Multifactorial Treatment for Preschool Children

The PCI approach rests on the research interpretation that physiological and linguistic factors may be significant in the onset and development of stuttering and that their interaction with emotional and environmental aspects contributes to the severity and persistence of the disorder (Kelman et al., 2005). The Yaruss et al. (2006) description of Family Focused Treatment describes children’s capacity for fluent speech as a bucket of water. The analogy shows that certain factors add water to the bucket and if the child’s bucket becomes too full, the water spills out and the child stutters. Factors causing this could include (1) aspects of the child’s overall development, such as perfectionism, high degree of sensitivity and genetic predisposition to stutter; (2) interpersonal stressors such as major changes or traumatic events in the child’s life, marital conflicts, unrealistic developmental demands being placed on the child or fast-paced lifestyle and (3) communicative stressors that can increase the child’s sense of time pressure, such as listener negative response to stuttering, frequent interruptions, rapid rate of conversation and competition for talking time. This analogy is supplemented with the explanation that the greater the child’s intrinsic motor and linguistic skill, the greater the bucket size.

Because children differ in the combination of factors that may influence the onset and persistence of stuttering (Rustin et al., 1996), both treatments emphasise the necessity for an individual approach. Also, as mentioned previously, both approaches contend that manipulating aspects of the environment increases the likelihood that the child will be able to speak more fluently. So, the parent-focused aspects of both treatments seek to recognise and change those behaviours that may influence the child’s stuttering. These may include reducing parent speech rate, increasing response time latency, reducing linguistic complexity, commenting rather than asking questions and following the child’s lead in play.

Both treatments incorporate reduced parent speech rate, with associated slower conversational turn-taking patterns. Meyers and Freeman (1985) reported that parents of stuttering children spoke faster than control mothers to stuttering and control preschoolers. Zebrowski et al. (1996) reported some effect on stuttering when parents reduced speech rate, but the effect was by no means consistent. Yaruss and Conture (1995) similarly reported that the relation between parent speech rate and stuttering of preschoolers was not straightforward. In short, there is some evidence that reduced speech rate and altering turn-taking patterns will facilitate fluency, but their mechanisms are poorly understood (Bernstein Ratner, 2004). By consciously slowing speech rate, parents also change behaviours such as making longer turn-taking latencies (Bernstein Ratner, 1992), which, in turn, have been linked to reductions in stuttering frequency (Newman and Smit, 1989).

Demonstrated value

Yaruss et al. (2006) conducted a file audit of the Family Focused Treatment and evaluated preschoolers stuttering before and after the treatment and at follow-up, and gave parents a questionnaire. For the 17 children involved, there were significant reductions in the children’s stuttering frequency in the clinical setting after the treatment. The mean stuttering frequency before treatment, in terms of stutters per 100 words, was 16.4% (SD = 6.6%) and at post-treatment was 3.2% (SD = 2.0%). Results of parents’ ratings of treatment helpfulness and satisfaction showed that most parents were pleased with the components of the parent–child training programme and judged parent education about stuttering to be the most valuable. Their ratings of the children’s speech showed that children spoke significantly more fluently following treatment at home and in new speaking situations.

Treatment outcomes and long-term follow-up indicate that 11 of the 17 children (64.7%) exhibited sufficient improvements in stuttering following the parent–child training programme and were dismissed from stuttering therapy entirely. Another six children (35%) continued to stutter and were enrolled in child-focused treatment, after which they were dismissed from treatment.

Millard et al. (2008) reported data from six young children who stutter who were involved in PCI. Stuttering severity was based on stuttering rate measured with percent syllables stuttered (%SS), duration of three longest stutters and the degree of tension and secondary behaviour present. This generated a score on a scale from 0 (normal speech) to 7 (very severe stuttering) (Yairi and Ambrose, 1992). The results of this Phase I clinical trial show that four of six children significantly reduced the frequency of their stuttering. Three of those four reduced stuttering severity to zero. One child reduced stuttering only with his father and continued with the therapy while the remaining child made significant progress when a direct approach was introduced.

Another study (Millard et al., 2009) investigating the efficacy of PCI showed results for 10 children. The study involved a Baseline Phase, followed by 6 weeks of clinic-based therapy and then 6 weeks of home-based therapy, then a Follow-up Phase. Six of the ten children received therapy and the other four did not. Millard et al. (2009) did not present the customary absolute measures of stuttering frequency, but analysed them with a cusum analysis. Results showed that there was a reduction in the trend of stuttering for four of the six children who received therapy. Three of four children remaining on the waiting list showed no systematic changes in stuttering, and the remaining one demonstrated a significant reduction, which the authors explained as spontaneous recovery. Parent ratings of the impact of stuttering on themselves and their confidence in managing the stuttering indicated improvements after the treatment.

Franken et al. (2005) compared the 12 weeks of Lidcombe Program (LP) treatment with 12 weeks of a treatment based on the Demands and Capacities model. The latter treatment was described as involving traditional Demands and Capacities components as needed, such as calming the household, modelling normal disfluencies and speaking to children with short, simple sentences. Three core components were presented to each family: (1) a special talking time each day dedicated exclusively to the child and designed to build self-confidence about speaking, (2) parent instruction to reduce speech rate and pause between utterances and (3) parent instruction not to overtly require any form of speech performance from children. Thirty children were randomly allocated to each of the therapies. Stuttering frequency and severity ratings were obtained immediately before and after 12 weeks of treatment. Both groups showed similar and significant reductions of stuttering frequency in %SS from 7.2% to 3.7% for the LP and from 7.9% to 3.1% for the Demands and Capacities model treatment. Similar results occurred as well for stuttering severity rated by parents: from 5.0 to 2.3 for the LP and from 4.8 to 2.1 for the Demands and Capacities model therapy. Overall, no differences between groups were found and parents rated both treatments favourably.

It is difficult to interpret the Franken et al. (2005) report because only portions of the treatments were presented and a no-treatment control group was not used to determine whether there was in fact any treatment effect or natural recovery was observed for the two experimental groups. Similar caveats apply to the results of the Yaruss et al. 2006 report and the two Phase I non-randomised clinical trials (Millard et al., 2008, 2009). Nonetheless, there is a strong suggestion that therapies based on the multifactorial model of early stuttering provide some stuttering reduction and parent understanding of their children’s stuttering, and their capacity to cope with it will improve with counselling

Advantages and disadvantages

Advantages

The strength of the treatment is linked to the value of its guiding theory. Since the multifactorial model is about what triggers stuttering during childhood speech development, the model is not threatened by recent advances in the understanding of the more distant physiological causes of stuttering. At the beginning of both treatments, parents are taught about the multifactorial nature of stuttering and that those factors vary from one child to another, from one situation to another and from one time to another. This way parents learn that there is no single cause of stuttering and that they are not to blame for their child’s stuttering. Hence, each child’s stuttering treatment is individualised. They also learn that there are factors which are not under their control, such as genetic and temperament factors, and factors that can be modified, such as negative responses to stuttering, rapid rate of speech and coping with children who are sensitive about stuttering. This way parents become less anxious and by being active participants in treatment, parents become competent in dealing with their child’s stuttering and helping the child to develop more effective communication skills and attitudes. The child, therefore, feels supported by parents. Also, as Kelman et al. (2005) suggest, the PCI approach – and indeed any approach based on a multifactorial model – can be implemented with very young children who still do not have well-developed attention and listening skills, or cognitive or meta-linguistic skills. Arguably, direct therapy would not be indicated for such children. The approach encourages parents to acknowledge their child’s stuttering, but because the focus of the initial stages is not on the child’s speech this could be useful for sensitive children who may interpret direct therapy as stuttering behaviour not being acceptable.

Disadvantages

Indirect approaches to early stuttering intervention, such as those overviewed here, could be criticised for an unproven theoretical basis and limited evidence of efficacy. Additionally, it is possible that such approaches may elicit parental blame about the disorder (Kelman et al., 2005). Obviously, this could have a negative impact on parents. Additionally, not all families are willing or able to participate in family-based approaches. As with some other family-based approaches it is hard to determine which components of the treatment contribute to any of its demonstrated efficacy and which are irrelevant. Another disadvantage is that the treatment is not overtly designed to remove children’s stuttering, only to improve it.

All that being said, however, any proposed early intervention is likely to be controversial, and this is no exception. So, to what extent is a multifactorial model acceptable? To what extent does it fulfil the requirements of a successful theory: testability, heuristic value and explanatory power as outlined by Packman and Attanasio (2004)? Perhaps that is not for me to posit but to invite later discussion about.

Conclusions and future directions

The results of the Yaruss et al. (2006) preliminary study, the Franken et al. (2005) experiment, and the two Phase I clinical trials by Millard et al. (2008, 2009), have shown that some children involved in the PCI programme achieve stuttering reductions. Additionally, such interventions clearly increase parent knowledge and understanding of their child’s stuttering, which helps them to deal with it. It is not clear at present, though, whether that intervention is better ultimately than no intervention. For some children, a more direct approach clearly is needed. This raises the question of the large-scale research that would eventually be needed to determine subgroups of children who are likely to benefit from such direct intervention rather than experiencing a delay in receiving it.

As with any treatment for early stuttering, the future of the treatments overviewed here, based on a multifactorial model of early stuttering, depends on research about that model itself and whether it can be successfully applied to early stuttering intervention. So, although the results of the preliminary clinical trials research show overall positive results, that is only the beginning. Franken et al. (2005) raised the prospect that parent involvement and time spent with children might be the critical elements of these indirect styles of treatment. That of course applies to any early stuttering intervention and beckons for experimental investigation. PCI has changed since it was initially developed. There is no reason to believe that treatment will continue to change and evolve over time in light of research and clinical experience. The same is true of Family Focused Treatment. Indeed, the authors of the report about that treatment reviewed here conclude that with further research such a treatment ‘may ultimately take its place alongside other empirically validated approaches for helping young children who stutter develop and maintain normally fluent speech’ (Yaruss et al., 2006, p. 130). Perhaps it is the most heartening feature of the work described here that the other two research groups involved (Franken et al., 2005; Millard et al., 2008, 2009) projected further empirical development of the treatments concerned.

Discussion1

Sheena Reilly
The first thing is to clarify that the main goal of the programme is to change the parents’ behaviour and if so, we had a number of questions about how you actually measure that. How do you know that you’ve actually changed the parent behaviour?
Mirjana Lasan
This clinical approach would not necessarily be my first choice. You are correct that the emphasis is on changing parent behaviour and their communication with children. Unlike child stuttering severity, that is not so easy to measure. What you are raising is treatment fidelity. All treatments require parents to do certain things outside the clinic and it is a difficult matter to determine whether they actually do them.
Joseph Attanasio
Your presentation resonated with many in my group because of the focus on the child and not on the stuttering, and not using techniques to modify stuttering but to increase the opportunity for fluency. That led though to an inevitable comparison to the LP. The LP and the approaches you describe make use of parent interaction with children, albeit different kinds of interaction and for different purposes. Do you think that parent interaction with children could be important as a supportive variable with either programme?
Mirjana Lasan
Yes, of course that is a possibility.
Joseph Attanasio
Perhaps for future clinical trials we need a control condition where parents are just interacting with their children in a positive way. So the LP and, say, PCI therapy could be compared to a control condition in a randomised trial. The control condition could just involve positive parent attention to speech.
Dave Rowley
Of course that scientifically makes sense. But I can’t really see how the two treatments could be combined in a clinical trial, because they have different primary outcomes. It seems to be a logical impossibility. The primary outcome for the LP is no stuttering or extremely mild stuttering, but that is not the case for PCI therapy, or any of the related treatments discussed.
Ann Packman
Our group was interested from a clinical point of view how the therapist decides what to work on with parents. From my recall of the Demands and Capacities model there are four demands and four capacities, and the combination of them is different for each child. How does the clinician decide which of them is relevant to any child?
Mirjana Lasan
The parent essentially makes the decision. A video recording of children and parents interacting is made in the clinic and reviewed. Parents and clinicians watch the video, and the parent decides, guided by the clinician, what the targets for therapy should be. Parents determine what would be the best thing to enhance the child’s fluency: whether, for example, they speak too fast or whether they ask too many questions. So together with parents they decide what would be the best thing to modify
Ann Packman
So then, if the initial choices did not produce any effects, the combination might be changed. Is that correct?
Mirjana Lasan
Yes, like any treatment, the entire process is constantly subject to potential revision.
Sheena Reilly
We had much discussion around the fact that there is limited evaluation for any of these styles of treatment, with small numbers of children. These treatments have been around for decades, so why do you think that more advanced and sophisticated clinical trials have never been done?
Dave Rowley
I think the answer is that these styles of treatment do not lend themselves to evaluation, because it is not perfectly clear what they are designed to attain. Hence, there has been no driving need to evaluate them. I think that is a shame because they may have real value. But if the question is posed ‘What is the evidence that it works?’, the only answer can be that there is limited evidence. The problem extends here to replication also, because the nature of the treatment is different for every child and that it is not fully clear what the treatment is designed to achieve. So it is difficult to imagine a future time when clinicians can be sure they are doing the same early intervention method that others have shown independently to be useful.
Joseph Attanasio
In terms of the Demands and Capacities model, perhaps we should look at capacity as performance. My reading of the approach suggests much emphasis on decreasing demand and but not much emphasis on increasing competence or performance. Perhaps it is just easier to decrease demand than to increase performance.
Mirjana Lasan
Definitely in my experience, in any treatment derived ultimately from multifactorial concepts, it is much easier to decrease demands. And that is why the clinical applications that I describe focus on just doing that.
Ann Packman
Another clinical question is that it is obvious, from the research that you presented, that some children will not respond to this style of treatment and will need a direct one. So when is the decision made to try a direct treatment?
Mirjana Lasan
Quite simply, when the indirect proves unsatisfactory.
Ann Packman
If they are still stuttering?
Mirjana Lasan
Yes.
Sheena Reilly
We were interested in a comment that you made about not making parents feel guilty and not adopting blame when using this sort of approach. We had much discussion about how do you manage to do that when what the clinician is really doing is removing parent behaviours that are implicated in the start and the continuation of stuttering.
Mirjana Lasan
Of course, feelings of guilt and blame might occur with all early stuttering interventions that involve the parents. I suppose that is the value of the parent education components of these treatments that tell parents about the multifactorial nature of what is happening and about how it is inevitable that this will trigger stuttering if a child is genetically prone to stuttering. With a proper clinical relationship with parents it should not be an issue.
Sheena Reilly
Surely then there must be a fine line there between being responsible and not being responsible for the child’s stuttering, because the treatment focuses on behaviours they must stop doing in order to treat the stuttering. It must be a clinical challenge to get that just right.
Mirjana Lasan
You are absolutely correct that this is one of the challenges with presenting this treatment, particularly for junior clinicians who may be attempting it for the first time.
Joseph Attanasio
How do you handle the problem you mentioned that parents may not want to be involved in multifactorial treatment? They may, for example, just want the clinician to fix the problem during each clinic visit. It would be by definition impossible to do a multifactorial treatment without full parent involvement.
Mirjana Lasan
I do hope that I did not imply that such a problem was unique to any multifactorial style treatment. It is by definition a problem with any early intervention I know of that has been shown in any way to be empirically viable. Of course, on some occasions such an issue is a barrier to treatment, but fortunately most parents are not like that. Otherwise, I don’t think early stuttering intervention would be possible.

References

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Bernstein Ratner, N. (2004) Caregiver–child interactions and their impact on children’s fluency: implications for treatment. Language, Speech, and Hearing Services in Schools, 35, 46–56.

Franken, M. C., Kielstra-Van der Schalka, C. J., & Boelens, H. (2005) Experimental treatment of early stuttering: a preliminary study. Journal of Fluency Disorders, 30, 189–199.

Guitar, B. (2006) Stuttering: An Integrated Approach to its Nature and Treatment (3rd ed.). Baltimore, MD: Lippincott Williams and Wilkins.

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Zebrowski, P. M., Weiss, A. L., Savelkoul, E. M., & Hammer, C. S. (1996) The effect of maternal rate reduction on the stuttering, speech rates and linguistic productions of children who stutter: evidence from individual dyads. Clinical Linguistics & Phonetics, 10(3), 189–206.

1 Dave Rowley joined Mirjana Lasan to respond to the leaders of the discussion groups.

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