Look at Mummy: challenges in training parents to deliver a home treatment program for childhood apraxia of speech in a rural Canadian community AUTHORS

Introduction:  Children requiring speech pathology services in rural and remote locations face many barriers in accessing adequate services. This has particular consequences for children who need intensive treatment for therapy to be effective, such those with childhood apraxia of speech (CAS). Parent training has been used to overcome speech pathology service delivery barriers for a range of other communication disorders. However, the effectiveness of training parents to deliver a motor-based treatment for CAS within rural and remote contexts has not been evaluated. This study examined the effectiveness and feasibility of training parents in a rural community to use the treatment approach of dynamic temporal and tactile cueing (DTTC) in order to provide more intensive treatment sessions at home. Methods:  The study used an experimental single case across behaviours design and parent interviews to evaluate outcomes both quantitatively and qualitatively. The study included four parent– child dyads from a mixed socioeconomic rural community in Canada. Child participants ranged in age from 3 years to 8 years. Child treatment outcomes were measured using an improvement rate difference (IRD) calculation based on percentage of phonemes correct. Fidelity to the treatment protocol was measured using a fidelity score. r Rural and Remote Health rrh.org.au James Cook University ISSN 1445-6354


Introduction
Children needing speech pathology intervention in rural and remote locations face barriers in accessing services . The reported barriers to service delivery include transportation issues, long waiting lists and lack of clinicians . Overall health inequities between rural/remote populations reflect these barriers and are well documented in countries with large rural geographic areas such as Australia and Canada . Barriers to service delivery are particularly problematic for children with long-term conditions such as childhood apraxia of speech (CAS), a severe speech disorder in which children experience difficulty in forming appropriate motor plans for speech sounds and that is often difficult to treat .
Current research into the treatment of CAS suggests that treatment is more effective if it is provided intensively . However, in the reality of clinical practice, particularly within a rural and remote context, it may be challenging for a child with CAS to access services that provide a similar level of treatment intensity . Furthermore, there are limited evidence based treatment options for CAS, and many clinicians often do not feel prepared enough in their training in providing services for children with CAS .
The relationship between high levels of treatment intensity and successful motor learning is one of the tenets of the principles of motor learning . Dynamic temporal and tactile cueing (DTTC) is one of the few evidence based and effective treatment strategies for CAS that utilises the principles .
Within speech pathology service delivery, many speech language pathologists (SLPs) train parents to provide home-based treatment as a means of overcoming service delivery barriers such as the inability to provide adequately intensive treatment . A survey of Canadian and Australian SLPs indicated that the majority of SLPs surveyed used parent training to overcome service delivery barriers . In addition to using parent training to overcoming barriers, several surveys of Australian SLPs have shown that 80-96% of SLPs involve parents in intervention, mainly through the provision of homework .
Currently there is limited research into the effectiveness in training parents to deliver a structured treatment program for CAS, particularly within the context of a rural/remote location. The only treatment study to date that has explored the effectiveness of training parents to deliver a home-based treatment program for CAS was conducted in an urban location and showed limited effectiveness in using parents to provide treatment . In that study, parents were trained to use the Rapid Syllable Transition Treatment with their child. In addition to the limited effectiveness, parents also reported many challenges in carrying out the program in the home . Nonetheless, with research evidence strongly supporting more intensive therapy for best intervention outcomes, documented inequities in rural and remote speech pathology service delivery, the effectiveness and feasibility of training parents to deliver therapy at home for their children with CAS in a rural/remote location needs to be further explored.
One paradigm for exploring how parent-led intervention can affect the therapeutic process is dynamic systems theory (DST) . DST proposes that development is a result of the bilateral and nonlinear interactions that exist between different developmental areas (eg speech and motor development) and between the child and their environment . Various internal (eg neuronal maturation, attention) and external factors (eg parent interactions and the environment) may impact the developmental process to produce long term change. In the case of the current study, a DST lens can be used to see how an exploration of external factors such as the dynamic influence of the parent-child interactions and the child's social environment may influence the outcome of a parent training program in a rural community.
The present study explored the effectiveness of a parent training program that used DTTC to improve the speech skills of children with CAS living in a remote location, and aimed to answer the following questions: Were the parents able to achieve good fidelity with the treatment and be able to complete the required number of treatment sessions?
What were the parents' experiences when delivering DTTC?
Did a parent-delivered DTTC home treatment program lead to increased speech accuracy in words targeted in therapy (target words)?

Methods
The study used sequential mixed methods research design comprising a single case experimental study followed by a qualitative thematic analysis of parent interviews. These methods were selected as appropriate for early trials of new or substantially changed behavioural interventions .

Community
The research was conducted in a remote Northern Canadian regional centre comprising three closely located communities that had a combined population of less 6000 people . This tricommunity acted as a regional centre for communities that were further north and more remote. There is only a single SLP employed by the local health region to service children of 0-5 years of age within the tri-community and the entire northern region. A single

Participants
Four parent-child dyads participated in the study. One of the dyads was an auntie-nephew combination but for the purposes of this study, all the adults will be referred to as 'parent' unless specifically discussing this dyad. (The term 'auntie' is used here because it is the culturally relevant term used in the community in which the participants lived. All parents participating were female, so 'she' and 'her' are used when referring to parents.) All participants lived within the tri-community. Three of the children lived with their parents and one child, whose auntie completed the study with him, lived with his grandmother. All children had received prior speech therapy from the health SLP at some time in their lives. However, this therapy was not intensive and did not involve any prior parent training related to their speech disorder. Further demographic information is found in  DTTC treatment protocol: DTTC relies on a hierarchy of cues that help support a child to say a targeted word with maximal success .
An outline of the cueing hierarchy used in this study is shown in Figure 2. If a child is unable to respond to the cues provided (eg to copy the parent saying the word or to say the word at the same time as the parent) and does not say the targeted word accurately, then the parent moves down the hierarchy and provides a more supportive cue (eg if the child cannot copy the word in direct imitation, the parent will then provide simultaneous cueing). If the child is able to respond to a cue with an accurate model, then the parent moves up the hierarchy and provides a less supportive cue (eg moving from tactile or gestural cues to a simultaneous and slowed rate of cueing).
A DTTC board game was designed and provided to each parent to help her follow the cueing hierarchy when working with her child.
The game provided the parent with instructions of what to do based on the child's response to the cue. Parents were also instructed to ask the child to repeat a target word three times if they gave a correct response to a cue. If a child made two consecutive errors at one level of the hierarchy, they were given another chance at saying the word with a lower level cue before the parent moved onto another target word. Each target word was written on a card with a picture of that word also on the card. The same picture was used in the probe data presentation.
At the start of each session, parents were asked to shuffle the target words. They were provided with a timer that they could set for 15 minutes and were also provided with short games that they could play with their child periodically throughout the session if their child was having difficulty complying with the tasks. During the two treatment phases, parents were asked to provide treatment at home for 15 minutes twice a day, five days a week. Parents were also provided with an audio-recorder and asked to record the treatment sessions with their child.  them. Parents were given the opportunity at the end of the interview to add any further comments. The semi-structured interviews were audio-recorded and later transcribed by the first researcher. The first researcher kept notes during the interview, and these were compared to the transcription of the audio-recording. Qualitative content analysis was used by the first author to analyse the parent interviews . Each idea communicated by each parent was recorded and then similar ideas were collated. Out of these collated similar ideas, emergent themes were identified and these emergent themes sorted as either representing challenges or benefits of the experience. These themes were then compared across all participants to determine any similarities or differences.

Outcome measures
The transcripts and themes were then compared by the second author to ensure there were no omissions or misinterpretations.
Parents were provided with copies of the transcribed interviews and invited to make any changes. None of the parents requested any changes to the transcripts.

Ethics approval
The research was approved by the University of Sydney Human Research Ethics Committee (2014/924).

Data for each child are shown in Figures 3a -d and a comparison of
their IRDs is shown in Figure 4.
Brandon: Brandon had a slightly altered treatment phase due to personal circumstances. His auntie provided eight sessions at variable time periods across 7 weeks followed by a 3-week break and then four more sessions over 2 weeks. Three weeks of maintenance data were collected after treatment ceased. His auntie was able to treat only one group of words (phase 1 target words). Therefore, probe data were split into target, similar and control words. For simplicity, the two treatment phases were still considered to be separate phases. Although Brandon's auntie received the same duration of parent training, due to her work commitments, it was spread over six weeks instead of four. See Figure 3a for

Figure 4: Improvement rate difference scores for all participants for all probe words.
A visual analysis of Nicholas' probes indicated that there was no treatment effect for his target words (Fig3b). There was an improvement with his target words but at 43% this was not reflected in a treatment effect. However, both target (43%) and similar (46%) words improved to a greater extent than his control words, which only had an IRD of 16% (Fig4).
Tony: Visual analysis indicated that Tony did not show any treatment effect across the study (Fig3c). This is reflected in IRDs less than 50% for all groups of words (Fig4). However, although control, similar and phase 2 target words showed low IRDs, phase 1 target words demonstrated a higher IRD of 46%. This indicated that, like Nicholas, although not demonstrating a high enough effect size to be considered effective, the phase 1 target words showed a greater degree of improvement than the other words.
Rosalind: Visual analysis indicates that Rosalind did not demonstrate any improvement in her target words (Fig3d). As seen in Figure 4, Rosalind did not demonstrate any treatment effect for any words during the study, with IRDs of less than 50% for all word groups (control: 21%; similar: 21%; phase 1 target: 36%; phase 2 target: 12%).

Parent measures
Data for all parents are summarised in Table 2.   Rosalind's responses correctly.

Themes from semi-structured interviews
The themes that emerged from the parent interviews have been divided into either the barriers to completing the therapy program or the benefits of the program. All themes were expressed by all parents during the semi-structured interview.
Barriers: Two themes related to the barriers of delivering the program at home. These were lifestyle challenges and behavioural challenges that the parent faced. Other commitments such as needing to travel out of town for medical appointments or to visit family also interfered in their ability to carry through with the program. As an example, Brandon sometimes was required to travel south to another town to visit his birth mother.
Behavioural challenges: Parents often had to spend time getting their child motivated to do the therapy because non-compliance was frequently an issue. This was despite attempts at developing a reward system to encourage better compliance. In addition, parents often had to manage tantrums from their child during the therapy session or when their child felt that they could not do what was required of them. Emma said, 'what I found hard was getting her to concentrate'. Anna also found it difficult to manage Brandon's behaviour, particularly after he returned from visiting his mother: … it was hard to go over there in the middle of the day when he had a bad day at school and then try and do with him, there was no progress 'cause he was in his mood ….

Benefits
The two themes that emerged for the benefits of therapy were positive relationship building and development of therapeutic skills.  Despite Brandon's challenging behaviour, Anna still found the oneto-one time a positive experience: 'Cause I got to spend that one on one time with him and we did do activities and stuff and that was good.

Development of skills:
The parents all considered that they developed increased confidence in using the cues as the study progressed, and they felt that they were able to use the skills in conversation with their child. As Nicholas' mother said, 'we'll always have the techniques'. The parents felt they could use the cues during normal conversation to help their child be more accurate in their speech production in the moment as opposed to during a structured therapy setting. The parents often found themselves using the cueing during the day with their child. Anna, Brandon's auntie, found that she was using some of the cues in interactions she was having with other children with unclear speech that she encountered during her work. She also reported that she was using the cues in conversation with Brandon when she did not understand him: Betty and Emma reported that as they became more comfortable with managing their child's behaviour, their child's compliance improved, and the parents were able to get them to work more effectively. For instance, Betty reported that Tony 'tried a little harder to talk, he didn't get as frustrated faster'.

Discussion
This research study aimed to explore the effectiveness of training parents in a rural and remote community to deliver a home treatment program for children with CAS in order to address the lack of adequate speech pathology services. All the parents, no matter their level of fidelity of the treatment protocol, had difficulty carrying out the program due to both social and behavioural challenges. Not surprisingly then, only one of the four children, Brandon, had a moderate treatment effect.
A better understanding of the therapeutic process and the impact of the social and behavioural challenges encountered by the four parents in this study may provide a possible explanation for the lack of treatment effect in three of the children. A DST paradigm can be used to explore these influences on treatment outcomes. DST proposes that development is the result of multiple factors interacting with each other . Therefore, in understanding why the parent training in the present study was not successful in developing the speech skills of participants, we need to look beyond just the act of therapy and explore the influence of multiple factors on this therapeutic process. For this study, the impact of the parent-child interactions and the influence of social factors on the therapy process were explored.

Parent-child interactions
Several findings from this study highlight how the parent-child interactions impacted the therapy process. For example, the qualitative data from this study suggests that when a parent attempts therapy with a child, the nature of the parent-child

REFERENCES:
has the potential to circumvent some of the barriers to service delivery that rural and remote clients face, may itself not be an appropriate strategy given the challenging social environment that may exist in such communities. Viewing speech pathology services within their social context may need to be considered by clinicians when deciding on the model of service delivery for their clients. This may be of particular importance in communities where there are societal challenges that may make a home treatment program more difficult to achieve.

Limitations
Given that this study only focused on four parent-child dyads within one rural location, there are limitations on the extent to which the results of the study can be generalised to other children with CAS and other locations. This is further compounded by the fact that two of the four parent-child dyads had truncated or altered treatment timelines; however, it is also possible that this truncation would be replicated in other communities due to equivalent social factors.
It is necessary, therefore, for such research to be conducted in other communities to determine if these results are replicated. This would include both rural and non-rural locations to determine if these challenges are experienced across the board or if rural parents face particular difficulties in implementing home treatment programs.
Further studies also may explore the role of target selection on the success of parent-implemented therapy. For instance, it is possible that the children in this study may have responded better to parent-led therapy if the targets selected were easier or if the goals were more about consolidating newly learnt sounds rather than introducing new sounds.
This study did not explore other possible outcomes such as the development of prosody (also a feature of CAS) or improvements in intelligibility. Therefore, the potential impact of parent-led treatment on these features is unknown.
The existing relationship between the first researcher and the parents may have led to parents to self-select the information they provided in the interview. However, this prior relationship may also have meant that parents felt more comfortable expressing their opinions about the process, particularly the challenges that they faced.

Conclusion
The present study highlights the complexities of training parents to deliver a motor-based treatment program, particularly within the context of a rural/remote community where social factors may create challenges in carrying out a home-based treatment program.
The complexities of living in a remote location may make regular and intensive home therapy sessions difficult. The nature of a drilllike program such as DTTC brings the parent-child relationship into somewhat uncharted waters. It may well be that parents are not always the best situated to carry out motor-based therapy with their child. It is possible that other people in a child's life such as early education workers or school tutors may be better placed to carry out regular and structured therapy programs. Therefore, further research into the efficacy of training other support professionals in a child's life to provide this therapy is required.

Declaration of interest
The first author was employed in the health region where this research was conducted.