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 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 produce optimal sphincter-like closure between the V excessive nasal resonance or hypernasality as oro- and nasopharynx.1 It occurs in a substantial the consequence of anatomical abnormalities and number of patients after cleft palate repair2-5 and can failure of the velum and the pharyngeal muscles to 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: [email protected]

El Barbary & Ghandour

result of the initial can shorten the following sphincter pharyngoplasty,27 a number of velum; making it impossible for the velum to reach patients have persistent unacceptable vocal resonance the posterior pharyngeal wall "target" during speech; and residual air escape postoperatively.21,28 Advances a deep nasopharynx relative to the position of the in patient selection and surgical technique to enhance velum; a poor velar movement despite an adequate successful valving of sphincter pharyngoplasty have length resulting from insufficient restoration of the been reported.8,28 palatal muscle sling at the time of primary repair.6 Velopharyngeal insufficiency results in the inability In an attempt to enhance the success of the sphincter of the cleft patients to communicate coherently and is palatoplasty and further improve the outcome, a considered the most disabling and devastating result modified sphincter pharyngoplasty is presented. It among the various secondary problems that may included the elevation of bilateral superiorly based follow cleft /palate repair.7 palatopharyngeus muscle with overlying mucosa that are sutured overlapped to each another and to a When surgical management is indicated for transverse incision on the posterior pharyngeal wall restoration of the velopharyngeal function, the at the level of attempted velopharyngeal closure. pharyngeal flap and the sphincter pharyngoplasty are among the most commonly used surgical The aim of this prospective study is to assess the procedures8. Considerable uncertainty of choice exists treatment outcome for patients with residual both within variations of flap and sphincter velopharyngeal insufficiency after palatal repair pharyngoplasty and between the two approaches. undergoing a modified technique of sphincter Authorities such as Riski9 agree that if surgical pharyngoplasty regardless of the pattern of intervention is needed, the procedure should be velopharyngeal closure. tailored to the size and nature of the velopharyngeal defect. However, reports of morbidity and mortality associated with pharyngeal flap surgery10-16 have led PATIENTS AND METHODS a lot of operators to adopt sphincterplasty instead. Several publications have advocated sphincter Patients diagnosed with residual velopharyngeal 17-19 pharyngoplasties citing their additional insufficiency after cleft palate repair presenting to the advantages as (1) technical ease of execution, (2) outpatient cleft palate clinic at Ain-Shams University superior speech results, (3) low complication rate, (4) Hospital from January of 2004 to December of 2006 reduced anaesthesia time, (5) non-obstruction of the had been considered potentially eligible for the study. nasal airway. Similar to other studies,8,29 the patients had to meet The sphincter pharyngoplasty operation is designed the following criteria to qualify for the study: (1) to form a ridge on the posterior pharyngeal wall, undergone a primary repair of the palate (with or narrow the from side to side, and to produce without a cleft lip or alveolus), (2) chronological age 20 a sphincteric type of closure. The objective of the between 4 and 16 years with apparent VPI diagnosed procedure is to create a muscular valve capable of by an experienced speech specialist, (3) had at least isolating the nasal cavity from the remainder of the 75% of normal language development for their age. vocal tract during appropriate speech tasks. This is Exclusion criteria comprised patients with: (1) size of necessary to eliminate hypernasality and to allow oral the velopharyngeal gap exceeding 2 cm in pressure to build in the oral cavity for the production anteroposterior dimension which necessitated a 21 of many consonant phonemes. Several lengthening procedure, (2) hearing impairment, (3) modifications of sphincter pharyngoplasty have been the cleft being part of a syndrome, (4) any preexisting 22-24 described since it was first introduced by Hynes palatal fistulae, (5) syndrome. who used superiorly based flaps from the salpingopharyngeus. Orticochea25 used The surgical procedure and study methods were palatopharyngeus instead and sutured them to an carefully explained to all parents. Patients underwent inferiorly based pharyngeal flap to below the palatal in-depth speech and endoscopic evaluations for plane. Jackson & Silverston26 replaced the inferiorly symptoms of velopharyngeal insufficiency. Six to based flap by a superiorly based posterior wall flap in twelve weeks, with a mean of two months, following an attempt to raise the flap insertion and improve the the surgical procedure they underwent the same outcome. Despite the reported high success rate thorough evaluation prior to receiving any speech

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therapy if they needed any. Evaluation was carried on a chair) a nasal loaded sentence /mama out using the protocol of assessment that is applied in betnajem mana:l/ and an oral sentence devoid of the phoniatric unit, Ain-Shams University which nasal sounds /ς ali rah jelς ab korah/ according to includes subjective as well as quasi-objective the protocol of assessment of VPI in the phoniatric measures of evaluation30.This protocol includes: unit, Ain-Shams University.30 The nasometer calculates the nasalance which is the ratio of the I- Preliminary Diagnostic Procedures: nasal to the nasal plus oral acoustic energy Auditory Perceptual Assessment (APA) was used multiplied by 100. The degree of hypernasality as a subjective tool for evaluation of patients' depends on the percent nasalance.31 Similar to language, speech and voice through listening to Abyholm et al,8 the nasalance results were reported every patient in a free conversation and a recorded as an overall measure of preoperative and speech sample. Passive and active aspects of postoperative mean changes as opposed to language were investigated including semantic, comparing each patient with the norm. syntactic and pragmatic aspects. Speech evaluation included the type and degree of open nasality, All patients included in the study received a consonant precision, the compensatory articulatory modified sphincter pharyngoplasty regardless of mechanisms (glottal articulation, pharyngealization the pattern of velopharyngeal closure or the of fricatives, and facial grimace), audible nasal air severity of the symptoms. The retractor and escape and overall unintelligibility of speech. All blade were adjusted to completely expose the these elements are graded along a 5-point scale in palatopharyngeus folds in the operating field. The which 0 = normal and 4 = severe affection. was retracted supoeriorly to expose the posterior pharyngeal wall as high as the adenoid. A II-Clinical Diagnostic Aids: submucous injection of 1:200,000 adrenaline was (A) Nasopharyngeal videofibroscopy: injected into the operative field. Vertical incisions were made in front of and behind the posterior All patients were examined using nasopharyngeal tonsillar pillar starting from the upper limit of the video-fibroscopy Henke-Sass-Wolf, type 10, tonsillar recess. With a Metzenbaum scissor, the connected to a Lemke video camera (MC 204) and palatopharyngeous muscle was elevated with its Panasonic video cassette recorder 357. The overlying mucosa. After obtaining as much vertical nasofibroscope was introduced through the nasal length as possible, the superiorly based cavity to a position superior to the soft palate. The myomucosal flap was divided inferiorly and velopharyngeal valve movement was recorded elevated with a right-angled scissor. The donor site while the patient repeated the speech samples was closed. The same steps were repeated to applied in the protocol of assessment of VPI in the elevate the contralateral flap. A transverse incision 30 phoniatric unit, Ain-Shams University. down to the prevertebral fascia is placed Movements of the velum, lateral, and posterior approximately 2 mm below the adenoidal pad to pharyngeal walls were traced on the monitor. The which the flaps are rotated 90 degrees. The flaps movement of each component was given a score were interdigitated and sutured together tip-to- from 0 to 4 as follow: 0 = the resting () base and to the posterior pharyngeal wall without position or no movement, 2 = half the distance to leaving any lateral ports. During recovery from the corresponding wall, 4 = the maximum general anaesthesia, the patients were placed in a movement reaching and touching the opposite compulsory posture with the head turned to one wall. Also,the pattern of closure of the side. They were encouraged to start liquid diet on velopharyngeal port, whether coronal, sagittal, the first day of operation as soon as they were fully circular or others were specified and recorded. recovered. Any perioperative complications in recovery or any later complications including (B) Nasometry: airway complications, readmission to hospital and Nasometery was performed to all patients using reoperation were recorded. Kay nasometer model 6200-2 with a software version 1.5. It is composed of a head set, Statistical analysis was done using paired t-test microprocessor and a printed circuit board. Every with P>0.05 indicating no significance. P<0.05 patient was asked to repeat (with a normal indicated significance, P<0.01 indicated high conversational loudness, while sitting comfortably

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significance, and P<0.001 indicated very high hyponasal postoperatively. The compensatory glottal significance. 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% RESULTS (Fig. 2). pharyngealization of fricatives remained unchanged in 25.5% of patients and improved in Forty three patients whom fulfilled the criteria were 74.5% of patients. Improvement of one scale point included in the study. They were twenty six males was seen for 37% of patients, and improvement of and seventeen females. Twenty seven patients had more than one scale point was observed for the cleft lip and palate with four being bilateral. The remaining 37.5% (Fig. 3). Facial grimace was not remaining sixteen patients had isolated cleft palate. present in 15 patients preoperatively. From those The mean age of the patients at the time of the presenting with facial grimace on evaluation operation was seven years and five months. preoperatively 60.5% did not show it post Intraoperatively, blood loss was minimal and the operatively. Audible nasal escape could not be mean length of surgery was 45 minutes. All patients verified in two patients preoperatively, and became experienced a smooth postoperative recovery and absent in 78% of those that had before surgery. were discharged from the hospital on the second Ratings of speech overall intelligibility (Fig. 4) postoperative day. There were no severe improved significantly in 86% of patients; one scale complications, such as obstructive dyspnea or point improvement in 30% of patients and 56% for bleeding. Almost all patients complained of two or more scale point improvement. Speech during sleep in the first couple of weeks. The intelligibility was judged as showing deterioration in postoperative snoring disappeared in an average of 4.5% of patients and no change in 7% of patients. 2-3 months following the operation. None of the A significant change was found in nasalance scores patients was reoperated upon for airway obstruction. between the mean preoperative and mean Only in one patient, flap dehiscence occurred and postoperative scores. With the normal value being was revised. 10.5 for oral sentence, a mean score of 44.3 Six speech variables were rated pre and preoperatively was calculated compared to a mean postoperatively. The degree of open nasality was score of 21.1 postoperatively with 23.2 difference. As reduced in 90.5% of patients. Of these patients 44% for the nasal sentence, a mean score of 64 improved only one scale point, 37% improved two preoperatively was recorded compared to a mean scale points, while the remaining 9.5% improved score of 51.5 postoperatively, with the normal value three scale points (Fig. 1). No patients were rated as being 48.5.

A B 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.

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A B 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 B 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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A B 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 the majority of patients and was categorized as nasopharyngeal videofibroscopy was categorized circular in thirty patients and coronal in 13. Out of the preoperatively as coronal in 28 patients, sagittal in 8, 8 sagittal closures preoperatively, 6 became coronal and circular in 7 patients. Postoperatively, while the remaining two became circular. The grade velopharyngeal port achieved functional closure in of movement in attempting closure improved in all

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except for one of the two that became circular with In 1968 Orticochea25 described the construction of a movement judged as remaining similar to the "dynamic" pharyngeal muscle sphincter in cleft palate preoperative grade of movement. All preoperative patients by suturing the tips of the lateral flaps, circular closures remained as such except for one that containing palatopharyngeus, onto the superior end became coronal. It was only in this one that the of a third low inferiorly based posterior pharyngeal degree of movement remained as preoperative, while flap. The flaps were not sutured to the posterior improving in the rest. The majority of coronal pharyngeal wall laterally. A modification of the latter closures (n=22) became circular and only 6 remained that has gained so much popularity was introduced as coronal in the postoperative evaluation. Two of the by Jackson & Silverton in 1977.26 They felt that the 22 that became circular were judged as deteriorating term "sphincter" was more appropriate for this type of in the degree of movement as opposed to an functional pharyngoplasy. A superiorly based midline improvement in the remaining 20. All of the 6 that flap, raised high on the posterior pharyngeal wall, remained coronal showed an improvement in the substituted for the low inferiorly based flap of degree of movement except for one that remained Orticochea. Further modifications of the technique with the same grade as preoperatively. The have been made. Most have centered on obtaining a nasopharyngeal sphincter showed remarkable superior placement and covering raw tissue areas.34 contraction during speech, but not to the extent of Ren & Wang35 Sutured half of the wounds on the closing the velopharyngeal port completely in all palatopharyngeus flaps to form a tubed pedicled flap patients (Fig. 5) 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. DISCUSSION The modification in the technique presented takes into The sphincter pharyngoplasty described here is a account the evolution and advantages of each of the variation of older concepts. Even though sphincter procedures and simplifies them. It avoided reconstruction of the velopharyngeal sphincter some of the intrinsic deficiencies that were present anatomically was attempted in 1935 by Browne32 through eliminating all raw areas of the earlier through placing a constricting suture around the procedures and raising the level of inset by entire oronasal port at the level of Passavant's ridge, discontinuing the use of the pharyngeal flap that Hynes22 was, undoubtedly, the first to introduce the complemented earlier modifications of all sphincter "lateral" pharyngoplasty as a method of treatment of procedure. The palatopharyngeal flaps were sutured velopharyngeal insufficiency in 1950. He raised flaps overlapped tip-to-base similar to the original from the lateral pharyngeal walls then closed the description by Hynes to further narrow the donor defects to narrow the pharynx. Medial velopharyngeal valve in a static manner. In agreement interpolation and crosslapping of the two flaps with Sie et al17 and Witt et al36, the width, length, and produced a horizontal shelf above Passavant's ridge level of insertion of the palatopharyngeal flaps, as to bring the posterior pharyngeal target closer to the well as the degree of overlap of the transposed flaps, velum. He believed that his technique would be more can be modified to suit the requirements of any functional since the nerve supply to the lateral individual patient. The degree of tightness and pharyngeal muscles comes from a superior origin. closure of the sphincter is therefore determined by all Any flap lifted laterally would contain neuromuscular these factors. elements and would contract. In his first paper, Reid37 and Abyholm38 suggested that large fistulas Hynes23 described the flaps containing the might be detrimental to speech. Cosman & Falk39 salpingopharyngeus muscles but in his Hunterian reported on general speech effects associated with lecture in 1953 he made it clear that he also raised the palatal fistulas. Isberg & Henningsson40 studied the palatopharyngeus and the fibers of the underlying influence of palatal fistulas on velopharyngeal superior constrictor. Further observations on this type movements and found a statistically significant of operation were made by Hynes24 in 1967 and the correlation between the fistula size and the degree of results were later reviewed by Pigott.20 Moore33 also lateral wall movement but not with the velar raised the salpigopharyngeus muscle but rather used movement. Furthermore, they demonstrated an it to augment the posterior margin of the soft palate. improvement in velopharyngeal movements when

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

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El Barbary & Ghandour reduce the lateral recess thus narrowing the have good mobility (coronal pattern of closure), and circumference with resultant decrease in therefore one shrinks from placing anything in the velopharyngeal port area.20 soft palate that might in any way interfere with this much valued movement. When the palate is not The size and shape of the residual velopharyngeal gap functioning properly (sagittal pattern of closure), an have been determinant factors in implementing the initial reconstruction of the soft palate levator different surgical modalities for the correction of mechanism should be carried out. If a degree of velopharyngeal insufficiency.52 Pharyngeal flaps have velopharyngeal insufficiency is still present, then been recommended when the residual patients can be very well rehabilitated with sphincter velopharyngeal gap is sagittal indicating a strong pharyngoplasty. Although this approach involves two lateral wall movement and weak palatal mobility, operations, it is certainly more anatomically and while sphincter pharyngoplasty is indicated when the physiologically sounds than opting immediately for a residual velopharyngeal gap is coronal indicating pharyngeal flap. poor lateral wall movement and a strong velar movement53-56. Armour and colleagues19 further It seems logical that when the soft palate is emphasized on this and confirmed that pharyngeal functioning adequately but is short and there is no flaps are less effective in treating velopharyngeal viable way to make the palate longer, then the answer insufficiency in patients with coronal closure. is to maintain the good palatal function and reduce Consequently, they changed the historic pattern of the dimensions of the velopharyngeal mechanism by treatment at The Hospital for Sick Children in Toronto performing a sphincter pharyngoplasty. This becomes that implied the pharyngeal flap for all even more logic if we add to it the further velopharyngeal insufficiencies regardless of their improvement in the palatal elevation that has been closure patterns; and became more inclined to treat considered to be one of the additional advantages of coronal pattern velopharyngeal insufficiencies with the sphincter pharyngoplasty.26,60 Georgantopoulou sphincter pharyngoplasty. Similarly, de Serres et al18 and coworkers61 studied in detail the effect on velar examined their experience with the sphincter mobility and demonstrated a significant increase in pharyngoplasty and pharyngeal flap procedures after the range of movement of the soft palate following their results seemed to indicate that less than optimal different types of sphincter pharyngoplasty. They results were being obtained with the pharyngeal flap explained their findings based on the fact that procedure. They found Sphincter pharyngoplasty to elevating the superiorly based posterior tonsillar have a higher success rate and tended to recommend pillar flaps divided the palatopharyngeous muscle. it more liberally while abandoning the pharyngeal This in turn liberated the levator palati from its flap procedure at their institution, although they still antagonist, the palatopharyngeous, to act consider it as a management option depending on unopposed62 resulting in an increased velar elevation. operator preference. It is well documented1,57,58 that Although, their explanation might have been a coronal closure is the commonest pattern of closure simplistic view to a very complex interaction between accounting for 55% while sagittal closure accounts for the muscles of the soft palate63, it still holds logic and only 10-15%. This was comparable with the findings validates their findings. Some of the closure patterns in this study; coronal closure represented 65% of the in this study changed into coronal postoperatively, cases while sagittal 18.5%. indicating a stronger velar mobility. Out of 8 sagittal closures preoperatively, 6 became coronal which Therefore, having the coronal closure as the concurs Georgantopoulou's et al61 explanation. commonest pattern together with the hazard of potential impairment of the lateral wall movement The concept of dynamic pharyngoplasty arose from when elevating a pharyngeal flap,49 offers the Orticochea's25,64 observations of the process of sphincter pharyngoplasty, which interferes less with deglutition. He speculated that the posterior tonsillar the pharyngeal wall anatomy and is indicated in the pillars with enclosed palatopharyngeus muscle which coronal pattern of closure, a better chance of outcome acts during the gag reflex could be engaged for speech in all circumstances. Jackson59 once said that "as soon production. Hence, sphincter pharyngoplasty was as surgeons become experienced and comfortable designed to change the lower insertion of the with sphincter pharyngoplasty, they will find it the posterior pillars from the lateral walls to the posterior most common rehabilitative measure employed". This wall on the pharynx. Witt et al65 tested this theoretical should be because properly repaired should advantage of dynamic activity of the

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El Barbary & Ghandour neovelopharyngeal port. They evaluated 58 patients results could be rather explained by factors other than who underwent sphincter pharyngoplasty by speech the pattern of closure. videofluroscopicy. Their results indicated a Huang et al68 examined the blood supply of the entire quantifiable and statistically significant excursion of velopharyngeal complex. They described the sphincteric closure concluding that sphincter pharyngeal flap, from a vascular standpoint and in pharyngoplasty works in a dynamic and active agreement with Mercer & MacCarthy69 as being manner. In another study, they66 found little evidence random in nature. The lateral incisions for superiorly to suggest that preexisting posterior pharyngeal wall or inferiorly based flaps invariably divide the motion caused the sphincteric movement. To the perforators from the ascending pharyngeal artery that contrary, Ysunza et al42 and Ysunza67 suggested that supplies the superior constrictor as these vessels run neither the lateral pharyngeal flaps in cases of transversely. Although this certainly does not sphincter pharyngoplasties nor the central pharyngeal preclude flap viability, it could be a contributing flap in cases of pharyngeal flaps created new factor to the common sequel of unpredictable flap sphincters for velopharyngeal closure by using shrinkage. On the other hand, the pattern of blood selective electromyography and simultaneous supply to the faucial portion of the palatopharyngeus videonasopharyngoscopy. The participation of these was observed to be segmental, with a number of structures is passive, increasing tissue volume in branches of the ascending palatine artery entering the specific areas, whereas their movements are caused by muscle throughout the length of the tonsilar pillar. the contraction of the superior constrictor pharyngeus Therefore, even if the flaps were raised up to or and the levator veli palatini. It is interesting to sight beyond the superior pole of the tonsil, the base of each that they were cautious about their observation and flap would probably contain at least the hamular stated that the small number of their study does not branch of the ascending palatine artery, ensuring an allow definite conclusions. In all circumstances, the adequate axial blood supply to the flap. This concured sphincter pharyngoplasty will work as described by with the opinions of Boorman & Freedlander70 and Pigott20 in any of three ways: as an active sphincter, or probably explains why flap necrosis in this procedure at least by advancing the posterior wall and by is a rare phenomenon. An intraoperative finding of reducing the lateral recess in a static manner. The flap retraction following its elevation was observed in nasopharyngoscopic findings of this study concurred all cases of this study indicating the preservation of its with Witt et al65,66 results and demonstrated a neurovascular supply. dynamic neosphincter in all cases. However, the origin of this activity was not tested for since no Although healing around the orifice of the electomyographic study was conducted. The majority velopharyngeal sphincter is not totally controllable of the cases demonstrated a circular pattern of closure following sphincter pharyngoplasty, it is still much indicating participation of velum and pharyngeal wall more controllable than healing around the lateral in the movement. While in the remaining cases, the ports of the pharyngeal flap. Jackson59 emphasizes velar movement was more significant than the that only one posterior vertical suture line is pharyngeal wall and was accounted in as coronal incorporated in the recreated sphincter and therefore pattern of closure. It is interesting to observe from the contracture of the sphincter due to scarring is unlikely results of this study that the five patients whom to occur. In the modification presented here, even this overall speech intelligibility were judged as posterior scar has been eliminated. The fact that the unsatisfactory following surgery were distributed velopharyngeal aperture does not retract by scarring, over the different patterns of closure. In three of the in this modification, because of the lack of raw areas patients whom had the same preoperative and and circumferential incisions is significant. postoperative overall speech intelligibility, one was a There have been several reports of disastrous total sagittal that became circular, the second was circular closure of the velopharyngeal area, airway and became coronal, while the last was the coronal obstruction and death associated with posterior that remained as such postoperatively. Speech pharyngeal flap surgery.10-16 Valnicek et al14 reviewed intelligibility was reviewed as showing deterioration a 7-year experience with superiorly based pharyngeal only in two patients out of the 22 patients that flap in a total of 219 children at The Hospital for Sick changed from coronal preoperatively into circular Children in Toronto. Complications included 18 postoperatively. Therefore, these few unsatisfactory children (8.2%) with bleeding, of whom 5 required transfusion; 20 children (9.1%) with airway

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El Barbary & Ghandour obstruction; and 9 (4.1%) with sleep apnea after It should be emphasized that the results in this study discharge from the hospital. Three patients required were evaluated early between 6 and 12 weeks reintubation in the early postoperative period, and 11 postoperatively and prior to establishment of speech required eventual surgical revision, including therapy to assess the role of the surgical procedure complete takedown of the flap in four patients. To the alone. Abyholm et al8 demonstrated that the sphincter contrary, Jackson59 stated that he has never seen a case pharyngoplasty achieved correction more slowly and of sleep apnea with sphincter pharyngoplasty and that one year follow ups provided better outcomes attribute this to the fact that scarring is minimal and than at three months. The original design of this study occurs in a vertical rather than a horizontal direction. was to carry a second follow up evaluation at one Obstructive sleep apnea seems to be more frequent, if year. However, because of the non compliance of the not almost exclusively, associated with posterior majority of patients, only a small sample size were pharyngeal flap surgery.62 The literature does not followed up after one year and no conclusive contain as many reports of airway problems with outcomes could be withdrawn in this regard. sphincter pharyngoplasty as with pharyngeal flap. Nevertheless, the results of this study indicates that a Extraordinarily, Witt et al71 demonstrated that airway modified sphincter pharyngoplasty improved speech dysfunction can occur following sphincter overall intelligibility significantly in 86%. This is pharyngoplasty. They reported perioperative and/or comparable with the 74% success rate of Ren & postoperative airway dysfunction in a minority of Wang,35 79% improvement of Witt et al,73 85% success their patients. However, these patients had either rate of Abyholm et al8, 91% of Jackson & Silverton26. Pierre Robin sequence, micrognathia, or histories of Orticochea48 reported his observations accumulated perinatal respiratory and/or feeding problems. In all over 40 years in treating velopharyngeal insufficiency. of them, airway dysfunction resolved within 3 days He recognized a variety of factors that influenced the postoperatively without the need for a surgical success rate of dynamic sphincter pharyngoplasty takedown of the sphincter pharyngoplasty to relieve outcomes. He considered the age of the patient as one the airway problems. In this study none of the of them; the older the patient, the more deeply patients suffered airway dysfunction nor sleep apnea; engraved will be the cerebral engram patterns of the only snoring at night that persisted for three months maternal language spoken through the nose with in some cases. velopharyngeal incompetence. Also, he believed that Finally, a very important issue is the ability to salvage the later the reconstruction, the less the mobility of the failures with further surgery. Jackson59 stated that sphincter. Moreover, he cited the ability of each sphincter pharyngoplasty can be used to rehabilitate patient to modify the learned speech abnormalities of the patient who has failed a pharyngeal flap. changing their nasal mother tongue to an oral mother Moreover, in patients requiring Le Fort I or Le Fort III tongue as one of the important factors. Any of these advancements, the sphincter has never caused any factors could have been a contributing source for the obstruction to advancement. This problem occurred few unsatisfactory results in this study. Jackson59 on several occasions with pharyngeal flaps, which, cautioned about the wrong position of palatal muscles when tight and scarred, have required division. Even in the primary repairs and suggested an initial then, rehabilitation has been performed using correction followed by sphincter pharyngoplasty if sphincter pharyngoplasty six months after the they ever needed it. The fact that our study maxillary advancement. Advocates of sphincter population was heterogeneous and included patients pharyngoplasty have listed the ability to easily and with their primary repairs conducted elsewhere, successfully revise port size as one of its advantages might have contained some patients with wrong over pharyngeal flap62. Revision surgeries were position of palatal muscles and accounted for the few described as being without difficulty72. The patients unsatisfactory results. whom required revisions because of persistent Ideally the level of the sphincter should be placed hypernasality were associated with flap dehiscence, where the velum is attempting contact with the low-lying flaps, and end-to-end (as opposed to end- posterior pharyngeal wall. However, some limitations to-tip) flap suturing72. In this study only one patient are encountered in the presence of a large or low required revision because of flap dehiscence. This was adenoid pad17. Abyholm et al8 reported a surgeon one of the patients that were operated upon early in who declined the procedure because he did not think this series and the flaps were set high into the adenoid that he could adequately carry out the procedure in tissues were the sutures did not hold.

Annals of Pediatric Surgery 32

El Barbary & Ghandour the presence of large adenoids. Some of the few 2. Shprintzen RJ, Golding-Kushner KJ. Evaluation of unsatisfactory results in this study could be explained velopharyngeal insufficiency. Otolarngol Clin North by the low level of placement of sphincter due to the Am. 22:519,1989. presence of large adenoid pad. The only patient in this 3. Pamplona M, Ysunza A, Guerrero M, Mayer I, García- study that was reoperated upon for flap dehiscence Velasco M. Surgical correction of velopharyngeal occurred when attempting on insetting the sphincter insufficiency with and without compensatory into a large adenoid pad to avoid its inset at a lower articulation. Int J Pediatr Otorhinolaryngol. 34:53,1996. position. One of the future implications that could be 4. Kuehn DP, Moller KT. Speech and language issues in drawn from this work is to design a pilot study were the cleft palate population: The state of the art. Cleft is carried out on a selective group of Palate Craniofac J. 37:348,2000. patients prior to sphincter pharyngoplasty in order to help in insetting the sphincter at its ideal level. 5. Ysunza A, Pamplona M, Mendoza M, Molina F, Martinez P, García-Velasco M, Prada N. Surgical treatment of submucous cleft palate: a comparative trial of two modalities for palatal closure. Plast Reconstr CONCLUSION Surg. 107:9,2001.

Sphincter pharyngoplasty is a physiological and 6. Billmire DA. Surgical management of clefts and anatomical substitution of the velopharyngeal valve. velopharyneal dysfunction. In Ann W. Kummer (ed): Not only does it reduce the velopharyngeal port by Cleft palate and craniofatial anomalies. Ohio. 401,2005. advancing the posterior pharyngeal wall and 7. Bardach J, Salyer KE, Jackson IT. Pharyngoplasty. In decreasing the lateral recess, but it also offers a Bardach J, Salyer KE (eds). Surgical techniques in cleft dymanic sphincter in the majority of cases, and lip and palate. St. Louis: Mosby. 274,1991. improves velar elevation. It relies on an axial pattern flap with fewer complications. When needed to be 8. Abyholm F, D'Antonio L, Davidson-Ward SL, Kjoll L, salvaged it can be easily revised. The modified Saeed M, Shaw W, Sloan G, Whitby D, Worthington H, sphincter pharyngoplasty that is presented herein Wyatt R. VPI Surgical Group: Pharyngeal flap and sphincterplasty for velopharyngeal insufficiency have eliminates the addition of pharyngeal flap as well as equal outcome at 1 year postoperatively: results of a all raw surfaces and is sutured with overlap. These randomized trial. Cleft Palate Craniofac J. 42:501,2005. modifications are more anatomically and physiologically sound and take into account the 9. Riski JE, Ruff GL, Georgiade GS, Barwick WJ, Edwards evolution and advantages of the sphincter PD. Evaluation of sphincter pharyngoplasty. Cleft pharyngoplasty. It is very easy and very quick to Palate Craniofac J. 29:254,1992. perform with minor postoperative symptoms. The 10. Kravath RE, Pollak CP, Borowiecki B, Weitzman ED. results demonstrate a satisfactory improvement of Obstructive sleep apnea and death associated with velopharyngeal function when applied in all patterns surgical correction of velopharyngeal incompetence. J of velopharyngeal closure following primary repair of Pediatr. 96:645,1980. the palate despite a heterogeneous population of the 11. Orr WC, Levine NS, Buchanan RT: Effects of cleft palate study. Caution should be practiced in patients with repair and pharyngeal flap surgery on upper airway extremely large defects, those accompanied by palatal obstruction during sleep. Plast Reconstr Surg. fistulae, and those with improper position of palatal 80:226,1987. muscles following their primary repair. 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.

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70. Boorman JG, Freedlander E. Surgical anatomy of the 73. Witt PD, D'Antonio LL, Zimmerman GJ, Marsh JL. velum and pharynx. Recent Adv Plast Surg. 4:17,1992. Sphincter pharyngoplasty: a preoperative and postoperativre analysis of perceptual speech 71. Witt PD, Marsh JL, Muntz HR, Marty-Grames L, characteristics and endoscopic studies of Watchmaker GP. Acute obstructive sleep apnea as a velopharyngeal function. Plast Reconstr Surg. complication of sphincter pharyngoplasty. Cleft Palate 93:1154,1994. 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.

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