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Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients
Int. J. Oral Maxillofac. Surg. 2013; 42: 192–197 http://dx.doi.org/10.1016/j.ijom.2012.09.022, available online at http://www.sciencedirect.com
Clinical Paper Cleft Lip and Palate
Influence of pharyngeal flap surgery on maxillary outgrowth § in cleft patients
I. E. Voshola, L. N. A. van Adrichemb, K. G. H. van der Wala, M. J. Koudstaala a Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, The Netherlands; bDepartment of Plastic and Reconstructive Surgery, Erasmus University Medical Center, Rotterdam, The Netherlands
I. E. Voshol, L. N. A. van Adrichem, K. G. H. van der Wal, M. J. Koudstaal: Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients. Int. J. Oral Maxillofac. Surg. 2013; 42: 192–197. # 2012 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons. Abstract. The aim of this study was to investigate the influence of the pharyngeal flap procedure on the frequency of Le Fort I osteotomies in full-grown nonsyndromic cleft patients. A retrospective review of 508 full-grown cleft patients born between 1 January 1983 and 31 December 1992 was performed. Following data analysis, 140 males older than 18 years and 111 females over the age of 16 years were included. 69 of the 251 included cleft patients required pharyngeal flap surgery (27.5%). Revision flap surgery was performed in 17.4% of the cases. A significantly lower age at time of the initial pharyngeal flap procedure was found in patients requiring revision surgery (5.6 years versus 6.8 years). The frequency of Le Fort I osteotomies was significantly higher in the patients with a pharyngeal flap (19%) compared to those without (8%) (p < 0.05). The results of this study point towards the pharyngeal flap procedure being one of the possible limiting factors for maxillary antero-posterior growth in cleft patients.
Introduction
The aim of cleft treatment is to obtain the best possible functional and aesthetic result when facial growth is completed; but the optimal timing and approach for the surgical management continues to be a source of controversy. Therefore, factors influencing the outcome need to be studied. In general, the presence of maxillary hypoplasia in repaired cleft patients is a § Presented at the Spring Meeting of the Dutch Association of Oral and Maxillofacial Surgery (NVMKA), in Maastricht, the Netherlands, 18 March 2011.
0901-5027/020192 + 06 $36.00/0
well-recognized phenomenon. Most authors advocate the theory that iatrogenic factors, especially primary lip and palate repair, cause the main restriction of maxillary growth.1–4 The Le Fort I advancement osteotomy (LFI) is the standard surgical procedure to treat Angle Class III relationships due to maxillary retrusion.5–7 The incidence of maxillary hypoplasia is often translated into the number of LFI osteotomies performed.8,9 The frequency of LFI varies from 0.0 to 69.6%.4,8–18 The influence of primary lip or palate repair on maxillary growth has been the subject of many reports, but the influence of pharyngeal
Keywords: Cleft; Pharyngeal flap surgery; Le Fort I osteotomy; Maxillary hypoplasia. Accepted for publication 26 September 2012 Available online 2 November 2012
flap surgery (PFS) is seldom discussed and the results are contradictory. This is remarkable, since 20–38% of cleft palate patients require this procedure.19,20 Oberoi et al. delineated factors possibly contributing to maxillary hypoplasia, one of them being PFS. In a group of 16 unilateral cleft lip and palate patients without maxillary hypoplasia only one patient had undergone (secondary) PFS surgery (6.25%), whereas in the group with maxillary hypoplasia 4 of 16 patients had PFS before during palate repair and 4 had PFS as a secondary (after palate repair) procedure (50%); these results were not supported by statistical investigations.4
# 2012 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.
Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients Although some small differences in growth pattern were seen in previous studies on the effect of pharyngeal flaps on maxillary growth, no conclusive evidence was provided.20,21 The sparse number of reports considering the influence of PFS on maxillary growth combined with the small number of patients studied and the short follow up period, show more studies are required. This report attempts to address the possible effect of the commonly performed PFS on subsequent maxillary growth in cleft patients. Additional risk factors for PFS and LFI, including cleft subtype, gender and number of previous surgical procedures, will be evaluated.4,8,9 Materials and methods
508 consecutive cleft patients born between 1 January 1983 and 31 December 1990 (male) and 31 December 1992 (female) and treated at the authors’ institution were evaluated retrospectively. The indication for a LFI was assessed when facial skeletal growth had been completed. Since this is on average 2 years earlier in females (at the age of 16 years) than in males (at the age of 18 years)22 the included birth dates of patients differed between genders. Inclusion criteria were lip and/or alveolar process and/or palate and/or submucous non-syndromal cleft patients, with maturity of the facial skeleton. The treatment was according to the institution protocol as shown in Table 1. Exclusion criteria were cleft patients with other congenital deformities, patients who died before reaching the age of 16 years (females)/18 years (males), midfacial clefts, previous radio- or chemotherapy, incomplete documentation and exceptions to the protocol. Although two-stage palate repair is reported to result in higher percentages of PFS compared to one-stage repair,23 the protocol includes a two-stage palate repair resulting in a superior maxillary antero-posterior growth, supported by a lower percentage of LFI in these patients compared to patients treated with a onestage palate repair.9 Since the cleft in the alveolar process is thought to be the main inhibiting factor for maxillary growth9,15 it was preferred, in agreement with the authors’ previous study,9 to divide the cleft lip group into a cleft lip group and a cleft lip and alveolus group. Overall, patients were subcategorized as shown in Table 2. The following information was collected from the clinical records for each
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Table 1. Cleft protocol Erasmus Medical Center Rotterdam. Lip repair (according to the Millard technique) 3–5 months 9–11 months Closure of the soft palate cleft in case of cleft lip, alveolus and palate (CLAP), closure of the soft and hard palate cleft in case of cleft palate (CP) (according to the Wardill-Kilner, Von Langenbeck, Perko or Kriens technique) 8–13 years Alveolar bone grafting and closure of the hard palate in case of cleft lip, alveolus and palate (CLAP) 16–18 years If necessary: orthognathic surgery after completion of facial skeletal development and secondary nose and/or lip corrections Treatment of submucous cleft is performed in the presence of complaints; an individual treatment plan is developed. Pharyngeal flap surgery is performed when velopharyngeal insufficiency is diagnosed (according to the Orticochea or Roselli technique). Table 2. Categorization of cleft subtypes. Name
Type of clefta
UCL UICL UCCL BCL BICL BACL BCCL UCLA UICLA UCCLA BCLA BICLA BACLA BCCLA UCLAP UICLAP UCCLAP BCLAP BICLAP BACLAP BCCLAP CP ICP CCP SM
Unilateral cleft lip Unilateral incomplete cleft lip Unilateral complete cleft lip Bilateral cleft lip Bilateral incomplete cleft lip Bilateral asymmetric cleft lip Bilateral complete cleft lip Unilateral cleft lip and alveolus Unilateral incomplete cleft lip and alveolus Unilateral complete cleft lip and alveolus Bilateral cleft lip and alveolus Bilateral incomplete cleft lip and alveolus Bilateral asymmetric cleft lip and alveolus Bilateral complete cleft lip and alveolus Unilateral cleft lip, alveolus and palate Unilateral incomplete cleft lip, alveolus and palate Unilateral cleft lip, alveolus and palate Bilateral cleft lip, alveolus and palate Bilateral incomplete cleft lip, alveolus and palate Bilateral asymmetric cleft lip, alveolus and palate Bilateral complete cleft lip, alveolus and palate Cleft palate Incomplete cleft palate (solitary soft palate affected) Complete cleft palate (soft and hard palate) Submucous cleft
a Classification of cleft type in incomplete (I), asymmetrical (A) or complete (C) and unilateral (U) or bilateral (B) only specifies the extent of the cleft lip and not the extent of the cleft alveolus or palate (Good et al. [8]).
case: date of birth, gender, cleft type and extent, and all surgical procedures related to the cleft (lip closure, anterior and posterior palatoplasty, alveolar bone graft surgery, closure of possible fistulas, pharyngeal flap procedure, surgically assisted rapid maxillary expansion). The indication to perform PFS was assessed by a team of ENT specialists and speech therapist. PFS was performed in case of communication problems (i.e. hypernasal speech) due to velopharyngeal incompetence. The criterion was hypernasal speech causing communication problems, which could not be improved (further) by speech therapy. The open nasality was diagnosed in clinical perceptual speech evaluation performed by the team’s speech therapist, by a standard repetitive text, speech with cartoons and
‘free’ speech, combined with a mirror-test (to check the nasal air passage). In cases where PFS was performed, additional information was collected regarding the type of pharyngeal flap procedure performed, age during initial procedure and the necessity for secondary procedures. A secondary procedure was defined as any revision of the sphincter, determined by the clinical evaluation by the treatment team and objective speech assessment by speech pathologist. The need for a LFI osteotomy was recorded by the number of LFI performed or if there had been an indication for LFI osteotomy when fully grown as stated by the multidisciplinary treatment team, even though the surgery had not yet been performed. The frequency of LFI in patients with a pharyngeal flap (PFS+) and without a
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pharyngeal flap (PFS ) was compared. Additional risk factors for both procedures were evaluated, such as cleft subtype, gender and number of previous surgical procedures.4,9,15 Since age at the time of cleft repair influences maxillary growth24,25 the possible correlation between age at initial pharyngeal flap procedure and frequency of LFI was studied. Cephalometric analysis was performed on patients with an indication for LFI osteotomy before and after treatment. Angular measurements recorded were SNA angle (degrees), SNB angle (degrees) and ANB (degrees). The measurements were performed using the digital dicom-data program Easy-ViewWeb (2005, Philips Medical Systems, Best, The Netherlands). Statistical analysis was performed using Microsoft Excel (version 2003) and the statistical Package of the Social Sciences (SPSS, version 17.0). Student’s test and the independent t sample test were used to evaluate data. The limit of significance considered was p = 0.05 (two-sided). For this study the principles outlined in the Declaration of Helsinki have been followed. The Medical Ethical Commission (METC, ethical board commission) of the Erasmus Medical Center accepted this study format and the intended publication on 7 July 2010 (MEC-2010-207). Results
Of the total of 508 patients, 86 had a congenital deformity, 8 patients died before reaching maturity, 2 patients received chemotherapy and in 111 cases the documentation was incomplete. 11 patients changed to another cleft team and 39 patients were not treated according to the institution’s protocol and were consequently excluded. This left 251 patients for inclusion in the study: 140 males (55.8%) and 111 females (44.2%). The miscellaneous group included patients with cleft types that could not be included in the other cleft subcategories, for example, cleft lip combined with a submucous cleft or cleft lip and a cleft palate, without an alveolar cleft. Of the 251 included cleft patients, 69 patients (27.5%) required PFS (Fig. 1). Since the authors’ institution is a tertiary care and teaching hospital, PFS was performed by different surgeons. The group of patients who underwent PFS consisted of 35 males and 34 females. The overall frequency of PFS in males was 25%, compared to 31% in females (p > 0.05). The mean age at the time of initial PFS was 6.6 years (range 3.1– 16.8 years, median 6.4). Of all PFS, 84%
Fig. 1. Number of pharyngeal flap procedures performed in each cleft subtype.
was performed before 9 years of age, and 93% before reaching the age of 10 years. Frequency of PFS was lowest in the unilateral incomplete cleft lip, alveolus and palate (UICLAP) group (20%) and highest in the bilateral asymmetric cleft lip, alveolus and palate (BACLAP) group (58.3%). Frequencies of PFS were 40.0% in the submucous cleft (SM) group, 34.0% in the cleft palate (CP) group, 46.5% in the unilateral cleft lip and palate (UCLAP) group and 51.7% in the bilateral cleft lip, alveolus and palate (BCLAP) as shown in Table 3. PFS was generally performed according to the Orticochea procedure (54 cases, 78.3%). In 8 cases the modification by Jackson was used. In 14 cases the procedure according to Roselli was performed (20.3%) and in one case the PFS procedure was not further specified. The number of revision flap procedures was 17.4% (n = 12), with 7 surgical revisionary procedures in males (20.0%) and 5 in females (14.7%) (p > 0.05). The revision rate varied per cleft subtype: 0% in SM patients, 15.2% in UCLAP patients, 17.6% in CP patients up to 26.7% in BCLAP patients as shown in Table 3. The revision led to a satisfactory outcome in 10 of these patients (97.1%). Two patients (both males) needed a secondary revision. A statistically significant difference was seen in the average age at the initial PFS in patients who later required revision (5.6 years) compared to those who did not need revision (6.8 years) (p < 0.05). In one male UCLAP patient a wound of dehiscence of the flap was repaired after 4 days; this was counted as a revision procedure. Looking at the type of initial pharyngeal flap surgery and the number of revision
procedures, 11 of 54 patients treated with a pharyngeal flap according to Orticochea needed revision (20.4%), whereas of the 14 patients treated with a pharyngeal flap according to Roselli only one pharyngeal flap needed to be revised (7.2%). This difference was not statistical significant (p > 0.05). Patients with PFS+ surgery had a significantly higher frequency of LFI (p < 0.05) compared to PFS patients. 13 patients (19%) of the 69 PFS+ patients required an LFI, but only 15 of 182 (8%) PFS patients required an LFI. The LFI advancements (measured by cephalometric tracing using the ANB) ranged from 2.2 to 19.0 degrees, with an average of 4.3 degrees in patients treated with conventional LFI osteotomy and 7.4 degrees in patients treated using distraction osteogenesis. On average, all patients treated with LFI had an ANB of 3.4 degrees before treatment, and 1.9 degrees following treatment. The frequency of LFI is slightly higher in patients with PFS compared to patients without. None of the differences between the PFS and PFS+ groups in each cleft subtype were significant (p > 0.05) (Table 4). The type of PFS also seems to have an influence on the frequency of LFI. Although the number of patients in each category was small, patients who underwent Orticochea sphincteroplasty (modified according to Jackson) needed an LFI more frequently (10 of 52 patients, 19.2%) than patients who underwent superiorly based flap surgery (e.g. Roselli) (1 of 14 patients, 7.14%). With regard to the age of the cleft patients at the time of initial PFS, no statistically significant difference was found between patients with (6.7 years) and
Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients Table 3. Pharyngeal flap and revisionary procedures in each cleft subtype. Number of patients
Cleft type
Number of patients with pharyngeal flap
9
Miscellaneous SM
10
ICP
24
CCP
26
UCL
34
BCL
5
UCLA
37
BCLA
6
UICLAP
10
UCCLAP
61
BICLAP
4
BACLAP
12
BCCLAP
13
0 0% 4 40.0% 7 29.2% 10 38.5% 0 0% 0 0% 0 0% 0 0% 2 20.0% 31 50.8% 2 50.0% 7 58.3% 6 46.2% 69 27.5%
251
Total
Number of revisionary procedures 0 0% 0 0% 1 14.3% 2 20% 0 0% 0 0% 0 0% 0 0% 0 0% 5 16.1% 1 50.0% 1 14.3% 2 33.3% 12 17.4%
Frequency of revisionary procedures in each cleft subtype – – 8.3% 16.7% – – – – – 41.7% 8.3% 8.3% 16.7% –
SM, submucous cleft; ICP, incomplete cleft palate; CCP, complete cleft palate; UCL, unilateral cleft lip; BCL, bilateral cleft lip; UCLA, unilateral cleft lip and alveolus; BCLA, bilateral cleft lip and alveolus; UICLAP, unilateral incomplete cleft lip, alveolus and palate; UCCLAP, unilateral complete cleft lip, alveolus and palate; BICLAP, bilateral incomplete cleft lip, alveolus and palate; BACLAP, bilateral asymmetric cleft lip, alveolus and palate; BCCLAP, bilateral complete cleft lip, alveolus and palate.
Table 4. Frequency of LFI in cleft patients with and without pharyngeal flap. Cleft subtype
Total number of patients
SM
10
CP
50
UCLAP
71
BCLAP
29
Pharyngeal flap surgery performed Yes No Yes No Yes No Yes No
Number of patients with/without pharyngeal flap
Number of patients with indication for LFI
4 6 17 33 33 38 15 14
0 0 1 1 7 8 5 4
Frequency of LFI (%) 0 0 5.9 3.0 21.2 21.1 33.3 28.6
SM, submucous cleft; CP, cleft palate; UCLAP, unilateral cleft lip, alveolus and palate; BCLAP, bilateral cleft lip, alveolus and palate.
without (6.2 years) an indication for LFI (p > 0.05). One male bilateral complete cleft lip, alveolus and palate (BCCLAP) patient without pharyngeal flap suffered velopharyngeal insufficiency after the LFI procedure, requiring late pharyngeal flap surgery.
Discussion
The frequency of PFS in cleft patients treated according to the protocol at he authors’ institution was 27.5%, in concordance with percentages reported in the literature. Vedung found frequencies of 20% in complete cleft palate (CCP), 11–
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12% in UCLAP and incomplete cleft palate (ICP) and 14–16% in BCLAP, De Buys describes frequencies of 15.6% in CP, 34.4% in unilateral cleft lip and palate (UCLP) and 48.6% in BCLAP.23,26 Bicknell found an overall frequency of PFS of 25% in cleft patients (28 of 114 patients), subdivided into 12% ICP, 44% CCP, 21% UCLAP, 50% BCLAP and 20% SM.27 These small differences can be explained by the high diversity in cleft types with a relatively large number of patients in the BCLAP/CP group, which have a considerably higher frequency of PFS. The two stage palate repair advocated in the authors’ institution has to be taken into account since this two stage palate repair is reported to result in higher percentages of PFS compared to one stage repair.23 Many authors assume the two stage palate repair has an advantage for maxillary growth so the two stage palate repair is justified in the opinion of the authors’ cleft team.9,24,25 Nevertheless, the authors are aware of recent reports that state that acceptable maxillary antero-posterior growth is accomplished in both single and two stage palate repair.28,29 A factor of major influence seems to be the involvement of the hard palate, since the frequency of PFS is 38.5% in CCP (10 of 26) against 29.2% in ICP (7 of 24) patients, this result is in accordance with the reports of De Buys Roessingh and Vedung.23,26 A possible explanation could be the attachment of the vomer, since a report by Bicknell et al. revealed that clefts with an unattached vomer are far more likely to require a PFS.27 The 17.4% frequency of revision flap procedures is in concordance with previous studies providing frequencies of 16–18.8% in case of a two-stage palate repair in the study by Vedung.23 The number of revision procedures differed among the cleft subtypes; it was lowest in SM patients (0%) and highest in BCLAP patients (26.7%), which is supported by previous studies.30,31 The age for initial PFS was significantly lower in patients who required a revision procedure (5.6 versus 6.8 years) (p < 0.05). Pryor et al. reported this factor to be of no significant influence on the revision rate.31 These results have to be taken into account when a decision regarding the need for and timing of PFS is made, weighed against the possible adverse effects on speech development. More studies are necessary to support this conclusion. With regard to the relation between PFS and LFI, a significantly higher frequency (19% versus 8%) of LFI at full-grown age
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was found in patients who underwent a PFS procedure (p < 0.05). Oberoi et al. reported that of 16 UCLAP patients without maxillary hypoplasia, only one patient had had (secondary) PFS (6.25%), compared to a group of 16 patients with maxillary hypoplasia of whom 4 had PFS during palatal repair and 4 underwent PFS as a secondary (following palatal repair) procedure (50%).4 These results were not supported by a statistical investigation. Since the cleft subtype is the factor that most influences the frequency of LFIs, these results have to be adjusted for cleft subtype. In this present study, the frequency of LFI in each cleft subtype is marginally higher in PFS+ patients. None of the differences between the PFS and PFS+ groups in each cleft subtype were significant (p > 0.05), which is most likely the result of the small number of patients in each subtype group. Since this trend is in concordance with previous reports, the PFS procedure can be regarded as one of the iatrogenic factors related to the maxillary growth restriction found in repaired cleft patients. There are many determinant factors influencing maxillary growth. In ideal circumstances, the results have to be corrected for all other potential influences, but since these factors are frequently correlated to each other, for example number of surgeries and cleft subtype, the solitary influence of one determinant is hard to determine. Cleft care is a complex treatment over multiple years and with a multidisciplinary approach. Although for some cleft repair procedures, such as palatal repair, the age at the time of surgery is of significant importance for maxillary antero-posterior growth,25 this is not the case in PFS as shown in this study. It does, however, have an influence on the revision rate. While not the subject of this research, only one of the patients needed PFS after the LFI procedure (n = 24). This fraction of 4.2% velopharyngeal insufficiency after LFI is remarkably lower than the fraction of 25.3% reported Maegawa et al. (n = 91).32 One of the obvious restrictions of this study is its retrospective character and even though it is one of the largest studies evaluating the influence of PFS on maxillary growth, the sample size is still too small to allow for an evidential conclusion. Comparable to previous studies, this study shows a tendency for a negative influence of PFS on maxillary growth in cleft patients when corrected for the influences of other interfering factors, such as
cleft subtype, gender and number of surgical procedures. Larger samples are necessary to maximize statistical credibility. Since the assessment of speech is largely subjective in nature, objective measurements in international accepted terms are difficult. In this study cohort, a team of speech therapist and ENT specialist made the decision on PFS. At present, the indication for PFS in the authors’ institution is assessed during nasoendoscopy. In conclusion, this study shows that the LFI frequency of 19% in cleft patients with pharyngeal flap surgery is significantly higher compared to the percentage of 8% LFI in patients without pharyngeal flap surgery. This finding, in concordance with previous reports, points towards PFS being an iatrogenic factor related to maxillary growth restriction found in repaired cleft patients.
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Funding
None. Competing interests
12.
None declared. Ethical approval
For this study the principles outlined in the Declaration of Helsinki have been followed. The Medical Ethical Commission (METC, ethical board commission) of the EMC accepted this study format and the intended publication on the 7th of July 2010. (MEC-2010-207).
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Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients 19. Morris HL. Velopharyngeal competence and primary cleft palate surgery, 1960–1971: a critical review. Cleft Palate J 1973;10:62–71. 20. Semb G, Shaw WC. Pharyngeal flap and facial growth. Cleft Palate J 1990;27:217– 24. 21. Kuijpers-Jagtman AM, Stoelinga PJ. State of the art in oral and maxillofacial surgery: treatment of maxillary hypoplasia and anterior palatal and alveolar clefts. Cleft Palate Craniofac J 2000;37:421–2. 22. Broadbent Jr BH, Golden WH. The value of an assessment of skeletal maturity in orthodontic diagnosis. Am J Phys Anthropol 1971;35:409–10. 23. Vedung S. Pharyngeal flaps after one- and two-stage repair of the cleft palate: a 25-year review of 520 patients. Cleft Palate Craniofac J 1995;32:206–15. discussion 215–206. 24. Friede H, Enemark H. Long-term evidence for favorable midfacial growth after delayed hard palate repair in UCLP patients. Cleft Palate Craniofac J 2001;38:323–9. 25. Mars M, Houston WJ. A preliminary study of facial growth and morphology in unoperated male unilateral cleft lip and palate sub-
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Corresponding author at: Department of Oral and Maxillofacial Surgery Erasmus University Medical Center Rotterdam Dr. Molewaterplein 40 3015 GD Rotterdam The Netherlands Tel.: +31 107033302 fax: +31 104633098 E-mails: [email protected] [email protected]
Clinical Paper Cleft Lip and Palate
Influence of pharyngeal flap surgery on maxillary outgrowth § in cleft patients
I. E. Voshola, L. N. A. van Adrichemb, K. G. H. van der Wala, M. J. Koudstaala a Department of Oral and Maxillofacial Surgery, Erasmus University Medical Center, Rotterdam, The Netherlands; bDepartment of Plastic and Reconstructive Surgery, Erasmus University Medical Center, Rotterdam, The Netherlands
I. E. Voshol, L. N. A. van Adrichem, K. G. H. van der Wal, M. J. Koudstaal: Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients. Int. J. Oral Maxillofac. Surg. 2013; 42: 192–197. # 2012 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons. Abstract. The aim of this study was to investigate the influence of the pharyngeal flap procedure on the frequency of Le Fort I osteotomies in full-grown nonsyndromic cleft patients. A retrospective review of 508 full-grown cleft patients born between 1 January 1983 and 31 December 1992 was performed. Following data analysis, 140 males older than 18 years and 111 females over the age of 16 years were included. 69 of the 251 included cleft patients required pharyngeal flap surgery (27.5%). Revision flap surgery was performed in 17.4% of the cases. A significantly lower age at time of the initial pharyngeal flap procedure was found in patients requiring revision surgery (5.6 years versus 6.8 years). The frequency of Le Fort I osteotomies was significantly higher in the patients with a pharyngeal flap (19%) compared to those without (8%) (p < 0.05). The results of this study point towards the pharyngeal flap procedure being one of the possible limiting factors for maxillary antero-posterior growth in cleft patients.
Introduction
The aim of cleft treatment is to obtain the best possible functional and aesthetic result when facial growth is completed; but the optimal timing and approach for the surgical management continues to be a source of controversy. Therefore, factors influencing the outcome need to be studied. In general, the presence of maxillary hypoplasia in repaired cleft patients is a § Presented at the Spring Meeting of the Dutch Association of Oral and Maxillofacial Surgery (NVMKA), in Maastricht, the Netherlands, 18 March 2011.
0901-5027/020192 + 06 $36.00/0
well-recognized phenomenon. Most authors advocate the theory that iatrogenic factors, especially primary lip and palate repair, cause the main restriction of maxillary growth.1–4 The Le Fort I advancement osteotomy (LFI) is the standard surgical procedure to treat Angle Class III relationships due to maxillary retrusion.5–7 The incidence of maxillary hypoplasia is often translated into the number of LFI osteotomies performed.8,9 The frequency of LFI varies from 0.0 to 69.6%.4,8–18 The influence of primary lip or palate repair on maxillary growth has been the subject of many reports, but the influence of pharyngeal
Keywords: Cleft; Pharyngeal flap surgery; Le Fort I osteotomy; Maxillary hypoplasia. Accepted for publication 26 September 2012 Available online 2 November 2012
flap surgery (PFS) is seldom discussed and the results are contradictory. This is remarkable, since 20–38% of cleft palate patients require this procedure.19,20 Oberoi et al. delineated factors possibly contributing to maxillary hypoplasia, one of them being PFS. In a group of 16 unilateral cleft lip and palate patients without maxillary hypoplasia only one patient had undergone (secondary) PFS surgery (6.25%), whereas in the group with maxillary hypoplasia 4 of 16 patients had PFS before during palate repair and 4 had PFS as a secondary (after palate repair) procedure (50%); these results were not supported by statistical investigations.4
# 2012 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.
Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients Although some small differences in growth pattern were seen in previous studies on the effect of pharyngeal flaps on maxillary growth, no conclusive evidence was provided.20,21 The sparse number of reports considering the influence of PFS on maxillary growth combined with the small number of patients studied and the short follow up period, show more studies are required. This report attempts to address the possible effect of the commonly performed PFS on subsequent maxillary growth in cleft patients. Additional risk factors for PFS and LFI, including cleft subtype, gender and number of previous surgical procedures, will be evaluated.4,8,9 Materials and methods
508 consecutive cleft patients born between 1 January 1983 and 31 December 1990 (male) and 31 December 1992 (female) and treated at the authors’ institution were evaluated retrospectively. The indication for a LFI was assessed when facial skeletal growth had been completed. Since this is on average 2 years earlier in females (at the age of 16 years) than in males (at the age of 18 years)22 the included birth dates of patients differed between genders. Inclusion criteria were lip and/or alveolar process and/or palate and/or submucous non-syndromal cleft patients, with maturity of the facial skeleton. The treatment was according to the institution protocol as shown in Table 1. Exclusion criteria were cleft patients with other congenital deformities, patients who died before reaching the age of 16 years (females)/18 years (males), midfacial clefts, previous radio- or chemotherapy, incomplete documentation and exceptions to the protocol. Although two-stage palate repair is reported to result in higher percentages of PFS compared to one-stage repair,23 the protocol includes a two-stage palate repair resulting in a superior maxillary antero-posterior growth, supported by a lower percentage of LFI in these patients compared to patients treated with a onestage palate repair.9 Since the cleft in the alveolar process is thought to be the main inhibiting factor for maxillary growth9,15 it was preferred, in agreement with the authors’ previous study,9 to divide the cleft lip group into a cleft lip group and a cleft lip and alveolus group. Overall, patients were subcategorized as shown in Table 2. The following information was collected from the clinical records for each
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Table 1. Cleft protocol Erasmus Medical Center Rotterdam. Lip repair (according to the Millard technique) 3–5 months 9–11 months Closure of the soft palate cleft in case of cleft lip, alveolus and palate (CLAP), closure of the soft and hard palate cleft in case of cleft palate (CP) (according to the Wardill-Kilner, Von Langenbeck, Perko or Kriens technique) 8–13 years Alveolar bone grafting and closure of the hard palate in case of cleft lip, alveolus and palate (CLAP) 16–18 years If necessary: orthognathic surgery after completion of facial skeletal development and secondary nose and/or lip corrections Treatment of submucous cleft is performed in the presence of complaints; an individual treatment plan is developed. Pharyngeal flap surgery is performed when velopharyngeal insufficiency is diagnosed (according to the Orticochea or Roselli technique). Table 2. Categorization of cleft subtypes. Name
Type of clefta
UCL UICL UCCL BCL BICL BACL BCCL UCLA UICLA UCCLA BCLA BICLA BACLA BCCLA UCLAP UICLAP UCCLAP BCLAP BICLAP BACLAP BCCLAP CP ICP CCP SM
Unilateral cleft lip Unilateral incomplete cleft lip Unilateral complete cleft lip Bilateral cleft lip Bilateral incomplete cleft lip Bilateral asymmetric cleft lip Bilateral complete cleft lip Unilateral cleft lip and alveolus Unilateral incomplete cleft lip and alveolus Unilateral complete cleft lip and alveolus Bilateral cleft lip and alveolus Bilateral incomplete cleft lip and alveolus Bilateral asymmetric cleft lip and alveolus Bilateral complete cleft lip and alveolus Unilateral cleft lip, alveolus and palate Unilateral incomplete cleft lip, alveolus and palate Unilateral cleft lip, alveolus and palate Bilateral cleft lip, alveolus and palate Bilateral incomplete cleft lip, alveolus and palate Bilateral asymmetric cleft lip, alveolus and palate Bilateral complete cleft lip, alveolus and palate Cleft palate Incomplete cleft palate (solitary soft palate affected) Complete cleft palate (soft and hard palate) Submucous cleft
a Classification of cleft type in incomplete (I), asymmetrical (A) or complete (C) and unilateral (U) or bilateral (B) only specifies the extent of the cleft lip and not the extent of the cleft alveolus or palate (Good et al. [8]).
case: date of birth, gender, cleft type and extent, and all surgical procedures related to the cleft (lip closure, anterior and posterior palatoplasty, alveolar bone graft surgery, closure of possible fistulas, pharyngeal flap procedure, surgically assisted rapid maxillary expansion). The indication to perform PFS was assessed by a team of ENT specialists and speech therapist. PFS was performed in case of communication problems (i.e. hypernasal speech) due to velopharyngeal incompetence. The criterion was hypernasal speech causing communication problems, which could not be improved (further) by speech therapy. The open nasality was diagnosed in clinical perceptual speech evaluation performed by the team’s speech therapist, by a standard repetitive text, speech with cartoons and
‘free’ speech, combined with a mirror-test (to check the nasal air passage). In cases where PFS was performed, additional information was collected regarding the type of pharyngeal flap procedure performed, age during initial procedure and the necessity for secondary procedures. A secondary procedure was defined as any revision of the sphincter, determined by the clinical evaluation by the treatment team and objective speech assessment by speech pathologist. The need for a LFI osteotomy was recorded by the number of LFI performed or if there had been an indication for LFI osteotomy when fully grown as stated by the multidisciplinary treatment team, even though the surgery had not yet been performed. The frequency of LFI in patients with a pharyngeal flap (PFS+) and without a
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pharyngeal flap (PFS ) was compared. Additional risk factors for both procedures were evaluated, such as cleft subtype, gender and number of previous surgical procedures.4,9,15 Since age at the time of cleft repair influences maxillary growth24,25 the possible correlation between age at initial pharyngeal flap procedure and frequency of LFI was studied. Cephalometric analysis was performed on patients with an indication for LFI osteotomy before and after treatment. Angular measurements recorded were SNA angle (degrees), SNB angle (degrees) and ANB (degrees). The measurements were performed using the digital dicom-data program Easy-ViewWeb (2005, Philips Medical Systems, Best, The Netherlands). Statistical analysis was performed using Microsoft Excel (version 2003) and the statistical Package of the Social Sciences (SPSS, version 17.0). Student’s test and the independent t sample test were used to evaluate data. The limit of significance considered was p = 0.05 (two-sided). For this study the principles outlined in the Declaration of Helsinki have been followed. The Medical Ethical Commission (METC, ethical board commission) of the Erasmus Medical Center accepted this study format and the intended publication on 7 July 2010 (MEC-2010-207). Results
Of the total of 508 patients, 86 had a congenital deformity, 8 patients died before reaching maturity, 2 patients received chemotherapy and in 111 cases the documentation was incomplete. 11 patients changed to another cleft team and 39 patients were not treated according to the institution’s protocol and were consequently excluded. This left 251 patients for inclusion in the study: 140 males (55.8%) and 111 females (44.2%). The miscellaneous group included patients with cleft types that could not be included in the other cleft subcategories, for example, cleft lip combined with a submucous cleft or cleft lip and a cleft palate, without an alveolar cleft. Of the 251 included cleft patients, 69 patients (27.5%) required PFS (Fig. 1). Since the authors’ institution is a tertiary care and teaching hospital, PFS was performed by different surgeons. The group of patients who underwent PFS consisted of 35 males and 34 females. The overall frequency of PFS in males was 25%, compared to 31% in females (p > 0.05). The mean age at the time of initial PFS was 6.6 years (range 3.1– 16.8 years, median 6.4). Of all PFS, 84%
Fig. 1. Number of pharyngeal flap procedures performed in each cleft subtype.
was performed before 9 years of age, and 93% before reaching the age of 10 years. Frequency of PFS was lowest in the unilateral incomplete cleft lip, alveolus and palate (UICLAP) group (20%) and highest in the bilateral asymmetric cleft lip, alveolus and palate (BACLAP) group (58.3%). Frequencies of PFS were 40.0% in the submucous cleft (SM) group, 34.0% in the cleft palate (CP) group, 46.5% in the unilateral cleft lip and palate (UCLAP) group and 51.7% in the bilateral cleft lip, alveolus and palate (BCLAP) as shown in Table 3. PFS was generally performed according to the Orticochea procedure (54 cases, 78.3%). In 8 cases the modification by Jackson was used. In 14 cases the procedure according to Roselli was performed (20.3%) and in one case the PFS procedure was not further specified. The number of revision flap procedures was 17.4% (n = 12), with 7 surgical revisionary procedures in males (20.0%) and 5 in females (14.7%) (p > 0.05). The revision rate varied per cleft subtype: 0% in SM patients, 15.2% in UCLAP patients, 17.6% in CP patients up to 26.7% in BCLAP patients as shown in Table 3. The revision led to a satisfactory outcome in 10 of these patients (97.1%). Two patients (both males) needed a secondary revision. A statistically significant difference was seen in the average age at the initial PFS in patients who later required revision (5.6 years) compared to those who did not need revision (6.8 years) (p < 0.05). In one male UCLAP patient a wound of dehiscence of the flap was repaired after 4 days; this was counted as a revision procedure. Looking at the type of initial pharyngeal flap surgery and the number of revision
procedures, 11 of 54 patients treated with a pharyngeal flap according to Orticochea needed revision (20.4%), whereas of the 14 patients treated with a pharyngeal flap according to Roselli only one pharyngeal flap needed to be revised (7.2%). This difference was not statistical significant (p > 0.05). Patients with PFS+ surgery had a significantly higher frequency of LFI (p < 0.05) compared to PFS patients. 13 patients (19%) of the 69 PFS+ patients required an LFI, but only 15 of 182 (8%) PFS patients required an LFI. The LFI advancements (measured by cephalometric tracing using the ANB) ranged from 2.2 to 19.0 degrees, with an average of 4.3 degrees in patients treated with conventional LFI osteotomy and 7.4 degrees in patients treated using distraction osteogenesis. On average, all patients treated with LFI had an ANB of 3.4 degrees before treatment, and 1.9 degrees following treatment. The frequency of LFI is slightly higher in patients with PFS compared to patients without. None of the differences between the PFS and PFS+ groups in each cleft subtype were significant (p > 0.05) (Table 4). The type of PFS also seems to have an influence on the frequency of LFI. Although the number of patients in each category was small, patients who underwent Orticochea sphincteroplasty (modified according to Jackson) needed an LFI more frequently (10 of 52 patients, 19.2%) than patients who underwent superiorly based flap surgery (e.g. Roselli) (1 of 14 patients, 7.14%). With regard to the age of the cleft patients at the time of initial PFS, no statistically significant difference was found between patients with (6.7 years) and
Influence of pharyngeal flap surgery on maxillary outgrowth in cleft patients Table 3. Pharyngeal flap and revisionary procedures in each cleft subtype. Number of patients
Cleft type
Number of patients with pharyngeal flap
9
Miscellaneous SM
10
ICP
24
CCP
26
UCL
34
BCL
5
UCLA
37
BCLA
6
UICLAP
10
UCCLAP
61
BICLAP
4
BACLAP
12
BCCLAP
13
0 0% 4 40.0% 7 29.2% 10 38.5% 0 0% 0 0% 0 0% 0 0% 2 20.0% 31 50.8% 2 50.0% 7 58.3% 6 46.2% 69 27.5%
251
Total
Number of revisionary procedures 0 0% 0 0% 1 14.3% 2 20% 0 0% 0 0% 0 0% 0 0% 0 0% 5 16.1% 1 50.0% 1 14.3% 2 33.3% 12 17.4%
Frequency of revisionary procedures in each cleft subtype – – 8.3% 16.7% – – – – – 41.7% 8.3% 8.3% 16.7% –
SM, submucous cleft; ICP, incomplete cleft palate; CCP, complete cleft palate; UCL, unilateral cleft lip; BCL, bilateral cleft lip; UCLA, unilateral cleft lip and alveolus; BCLA, bilateral cleft lip and alveolus; UICLAP, unilateral incomplete cleft lip, alveolus and palate; UCCLAP, unilateral complete cleft lip, alveolus and palate; BICLAP, bilateral incomplete cleft lip, alveolus and palate; BACLAP, bilateral asymmetric cleft lip, alveolus and palate; BCCLAP, bilateral complete cleft lip, alveolus and palate.
Table 4. Frequency of LFI in cleft patients with and without pharyngeal flap. Cleft subtype
Total number of patients
SM
10
CP
50
UCLAP
71
BCLAP
29
Pharyngeal flap surgery performed Yes No Yes No Yes No Yes No
Number of patients with/without pharyngeal flap
Number of patients with indication for LFI
4 6 17 33 33 38 15 14
0 0 1 1 7 8 5 4
Frequency of LFI (%) 0 0 5.9 3.0 21.2 21.1 33.3 28.6
SM, submucous cleft; CP, cleft palate; UCLAP, unilateral cleft lip, alveolus and palate; BCLAP, bilateral cleft lip, alveolus and palate.
without (6.2 years) an indication for LFI (p > 0.05). One male bilateral complete cleft lip, alveolus and palate (BCCLAP) patient without pharyngeal flap suffered velopharyngeal insufficiency after the LFI procedure, requiring late pharyngeal flap surgery.
Discussion
The frequency of PFS in cleft patients treated according to the protocol at he authors’ institution was 27.5%, in concordance with percentages reported in the literature. Vedung found frequencies of 20% in complete cleft palate (CCP), 11–
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12% in UCLAP and incomplete cleft palate (ICP) and 14–16% in BCLAP, De Buys describes frequencies of 15.6% in CP, 34.4% in unilateral cleft lip and palate (UCLP) and 48.6% in BCLAP.23,26 Bicknell found an overall frequency of PFS of 25% in cleft patients (28 of 114 patients), subdivided into 12% ICP, 44% CCP, 21% UCLAP, 50% BCLAP and 20% SM.27 These small differences can be explained by the high diversity in cleft types with a relatively large number of patients in the BCLAP/CP group, which have a considerably higher frequency of PFS. The two stage palate repair advocated in the authors’ institution has to be taken into account since this two stage palate repair is reported to result in higher percentages of PFS compared to one stage repair.23 Many authors assume the two stage palate repair has an advantage for maxillary growth so the two stage palate repair is justified in the opinion of the authors’ cleft team.9,24,25 Nevertheless, the authors are aware of recent reports that state that acceptable maxillary antero-posterior growth is accomplished in both single and two stage palate repair.28,29 A factor of major influence seems to be the involvement of the hard palate, since the frequency of PFS is 38.5% in CCP (10 of 26) against 29.2% in ICP (7 of 24) patients, this result is in accordance with the reports of De Buys Roessingh and Vedung.23,26 A possible explanation could be the attachment of the vomer, since a report by Bicknell et al. revealed that clefts with an unattached vomer are far more likely to require a PFS.27 The 17.4% frequency of revision flap procedures is in concordance with previous studies providing frequencies of 16–18.8% in case of a two-stage palate repair in the study by Vedung.23 The number of revision procedures differed among the cleft subtypes; it was lowest in SM patients (0%) and highest in BCLAP patients (26.7%), which is supported by previous studies.30,31 The age for initial PFS was significantly lower in patients who required a revision procedure (5.6 versus 6.8 years) (p < 0.05). Pryor et al. reported this factor to be of no significant influence on the revision rate.31 These results have to be taken into account when a decision regarding the need for and timing of PFS is made, weighed against the possible adverse effects on speech development. More studies are necessary to support this conclusion. With regard to the relation between PFS and LFI, a significantly higher frequency (19% versus 8%) of LFI at full-grown age
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was found in patients who underwent a PFS procedure (p < 0.05). Oberoi et al. reported that of 16 UCLAP patients without maxillary hypoplasia, only one patient had had (secondary) PFS (6.25%), compared to a group of 16 patients with maxillary hypoplasia of whom 4 had PFS during palatal repair and 4 underwent PFS as a secondary (following palatal repair) procedure (50%).4 These results were not supported by a statistical investigation. Since the cleft subtype is the factor that most influences the frequency of LFIs, these results have to be adjusted for cleft subtype. In this present study, the frequency of LFI in each cleft subtype is marginally higher in PFS+ patients. None of the differences between the PFS and PFS+ groups in each cleft subtype were significant (p > 0.05), which is most likely the result of the small number of patients in each subtype group. Since this trend is in concordance with previous reports, the PFS procedure can be regarded as one of the iatrogenic factors related to the maxillary growth restriction found in repaired cleft patients. There are many determinant factors influencing maxillary growth. In ideal circumstances, the results have to be corrected for all other potential influences, but since these factors are frequently correlated to each other, for example number of surgeries and cleft subtype, the solitary influence of one determinant is hard to determine. Cleft care is a complex treatment over multiple years and with a multidisciplinary approach. Although for some cleft repair procedures, such as palatal repair, the age at the time of surgery is of significant importance for maxillary antero-posterior growth,25 this is not the case in PFS as shown in this study. It does, however, have an influence on the revision rate. While not the subject of this research, only one of the patients needed PFS after the LFI procedure (n = 24). This fraction of 4.2% velopharyngeal insufficiency after LFI is remarkably lower than the fraction of 25.3% reported Maegawa et al. (n = 91).32 One of the obvious restrictions of this study is its retrospective character and even though it is one of the largest studies evaluating the influence of PFS on maxillary growth, the sample size is still too small to allow for an evidential conclusion. Comparable to previous studies, this study shows a tendency for a negative influence of PFS on maxillary growth in cleft patients when corrected for the influences of other interfering factors, such as
cleft subtype, gender and number of surgical procedures. Larger samples are necessary to maximize statistical credibility. Since the assessment of speech is largely subjective in nature, objective measurements in international accepted terms are difficult. In this study cohort, a team of speech therapist and ENT specialist made the decision on PFS. At present, the indication for PFS in the authors’ institution is assessed during nasoendoscopy. In conclusion, this study shows that the LFI frequency of 19% in cleft patients with pharyngeal flap surgery is significantly higher compared to the percentage of 8% LFI in patients without pharyngeal flap surgery. This finding, in concordance with previous reports, points towards PFS being an iatrogenic factor related to maxillary growth restriction found in repaired cleft patients.
6.
7.
8.
9.
10.
11.
Funding
None. Competing interests
12.
None declared. Ethical approval
For this study the principles outlined in the Declaration of Helsinki have been followed. The Medical Ethical Commission (METC, ethical board commission) of the EMC accepted this study format and the intended publication on the 7th of July 2010. (MEC-2010-207).
13.
14.
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Corresponding author at: Department of Oral and Maxillofacial Surgery Erasmus University Medical Center Rotterdam Dr. Molewaterplein 40 3015 GD Rotterdam The Netherlands Tel.: +31 107033302 fax: +31 104633098 E-mails: [email protected] [email protected]