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Experimental treatment of early stuttering: A preliminary study
Journal of Fluency Disorders 30 (2005) 189–199
Experimental treatment of early stuttering: A preliminary study Marie-Christine J. Franken a,∗ , Carine J. Kielstra-Van der Schalk a , Harrie Boelens b a
ENT Department, Hearing and Speech Center, Erasmus MC-Sophia, Rotterdam, Netherlands b Department of Psychology, Leiden University, Leiden, Netherlands Received 1 December 2004; received in revised form 15 April 2005; accepted 9 May 2005
Abstract This pilot study compared two treatments for stuttering in preschool-age children. Thirty children were randomly assigned to either a Lidcombe Program (LP) treatment or a Demands and Capacities Model (DCM) treatment. Stuttering frequencies and severity ratings were obtained immediately before and after treatment (12 weeks). The stuttering frequencies and severity ratings significantly decreased for both treatment groups. No differences between groups were found. Parents of children in both groups were cooperative in many respects, and there were no differences between them on scales that measured their satisfaction with the two treatments. The findings suggest that randomized controlled trials of LP versus DCM treatments are feasible, and they underline the need for experimental analyses of the two treatments. Educational objectives: The reader will be able to: (1) describe the principles and methods of Lidcombe treatment for early stuttering; (2) delineate principles and methods of Demands and Capacities Model treatment; and (3) summarize results of an investigation that compared these programs’ relative effects in a pilot study. © 2005 Elsevier Inc. All rights reserved. Keywords: Stuttering; Early stuttering; Lidcombe Program treatment; Demands and Capacities Model; Short-term evaluation; Randomized controlled trial
∗
Corresponding author. Tel.: +31 10 463 6073. E-mail address: [email protected] (M.-C.J. Franken).
0094-730X/$ – see front matter © 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.jfludis.2005.05.002
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The appearance of stuttering in a young child confronts parents and speech-language pathologists with a difficult decision. On the one hand, the child’s stuttering may disappear without formal treatment, which could make one inclined to wait and see (phenomenon of natural recovery; Andrews & Harris, 1964; Ryan, 2001; Yairi & Ambrose, 1999). On the other hand, the outcomes of treatment at some later time may be less favorable than those of treatment in early childhood. In later years, longer treatment may be required for success, the speech that results from treatment may sound less natural, and the probability of relapse may be greater (for review and discussion, see Ingham & Riley, 1998; Lincoln & Onslow, 1997; Starkweather, 1997). These less favorable outcomes have been used as arguments in favor of early intervention in stuttering exhibited by preschool age children. Many treatment procedures have been developed for early childhood stuttering (Coppolla & Yairi, 1982; Costello, 1983; Culp, 1984; Gregory, 1995; Martin, Kuhl, & Haroldson, 1972; Onslow, Costa, & Rue, 1990; Reed & Godden, 1977; Riley & Riley, 1984; Rustin, Botteril, & Kelman, 1996; Ryan & Ryan, 1983; Shine, 1984; Starkweather, Gottwald, & Halfond, 1990; St. Louis, Clausell, Thompson, & Rife, 1982; Wakaba, 1983). The most extensively investigated treatment is the Lidcombe Program (LP; Onslow et al., 1990). In LP treatment, parents respond in different ways to fluent and disfluent speech. Fluent speech is followed by praise, or by an acknowledgment of its fluency. Disfluent speech is followed by an attempt to encourage the child to produce a fluent correction, or by an acknowledgment of its disfluency. Children treated this way often show large reductions of stuttering within 12 weeks after the beginning of treatment (Harris, Onslow, Packman, Harrison, & Menzies, 2002; Onslow, Andrews, & Lincoln, 1994; Onslow et al., 1990). Furthermore, follow-up observations one to seven years after treatment have revealed a full recovery of fluency in many cases (Lincoln & Onslow, 1997). The empirical support for LP treatment is impressive, but it consists primarily of a large collection of successful cases. The only experimental evidence for the effectiveness of LP treatment was obtained by Harris et al. (2002). Twenty-three children, aged two to five years, who had been stuttering for at least six months, participated in this study. After a pre-test, the children were assigned to LP treatment or to a no-treatment control group. The treatment lasted for 12 weeks. It was immediately followed by a post-test for both groups. The stuttering frequency (percentage of syllables stuttered) decreased from pre-test to post-test, but more so in the experimental group, indicating that LP treatment is better than no treatment. The Lidcombe therapy program should also be compared with other treatments for early stuttering. Especially interesting would seem to be treatments that have become popular and commonly practiced. In the Netherlands, the current standard practice is based on Starkweather’s Demands and Capacities Model of stuttering (DCM; see Adams, 1990; Gottwald & Starkweather, 1999; Starkweather, 1987, 1997; Starkweather et al., 1990). The key assumption of this model is that stuttering is promoted when demands on fluency exceed the child’s capacities for fluency. Demands and capacities are categorized as motoric, emotional, linguistic, or cognitive. Treatment aims at decreasing demands and increasing capacities in these four domains of development. As with LP, DCM treatment is carried out by the parents. For example, parents speak more slowly and they give their child more time to speak to reduce demands on speech motor behavior. The only evidence to support the effectiveness of DCM treatment has been provided in the form of subjective parental
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ratings obtained after treatment (Gottwald & Starkweather, 1995; Starkweather et al., 1990). The difference in empirical support for LP and DCM treatments suggests that LP treatment might be more effective than DCM treatment. This possibility can be investigated with randomized controlled trials of the two treatments. A trial with sufficient power to detect a difference, however, will be time-consuming and expensive, and it will depend in many ways on the cooperativeness of families. We therefore decided to conduct a pilot study. Four questions about the involvement and perceptions of parents were addressed: (1) Will parents accept random assignment to (LP or DCM) treatment conditions? (2) Will parents collect the data required for the evaluation of the two treatments? (3) Will parents complete the treatments? (4) How do parents rate the acceptability of the two treatments? When parents proved cooperative, some preliminary evidence on the issue of the effectiveness of LP versus DCM treatments would also be obtained. We did a relatively large pilot study (30 children) and examined short-term (12-week) outcomes of LP and DCM treatments.
1. Method 1.1. Participants Children could participate when seven conditions (inclusion criteria) were met. Briefly, the inclusion criteria were: (1) child younger than six years of age; (2) time since onset of stuttering at least six months; (3) severity of stuttering, as rated by both parents and therapist, at least 2 on the scale for stuttering severity developed by Yairi and Ambrose (1992, 1999); (4) stuttering frequency at least 3% syllables stuttered (SS) during free-play at intake; (5) no diagnosed emotional, behavioral, learning, or neurological disorders; (6) both parents in favor of the assigned treatment; (7) parent responsible for treatment fluent in Dutch. Fifty families responded. In 2 of these 50 cases, the child had stuttered for fewer than six months. Seventeen other children had stuttering frequencies below 3% SS at intake, and one couple refused random assignment to treatment conditions. The remaining 30 families were randomly assigned to treatment groups (LP, n = 15; or DCM, n = 15), with equal numbers of boys in each group. Four families (three in the LP group, one in the DCM group) terminated treatment prematurely. The reasons given for this were: difficulties in traveling to the clinic, more pressing treatment of the child’s language problems, complete cessation of stuttering after ventilation tubes had been inserted in the ears, and marital problems between the parents. Three other families (one LP, two DCM) did not collect audiorecordings after the termination of treatment. Consequently, 11 children were retained in the LP group and 12 children in the DCM group. Table 1 shows the mean age, the mean age at onset, the gender ratio, and the number of children with a family history of stuttering for each group. 1.2. Procedure Families were recruited via speech-language pathologists and general practitioners. All registered speech-language pathologists who practiced in the environment of Rotterdam,
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Table 1 Make up of the Lidcombe Program (LP, n = 11) and Demands and Capacities Model (DCM, n = 12) treatment groups Statistic
Age (years;months) Age at onset (years;months) Number of boys Number of children with a family history of stuttering
Treatment LP
DCM
4;3 2;10 9 6
4;2 2;8 8 6
Netherlands, and all general practitioners who had previously referred children to the Erasmus MC-Sophia Speech and Hearing Center, were approached. Most families were found via the speech-language pathologists. All interested parents received a screening questionnaire with questions about the age of the child, the age at onset of stuttering, the family history of stuttering, the medical history of the child, and family circumstances. Families were invited for an assessment if responses in the questionnaire indicated that they met the inclusion criteria. The assessment began with a videorecording of the family during freeplay. The child’s stuttering frequency was determined concurrently. Parents were requested to rate the severity of the child’s stuttering (range 0–7; Yairi & Ambrose, 1992, 1999). The therapist made an independent rating of the child’s stuttering using the same scale. Informed consent was obtained upon admission to the study. One parent became the parent responsible for treatment. This parent received an audio-cassette recorder, and was asked to make three 10-min recordings of natural conversations at home, with a week between successive recordings. The parent and the child returned to the clinic two weeks later, at which time they were randomly assigned to a treatment group, and treatment began. Parents received training in treatment procedures during weekly visits to the clinic. The first author counseled four families (two in each group), and the second author 26 families (13 in each group). The first author had 12 years of experience in DCM treatment, and one year of experience in LP treatment. The second author had no experience in either treatment. She observed treatment sessions conducted by the first author, both before and during the present study, was observed by the first author in the treatment of six families (three in each group of the present study), and conducted the other treatment sessions under the first author’s supervision. The treatment was terminated after 12 weeks, or sooner if certain criteria had been met. Post-treatment data were collected immediately after the termination of treatment. The data included audiorecordings and severity ratings, as had been collected before treatment. In addition, treatment acceptability ratings were obtained. 1.3. Treatments 1.3.1. LP treatment The main intervention was to administer verbal contingencies during conversation with the child. Fluent speech was followed by praise, or by an acknowledgment of its fluency. Disfluent speech was followed by an attempt to induce the child to produce a fluent correction, or by an acknowledgment of its disfluency. This was done by the parent responsible for
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treatment. The parent was instructed to maintain a ratio of at least five to one in the numbers of responses to fluent and disfluent speech. Initially, these responses were to be delivered during daily training sessions lasting approximately 15 min and taking place in the home. Later, they were also to be delivered at other times, and outside of the home. This depended on ratings of the severity of the child’s stuttering. The parent rated stuttering severity on a discrete 10-point scale, with 1 = No stuttering, and 10 = Extremely severe stuttering. The parent gave these ratings every day. Parental responses to speech at other times and outside the home were to be given when severity ratings had dropped to the level of four. The therapist taught the parent how to carry out treatment during weekly sessions in the clinic, according to the following pattern. Initially, the parent and the child played for 10 min. The therapist determined the child’s stuttering frequency during this period. The parent and the therapist then independently rated the child’s stuttering severity during the play period. Next, the therapist and the parent compared their ratings. They discussed the ratings the parent had given at home, and the parent demonstrated the procedures that had been carried out at home. These procedures were evaluated, and revised procedures were adopted if necessary. Finally, the therapist demonstrated the revised procedures, the parent practiced them, and the therapist gave feedback. LP treatment was terminated after 12 weeks, or sooner if (1) the stuttering frequency during parent–child conversation in the clinic was below 1%; (2) stuttering severities at home during the past three weeks had been rated to be one or two; and (3) the parent was confident about how to maintain the procedures at home. The mean number of treatment sessions was 11.5. Treatment lasted fewer than 12 weeks for 2 of the 11 children. 1.3.2. DCM treatment Treatment aimed at decreasing demands for, and increasing capacities in, motoric, emotional, linguistic, or cognitive domains of development. Three procedures were arranged for all children: (1) Daily special interaction time: The responsible parent sat down for a period of 15 min with undivided attention to the child. The aim was to boost the child’s self-confidence during activities that involved talking, quiet games, or reading. (2) Changing parents’ speech rate: The parent talked more slowly, with longer pauses as well as slower speech movements, but maintained a natural intonation. (3) Substituting modeling and self-talk for demand speech: The parent was instructed not to ask the child for speech performance. Instead, the parent talked, and thereby provided a model (see Starkweather et al., 1990, pp. 71–77; Starkweather, 1997, pp. 274–276). Other DCM procedures were taught when deemed relevant, based on information provided by the parent and on observations of parent and child at intake and at the beginning of treatment sessions. They aimed at decreasing emotional demands (i.e., slow down pace of activity in the household, calm situations that can be calmed, avoid excitement, model normal disfluencies), decreasing motoric demands (insert a pause between conversation turns, establish turn-taking rules), decreasing cognitive-linguistic demands (speak in short, simple sentences; do not ask questions that require long, complex answers), and increasing emotional capacities (reduce emotional reactions, talk openly about stuttering). Again, as in LP treatment, the therapist provided the parent with treatment instructions during weekly sessions in the clinic. Each session began with a 10-min play period. The therapist and the parent then evaluated the child’s speech during the play period and during
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the previous week, and they selected a target procedure. This was something the parent could do to decrease demands, or increase capacities, as related to fluency. Finally, the therapist modeled the target procedure, the parent practiced it, and the therapist provided feedback. DCM treatment was terminated after 12 weeks, or sooner if (1) the parent had demonstrated mastery of all DCM treatment procedures in the clinic; (2) the parent was confident about how to maintain the procedures at home; and (3) both parent and therapist detected only minimally abnormal disfluencies in the child’s speech. The mean number of treatment sessions was 11.0. Treatment lasted fewer than 12 weeks for 3 of the 12 children. 1.4. Dependent variables and reliability Dependent variables were based on audiorecordings collected by the parent at home, stuttering severity ratings, and the Bristol Stammering Questionnaire (Hayhow, Enderby, & Kingston, 2000). The audiorecordings were used to derive stuttering frequencies (percentage of syllables stuttered). Part-word repetitions, abnormal-sounding one-syllable word repetitions, silent prolongations, and prolongations of speech sounds were counted as instances of stuttering (see Riley, 1994). Stuttering frequencies were derived before as well as after treatment. Stuttering severity ratings were made on a continuous scale from 0 to 7 (Yairi & Ambrose, 1992, 1999). The values corresponding to the whole numbers on this scale were Normally fluent (0), Borderline, Mild, Mild to moderate, Moderate, Moderate to severe, Severe, and Very severe (7). This rating was done for each child, by parent and therapist, before and after treatment. The Bristol Stammering Questionnaire asks parents for their views about changes in their stuttering child, and about acceptability of treatment. Below, outcomes will be presented for the treatment acceptability items only (five items; discrete 1–5 scale). Each item presented a statement. The statements were: (a) Therapy sessions in the clinic were well structured. (b) This approach expects too much from the parents. (c) This approach is disruptive for the rest of the family. (d) I would recommend this approach to other parents whose young children stammer. (e) I was pleased to be able to help my child myself. The values were Agree (1), Mostly agree (2), Not sure (3), Mostly disagree (4), and Disagree (5). An overall acceptability score was obtained with reverse scoring on items a, d, and e, followed by averaging across the five items. Hayhow et al. (2000) presented the questionnaire to 59 parents whose child had reached the maintenance phase in the LP program (stuttering frequencies at or below 1%). A total of 52 questionnaires were returned. The mean ratings on items (a–e) were 1.2, 4.5, 4.5, 1.2, and 1.1, respectively, and the overall acceptability was 4.7. No psychometric properties have been established for these items. Fifteen audiorecordings were number-coded and presented in a random order to two research assistants, who independently determined stuttering frequencies for each sample. The treatment assigned to the child, the time of data collection (pre or post), and the identity of the child were unknown to the assistants. Agreement scores were obtained by dividing the lower by the higher stuttering frequency (see Ingham & Riley, 1998, for a similar procedure). The mean across the 15 agreement scores was 85.5%.
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Fig. 1. Mean pre- and post-stuttering frequencies and standard deviations for the 11 Lidcombe Program (LP) participants and the 12 Demands and Capacities Model (DCM) participants.
2. Results Fig. 1 shows the stuttering frequencies for the LP and DCM treatments. The data shown are means across 11 (LP) and 12 (DCM) children. For LP treatment, the means decreased from 7.2% (S.D. = 2.0) to 3.7% (S.D. = 2.1). For DCM treatment, the means decreased from 7.9% (S.D. = 7.1) to 3.1% (S.D. = 2.1). A mixed-design ANOVA with one between-subjects factor (Treatment: LP versus DCM) and one within-subjects factor (Time: Pre versus Post) was used to assess effects of treatment on the stuttering frequencies. The ANOVA revealed a significant effect of Time, F (1, 21) = 15.18, p < .01, but no effect of Treatment, and no Treatment × Time interaction (p > .10). Table 2 shows the mean stuttering severity ratings. The severity ratings showed a pattern similar to that of the stuttering frequencies, both for parent ratings and for therapist ratings. Mixed-design ANOVAs revealed effects of Time for the parent, F (1, 21) = 85.50, p < .01, and for the therapist, F (1, 21) = 73.73, p < .01, but no effects that involved Treatment (p > .10). Table 2 Mean stuttering pre- and post-severity ratings for both parents and therapists for the Lidcombe Program (LP) and Demands and Capacities Model (DCM) treatments Measure
Treatment LP
Severity rating Parent Therapist
DCM
Pre
Post
Pre
Post
5.0 3.9
2.3 1.5
4.8 4.1
2.1 1.6
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Table 3 Means for the five treatment acceptability items on the Bristol Stammmering Questionnaire for the Lidcombe Program (LP) and Demands and Capacities Model (DCM) treatments Item
Treatment
structureda
Well Expects too much Disruptive Recommend Pleased to be able to help a b
LP
DCM
1.5b
1.1b 4.0 4.4 1.6 1.2
3.9 4.2 1.9 1.3
See Section 1 for complete statements. 1 = Agree (Minimum); 5 = Disagree (Maximum).
Table 3 shows the ratings of treatment acceptability given by the parents. Both treatments were found to be highly acceptable on all dimensions. The five means were each slightly better for the DCM treatment, but Mann–Whitney U tests revealed no differences between treatments (p > .05). The overall acceptability measures were 4.3 (LP) and 4.5 (DCM), on average.
3. Discussion Four findings are directly relevant to the feasibility of a randomized controlled trial of LP and DCM treatments. First, nearly all parents (30 of 31 cases) accepted random assignment to treatment conditions. Second, most parents (26 of 30 cases) completed the treatment they were assigned to. Third, most parents (23 of 26 cases) continued to collect data after treatment. Finally, both treatments were rated favorably by the parents who completed treatment and continued to collect data. These findings suggest that a randomized controlled trial of the two treatments may indeed be feasible. Reductions in both stuttering frequencies and severity ratings did not differ between treatments and provide no evidence for the superiority of either of the two treatments. Thus, this preliminary comparative study provides no evidence for the superiority of a treatment (LP) that would be new in the Netherlands. Furthermore, pooling the stuttering frequencies found in our study with those found in the study carried out by Harris et al. (2002) provides additional support for this view. The two studies were similar in many ways. Similarities are found in the children studied (preschool age, stuttering present at least six months, stuttering frequencies greater than 3%) and in the timing and intensity of treatment (12-week program, weekly sessions, short-term effects measured). The mean change from pre to post in the LP groups of the two studies was 7.9%–3.6% (means based on 21 children in total; Harris et al.: 10 children; present study: 11 children). This change is similar to that observed in the DCM treatment group of the present study (7.9%–3.1%; means based on 12 children). Therefore, the pooled evidence also suggests that the protocols provided by the LP and DCM programs provide a similar treatment outcome. Future research of the LP and DCM treatments should not only establish their relative effectiveness, but also analyze the two treatments. Analytic work could establish the effec-
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tiveness of the two treatments compared to no treatment, the roles of treatment components, and the importance of choosing a treatment that is appropriate for an individual family. The findings of Harris et al. (2002) indicate that LP treatment is better than no treatment. Is this also true for DCM treatment? This question can be answered in experiments with two (DCM versus no treatment) or three (LP versus DCM versus no treatment) groups. The roles of treatment components also deserve further study. LP and DCM treatment both consist of many components, and it is unknown which components contribute to the treatment effects. It may even be that LP and DCM treatments are equally effective because of procedures common to both treatments (e.g., daily extra time with the undivided attention of a parent who boosts the child’s self-confidence). Finally, very little is known about the importance of choosing a treatment that fits an individual family. Some families might benefit more from LP treatment and others more from DCM treatment. Could it be, for example, that DCM is more suitable than LP when the speech rates of parent and child differ strongly? In empirical work directed at these questions, it will be worthwhile to observe the treatments as they are carried out in the homes. In conclusion, the findings of the present study suggest that randomized controlled trials of LP versus DCM treatments are feasible, and they underline the need for experimental analyses of the two treatments.
Acknowledgments This study was supported by the Revolving Fund of the Erasmus MC-Sophia. Additional funding was received from ‘Vereniging Trustfonds Erasmus Universiteit Rotterdam’ and ‘Stichting Bevordering van Volkskracht’. The authors would like to thank Theo Stijnen for his statistical contributions, Michael Brocaar for his technical assistance, and David Heyne for his comments on a previous version of the manuscript. CONTINUING EDUCATION Experimental treatment of early stuttering: A preliminary study QUESTIONS 1. The essence of Lidcombe Program (LP) treatment is: a. children overcorrect stuttered words b. demands on fluency are reduced, and capacities for fluency are increased c. severity ratings are used to monitor progress d. parents respond differentially to fluent and disfluent speech 2. The key assumption of the Demands and Capacities Model (DCM) is: a. stuttering is promoted if demands on fluency exceed the child’s capacities for fluency b. experiences that boost self-esteem will decrease stuttering c. stuttering will develop if parents speak faster than their child
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d. fluency will be promoted if parents substitute modeling and self-talk for demand speech 3. The study reported above was designed to: a. compare LP and DCM treatments b. determine whether parents would accept random assignment to LP and DCM treatments c. assess acceptability of LP and DCM treatments d. assess the feasibility of a randomized controlled trial of LP versus DCM treatments 4. The study reported above revealed: a. an effect of time (Pre versus Post) on stuttering frequency b. an effect of treatment (LP versus DCM) on stuttering frequency c. a Time x Treatment interaction in stuttering frequency d. all of the above 5. The conclusion of the study reported above is: a. randomized controlled trials of LP versus DCM treatments are feasible b. experimental analyses of the two treatments are needed c. both a. and b. d. neither a. nor b.
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Onslow, M., Andrews, C., & Lincoln, M. (1994). A control/experimental trial of an operant treatment for early stuttering. Journal of Speech and Hearing Research, 37, 1244–1259. Onslow, M., Costa, L., & Rue, S. (1990). Direct early intervention with stuttering: Some preliminary data. Journal of Speech and Hearing Disorders, 55, 405–416. Reed, C. G., & Godden, A. L. (1977). An experimental treatment using verbal punishment with two preschool stutterers. Journal of Fluency Disorders, 2, 225–233. Riley, G. (1994). Stuttering severity instrument for children and adults (3rd ed.). Austin, TX: Pro-Ed. Riley, G. D., & Riley, J. (1984). A component model for treating stuttering in children. In M. Peins (Ed.), Contemporary approaches in stuttering therapy (pp. 36–69). Boston: Little, Brown. Rustin, L., Botterill, L., & Kelman, E. (1996). Assessment and therapy for young dysfluent children: Family interaction. London: Whurr Publishers Ltd. Ryan, B. P. (2001). A longitudinal study of articulation, language, rate, and fluency of 22 preschool children who stutter. Journal of Fluency Disorders, 26, 107–127. Ryan, B. P., & Ryan, B. (1983). Programmed stuttering therapy for children: Comparison of four establishment programs. Journal of Fluency Disorders, 8, 291–321. Shine, R. E. (1984). Assessment and fluency training with the young stutterer. In M. Peins (Ed.), Contemporary approaches in stuttering therapy (pp. 173–216). Boston: Little, Brown. Starkweather, C. W. (1987). Fluency and stuttering. Englewood Cliffs, NJ: Prentice-Hall. Starkweather, C. W. (1997). Therapy for younger children. In R. F. Curlee & G. M. Siegel (Eds.), Nature and treatment of stuttering (2nd ed., pp. 257–279). Boston: Allyn and Bacon. Starkweather, C. W., Gottwald, S. R., & Halfond, M. M. (1990). Stuttering prevention: A clinical method. Englewood Cliffs, NJ: Prentice-Hall. St. Louis, K. O., Clausell, P. L., Thompson, J. N., & Rife, C. C. (1982). Preliminary investigation of EMG biofeedback induced relaxation with a preschool aged stutterer. Perceptual and Motor Skills, 55, 195–199. Wakaba, Y. Y. (1983). Group play therapy for Japanese children who stutter. Journal of Fluency Disorders, 8, 93–118. Yairi, E., & Ambrose, N. (1992). A longitudinal study of stuttering in children: A preliminary report. Journal of Speech and Hearing Research, 36, 521–528. Yairi, E., & Ambrose, N. (1999). Early childhood stuttering I: Persistency and recovery rates. Journal of Speech Language and Hearing Research, 42, 1097–1112. Marie-Christine J. Franken (1960) graduated in General Linguistics and Logopedics. In 1997, she obtained her Ph.D. degree at Nijmegen University. At the ErasmusMC-Sophia in Rotterdam she combines clinical work in stuttering with applied research in stuttering and linguistics. Carine J. Kielstra-van der Schalk (1972) is a developmental psychologist and a physical therapist specializing in children (Child PT). Before she started this research project, she worked as a Child PT in the Amsterdam Medical Center. Harrie Boelens (1956) obtained his Ph.D. degree at Utrecht University in 1988. He is lecturer in developmental psychology at Leiden University since 1987. His research concerns the roles of operant conditioning in children’s language and speech. This has led to publications on basic as well applied issues in children’s learning.
Experimental treatment of early stuttering: A preliminary study Marie-Christine J. Franken a,∗ , Carine J. Kielstra-Van der Schalk a , Harrie Boelens b a
ENT Department, Hearing and Speech Center, Erasmus MC-Sophia, Rotterdam, Netherlands b Department of Psychology, Leiden University, Leiden, Netherlands Received 1 December 2004; received in revised form 15 April 2005; accepted 9 May 2005
Abstract This pilot study compared two treatments for stuttering in preschool-age children. Thirty children were randomly assigned to either a Lidcombe Program (LP) treatment or a Demands and Capacities Model (DCM) treatment. Stuttering frequencies and severity ratings were obtained immediately before and after treatment (12 weeks). The stuttering frequencies and severity ratings significantly decreased for both treatment groups. No differences between groups were found. Parents of children in both groups were cooperative in many respects, and there were no differences between them on scales that measured their satisfaction with the two treatments. The findings suggest that randomized controlled trials of LP versus DCM treatments are feasible, and they underline the need for experimental analyses of the two treatments. Educational objectives: The reader will be able to: (1) describe the principles and methods of Lidcombe treatment for early stuttering; (2) delineate principles and methods of Demands and Capacities Model treatment; and (3) summarize results of an investigation that compared these programs’ relative effects in a pilot study. © 2005 Elsevier Inc. All rights reserved. Keywords: Stuttering; Early stuttering; Lidcombe Program treatment; Demands and Capacities Model; Short-term evaluation; Randomized controlled trial
∗
Corresponding author. Tel.: +31 10 463 6073. E-mail address: [email protected] (M.-C.J. Franken).
0094-730X/$ – see front matter © 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.jfludis.2005.05.002
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The appearance of stuttering in a young child confronts parents and speech-language pathologists with a difficult decision. On the one hand, the child’s stuttering may disappear without formal treatment, which could make one inclined to wait and see (phenomenon of natural recovery; Andrews & Harris, 1964; Ryan, 2001; Yairi & Ambrose, 1999). On the other hand, the outcomes of treatment at some later time may be less favorable than those of treatment in early childhood. In later years, longer treatment may be required for success, the speech that results from treatment may sound less natural, and the probability of relapse may be greater (for review and discussion, see Ingham & Riley, 1998; Lincoln & Onslow, 1997; Starkweather, 1997). These less favorable outcomes have been used as arguments in favor of early intervention in stuttering exhibited by preschool age children. Many treatment procedures have been developed for early childhood stuttering (Coppolla & Yairi, 1982; Costello, 1983; Culp, 1984; Gregory, 1995; Martin, Kuhl, & Haroldson, 1972; Onslow, Costa, & Rue, 1990; Reed & Godden, 1977; Riley & Riley, 1984; Rustin, Botteril, & Kelman, 1996; Ryan & Ryan, 1983; Shine, 1984; Starkweather, Gottwald, & Halfond, 1990; St. Louis, Clausell, Thompson, & Rife, 1982; Wakaba, 1983). The most extensively investigated treatment is the Lidcombe Program (LP; Onslow et al., 1990). In LP treatment, parents respond in different ways to fluent and disfluent speech. Fluent speech is followed by praise, or by an acknowledgment of its fluency. Disfluent speech is followed by an attempt to encourage the child to produce a fluent correction, or by an acknowledgment of its disfluency. Children treated this way often show large reductions of stuttering within 12 weeks after the beginning of treatment (Harris, Onslow, Packman, Harrison, & Menzies, 2002; Onslow, Andrews, & Lincoln, 1994; Onslow et al., 1990). Furthermore, follow-up observations one to seven years after treatment have revealed a full recovery of fluency in many cases (Lincoln & Onslow, 1997). The empirical support for LP treatment is impressive, but it consists primarily of a large collection of successful cases. The only experimental evidence for the effectiveness of LP treatment was obtained by Harris et al. (2002). Twenty-three children, aged two to five years, who had been stuttering for at least six months, participated in this study. After a pre-test, the children were assigned to LP treatment or to a no-treatment control group. The treatment lasted for 12 weeks. It was immediately followed by a post-test for both groups. The stuttering frequency (percentage of syllables stuttered) decreased from pre-test to post-test, but more so in the experimental group, indicating that LP treatment is better than no treatment. The Lidcombe therapy program should also be compared with other treatments for early stuttering. Especially interesting would seem to be treatments that have become popular and commonly practiced. In the Netherlands, the current standard practice is based on Starkweather’s Demands and Capacities Model of stuttering (DCM; see Adams, 1990; Gottwald & Starkweather, 1999; Starkweather, 1987, 1997; Starkweather et al., 1990). The key assumption of this model is that stuttering is promoted when demands on fluency exceed the child’s capacities for fluency. Demands and capacities are categorized as motoric, emotional, linguistic, or cognitive. Treatment aims at decreasing demands and increasing capacities in these four domains of development. As with LP, DCM treatment is carried out by the parents. For example, parents speak more slowly and they give their child more time to speak to reduce demands on speech motor behavior. The only evidence to support the effectiveness of DCM treatment has been provided in the form of subjective parental
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ratings obtained after treatment (Gottwald & Starkweather, 1995; Starkweather et al., 1990). The difference in empirical support for LP and DCM treatments suggests that LP treatment might be more effective than DCM treatment. This possibility can be investigated with randomized controlled trials of the two treatments. A trial with sufficient power to detect a difference, however, will be time-consuming and expensive, and it will depend in many ways on the cooperativeness of families. We therefore decided to conduct a pilot study. Four questions about the involvement and perceptions of parents were addressed: (1) Will parents accept random assignment to (LP or DCM) treatment conditions? (2) Will parents collect the data required for the evaluation of the two treatments? (3) Will parents complete the treatments? (4) How do parents rate the acceptability of the two treatments? When parents proved cooperative, some preliminary evidence on the issue of the effectiveness of LP versus DCM treatments would also be obtained. We did a relatively large pilot study (30 children) and examined short-term (12-week) outcomes of LP and DCM treatments.
1. Method 1.1. Participants Children could participate when seven conditions (inclusion criteria) were met. Briefly, the inclusion criteria were: (1) child younger than six years of age; (2) time since onset of stuttering at least six months; (3) severity of stuttering, as rated by both parents and therapist, at least 2 on the scale for stuttering severity developed by Yairi and Ambrose (1992, 1999); (4) stuttering frequency at least 3% syllables stuttered (SS) during free-play at intake; (5) no diagnosed emotional, behavioral, learning, or neurological disorders; (6) both parents in favor of the assigned treatment; (7) parent responsible for treatment fluent in Dutch. Fifty families responded. In 2 of these 50 cases, the child had stuttered for fewer than six months. Seventeen other children had stuttering frequencies below 3% SS at intake, and one couple refused random assignment to treatment conditions. The remaining 30 families were randomly assigned to treatment groups (LP, n = 15; or DCM, n = 15), with equal numbers of boys in each group. Four families (three in the LP group, one in the DCM group) terminated treatment prematurely. The reasons given for this were: difficulties in traveling to the clinic, more pressing treatment of the child’s language problems, complete cessation of stuttering after ventilation tubes had been inserted in the ears, and marital problems between the parents. Three other families (one LP, two DCM) did not collect audiorecordings after the termination of treatment. Consequently, 11 children were retained in the LP group and 12 children in the DCM group. Table 1 shows the mean age, the mean age at onset, the gender ratio, and the number of children with a family history of stuttering for each group. 1.2. Procedure Families were recruited via speech-language pathologists and general practitioners. All registered speech-language pathologists who practiced in the environment of Rotterdam,
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Table 1 Make up of the Lidcombe Program (LP, n = 11) and Demands and Capacities Model (DCM, n = 12) treatment groups Statistic
Age (years;months) Age at onset (years;months) Number of boys Number of children with a family history of stuttering
Treatment LP
DCM
4;3 2;10 9 6
4;2 2;8 8 6
Netherlands, and all general practitioners who had previously referred children to the Erasmus MC-Sophia Speech and Hearing Center, were approached. Most families were found via the speech-language pathologists. All interested parents received a screening questionnaire with questions about the age of the child, the age at onset of stuttering, the family history of stuttering, the medical history of the child, and family circumstances. Families were invited for an assessment if responses in the questionnaire indicated that they met the inclusion criteria. The assessment began with a videorecording of the family during freeplay. The child’s stuttering frequency was determined concurrently. Parents were requested to rate the severity of the child’s stuttering (range 0–7; Yairi & Ambrose, 1992, 1999). The therapist made an independent rating of the child’s stuttering using the same scale. Informed consent was obtained upon admission to the study. One parent became the parent responsible for treatment. This parent received an audio-cassette recorder, and was asked to make three 10-min recordings of natural conversations at home, with a week between successive recordings. The parent and the child returned to the clinic two weeks later, at which time they were randomly assigned to a treatment group, and treatment began. Parents received training in treatment procedures during weekly visits to the clinic. The first author counseled four families (two in each group), and the second author 26 families (13 in each group). The first author had 12 years of experience in DCM treatment, and one year of experience in LP treatment. The second author had no experience in either treatment. She observed treatment sessions conducted by the first author, both before and during the present study, was observed by the first author in the treatment of six families (three in each group of the present study), and conducted the other treatment sessions under the first author’s supervision. The treatment was terminated after 12 weeks, or sooner if certain criteria had been met. Post-treatment data were collected immediately after the termination of treatment. The data included audiorecordings and severity ratings, as had been collected before treatment. In addition, treatment acceptability ratings were obtained. 1.3. Treatments 1.3.1. LP treatment The main intervention was to administer verbal contingencies during conversation with the child. Fluent speech was followed by praise, or by an acknowledgment of its fluency. Disfluent speech was followed by an attempt to induce the child to produce a fluent correction, or by an acknowledgment of its disfluency. This was done by the parent responsible for
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treatment. The parent was instructed to maintain a ratio of at least five to one in the numbers of responses to fluent and disfluent speech. Initially, these responses were to be delivered during daily training sessions lasting approximately 15 min and taking place in the home. Later, they were also to be delivered at other times, and outside of the home. This depended on ratings of the severity of the child’s stuttering. The parent rated stuttering severity on a discrete 10-point scale, with 1 = No stuttering, and 10 = Extremely severe stuttering. The parent gave these ratings every day. Parental responses to speech at other times and outside the home were to be given when severity ratings had dropped to the level of four. The therapist taught the parent how to carry out treatment during weekly sessions in the clinic, according to the following pattern. Initially, the parent and the child played for 10 min. The therapist determined the child’s stuttering frequency during this period. The parent and the therapist then independently rated the child’s stuttering severity during the play period. Next, the therapist and the parent compared their ratings. They discussed the ratings the parent had given at home, and the parent demonstrated the procedures that had been carried out at home. These procedures were evaluated, and revised procedures were adopted if necessary. Finally, the therapist demonstrated the revised procedures, the parent practiced them, and the therapist gave feedback. LP treatment was terminated after 12 weeks, or sooner if (1) the stuttering frequency during parent–child conversation in the clinic was below 1%; (2) stuttering severities at home during the past three weeks had been rated to be one or two; and (3) the parent was confident about how to maintain the procedures at home. The mean number of treatment sessions was 11.5. Treatment lasted fewer than 12 weeks for 2 of the 11 children. 1.3.2. DCM treatment Treatment aimed at decreasing demands for, and increasing capacities in, motoric, emotional, linguistic, or cognitive domains of development. Three procedures were arranged for all children: (1) Daily special interaction time: The responsible parent sat down for a period of 15 min with undivided attention to the child. The aim was to boost the child’s self-confidence during activities that involved talking, quiet games, or reading. (2) Changing parents’ speech rate: The parent talked more slowly, with longer pauses as well as slower speech movements, but maintained a natural intonation. (3) Substituting modeling and self-talk for demand speech: The parent was instructed not to ask the child for speech performance. Instead, the parent talked, and thereby provided a model (see Starkweather et al., 1990, pp. 71–77; Starkweather, 1997, pp. 274–276). Other DCM procedures were taught when deemed relevant, based on information provided by the parent and on observations of parent and child at intake and at the beginning of treatment sessions. They aimed at decreasing emotional demands (i.e., slow down pace of activity in the household, calm situations that can be calmed, avoid excitement, model normal disfluencies), decreasing motoric demands (insert a pause between conversation turns, establish turn-taking rules), decreasing cognitive-linguistic demands (speak in short, simple sentences; do not ask questions that require long, complex answers), and increasing emotional capacities (reduce emotional reactions, talk openly about stuttering). Again, as in LP treatment, the therapist provided the parent with treatment instructions during weekly sessions in the clinic. Each session began with a 10-min play period. The therapist and the parent then evaluated the child’s speech during the play period and during
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the previous week, and they selected a target procedure. This was something the parent could do to decrease demands, or increase capacities, as related to fluency. Finally, the therapist modeled the target procedure, the parent practiced it, and the therapist provided feedback. DCM treatment was terminated after 12 weeks, or sooner if (1) the parent had demonstrated mastery of all DCM treatment procedures in the clinic; (2) the parent was confident about how to maintain the procedures at home; and (3) both parent and therapist detected only minimally abnormal disfluencies in the child’s speech. The mean number of treatment sessions was 11.0. Treatment lasted fewer than 12 weeks for 3 of the 12 children. 1.4. Dependent variables and reliability Dependent variables were based on audiorecordings collected by the parent at home, stuttering severity ratings, and the Bristol Stammering Questionnaire (Hayhow, Enderby, & Kingston, 2000). The audiorecordings were used to derive stuttering frequencies (percentage of syllables stuttered). Part-word repetitions, abnormal-sounding one-syllable word repetitions, silent prolongations, and prolongations of speech sounds were counted as instances of stuttering (see Riley, 1994). Stuttering frequencies were derived before as well as after treatment. Stuttering severity ratings were made on a continuous scale from 0 to 7 (Yairi & Ambrose, 1992, 1999). The values corresponding to the whole numbers on this scale were Normally fluent (0), Borderline, Mild, Mild to moderate, Moderate, Moderate to severe, Severe, and Very severe (7). This rating was done for each child, by parent and therapist, before and after treatment. The Bristol Stammering Questionnaire asks parents for their views about changes in their stuttering child, and about acceptability of treatment. Below, outcomes will be presented for the treatment acceptability items only (five items; discrete 1–5 scale). Each item presented a statement. The statements were: (a) Therapy sessions in the clinic were well structured. (b) This approach expects too much from the parents. (c) This approach is disruptive for the rest of the family. (d) I would recommend this approach to other parents whose young children stammer. (e) I was pleased to be able to help my child myself. The values were Agree (1), Mostly agree (2), Not sure (3), Mostly disagree (4), and Disagree (5). An overall acceptability score was obtained with reverse scoring on items a, d, and e, followed by averaging across the five items. Hayhow et al. (2000) presented the questionnaire to 59 parents whose child had reached the maintenance phase in the LP program (stuttering frequencies at or below 1%). A total of 52 questionnaires were returned. The mean ratings on items (a–e) were 1.2, 4.5, 4.5, 1.2, and 1.1, respectively, and the overall acceptability was 4.7. No psychometric properties have been established for these items. Fifteen audiorecordings were number-coded and presented in a random order to two research assistants, who independently determined stuttering frequencies for each sample. The treatment assigned to the child, the time of data collection (pre or post), and the identity of the child were unknown to the assistants. Agreement scores were obtained by dividing the lower by the higher stuttering frequency (see Ingham & Riley, 1998, for a similar procedure). The mean across the 15 agreement scores was 85.5%.
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Fig. 1. Mean pre- and post-stuttering frequencies and standard deviations for the 11 Lidcombe Program (LP) participants and the 12 Demands and Capacities Model (DCM) participants.
2. Results Fig. 1 shows the stuttering frequencies for the LP and DCM treatments. The data shown are means across 11 (LP) and 12 (DCM) children. For LP treatment, the means decreased from 7.2% (S.D. = 2.0) to 3.7% (S.D. = 2.1). For DCM treatment, the means decreased from 7.9% (S.D. = 7.1) to 3.1% (S.D. = 2.1). A mixed-design ANOVA with one between-subjects factor (Treatment: LP versus DCM) and one within-subjects factor (Time: Pre versus Post) was used to assess effects of treatment on the stuttering frequencies. The ANOVA revealed a significant effect of Time, F (1, 21) = 15.18, p < .01, but no effect of Treatment, and no Treatment × Time interaction (p > .10). Table 2 shows the mean stuttering severity ratings. The severity ratings showed a pattern similar to that of the stuttering frequencies, both for parent ratings and for therapist ratings. Mixed-design ANOVAs revealed effects of Time for the parent, F (1, 21) = 85.50, p < .01, and for the therapist, F (1, 21) = 73.73, p < .01, but no effects that involved Treatment (p > .10). Table 2 Mean stuttering pre- and post-severity ratings for both parents and therapists for the Lidcombe Program (LP) and Demands and Capacities Model (DCM) treatments Measure
Treatment LP
Severity rating Parent Therapist
DCM
Pre
Post
Pre
Post
5.0 3.9
2.3 1.5
4.8 4.1
2.1 1.6
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Table 3 Means for the five treatment acceptability items on the Bristol Stammmering Questionnaire for the Lidcombe Program (LP) and Demands and Capacities Model (DCM) treatments Item
Treatment
structureda
Well Expects too much Disruptive Recommend Pleased to be able to help a b
LP
DCM
1.5b
1.1b 4.0 4.4 1.6 1.2
3.9 4.2 1.9 1.3
See Section 1 for complete statements. 1 = Agree (Minimum); 5 = Disagree (Maximum).
Table 3 shows the ratings of treatment acceptability given by the parents. Both treatments were found to be highly acceptable on all dimensions. The five means were each slightly better for the DCM treatment, but Mann–Whitney U tests revealed no differences between treatments (p > .05). The overall acceptability measures were 4.3 (LP) and 4.5 (DCM), on average.
3. Discussion Four findings are directly relevant to the feasibility of a randomized controlled trial of LP and DCM treatments. First, nearly all parents (30 of 31 cases) accepted random assignment to treatment conditions. Second, most parents (26 of 30 cases) completed the treatment they were assigned to. Third, most parents (23 of 26 cases) continued to collect data after treatment. Finally, both treatments were rated favorably by the parents who completed treatment and continued to collect data. These findings suggest that a randomized controlled trial of the two treatments may indeed be feasible. Reductions in both stuttering frequencies and severity ratings did not differ between treatments and provide no evidence for the superiority of either of the two treatments. Thus, this preliminary comparative study provides no evidence for the superiority of a treatment (LP) that would be new in the Netherlands. Furthermore, pooling the stuttering frequencies found in our study with those found in the study carried out by Harris et al. (2002) provides additional support for this view. The two studies were similar in many ways. Similarities are found in the children studied (preschool age, stuttering present at least six months, stuttering frequencies greater than 3%) and in the timing and intensity of treatment (12-week program, weekly sessions, short-term effects measured). The mean change from pre to post in the LP groups of the two studies was 7.9%–3.6% (means based on 21 children in total; Harris et al.: 10 children; present study: 11 children). This change is similar to that observed in the DCM treatment group of the present study (7.9%–3.1%; means based on 12 children). Therefore, the pooled evidence also suggests that the protocols provided by the LP and DCM programs provide a similar treatment outcome. Future research of the LP and DCM treatments should not only establish their relative effectiveness, but also analyze the two treatments. Analytic work could establish the effec-
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tiveness of the two treatments compared to no treatment, the roles of treatment components, and the importance of choosing a treatment that is appropriate for an individual family. The findings of Harris et al. (2002) indicate that LP treatment is better than no treatment. Is this also true for DCM treatment? This question can be answered in experiments with two (DCM versus no treatment) or three (LP versus DCM versus no treatment) groups. The roles of treatment components also deserve further study. LP and DCM treatment both consist of many components, and it is unknown which components contribute to the treatment effects. It may even be that LP and DCM treatments are equally effective because of procedures common to both treatments (e.g., daily extra time with the undivided attention of a parent who boosts the child’s self-confidence). Finally, very little is known about the importance of choosing a treatment that fits an individual family. Some families might benefit more from LP treatment and others more from DCM treatment. Could it be, for example, that DCM is more suitable than LP when the speech rates of parent and child differ strongly? In empirical work directed at these questions, it will be worthwhile to observe the treatments as they are carried out in the homes. In conclusion, the findings of the present study suggest that randomized controlled trials of LP versus DCM treatments are feasible, and they underline the need for experimental analyses of the two treatments.
Acknowledgments This study was supported by the Revolving Fund of the Erasmus MC-Sophia. Additional funding was received from ‘Vereniging Trustfonds Erasmus Universiteit Rotterdam’ and ‘Stichting Bevordering van Volkskracht’. The authors would like to thank Theo Stijnen for his statistical contributions, Michael Brocaar for his technical assistance, and David Heyne for his comments on a previous version of the manuscript. CONTINUING EDUCATION Experimental treatment of early stuttering: A preliminary study QUESTIONS 1. The essence of Lidcombe Program (LP) treatment is: a. children overcorrect stuttered words b. demands on fluency are reduced, and capacities for fluency are increased c. severity ratings are used to monitor progress d. parents respond differentially to fluent and disfluent speech 2. The key assumption of the Demands and Capacities Model (DCM) is: a. stuttering is promoted if demands on fluency exceed the child’s capacities for fluency b. experiences that boost self-esteem will decrease stuttering c. stuttering will develop if parents speak faster than their child
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d. fluency will be promoted if parents substitute modeling and self-talk for demand speech 3. The study reported above was designed to: a. compare LP and DCM treatments b. determine whether parents would accept random assignment to LP and DCM treatments c. assess acceptability of LP and DCM treatments d. assess the feasibility of a randomized controlled trial of LP versus DCM treatments 4. The study reported above revealed: a. an effect of time (Pre versus Post) on stuttering frequency b. an effect of treatment (LP versus DCM) on stuttering frequency c. a Time x Treatment interaction in stuttering frequency d. all of the above 5. The conclusion of the study reported above is: a. randomized controlled trials of LP versus DCM treatments are feasible b. experimental analyses of the two treatments are needed c. both a. and b. d. neither a. nor b.
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