UNIVERSITY OF CINCINNATI

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1 UNIVERSITY OF CINCINNATI Date: I,, hereby submit this work as part of the requirements for the degree of: in: It is entitled: This work and its defense approved by: Chair:

2 Speech Outcomes following Surgical Management of Velopharyngeal Dysfunction: A Survey of Craniofacial Anomalies Teams A thesis submitted to the Division of Research and Advanced Studies of the University of Cincinnati In partial fulfillment of the Requirements for the degree of MASTERS OF ARTS in the Department of Communication Sciences and Disorders of the College of Allied Health Sciences 2005 by Leisa C. Lauck B.S., Purdue University Committee Chair: Linda Lee, Ph.D.

3 ABSTRACT Objective: To determine how craniofacial teams are evaluating and reporting outcomes of surgery for velopharyngeal dysfunction (VPD). Methods: A survey was distributed to all speech-language pathologists, otolaryngologists, plastic and oral surgeons in the American Cleft Palate Association (ACPA) and the American Speech- Language Hearing Association (ASHA) Division 5 listserves. Results: Ninety-three questionnaires were completed. The majority of respondents reported the pharyngeal flap as the most often performed VPI surgery. Most respondents reported evaluations to include: intra-oral examination, perceptual evaluation, and nasopharyngoscopy. Reported criteria for surgical success included normal and acceptable. Hyponasality was considered indicative of success by 43%. Thirty-one percent listed improved as a criterion for success. The majority believe that surgical success should be determined by a speech-language pathologist and the patient/family. Conclusion: These results suggest that there is no standard for reporting post-surgical success following VPI surgery. The definition of success should be addressed by the craniofacial community.

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5 ACKNOWLEDGMENTS I would like to take this opportunity to thank Dr. Linda Lee for her guidance and patient assistance in completing this study. Special thanks also to Dr. Ann Kummer, a wonderful role model and teacher, without whom the success of this project would have not been possible. Thank you to Dakshika Bandaranayake, friend and mentor, for her wonderful comments and assistance. Thanks especially to Dr. David Billmire for the idea that inspired this study. To Ryan Erb, who helped create the online survey and collect data you saved me more time and frustration than you will ever know! To all of the people who helped me with charts, graphs, editing, and revising this project thank you so much! Lastly, thank you to my friends and family for your support and your wonderful patience over the past two years.

6 TABLE OF CONTENTS LIST OF TABLES AND FIGURES... 3 CHAPTER 1: INTRODUCTION 4 Definition and etiology of VPI 4 Surgical Treatment...5 Assessment Procedures...7 Research Questions..8 CHAPTER 2: LITERATURE REVIEW... 8 Goals of VPI Surgery...9 Speech Outcomes...10 Instrumental Assessment...17 Perceptual Assessment...20 Craniofacial Team Member Roles.21 Purpose of the Study..23 Hypotheses.24 CHAPTER 3: METHODS.25 Subjects..25 Distribution of survey 25 CHAPTER 4: RESULTS...26 Demographics 26 Surgical Preferences..27 VPI Assessment Procedures..28 Speech Outcomes..30 1

7 Craniofacial Team Member Roles. 31 CHAPTER 5: DISCUSSION.31 Surgical Preferences..32 VPI Assessment Procedures..32 Defining Surgical Success.35 Limitations.37 Implications 37 REFERENCES.39 2

8 LIST OF TABLES AND FIGURES Table Table Figure 1.26 Figure 2.27 Figure 3.27 Figure 4.28 Figure 5.29 Figure 6.29 Figure 7.30 Figure

9 CHAPTER 1: INTRODUCTION One in approximately every 750 children in the United States is born with a cleft palate. Reports estimate that 20% to 43% of these children will develop a condition called velopharyngeal dysfunction, or VPD (Cable, Canady, M. Karnell, L. Karnell, and Malick, 2004; Kummer, 2001). VPD caused primarily by a short velum is called velopharyngeal insufficiency, while VPD caused by poor velar movement during speech is called velopharyngeal incompetence. In a typical speaker, the velum, or soft palate, elevates and moves posteriorly in coordination with medial movement of the lateral pharyngeal walls and anterior excursion of the posterior pharyngeal wall. These coordinated movements create a seal in the back of the nasopharyngeal cavity, thus closing off the nasal cavity from the oral cavity during most speech sounds (Boseley and Hartnick, 2004). However, if a person s velum is not long enough to create the necessary seal, air pressure and acoustic sound energy enter the nasal cavity during speech (Pannbacker, 2004). This is referred to by many professionals as velopharyngeal insufficiency. Similarly, if a person s velum is not functioning properly, regardless of its length, the same results may occur. This situation of normal anatomical structures with a disorder of innervation is called velopharyngeal incompetence. The general term of VPD or VPI can include both of these conditions. When too much sound enters and exits the nasal cavity during productions of nonnasal sounds, hypernasality exists. If sound is restricted from entering into the nasal cavity during speech, specifically during production of the nasal sounds m, n, and ng, a condition called hyponasality occurs. Other abnormal resonance conditions 4

10 which affect a speaker s intelligibility include nasal air emission and nasal rustle. Nasal air emissions occur due to an inappropriate flow of air through the nose causing distorted speech. This can also be referred to as nasal escape. Nasal rustle (also referred to as nasal turbulence) describes a fricative sound that occurs when air is pushed through a small velopharyngeal opening during speech. This results in a bubbling of oral and nasal secretions above the velopharyngeal opening which can be viewed during a nasopharyngoscopy (Kummer, 2001). Speech therapy can aid in some cases of VPI, particularly when velopharyngeal closure is inconsistent, hypernasality is phoneme specific, or when compensatory patterns such as glottal articulation are present (Dixon-Wood et al., 1991). Speech therapy should not be considered an option for those individuals for whom VPI is being caused by structural abnormalities alone, because if the structure of the velopharyngeal mechanism is inadequate, no amount of speech therapy can correct the resonance disorder (Kummer, 2001). Most individuals with VPI will require surgical intervention to correct the condition. A number of techniques have been developed and are currently being used in craniofacial centers across the United States. These surgeries include pharyngeal flap, the sphincter pharyngoplasty or Orticochia, pharyngeal wall augmentation, and the Furlow Z-plasty. Controversy over which surgical methods are most ideal for managing VPI is widespread (Lin, Goldberg, Williams, Borowitz, Persing, and Edgerton, 1999; Pannbacker, 2004). Currently, the most common surgical treatments across the United States are the pharyngeal flap and the sphincter pharyngoplasty (Boseley and Hartnick, 2004; De Serres, Deleyiannis., Eblen, Gruss, Richardson, and Sie, 1999; Gosain, Conley, 5

11 Marks, and Larson, 1996; Sloan, 2000; Ysunza, Pamplona,, Molina,, Drucker, Felemovicius, Ramirez, and Patino, 2004). Although experience and preference of the surgeon performing the procedure are essential factors in determining surgical outcome, anatomy and physiology of the velopharyngeal port must be considered in order to choose the optimal surgery for each individual (Amour, Fischbach, Klaiman, and Fisher, 2005; Seagle, Mazaheri, Dixon-Wood, and Williams, 2002; Sie et al., 1998, Riski et al., 1992). Specifically, the VP closure pattern is important to assess before deciding on the appropriate surgical procedure. There are three common identified velopharyngeal closure patterns. Coronal is the most commonly seen closure pattern, and is characterized by velar closure to a broad area of the posterior pharyngeal wall, with little medial movement of the lateral pharyngeal walls. Circular is the next most common closure pattern, and is characterized by movement of both the lateral pharyngeal walls medially and the velum moving posteriorly to the posterior pharyngeal wall. The least common closure pattern is sagittal, in which velopharyngeal closure is achieved primarily through medial movement of the lateral pharyngeal walls, with little movement for closure by the velum and posterior pharyngeal wall (Kummer, 2001). Variations and modifications of the pharyngeal flap and sphincter pharyngoplasty surgeries exist, but the general procedures are consistent. A pharyngeal flap consists of a medial tissue bridge connecting the velum to the posterior pharyngeal wall, thereby decreasing the size of the velopharyngeal gap and increasing the likelihood that velopharyngeal closure can be completed during speech tasks (Sloan, 2000). With this procedure, lateral pharyngeal walls must have adequate medial movement so that they connect with the flap tissue and seal off the velopharyngeal port. The width of the flap is 6

12 dependent upon the extent of the medial lateral pharyngeal wall displacement, which can be assessed during a nasopharyngoscopic procedure (Golding-Kushner et al., 1990). Sphincter pharyngoplasty (SP) consists of 2 bilateral flaps of tissue cut from the posterior tonsillar pillars and attached to the undersurface of a superior-based pharyngeal flap, creating a sphincter in the oropharynx (Sie, Tampakopoulou, De Serres, Gruss, Eblen, and Yonick, 1998; Sloan, 2000; Ysunza et al., 2004). Greater pre-surgical posterior movement of the velum and anterior movement of the posterior pharyngeal wall (coronal closure pattern) are required for an adequate seal to be formed after the SP procedure. Visual assessment of velopharyngeal closure pattern and determination of velopharyngeal gap type and size can be acquired best through use of a nasopharyngoscopy, because it allows visualization of the actual structures and functioning of the velopharyngeal port. Videofluoroscopy is an additional instrumental measure that can provide visualization of velar, posterior pharyngeal wall, and lateral pharyngeal walls movement through x-ray. Other instrumental measures such as nasometry and aerodynamic measures can also provide information about the size of the VP gap and amount of escaping air. The American Cleft Palate Association (ACPA) proposed Evaluation and Treatment Parameters (1993) and Parameters for Evaluation and Treatment of Patients with Cleft Lip/Palate or Other Craniofacial Anomalies (2000), as guidelines for cleft palate and craniofacial anomaly teams. They state that pre-operative and post-operative speech assessments are necessary in determining candidacy for and outcomes of surgical, behavioral, and prosthetic management of the VP system. Ideally, according to the 7

13 ACPA, these assessments should include analysis of articulation, aerodynamic measures, videofluoroscopy, nasopharyngoscopy, and nasometry. These documents further state that the measures should be obtained by the cleft palate/craniofacial team s speechlanguage pathologist. Often, hospital centers and craniofacial teams prefer one type of surgical intervention over others because they believe it will provide better results. However, it is difficult to compare success rates of surgical techniques from different craniofacial teams and centers when the methods for measuring and reporting the outcomes are different. The purpose of the present investigation is to determine the ways in which craniofacial teams decide on the appropriate treatment for VPI and how they measure and report speech outcomes following treatment. What do craniofacial teams consider the goals of successful VPI surgery (i.e. elimination of hypernasality or increase in intelligibility)? Are craniofacial teams utilizing the available tools and resources to decide on the best surgical treatment for each individual? Who is involved in determining the outcomes of VPI surgery? What are the stipulations for determining a surgical procedure successful or unsuccessful? CHAPTER 2: LITERATURE REVIEW This chapter will review past studies and reports about surgical intervention for VPI and how speech outcomes were measured and reported. Subjects such as the role of the speech-pathologist, the types of measurements and procedures for gathering information related to speech, perceptual rating scales, and definitions of successful surgeries will be highlighted. 8

14 Goals of VPI Surgery Reports focusing on speech and resonance outcomes following surgeries to correct VPI are extensive. However, most of the current research varies widely across different craniofacial teams and medical centers (Cable et al., 2004). No current standard or protocol could be found that provided a template for reporting speech outcomes or defining success following surgery to correct or improve VPI. Some teams and centers use subjective perceptual data alone to differentiate between a successful versus an unsuccessful VPI surgery, while others employ objective measures through the use of acoustic and aerodynamic measures to qualify their surgeries as successful or unsuccessful. Even the definitions of successful and acceptable are open to interpretation, depending upon the individual craniofacial team or medical center. Therefore, success rates reported in the current research and literature vary widely, depending on the standards of that specific team or medical center. Most professionals agree that the ultimate goal of VPI surgery is to eliminate hypernasality in order to improve speech communication. However, slight variations in individual centers goals can reflect what they believe to be the most important outcome of surgery, and that outcome is defined differently across centers. Boseley and Hartnick (2004) claim that when they are performing VPI surgery, the ultimate goal should be to restore adequate function of the velopharyngeal sphincter and thereby improve the child s ability to communicate (p. 1430). This definition mentions nothing of how any degree of remaining hypernasality or hyponasality would affect their judgment of a successful or unsuccessful surgical outcome. In their study comparing results of the pharyngeal flap and sphincter pharyngoplasty, de Serres et al. (1999) considered the goals of VPI surgery 9

15 to be achievement of an appropriate blend of oronasal resonance and elimination of nasal air emissions (p. 18). In their discussion, they reported that surgical outcomes were only considered to be successful if oronasal resonance was normal, which was defined as no remaining hyper or hyponasality, regardless of the degree, and no nasal emission. Riski et al. (1992) considered any degree of hypernasal resonance or air escape to be a sign of surgical failure; however, they counted hyponasal results as successful. Other researchers considered the presence of hyponasality following VPI surgery to be an unacceptable outcome of surgery (Cable et al., 2004; Grobbelaar, Hudson, Fernandes, and Lentin, 1995; Sie et al., 1998). It appears that the methods for measuring the success of the speech goals are often subjective and nonstandardized. Speech Outcomes Table I compares the results of VPI surgery as well as a comparison of definitions of success that have been reported in the literature. Data presented in this table indicate a lack of consistency in evaluating and reporting outcomes following surgery. Their primary results will be discussed in this section. Amour, Fischbach, Klaiman, and Fisher (2005) recently published a study examining the effectiveness of the pharyngeal flap as related to the individual s type of velopharyngeal closure pattern (coronal, sagittal, or circular). Fifty-seven percent of the patients with non-coronal closure pattern achieved normal nasalance scores post surgery, while only 35% of the coronal group achieved normal nasalance. In conclusion, they found that the pharyngeal flap was more effective in correcting hypernasality in individuals with non-coronal closure patterns. They also found that the non-coronal closure pattern group had less post-operative hyponasality (7%) than did the coronal 10

16 group (13%). Because of these results, patients at their center with coronal closure patterns are now more inclined to receive a sphincter pharyngoplasty to treat VPI rather than a pharyngeal flap. In 2002, Seagle, Mazaheri, Dixon-Wood, and Williams conducted a retrospective study on 75 patients who had received on of four surgical interventions for VPI. The surgeries included: palatal pushback with pharyngeal flap lining, sphincter pharyngoplasty, superiorly-based pharyngeal flap, or a Furlow palatoplasty. Surgical decisions were based on a thorough pre-surgical evaluation that included perceptual speech ratings, videofluoroscopy, and nasendoscopy. Ninety-five percent of the subjects were reported to have normal, non-nasal speech following surgery. In the discussion, the authors reiterated the importance of individualizing each surgical procedure to meet the patient s needs. In Sloan s (2000) Posterior Pharyngeal flap and Sphincter Pharyngoplasty: The State of the Art, the debate over which surgical technique provides the best results was examined. While Sloan found no clear consensus on which technique was better or worse, his review of the literature showed that neither give uniformly successful results, and both can result in negative consequences. He concluded the pharyngeal flap to be a more effective procedure, in general, for correcting severe VPI. However, the risk of patients developing hyponasality and obstructive sleep apnea following a pharyngeal flap procedure is much more prevalent than in patients receiving a sphincter pharyngoplasty. A recent study by Cable, Canady, Karnell, Karnell, and Malick (2004) on longterm speech outcomes following pharyngeal flap surgery supported the use of pharyngeal flap as surgical treatment for VPI. Speech outcomes were measured subjectively by 11

17 speech-language pathologists using two numeric scales. Hypernasality was rated perceptually on a scale of 1 through 6 (1 indicating no involvement, 6 indicating severe effect on resonance) and velopharyngeal insufficiency on a scale of 1 through 3 (1 indicating competence, 3 indicating incompetence). Results of the study determined the pharyngeal flap to be an adequate treatment to correct VPI, although the authors failed to define what adequate in this case represents. No percentages of success were given and the authors noted at the end of the report that a large limitation of the study was that the findings were based on subjective measures only. Meek, Coert, Hofer, Goorhuis-Brouwer, and Nicolai (2003) also reported on short and long-term speech results following pharyngeal flap surgery. Additionally, they analyzed the 93 patients for any differences correlated with age (less than 6 years old, over 6 years old). While speech improved in all of the patients, there was better improvement in children who received the pharyngeal flap younger than age 6. David, Blalock, and Argenta (1999) reported speech outcomes following a uvular transposition to repair cleft palate. While the surgery performed was not to correct or repair VPI, the methods used for measuring speech outcomes apply to VPI surgery. A speech-language pathologist evaluated speech and resonance using perceptual ratings. The rating system that was used is not explained within the study. Nasometry was also used to objectively measure success of surgery. A nasalance score of less than or equal to 25% was considered within normal limits. It is important to note that less than 25% would also include hyponasal resonance. Dejonckere and Van Wijngaarden (2001) used both subjective measures through speech-language pathologist s perceptual evaluation and objective measures through 12

18 acoustic nasometry to report speech outcomes following a fat transplantation surgical procedure to correct VPI. Seventeen subjects with slight to moderate VPI, as determined by an unspecified perceptual evaluation by the SLP, nasopharyngoscopy, and x-ray contrast pharyngoplasty, and aerodynamic tests, qualified for this study. All subjects read a standardized phonetically selected normal Dutch spoken passage, and a standardized phonetically selected denasal Dutch passage of comparable length. Perceptual evaluations, nasometry, and a parent/child satisfaction questionnaire following surgery were used to determine success. All children were rated perceptually as less nasal following the fat transplant. It was not reported whether or not any remaining hypernasality or hyponasality was acceptable. Questionnaire results revealed that all patients or parents in this study were satisfied with the results. The mean nasalance score of the seventeen patients involved in this study reached nasometer values that fell in the normal range (31.95%), as standardized by speakers without VPI reading the same passage. While these results are encouraging, a limitation of the study is that there is not a perfect correlation between nasalance scores and perceptual features (Nellis, Neiman, and Lehman, 1992; Vallino-Napoli and Montgomery, 1997). De Serres, Deleyiannis, Eblen, Gruss, Richardson, and Sie (1999) did a retrospective study comparing speech outcome results of patients receiving either a sphincter pharyngoplasty or a pharyngeal flap in a five-year period. Pre-surgery evaluations included perceptual ratings by two speech pathologists, nasendoscopy, and videofluoroscopy to determine the extent of the VPI. Post-surgery results showed a slightly higher success rate with lower incidence of complications for patients undergoing pharyngoplasty versus pharyngeal flap surgery, although this difference was not 13

19 statistically significant. Fifty percent of the patients who received the pharyngoplasty had resolution, and only 22.2% of those receiving a pharyngeal flap had complete resolution. However, success was rated solely by perception of two speech pathologists, using a four-point rating scale for VPI severity, a three point rating scale for nasal air emissions, a five-point rating scale for nasal resonance, and a four-point rating scale for overall speech intelligibility. No objective measurements were used in the determination of surgical success. Outcomes were only deemed successful if there was complete resolution of symptoms (resonance was rated as normal and there were no nasal air emissions.) Hyponasality was not considered to be an acceptable outcome of surgery. Mehendale and Sommerlad (2002) did a study describing the use of the Moore pharyngoplasty modification technique in treatment of asymmetric VPI. In this study, 18 patients underwent a unilateral Moore pharyngoplasty to treat asymmetric VPI. Pre and post surgery speech assessment included recorded speech samples of subjects counting, producing repeated syllables and sentences, and conversation. Tapes were then analyzed and scored according to the Cleft Audit Protocol for Speech (CAPS) (Razzell, Harding, and Harland, 1997). This scoring system ranks nasality and nasal airflow errors on a scale of 0-3 (0 indicating normal, 3 indicating severe). Nasendoscopy was also performed in most cases, and a four-point scale was used to assess VP gap size (range from no gap, small gap, bubbling, medium gap and large gap). Results showed a significant improvement in hypernasality, and a trend toward improvements in nasal emission and nasal turbulence. Four patients had postoperative mild hyponasality and two patients had persistent dysphonia following surgery. No percentage of success was 14

20 given and it is unclear whether any remaining hypernasal or other symptoms were determined to be a success. Mehendale et al. (2003) reported on speech outcomes in 42 patients with Velocardiofacial Syndrome (VCF) who received either a radical dissection and retropositioning of velar muscles (submucous cleft palate [SMCP] repair), a Hynes pharyngoplasty (type of sphincter pharyngoplasty), or a combination of the two procedures. Patients received pre-surgery speech assessment which included a perceptual speech evaluation, recordings of speech, lateral videofluoroscopy, and nasendoscopy. Perceptual speech ratings were made by two speech-language pathologists using the CAPS scoring system. In the group who received the SMCP Repair, there was a statistically significant improvement in hypernasality, a trend toward improvement of nasal emission, and no significant development of hyponasality. In patients who received the Hynes pharyngoplasty, there was a significant improvement in hypernasality, improvement (but not statistically significant) in nasal emission, and no significant development of hyponasality (2 of 16 subjects developed mild hyponasality). In this second group, 3 of 16 patients required further surgery. Patients who received the SMCP+Hynes procedure showed significant improvements in hypernasality and nasal emission, but 25% (3/12) developed mild hyponasality. Sie et al. (1998) retrospectively studied the speech outcomes and complications of 24 children who underwent a sphincter pharyngoplasty for management of VPI. Presurgery perceptual evaluations conducted by the team SLP included a 5-point scale for judging nasal resonance and a 3-point scale for judging nasal emissions. Objective measures also taken pre-surgery included videofluoroscopy and nasendoscopy. A 5-point 15

21 scale was utilized to rate lateral wall closure (scores of 4 or 5 considered normal ), palatal elevation was rated using a 5-point scale, and gap shape and size were categorized as coronal, round or sagittal (shape) and small, moderate, or large (size). All subjects received the same perceptual evaluation four times post-surgery. Nasendoscopy and videofluoroscopy were performed following surgery only if the perceptual evaluation revealed persistent VPI. Results of the perceptual evaluation by the SLP revealed that 62.5% of subjects had complete resolution of symptoms, 20.8% had mildly remaining VPI, 4.2% had moderate VPI, 12.5% were mildly hyponasal, and none of the subjects had post-operative obstructive sleep apnea. Successful surgery was defined clearly by these authors as normal resonance and absence of nasal air emissions. By their standards, no subjects with any remaining VPI or post-surgical hyponasality could be considered to have a successful surgical outcome. Witt, Myckatyn, and Marsh (1998) reported on speech outcomes following the revision of failed pharyngeal flaps and sphincter pharyngoplasties. Pre-treatment and post-treatment (3 and 12 months after surgery) evaluations included: nasendoscopy, videofluoroscopy, standard upper airway assessment, and perceptual speech evaluation by an SLP. Sixty-five subjects received a pharyngeal flap, of which 13 (20%) required revisions. Following the revision, 5 subjects still had unfavorable speech results, so the overall success rate of the pharyngeal flap was 93%. However, 7% of the successful subjects presented with post-operative hyponasality. Sphincter pharyngoplasty was originally performed on 123 subjects studied by these authors. Of the 123 total subjects, 20 (16%) required revisions. Following the revisions, only 3 patients had unfavorable speech results, producing an overall success rate of 97%. One subject had post-surgical 16

22 hyponasality, which was less than 1% of the total population. This was considered successful. The authors provide a clear definition of what they consider success: normal perceptual resonance, complete velopharyngeal closure documented by instrumental assessments, upper airway patency, and absence of sleep apnea" (p. 452). Although some hyponasal results were considered to be successful, the authors noted that hyponasality would not be considered to be a successful criterion if any morbidity such as upper airway patency or obstructive sleep apnea were also present with the hyponasality. Yzuna et al. (2004) reported on the speech outcomes of seventy subjects who received either a sphincter pharyngoplasty (n=35) or a pharyngeal flap (n=35) for the correction of VPI. Pre-operative evaluation included articulation assessment, nasopharyngoscopy, multi-view videofluoroscopy, and a perceptual speech evaluation. These evaluation measures were repeated four months following the operations. VPI was reported to be completely corrected in 89% of the individuals in the pharyngeal flap group and 85% of the sphincter pharyngoplasty group. The authors did not comment on the degree or type of residual VPI following surgery, or if any patients presented with post-operative hyponasality (and whether or not this was included as success ). Instrumental Assessment Table II summarizes the VPI assessment procedures as reported in the literature. The next two sections will review instrumental and perceptual assessment procedures that have been recommended by the ACPA and are currently being used across cleft palate and craniofacial teams. A 1993 survey of speech-language pathologists on cleft palate/craniofacial teams was conducted by Pannbacker et al. Results revealed the majority opinion that 17

23 nasopharyngoscopy should be included as an important piece of assessment of VPI. Sixty-four percent of these respondents also believed that nasopharyngoscopy should be performed by a speech-language pathologist, and 78.5% of respondents believed this should be done under medical supervision. However, only 35.5% of the speech-language pathologists surveyed at the time actually performed nasopharyngoscopic examinations (Pannbacker, Lass, Hansen, Mussa, and Robison, 1993). These data reveal an inconsistency in what speech language pathologists believed their role should be and what their role actually was at the time. A similar survey was conducted by D Antonio, Achauer, and Vander Kam (1993). A questionnaire was sent to all ACPA craniofacial/cleft palate teams, for which 90% of the respondents indicated that nasendoscopy was available to their teams. Of the responding teams, 90% reported that they believed nasendoscopy to be necessary for difficult diagnostic problems, but only 50% of those who responded believed that nasendoscopy was appropriate for all patients with velopharyngeal insufficiency, and 41% believed that nasendoscopy was an appropriate instrumental measure for patients for whom secondary palatal management is planned. Golding-Kushner and her international working group stressed the importance of utilizing videofluoroscopy in addition to nasopharyngoscopy when conducting an evaluation of velopharyngeal structure and function. At the end of their 1993 report, they concluded that the evaluation of velopharyngeal movement must be comprehensive and include, at the very least, frontal (P-A) and lateral view videofluoroscopy, and en face view or nasopharyngoscopy (p. 345). 18

24 Henningson and Isberg (1991) reiterated the importance of performing videofluoroscopy during assessment of velopharyngeal function in their comparison of instrumental measurements in 80 subjects with hypernasal speech. During both videofluoroscopy and nasendoscopy, subjects read sentences and consonant-vowel sequences containing a variety of pressure consonants and stops at various places of articulation and a variety of vowels. Lateral pharyngeal wall movements were then rated on a 5-point scale during both videofluoroscopy and nasendoscopy. Results showed that in a third of the patients, nasopharyngoscopy failed to show full degrees of lateral wall movement that were seen through videofluoroscopy. By combining videofluoroscopy with nasendoscopy, they found that the chance of misinterpretation of velopharyngeal movements was decreased. Another advantage of videofluoroscopy over nasendoscopy is that it is more often easier to perform on small children. They concluded their study by reporting that videofluoroscopy in multiviews is indispensable in assessing velopharyngeal function (p. 416). Nellis, Neiman, and Lehman (1992) conducted a study examining the relationship between nasalance scores obtained from the Model 6200 Nasometer and listener judgments when rating 16 subjects who had received pharyngeal flaps. Each subject s speech sample was composed of the same seven sentences. The speech samples were audio recorded and then judged by ten trained speech-pathology graduate students. Samples were rated for both hypernasality and hyponasality (scale: 1 = no resonance disorder; 6 = severe resonance disorder). Results revealed that listener ratings of increasing hypernasality were not significantly correlated with increasing nasalance scores. 19

25 Perceptual Assessment Although review of the current literature shows no universally consistent method of judging speech and defining speech outcomes following surgery to correct VPI, several different methods of success measurement have been used by individual craniofacial anomalies teams. Rating systems for perceptual measures of speech characteristics are almost always used during pre and post surgery assessments. Rating systems that have been reported in the literature include the Cleft Audit Protocol for Speech, or CAPS (Mehendale and Sommerlad, 2002; Mehendale, Birch, Birkett, Sell, and Sommerlad, 2003), the Borel-Maisonny classification (Zanzi, Cherpillod, and Hohlfeld, 2002), the Base-10 Index (Dixon-Wood et al., 1991), and numerous similar rating systems developed by individual centers (Cable et al., 2004; De Serres et al., 1999; Lin, Goldberg, Williams, Borowitz, Persing, and Edgerton, 1999; Haapanen, 1995; Riski et al., 1992; Sie et al., 1998; Witt, Marsh, Marty-Grames, Muntz, and Gay, 1995; Witt, Myckatyn, and Marsh, 1998). Boseley and Hartnick (2004) published a study supporting the use of the pediatric voice outcomes survey (PVOS) as a functional outcome assessment to measure success following surgery for VPI. These authors proposed the idea that success of surgery can only be determined by the quality of the patient s communication performance in everyday situations as compared to their pre-surgery status. Although this survey tool was originally designed to evaluate the progress of children being treated for voice disorders, the questions can be applied to resonance disorders as well. In this study, 12 pediatric patients underwent surgery for treatment of VPI. Treatments included either a sphincterplasty or superior-based pharyngeal flap, depending on the velopharyngeal 20

26 closure pattern exhibited by each individual patient. Parents of the twelve patients completed the PVOS before and after surgical treatment was performed. Results showed a mean preoperative score of 38.3% (mean of normative population is 80.51). Following surgery, mean score of the PVOS increased to 72.3%. While this indicates a satisfaction rate that is lower than the normative population, it is a significant increase from the preoperative mean status. Three patients (25%) from this study required additional surgery for VPI. Although the there were few subjects in this study, results indicate that the PVOS can be a useful resource for determining a positive change in the quality of speech communication. Citing that the most important goal of treatment is to improve the patient s ability to communicate, Boseley and Hartnick (2004) concluded that the PVOS more accurately defines successful treatment when compared to the existing measures of successful outcome (p. 1432). Craniofacial Team Member Roles Outcomes following VPI surgery can vary not only depending upon what types of measures are taken, but also depending upon the team member who recommends the treatment method. Dixon-Wood, Williams, and Seagle (1991) conducted a retrospective study on 100 people who had received surgical treatment for VPI. The purpose of the study was to compare speech outcomes of patients who had received treatment based on the speech-language pathologist s recommendation versus patients who received treatment other than that which was recommended by the speech-language pathologist. Results indicated that when recommendations of the speech-language pathologist were followed, significantly more patients were successfully treated and free of any speech 21

27 disorders associated with VPI than when the recommendations were not or could not be accomplished (p. 288). Results from the study by Dixon-Wood, et al. (1991) highlight the fact that speech-language pathologists on craniofacial teams are the most knowledgeable and qualified team members to recommend appropriate treatment for VPI, since they should understand best how the velopharyngeal mechanisms affect speech production, both in terms of anatomy and physiology. Pannbacker, Lass, and Stout (1990) conducted a survey of speech-language pathologists who were members of ACPA focusing on their opinions about the management of VPI. Results showed that 98.2% of respondents believed that obtained information about lateral pharyngeal wall movement is valuable in surgical decisionmaking. Of these respondents, 80.3% always or frequently refer patients with VPI for instrumental assessment. The survey did not address what the role of the team speechlanguage pathologist was in determining the success of speech outcomes following surgery. In the D Antonio et al. (1993) survey of SLP s on cleft palate/craniofacial teams, 62% of responding teams reported that a nasendoscopy was performed solely by the physician (otolaryngologist or plastic reconstructive surgeon). Speech pathologists on these craniofacial teams were reported to either perform (8%) or be present for (27%) the nasendoscopy only 35% of the time during standard VPI assessments. This conflicts with the ACPA s recommendation that VPI assessments should include in-depth analysis of articulatory performance, aerodynamic measures, videofluoroscopy, nasopharyngoscopy, 22

28 and nasometric studies, all of which should be conducted with the participation of the team speech-language pathologist (pp ). Pannbacker (2004) highlighted the need for better standards when evaluating speech outcomes following surgery to correct VPI. A review of speech pathology standards and qualifications was done with the purpose of identifying what makes a speech-language pathologist qualified to diagnose and treat VPI and identify strategies for reduction of risks involved in delivering speech-language pathology services for people with VPI. Results of her review revealed that there is currently a lack of knowledge and skills among speech-language pathologists regarding the diagnosis and treatment of VPI. It is important to note that Pannbacker based her findings on the experience of speech pathologists in general, and not only those who have specialized in VPI. This study highlighted the need for increased awareness of risks related to assessment and treatment, a better understanding of strategies to decrease risks, and a strong knowledge of acceptable clinical practice. She proposed the idea of creating a mechanism to identify speech-language pathologists who have the education and clinical skills to provide these [VPI] services as well as meet these standards (p. 200). Purpose of the Present Investigation The current literature reveals that many different surgical treatments are being utilized by craniofacial teams to treat VPI. Reported surgical outcomes are variable and inconsistent, due largely to the fact that there is no standard for defining and reporting surgical success. Opinions on appropriate surgical goals, assessment procedures, and outcome measurements seem to differ from team to team. 23

29 The purpose of the present study was to collect data from members of craniofacial teams across the country in an attempt to analyze the differences and commonalities in how they measure speech outcomes of VPI surgery. The ultimate objective is to determine whether or not speech and resonance outcomes following VPI surgery are reliable, and how reliability can be increased. Efficacy data would be easier to gather with a more consistent approach to measuring outcomes following surgery. Hypotheses Based on the information currently present in the literature, the following hypotheses were developed: Preferred Surgical Methods There will be a range of reported most often used surgical methods for treatment of VPI, with pharyngeal flap and sphincter pharyngoplasty being the most commonly performed surgeries, with near equal frequency. Assessment The majority of cleft palate/craniofacial teams will report that assessments include at least an intra-oral examination, nasendoscopy, videofluoroscopy, and some kind of perceptual rating system. It is expected that the rating systems used will vary from team to team, because a standard rating system could not be found in the literature or ACPA documents. Based on the recommendations from the ACPA, teams should also be utilizing nasometry and aerodynamic measures during assessment. Team Member Roles 24

30 The team speech-language pathologists will have the greatest role in determining successful speech outcomes following VPI surgery, because this is clearly outlined in the ACPA documents. Definition of success Definitions of successful surgical results will vary considerably. Some teams will likely consider mild residual hypernasality to be included in the definition of success even though the condition of VPI may not be totally resolved. Other teams will consider mild hyponasality to be included in the definition of surgical success. CHAPTER 3: METHODS A ten-question survey was developed with the collaboration of a craniofacial team speech-language pathologist and plastic surgeon. The survey was also formatted into a website. Survey questions were designed for members of craniofacial teams. Questions related to the following: preferred methods of surgical intervention for VPI; assessment procedures; perceptual rating systems for speech; considerations for successful VPI surgery; opinions on how successful surgery should be determined; roles of craniofacial team members; demographic information; and experience working with VPI. The 10-item questionnaire was distributed to all speech-language pathologists attending the craniofacial short course at the American Speech-Language Hearing Association (ASHA) conference in November Their written responses were entered into the web-based survey by the primary investigator, so that all data could be analyzed together. A link to the survey website was sent by to the ACPA list serve, 25

31 the ASHA Division 5 Orofacial and Myofunctional Disorders list serve, and the clinical craniofacial team leaders as listed in the 2003 ACPA directory. Individuals providing data at the ASHA conference were asked not to complete the survey a second time. CHAPTER 4: RESULTS Ninety-three surveys (13%) were completed within a two-month period. Distributions of raw data for each question on the survey can be found in the figures under each subheading. Demographics Of the respondents, the majority (80%) were speech pathologists, followed by plastic surgeons, otolaryngologists, and maxillofacial surgeons (see Figure 1). Half of the respondents had 16 or more years of experience working with cleft lip and palate. The distribution of years of experience is contained in Figure 2. Figure 1. Occupation distribution for professionals completing the survey. Figure 2. Distribution of the number of years of experience for professionals completing the survey. 26

32 Surgical Preferences Figure 3 contains the distribution of the percent response for preferred surgical techniques used to correct VPI. Although the pharyngeal flap and sphincter pharyngoplasty made up the majority (76%) of most often used procedures to correct VPI, the hypothesis that the use of these two procedures would be equally reported was rejected. Approximately half (55%) of the respondents reported that the pharyngeal flap was the procedure most often used at their centers for correcting VPI. Sphincterplasty was the preferred procedure of 21% of respondents. Other procedures (Furlow Z-plasty, Pharyngeal wall augmentation) comprised a very small percentage of the most often used procedures. Levator sling repositioning was also listed by one responder. Figure 3. Distribution of percent response for preferred surgical techniques used to correct VPI. VPI Assessment Procedures 27

33 Figure 4 contains the distribution of the percent response regarding patients that have a speech pathology evaluation before and after surgery. Almost all respondents (96%) reported that patients receive a speech pathology evaluation both before and after. Figure 4. Distribution of the percent response regarding patients that have a speech pathology evaluation before and after surgery. Figure 5 contains the distribution of the percent response for components of a typical speech evaluation. The majority of respondents reported that a speech evaluation for VPI surgical candidates at their respective centers includes perceptual assessment, intra-oral examination, and nasopharyngoscopy. This was consistent with the hypothesis. Less than half of respondents reported including videofluoroscopy in their VPI evaluations, which was lower than hypothesized. Additionally, less than half of respondents reported including nasometric measures when assessing hypernasality. Only a small percentage of respondents reported including aerodynamic measures. Other tools that a few teams reported using in assessments included articulation evaluation and cephlometric x-ray during phonation and at rest. Figure 5. Distribution of the percent response for components of a typical speech evaluation. 28

34 Figure 6 contains the distribution of the percent response for rating scales used to assess hypernasality and nasal emission. As hypothesized, the perceptual rating scales reported varied across teams. The majority (87%) rate severity of hypernasality on a mild, moderate, severe scale. Other rating scales reported by respondents included: 0-4, 0-7, 1-3, 1-4, 1-5, 1-6, 1-10, mild intermittent, mild pervasive, and marked pervasive, speech naturalness scale, Pittsburgh rating scale, Bzoch (U-F scale), and the Weighted Values of Speech Systems associated with VPI. For severity levels of nasal emission, 67% of respondents reported using both a consistent, inconsistent, variable and a mild, moderate, severe rating scale. Over half (58%) also reported rating nasal emission as present or absent. Other rating scales reported for rating nasal emission included: 0-2, 0-4, 0-7, 1-3, 1-5, 1-6, 10%-60%, Pittsburgh scale, Weighted Values of Speech Symptoms associated with VPI. Figure 6. Distribution of the percent response for rating scales used to assess hypernasality and nasal emission. 29

35 Speech Outcomes Figure 7 contains the distribution of the percent response for definitions of treatment success. The hypothesis that the definition of success would vary from team to team was accepted. The majority of respondents (84%) reported surgical success criteria to include normal (see appendix for definition), and two-thirds of respondents also include acceptable speech to be a criteria of successful surgical outcomes. Almost half of the respondents also included mild hyponasality as a criteria of surgical success, while 31% of respondents reported that their centers consider improved speech to be a sign of success. When asked how they believe success should be defined when comparing results across centers or between procedures, the majority chose normal or acceptable. Only 24% believed mild hyponasality should be used to define success, and 21% believed improved speech should define success. Figure 7. Distribution of the percent response for definitions of treatment success. 30

36 Craniofacial Team Member Roles Figure 8 contains the distribution of the percent response indicating the team individual best qualified to define treatment success. As hypothesized, the overwhelming majority of respondents (96%) believed that success of surgery should be judged by the team speech-language pathologist. Two thirds of respondents thought the patient or family should also judge success, which was much higher than originally expected. Less than half of respondents believed that success should be judged by the treating surgeon. Other persons listed as being included in the judgment of success included: external assessor, external peer review, ENT, unfamiliar listener and classroom teacher, peers, and entire craniofacial team. Figure 8. Distribution of the percent response indicating the team individual best qualified to define treatment success. CHAPTER 5: DISCUSSION 31

37 The results of the present survey indicate a number of discrepancies between the assessment procedures, surgical decisions, methods of reporting speech outcomes, and definitions of surgical success among cleft palate/craniofacial teams throughout the United States at the present time. Comparison of the results from the present study and previous literature will be completed in this chapter. Surgical Preferences The most commonly performed surgery for VPI was the pharyngeal flap, although the literature tended to favor the sphincter pharyngoplasty for correction of VPI, because of its lesser risk of leading to hyponasality and obstructive sleep apnea (de Serres et al., 1999; Riski et al., 1992; Sie et al., 1998; Witt et al., 1998). However, since almost half of the responding teams are currently including mild hyponasality as a criterion of surgical success, the surgical preferences reported in this survey may be reflective of the success that they are reporting in their individual centers. Also, over half of the respondents (56%) reported not including videofluoroscopy as a typical assessment tool, and 16% did not report using nasopharyngoscopy. If teams are not utilizing the necessary instruments for visualization of the velopharyngeal mechanisms, then surgical decisions are likely being made solely based on subjective speech perceptual evaluations or surgeon experience and preference, despite the fact that basing surgical decisions on velopharyngeal closure pattern can often predict higher speech success rates (Seagle et al., 2002; Amour et al., 2005). VPI Assessment Procedures Approximately 84% of respondents indicated that nasopharyngoscopy is currently used as an assessment tool for patients undergoing VPI surgery. This shows an increase 32