the GOSLON score (Table 4). The highest value of R2 was found in the model with ANB as an independent variable (R2 ¼ 0.265), which means that 26.5% of variance of the GOSLON scores can be explained by variance of the ANB angle. 4. Discussion An evaluation of the outcome of treatment of cleft lip and palate should be multidimensional. This means that it ought to include assessment of facial appearance, speech, and the patient's psychological adjustment/satisfaction, in addition to examination of craniofacial form and dental arch relationship. Multifaceted evaluation is indispensible because of the influence of cleft deformity on speech, dentofacial growth, facial appearance, and psychosocial adjustment. Therefore if a CLP team wants to make a rational decision regarding continuation, modification, or discontinuation of the current treatment regimen it has to have a full and balanced picture of its own results. Our main finding is that the maxillary prominence and maxillomandibular relationship in patients treated in Warsaw were less favorable than in patients treated in Oslo. An almost 4 difference in the SNA angle roughly corresponds to a reduction of 4 mm in maxillary convexity. The ANB angle representing the anteroposterior relationship of the maxilla and mandible was, in turn, smaller by 2 in patients from Warsaw. A recent overview of the association between the type of surgical management and facial growth (Semb and Shaw, 2013) indicated surgical skill may be one of the major factors. It means that centers in which cleft repairs are done by high-volume operators, usually show better outcomes
Fig. 1. Reference points and lines on lateral cephalometric radiograph. Skeletal reference points: S (sella), the center of the sella turcica; N (nasion), the most anterior point of the frontonasal suture; ANS (spina nasalis anterior), the apex of the anterior nasal spine; PNS (spina nasalis posterior), the apex of the posterior nasal spine; A (A point), the deepest point on the anterior contour of the upper alveolar arch; B (B point), the deepest point on the anterior contour of the lower anterior process; Pog (pogonion), the most anterior point on the mandibular symphysis; Me (menton), the most inferior point on the mandibular symphysis. Soft tissue reference points: sG (soft tissue glabella), the most anterior point on the soft tissue glabella; sN (soft tissue nasion), the deepest point on the frontonasal curvature; Prn (pronasale), the most prominent point on the apex of the nose; NSt (nasal septum tangent point), the anterior tangent point to the tangent to the nasal septum through subnasale; Subn (subnasale), the deepest point in the nasolabial curvature; sA (soft tissue A point), the point of greatest concavity in the midline of the upper lip; Lab sup (labrale superius), the most prominent point on the prolabium of the upper lip; sB (soft tissue B point), the point of the greatest concavity in the midline of the lower lip; sPog (soft tissue pogonion), the most prominent point on the chin; sMe (soft tissue menton), the lowest point on the chin. Reference lines: SN (sella-nasion line), the line through sella and nasion; PP (palatal plane), the line through ANS and PNS; MP (mandibular plane), the tangent to the lower border of the mandible through the menton; U1, axis of upper incisors; L1, axis of lower incisors € m et al., 2005; modified). (from Brattstro
than centers in which less experienced surgeons perform cleft operations. In both Warsaw and Oslo, closures of CUCLP were done by operators with long experience in cleft surgery. In particular, a single surgeon with more than 10 years' experience operated on all patients in Warsaw. Thus, a surgeon's experience probably was not the cause of the difference in maxillary convexity between groups.
Table 1 Summary of treatment protocols employed in the two cleft centers.
PSOT 3 months 6e12 months 18 months 8e12 years PSOT: pre-surgical orthopedic treatment.
Warsaw
Oslo
No
No Lip (Millard) and hard palate closure (single layer vomer flap)
Lip, soft, and hard palate closure Alveolar bone grafting
Soft palate closure (modified von Langenbeck) Alveolar bone grafting
P.S. Fudalej et al. / Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1224e1231
However, the Warsaw and Oslo centers differed in surgical methods e in the former, a one-stage closure of the entire cleft at 9 months was used and, in the latter, lip and anterior palate repair at 3 months was followed by repair of the remaining cleft palate at 18
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months. Unfortunately, effects of the various surgical techniques and timings on maxillary growth are difficult to assess, because of their concerted action and the possible interplay between them. For example, lip repair has a molding (ie, growth changing) effect
Fig. 2. BlandeAltman plots demonstrating the bias for (a) hard tissue and (b) soft tissue cephalometric variables.
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Fig. 2. (continued).
on the anterior maxilla (Liao and Mars, 2005). If a lip repair is performed at a later time (9 vs. 3 months) maxillary growth restriction can be less. On the other hand, palatal repair carried out along with lip closure (as practiced in Warsaw) can cancel the beneficial effect of late lip closure, or even result in more maxillary inhibition when compared with the Oslo two-stage surgical protocol. However, this is only speculation because elucidation of which element of treatment was responsible for a smaller maxillary
prominence in children from Warsaw is not possible through the current study design. As explained by Shaw et al. (2005) a limitation of inter-center comparison is that they cannot distinguish between the relationship of different elements of a center's protocol and its outcome or between its protocols and the influence of the personnel who deliver that protocol. However, an important factor to consider is the distinctiveness of the background populations from which samples come from.
P.S. Fudalej et al. / Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1224e1231
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Table 2 Differences between Warsaw and Oslo cleft centers in hard tissue morphology. Variable
Warsaw
S-N-A ( ) A-N-B ( ) S-N-Pog ( ) SN/PP ( ) SN/MP ( ) N-PP/N-Me*100% U1/PP ( ) U1/L1 ( )
Oslo
95% CI
P value
Mean
SD
Mean
SD
Difference
Lower limit
Upper limit
75.31 0.79 75.73 12.2 36.3 43.58 107.01 142.72
3.67 2.83 4.26 4.01 6.68 2.04 8.7 11.41
79.14 2.75 77.01 9.05 36.23 42.75 104.1 139.63
4.14 2.41 4.94 3.56 4.73 2.24 7.54 15.16
3.83 1.96 1.29 3.15 0.08 0.82 2.9 3.09
5.19 2.87 2.89 1.85 1.9 0.08 0.09 1.6
2.47 1.06 0.32 4.46 2.06 1.57 5.71 7.78
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
One-stage (Warsaw) and two-stage (Oslo) repair of unilateral cleft lip and palate: Craniofacial outcomes Piotr Stanislaw Fudalej a, b, *, Ewa Wegrodzka c, Gunvor Semb d, Maria Hortis-Dzierzbicka e, f a
Department of Orthodontics and Dentofacial Orthopedics, University of Bern, (Head: Prof. Christos Katsaros), Freiburgstrasse 7, CH-3010 Bern, Switzerland Department of Orthodontics, Medical Faculty of Palacký University, (Head: Assoc. Prof. Milos Spidlen), Palackeho Str. 12, 772 00 Olomouc, Czech Republic Private Practice, Warsaw, Poland d School of Dentistry, University of Manchester, (Head: Prof. Paul Coulthard), Higher Cambridge Street, Manchester M15 6FH, United Kingdom e Laboratory of Speech Pathology and Upper Airway Endoscopy, Institute of Mother and Child, (Head: Assoc. Prof. Maria Hortis-Dzierzbicka), Kasprzaka Str. 17A, 01-211 Warsaw, Poland f Department of Otolaryngology and Maxillofacial Surgery, Universitary Clinical Hospital, (Head: Prof. Andrzej Kukwa), Warszawska Str. 30, 10-082 Olsztyn, Poland b c
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 11 November 2014 Accepted 30 April 2015 Available online 9 May 2015
The aim of this study was to compare facial development in subjects with complete unilateral cleft lip and palate (CUCLP) treated with two different surgical protocols. Lateral cephalometric radiographs of 61 patients (42 boys, 19 girls; mean age, 10.9 years; SD, 1) treated consecutively in Warsaw with one-stage repair and 61 age-matched and sex-matched patients treated in Oslo with two-stage surgery were selected to evaluate craniofacial morphology. On each radiograph 13 angular and two ratio variables were measured in order to describe hard and soft tissues of the facial region. The analysis showed that differences between the groups were limited to hard tissues e the maxillary prominence in subjects from the Warsaw group was decreased by almost 4 in comparison with the Oslo group (sella-nasion-Apoint (SNA) ¼ 75.3 and 79.1, respectively) and maxillo-mandibular morphology was less favorable in the Warsaw group than the Oslo group (ANB angle ¼ 0.8 and 2.8 , respectively). The soft tissue contour was comparable in both groups. In conclusion, inter-group differences suggest a more favorable outcome in the Oslo group. However, the distinctiveness of facial morphology in background populations (ie, in Poles and Norwegians) could have contributed to the observed results. © 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
Keywords: Cephalometrics Cleft lip Cleft palate Morphology Treatment
1. Introduction The treatment of cleft lip and palate (CLP) is far from uniform. Despite ongoing experimental and clinical research, numerous aspects of CLP care, such as the influence of non-surgical factors on maxillofacial morphology (Xu et al., 2014), the extension of primary surgery (Janiszewska-Olszowska, 2013), the role of various types of palatoplasties on facial growth (Xu et al., 2015) or who should judge the final outcome (Gkantidis et al., 2013), continue to be discussed. The results of two large multi-center comparative outcome studies: Eurocleft (Shaw et al., 2005) and Americleft (Russell et al.,
* Corresponding author. Department of Orthodontics and Dentofacial Orthopedics, University of Bern, Freiburgstrasse 7, CH-3010 Bern, Switzerland. E-mail address: [email protected] (P.S. Fudalej).
2011) showed a diversity of outcomes e some participating centers produced more favorable results and some produced less favorable results. Due to the retrospective design of the first 9-year comparisons of Eurocleft (with prospective follow-up at 12 and 17 years) and Americleft, it was not possible to identify those elements of treatment which were responsible for the final outcome. Nevertheless, relatively good results obtained by centers which employ simpler and less burdensome protocols without the use of presurgical orthopedics or primary bone grafting, and with fewer operating surgeons, imply that demanding protocols may be unjustified. Treatment of complete unilateral cleft lip and palate (CUCLP) using a one-stage simultaneous repair of the entire cleft exemplifies a simple and economic therapeutical method. The concept of one-stage repair dates back to 1958, when Farina (1958) described the surgical technique employed during one-stage closure of
http://dx.doi.org/10.1016/j.jcms.2015.04.027 1010-5182/© 2015 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
P.S. Fudalej et al. / Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1224e1231
CUCLP. He listed a decreased risk associated with general anesthesia, better healing of soft tissues, lower incidence of fistulae, and reduction of costs of hospitalization as the rationale for performing this type of closure of the cleft. Farina also mentioned that no postoperative complications had been encountered; however, the reliability of his findings is uncertain. Despite the long history, only 9 out of 201 European cleft teams have adopted this method (Shaw et al., 2000) and few studies have so far examined the long-term outcome (De Mey et al., 2009; Savaci et al., 2005; Fudalej et al., 2009a). Overall, the cleft teams from Brussels, Belgium (De Mey et al., 2009), Konya and Zonguldak, Turkey (Savaci et al., 2005) and Warsaw, Poland (Fudalej et al., 2010) found that their protocols employing one-stage closure of UCLP produced favorable morphological results. As in other studies, the authors observed a retrognathic maxillo-mandibular complex in cleft groups compared with non-cleft controls. However, no significant differences in cephalometric measurements between patients with CUCLP treated with the one-stage and alternative protocols were found. At the Warsaw Institute of Mother and Child (IMC) the one-stage approach of treatment of UCLP was developed in the early 1980s by Dudkiewicz (1991) in response to previously unsatisfactory results, as a large proportion of patients treated in the 1970s demonstrated poor facial development and had speech deficiencies. In 2009, the dental arch relationship in patients treated in Warsaw and Oslo was compared (Fudalej et al., 2009a). The Oslo Cleft Center was chosen as a reference center for Warsaw because Oslo had participated in numerous intercentre comparison studies, most of the time with € m et al., 2005; Meazzini et al., 2008, favorable results (Brattstro 2010; Del Guercio et al., 2010; Bartzela et al., 2012) and it has a long-established protocol, a large record collection of consecutively treated patients, and a willingness to participate in research. The WarsaweOslo comparison of dental arch relationships showed that the one-stage protocol practiced in Warsaw and two-stage protocol employed in Oslo were equally successful (Fudalej et al., 2009a). However, some patients from Warsaw and Oslo had been treated orthodontically before study models used in the comparison were prepared, but the duration and intensity of orthodontic treatment could not be confirmed in all Polish subjects. Although the GOSLON (Great Ormond Street London and Oslo, Norway) Yardstick, which was used for rating, has for experienced cleft orthodontists some capacity to compensate for effects of an initial phase of orthodontic therapy, a recent study (Southall et al., 2012) demonstrated that GOSLON scores could be improved with orthodontic treatment. In other words, the inclusion of patients who had received more extensive orthodontic treatment prior to the taking of study models being used for GOSLON Yardstick scoring, can result in a more favorable outcome. The orthodontic appliances used in the initial phase of orthodontic therapy in patients from Warsaw and Oslo were simple removable plates or fixed appliances to align incisors. They would have affected the alignment of the front teeth but probably had little or no effect on the antero-posterior position of the maxilla. The objective of this study was to compare facial morphology in a sample of patients with UCLP treated with the one-stage repair and a control group treated by the Oslo Cleft Team with the two-stage repair. The null hypothesis is that facial morphology in both groups is comparable.
1225
treated with two different protocols. The Warsaw group consisted of 61 subjects (42 male, 19 female) consecutively treated at the Warsaw Institute of Mother and Child, Poland. All subjects in the Polish sample were born between May 1992 and January 1996 and were operated on by the same experienced surgeon. The mean age when radiographic assessment was carried out was 10.9 years (SD ¼ 1.0). The Oslo group was taken from a consecutive series of patients with CUCLP born between 1975 and 1980 treated by the Oslo Cleft Team (and a part of the Eurocran Good Practice Archive) and matched with the Polish sample for age, sex, and soft tissue band. Three senior surgeons did the first operations (lip and hard palate closure) while two additional surgeons were involved in the closure of the soft palate. The mean age when radiographic assessment was carried out was 11 years (SD ¼ 1.9). 2.2. Surgical management 2.2.1. Warsaw sample No pre-surgical orthopedic treatment (PSOT) was carried out. The lip, and hard and soft palate were all closed in a single operation according to the following protocol: lip closure was undertaken using a triangular flap; for hard palate repair an extended vomer flap with tight closure of all surgical wounds on the anterior palate was performed. Relaxing incisions along the alveoli were small and limited to the premolar region. Soft palate closure was done by one-cut dissection of all abnormal muscle insertions from the posterior margin of the hard palate up to the pterygoid hamuli, which were always fractured. The mean age at surgery was 9.2 months (range: 6.0e15.8; SD ¼ 2.0). Alveolar bone grafting was performed between 9 and 12 years. 2.2.2. Oslo sample No PSOT was performed. In the first operation: the lip was closed using the Millard technique and simultaneous hard palate closure was done using a single layer vomer flap. The mean age for this operation was 3.3 months (range 2.1e4.3). The soft palate was closed at a mean age of 17.2 months (range 15.7e31.0 months) using a modified von Langenbeck technique. Alveolar bone grafting was performed at a mean age of 9.7 years (range 8.8e12.6). Details of the surgical protocol can be found in Åbyholm (1996). A summary of the Warsaw and Oslo protocols is shown in Table 1. 2.3. Methods Lateral cephalograms taken in centric occlusion were analyzed € m et al., 2005) and Americleft according to the Eurocleft (Brattstro (Daskalogiannakis et al., 2011) protocols. Cephalograms were scanned with the PowerLook III (UMAX, Taipei, Taiwan) scanner and subsequently analyzed with the NemoCeph NX 2005 software (Nemotec, Madrid, Spain). Eight hard and 10 soft tissue landmarks were digitized by one observer (Fig. 1). Thirteen angular and two ratio variables were calculated. To determine the method reliability, 30 cephalograms were selected at random and measured twice. The bias was assessed with BlandeAltman plots (Fig. 2). 2.4. Statistical analysis
2. Material and methods 2.1. Subjects In this retrospective comparative study, facial morphology was examined in two groups of children with non-syndromic CUCLP
Descriptive statistics (means and standard deviation) were computed for each group. Independent t-tests were used to assess differences in craniofacial morphology between the Warsaw and Oslo groups. Regression analyses were performed to investigate an association between the mean GOSLON score (dependent variable;
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GOSLON scores derived from a study by Fudalej et al. (2009a)) and cephalometric parameters (independent variables e SNA: sellanasion-A-point; ANB: A-point-nasion-B-point; SNPog: sellanasion-pogonion; sANB: soft-tissue-A-point-nasion-B-point; and sANPog: soft-tissue-A-point-nasion-pogonion). All statistical analyses were done with SPSS software (version 20). 3. Results The morphology of the facial skeleton in patients treated with one-stage and two-stage protocols was different (Table 2). Maxillary prominence in subjects from the Warsaw group was decreased by almost 4 in comparison to the Oslo group (SNA ¼ 75.3 and 79.1, respectively), which likely led to the reduction of the ANB angle (0.8 and 2.8 in Warsaw and Oslo groups, respectively). In the Warsaw group, the maxillary plane was steeper and the anterior teeth were at higher inclination relative to the maxillary plane than in the Oslo group (p < 0.05). The interincisal angle, however, was comparable in both groups. Inter-group differences were limited to hard tissues e the soft tissue contour in the groups was comparable. The convexity of the subnasal region, chin, and nose did not differ between the groups (p > 0.05) (Table 3). The regression analysis with the GOSLON rating as a dependent variable and SNA, ANB, SNPog, sANB, and sANPog as independent variables showed that only ANB, sANB and sANPog had an effect on the GOSLON score (Table 4). The highest value of R2 was found in the model with ANB as an independent variable (R2 ¼ 0.265), which means that 26.5% of variance of the GOSLON scores can be explained by variance of the ANB angle. 4. Discussion An evaluation of the outcome of treatment of cleft lip and palate should be multidimensional. This means that it ought to include assessment of facial appearance, speech, and the patient's psychological adjustment/satisfaction, in addition to examination of craniofacial form and dental arch relationship. Multifaceted evaluation is indispensible because of the influence of cleft deformity on speech, dentofacial growth, facial appearance, and psychosocial adjustment. Therefore if a CLP team wants to make a rational decision regarding continuation, modification, or discontinuation of the current treatment regimen it has to have a full and balanced picture of its own results. Our main finding is that the maxillary prominence and maxillomandibular relationship in patients treated in Warsaw were less favorable than in patients treated in Oslo. An almost 4 difference in the SNA angle roughly corresponds to a reduction of 4 mm in maxillary convexity. The ANB angle representing the anteroposterior relationship of the maxilla and mandible was, in turn, smaller by 2 in patients from Warsaw. A recent overview of the association between the type of surgical management and facial growth (Semb and Shaw, 2013) indicated surgical skill may be one of the major factors. It means that centers in which cleft repairs are done by high-volume operators, usually show better outcomes
Fig. 1. Reference points and lines on lateral cephalometric radiograph. Skeletal reference points: S (sella), the center of the sella turcica; N (nasion), the most anterior point of the frontonasal suture; ANS (spina nasalis anterior), the apex of the anterior nasal spine; PNS (spina nasalis posterior), the apex of the posterior nasal spine; A (A point), the deepest point on the anterior contour of the upper alveolar arch; B (B point), the deepest point on the anterior contour of the lower anterior process; Pog (pogonion), the most anterior point on the mandibular symphysis; Me (menton), the most inferior point on the mandibular symphysis. Soft tissue reference points: sG (soft tissue glabella), the most anterior point on the soft tissue glabella; sN (soft tissue nasion), the deepest point on the frontonasal curvature; Prn (pronasale), the most prominent point on the apex of the nose; NSt (nasal septum tangent point), the anterior tangent point to the tangent to the nasal septum through subnasale; Subn (subnasale), the deepest point in the nasolabial curvature; sA (soft tissue A point), the point of greatest concavity in the midline of the upper lip; Lab sup (labrale superius), the most prominent point on the prolabium of the upper lip; sB (soft tissue B point), the point of the greatest concavity in the midline of the lower lip; sPog (soft tissue pogonion), the most prominent point on the chin; sMe (soft tissue menton), the lowest point on the chin. Reference lines: SN (sella-nasion line), the line through sella and nasion; PP (palatal plane), the line through ANS and PNS; MP (mandibular plane), the tangent to the lower border of the mandible through the menton; U1, axis of upper incisors; L1, axis of lower incisors € m et al., 2005; modified). (from Brattstro
than centers in which less experienced surgeons perform cleft operations. In both Warsaw and Oslo, closures of CUCLP were done by operators with long experience in cleft surgery. In particular, a single surgeon with more than 10 years' experience operated on all patients in Warsaw. Thus, a surgeon's experience probably was not the cause of the difference in maxillary convexity between groups.
Table 1 Summary of treatment protocols employed in the two cleft centers.
PSOT 3 months 6e12 months 18 months 8e12 years PSOT: pre-surgical orthopedic treatment.
Warsaw
Oslo
No
No Lip (Millard) and hard palate closure (single layer vomer flap)
Lip, soft, and hard palate closure Alveolar bone grafting
Soft palate closure (modified von Langenbeck) Alveolar bone grafting
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However, the Warsaw and Oslo centers differed in surgical methods e in the former, a one-stage closure of the entire cleft at 9 months was used and, in the latter, lip and anterior palate repair at 3 months was followed by repair of the remaining cleft palate at 18
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months. Unfortunately, effects of the various surgical techniques and timings on maxillary growth are difficult to assess, because of their concerted action and the possible interplay between them. For example, lip repair has a molding (ie, growth changing) effect
Fig. 2. BlandeAltman plots demonstrating the bias for (a) hard tissue and (b) soft tissue cephalometric variables.
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Fig. 2. (continued).
on the anterior maxilla (Liao and Mars, 2005). If a lip repair is performed at a later time (9 vs. 3 months) maxillary growth restriction can be less. On the other hand, palatal repair carried out along with lip closure (as practiced in Warsaw) can cancel the beneficial effect of late lip closure, or even result in more maxillary inhibition when compared with the Oslo two-stage surgical protocol. However, this is only speculation because elucidation of which element of treatment was responsible for a smaller maxillary
prominence in children from Warsaw is not possible through the current study design. As explained by Shaw et al. (2005) a limitation of inter-center comparison is that they cannot distinguish between the relationship of different elements of a center's protocol and its outcome or between its protocols and the influence of the personnel who deliver that protocol. However, an important factor to consider is the distinctiveness of the background populations from which samples come from.
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Table 2 Differences between Warsaw and Oslo cleft centers in hard tissue morphology. Variable
Warsaw
S-N-A ( ) A-N-B ( ) S-N-Pog ( ) SN/PP ( ) SN/MP ( ) N-PP/N-Me*100% U1/PP ( ) U1/L1 ( )
Oslo
95% CI
P value
Mean
SD
Mean
SD
Difference
Lower limit
Upper limit
75.31 0.79 75.73 12.2 36.3 43.58 107.01 142.72
3.67 2.83 4.26 4.01 6.68 2.04 8.7 11.41
79.14 2.75 77.01 9.05 36.23 42.75 104.1 139.63
4.14 2.41 4.94 3.56 4.73 2.24 7.54 15.16
3.83 1.96 1.29 3.15 0.08 0.82 2.9 3.09
5.19 2.87 2.89 1.85 1.9 0.08 0.09 1.6
2.47 1.06 0.32 4.46 2.06 1.57 5.71 7.78
