Annals of Pediatric Surgery, Vol 4, No 1,2, January-April, 2008 PP 22-36

Original Article

Sphincter Pharyngoplasty: the One Procedure That Fits All Patterns of Closure in Velopharyngeal Insufficiencies*
Amir Elbarbary, MD, Hassan Ghandour, MD
Plastic & Reconstructive Surgery Department & Phoniatric Unit, ENT Department, Faculty of Medicine, Ain-Shams University

Abstract Background/ Purpose: Velopharyngeal insufficiency occurs in a considerable number of patients following cleft palate repair. It disrupts speech intelligibility leading to breakdown of the ability to communicate verbally. Substantial uncertainty occurs in choosing between sphincter pharyngoplasty and pharyngeal flap in restoring the velopharyngeal function. This prospective study aimed to assess the treatment outcome of modified sphincter pharyngoplasty applied to patients with residual velopharyngeal insufficiency following palatal repair regardless of their pattern of velopharyngeal closure. Materials & Methods: Preoperatively, patients underwent in-depth speech and endoscopic evaluations for symptoms of velopharngeal insufficiency. Six to twelve weeks following the surgical procedure they underwent the same thorough evaluation prior to receiving any speech therapy. Speech evaluation was carried out using the protocol of assessment that is applied in the phoniatric unit, Ain-Shams University which included auditory perceptual assessment (APA), nasopharyngeal videofibroscopy and nasometry. Results: Forty three patients were included in this study. Statistical analysis of the results documented a significant reduction in the degree of open nasality, glottal articulation and pharyngalization following a modified sphincter pharyngoplasty. A significant increase in the overall intelligibility was delineated regardless of the pattern of velopharyngeal closure. Postoperatively, velopharyngeal port achieved functional closure in the majority of patients as detected by nasopharyngeal videofibroscopy and was categorized as circular in thirty patients and coronal in thirteen. Conclusion: The results of this study demonstrated that sphincter pharyngoplasty could be applied effectively to patients with velopharyngeal insufficiency following cleft palate repair regardless of their velopharyngeal pattern of closure. Index Word: Velopharyngeal inefficiency, sphincter pharyngoplasty, closure pattern.

INTRODUCTION

elopharyngeal insufficiency (VPI) refers to excessive nasal resonance or hypernasality as the consequence of anatomical abnormalities and failure of the velum and the pharyngeal muscles to

produce optimal sphincter-like closure between the oro- and nasopharynx.1 It occurs in a substantial number of patients after cleft palate repair2-5 and can be attributed to a variety of factors: scarring as a

*Presented at the 22nd annual meeting of Egyptian Pediatric Surgical Association (EPSA) 14-16 December 2006

Correspondence to: Amir Elbarbary, MD, Plastic & Reconstructive Surgery Department, Faculty of Medicine, Ain-Sham University, Phone: + 20 12 228 7582, Email: amir_elbarbary@yahoo.com

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result of the initial palatoplasty can shorten the velum; making it impossible for the velum to reach the posterior pharyngeal wall "target" during speech; a deep nasopharynx relative to the position of the velum; a poor velar movement despite an adequate length resulting from insufficient restoration of the palatal muscle sling at the time of primary repair.6 Velopharyngeal insufficiency results in the inability of the cleft patients to communicate coherently and is considered the most disabling and devastating result among the various secondary problems that may follow cleft lip/palate repair.7 When surgical management is indicated for restoration of the velopharyngeal function, the pharyngeal flap and the sphincter pharyngoplasty are among the most commonly used surgical procedures8. Considerable uncertainty of choice exists both within variations of flap and sphincter pharyngoplasty and between the two approaches. Authorities such as Riski9 agree that if surgical intervention is needed, the procedure should be tailored to the size and nature of the velopharyngeal defect. However, reports of morbidity and mortality associated with pharyngeal flap surgery10-16 have led a lot of operators to adopt sphincterplasty instead. Several publications have advocated sphincter pharyngoplasties17-19 citing their additional advantages as (1) technical ease of execution, (2) superior speech results, (3) low complication rate, (4) reduced anaesthesia time, (5) non-obstruction of the nasal airway. The sphincter pharyngoplasty operation is designed to form a ridge on the posterior pharyngeal wall, narrow the pharynx from side to side, and to produce a sphincteric type of closure.20 The objective of the procedure is to create a muscular valve capable of isolating the nasal cavity from the remainder of the vocal tract during appropriate speech tasks. This is necessary to eliminate hypernasality and to allow oral pressure to build in the oral cavity for the production of many consonant phonemes.21 Several modifications of sphincter pharyngoplasty have been described since it was first introduced by Hynes22-24 who used superiorly based flaps from the salpingopharyngeus. Orticochea25 used palatopharyngeus instead and sutured them to an inferiorly based pharyngeal flap to below the palatal plane. Jackson & Silverston26 replaced the inferiorly based flap by a superiorly based posterior wall flap in an attempt to raise the flap insertion and improve the outcome. Despite the reported high success rate
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following sphincter pharyngoplasty,27 a number of patients have persistent unacceptable vocal resonance and residual air escape postoperatively.21,28 Advances in patient selection and surgical technique to enhance successful valving of sphincter pharyngoplasty have been reported.8,28 In an attempt to enhance the success of the sphincter palatoplasty and further improve the outcome, a modified sphincter pharyngoplasty is presented. It included the elevation of bilateral superiorly based palatopharyngeus muscle with overlying mucosa that are sutured overlapped to each another and to a transverse incision on the posterior pharyngeal wall at the level of attempted velopharyngeal closure. The aim of this prospective study is to assess the treatment outcome for patients with residual velopharyngeal insufficiency after palatal repair undergoing a modified technique of sphincter pharyngoplasty regardless of the pattern of velopharyngeal closure.

PATIENTS AND METHODS

Patients diagnosed with residual velopharyngeal insufficiency after cleft palate repair presenting to the outpatient cleft palate clinic at Ain-Shams University Hospital from January of 2004 to December of 2006 had been considered potentially eligible for the study. Similar to other studies,8,29 the patients had to meet the following criteria to qualify for the study: (1) undergone a primary repair of the palate (with or without a cleft lip or alveolus), (2) chronological age between 4 and 16 years with apparent VPI diagnosed by an experienced speech specialist, (3) had at least 75% of normal language development for their age. Exclusion criteria comprised patients with: (1) size of the velopharyngeal gap exceeding 2 cm in anteroposterior dimension which necessitated a lengthening procedure, (2) hearing impairment, (3) the cleft being part of a syndrome, (4) any preexisting palatal fistulae, (5) obstructive sleep apnea syndrome. The surgical procedure and study methods were carefully explained to all parents. Patients underwent in-depth speech and endoscopic evaluations for symptoms of velopharyngeal insufficiency. Six to twelve weeks, with a mean of two months, following the surgical procedure they underwent the same thorough evaluation prior to receiving any speech
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therapy if they needed any. Evaluation was carried out using the protocol of assessment that is applied in the phoniatric unit, Ain-Shams University which includes subjective as well as quasi-objective measures of evaluation30.This protocol includes: I- Preliminary Diagnostic Procedures: Auditory Perceptual Assessment (APA) was used as a subjective tool for evaluation of patients' language, speech and voice through listening to every patient in a free conversation and a recorded speech sample. Passive and active aspects of language were investigated including semantic, syntactic and pragmatic aspects. Speech evaluation included the type and degree of open nasality, consonant precision, the compensatory articulatory mechanisms (glottal articulation, pharyngealization of fricatives, and facial grimace), audible nasal air escape and overall unintelligibility of speech. All these elements are graded along a 5-point scale in which 0 = normal and 4 = severe affection. II-Clinical Diagnostic Aids: (A) Nasopharyngeal videofibroscopy: All patients were examined using nasopharyngeal video-fibroscopy Henke-Sass-Wolf, type 10, connected to a Lemke video camera (MC 204) and Panasonic video cassette recorder 357. The nasofibroscope was introduced through the nasal cavity to a position superior to the soft palate. The velopharyngeal valve movement was recorded while the patient repeated the speech samples applied in the protocol of assessment of VPI in the phoniatric unit, Ain-Shams University.30 Movements of the velum, lateral, and posterior pharyngeal walls were traced on the monitor. The movement of each component was given a score from 0 to 4 as follow: 0 = the resting (breathing) position or no movement, 2 = half the distance to the corresponding wall, 4 = the maximum movement reaching and touching the opposite wall. Also,the pattern of closure of the velopharyngeal port, whether coronal, sagittal, circular or others were specified and recorded. (B) Nasometry: Nasometery was performed to all patients using Kay nasometer model 6200-2 with a software version 1.5. It is composed of a head set, microprocessor and a printed circuit board. Every patient was asked to repeat (with a normal conversational loudness, while sitting comfortably
Annals of Pediatric Surgery

on a chair) a nasal loaded sentence /mama betnajem mana:l/ and an oral sentence devoid of nasal sounds / ali rah jel ab korah/ according to the protocol of assessment of VPI in the phoniatric unit, Ain-Shams University.30 The nasometer calculates the nasalance which is the ratio of the nasal to the nasal plus oral acoustic energy multiplied by 100. The degree of hypernasality depends on the percent nasalance.31 Similar to Abyholm et al,8 the nasalance results were reported as an overall measure of preoperative and postoperative mean changes as opposed to comparing each patient with the norm. All patients included in the study received a modified sphincter pharyngoplasty regardless of the pattern of velopharyngeal closure or the severity of the symptoms. The retractor and tongue blade were adjusted to completely expose the palatopharyngeus folds in the operating field. The soft palate was retracted supoeriorly to expose the posterior pharyngeal wall as high as the adenoid. A submucous injection of 1:200,000 adrenaline was injected into the operative field. Vertical incisions were made in front of and behind the posterior tonsillar pillar starting from the upper limit of the tonsillar recess. With a Metzenbaum scissor, the palatopharyngeous muscle was elevated with its overlying mucosa. After obtaining as much vertical length as possible, the superiorly based myomucosal flap was divided inferiorly and elevated with a right-angled scissor. The donor site was closed. The same steps were repeated to elevate the contralateral flap. A transverse incision down to the prevertebral fascia is placed approximately 2 mm below the adenoidal pad to which the flaps are rotated 90 degrees. The flaps were interdigitated and sutured together tip-tobase and to the posterior pharyngeal wall without leaving any lateral ports. During recovery from general anaesthesia, the patients were placed in a compulsory posture with the head turned to one side. They were encouraged to start liquid diet on the first day of operation as soon as they were fully recovered. Any perioperative complications in recovery or any later complications including airway complications, readmission to hospital and reoperation were recorded. Statistical analysis was done using paired t-test with P>0.05 indicating no significance. P<0.05 indicated significance, P<0.01 indicated high

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significance, and P<0.001 indicated very high significance.

RESULTS
Forty three patients whom fulfilled the criteria were included in the study. They were twenty six males and seventeen females. Twenty seven patients had cleft lip and palate with four being bilateral. The remaining sixteen patients had isolated cleft palate. The mean age of the patients at the time of the operation was seven years and five months. Intraoperatively, blood loss was minimal and the mean length of surgery was 45 minutes. All patients experienced a smooth postoperative recovery and were discharged from the hospital on the second postoperative day. There were no severe complications, such as obstructive dyspnea or bleeding. Almost all patients complained of snoring during sleep in the first couple of weeks. The postoperative snoring disappeared in an average of 2-3 months following the operation. None of the patients was reoperated upon for airway obstruction. Only in one patient, flap dehiscence occurred and was revised. Six speech variables were rated pre and postoperatively. The degree of open nasality was reduced in 90.5% of patients. Of these patients 44% improved only one scale point, 37% improved two scale points, while the remaining 9.5% improved three scale points (Fig. 1). No patients were rated as

hyponasal postoperatively. The compensatory glottal articulation showed no change in 25.5% of patients, improved one scale point in 28% of patients, and more than two scale points in the remaining 46.5% (Fig. 2). pharyngealization of fricatives remained unchanged in 25.5% of patients and improved in 74.5% of patients. Improvement of one scale point was seen for 37% of patients, and improvement of more than one scale point was observed for the remaining 37.5% (Fig. 3). Facial grimace was not present in 15 patients preoperatively. From those presenting with facial grimace on evaluation preoperatively 60.5% did not show it post operatively. Audible nasal escape could not be verified in two patients preoperatively, and became absent in 78% of those that had before surgery. Ratings of speech overall intelligibility (Fig. 4) improved significantly in 86% of patients; one scale point improvement in 30% of patients and 56% for two or more scale point improvement. Speech intelligibility was judged as showing deterioration in 4.5% of patients and no change in 7% of patients. A significant change was found in nasalance scores between the mean preoperative and mean postoperative scores. With the normal value being 10.5 for oral sentence, a mean score of 44.3 preoperatively was calculated compared to a mean score of 21.1 postoperatively with 23.2 difference. As for the nasal sentence, a mean score of 64 preoperatively was recorded compared to a mean score of 51.5 postoperatively, with the normal value being 48.5.

Fig 1. (A) Improvement in open nasality in scale points from preoperative to the postoperative condition. (B) Comparison between preoperative and postoperative degree of open nasality. There was a very highly significant increase in the number of patients with grade 0 open nasality, a highly significant increase in the number of patients with grade 1, and a significant increase in the number of patients with grade 2. A highly significant decrease in number of patients with grades 3 and 4 open nasality was found. 25 Vol 4, No 1,2, January-April, 2008

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Fig 2. (A) Improvement in glottal articulation as a compensatory mechanism in scale points from preoperative to the postoperative condition. (B) Comparison between preoperative and postoperative glottal articulation. A highly significant increase in the number of patients with grade 1 and a significant increase in the number of patients with grades 0 & 2. A highly significant decrease in the number of patients with grade 3, and a ver highly significant decrease in the number of patients with grade 4 glottal articulation.

A
Fig 3.

(A) Improvement in pharyngalization as a compensatory mechanism in scale points from preoperative to the postoperative condition. (B) Comparison between preoperative and postoperative degree of pharyngalization. There was a significant increase in the number of patients with grade 0 and a very highly significant increase in the number of patients with grade 1. No statistical significance was found among the increase in number of patients with grade 2. A highly significant decrease in the number of patients with grades 3 & 4 pharyngalization was detected.

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Fig 4. (A) Overall speech intelligibility was rated on a 6-point scale. Ratings showed improvement from preoperative to the postoperative condition in 86% of patients. (B) Comparison between the preoperative and postoperative grade revealed that there was a significant decrease in the number of patients with grade 0, and a very high significance decrease in the number of patients with grade 1. There was a significant increase in the number of patients with grades 2 & 4, and a very high significant increase in the number of patients with grade 3 overall intelligibility.

A B C Fig 5. Nasofiboscopic view of one of the cases classified with excellent postoperative overall intelligibility. (A) Preoperative view demonstrating the gap at rest. (B) postoperative view demonstrating an open sphincter in repose and during pronunciation of nasal phonemes (C) sphincter with circular pattern of closure upon pronunciations of oral phonemes. (* asterisk denotes posterior pharyngeal wall, and the arrow refers to the area of velopharyngeal port.

The velopharyngeal port, as revealed by the nasopharyngeal videofibroscopy was categorized preoperatively as coronal in 28 patients, sagittal in 8, and circular in 7 patients. Postoperatively, velopharyngeal port achieved functional closure in
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the majority of patients and was categorized as circular in thirty patients and coronal in 13. Out of the 8 sagittal closures preoperatively, 6 became coronal while the remaining two became circular. The grade of movement in attempting closure improved in all
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except for one of the two that became circular with movement judged as remaining similar to the preoperative grade of movement. All preoperative circular closures remained as such except for one that became coronal. It was only in this one that the degree of movement remained as preoperative, while improving in the rest. The majority of coronal closures (n=22) became circular and only 6 remained as coronal in the postoperative evaluation. Two of the 22 that became circular were judged as deteriorating in the degree of movement as opposed to an improvement in the remaining 20. All of the 6 that remained coronal showed an improvement in the degree of movement except for one that remained with the same grade as preoperatively. The nasopharyngeal sphincter showed remarkable contraction during speech, but not to the extent of closing the velopharyngeal port completely in all patients (Fig. 5)

DISCUSSION
The sphincter pharyngoplasty described here is a variation of older concepts. Even though reconstruction of the velopharyngeal sphincter anatomically was attempted in 1935 by Browne32 through placing a constricting suture around the entire oronasal port at the level of Passavant's ridge, Hynes22 was, undoubtedly, the first to introduce the "lateral" pharyngoplasty as a method of treatment of velopharyngeal insufficiency in 1950. He raised flaps from the lateral pharyngeal walls then closed the donor defects to narrow the pharynx. Medial interpolation and crosslapping of the two flaps produced a horizontal shelf above Passavant's ridge to bring the posterior pharyngeal target closer to the velum. He believed that his technique would be more functional since the nerve supply to the lateral pharyngeal muscles comes from a superior origin. Any flap lifted laterally would contain neuromuscular elements and would contract. In his first paper, Hynes23 described the flaps containing the salpingopharyngeus muscles but in his Hunterian lecture in 1953 he made it clear that he also raised the palatopharyngeus and the fibers of the underlying superior constrictor. Further observations on this type of operation were made by Hynes24 in 1967 and the results were later reviewed by Pigott.20 Moore33 also raised the salpigopharyngeus muscle but rather used it to augment the posterior margin of the soft palate.

In 1968 Orticochea25 described the construction of a "dynamic" pharyngeal muscle sphincter in cleft palate patients by suturing the tips of the lateral flaps, containing palatopharyngeus, onto the superior end of a third low inferiorly based posterior pharyngeal flap. The flaps were not sutured to the posterior pharyngeal wall laterally. A modification of the latter that has gained so much popularity was introduced by Jackson & Silverton in 1977.26 They felt that the term "sphincter" was more appropriate for this type of functional pharyngoplasy. A superiorly based midline flap, raised high on the posterior pharyngeal wall, substituted for the low inferiorly based flap of Orticochea. Further modifications of the technique have been made. Most have centered on obtaining a superior placement and covering raw tissue areas.34 Ren & Wang35 Sutured half of the wounds on the palatopharyngeus flaps to form a tubed pedicled flap and left only distal free ends. These ends were sutured together in a "lateral to lateral" way, then joined the raw surface of the superiorly based posterior pharyngeal flap. The modification in the technique presented takes into account the evolution and advantages of each of the sphincter procedures and simplifies them. It avoided some of the intrinsic deficiencies that were present through eliminating all raw areas of the earlier procedures and raising the level of inset by discontinuing the use of the pharyngeal flap that complemented earlier modifications of all sphincter procedure. The palatopharyngeal flaps were sutured overlapped tip-to-base similar to the original description by Hynes to further narrow the velopharyngeal valve in a static manner. In agreement with Sie et al17 and Witt et al36, the width, length, and level of insertion of the palatopharyngeal flaps, as well as the degree of overlap of the transposed flaps, can be modified to suit the requirements of any individual patient. The degree of tightness and closure of the sphincter is therefore determined by all these factors. Reid37 and Abyholm38 suggested that large fistulas might be detrimental to speech. Cosman & Falk39 reported on general speech effects associated with palatal fistulas. Isberg & Henningsson40 studied the influence of palatal fistulas on velopharyngeal movements and found a statistically significant correlation between the fistula size and the degree of lateral wall movement but not with the velar movement. Furthermore, they demonstrated an improvement in velopharyngeal movements when

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the fistula was covered and concluded that even small fistulas impair the velopharyngeal activity. Therefore, any preexisting palatal fistula/fistulae was repaired first before the patient was included in this study. This is similar to the designs of other studies.8,29 While several studies have compared posterior pharyngeal flap and sphincter pharyngoplasty in terms of speech outcome or complications, there is not, as yet, a consensus regarding the specific choice of one versus the other for surgical management of velopharyngeal insufficiency. Several reports have suggested that there cannot be one single approach because velopharyngeal physiology varies from one individual to another. Thus, a single operation is not likely to correct all cases of velopharyngeal insufficiency because closure defects at the velopharyngeal sphincter have been noted to vary in size, position, shape, and consistency.5,28,29,41,42 Other reports have demonstrated superior results of one approach over the other.17-19 The evidence for all these contradicting views is generally weak and difficult to resolve. Even the reliability of the few randomized trials that has been performed and found no statistical difference between the different procedures29,43,44 is inevitably prejudiced by important sources of bias.8 These could include small number of patients and homogenecity of sample included in the studies45 as well as comparisons among groups of cases without baseline equivalence in the degree of VPI, age of the patients, ability of patients to modify the learned speech abnormalities, variables in closure defects, secondary deformity, or among cases that received surgery from operators with different levels of skill. In addition, false conclusions may arise from group differences in follow-up, diagnostic measurement, and reporting.46 Having these limitations in mind, this study was designed to evaluate one surgical technique in different patterns of closure rather than comparing different techniques in the presence of a lot of variables. It should be noted though that unlike other studies, cases in this study were not confined to those whom received their primary repair at our institution but rather included a heterogeneous group of patients to increase the sample size and to further validate the outcomes. Choosing sphincter pharyngoplasty was based on its increasing popularity and its numerous advantages cited in literature. The velopharyngeal sphincter as described by Passavant in 186247 is formed bilaterally by the superior constrictor muscles, the palatopharyngeal muscle, and the levator veli
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palatine. Its function is to distribute the column of air leaving the pharynx during speech and direct it through the nose or mouth. Orticochea48 believes that movements and functions of the Passavant sphincter are represented in the brain rather than the muscles. Therefore, the palatopharyngeal muscle that is elevated and relocated into a transverse incision at the posterior pharyngeal wall is considered more physiological in substituting the Passavant sphincter because it will have the similar cerebral representation and mimics the same pattern of closure. Supporters of the sphincter pharyngoplasty found that the pharyngeal flap divides the velopharyngeal area into two lateral ports. They believe that it disturbs the superior constrictor function, disrupting the palatopharyngeus superior constrictor sphincter with loss of its mechanical advantage. Ultimately, the pharyngeal flap weakens the posterior and lateral pharyngeal wall movement49. Shprintzen et al50 found that unless the pharyngeal flaps are tailored to the size of the gap, the velopharyngeal insufficiency and hypernasality are likely to persist. They demonstrated that, most often, the lateral wall movement does not adapt to the presence of this new structure in the pharynx. To the contrary, surgeons who prefer the pharyngeal flap believe that the sphincter mechanism is not disrupted since the posterior pharyngeal wall is always repaired after raising the flap. In agreement, Karling et al51 found that the magnitude and character of change in pharyngeal wall adduction was significantly correlated with the degree of preoperative adduction and with the width of the flap. Although they verified an increase in lateral wall activity when narrow pharyngeal flaps were elevated, they also documented a decrease in the lateral wall activity when the flaps were wide. They attributed this to the mechanical hindrance by the large flap, which proves in a way the argument of the sphincterpharyngoplasty advocates. Moreover, they stated51 in their conclusion, that their results cannot be interpreted as generally applicable because of the strict selection of patients. Regardless of these conflicting reports, it doesn't seem logical to base the surgical plan of a patient on his lateral wall mobility when this postoperative movement is uncertain and could be affected in any form or degree following the pharyngeal flap elevation; it simply defeats the purpose. Especially when there is a more physiological option; namely the sphincter pharyngoplasty that is known to preserve the sphincter with minimal interference of the pharyngeal wall anatomy,49 advance the posterior wall and
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reduce the lateral recess thus circumference with resultant velopharyngeal port area.20

narrowing decrease

the in

The size and shape of the residual velopharyngeal gap have been determinant factors in implementing the different surgical modalities for the correction of velopharyngeal insufficiency.52 Pharyngeal flaps have been recommended when the residual velopharyngeal gap is sagittal indicating a strong lateral wall movement and weak palatal mobility, while sphincter pharyngoplasty is indicated when the residual velopharyngeal gap is coronal indicating poor lateral wall movement and a strong velar movement53-56. Armour and colleagues19 further emphasized on this and confirmed that pharyngeal flaps are less effective in treating velopharyngeal insufficiency in patients with coronal closure. Consequently, they changed the historic pattern of treatment at The Hospital for Sick Children in Toronto that implied the pharyngeal flap for all velopharyngeal insufficiencies regardless of their closure patterns; and became more inclined to treat coronal pattern velopharyngeal insufficiencies with sphincter pharyngoplasty. Similarly, de Serres et al18 examined their experience with the sphincter pharyngoplasty and pharyngeal flap procedures after their results seemed to indicate that less than optimal results were being obtained with the pharyngeal flap procedure. They found Sphincter pharyngoplasty to have a higher success rate and tended to recommend it more liberally while abandoning the pharyngeal flap procedure at their institution, although they still consider it as a management option depending on operator preference. It is well documented1,57,58 that coronal closure is the commonest pattern of closure accounting for 55% while sagittal closure accounts for only 10-15%. This was comparable with the findings in this study; coronal closure represented 65% of the cases while sagittal 18.5%. Therefore, having the coronal closure as the commonest pattern together with the hazard of potential impairment of the lateral wall movement when elevating a pharyngeal flap,49 offers the sphincter pharyngoplasty, which interferes less with the pharyngeal wall anatomy and is indicated in the coronal pattern of closure, a better chance of outcome in all circumstances. Jackson59 once said that "as soon as surgeons become experienced and comfortable with sphincter pharyngoplasty, they will find it the most common rehabilitative measure employed". This should be because properly repaired palates should
Annals of Pediatric Surgery

have good mobility (coronal pattern of closure), and therefore one shrinks from placing anything in the soft palate that might in any way interfere with this much valued movement. When the palate is not functioning properly (sagittal pattern of closure), an initial reconstruction of the soft palate levator mechanism should be carried out. If a degree of velopharyngeal insufficiency is still present, then patients can be very well rehabilitated with sphincter pharyngoplasty. Although this approach involves two operations, it is certainly more anatomically and physiologically sounds than opting immediately for a pharyngeal flap. It seems logical that when the soft palate is functioning adequately but is short and there is no viable way to make the palate longer, then the answer is to maintain the good palatal function and reduce the dimensions of the velopharyngeal mechanism by performing a sphincter pharyngoplasty. This becomes even more logic if we add to it the further improvement in the palatal elevation that has been considered to be one of the additional advantages of the sphincter pharyngoplasty.26,60 Georgantopoulou and coworkers61 studied in detail the effect on velar mobility and demonstrated a significant increase in the range of movement of the soft palate following different types of sphincter pharyngoplasty. They explained their findings based on the fact that elevating the superiorly based posterior tonsillar pillar flaps divided the palatopharyngeous muscle. This in turn liberated the levator palati from its antagonist, the palatopharyngeous, to act unopposed62 resulting in an increased velar elevation. Although, their explanation might have been a simplistic view to a very complex interaction between the muscles of the soft palate63, it still holds logic and validates their findings. Some of the closure patterns in this study changed into coronal postoperatively, indicating a stronger velar mobility. Out of 8 sagittal closures preoperatively, 6 became coronal which concurs Georgantopoulou's et al61 explanation. The concept of dynamic pharyngoplasty arose from Orticochea's25,64 observations of the process of deglutition. He speculated that the posterior tonsillar pillars with enclosed palatopharyngeus muscle which acts during the gag reflex could be engaged for speech production. Hence, sphincter pharyngoplasty was designed to change the lower insertion of the posterior pillars from the lateral walls to the posterior wall on the pharynx. Witt et al65 tested this theoretical advantage of dynamic activity of the

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neovelopharyngeal port. They evaluated 58 patients who underwent sphincter pharyngoplasty by speech videofluroscopicy. Their results indicated a quantifiable and statistically significant excursion of sphincteric closure concluding that sphincter pharyngoplasty works in a dynamic and active manner. In another study, they66 found little evidence to suggest that preexisting posterior pharyngeal wall motion caused the sphincteric movement. To the contrary, Ysunza et al42 and Ysunza67 suggested that neither the lateral pharyngeal flaps in cases of sphincter pharyngoplasties nor the central pharyngeal flap in cases of pharyngeal flaps created new sphincters for velopharyngeal closure by using selective electromyography and simultaneous videonasopharyngoscopy. The participation of these structures is passive, increasing tissue volume in specific areas, whereas their movements are caused by the contraction of the superior constrictor pharyngeus and the levator veli palatini. It is interesting to sight that they were cautious about their observation and stated that the small number of their study does not allow definite conclusions. In all circumstances, the sphincter pharyngoplasty will work as described by Pigott20 in any of three ways: as an active sphincter, or at least by advancing the posterior wall and by reducing the lateral recess in a static manner. The nasopharyngoscopic findings of this study concurred with Witt et al65,66 results and demonstrated a dynamic neosphincter in all cases. However, the origin of this activity was not tested for since no electomyographic study was conducted. The majority of the cases demonstrated a circular pattern of closure indicating participation of velum and pharyngeal wall in the movement. While in the remaining cases, the velar movement was more significant than the pharyngeal wall and was accounted in as coronal pattern of closure. It is interesting to observe from the results of this study that the five patients whom overall speech intelligibility were judged as unsatisfactory following surgery were distributed over the different patterns of closure. In three of the patients whom had the same preoperative and postoperative overall speech intelligibility, one was a sagittal that became circular, the second was circular and became coronal, while the last was the coronal that remained as such postoperatively. Speech intelligibility was reviewed as showing deterioration only in two patients out of the 22 patients that changed from coronal preoperatively into circular postoperatively. Therefore, these few unsatisfactory

results could be rather explained by factors other than the pattern of closure. Huang et al68 examined the blood supply of the entire velopharyngeal complex. They described the pharyngeal flap, from a vascular standpoint and in agreement with Mercer & MacCarthy69 as being random in nature. The lateral incisions for superiorly or inferiorly based flaps invariably divide the perforators from the ascending pharyngeal artery that supplies the superior constrictor as these vessels run transversely. Although this certainly does not preclude flap viability, it could be a contributing factor to the common sequel of unpredictable flap shrinkage. On the other hand, the pattern of blood supply to the faucial portion of the palatopharyngeus was observed to be segmental, with a number of branches of the ascending palatine artery entering the muscle throughout the length of the tonsilar pillar. Therefore, even if the flaps were raised up to or beyond the superior pole of the tonsil, the base of each flap would probably contain at least the hamular branch of the ascending palatine artery, ensuring an adequate axial blood supply to the flap. This concured with the opinions of Boorman & Freedlander70 and probably explains why flap necrosis in this procedure is a rare phenomenon. An intraoperative finding of flap retraction following its elevation was observed in all cases of this study indicating the preservation of its neurovascular supply. Although healing around the orifice of the velopharyngeal sphincter is not totally controllable following sphincter pharyngoplasty, it is still much more controllable than healing around the lateral ports of the pharyngeal flap. Jackson59 emphasizes that only one posterior vertical suture line is incorporated in the recreated sphincter and therefore contracture of the sphincter due to scarring is unlikely to occur. In the modification presented here, even this posterior scar has been eliminated. The fact that the velopharyngeal aperture does not retract by scarring, in this modification, because of the lack of raw areas and circumferential incisions is significant. There have been several reports of disastrous total closure of the velopharyngeal area, airway obstruction and death associated with posterior pharyngeal flap surgery.10-16 Valnicek et al14 reviewed a 7-year experience with superiorly based pharyngeal flap in a total of 219 children at The Hospital for Sick Children in Toronto. Complications included 18 children (8.2%) with bleeding, of whom 5 required transfusion; 20 children (9.1%) with airway
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obstruction; and 9 (4.1%) with sleep apnea after discharge from the hospital. Three patients required reintubation in the early postoperative period, and 11 required eventual surgical revision, including complete takedown of the flap in four patients. To the contrary, Jackson59 stated that he has never seen a case of sleep apnea with sphincter pharyngoplasty and attribute this to the fact that scarring is minimal and occurs in a vertical rather than a horizontal direction. Obstructive sleep apnea seems to be more frequent, if not almost exclusively, associated with posterior pharyngeal flap surgery.62 The literature does not contain as many reports of airway problems with sphincter pharyngoplasty as with pharyngeal flap. Extraordinarily, Witt et al71 demonstrated that airway dysfunction can occur following sphincter pharyngoplasty. They reported perioperative and/or postoperative airway dysfunction in a minority of their patients. However, these patients had either Pierre Robin sequence, micrognathia, or histories of perinatal respiratory and/or feeding problems. In all of them, airway dysfunction resolved within 3 days postoperatively without the need for a surgical takedown of the sphincter pharyngoplasty to relieve the airway problems. In this study none of the patients suffered airway dysfunction nor sleep apnea; only snoring at night that persisted for three months in some cases. Finally, a very important issue is the ability to salvage failures with further surgery. Jackson59 stated that sphincter pharyngoplasty can be used to rehabilitate the patient who has failed a pharyngeal flap. Moreover, in patients requiring Le Fort I or Le Fort III advancements, the sphincter has never caused any obstruction to advancement. This problem occurred on several occasions with pharyngeal flaps, which, when tight and scarred, have required division. Even then, rehabilitation has been performed using sphincter pharyngoplasty six months after the maxillary advancement. Advocates of sphincter pharyngoplasty have listed the ability to easily and successfully revise port size as one of its advantages over pharyngeal flap62. Revision surgeries were described as being without difficulty72. The patients whom required revisions because of persistent hypernasality were associated with flap dehiscence, low-lying flaps, and end-to-end (as opposed to endto-tip) flap suturing72. In this study only one patient required revision because of flap dehiscence. This was one of the patients that were operated upon early in this series and the flaps were set high into the adenoid tissues were the sutures did not hold.
Annals of Pediatric Surgery

It should be emphasized that the results in this study were evaluated early between 6 and 12 weeks postoperatively and prior to establishment of speech therapy to assess the role of the surgical procedure alone. Abyholm et al8 demonstrated that the sphincter pharyngoplasty achieved correction more slowly and that one year follow ups provided better outcomes than at three months. The original design of this study was to carry a second follow up evaluation at one year. However, because of the non compliance of the majority of patients, only a small sample size were followed up after one year and no conclusive outcomes could be withdrawn in this regard. Nevertheless, the results of this study indicates that a modified sphincter pharyngoplasty improved speech overall intelligibility significantly in 86%. This is comparable with the 74% success rate of Ren & Wang,35 79% improvement of Witt et al,73 85% success rate of Abyholm et al8, 91% of Jackson & Silverton26. Orticochea48 reported his observations accumulated over 40 years in treating velopharyngeal insufficiency. He recognized a variety of factors that influenced the success rate of dynamic sphincter pharyngoplasty outcomes. He considered the age of the patient as one of them; the older the patient, the more deeply engraved will be the cerebral engram patterns of the maternal language spoken through the nose with velopharyngeal incompetence. Also, he believed that the later the reconstruction, the less the mobility of the sphincter. Moreover, he cited the ability of each patient to modify the learned speech abnormalities of changing their nasal mother tongue to an oral mother tongue as one of the important factors. Any of these factors could have been a contributing source for the few unsatisfactory results in this study. Jackson59 cautioned about the wrong position of palatal muscles in the primary repairs and suggested an initial correction followed by sphincter pharyngoplasty if they ever needed it. The fact that our study population was heterogeneous and included patients with their primary repairs conducted elsewhere, might have contained some patients with wrong position of palatal muscles and accounted for the few unsatisfactory results. Ideally the level of the sphincter should be placed where the velum is attempting contact with the posterior pharyngeal wall. However, some limitations are encountered in the presence of a large or low adenoid pad17. Abyholm et al8 reported a surgeon who declined the procedure because he did not think that he could adequately carry out the procedure in

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the presence of large adenoids. Some of the few unsatisfactory results in this study could be explained by the low level of placement of sphincter due to the presence of large adenoid pad. The only patient in this study that was reoperated upon for flap dehiscence occurred when attempting on insetting the sphincter into a large adenoid pad to avoid its inset at a lower position. One of the future implications that could be drawn from this work is to design a pilot study were adenoidectomy is carried out on a selective group of patients prior to sphincter pharyngoplasty in order to help in insetting the sphincter at its ideal level.

2.

Shprintzen RJ, Golding-Kushner KJ. Evaluation of velopharyngeal insufficiency. Otolarngol Clin North Am. 22:519,1989. Pamplona M, Ysunza A, Guerrero M, Mayer I, GarcaVelasco M. Surgical correction of velopharyngeal insufficiency with and without compensatory articulation. Int J Pediatr Otorhinolaryngol. 34:53,1996. Kuehn DP, Moller KT. Speech and language issues in the cleft palate population: The state of the art. Cleft Palate Craniofac J. 37:348,2000. Ysunza A, Pamplona M, Mendoza M, Molina F, Martinez P, Garca-Velasco M, Prada N. Surgical treatment of submucous cleft palate: a comparative trial of two modalities for palatal closure. Plast Reconstr Surg. 107:9,2001. Billmire DA. Surgical management of clefts and velopharyneal dysfunction. In Ann W. Kummer (ed): Cleft palate and craniofatial anomalies. Ohio. 401,2005. Bardach J, Salyer KE, Jackson IT. Pharyngoplasty. In Bardach J, Salyer KE (eds). Surgical techniques in cleft lip and palate. St. Louis: Mosby. 274,1991. Abyholm F, D'Antonio L, Davidson-Ward SL, Kjoll L, Saeed M, Shaw W, Sloan G, Whitby D, Worthington H, Wyatt R. VPI Surgical Group: Pharyngeal flap and sphincterplasty for velopharyngeal insufficiency have equal outcome at 1 year postoperatively: results of a randomized trial. Cleft Palate Craniofac J. 42:501,2005. Riski JE, Ruff GL, Georgiade GS, Barwick WJ, Edwards PD. Evaluation of sphincter pharyngoplasty. Cleft Palate Craniofac J. 29:254,1992.

3.

4.

5.

CONCLUSION
Sphincter pharyngoplasty is a physiological and anatomical substitution of the velopharyngeal valve. Not only does it reduce the velopharyngeal port by advancing the posterior pharyngeal wall and decreasing the lateral recess, but it also offers a dymanic sphincter in the majority of cases, and improves velar elevation. It relies on an axial pattern flap with fewer complications. When needed to be salvaged it can be easily revised. The modified sphincter pharyngoplasty that is presented herein eliminates the addition of pharyngeal flap as well as all raw surfaces and is sutured with overlap. These modifications are more anatomically and physiologically sound and take into account the evolution and advantages of the sphincter pharyngoplasty. It is very easy and very quick to perform with minor postoperative symptoms. The results demonstrate a satisfactory improvement of velopharyngeal function when applied in all patterns of velopharyngeal closure following primary repair of the palate despite a heterogeneous population of the study. Caution should be practiced in patients with extremely large defects, those accompanied by palatal fistulae, and those with improper position of palatal muscles following their primary repair. .
6.

7.

8.

9.

10. Kravath RE, Pollak CP, Borowiecki B, Weitzman ED. Obstructive sleep apnea and death associated with surgical correction of velopharyngeal incompetence. J Pediatr. 96:645,1980. 11. Orr WC, Levine NS, Buchanan RT: Effects of cleft palate repair and pharyngeal flap surgery on upper airway obstruction during sleep. Plast Reconstr Surg. 80:226,1987. 12. Ysunza A, Garcia-Velasco M, Garcia-Garcia M, Haro R, Valencia M: Obstructive sleep apnea secondary to surgery for velopharyngeal insufficiency. Cleft Palate Craniofac J. 30:387,1993. 13. Sirois M, Caouette LL, Spier S, Larocque G, Egerszegi EP: Sleep apnea following a pharyngeal flap: a feared complication. Plast Reconstr Surg. 93:943,1994. 14. Valnicek SM, Zuker RM, Halpern LM, Roy WL. Perioperative complications of superior pharyngeal flap surgery in children. Plast Reconstr Surg. 93:954,1994.

REFERENCES
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El Barbary & Ghandour 70. Boorman JG, Freedlander E. Surgical anatomy of the velum and pharynx. Recent Adv Plast Surg. 4:17,1992. 71. Witt PD, Marsh JL, Muntz HR, Marty-Grames L, Watchmaker GP. Acute obstructive sleep apnea as a complication of sphincter pharyngoplasty. Cleft Palate Craniofac J. 33:183,1996. 72. Kasten SJ, Buchman SR, Stevenson C, Berger M. A retrospective analysis of revision sphincter pharyngoplasty. Ann Plast Surg. 39:583,1997. 73. Witt PD, D'Antonio LL, Zimmerman GJ, Marsh JL. Sphincter pharyngoplasty: a preoperative and postoperativre analysis of perceptual speech characteristics and endoscopic studies of velopharyngeal function. Plast Reconstr Surg. 93:1154,1994.

Annals of Pediatric Surgery

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