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Cerclage sphincter pharyngoplasty: A new technique for velopharyngeal insufficiency
International Journal of Pediatric Otorhinolaryngology (2007) 71, 793—800
www.elsevier.com/locate/ijporl
Cerclage sphincter pharyngoplasty: A new technique for velopharyngeal insufficiency Ahmed Ragab * ORL department, Menoufiya University Hospital, Egypt Received 12 November 2006; received in revised form 24 January 2007; accepted 30 January 2007
KEYWORDS Velopharyngeal insufficiency; Velopharyngeal impairment; Velopharyngeal dysfunction; Pharyngoplasty
Summary Objectives: Several surgical techniques are available for the treatment of velopharyngeal insufficiency (VFI). Each method has its own complications and non-dynamic roles. So the aim of this study was to present a novel physiological surgical technique designed by the author for reconstruction of the velopharyngeal sphincter in VFI. Methods: This prospective study included six patients with VFI (two males and four females) with ages from 5 to 20 years (mean: 12.50 years). Speeches, nasopharyngeal and oral endoscopies for velopharyngeal valve closure were measured according to a 5-point scale where 0 was equivalent to normal and 4 meant a severe (constant) deviation. They were scheduled for cerclage sphincter pharyngoplasty after failure of appropriate speech therapy. Under general anaesthesia and the patient in semiflower’s position; two level cerclages (1-0 polypropylene suture materials) were inserted behind the muscles of the velopharynx. The first at the level of junction of posterior and middle one-thirds of the soft palate passing through soft palate, left lateral pharyngeal wall, posterior pharyngeal wall, right lateral pharyngeal wall and the soft palate. The second was at 3 mm in front of the latter. The surgical technique was described in details. Results: Before surgery five patients (83.3%) had sever hypernasality (rating scale 3). After the cerclage operation and speech therapy four patients (66.6%) significantly improved to normal nasality (rating scale 0) and the remaining two patients improved to mild and moderate hypernasality (rating scale 1 and 2), respectively ( p < 0.05). By endoscopy the closing activity was (rating scale 3) in five patients (83.3%) and (rating scale 2) in one patient (18%). After the cerclage operation and speech therapy five patients (83.3%) changed significantly to complete closure (rating scale 0) and to (rating scale 1) in one patient ( p < 0.05).
* Correspondence address: Lecturer and consultant of ORL Head & Neck Surgery, Menoufiyia university Hospital, Shibin Elkom, 73, Sayed St., Tanta, Egypt. Tel.: +20 40 3420114/20 0 101709898; fax: +20 40 3315000. E-mail address: [email protected]. 0165-5876/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2007.01.020
794
A. Ragab Conclusions: Cerclage sphincter pharyngoplasty is a new procedure designed by the author in VFI. It helps the velopharynx to function physiologically in three-dimensional patterns without dependency on the type of closure. Also it is an easy technique; without tissue flaps transfer, upper airway obstruction or hyponasality. # 2007 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Several components create velopharyngeal (VP) port closure, namely the velum, the lateral pharyngeal wall (LPW) and the posterior pharyngeal wall (PPW), which together forms a three-dimensional muscular valve known as the velopharyngeal sphincter [1]. The pattern of velopharyngeal closure depends on the degree to which each of these components of the sphincter is active during closure [2]. More than four closure patterns were described [3]. Closure patterns vary among individuals, but an individual will tend to have a consistent closure pattern across varied consonant utterances [2]. Complete velopharyngeal closure is required during swallowing and production of all consonants except for the nasal consonants [4]. Velopharyngeal insufficiency (VFI) refers to the inability of the velopharyngeal sphincter to close completely during production of the oral (non-nasal) sounds of speech [5]. The primary effects of VFI are nasal air escape and hypernasality. Secondary effects of VFI include speech articulation errors (distortions, substitutions and omissions). These effects combine to reduce intelligibility of speech [4]. Although speech articulation errors may be corrected through speech therapy, the primary effects of VFI — nasal air escape and hypernasality — can only be corrected by restoring velopharyngeal competence [6]. Several surgical techniques are available for the treatment of VFI, e.g. posterior pharyngeal wall augmentation, posterior pharyngeal flaps and sphincter pharyngoplasty [7]. The common goal of these surgical techniques is to create a permanent partial obstruction of the velopharyngeal space [7]. Every method has its own complications and nondynamic roles. Posterior pharyngeal wall augmentation is appropriate in the presence of a persistent gap in the central velopharyngeal port measuring at most 1— 3 mm but it is not suitable for larger defects [8]. Also a wide variety of implantable materials has been used to augment the posterior wall. Problems with extrusion, migration, resorption and infection have been reported [9]. Avoiding implantable materials in the design of operative techniques for VFI can prevent such complications.
Pharyngeal flap is the mainstay of surgical therapy for velopharyngeal insufficiency in many facilities. In this procedure, a flap of tissue is pedicled superiorly or inferiorly to the post-pharyngeal wall and sutured to the palate. Most reports describe good speech after pharyngeal flap surgery [10]. However, there are also studies that report unpleasant side effects such as nasal obstruction, hyponasality, sleep apnea and lateral port stenosis because the flap obstructs the air passage [11]. Another draw back is its limitation to obdurate mainly a midline VF defects and mobile lateral pharyngeal walls are needed for ports to be open during breathing and closing during speech [10]. Also it limits mobility of the soft palate. A technique that avoids altering and dividing the VF into two lateral ports susceptible to stenosis and obstruction can rescue the airway. Sphincter pharyngoplasty also called lateral pharyngoplasty is another common surgical treatment for VFI. The tonsillar pillars are used as donor flaps to be sutured across an incision in the nasopharyngeal posterior wall [7]. These procedures create a transverse sphincter of variable diameter with preservation of mobility of the soft palate. This approach is advantageous when coronal or circular closure is present and lateral pharyngeal wall motion is deficient. Several studies have reported a success rate (i.e. correction or significant reduction in hypernasality) from 78 to 90% with pharyngoplasty [10]. The incidence of post-operative hyponasality is estimated to be 12—17% [12]. More superiorly raised palatopharyngeal flaps lead to a greater degree of velopharyngeal obturation. If this is excessive, an obstructive pattern of breathing can develop. Some patients still develop some mild temporary obstructive sleep patterns in the immediate post-operative period [12]. Using a novel dynamic approach suite in any type of VF defects (different sizes and shapes) without such complications of upper airway obstruction during day or sleep and without hyponasality is advantageous. Lateral pharyngeal flaps in cases of sphincter pharyngoplasties and the central pharyngeal flap in cases of pharyngeal flaps; do not create new sphincters for velopharyngeal closure. The participation of these structures is passive (absent electromyographic activity in the flaps in both techniques) [13], increasing tissue volume in speci-
Cerclage sphincter pharyngoplasty fic areas, whereas their movements are caused by the contraction of the superior constrictor pharyngeus and the levator veli palatini [13]. A new procedure with active participation of all surrounding muscle without muscle transposition will add a valuable technique in surgical treatment of VFI. So the aim of this work is to present a novel physiological surgical technique designed by the author for reconstruction of the velopharyngeal sphincter in VFI.
2. Patient and methods 2.1. Patients Six patients with VFI were enrolled in this prospective study. They were scheduled for surgical treatment of VFI after appropriate speech therapy conducted by a trained orthophonist. Patients received speech therapy, including phonologic approach and whole language intervention. The study population included two males and four females with their ages from 6 to 20 years (mean: 13 years). The eligibility criteria for the study included: velopharyngeal insufficiency with or without compensatory articulation disorder as demonstrated by phoniatric assessment, nasopharyngoscopy, oral endoscopy and lateral radiography; normal hearing as demonstrated by conventional pure-tone audiometry and Speech Discrimination Score and language development within reference limits. The study approved by the Ethics Committee of the hospital (Menoufiya University Hospital, Egypt). The parents and legal guardians of all selected patients agreed to participate in the study with an informed consent. All patients received speech therapy at least 6 months before the operation and 3 months after the operation.
2.2. Analyses 2.2.1. Speech Speech was assessed through perceptual ratings of eight speech variables by speech/language pathologist (SLP) trained in assessing speech in patients with VFI and who was blinded to whether the test stimuli were obtained pre- or post-surgery. These variables included hypernasality, hyponasality, weak pressure consonants, glottal stops, pharyngeal fricatives, nasal fricatives, velar for dental plosives and palatal for dental plosives. Each variable was rated on a 5-point scale from 0 to 4, where 0 was equivalent to normal articulation or resonance and 4
795 Table 1 The speech variables and rating scale used for the assessment of speech Hypernasality, hyponasality and weak pressure consonants 0_/Normal resonance/adequate pressure 1_/Mildly hyper/hyponasal/weak (reduced pressure) 2_/Moderately hyper/hyponasal/weak (reduced pressure) 3_/Severely hyper/hyponasal/weak (reduced pressure) 4_/Very severely hyper/hyponasal/weak (reduced pressure) Glottal stop articulation of plosives, pharyngeal fricative articulation of fricatives and retracted oral (to palatal/velar) articulation of dental stop consonants 0_/No misarticulation 1_/Occurs once or twice 2_/Occurs often 3_/Occurs very often but not all the time 4_/Occurs all the time
meant a severe (constant) deviation (Table 1). Speech samples include vowels /a/ and /i/. It includes [ma ma ma], [pI pI pI], [tI tI tI], [kI kI kI], [ka ka ka]. Also to repeat or read aloud standardized passages in Arabic language that is totally free of nasal consonants and passages loaded with nasal consonants. These were evaluated in a quiet room by the same SLP on two different occasions within a 2-week period on recorded voice. Each recording was given a number that could be heard on the tape. 2.2.2. Nasopharyngeal and oral endoscopy for velopharyngeal valve closure The visual perceptual judgments were made by the same SLPs who evaluated the speech and the Otorhinolaryngology surgeon (judges 1 and 2). In this analysis, the judges independently estimated the closing pattern activity according to a fivepoint scale, from zero to four, where zero represented a complete closure in the midline and four was equivalent to little or no activity (Table 2). Rating obtained by consensus through discussion amongst judges. Judgments were each made on three different occasions. In addition to the closing pattern also the movements of the lateral pharyngeal walls, posterior walls and velum were assessed. When judging asymmetry, however, both lateral pharyngeal walls were observed together. Analysis was obtained before speech therapy, 2 weeks before the operation, 1 month after the operation (3 and 6 months) after speech therapy and at the same week of recording the study results (Table 3).
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Table 2 The rating scales for the assessment of velopharyngeal valve closure before and after cercelage sphicter surgery (a) Closing activity 0_/100% (complete) 1_/75% 2_/50% 3_/25% 4_/None or hardly any (b) Asymmetry Yes/No Right/Left (c) Shape: coronal, sagittal, circular, others The activity in each side was assessed separately. (a) Closing activity and (b) asymmetry (which was assessed by observing both sides together).
2.3. Statistics The data was collected, tabulated and analyzed by SPSS statistical package Version II on IBM compatible computer. Qualitative data expressed as number and corresponding percentages were analyzed by applying Chi-Square test to compare preoperative, post-operative and post-speech variables. Spearman rank correlation test was used to detect the association between quantitative variables. All tests were used as tests of significance at 0.05 levels.
3. Operation After induction of general orotracheal anaethesthia, the patient was placed in semiflower’s position with shoulder roll in place. The patient was prepared and draped in the normal manner. A self-retaining mouth retractor was placed in the mouth for exposure. Lidocaine epinephrine 1/ 100,000 was administered in the proposed sites of suture cercelage insertion. A catheter was inserted through the nose to aid in palatal elevation during LPW and PPW sutures or using a retractor to elevate the soft palate. Using methelyne blue allowed marking of the proposed posterior pharyngeal wall closure site. Two levels cerclage: the first at the level of junction of posterior and middle one-thirds of the soft palate passing through soft palate left LPW, PPW, right LPW and the soft palate. The second was at 3 mm in front of the first level (Fig. 1). A special needle designed for this purpose was breaded with 1-0 polypropylene suture material. Starting from the right side of the soft palate the needle was inserted at the junction of the soft
Fig. 1 Cerclage sphincter pharyngoplasty technique: (A) starting from the right side of the soft palate a special needle breaded with 1-0 polypropylene is inserted laterally behind the muscles of the lateral and posterior pharyngeal walls. (B) Passing behind the muscles of left lateral pharyngeal wall and the soft palate, the needle comes out at the same point of insertion. (C) One cerclage with its two ends suture is formed. (D) The suture ends of each ring are tied together. (E) With insertion of the second circle, two levels (1-0 polypropylene suture materials) circles around the muscles of the velopharynx are formed. Each cerclage brings the four walls near each other like a sphincter.
palate and lateral pharyngeal wall (at the level of the junction between the posterior and middle one-thirds of the soft palate) (Fig. 1A). Passing behind the muscles of the lateral and posterior pharyngeal walls the needle came out at the left corner of the posterior pharyngeal wall (Fig. 1A). Then passing behind the muscles of left LPW and the soft palate, the needle came out at the same
Cerclage sphincter pharyngoplasty
797
point of insertion (Fig. 1B). A second cerclage suture was repeated 3 mm in front of the first. After adjusting the size of the VF valve closure, each suture ends of each ring was tied together (Fig. 1C and D). The degree of tightness depends on the degree of preoperative closing activity. Using nasal endoscopy can allow observing the needed tightness degree. Each cerclage would bring the four walls near each other like a sphincter (Fig. 1C and D). The knots were secured and buried. Buried site was closed with absorbable suture (coated vicryl 4-0).
Fig. 2 Speech analysis result: hypernasality rating scale pre- and post-sphincter cerclage pharyngoplasty. The difference between the hypernasality before and after operation was significant ( p < 0.05).
4. Results The study population included two males and four females with ages ranging from 6 to 20 years and a mean of 13 years. Distribution of patients according to the cause of VPI was clarified (Table 3).
Using Spearman rank test, the more months postoperatively the less the degree of hypernasality ( p = 0.03). 4.1.2. Hyponasality No patient had hyponasality post-operative.
4.1. Speech analysis 4.1.1. Hypernasality Before surgery five patients (83.3%) had sever hypernasality (rating scale 3). Only one patient (16.7%) had very sever hypernasality (rating scale 4). After the cerclage operation, five patients (83%) improved to moderate hypernasality (rating scale 2) (Fig. 2). Only one patient improved from rating scale 4 to 3. The difference between the hypernasality before and after operation was significant ( p < 0.05). After speech therapy (after the operation) four patients improved to normal (rating scale 0) (66.6%), one improved to mild hypernasality (rating scale 1) (16.7%) and the other one improved to moderate hypernasality (rating scale 2) (17%).
4.1.3. Weak pressure consonants One patient could not be evaluated because she had glottal articulation. After the cerclage operation, the number of patients with weak pressure consonants was changed significantly from four (66.6%) to none (0%) ( p < 0.05). Using Spearman rank test, the more months post-operatively the less the degree of weak pressure consonants ( p = 0.02). 4.1.4. Glottal stops One patient (16.7%) had glottal articulation before surgery and none had it afterwards, the difference being significant ( p < 0.05).
Table 3 Distribution of patients according to the cause of velopharyngeal insufficiency (VPI)), sex, age at the pharyngeal surgery, number of months at recording, presence of additional malformations/syndrome and of speech therapy preoperatively and post-operatively Cause of VFI
Submucus cleft palate repaired with furlow palatoplasty Post-adenoidectomy Adenoid atrophy with short velum Facioscapulohumoral (LandouzyDejerine) muscular dystrophy and adenoidectomy Occult submucus cleft palate with adenoidectomy Post-adenotonsillectomy
Sex
Number of months after operation
Additional anomaly
Duration of speech therapy before operation
Duration of speech therapy after operation
9
Female
15
No
6
3
10 5 20
Female Male Female
11 9 8
6 6 6
2 2 4
17
Male
5
No No Anterior tibial weakness No
6
3
14
Female
4
No
6
2
Age
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A. Ragab 1) in one patient ( p < 0.05) (Fig. 4). Using Spearman rank test, the more months post-operatively the better the degree of velopharyngeal closure ( p = 0.03).
4.3. Complications
Fig. 3 VF valve closure rating scale before and after cerclage sphincter pharyngoplasty. The difference between VF valve closure before and after operation was significant ( p < 0.05).
4.2. Nasopharyngeal and oral endoscopy 4.2.1. Velopharyngeal valve closure Before the surgery the pattern of closure was three sagittal (50%), two circular (33.3%) and one coronal (16.7%). The closing activity was (rating scale 3) in five patients (83.3%) and (rating scale 2) in one patient (18%). After the cerclage operation the closing activities changed significantly to (rating scale 2) in five patients (83%) ( p < 0.05) and to (rating scale 3) in one patient (16.7%) (Fig. 3). After post-operative speech therapy the closure pattern changed significantly to complete closure (rating scale 0) in five patients (83.3%) and (rating scale
Fig. 4 Nasopharyngeal endoscopy velopharyngeal valve closure in patient number (4): the post-operative closing pattern after 8 months was complete closure (rating scale 0).
No complications were observed in all the patients except for throat pain. It was mainly unilateral at the suture tie and disappeared maximally after 3 weeks.
5. Discussion Using the physiological principles of VF valve closure allowed the development of this new procedure for VFI. Normally the velum moves in a superior and posterior direction until it makes firm contact with the posterior pharyngeal wall or adenoid pad [3]. The velum must be of sufficient length, once elevation and stretching has occurred, to span the entire depth of the pharynx [14]. Cerclage suturing technique is a novel technique designed by the author. After cerclage pharyngoplasty, VF function simulates the normal velopharyngeal function. It allows backward displacement and stretching of the velum posterioly, the lateral pharyngeal walls medially and the posterior pharyngeal wall anteriorly. Thus, the cerclage permits stretching of the main velum closure muscles (the palatal tensor, the palatal levator and the superior pharyngeal constrictor). At the same time it decreasess the needed gap for closure. Thus, an easy and physiological closure pattern can be seen. Subsequently the coordinated movement of these structures results in complete closure of the valve at appropriate times for normal speech. As the velum does not move like a hinged trap door but only the anterior part of a complex velopharyngeal valve which functions as a circular sphincter [14]. Also as the Levator veli palatni muscle occupies the intermediate 40% of the length of the soft palate [15,16]. So selection of the cerclage site to be between the middle and the posterior one-thirds of the soft palate facilitates the velum to close at the active site of closure. Also it lends a handed traction of the Levator veli palatni muscle, which is the major elevator of the velum. Using two levels cerclage with the above physiological mechanism permits closure of the VF valve in a three-dimensional pattern [3]. Partial obstruction, either temporary or permanent, of the velopharyngeal port is the unifying feature of most current operative management strategies for VPD. The choice of either of the two broad categories options for VPI depends on
Cerclage sphincter pharyngoplasty the patient’s specific diagnosis. The first option category is [1] lengthening the palate by retropositioning the velum, which can be achieved with a V-Y pushback procedure, an intravelar veloplasty [17] or a double-opposing Z-plasty [18], and palatal rerepair [19]. The second option category is reduction of the static opening between the nasopharynx and the oropharynx [10,20], which is considered a velopharyngeal narrowing procedure. It may be accomplished with a pharyngeal flap, sphincter pharyngoplasty or retropharyngeal and velar augmentations. Different from all the above velopharyngeal procedures cerclage sphincter pharyngoplasty is not a passive narrowing procedure. It is a dynamic one using the normal muscles in place with no disturbance of different surrounding structures. Also the sizes of the gaps were reduced in all the cases significantly. During the last 10 years, there had been a movement toward tailoring the choice of surgical procedures for the correction of VPI to the defect exhibited by the patient and manner of closure [6]. This is not universally accepted and remains a debatable issue. Many centers have adopted this philosophy with satisfactory speech outcome. The University of Florida experience outlines the specific prospective criteria for the use of four surgical procedures for the correction of VPI. Their success rates for each of the four procedures were all similar and are near 90% [9]. They attributed their successful outcomes to matching the functional capability of the patient with the surgical procedure that would capitalize on the residual function of the velum [9]. On the contrary cerclage sphincter phayngoplasty could be applied to different types VFI closure with high success rate. As the most important issue is to find out if speech has improved or not after any VFI operation [21], our results showed that speech was improved significantly after cerclage operation especially when followed by speech therapy. Post-operative speech therapy helps such patients to use their VF and the surrounding muscles in the right manner. This can be seen clearly with the significant more improvement with time. Except for throat pain no complications such as post-operative bleeding, fistulas, painful stiff neck (Grisel’s syndrome), dehiscence or upper airway obstruction were observed in any of the patients operated upon using our procedure. This is in comparison to the reported complications in other velopharyngeal narrowing procedures [8]. Using cerclage procedure, the normal VP muscles can accommodate during different normal tasks. So
799 no sleep disturbance or hyponasality was observed subjectively, although it was not confirmed objectively with polsomnography. On the contrary, pharyngeal flap procedures were associated with the development of hyponasal speech (5—10%) [8] and obstructive sleep apnea (2—10%) [11]. Sphincter pharyngoplasty, although creating a clinically obvious reduction of velopharyngeal diameter, generally did not lead to the occurrence of an obstructive sleep apnea syndrome. However, a significant reduction of slow wave sleep quantity and a trend toward an increase in the number of cortical microarousals was observed [12]. One of the limitation of this study was the limited number of the patients without a control group, although this study was just to present the author new procedure. Also the long-term follow up was another limitation.
6. Conclusion Cerclage sphincter pharyngoplasty is a new procedure designed by the author in velopharyngeal insufficiency with the following advantages: Easy with no tissue flaps transfer. Physiological and functional in three-dimensional patterns. Not dependent on the velopharyngeal type of closure. No upper airway obstruction. No hyponasality.
Acknowledgments The skilful assistance of Dr. Eman Ezat in speech analysis and therapy, the valuable statistical analysis of data by Dr. Manal Elbatanony Ph.D. and the nice drawing of the procedure by Mr. Esam Ibrhim is greatly acknowledged.
References [1] D.J. Zajac, R. Mayo, R. Kataoka, Nasal coarticulation in normal speakers: a re-examination of the effects of gender, J. Speech Lang. Hear. Res. 41 (3) (1998) 503—510. [2] L. Skolnick, G.N. McCall, M. Barnes, The sphincteric mechanism of velopharyngeal closure, Cleft Palate J. 10 (1973) 286—305. [3] C.B. Croft, R.J. Shprintzen, S.J. Rakoff, Patterns of velopharyngeal valving in normal and cleft palate subjects: a multi-view video-endoscopic and nasendoscopic study, Laryngoscope 91 (1981) 265—271.
800 [4] D.F. Johns, R.J. Rohrich, M. Awada, Velopharyngeal incompetence: a guide for clinical evaluation, Plast. Reconstr. Surg. J. 112 (7) (2003) 1890—1897. [5] A.W. Kummer, M. Briggs, L. Lee, The relationship between the characteristics of speech and velopharyngeal gap size, Cleft Palate Craniofac. J. 40 (6) (2003) 590— 596. [6] V.L. Dixon-Wood, W.N. Williams, M.B. Seagle, Team acceptance of specific recommendations for the treatment of VPI as provided by speech pathologists, Cleft Palate Craniofac. J. 28 (1991) 285—290. [7] J. Marsh, Management of velopharyngeal dysfunction: differential diagnosis for differential management, Craniofac. Surg. J. 14 (5) (2003) 621—629. [8] J. Paul Willging, Velopharyngeal insufficiency, Curr. Opin. Otolaryngol. Head Neck Surg. 11 (2003) 452—455. [9] M.B. Seagle, M.K. Mazaheri, V.L. Dixon-Wood, W.N. Williams, Evaluation and treatment of velopharyngeal insufficiency: the University of Florida experience, Ann. Plast. Surg. 5 (2002) 464—470. [10] G.M. Sloan, Posterior pharyngeal flap and sphincter pharyngoplasty: the state of the art, Cleft Palate Craniofac. J. 37 (2) (2000) 112—122. [11] Y.F. Liao, M.L. Chuang, P.K. Chen, N.H. Chen, C. Yun, C.S. Huang, Incidence and severity of obstructive sleep apnea following pharyngeal flap surgery in patients with cleft palate, Cleft Palate Craniofac. J. 3 (2002) 312— 316. [12] C. Saint Raymond, G. Bettega, C. Deschaux, J. Lebeau, B. Raphael, P. Levy, et al., Sphincter pharyngoplasty as a treatment of velopharyngeal incompetence in young people: a prospective evaluation of effects on sleep structure
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[13]
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[16]
[17] [18]
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[20] [21]
and sleep respiratory disturbances, Chest 125 (3) (2004) 864—871. A. Ysunza, Physiology of pharyngeal muscles after surgical restoration of the velopharyngeal sphincter, Gac. Med. Mex. 141 (3) (2005) 195—199. A.W. Kummer, Anatomy and physiology: the orofacial structures and velopharyngeal valve, in: A.W. Kummer (Ed.), Cleft Palate and Craniofacial Anomalies: The Effects on Speech and Resonance, Singular Thompson Learning, Albany, 2001, pp. 3—32. F.V. Mehendale, Surgical anatomy of the levator veli palatini: a previously undescribed tendinous insertion of the anterolateral fibers, Plast. Reconstr. Surg. 114 (2) (2004) 307—315. J.G. Boorman, B.C. Sommerlad, Levator palatine and palatal Dimples: their anatomy, relationship and clinical significance, Br. J. Plast. Surg. 38 (1985) L326. J.L. Marsh, L.M. Grames, B. Holtman, Intravelar veloplasty: a prospective study, Cleft Palate J. 26 (1) (1989) 46—50. L.L. D’Antonio, Correction of velopharyngeal insufficiency using the Furlow double-opposing Z-plasty, West J. Med. 167 (2) (1997) 101—102. B.C. Sommerlad, F.V. Mehendale, M.J. Birch, D. Sell, C. Hattee, K. Harland, Palate re-repair revisited, Cleft Palate Craniofac. J. 39 (3) (2002) 295—307. D. LaRossa, The state of the art in cleft palate surgery, Cleft Palate Craniofac. J. 37 (3) (2000) 225—228. J. Liedman-Boshko, A. Lohmander, C. Persson, A. Lith, A. Elander, Perceptual analysis of speech and the activity in the lateral pharyngeal walls before and after velopharyngeal flap surgery, Scand. J. Plast. Reconstr. Surg. Hand Surg. 39 (2005) 22—32.
www.elsevier.com/locate/ijporl
Cerclage sphincter pharyngoplasty: A new technique for velopharyngeal insufficiency Ahmed Ragab * ORL department, Menoufiya University Hospital, Egypt Received 12 November 2006; received in revised form 24 January 2007; accepted 30 January 2007
KEYWORDS Velopharyngeal insufficiency; Velopharyngeal impairment; Velopharyngeal dysfunction; Pharyngoplasty
Summary Objectives: Several surgical techniques are available for the treatment of velopharyngeal insufficiency (VFI). Each method has its own complications and non-dynamic roles. So the aim of this study was to present a novel physiological surgical technique designed by the author for reconstruction of the velopharyngeal sphincter in VFI. Methods: This prospective study included six patients with VFI (two males and four females) with ages from 5 to 20 years (mean: 12.50 years). Speeches, nasopharyngeal and oral endoscopies for velopharyngeal valve closure were measured according to a 5-point scale where 0 was equivalent to normal and 4 meant a severe (constant) deviation. They were scheduled for cerclage sphincter pharyngoplasty after failure of appropriate speech therapy. Under general anaesthesia and the patient in semiflower’s position; two level cerclages (1-0 polypropylene suture materials) were inserted behind the muscles of the velopharynx. The first at the level of junction of posterior and middle one-thirds of the soft palate passing through soft palate, left lateral pharyngeal wall, posterior pharyngeal wall, right lateral pharyngeal wall and the soft palate. The second was at 3 mm in front of the latter. The surgical technique was described in details. Results: Before surgery five patients (83.3%) had sever hypernasality (rating scale 3). After the cerclage operation and speech therapy four patients (66.6%) significantly improved to normal nasality (rating scale 0) and the remaining two patients improved to mild and moderate hypernasality (rating scale 1 and 2), respectively ( p < 0.05). By endoscopy the closing activity was (rating scale 3) in five patients (83.3%) and (rating scale 2) in one patient (18%). After the cerclage operation and speech therapy five patients (83.3%) changed significantly to complete closure (rating scale 0) and to (rating scale 1) in one patient ( p < 0.05).
* Correspondence address: Lecturer and consultant of ORL Head & Neck Surgery, Menoufiyia university Hospital, Shibin Elkom, 73, Sayed St., Tanta, Egypt. Tel.: +20 40 3420114/20 0 101709898; fax: +20 40 3315000. E-mail address: [email protected]. 0165-5876/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2007.01.020
794
A. Ragab Conclusions: Cerclage sphincter pharyngoplasty is a new procedure designed by the author in VFI. It helps the velopharynx to function physiologically in three-dimensional patterns without dependency on the type of closure. Also it is an easy technique; without tissue flaps transfer, upper airway obstruction or hyponasality. # 2007 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Several components create velopharyngeal (VP) port closure, namely the velum, the lateral pharyngeal wall (LPW) and the posterior pharyngeal wall (PPW), which together forms a three-dimensional muscular valve known as the velopharyngeal sphincter [1]. The pattern of velopharyngeal closure depends on the degree to which each of these components of the sphincter is active during closure [2]. More than four closure patterns were described [3]. Closure patterns vary among individuals, but an individual will tend to have a consistent closure pattern across varied consonant utterances [2]. Complete velopharyngeal closure is required during swallowing and production of all consonants except for the nasal consonants [4]. Velopharyngeal insufficiency (VFI) refers to the inability of the velopharyngeal sphincter to close completely during production of the oral (non-nasal) sounds of speech [5]. The primary effects of VFI are nasal air escape and hypernasality. Secondary effects of VFI include speech articulation errors (distortions, substitutions and omissions). These effects combine to reduce intelligibility of speech [4]. Although speech articulation errors may be corrected through speech therapy, the primary effects of VFI — nasal air escape and hypernasality — can only be corrected by restoring velopharyngeal competence [6]. Several surgical techniques are available for the treatment of VFI, e.g. posterior pharyngeal wall augmentation, posterior pharyngeal flaps and sphincter pharyngoplasty [7]. The common goal of these surgical techniques is to create a permanent partial obstruction of the velopharyngeal space [7]. Every method has its own complications and nondynamic roles. Posterior pharyngeal wall augmentation is appropriate in the presence of a persistent gap in the central velopharyngeal port measuring at most 1— 3 mm but it is not suitable for larger defects [8]. Also a wide variety of implantable materials has been used to augment the posterior wall. Problems with extrusion, migration, resorption and infection have been reported [9]. Avoiding implantable materials in the design of operative techniques for VFI can prevent such complications.
Pharyngeal flap is the mainstay of surgical therapy for velopharyngeal insufficiency in many facilities. In this procedure, a flap of tissue is pedicled superiorly or inferiorly to the post-pharyngeal wall and sutured to the palate. Most reports describe good speech after pharyngeal flap surgery [10]. However, there are also studies that report unpleasant side effects such as nasal obstruction, hyponasality, sleep apnea and lateral port stenosis because the flap obstructs the air passage [11]. Another draw back is its limitation to obdurate mainly a midline VF defects and mobile lateral pharyngeal walls are needed for ports to be open during breathing and closing during speech [10]. Also it limits mobility of the soft palate. A technique that avoids altering and dividing the VF into two lateral ports susceptible to stenosis and obstruction can rescue the airway. Sphincter pharyngoplasty also called lateral pharyngoplasty is another common surgical treatment for VFI. The tonsillar pillars are used as donor flaps to be sutured across an incision in the nasopharyngeal posterior wall [7]. These procedures create a transverse sphincter of variable diameter with preservation of mobility of the soft palate. This approach is advantageous when coronal or circular closure is present and lateral pharyngeal wall motion is deficient. Several studies have reported a success rate (i.e. correction or significant reduction in hypernasality) from 78 to 90% with pharyngoplasty [10]. The incidence of post-operative hyponasality is estimated to be 12—17% [12]. More superiorly raised palatopharyngeal flaps lead to a greater degree of velopharyngeal obturation. If this is excessive, an obstructive pattern of breathing can develop. Some patients still develop some mild temporary obstructive sleep patterns in the immediate post-operative period [12]. Using a novel dynamic approach suite in any type of VF defects (different sizes and shapes) without such complications of upper airway obstruction during day or sleep and without hyponasality is advantageous. Lateral pharyngeal flaps in cases of sphincter pharyngoplasties and the central pharyngeal flap in cases of pharyngeal flaps; do not create new sphincters for velopharyngeal closure. The participation of these structures is passive (absent electromyographic activity in the flaps in both techniques) [13], increasing tissue volume in speci-
Cerclage sphincter pharyngoplasty fic areas, whereas their movements are caused by the contraction of the superior constrictor pharyngeus and the levator veli palatini [13]. A new procedure with active participation of all surrounding muscle without muscle transposition will add a valuable technique in surgical treatment of VFI. So the aim of this work is to present a novel physiological surgical technique designed by the author for reconstruction of the velopharyngeal sphincter in VFI.
2. Patient and methods 2.1. Patients Six patients with VFI were enrolled in this prospective study. They were scheduled for surgical treatment of VFI after appropriate speech therapy conducted by a trained orthophonist. Patients received speech therapy, including phonologic approach and whole language intervention. The study population included two males and four females with their ages from 6 to 20 years (mean: 13 years). The eligibility criteria for the study included: velopharyngeal insufficiency with or without compensatory articulation disorder as demonstrated by phoniatric assessment, nasopharyngoscopy, oral endoscopy and lateral radiography; normal hearing as demonstrated by conventional pure-tone audiometry and Speech Discrimination Score and language development within reference limits. The study approved by the Ethics Committee of the hospital (Menoufiya University Hospital, Egypt). The parents and legal guardians of all selected patients agreed to participate in the study with an informed consent. All patients received speech therapy at least 6 months before the operation and 3 months after the operation.
2.2. Analyses 2.2.1. Speech Speech was assessed through perceptual ratings of eight speech variables by speech/language pathologist (SLP) trained in assessing speech in patients with VFI and who was blinded to whether the test stimuli were obtained pre- or post-surgery. These variables included hypernasality, hyponasality, weak pressure consonants, glottal stops, pharyngeal fricatives, nasal fricatives, velar for dental plosives and palatal for dental plosives. Each variable was rated on a 5-point scale from 0 to 4, where 0 was equivalent to normal articulation or resonance and 4
795 Table 1 The speech variables and rating scale used for the assessment of speech Hypernasality, hyponasality and weak pressure consonants 0_/Normal resonance/adequate pressure 1_/Mildly hyper/hyponasal/weak (reduced pressure) 2_/Moderately hyper/hyponasal/weak (reduced pressure) 3_/Severely hyper/hyponasal/weak (reduced pressure) 4_/Very severely hyper/hyponasal/weak (reduced pressure) Glottal stop articulation of plosives, pharyngeal fricative articulation of fricatives and retracted oral (to palatal/velar) articulation of dental stop consonants 0_/No misarticulation 1_/Occurs once or twice 2_/Occurs often 3_/Occurs very often but not all the time 4_/Occurs all the time
meant a severe (constant) deviation (Table 1). Speech samples include vowels /a/ and /i/. It includes [ma ma ma], [pI pI pI], [tI tI tI], [kI kI kI], [ka ka ka]. Also to repeat or read aloud standardized passages in Arabic language that is totally free of nasal consonants and passages loaded with nasal consonants. These were evaluated in a quiet room by the same SLP on two different occasions within a 2-week period on recorded voice. Each recording was given a number that could be heard on the tape. 2.2.2. Nasopharyngeal and oral endoscopy for velopharyngeal valve closure The visual perceptual judgments were made by the same SLPs who evaluated the speech and the Otorhinolaryngology surgeon (judges 1 and 2). In this analysis, the judges independently estimated the closing pattern activity according to a fivepoint scale, from zero to four, where zero represented a complete closure in the midline and four was equivalent to little or no activity (Table 2). Rating obtained by consensus through discussion amongst judges. Judgments were each made on three different occasions. In addition to the closing pattern also the movements of the lateral pharyngeal walls, posterior walls and velum were assessed. When judging asymmetry, however, both lateral pharyngeal walls were observed together. Analysis was obtained before speech therapy, 2 weeks before the operation, 1 month after the operation (3 and 6 months) after speech therapy and at the same week of recording the study results (Table 3).
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Table 2 The rating scales for the assessment of velopharyngeal valve closure before and after cercelage sphicter surgery (a) Closing activity 0_/100% (complete) 1_/75% 2_/50% 3_/25% 4_/None or hardly any (b) Asymmetry Yes/No Right/Left (c) Shape: coronal, sagittal, circular, others The activity in each side was assessed separately. (a) Closing activity and (b) asymmetry (which was assessed by observing both sides together).
2.3. Statistics The data was collected, tabulated and analyzed by SPSS statistical package Version II on IBM compatible computer. Qualitative data expressed as number and corresponding percentages were analyzed by applying Chi-Square test to compare preoperative, post-operative and post-speech variables. Spearman rank correlation test was used to detect the association between quantitative variables. All tests were used as tests of significance at 0.05 levels.
3. Operation After induction of general orotracheal anaethesthia, the patient was placed in semiflower’s position with shoulder roll in place. The patient was prepared and draped in the normal manner. A self-retaining mouth retractor was placed in the mouth for exposure. Lidocaine epinephrine 1/ 100,000 was administered in the proposed sites of suture cercelage insertion. A catheter was inserted through the nose to aid in palatal elevation during LPW and PPW sutures or using a retractor to elevate the soft palate. Using methelyne blue allowed marking of the proposed posterior pharyngeal wall closure site. Two levels cerclage: the first at the level of junction of posterior and middle one-thirds of the soft palate passing through soft palate left LPW, PPW, right LPW and the soft palate. The second was at 3 mm in front of the first level (Fig. 1). A special needle designed for this purpose was breaded with 1-0 polypropylene suture material. Starting from the right side of the soft palate the needle was inserted at the junction of the soft
Fig. 1 Cerclage sphincter pharyngoplasty technique: (A) starting from the right side of the soft palate a special needle breaded with 1-0 polypropylene is inserted laterally behind the muscles of the lateral and posterior pharyngeal walls. (B) Passing behind the muscles of left lateral pharyngeal wall and the soft palate, the needle comes out at the same point of insertion. (C) One cerclage with its two ends suture is formed. (D) The suture ends of each ring are tied together. (E) With insertion of the second circle, two levels (1-0 polypropylene suture materials) circles around the muscles of the velopharynx are formed. Each cerclage brings the four walls near each other like a sphincter.
palate and lateral pharyngeal wall (at the level of the junction between the posterior and middle one-thirds of the soft palate) (Fig. 1A). Passing behind the muscles of the lateral and posterior pharyngeal walls the needle came out at the left corner of the posterior pharyngeal wall (Fig. 1A). Then passing behind the muscles of left LPW and the soft palate, the needle came out at the same
Cerclage sphincter pharyngoplasty
797
point of insertion (Fig. 1B). A second cerclage suture was repeated 3 mm in front of the first. After adjusting the size of the VF valve closure, each suture ends of each ring was tied together (Fig. 1C and D). The degree of tightness depends on the degree of preoperative closing activity. Using nasal endoscopy can allow observing the needed tightness degree. Each cerclage would bring the four walls near each other like a sphincter (Fig. 1C and D). The knots were secured and buried. Buried site was closed with absorbable suture (coated vicryl 4-0).
Fig. 2 Speech analysis result: hypernasality rating scale pre- and post-sphincter cerclage pharyngoplasty. The difference between the hypernasality before and after operation was significant ( p < 0.05).
4. Results The study population included two males and four females with ages ranging from 6 to 20 years and a mean of 13 years. Distribution of patients according to the cause of VPI was clarified (Table 3).
Using Spearman rank test, the more months postoperatively the less the degree of hypernasality ( p = 0.03). 4.1.2. Hyponasality No patient had hyponasality post-operative.
4.1. Speech analysis 4.1.1. Hypernasality Before surgery five patients (83.3%) had sever hypernasality (rating scale 3). Only one patient (16.7%) had very sever hypernasality (rating scale 4). After the cerclage operation, five patients (83%) improved to moderate hypernasality (rating scale 2) (Fig. 2). Only one patient improved from rating scale 4 to 3. The difference between the hypernasality before and after operation was significant ( p < 0.05). After speech therapy (after the operation) four patients improved to normal (rating scale 0) (66.6%), one improved to mild hypernasality (rating scale 1) (16.7%) and the other one improved to moderate hypernasality (rating scale 2) (17%).
4.1.3. Weak pressure consonants One patient could not be evaluated because she had glottal articulation. After the cerclage operation, the number of patients with weak pressure consonants was changed significantly from four (66.6%) to none (0%) ( p < 0.05). Using Spearman rank test, the more months post-operatively the less the degree of weak pressure consonants ( p = 0.02). 4.1.4. Glottal stops One patient (16.7%) had glottal articulation before surgery and none had it afterwards, the difference being significant ( p < 0.05).
Table 3 Distribution of patients according to the cause of velopharyngeal insufficiency (VPI)), sex, age at the pharyngeal surgery, number of months at recording, presence of additional malformations/syndrome and of speech therapy preoperatively and post-operatively Cause of VFI
Submucus cleft palate repaired with furlow palatoplasty Post-adenoidectomy Adenoid atrophy with short velum Facioscapulohumoral (LandouzyDejerine) muscular dystrophy and adenoidectomy Occult submucus cleft palate with adenoidectomy Post-adenotonsillectomy
Sex
Number of months after operation
Additional anomaly
Duration of speech therapy before operation
Duration of speech therapy after operation
9
Female
15
No
6
3
10 5 20
Female Male Female
11 9 8
6 6 6
2 2 4
17
Male
5
No No Anterior tibial weakness No
6
3
14
Female
4
No
6
2
Age
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A. Ragab 1) in one patient ( p < 0.05) (Fig. 4). Using Spearman rank test, the more months post-operatively the better the degree of velopharyngeal closure ( p = 0.03).
4.3. Complications
Fig. 3 VF valve closure rating scale before and after cerclage sphincter pharyngoplasty. The difference between VF valve closure before and after operation was significant ( p < 0.05).
4.2. Nasopharyngeal and oral endoscopy 4.2.1. Velopharyngeal valve closure Before the surgery the pattern of closure was three sagittal (50%), two circular (33.3%) and one coronal (16.7%). The closing activity was (rating scale 3) in five patients (83.3%) and (rating scale 2) in one patient (18%). After the cerclage operation the closing activities changed significantly to (rating scale 2) in five patients (83%) ( p < 0.05) and to (rating scale 3) in one patient (16.7%) (Fig. 3). After post-operative speech therapy the closure pattern changed significantly to complete closure (rating scale 0) in five patients (83.3%) and (rating scale
Fig. 4 Nasopharyngeal endoscopy velopharyngeal valve closure in patient number (4): the post-operative closing pattern after 8 months was complete closure (rating scale 0).
No complications were observed in all the patients except for throat pain. It was mainly unilateral at the suture tie and disappeared maximally after 3 weeks.
5. Discussion Using the physiological principles of VF valve closure allowed the development of this new procedure for VFI. Normally the velum moves in a superior and posterior direction until it makes firm contact with the posterior pharyngeal wall or adenoid pad [3]. The velum must be of sufficient length, once elevation and stretching has occurred, to span the entire depth of the pharynx [14]. Cerclage suturing technique is a novel technique designed by the author. After cerclage pharyngoplasty, VF function simulates the normal velopharyngeal function. It allows backward displacement and stretching of the velum posterioly, the lateral pharyngeal walls medially and the posterior pharyngeal wall anteriorly. Thus, the cerclage permits stretching of the main velum closure muscles (the palatal tensor, the palatal levator and the superior pharyngeal constrictor). At the same time it decreasess the needed gap for closure. Thus, an easy and physiological closure pattern can be seen. Subsequently the coordinated movement of these structures results in complete closure of the valve at appropriate times for normal speech. As the velum does not move like a hinged trap door but only the anterior part of a complex velopharyngeal valve which functions as a circular sphincter [14]. Also as the Levator veli palatni muscle occupies the intermediate 40% of the length of the soft palate [15,16]. So selection of the cerclage site to be between the middle and the posterior one-thirds of the soft palate facilitates the velum to close at the active site of closure. Also it lends a handed traction of the Levator veli palatni muscle, which is the major elevator of the velum. Using two levels cerclage with the above physiological mechanism permits closure of the VF valve in a three-dimensional pattern [3]. Partial obstruction, either temporary or permanent, of the velopharyngeal port is the unifying feature of most current operative management strategies for VPD. The choice of either of the two broad categories options for VPI depends on
Cerclage sphincter pharyngoplasty the patient’s specific diagnosis. The first option category is [1] lengthening the palate by retropositioning the velum, which can be achieved with a V-Y pushback procedure, an intravelar veloplasty [17] or a double-opposing Z-plasty [18], and palatal rerepair [19]. The second option category is reduction of the static opening between the nasopharynx and the oropharynx [10,20], which is considered a velopharyngeal narrowing procedure. It may be accomplished with a pharyngeal flap, sphincter pharyngoplasty or retropharyngeal and velar augmentations. Different from all the above velopharyngeal procedures cerclage sphincter pharyngoplasty is not a passive narrowing procedure. It is a dynamic one using the normal muscles in place with no disturbance of different surrounding structures. Also the sizes of the gaps were reduced in all the cases significantly. During the last 10 years, there had been a movement toward tailoring the choice of surgical procedures for the correction of VPI to the defect exhibited by the patient and manner of closure [6]. This is not universally accepted and remains a debatable issue. Many centers have adopted this philosophy with satisfactory speech outcome. The University of Florida experience outlines the specific prospective criteria for the use of four surgical procedures for the correction of VPI. Their success rates for each of the four procedures were all similar and are near 90% [9]. They attributed their successful outcomes to matching the functional capability of the patient with the surgical procedure that would capitalize on the residual function of the velum [9]. On the contrary cerclage sphincter phayngoplasty could be applied to different types VFI closure with high success rate. As the most important issue is to find out if speech has improved or not after any VFI operation [21], our results showed that speech was improved significantly after cerclage operation especially when followed by speech therapy. Post-operative speech therapy helps such patients to use their VF and the surrounding muscles in the right manner. This can be seen clearly with the significant more improvement with time. Except for throat pain no complications such as post-operative bleeding, fistulas, painful stiff neck (Grisel’s syndrome), dehiscence or upper airway obstruction were observed in any of the patients operated upon using our procedure. This is in comparison to the reported complications in other velopharyngeal narrowing procedures [8]. Using cerclage procedure, the normal VP muscles can accommodate during different normal tasks. So
799 no sleep disturbance or hyponasality was observed subjectively, although it was not confirmed objectively with polsomnography. On the contrary, pharyngeal flap procedures were associated with the development of hyponasal speech (5—10%) [8] and obstructive sleep apnea (2—10%) [11]. Sphincter pharyngoplasty, although creating a clinically obvious reduction of velopharyngeal diameter, generally did not lead to the occurrence of an obstructive sleep apnea syndrome. However, a significant reduction of slow wave sleep quantity and a trend toward an increase in the number of cortical microarousals was observed [12]. One of the limitation of this study was the limited number of the patients without a control group, although this study was just to present the author new procedure. Also the long-term follow up was another limitation.
6. Conclusion Cerclage sphincter pharyngoplasty is a new procedure designed by the author in velopharyngeal insufficiency with the following advantages: Easy with no tissue flaps transfer. Physiological and functional in three-dimensional patterns. Not dependent on the velopharyngeal type of closure. No upper airway obstruction. No hyponasality.
Acknowledgments The skilful assistance of Dr. Eman Ezat in speech analysis and therapy, the valuable statistical analysis of data by Dr. Manal Elbatanony Ph.D. and the nice drawing of the procedure by Mr. Esam Ibrhim is greatly acknowledged.
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