Incongruous Movements of the Velum and Lateral Pharyngeal Walls

ROBERT J. SHPRINTZEN, Ph.D. SAUL J. RAKOFF, M.D. M. LEON SKOLNICK, M.D. ALFRED S. LAVORATO, Ph.D. Bronx, New York 10467

Five patients evaluated via multi-view videofluoroscopy were found to have incongruous movements between the velum and lateral aspects of the pharyngeal walls. All five patients had velopharyngeal insufficiency resulting from either absent lateral pharyngeal wall motion in the presence of velar mobility or absent velar mobility in the presence of lateral pharyngeal wall motion. The data indicates that these valving patterns are not rare phenomena of velopharyngeal valving. Treatment by pharyngeal flap or other methods for velopharyngeal insufficiency must be suited to these peculiar valving patterns based on adequate diagnostic information.

The physiology of velopharyngeal closure has been debated and questioned on numerous occasions and as is indicated by Dickson, et al. (1974): "Opinions regarding the specific muscles responsible for these patterns of movement are almost as numerous as authors who discuss them (p. 477)." A multiplicity of explanations have been offered in discussing the posterosuperior movement of the velum and medial movements of the lateral pharyngeal walls during speech. There is general agreement that the levator muscle is responsible for observed velar mobility (Dickson, et al., 1974), but the muscles contributing to movements of the pharyngeal walls remain a point of contention. The purpose of this paper is to present some case reports of subjects who showed unusual patterns of velopharyngeal valving which raise questions concerning the physiology of velopharyngeal closure and methods for treating velopharyngeal insufficiency. It must be emphasized that the multi-view videofluoroscopic data presented below do not answer any existing questions but may aid in constructing further hy- potheses for future investigations. It has been well established that medial motion of the lateral pharyngeal walls is essential to velopharyngeal closure (Calnan, 1953; Harring-

Robert J. Shprintzen, Ph.D., is Co-ordinator and Deputy Director of the Center for Craniofacial Disorders at Montefiore Hospital and Medical Center, Bronx, N.Y., Adjunct Associate Professor of Speech Pathology at C. W. Post Center of Long Island University and at Kean College of New Jersey. Alfred S. Lavorato, Ph.D., is Supervisor of the Division of Speech Pathology in the Department of Audiology, Eye and Ear Hospital of Pittsburgh, and Research Associate at the University of Pittsburgh Cleft Palate Center. Saul J. Rakoff, M.D., is Associate Attending, Department of Radiology at Montefiore Hospital and Medical Center and Associate Professor of Radiology at the Albert Einstein College of Medicine. M. Leon Skolnick, M.D., is Associate Professor of Radiology at the University of Pittsburgh School of Medicine. 148 Shprintzen et al., inconcruvous movements 149 ton, 1945; Skolnick, 1969; Takahasi, 1962). Medial motion of the pharyngeal walls has been variously attributed to the salpingopharyngeus, levator, and superior constrictor muscles. While the salpingopharyngeus has been described as a contributor to lateral pharyngeal wall movement (Bloomer, 1953; Harrington, 1944; Zemlin, 1968), more re- cent findings tend to indicate that the salpingopharyngeus is only incon- sistently present in man and typically not well defined nor large enough to account for such motion (Dickson and Dickson, 1972; Dickson, et al., 1974; McMyn, 1940; Strong, 1949). It has been hypothesized that the levator is the sole muscle of velopharyngeal closure, producing both velar and pharyngeal wall motion components (Dickson, 1972; Dickson and Dickson, 1972). Other reported data from multi-view fluoroscopic analyses (Shprintzen, et al., 1974; Shprintzen, et al., 1975b), electro- myography (Fritzell, 1969), and ultra-sonic studies (Ewanowski, et al., 1974; Zagzebski, 1975) would seem to indicate that there is a separate muscular component involved in lateral pharyngeal wall movement, most likely the superior constrictor. The present study was designed to evaluate the relationship between palatal and pharyngeal wall movements.

Method |_ _- -

SuBjEctTs. The subjects selected were five patients evaluated at the Center for Craniofacial Disorders of Montefiore Hospital and Medical Center. Each patient exhibited velopharyngeal incompetence (not nec- essarily cleft palate) and was chosen because he demonstrated what was considered to be an atypical pattern of velopharyngeal valving. The subjects included: (A) a four-year-old female, (B) a five-year-old male, (C) a five-year-old female, (D) a thirty-one-year-old male, and (E) a thirty-three-year-old male. ExPERIMENTAL PrRocEDURE. The VldCOﬂUOI‘OSCOplC procedure utilized to examine the subjects has been reported in detail elsewhere (Shprintzen, et al., 1975b; Skolnick, 1969). All subjects were examined in lateral, frontal, base, and left and right oblique projections. The subjects repeated a series of speech tasks designed to elicit speech in varying phonemic contexts and engaged in non-speech activities as well. The tasks used were: , ma-ma-ma-ma-ma ’ Kitty-cat pa-pa-pa-pa-pa Give Kate the cake ta-ta-ta-ta-ta Gerry's slippers ka-ka-ka-ka-ka Stop the bus ssssss (sustained /s/ phoneme) Catch a fish Suzie | One, two,. .. ., ten (counting to ten) Suzie sees Sally Blowing Popeye ' Whistling Popeye plays baseball Swallowing

The total time under fluoroscopy was typically between two and three minutes with less than two rads irradiation. Resulting video images were recorded on a one-inch video tape recorder (Sony EV 210) and were 150 Cleft Palate Journal, April 1977, Vol. 14 No. 2 reviewed in full speed, slow motion, and stopped frames (60 visual fields per second) from a 14-inch TV monitor (Conrac) w1th a line scan rate of 945.

Results SumBjEct A. Subject A was referred to the center at the age of four years, eight months, because of poor speech development secondary to a complete bilateral cleft lip and palate. She had had a one-stage lip repair at one month of age and a repair of the soft palate at 18 months of age at another institution. She had an unrepaired cleft of the hard palate which had never been obturated prosthetically. Her speech was characterized by severe hypernasal resonance, audible nasal air flow, and numerous articulatory omissions, distortions, and substitions. Much of her articulation was characterized by glottal stops. Language, however, was age appropriate. Multi-view videofluoroscopic examination showed fair mobility of the velum as observed in lateral view. However, no contact was made between the velum and either the posterior pharyngeal wall or a small adenoid mass in the nasopharynx. No movement was observed in the posterior pharyngeal wall. The velum formed a velar eminence during speech, but a consistent two mm gap was observed between the velum and adenoid mass. Frontal view showed no observa- ble medial movement in the lateral pharyngeal walls. Base and left and right oblique views confirmed that the velum was moving but that the lateral pharyngeal walls were not (Figure 1). SumBpEct B. This five-year-old male subject was referred to the center at the age of four years, one month, because of a life-long history of hypernasal resonance and poor articulation in the absence of an overt or submucous cleft palate. Speech was severely hypernasal and articulation was characterized by numerous articulatory omissions, substitutions,

REST / AND SPEECH

LATERAL VIEW FRONTAL VIEW FIGURE 1. Videofluoroscopic tracings for Subject A showing velar motion in lateral view during speech but absence of lateral wall motion in frontal view. Shprintzen et al., INCONGRUOUS MOVEMENTS 151

and distortions, and glottal stops. Language skills were slightly de- pressed. Significant factors in his history included congenital hypothy- roidism and possible neurological impairment. (Neurological examination was within normal limits, but psychometric evaluation revealed some specific learning disabilities.) Both gross and fine motor coordina- tion were within normal limits. Videofluoroscopic examination revealed that the velum was within normal limits in thickness and length, but there was no velar motion observed during speech in lateral view. No motion was observed in the posterior pharyngeal wall in lateral view , but in frontal view, excellent movement was observed in the lateral aspects of the pharyngeal walls as described by Shprintzen et al., (1975b) with a sharp shelf-like pattern at the level of the hard palate (Flgure 2). C. This five-year-old female child was referred to the center at the age of four years, ten months, by a neurologist because of a life- long history of hypernasal resonance in the absence of an overt or submucous cleft palate. Her speech was characterized by severe hypernasal resonance and numerous articulatory distortions and substitutions. Language skills were significantly disordered but had improved over the previous year. History was remarkable in that, at birth; follow- ing a full-term pregnancy, there was no sucking reflex until five days of age. All developmental landmarks were somewhat delayed, but by the age of four, she had no apparent deficits. All neurological examinations were negative and intelligence was within normal limits. Multi-view videofluoroscopic examination revealed a palate and nasopharynx of normal proportions. Lateral view showed some limited velar mobility in a posterosuperior direction. Velar contact was not made with a moder- ate sized adenoid mass nor with the posterior pharyngeal wall. In frontal

LATEML VIE FRONTAL VIEW FIGURE 2. Vldeoﬂuoroscoplc tracings for Subject B showmg absence of velar motlon during speech in lateral view but good lateral wall movement in frontal view. 152 Cleft Palate Journal, April 1977, Vol. 14 No. 2 view, there was no demonstrable lateral pharyngeal wall motion, an observation which was confirmed in base and oblique views (Figure 3). - SuBpECT D. This thirty-one-year-old male was referred to the center at the age of thirty years, nine months, by a prosthodontist. He was born with a complete left sided unilateral cleft lip and palate which were unrepaired until his lip and hard palate were surgically treated at six and seven years respectively. The soft palate was not repaired. His speech was characterized by severe hypernasal resonance with audible nasal air flow and several articulatory distortions and substitutions with frequent glottal stops. Lateral view showed no movement in the fairly substantial lateral segments of the velum or in the posterior pharyngeal wall. Frontal and oblique views showed excellent movement of the lateral pharyngeal walls, which was confirmed by the base view. There was a gross incompetence resulting from the velar cleft. While the lack of movement of the velar tags for this subject may be partially related to the fact that the soft palate was unrepaired, we have seen many patients with unrepaired palates with good movement in the lateral velar segments (Figure 4). SUBJECT E. This thirty-three year-old male was self-referred to the center at the age of thirty-two because of his desire to remediate his hypernasal speech. He had been born with a complete right unilateral cleft lip and palate. His lip had been repaired at six months of age, the soft palate at four years of age, and the hard palate at ten years of age. His speech was severely hypernasal, though articulation was essentially within normal limits except for nasal distortion of pressure consonants. Multi-view videofluoroscopy showed a velum of normal length and thickness but with no movement evident during speech in lateral view. Frontal, oblique and base views did show good movement in the lateral

Tee

SPEECH

LATERAL VIEW - FRONTAL VIEW _ FIGURE 3. Videoﬂuoroscopic tracings for Subject C showing velar motion in lateral view during speech but absence of lateral wall motion in frontal view. _ Shprintzen et al., inconcrvous movemEnts 153 pharyngeal walls, but velopharyngeal insufficiency was evident because the velar component of closure was not present (Figure 5). °

Discussion

Multi-view videofluoroscopic examinations of each of the subjects demonstrated generally good movement in one component of velopharyngeal closure but none in the other. Three of the patients (Subjects B, D, and E) exhibited no velar movement but good medial motion of the

REST AND SPEECH

LATERAL VIEW FRONTAL VIEW FIGURE 4. Vedeofluoroscopic tracings for Subject D showing absence of motion in velar segments in lateral view but good lateral wall motion in frontal view. es

REST AND SPEECH

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| LATERAL Vien FRONTAL View FIGURE 5. Videofluoroscopic tracings for Subject E showing absence of velar motion in lateral view but lateral wall motion in frontal view. 154 Cleft Palate Journal, April 1977, Vol. 14 No. 2 _ lateral pharyngeal walls. The other two subjects had velar mobility but no lateral pharyngeal wall motion. All five subjects had significant velopharyngeal insufficiencies. The question arises as to why these particular individuals display incongruities of motion between the velum and pharyngeal walls. In his EMG studies of normals, Fritzell (1969) found a close synchronization between the potentials recorded from the levator and superior constrictor. If velar motion is a result of levator activity and lateral wall motion a result of superior constrictor activity, (Shprintzen, et al., 1974; Shprintzen, et al., 1975b; Zagzebski, 1975), then the lack of motion in one component or the other for these subjects may be explained on the basis of possible neuromuscular difficulties (Subjects B and C), post-surgical complications (Subjects A and E.), anatomical anomalies (Subject D), a combination of these factors, or possible failures in function because of faulty learning. Another theory of normal velopharyngeal closure postulates that only one muscle-levator-is responsible for generating both velar and lateral wall components of such closure. Based upon this theory, the presence of motion in the lateral walls of the pharynx even though velar motion is absent in individuals such as subjects B, D and E is difficult to explain without concomittant accounting of the attachments and inner- vation of relevant musculature. In recognition of the limited data presented here and of the limita- tions of generalizing about normal mechanisms from pathological ones, no single physiological theory regarding the mechanism of velopharyngeal closure is being advocated. For example, with respect to the generalization about normal mechanisms from pathological ones, it is recognized that the level of activity and shape of the lateral walls in any of the subjects presented here might differ from a normal individual by virtue of the abberant nature of the subject's mechanism. Thus, it might be that, if both levator and superior constrictor are normally active, features of the lateral pharyngeal walls might differ in pathological cases if only one or the other of these muscles were active. The observations of this study are not meant to support any single physiological model simply because the data presented are not complete enough to do so. While it might be suggested that the observed phenomena described in these five subjects point to a two-muscle model, it is possible that another explanation is more appropriate. Firstly, it must be pointed out that the data here do not tell us what these subjects should have, but rather what they do not have. Secondly, what has become evident to these investigators is the importance of not being misled into thinking that all individuals, normal or pathological, valve the same way. It should be strongly emphasized that we may be in error by searching for one physiological model to explain velopharyngeal closure. Skolnick, et al., (1973) described multiple patterns of valving both in individuals with velopharyngeal competence and in those with incompetence. Ana- tomical variability in the velopharyngeal apparatus (Dickson, 1972) is Shprintzen et al., inconcruvous movements 155 probably enough to contribute to the multiple patterns of valving observed by Skolnick et al., (1973) and by these investigators in a series of over 20 normal subjects, but physiological variation undoubtedly ac- counts for most of the observed differences in velopharyngeal closure. Thus, some individuals may valve with levator plus the superior-most fibers of superior constrictor and some primarily with levator. In addi- ton, the muscles contributing to valving probably vary among individuals in the percentage of the valving effort they perform. In other words, variability in the physiology of valving may be the rule, and observing small samples of individuals with a particular anatomical and physiological pattern may have misled us into believing that the entire population operaties in a singular fashion. > An important factor to note as a result of this study is the importance of multi-view videofluoroscopy in the evaluation of the velopharyngeal sphincter and recommendations for treatment of velopharyngeal insufficiency. The unusual patterns of velopharyngeal valving employed by the five subjects would not have been discovered unless at least lateral and frontal projections had been utilized to evaluate their mechanism.. Moreover, these subjects probably represent a small but significant portion of individuals with velopharyngeal incompetence. For example, these five subjects represent approximately 5% of the new patients with diagnosed velopharyngeal incompetence undergoing multi-view videofluoroscopic examination in a one-year period (July 1, 1974 through July 1, 1975) at Montefiore's Center for Craniofacial Disorders. If the unusual valving patterns in these subjects had not been adequately diagnosed, possible errors in treatment could have resulted for this 5 per cent of the new case load. For example, if lateral views alone had been used for Subjects A and C, it would not have been known that the lateral pharyngeal walls were motionless. In constructing a pharyngeal flap to eliminate hypernasality , if the flap were not of the type which would obstruct the velopharyngeal portal, then the hypernasality would most probably persist (Kelsey, et al., 1972). However, the observation of good velar motion in lateral view might not prompt a recommendation for an obstructing type of pharyngeal flap. Or, it is possible that a push-back procedure, rather than a pharyngeal flap, could have been recommended because of the observed velar mobility. However with no lateral pharyngeal wall motion, this, too would probably fail. Lateral-view observations for Subjects B, D, and E who had no velar motion might lead to a recommendation for a broad obstructing pharyngeal flap which might lead to hyponasality. Since these subjects did show good lateral wall motion, obstruction of the portal was not necessary. Therefore, in these cases, multidimensional analysis becomes particularly important for accuracy of treatment plan- ning. Since it has been suggested that variability in valving may be the rule and not the exception and since there does not appear to be any way to predict how any particular individual will valve, multiview videofluo- 156 Cleft Palate Journal, April 1977, Vol. 14 No. 2

roscopy with an adequate barium coat throughout the nasopharynx (Lavornto, 1975) would appear to be quite important to assure an accurate diagnosis of the mechanism.

Summary Five subjects with incongruous movements of the velum and pharyngeal walls were examined via mutli-view videofluoroscopy. Each had velopharyngeal insufficiency related to lack of movement in one of the two components of closure of the sphincter. These observed phenomena raise questions concerning the physiology of velopharyngeal valving which need to be investigated more fully in the future. In addition, the need for multi-view video- or cinefluoroscopy is clearly demonstrated by these valving patterns. Acknowledgements: Appreciation to Deborah Shprintzen and Muriel Schwartz for assistance in the preparation of the manuscript. reprints: Robert J. Shprintzen, Ph.D. Center for Craniofacial Disorders Montefiore Hospital and Medical Center 111 E. 210th St. Bronx, N.Y. 10467

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