Relationships between nasalance scores and nasopharyngeal shapes in cleft palate patients

Journal of Cranio-Maxillofacial Surgery (2008) 36, 11e14 Ó 2007 European Association for Cranio-Maxillofacial Surgery doi:10.1016/j.jcms.2007.07.009, available online at http://www.sciencedirect.com

Relationships between*nasalance scores and nasopharyngeal shapes in cleft palate patients Katsuaki MISHIMA, Assistant Professor, DDS, PhD, Tomohiro YAMADA, Assistant Professor, DDS, PhD, Asuka SUGII, Resident, DDS, Hideto IMURA, Resident, DDS, Toshio SUGAHARA, Professor, DDS, PhD Department of Oral and Maxillofacial Reconstructive Surgery (Chairman: Prof. T. Sugahara), Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, 2-5-1 Shikata-cho, Okayama 700-8525, Japan

Objectives: The aim of the present study is to clarify the relationship between nasalance scores and nasopharyngeal shapes obtained by lateral cephalograms. Patients: Eight patients who underwent a WardilleKilner push-back palatoplasty were included in this study. Perceptual judgment by a speech pathologist indicated that these patients had no hypernasality and no nasal emission at blowing. As normal controls, 33 non-cleft individuals, 4 boys and 10 girls aged 6 years old and 5 boys and 14 girls aged 7 years old, were investigated. Methods: Lateral cephalograms at rest were taken for both groups. For the cleft (palate) patients, lateral cephalograms at phonation /a/ and blowing were analyzed and nasometries were also performed using a kitsutsuki passage. Results and conclusion: There was no significant difference in the velar length, the pharyngeal depth, the ratio of the velar length to the pharyngeal depth and the velar angle between the cleft patients and the non-cleft individuals. Multiple regression analyses indicated that standardized regression coefficients of ratios for the velar length to the pharyngeal depth and the velar ascent at blowing had higher nasalance scores for sentences 1 and 3, which had high coefficients of determination, respectively. Ó 2007 European Association for CranioMaxillofacial Surgery

SUMMARY.

Keywords: cephalogram, cleft palate speech, nasalance score, velopharyngeal shape

function of the nasopharynx. However, there are few reports that discuss the relationship between nasalance scores and nasopharyngeal shapes, excluding descriptions from Haapanen et al. (1996) and Stellzig-Eisenhauer (2001). Thus, the aim of the present study is to clarify the relationship between nasalance scores and nasopharyngeal shapes obtained by lateral cephalograms.

INTRODUCTION When diagnosing cleft palate (CP) speech (patterns), perceptual judgments, aerodynamic examinations, nasopharyngeal fiberscopes, cephalograms and nasometers are clinically performed and their data combined. Since nasometry was developed by Fletcher and Bishop (1970), it has established a firm position as a diagnostic tool (Dalston et al., 1991). Nasometry is a quick, non-invasive and objective procedure (Hirschberg et al., 2006), and is widespread in many countries, in which various languages are spoken. Normative data on nasalance scores are reported in English (Fletcher et al., 1989), in Finnish (Haapanen et al., 1991), in Japanese (Tachimura et al., 2000), in Flemish (Van Lierde et al., 2001), and so on. Many studies have discussed relationships between nasalance scores and perceptual judgments (Dalston et al., 1991; Hardin et al., 1992; Keuning et al., 2002; Hirschberg et al., 2006), aerodynamic measurements (Laine et al., 1988; Dalston et al., 1991; Smith and Guyette, 1996) and nasopharyngeal fiberscopes (Kummer et al., 1992; Karnell and Seaver, 1993; Karnell et al., 2001). Clinically, objective data can be obtained non-invasively and quickly using nasometry, and cephalograms can easily provide two-dimensional information on the shape and

PATIENTS AND METHODS Patients Eight patients who underwent a WardilleKilner pushback palatoplasty by two surgeons were included in this study (cleft group, Table 1). From perceptual judgments by a speech pathologist, these patients were found to have no hypernasality and no nasal emission at blowing. None of the patients had undergone an adenoidectomy or a secondary surgery that may have affected their velopharyngeal function. As normal controls, 33 non-cleft individuals (non-cleft group), 4 boys and 10 girls aged 6 years old and 5 boys and 14 girls aged 7 years old, were included in the analysis. Methods Lateral cephalograms at rest were taken for both groups. The cephalograms of the cleft patients were routinely taken for diagnosis of velopharyngeal

*

This study is supported by a Grant-in-Aid for Scientific Research (B) 18390540 from the Japan Society for the Promotion of Science. 11

12 Journal of Cranio-Maxillofacial Surgery Table 1 e Patient distribution

Table 2 e Comparisons between 6- and 7-year-old subjects in the noncleft group

Pt Type

Gender Age at palatoplasty

Age at examination

1 2 3 4 5 6 7 8

Female Male Male Female Male Female Female Female

6 years and 5 months 6 years and 4 months 5 years and 1 month 6 years and 11 months 6 years and 5 months 4 years and 4 months 4 years and 0 month 6 years and 2 months

l-UCLP l-UCLP l-UCLP l-UCLP r-UCLP r-UCLP CP CP

1 year and 5 months 1 year and 5 months 1 year and 4 months 1 year and 6 months 1 year and 11 months 1 year and 5 months 2 years and 0 month 1 year and 5 months

Adjusted PMPeU (mm) Adjusted PPWePMP (mm) PMPeU/PPWePMP Velar angle ( )

6 years of age (n ¼ 14)

7 years of age (n ¼ 19)

p Value

38.1 ^ 3.0 31.8 ^ 3.8 1.21 ^ 0.14 43.1 ^ 4.6

40.4 ^ 3.8 34.3 ^ 4.9 1.19 ^ 0.15 40.7 ^ 6.1

0.25 0.59 0.54 0.49

Table 3 e Comparisons between cleft and non-cleft groups

N

Adjusted PMPeU (mm) Adjusted PPWePMP (mm) PMPeU/PPWePMP Velar angle ( )

Normal sample (n ¼ 33)

Cleft sample (n ¼ 8)

p Value

39.4 ^ 3.6 33.3 ^ 4.6 1.20 ^ 0.15 41.7 ^ 5.6

43.2 ^ 5.7 32.7 ^ 5.4 1.34 ^ 0.24 40.0 ^ 7.6

0.06 0.69 0.08 0.51

S VPC

PMP

ANS

PPW

Table 4 e Nasalance scores (%) Sentence no.

1

2

3

4

Nasalance score

18.3 ^ 8.3

17.6 ^ 7.0

12.4 ^ 3.5

18.1 ^ 6.9

U

Fig. 1 e Cephalometric landmarks and measurements. N e nasion; S e sella; ANS e tip of the anterior nasal spine; PMP e posterior maxillary point, a foot of a perpendicular line from the pterygomaxillary fissure to the palatal plane; PPW e posterior pharyngeal wall at the intersection with the palatal plane; U e tip of the velum; VPC e midpoint of velar pharyngeal contact; pharyngeal depth: PPWePMP e distance from PPW to PMP; velar length: PMPeU e distance from PMP to U; velar angle: :PPWePMPeU; velar ascent: :UePMPeVPC.

and are illustrated in Fig. 1. According to a method from Nakamura et al. (2003), variables that indicated size, velar length and pharyngeal depth, were adjusted so that the SeN distance was converted to 100 mm. In the non-cleft group, cephalometric measurements from 6- to 7-year-old subjects were compared statistically by means of Student’s t-test. Differences between cephalometric measurements from the cleft group and the non-cleft group including both 6- and 7-year-old subjects were investigated statistically by means of ManneWhitney U-tests. By applying multiple regression analyses, relationships between nasalance scores and cephalometric measurements were investigated after correlations among variables were checked. The statistical package SPSS (ver. 11.0) was used, and differences with p value of less than 0.05 were considered statistically significant. RESULTS

function. In the non-cleft group, cephalograms which were taken for orthodontical diagnosis of skeletal pattern were used. All subjects in the non-cleft group had a profile of skeletal Class I. In addition, for the cleft group, lateral cephalograms at phonation /a/ and blowing were analyzed and nasometry was also applied with a Kay 6400 nasometer (RION Co. Ltd, Tokyo, Japan). Using a kitsutsuki passage, which consisted of Japanese non-nasal consonants and vowels (Tachimura et al., 2000), each sentence was read 3 times, and the mean nasalance score was obtained for each sentence (sentence 1: Kitsutsuki Ga Ki Wo Tsutsuku, sentence 2: Suku Suku Sodatsu, sentence 3: Te Wo Tataku, sentence 4: Te Ga Todoku). Cephalograms were measured with an analyzing software package, WinCeph (ver. 8.0, RISE Co. Ltd, Sendai, Japan). Cephalometric landmarks were defined according to Satoh et al. (2005)

In the non-cleft group, there was no significant difference in the velar length, the pharyngeal depth, the ratio of the velar length to the pharyngeal depth and the velar angle between 6- and 7-year-old subjects (Table 2). There was also no significant difference in the velar length, the pharyngeal depth, the ratio of the velar length to the pharyngeal depth and the velar angle between the cleft group and the non-cleft group that included both 6- and 7year-old subjects (Table 3). The mean nasalance scores for sentences 1, 2, 3 and 4 were 18.3%, 17.6%, 12.4% and 18.1%, respectively (Table 4). Pearson’s correlation coefficients between nasalance scores for sentences 1 and 2 were high (r ¼ 0.79, p ¼ 0.02) (Table 5). Pearson’s correlation coefficients between the pharyngeal depth and the ratio of the velar length to the pharyngeal depth (r ¼ 0.71, p ¼ 0.05), between the velar angle and

Relationships between nasalance scores and nasopharyngeal shapes 13 Table 5 e Pearson’s correlation coefficients Velar angle Velar ascent Velar ascent PMPeU/ PMPeU PPWePMP Nasalance Nasalance Nasalance at phonation /a/ at blowing PPWePMP (adjusted) (adjusted) score 1 score 2 score 3 Velar ascent at phonation /a/ 0.73 0.04* Velar ascent at blowing 0.70 0.06* PMPeU/PPWePMP 0.06 0.88* PMP-U (adjusted) 0.06 0.88* PPWePMP (adjusted) 0.00 0.99* Nasalance score 1 0.49 0.21* Nasalance score 2 0.36 0.38* Nasalance score 3 0.49 0.21* Nasalance score 4 0.26 0.53*

0.90 0.00* 0.26 0.53* 0.51 0.90* 0.32 0.45* 0.17 0.69* 0.41 0.31* 0.65 0.08* 0.04 0.93*

0.29 0.48* 0.02 0.97* 0.40 0.33* 0.17 0.68* 0.45 0.27* 0.69 0.06* 0.12 0.79*

0.51 0.20* 0.71 0.05* 0.75 0.03* 0.38 0.35* 0.28 0.49* 0.23 0.59*

0.23 0.58* 0.25 0.55* 0.03 0.94* 0.40 0.33* 0.41 0.31*

0.57 0.14* 0.36 0.38* 0.00 1.00* 0.59 0.13*

0.79 0.02* 0.54 0.17* 0.15 0.73*

0.57 0.14* 0.16 0.70*

0.45 0.92*

*Represents p values. Table 6 e Multiple regression analyses Nasalance score 1

PMPeU/PPWePMP Velar ascent at blowing R2

Nasalance score 2

Nasalance score 3

Nasalance score 4

Standardized regression coefficient

p Value

Standardized regression coefficient

p Value

Standardized regression coefficient

p Value

Standardized regression coefficient

p Value

0.88 0.43

0.02 0.14

0.56 0.61

0.15 0.13

0.53 0.85

0.08 0.02

0.28 0.20

0.55 0.68

0.73

0.04

0.49

0.19

0.74

0.04

0.09

0.80

the velar ascent at phonation /a/ (r ¼ 0.73, p ¼ 0.04), between the velar angle and the velar ascent blowing (r ¼ 0.70, p ¼ 0.06), and between the velar ascent at phonation /a/ and blowing (r ¼ 0.90, p ¼ 0.00) were also high (Table 5). Therefore, multiple regression analyses were carried out with the mean nasalance score for each sentence as a dependent variable and with the ratio of the velar length to the pharyngeal depth and the velar ascent at blowing as independent variables. Coefficients of determination (R2) in nasalance scores for sentences 1, 2, 3 and 4 were 0.73, 0.49, 0.74, 0.09, respectively (Table 6). In the nasalance scores for sentences 1 and 3, which had high coefficients of determination, the standardized regression coefficients of the ratio of the velar length to the pharyngeal depth and the velar ascent at blowing were high (0.88, p ¼ 0.02 and 0.85, p ¼ 0.02), respectively. DISCUSSION Lateral cephalograms were useful for evaluating velopharyngeal function (Mazaheri et al., 1994). Haapanen et al. (1991) described CP with short nasopharyngeal sagittal depths as having normal resonance, and that CP with long nasopharyngeal sagittal depths had reversely spoke with hypernasality. Later, Haapanen et al. (1996) reported that the correlation between pharyngeal depth and hypernasality and speech outcome is moderate,

but that the correlation between pharyngeal depth and nasalance scores is low. Similarly, the correlations between pharyngeal depths and nasalance scores were low in our present study. Stellzig-Eisenhauer (2001) stressed that the influence of age should be taken into account on cephalometric analyses of velopharyngeal variables. In our study, size parameters were adjusted according to a method reported by Nakamura et al. (2003). As described by Satoh et al. (2005), there is a tendency for the absolute value of the velar angle at rest to increase as one grows. So the influence of age should be taken into account on angle variables as well as distances. On the other hand, the relationship between velar angles at blowing and age is low (Satoh et al., 2005). Since the age of CP patients included in our study ranged from 4 to 6 years of age, we considered that these subjects could be combined into one category. It was suggested that speech outcomes may be influenced by the type and extent of the clefts (Scho¨nweiler et al., 1999). In this study, 6 patients with unilateral cleft lip and palate (UCLP) and 2 patients with hard and soft CP were included into the same cleft group, because none of them had hypernasality and nasal emission at blowing. If patients with different outcomes of speech are compared, the severities of the cleft may become important parameters. Multiple regression analyses indicated that the nasalance score of sentence 1 was higher as the ratio of the

14 Journal of Cranio-Maxillofacial Surgery

velar length to the pharyngeal depth increased, and that the nasalance score of sentence 3 was higher as the velar ascent at blowing increased. Namely, the ratio of the velar length to the pharyngeal depth and the velar ascent at blowing were important variables in determining nasalance scores. Stellzig-Eisenhauer (2001) also investigated the relationship between velopharyngeal variables obtained from lateral cephalograms and nasalance scores using multiple linear regression analyses, and reported that the ratio of the velar length to the pharyngeal depth and the area of the adenoids are included as predictor parameters. In the present study, CP patients who have no hypernasality and no nasal emission at blowing were included and investigated, and many patients had a relatively long velar. Therefore, patients who tended to have greater nasal resonance may be compensated by the smaller pharyngeal depth or by the greater velar ascent. However, the present study is limited by the small sample size and none of the CP patients presented hypernasality. Thus, further studies will help to clarify the relationship between pharyngeal variables and nasalance scores. CONCLUSION By investigating relationships between nasalance scores and cephalometrical measurements, the following results were obtained: There was no significant difference in the velar length, the pharyngeal depth, the ratio of the velar length to the pharyngeal depth and the velar angle between non-cleft individuals and cleft patients who had no hypernasality and no nasal emission at blowing. Multiple regression analyses indicated that the ratio of the velar length to the pharyngeal depth and the velar ascent at blowing were significant independent variables that determine nasalance scores. References Dalston RM, Warren DW, Dalston ET: Use of nasometry as a diagnostic tool for identifying patients with velopharyngeal impairment. Cleft Palate Craniofac J 28: 184e188, 1991 Fletcher SG, Bishop ME: Measurement of nasality with Tonar. Cleft Palate J 7: 610e621, 1970 Fletcher SG, Adams LE, McCutcheon MJ: Cleft palate speech assessment through oral-nasal acoustic measures. In: Bzoch K (ed.), Communicative disorders related to cleft lip and palate. Boston: College Hill Press, pp. 246e257, 1989 Haapanen ML, Helio¨vaara A, Ranta R: Hypernasality and the nasopharyngeal apace: a cephalometric study. J Craniomaxillofac Surg 19: 77e80, 1991 Haapanen ML, Laitinen S, Paaso M, Ranta R: Quality of speech correlated to craniofacial characteristics of cleft palate patients with the Pierre Robin sequence. Folia Phoniatr Logop 48: 215e222, 1996 Hardin MA, Van Demark DR, Morris HL, Payne MM: Correspondence between nasalance scores and listener judgments of hypernasality and hyponasality. Cleft Palate Craniofac J 29: 346e351, 1992 Hirschberg J, Bo´k S, Juha´sz M, Trenovszki Z, Votisky P, Hirschberg A: Adaptation of nasometry to Hungarian language and

experiences with its clinical application. Int J Pediatr Otorhinolaryngol 70: 785e798, 2006 Karnell MP, Seaver EJ: Integrated endoscopic/photodetector evaluation of velopharyngeal function. Cleft Palate Craniofac J 30: 337e342, 1993 Karnell MP, Schultz K, Canady J: Investigation of a pressure-sensitive theory of marginal velopharyngeal inadequacy. Cleft Palate Craniofac J 38: 346e357, 2001 Kummer AW, Curtis C, Wiggs M, Lee L, Strife JL: Comparison of velopharyngeal gap size in patients with hypernasality, hypernasality and nasal emission, or nasal turbulence (rustle) as the primary speech characteristic. Cleft Palate Craniofac J 29: 152e155, 1992 Keuning KHDM, Wieneke GH, van Wijngaarden HA, Dejonckere PH: The correlation between nasalance and a differentiated perceptual rating of speech in Dutch patients with velopharyngeal insufficiency. Cleft Palate Craniofac J 39: 277e284, 2002 Laine T, Warren DW, Dalston RM, Morr KE: Screening of velopharyngeal closure based on nasal airflow rate measurements. Cleft Palate J 25: 220e225, 1988 Mazaheri M, Athanasiou AE, Long Jr RE: Comparison of velopharyngeal growth patterns between cleft lip and/or palate patients requiring or not requiring pharyngeal flap surgery. Cleft Palate Craniofac J 31: 452e460, 1994 Nakamura N, Ogata Y, Sasaguri M, Suzuki A, Kikuta R, Ohishi M: Aerodynamic and cephalometric analyses of velopharyngeal structure and function following re-pushback surgery for secondary correction in cleft palate. Cleft Palate Craniofac J 40: 46e53, 2003 Satoh K, Wada T, Tachimura T, Fukuda J: Velar ascent and morphological factor affecting velopharyngeal function in patients with cleft palate and noncleft controls: a cephalometric study. Int J Oral Maxillofac Surg 34: 122e126, 2005 Smith BE, Guyette TW: Pressure-flow differences in performance during production of the CV syllables /pi/ and /pa/. Cleft Palate Craniofac J 33: 74e75, 1996 Scho¨nweiler R, Lisson JA, Scho¨nweiler B, Eckardt A, Ptok M, Tra¨nkmann J, Hausamen JE: A retrospective study of hearing, speech and language function in children with clefts following palatoplasty and veloplasty procedures at 18e24 months of age. Int J Pediatr Otorhinolaryngol 50: 205e217, 1999 Stellzig-Eisenhauer A: The influence of cephalometric parameters on resonance of speech in cleft lip and palate patients. J Orofac Orthop 62: 202e223, 2001 Tachimura T, Mori C, Hirata S, Wada T: Nasalance score variation in normal adult Japanese speakers of mid-west Japanese Dialect. Cleft Palate Craniofac J 37: 463e467, 2000 Van Lierde KM, Wuyts FL, De Bodt M, Van Cauwenberge P: Nasometric values for normal nasal resonance in the speech of young Flemish adults. Cleft Palate Craniofac J 38: 112e118, 2001

Dr. Katsuaki MISHIMA Oral and Maxillofacial Reconstructive Surgery Graduate School of Medicine Dentistry and Pharmaceutical Sciences Okayama University 2-5-1 Shikata-cho Okayama 700-8525 Japan Tel.: +81 86 235 6697 Fax: +81 86 235 6699 E-mail: [email protected] Paper received 6 March 2007 Accepted 23 July 2007