Journal of Plastic Surgery and Hand Surgery
ISSN: 2000-656X (Print) 2000-6764 (Online) Journal homepage: http://www.tandfonline.com/loi/iphs20
Subjective outcomes after treatment for velopharyngeal dysfunction Mia Stiernman, Kristina Klintö, Ahmed D. Al Qatani, Björn Schönmeyr & Magnus Becker To cite this article: Mia Stiernman, Kristina Klintö, Ahmed D. Al Qatani, Björn Schönmeyr & Magnus Becker (2015) Subjective outcomes after treatment for velopharyngeal dysfunction, Journal of Plastic Surgery and Hand Surgery, 49:4, 198-203 To link to this article: http://dx.doi.org/10.3109/2000656X.2014.988219
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J Plast Surg Hand Surg, 2015; 49: 198–203 © 2014 Informa Healthcare ISSN: 2000-656X print / 2000-6764 online DOI: 10.3109/2000656X.2014.988219
ORIGINAL ARTICLE
Subjective outcomes after treatment for velopharyngeal dysfunction Mia Stiernman1,2, Kristina Klintö3,4, Ahmed D. Al Qatani1,2, Björn Schönmeyr1,2 & Magnus Becker1,2 Department of Clinical Sciences in Malmö, University of Lund, Sweden, 2Department of Plastic and Reconstructive Surgery, 3Department of Otorhinolaryngology, Skåne University Hospital, Malmö, Sweden and 4Clinical Department of Speech Pathology, Karolinska University Hospital, Stockholm, Sweden
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1
Abstract Background: Velopharyngeal dysfunction (VPD) can have various causes and may be a significant disability for the affected patient. Treatment options include surgery and speech therapy, but the success rates are often inconsistent. Methods: In this study, self-assessment questionnaires were sent out to 222 Swedish patients with VPD. The questionnaire included questions about satisfaction with speech, perceived speech quality, perceived improvement from VPD-surgery, and/or speech therapy. Out of 117 (52.7%) respondents, 114 (51.4%) patients were included in the study. The participants were 7–71 years of age (median = 14 years), diagnosed with cleft palate, neurological/developmental delay, congenital hypernasality, or acquired VPD. All patients had previously undergone videofluoroscopy, and 61.4% had undergone VPD-surgery. Results: Seventy-one per cent of the patients perceived their speech to be normal or slightly deviant, but only 55% were satisfied with their speech. Sixty per cent of the operated on patients felt that the treatment had improved their speech much or very much, 10% thought that they had moderate improvement, and 30% stated that they had no or little improvement. Out of the patients that had received speech therapy, 41% felt that the treatment had improved their speech much or very much, 21% thought that they had moderate improvement, and 33% stated that they had no or little improvement. Conclusion: In conclusion, most patients with VPD in this study who underwent evaluation and treatment felt that surgery and speech therapy had improved their speech, but only about half of them were in the end satisfied with the quality of their speech. Key Words: velopharyngeal dysfunction, speech, velopharyngeal flap-surgery, speech therapy, self-assessment questionnaire, satisfaction
Introduction Velopharyngeal dysfunction (VPD) implies difficulties in closing of the passage between the oral and nasal cavities during speech, using the soft palate (velum) and the pharyngeal walls [1]. VPD may result in varying degrees of hypernasality, audible nasal air leakage, and weak articulation [2-5]. In severe cases the speech may be unintelligible. Of patients treated for cleft lip and palate, ~ 20% suffer from varying degrees of VPD [4]. Other aetiologies of non-cleft VPD are occult submucosal cleft palate (i.e. congenital reduced muscle mass in velum midline), congenital disproportion between the length of velum and the depth of the posterior pharyngeal wall, neurological diseases, and adenoidectomy. In certain syndromes, such as 22q11 deletion syndrome, the VPD may be caused by a combination of neurogenic and structural deviancies [6]. In this study, all patients presenting with VPD from the southern region of Sweden who were examined with videofluoroscopy between the years 2002–2010 were included, regardless of aetiology. Typical speech development of the child and socially acceptable speech is the main goal of treating VPD patients. The most commonly used surgical therapy for VPD is pharyngeal flapsurgery [7,8]. If there are articulation problems (i.e. problems with production of speech sounds), speech therapy may be necessary, both before and after surgery. However, there is no evidence for speech therapy improving VPD if there are no problems related to articulation [1]. It is important to note that
patients operated on for VPD cannot be expected to be 100% cured of their speech impairment [5]. Results of studies on VPD-surgery show high success rates; however, the results are incomparable due to different methods of evaluation [9,10]. For example, videofluoroscopy [7,11], endoscopic examination [2,12,13], nasometry [5,14,15] or perceptual assessment by specialised speech language pathologists [2,5,7,8,11-16] were used for speech evaluation in different studies. In addition, the studies based on perceptual evaluation used different variables of speech qualities and essential information on the methodology of perceptual assessment was lacking in several studies. For example, only two of the studies reported measures on the reliability of the perceptual assessments and if the raters were blinded as to whether the patient had been treated surgically or not [7,16]. Furthermore, in one study the perceptual assessment appears to have been performed live and not from recordings [11]. Outcome after surgery may also be evaluated with quality-oflife (QoL) instruments [4] in which the patient or, in the case of small children, the parents report the patient’s speech function in daily life. To date there are no valid questionnaires for evaluating the QoL related to speech in patients with VPD or cleft lip and palate [10]. The only article, to the authors’ knowledge, where a self-assessment questionnaire has been used to evaluate the effect of treatment on speech in VPD-patients shows an improvement in quality-of-life and speech quality [4]. The use of a questionnaire also appears to be the most appropriate way to measure QoL [4].
Correspondence: Mia Stiernman, MD, Department of Clinical Sciences in Malmö, University of Lund, Plastikkirurgiska kliniken, Skånes Universitetssjukhus, Jan Waldenströms gata 18, plan 2, 205 02 Malmö, Sweden. E-mail: [email protected] (Received 26 March 2014; accepted 12 November 2014)
Outcomes velopharyngeal dysfunction
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0 Isolated cleft palate
UCLP
BCLP
Submucosal Neurological/ developemental CP delay
Congential hypernasality
Aquired
Diagnosis
Figure 1. Numbers of patients with different diagnoses in this study.
In many studies, some patient groups with VPD were excluded (for example: Pierre Robin sequence, non-cleft patients, cleft associated with syndromes, cognitive deficiencies, and neuromotor dysfunctions) [5,12,14,17-19]. As shown in the study by Engström et al. [16], the exclusion of specific patient groups (in this case patients who used obturators prior to surgery) may alter the computed outcome of speech. The regular exclusion of specific patient groups makes it more difficult to understand the benefit of surgery for these patients. To summarise, it is difficult to estimate the success rate of surgery to correct VPD and it is also difficult to determine if there are any sub-groups of patients who are particularly benefitted. In this study, the aim was to evaluate the satisfaction with speech, perceived speech quality, and perceived improvement from the given treatment in patients with VPD. Materials and methods Participants The target group in this study was 222 patients with VPD from the southern region of Sweden, consecutively examined with videofluoroscopy between September 2002 and May 2010. The different aetiologies of VPD were cleft palate, neurological/ developmental delay, congenital hypernasality, and acquired VPD. Both patients who were treated with surgery and patients who were not referred for surgery were included in the study. A total of 142 (63.9%) patients/patient’s parents responded. Of these, the number of patients who chose not to participate in the study was 23 (16.2%). One patient was excluded because she had accidentally received the questionnaire and answered it, even though she was not a part of the target group. Another patient was deceased at the time of the study and could, therefore, not answer the questionnaire. In total, the amount responding the questionnaire was 117 (52.7%). A background check in hospital journals revealed that three patients who
answered that they had not been operated on had indeed been operated on. These were excluded from the statistical tests. Of the remaining 114 patients, 70 (61.4%) had been operated on for VPD and 44 (38.6%) had not (Figure 1). The patients who responded were between 7–71 years of age. The median age was 14 years. Sixty-six (57.9%) were males and 48 (42.1%) were females. The diagnosis composition is shown in Figure 2. Questionnaire The questionnaire contained questions concerning the patient’s background, satisfaction with speech, perceived speech quality, and perceived improvement from treatment, as shown in Appendix. The questionnaires were sent to the patients in September 2013. The author attempted to contact, by telephone, all patients who had not answered within 3 weeks to remind them to answer. Statistics The Mann-Whitney U-test or the Jonckheere-Terpstra test was used for comparing two or more variables. Spearman’s test, twotailed, was used to find correlations between variables. For all statistical analyses, p < 0.05 (two-tailed) was considered to indicate significant differences. All calculations were made in SPSS (Version 22). Results Satisfaction with speech and perceived speech quality One hundred and thirteen patients answered the question How satisfied are you with your speech? Out of these, 18 (15.9%) were very satisfied with their speech, 44 (38.9%) were satisfied, 39 (34.5%) were not completely satisfied, and 12 (10.6%) were not satisfied at all. There was no significant difference in satisfaction with speech between the patients who had or had not been treated with surgery (p = 0.318) (Figure 3).
200 M. Stiernman et al. Consecutive patients investigated for VPD: 222 subjects
Responded: 142 subjects
Participated in study: 114 subjects
23 subjectsdid not want to participate
44 (38.6%) Alternative treatment for VPD
3 subjects were not aware of operation
70 (61.4%) Operated on for VPD
1 subject was not part of the target group
Figure 2. Number of patients who participated in this study.
Out of the 109 patients who rated their own speech quality, 37 (33.9%) perceived their speech quality as normal, 40 (36.7%) perceived it as slightly deviant, 17 (15.6%) perceived it as deviant but understandable, 13 (11.9%) perceived it as deviant and hard to understand, and two (1.8%) perceived it as unintelligible. Patients who had been treated with surgery perceived their speech quality as more normal than patients who had not been treated with surgery (p = 0.003) (Figure 4). Of the patients
who had been operated on, 51.4% perceived their speech as normal or slightly deviant. Of the patients who had not been operated on, 19.3% perceived their speech as normal or slightly deviant. The satisfaction with speech and perceived speech quality correlated significantly with each other. However, none of the variables correlated significantly with the amount of speech therapy after the videofluoroscopy, the amount of speech
Has the patient been operated?
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20.35% 10 14.16% 12.39% 10.62% 6.19%
5.31%
4.42% 0 Not satisfied at all
Not comletely satisfied
Satisfied
Very satisfied
Are you satisfied with your speech today?
Figure 3. Results of patient satisfaction of speech. Patients divided into groups of whether they had been operated on or not.
Outcomes velopharyngeal dysfunction
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Has the patient been operated?
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Count
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10
11.93% 8.26%
7.34%
7.34%
6.42%
5.50% 1.83%
0
0.00% Normal
Slightly deviant
Deviant but understandable
Deviant and hard to understand
Unintelligable
How do you percieve your speech?
Figure 4. Results of patients’ perception of speech. Patients divided into groups of whether they had been operated on or not.
therapy at home, the amount of speech therapy after operation, or the age of the patient (Table I). Patients’ perception of speech improvement from surgery and speech therapy A total of 70 (61.4%) patients had been treated with surgery. Sixty-three patients answered the question Do you think the operation improved your speech? The perceived improvement from surgery was: none = 5 (7.9%), small = 14 (22.2%), moderate = 6 (9.5%), much = 20 (31.7%), very much = 18 (28.6%). One hundred and one patients answered the question Do you think the speech therapy helped you? The perceived improvement from speech therapy was: none = 12 (11.9%), small = 21 (20.8%), moderate = 21 (20.8%), much = 24 (23.8%), very much = 18 (17.8%). Five patients (5.0%) answered that they did not know how much speech therapy had improved their speech. The group “do not know” was not included in the statistical analyses. A greater perceived improvement from surgery or speech therapy correlated with a greater satisfaction with speech and more normal perception of speech. Greater perceived improvement from surgery also correlated with lower age (Table II).
Discussion The studied population was very heterogeneous considering age. This may be a limitation since it is difficult to make conclusions based on the statistics from the entire population. A child’s speech also naturally develops with age. On the other hand, the 7-year-old and the 71-year-old are both actual patients of the same treatment and both need to be studied if treatment is
Table I. p-values and correlation coefficients (r) patient satisfaction, perceived speech quality, and variables considered in this study that did not have a significant correlation. The Spearmans test for nonparametric correlation was used.
Amount of speech therapy after investigation Amount of speech therapy at home Amount of speech therapy after operation Age
Satisfaction p = 0.631 r = 0.052 p = 0.909 r = 0.011 p = 0.620 r = -0.063 p = 0.996 r = 0.000
Perceived speech quality p = 0.397 r = 0.093 p = 0.196 r = 0.128 p = 0.272 r = 0.138 p = 0.934 r = 0.008
202 M. Stiernman et al. Table II. p-values and correlation coefficients between perceived improvement from surgery and speech therapy and other variables considered in this study. The Spearmans test for non-parametric correlation was used.
Satisfaction Perceived speech quality
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Age
Perceived improvement from surgery p = 0.005 r = 0.350 p = 0.009 r = 0.327 p = 0.037 r = 0.263
Perceived improvement from speech therapy p = 0.001 r = 0.321 p = 0.005 r = 0.283 p = 0.176 r = 0.136
to be developed and improved. There was a statistically significant correlation between age and perceived improvement from surgery; younger patients perceived their improvement as greater. Even though the response rate in this study only was 63.9%, there was no significant difference between the studied sample and the entire 222 patients in diagnosis (p = 0.809). Neither was there any significant difference in rate of surgery (p = 0.613), sex (p = 0.688), or age (p = 0.282) between the studied population and the entire sample. As mentioned earlier, there is no evidence for speech therapy improving VPD if there are no problems related to articulation [1]. According to this study, the amount of speech therapy both before and after the operation did not affect the satisfaction with speech or perceived speech quality. As reported in the results, 32.7% of the patients thought that the speech therapy had no or little effect. One reason for the low rate of perceived improvement from speech therapy may be the difficulty for patients to distinguish between problems from VPD and articulatory problems. Patients may have deemed the speech therapy as ineffective due to the fact that it did not improve their VPD, even if it may have improved their articulation. Consequently, the improvement from speech therapy might be greater than reported in this study. Studies on speech therapy of patients with VPD-related speech problems are highly warranted. There was no significant difference in satisfaction with speech between the sub-groups of patients who had been operated on and those who had not. However, there was a significant difference in perceived speech quality, where the operated on patients perceived their speech as more normal. One could assume that satisfaction with speech would be because of a high speech quality, but, if so, the group of patients who were not operated on should have been less satisfied. One interpretation of this information could be that the operation improved the speech quality substantially, but the patients had expected to be 100% cured of their VPD and, therefore, still felt that their speech affected their QoL. Another interpretation would be that the patients selected to the group who were not operated on may have had worse conditions for improvement from the beginning and, even though they were on average on a lower level of speech, they were still satisfied with the result. However, these two assumptions cannot fully be supported in this study, since
no perceptual rating of the patients’ speech quality before and after the eventual operation was performed. This study was based on the subjective outcomes of the patients treated for VPD. An interesting approach for future studies would be to evaluate patients before and after treatment for VPD with a QoL-instrument and, in addition, compare the results to evaluation with perceptual speech assessment. With both measures of subjective satisfaction and outcomes of perceptual evaluation, we would get a more complete picture of improvement from treatment. From that knowledge, a more informed selection of patients to different treatment strategies could be made. A first step toward this goal would be to create a valid QoL-instrument for this patient group. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and the writing of the paper. The procedures followed while conducting this article were in accordance with the ethical standards of the Helsinki Declaration of 1975, as revised in 1983. References [1] Sweeney T. Nasality - assessment and intervention. In Cleft palate speech. Wiley; 2012. [2] Paal S, Reulbach U, Strobel-Schwarthoff K, et al. Evaluation of speech disorders in children with cleft lip and palate. J Orofac Orthop 2005;66:270–8. [3] Schuster T, Rustemeyer J, Bremerich A, et al. Analysis of patients with a cleft of the soft palate with special consideration to the problem of velopharyngeal insufficiency. J Craniomaxillofac Surg 2013;41:245–8. [4] Boseley ME, Hartnick CJ. Assessing the outcome of surgery to correct velopharyngeal insufficiency with the pediatric voice outcomes survey. Int J Pediatr Otorhinolaryngol 2004;68:1429–33. [5] Van Lierde KM, Bonte K, Baudonck N, et al. Speech outcome regarding overall intelligibility, articulation, resonance and voice in Flemish children a year after pharyngeal flap surgery. A pilot study. Folia Phoniatr Logop 2008;60:223–32. [6] Persson C. Background. In Speech and language in patients with an isolated cleft palate and/or 22q11 Deletion Syndrome. Universitetet; 2004. p 14. [7] Liedman-Boshko J, Lohmander A, Persson C, et al. 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 2005;39:22–32. [8] Cable BB, Canady JW, Karnell MP, et al. Pharyngeal flap surgery: long-term outcomes at the University of Iowa. Plast Reconstr Surg 2004;113:475–8. [9] John A, Sell D, Sweeney T, et al. The cleft audit protocol for speech-augmented: a validated and reliable measure for auditing cleft speech. Cleft Palate Craniofac J 2006;43:272–88. [10] Eckstein DA, Wu RL, Akinbiyi T, et al. Measuring quality of life in cleft lip and palate patients: currently available patientreported outcomes measures. Plast Reconstr Surg 2011;128: 518e–26e. [11] Patel KB, Sullivan SR, Murthy AS, et al. Speech outcome after palatal repair in nonsyndromic versus syndromic Robin sequence. Plast Reconstr Surg 2012;130:577e–84e. [12] Hortis-Dzierzbicka M, Radkowska E, Fudalej PS. Speech outcomes in 10-year-old children with complete unilateral cleft lip and palate after one-stage lip and palate repair in the first year of life. J Plast Reconstr Aesthet Surg 2012;65:175–81. [13] Goudy S, Ingraham C, Canady J. Noncleft velopharyngeal insufficiency: etiology and need for surgical treatment. Int J Otolaryngol 2012;2012:296073. [14] de Buys Roessingh AS, Cherpillod J, Trichet-Zbinden C, Hohlfeld J. Speech outcome after cranial-based pharyngeal
Outcomes velopharyngeal dysfunction flap in children born with total cleft, cleft palate, or primary velopharyngeal insufficiency. J Oral Maxillofac Surg 2006;64: 1736–42. [15] Wojcicki P, Wojcicka K. Prospective evaluation of the outcome of velopharyngeal insufficiency therapy after pharyngeal flap, a sphincter pharyngoplasty, a double Z-plasty and simultaneous Orticochea and Furlow operations. J Plast Reconstr Aesthet Surg 2011;64:459–61. [16] Engstrom K, Fritzell B, Johanson B. A study of speech improvement following palatopharyngeal flap surgery. Cleft Palate J 1970;7:419–31.
Supplementary material available online
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Appendix
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[17] Enemark H, Bolund S, Jorgensen I. Evaluation of unilateral cleft lip and palate treatment: long term results. Cleft Palate J 1990; 27:354–61. [18] Van Lierde KM, Dhaeseleer E, Luyten A, et al. Parent and child ratings of satisfaction with speech and facial appearance in Flemish pre-pubescent boys and girls with unilateral cleft lip and palate. Int J Oral Maxillofac Surg 2012;41:192–9. [19] Farzaneh F, Becker M, Peterson AM, Svensson H. Speech results in adult Swedish patients born with bilateral complete cleft lip and palate. Scand J Plast Reconstr Surg Hand Surg 2009;43:207–13.
ISSN: 2000-656X (Print) 2000-6764 (Online) Journal homepage: http://www.tandfonline.com/loi/iphs20
Subjective outcomes after treatment for velopharyngeal dysfunction Mia Stiernman, Kristina Klintö, Ahmed D. Al Qatani, Björn Schönmeyr & Magnus Becker To cite this article: Mia Stiernman, Kristina Klintö, Ahmed D. Al Qatani, Björn Schönmeyr & Magnus Becker (2015) Subjective outcomes after treatment for velopharyngeal dysfunction, Journal of Plastic Surgery and Hand Surgery, 49:4, 198-203 To link to this article: http://dx.doi.org/10.3109/2000656X.2014.988219
View supplementary material
Published online: 26 Jan 2015.
Submit your article to this journal
Article views: 63
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=iphs20 Download by: [University of Birmingham]
Date: 05 November 2015, At: 23:56
J Plast Surg Hand Surg, 2015; 49: 198–203 © 2014 Informa Healthcare ISSN: 2000-656X print / 2000-6764 online DOI: 10.3109/2000656X.2014.988219
ORIGINAL ARTICLE
Subjective outcomes after treatment for velopharyngeal dysfunction Mia Stiernman1,2, Kristina Klintö3,4, Ahmed D. Al Qatani1,2, Björn Schönmeyr1,2 & Magnus Becker1,2 Department of Clinical Sciences in Malmö, University of Lund, Sweden, 2Department of Plastic and Reconstructive Surgery, 3Department of Otorhinolaryngology, Skåne University Hospital, Malmö, Sweden and 4Clinical Department of Speech Pathology, Karolinska University Hospital, Stockholm, Sweden
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1
Abstract Background: Velopharyngeal dysfunction (VPD) can have various causes and may be a significant disability for the affected patient. Treatment options include surgery and speech therapy, but the success rates are often inconsistent. Methods: In this study, self-assessment questionnaires were sent out to 222 Swedish patients with VPD. The questionnaire included questions about satisfaction with speech, perceived speech quality, perceived improvement from VPD-surgery, and/or speech therapy. Out of 117 (52.7%) respondents, 114 (51.4%) patients were included in the study. The participants were 7–71 years of age (median = 14 years), diagnosed with cleft palate, neurological/developmental delay, congenital hypernasality, or acquired VPD. All patients had previously undergone videofluoroscopy, and 61.4% had undergone VPD-surgery. Results: Seventy-one per cent of the patients perceived their speech to be normal or slightly deviant, but only 55% were satisfied with their speech. Sixty per cent of the operated on patients felt that the treatment had improved their speech much or very much, 10% thought that they had moderate improvement, and 30% stated that they had no or little improvement. Out of the patients that had received speech therapy, 41% felt that the treatment had improved their speech much or very much, 21% thought that they had moderate improvement, and 33% stated that they had no or little improvement. Conclusion: In conclusion, most patients with VPD in this study who underwent evaluation and treatment felt that surgery and speech therapy had improved their speech, but only about half of them were in the end satisfied with the quality of their speech. Key Words: velopharyngeal dysfunction, speech, velopharyngeal flap-surgery, speech therapy, self-assessment questionnaire, satisfaction
Introduction Velopharyngeal dysfunction (VPD) implies difficulties in closing of the passage between the oral and nasal cavities during speech, using the soft palate (velum) and the pharyngeal walls [1]. VPD may result in varying degrees of hypernasality, audible nasal air leakage, and weak articulation [2-5]. In severe cases the speech may be unintelligible. Of patients treated for cleft lip and palate, ~ 20% suffer from varying degrees of VPD [4]. Other aetiologies of non-cleft VPD are occult submucosal cleft palate (i.e. congenital reduced muscle mass in velum midline), congenital disproportion between the length of velum and the depth of the posterior pharyngeal wall, neurological diseases, and adenoidectomy. In certain syndromes, such as 22q11 deletion syndrome, the VPD may be caused by a combination of neurogenic and structural deviancies [6]. In this study, all patients presenting with VPD from the southern region of Sweden who were examined with videofluoroscopy between the years 2002–2010 were included, regardless of aetiology. Typical speech development of the child and socially acceptable speech is the main goal of treating VPD patients. The most commonly used surgical therapy for VPD is pharyngeal flapsurgery [7,8]. If there are articulation problems (i.e. problems with production of speech sounds), speech therapy may be necessary, both before and after surgery. However, there is no evidence for speech therapy improving VPD if there are no problems related to articulation [1]. It is important to note that
patients operated on for VPD cannot be expected to be 100% cured of their speech impairment [5]. Results of studies on VPD-surgery show high success rates; however, the results are incomparable due to different methods of evaluation [9,10]. For example, videofluoroscopy [7,11], endoscopic examination [2,12,13], nasometry [5,14,15] or perceptual assessment by specialised speech language pathologists [2,5,7,8,11-16] were used for speech evaluation in different studies. In addition, the studies based on perceptual evaluation used different variables of speech qualities and essential information on the methodology of perceptual assessment was lacking in several studies. For example, only two of the studies reported measures on the reliability of the perceptual assessments and if the raters were blinded as to whether the patient had been treated surgically or not [7,16]. Furthermore, in one study the perceptual assessment appears to have been performed live and not from recordings [11]. Outcome after surgery may also be evaluated with quality-oflife (QoL) instruments [4] in which the patient or, in the case of small children, the parents report the patient’s speech function in daily life. To date there are no valid questionnaires for evaluating the QoL related to speech in patients with VPD or cleft lip and palate [10]. The only article, to the authors’ knowledge, where a self-assessment questionnaire has been used to evaluate the effect of treatment on speech in VPD-patients shows an improvement in quality-of-life and speech quality [4]. The use of a questionnaire also appears to be the most appropriate way to measure QoL [4].
Correspondence: Mia Stiernman, MD, Department of Clinical Sciences in Malmö, University of Lund, Plastikkirurgiska kliniken, Skånes Universitetssjukhus, Jan Waldenströms gata 18, plan 2, 205 02 Malmö, Sweden. E-mail: [email protected] (Received 26 March 2014; accepted 12 November 2014)
Outcomes velopharyngeal dysfunction
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11
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0 Isolated cleft palate
UCLP
BCLP
Submucosal Neurological/ developemental CP delay
Congential hypernasality
Aquired
Diagnosis
Figure 1. Numbers of patients with different diagnoses in this study.
In many studies, some patient groups with VPD were excluded (for example: Pierre Robin sequence, non-cleft patients, cleft associated with syndromes, cognitive deficiencies, and neuromotor dysfunctions) [5,12,14,17-19]. As shown in the study by Engström et al. [16], the exclusion of specific patient groups (in this case patients who used obturators prior to surgery) may alter the computed outcome of speech. The regular exclusion of specific patient groups makes it more difficult to understand the benefit of surgery for these patients. To summarise, it is difficult to estimate the success rate of surgery to correct VPD and it is also difficult to determine if there are any sub-groups of patients who are particularly benefitted. In this study, the aim was to evaluate the satisfaction with speech, perceived speech quality, and perceived improvement from the given treatment in patients with VPD. Materials and methods Participants The target group in this study was 222 patients with VPD from the southern region of Sweden, consecutively examined with videofluoroscopy between September 2002 and May 2010. The different aetiologies of VPD were cleft palate, neurological/ developmental delay, congenital hypernasality, and acquired VPD. Both patients who were treated with surgery and patients who were not referred for surgery were included in the study. A total of 142 (63.9%) patients/patient’s parents responded. Of these, the number of patients who chose not to participate in the study was 23 (16.2%). One patient was excluded because she had accidentally received the questionnaire and answered it, even though she was not a part of the target group. Another patient was deceased at the time of the study and could, therefore, not answer the questionnaire. In total, the amount responding the questionnaire was 117 (52.7%). A background check in hospital journals revealed that three patients who
answered that they had not been operated on had indeed been operated on. These were excluded from the statistical tests. Of the remaining 114 patients, 70 (61.4%) had been operated on for VPD and 44 (38.6%) had not (Figure 1). The patients who responded were between 7–71 years of age. The median age was 14 years. Sixty-six (57.9%) were males and 48 (42.1%) were females. The diagnosis composition is shown in Figure 2. Questionnaire The questionnaire contained questions concerning the patient’s background, satisfaction with speech, perceived speech quality, and perceived improvement from treatment, as shown in Appendix. The questionnaires were sent to the patients in September 2013. The author attempted to contact, by telephone, all patients who had not answered within 3 weeks to remind them to answer. Statistics The Mann-Whitney U-test or the Jonckheere-Terpstra test was used for comparing two or more variables. Spearman’s test, twotailed, was used to find correlations between variables. For all statistical analyses, p < 0.05 (two-tailed) was considered to indicate significant differences. All calculations were made in SPSS (Version 22). Results Satisfaction with speech and perceived speech quality One hundred and thirteen patients answered the question How satisfied are you with your speech? Out of these, 18 (15.9%) were very satisfied with their speech, 44 (38.9%) were satisfied, 39 (34.5%) were not completely satisfied, and 12 (10.6%) were not satisfied at all. There was no significant difference in satisfaction with speech between the patients who had or had not been treated with surgery (p = 0.318) (Figure 3).
200 M. Stiernman et al. Consecutive patients investigated for VPD: 222 subjects
Responded: 142 subjects
Participated in study: 114 subjects
23 subjectsdid not want to participate
44 (38.6%) Alternative treatment for VPD
3 subjects were not aware of operation
70 (61.4%) Operated on for VPD
1 subject was not part of the target group
Figure 2. Number of patients who participated in this study.
Out of the 109 patients who rated their own speech quality, 37 (33.9%) perceived their speech quality as normal, 40 (36.7%) perceived it as slightly deviant, 17 (15.6%) perceived it as deviant but understandable, 13 (11.9%) perceived it as deviant and hard to understand, and two (1.8%) perceived it as unintelligible. Patients who had been treated with surgery perceived their speech quality as more normal than patients who had not been treated with surgery (p = 0.003) (Figure 4). Of the patients
who had been operated on, 51.4% perceived their speech as normal or slightly deviant. Of the patients who had not been operated on, 19.3% perceived their speech as normal or slightly deviant. The satisfaction with speech and perceived speech quality correlated significantly with each other. However, none of the variables correlated significantly with the amount of speech therapy after the videofluoroscopy, the amount of speech
Has the patient been operated?
30
No Yes
20
Count
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1 subject was deceased
26.55%
20.35% 10 14.16% 12.39% 10.62% 6.19%
5.31%
4.42% 0 Not satisfied at all
Not comletely satisfied
Satisfied
Very satisfied
Are you satisfied with your speech today?
Figure 3. Results of patient satisfaction of speech. Patients divided into groups of whether they had been operated on or not.
Outcomes velopharyngeal dysfunction
201
Has the patient been operated?
30
No Yes
Count
20
26.61%
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24.77%
10
11.93% 8.26%
7.34%
7.34%
6.42%
5.50% 1.83%
0
0.00% Normal
Slightly deviant
Deviant but understandable
Deviant and hard to understand
Unintelligable
How do you percieve your speech?
Figure 4. Results of patients’ perception of speech. Patients divided into groups of whether they had been operated on or not.
therapy at home, the amount of speech therapy after operation, or the age of the patient (Table I). Patients’ perception of speech improvement from surgery and speech therapy A total of 70 (61.4%) patients had been treated with surgery. Sixty-three patients answered the question Do you think the operation improved your speech? The perceived improvement from surgery was: none = 5 (7.9%), small = 14 (22.2%), moderate = 6 (9.5%), much = 20 (31.7%), very much = 18 (28.6%). One hundred and one patients answered the question Do you think the speech therapy helped you? The perceived improvement from speech therapy was: none = 12 (11.9%), small = 21 (20.8%), moderate = 21 (20.8%), much = 24 (23.8%), very much = 18 (17.8%). Five patients (5.0%) answered that they did not know how much speech therapy had improved their speech. The group “do not know” was not included in the statistical analyses. A greater perceived improvement from surgery or speech therapy correlated with a greater satisfaction with speech and more normal perception of speech. Greater perceived improvement from surgery also correlated with lower age (Table II).
Discussion The studied population was very heterogeneous considering age. This may be a limitation since it is difficult to make conclusions based on the statistics from the entire population. A child’s speech also naturally develops with age. On the other hand, the 7-year-old and the 71-year-old are both actual patients of the same treatment and both need to be studied if treatment is
Table I. p-values and correlation coefficients (r) patient satisfaction, perceived speech quality, and variables considered in this study that did not have a significant correlation. The Spearmans test for nonparametric correlation was used.
Amount of speech therapy after investigation Amount of speech therapy at home Amount of speech therapy after operation Age
Satisfaction p = 0.631 r = 0.052 p = 0.909 r = 0.011 p = 0.620 r = -0.063 p = 0.996 r = 0.000
Perceived speech quality p = 0.397 r = 0.093 p = 0.196 r = 0.128 p = 0.272 r = 0.138 p = 0.934 r = 0.008
202 M. Stiernman et al. Table II. p-values and correlation coefficients between perceived improvement from surgery and speech therapy and other variables considered in this study. The Spearmans test for non-parametric correlation was used.
Satisfaction Perceived speech quality
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Age
Perceived improvement from surgery p = 0.005 r = 0.350 p = 0.009 r = 0.327 p = 0.037 r = 0.263
Perceived improvement from speech therapy p = 0.001 r = 0.321 p = 0.005 r = 0.283 p = 0.176 r = 0.136
to be developed and improved. There was a statistically significant correlation between age and perceived improvement from surgery; younger patients perceived their improvement as greater. Even though the response rate in this study only was 63.9%, there was no significant difference between the studied sample and the entire 222 patients in diagnosis (p = 0.809). Neither was there any significant difference in rate of surgery (p = 0.613), sex (p = 0.688), or age (p = 0.282) between the studied population and the entire sample. As mentioned earlier, there is no evidence for speech therapy improving VPD if there are no problems related to articulation [1]. According to this study, the amount of speech therapy both before and after the operation did not affect the satisfaction with speech or perceived speech quality. As reported in the results, 32.7% of the patients thought that the speech therapy had no or little effect. One reason for the low rate of perceived improvement from speech therapy may be the difficulty for patients to distinguish between problems from VPD and articulatory problems. Patients may have deemed the speech therapy as ineffective due to the fact that it did not improve their VPD, even if it may have improved their articulation. Consequently, the improvement from speech therapy might be greater than reported in this study. Studies on speech therapy of patients with VPD-related speech problems are highly warranted. There was no significant difference in satisfaction with speech between the sub-groups of patients who had been operated on and those who had not. However, there was a significant difference in perceived speech quality, where the operated on patients perceived their speech as more normal. One could assume that satisfaction with speech would be because of a high speech quality, but, if so, the group of patients who were not operated on should have been less satisfied. One interpretation of this information could be that the operation improved the speech quality substantially, but the patients had expected to be 100% cured of their VPD and, therefore, still felt that their speech affected their QoL. Another interpretation would be that the patients selected to the group who were not operated on may have had worse conditions for improvement from the beginning and, even though they were on average on a lower level of speech, they were still satisfied with the result. However, these two assumptions cannot fully be supported in this study, since
no perceptual rating of the patients’ speech quality before and after the eventual operation was performed. This study was based on the subjective outcomes of the patients treated for VPD. An interesting approach for future studies would be to evaluate patients before and after treatment for VPD with a QoL-instrument and, in addition, compare the results to evaluation with perceptual speech assessment. With both measures of subjective satisfaction and outcomes of perceptual evaluation, we would get a more complete picture of improvement from treatment. From that knowledge, a more informed selection of patients to different treatment strategies could be made. A first step toward this goal would be to create a valid QoL-instrument for this patient group. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and the writing of the paper. The procedures followed while conducting this article were in accordance with the ethical standards of the Helsinki Declaration of 1975, as revised in 1983. References [1] Sweeney T. Nasality - assessment and intervention. In Cleft palate speech. Wiley; 2012. [2] Paal S, Reulbach U, Strobel-Schwarthoff K, et al. Evaluation of speech disorders in children with cleft lip and palate. J Orofac Orthop 2005;66:270–8. [3] Schuster T, Rustemeyer J, Bremerich A, et al. Analysis of patients with a cleft of the soft palate with special consideration to the problem of velopharyngeal insufficiency. J Craniomaxillofac Surg 2013;41:245–8. [4] Boseley ME, Hartnick CJ. Assessing the outcome of surgery to correct velopharyngeal insufficiency with the pediatric voice outcomes survey. Int J Pediatr Otorhinolaryngol 2004;68:1429–33. [5] Van Lierde KM, Bonte K, Baudonck N, et al. Speech outcome regarding overall intelligibility, articulation, resonance and voice in Flemish children a year after pharyngeal flap surgery. A pilot study. Folia Phoniatr Logop 2008;60:223–32. [6] Persson C. Background. In Speech and language in patients with an isolated cleft palate and/or 22q11 Deletion Syndrome. Universitetet; 2004. p 14. [7] Liedman-Boshko J, Lohmander A, Persson C, et al. 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 2005;39:22–32. [8] Cable BB, Canady JW, Karnell MP, et al. Pharyngeal flap surgery: long-term outcomes at the University of Iowa. Plast Reconstr Surg 2004;113:475–8. [9] John A, Sell D, Sweeney T, et al. The cleft audit protocol for speech-augmented: a validated and reliable measure for auditing cleft speech. Cleft Palate Craniofac J 2006;43:272–88. [10] Eckstein DA, Wu RL, Akinbiyi T, et al. Measuring quality of life in cleft lip and palate patients: currently available patientreported outcomes measures. Plast Reconstr Surg 2011;128: 518e–26e. [11] Patel KB, Sullivan SR, Murthy AS, et al. Speech outcome after palatal repair in nonsyndromic versus syndromic Robin sequence. Plast Reconstr Surg 2012;130:577e–84e. [12] Hortis-Dzierzbicka M, Radkowska E, Fudalej PS. Speech outcomes in 10-year-old children with complete unilateral cleft lip and palate after one-stage lip and palate repair in the first year of life. J Plast Reconstr Aesthet Surg 2012;65:175–81. [13] Goudy S, Ingraham C, Canady J. Noncleft velopharyngeal insufficiency: etiology and need for surgical treatment. Int J Otolaryngol 2012;2012:296073. [14] de Buys Roessingh AS, Cherpillod J, Trichet-Zbinden C, Hohlfeld J. Speech outcome after cranial-based pharyngeal
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