Parent reflections of experiences of participating in a randomized controlled trial of a behavioral intervention for infants at risk of autism spectrum disorders

Abstract

Background:

Despite the mounting evidence of efficacy of early intervention for children with autism spectrum disorders, there is little research that considers the various perceptions and resources with which parents respond to the pressures and opportunities associated with participation in early intervention. Research is particularly lacking surrounding experiences of parents with infants who are at risk of autism spectrum disorders but do not (yet) have a diagnosed condition.

Objectives:

This qualitative study aimed to explore the experiences of caregivers following their participation in a randomized controlled trial of Adapted Responsive Teaching, a parent–infant relationship-focused intervention for infants at risk of autism spectrum disorders in a community sample. Parents were randomized into either the treatment group, in which they participated in the Adapted Responsive Teaching intervention, or the community services group, in which they were provided with information regarding local early intervention services and were encouraged, but not required to, seek community services as part of their inclusion in the randomized controlled trial.

Methods:

Semistructured interviews were conducted with families following the completion of the randomized controlled trial. Participants consisted of 13 mothers and 4 fathers. Five dyads were interviewed together for a total of 14 families. Child ages ranged from 39 to 46 months at the time of interview. Analysis was conducted on 14 interviews from 10 families who were randomized into the treatment group and 4 families randomized into the community services group. Analysis was informed by a thematic analysis approach, which involved a systematic process of coding and theme identification both across and within groups.

Results:

Themes that emerged across groups included Working against all odds, Value of the personal relationship, Getting the ball rolling, and Getting dad on board. One broad theme represented the data within the groups: Win-win (Adapted Responsive Teaching group) and Navigating amidst ambiguity (community services group).

Conclusions:

This study illuminates the personal experiences and contextual influences affecting families who are participating in the randomized controlled trial through early identification of “risk” status for autism spectrum disorders in their infants. Insights gained from these interviews may serve to refine and enhance intervention models and to enhance early intervention services for families.

Keywords

autism early intervention parent experience qualitative

Background

Recent literature has shown that children may be identified “at risk” of autism spectrum disorder (ASD) as early as 12 months (Pierce et al., 2011; Turner-Brown et al., 2013). This early recognition of “risk” status through screening facilitates early exposure to intervention, which has the potential to impact developmental outcomes in young children with ASD (Dawson et al., 2010).

While there is little information about the early experiences of parents who have infants newly determined to be “at risk” of a later diagnosis of ASD, literature has explored the experience of caring for a child with an ASD diagnosis (e.g. DeGrace, 2004; Myers et al., 2009). Caregivers of children with ASD report higher rates of stress as compared to those with other developmental disabilities (DDs) and those of children of typically developing peers (Baker-Ericzen et al., 2005; Estes et al., 2009). Additionally, there is evidence to suggest that caregivers of children with ASD report higher levels of marital discord (Dunn et al., 2001), increased isolation (Woodgate et al., 2008), as well as little time for family activities and outings (Hutton and Caron, 2005). Caregiver stress has also been shown to counteract positive outcomes in early intervention (EI) models for children with ASD (Osborne et al., 2008).

Although parent stress and depression have been studied extensively in the literature, other factors influencing early experiences of parents, particularly those with infants at risk of ASD, have been overlooked. Furthermore, there is limited research on how families’ experiences impact decision making surrounding uptake of services and participation in various interventions as well as differential outcomes for children with ASD (Dawson et al., 2010; Patterson and Smith, 2011). Overall, a dearth of research focuses on family experiences of participating in early screening and intervention for infants at risk of ASD in a community sample. Based on this scant literature, the potential value of understanding parent experiences amidst participation in a randomized controlled trial (RCT) focusing on parent training and implementation of strategies in the context of everyday family life is salient.

This qualitative study aimed to explore the experiences of caregivers following their participation in an RCT of a parent-mediated intervention for infants at risk of ASD (Baranek et al., in press; Wakeford et al., in press). We sought to generate novel insights into parent’s choices (to fill out an early screening measure, uptake of community services (CS)), perceptions (of the intervention team and CS providers, surrounding evaluation outcomes), and experiences (participating in intervention, navigating CS). It is hoped that this study would provide information to generate hypotheses for future research studies as well as to improve EI experiences for families of infants at risk of ASD and ultimately optimize child outcomes.

Methods

Overview of the Early Development Project

Families who consented to interviews were recruited from the “Early Development Project (EDP),” a grant funded by Autism Speaks. EDP consisted of an RCT testing the effects of an experimental treatment (Adapted Responsive Teaching (ART)) versus a control condition (CS) with infants who were screened to be at risk of ASD at 12 months on the First Year Inventory (FYI; Baranek et al., 2003). The FYI was distributed on the basis of birth records to families of infants approaching their first birthday in a community sample across rural and urban areas throughout central North Carolina. Families of infants who met risk criteria on the FYI were invited to participate in a comprehensive developmental evaluation (Time 1—baseline) to confirm risk status. Risk on the FYI for the present study was determined by using a total risk score for all FYI items (for a description of items and scoring, see Reznick et al., 2007). A second screening was completed as part of the Time 1 evaluation, and only those children demonstrating concerns in both social-communication and sensory-regulatory domains were deemed eligible to participate in the RCT. These concerns were communicated to parents at the Time 1 evaluation. Risk status was not defined by having an older sibling with an ASD diagnosis. Of the 24 families who completed the Time 1 evaluation, 16 enrolled in the RCT. Due to the recognition that the overall sample would be small and the interest of the investigators to evaluate the intervention qualitatively as well as quantitatively, the investigators chose a 2:1 randomization schedule to increase the number of families in the ART intervention (see Dumville et al., 2006, for a review of the use of unequal randomization ratios). Eleven families were randomized into ART and five were randomized into the CS group. Following the 6-month treatment phase, families participated in a posttreatment evaluation when the child was around 22–24 months (Time 2), followed by a diagnostic evaluation 6 months later (Time 3).

Families in the experimental group participated in a 6-month intervention utilizing ART. ART is a manualized parent–infant intervention (Wakeford et al., in press), adapted from the Responsive Teaching curriculum (Mahoney and MacDonald, 2007). ART consists of a relationship-based approach to EI that employs modeling and coaching to encourage parents to use responsive strategies during daily routines and interactions with their children. An ART interventionist makes 1-h home visits to each family, twice a week during the first 6 weeks, and once a week thereafter. The ART intervention is organized around two broad domains of development, social-communication and sensory-regulatory. Pivotal behaviors, that is, child behaviors considered foundational to optimal growth and learning, are specified under each developmental domain. The interventionist coaches the parent(s) in using responsive strategies to promote the child’s development of the targeted pivotal behaviors. Responsive strategies include “follow my child’s lead,” “be sensitive to my child’s state,” “be more interesting than my child’s distractions,” and “change the activity or environment to meet my child’s sensory needs,” among many possible strategies. The families in the ART group were also given information about local resources, providing them with the opportunity to simultaneously participate in services in the community, and were contacted monthly by the project coordinator to track services received.

Similar to the families in the ART group, families in the control (CS) group were provided with information regarding EI services found in the local community and were contacted and tracked monthly by the project coordinator. Parents in the CS group, just as in the ART group, were encouraged, but not required, to seek CS as part of their inclusion in the RCT.

Each month, the project coordinator contacted the parents of all children, those in the RCT, and those not randomized to the RCT. The project coordinator asked parents for a general update on their child, whether they had all the supports they needed, and whether they had any questions or concerns that the EDP team could possibly assist with answering. If a family was receiving any CS, the project coordinator asked about the frequency, duration, and location of the sessions.

Participants

In the midst of the early phases of the EDP study, the research team saw an opportunity to retrospectively gain knowledge about families’ experiences following the RCT process. Therefore, participants were offered interviews following their completion of the RCT. Interviews occurred approximately 3–6 months after the Time 3 evaluation and spanned an 11-month period across participants. Of the 24 families evaluated in the EDP study, 15 families consented to interviews, including 10 from the ART group, 4 from the CS group, and 1 family whose child was evaluated but was ineligible for randomization. The ineligible family was interviewed but was excluded from analysis on the basis of only having one family in this group. All families in the ART and CS groups were included in the analysis (n = 14). Participants consisted of 13 mothers and 4 fathers, including 5 dyads interviewed together. Participants resided in five counties in central North Carolina. Our participants were primarily Caucasian (87%), and mothers’ education levels ranged from some college to graduate degrees. Children in families who participated included 11 boys and 3 girls, 8 were first born and 1 was a fraternal twin. Of the six older siblings, two had a diagnosis of ASD. A third older sibling did not have an ASD diagnosis, but the mother reported “Asperger qualities.” No other sibling disabilities were reported.

Children were assessed at Time 1 between the ages of 13 and 17 months. Child ages ranged from 3 years 3 months to 3 years 10 months at the time of interview. See Table 1 for the demographics of the children.

Table 1.

Participant demographics by child.

Child	Group	Birth order	Received CS?	Age at Time 3 (months)	Age at time of interview (months)	Mother’s education	Mullen ELC (T1)	Mullen ELC (T3)	Diagnosis (T3)
1	ART	2/2	No	31	46	College	63	104	Other concerns
2	ART	2/2	No	33	40	College	106	92	Autism
3	ART	3/3	Yes	32	39	Vocational/trade	96	90	Other concerns
4	ART	1/2	No	33	45	Masters	109	130	Concerns
5	ART	1/2	No	32	39	College	73	117	Other concerns
6	ART	1/1	Yes	34	41	Master’s	72	60	PDD-NOS
7	ART	2/2	Yes	32	39	College	58	96	Autism
8	ART	1/1	Yes	35	43	Master’s	80	110	Other concerns
9	ART	1/2	No	30	41	College	85	95	Other concerns
10	ART	1/2	No	33	39	College	94	63	Autism
11	CS	2/3	Yes	32	40	College	102	105	Other concerns
12	CS	2/2	No	34	43	Doctoral	80	109	Other DDs
13	CS	1/2	Yes	35	40	Master’s	88	112	Other concerns
14	CS	1/1	Yes	31	43	Master’s	65	63	Autism

CS: community services; ELC: Early Learning Composite; ART: Adapted Responsive Teaching; PDD-NOS: pervasive developmental disorder–not otherwise specified; DDs: developmental disabilities.

“Other concerns” included one of the following: repetitive behavior, sensory processing, or pragmatic language.

Interview procedures

Semistructured interviews were used in order to identify new ways of understanding the phenomena of parent experiences in participating in this specific RCT testing an intervention for infants at risk of ASD (but not diagnosed at the time of randomization). One interview was conducted with each family following the completion of the RCT ranging from 3 to 6 months (mean = 5.2 months) after Time 3 evaluation.

Interviews consisted of a series of approximately 22 open-ended questions that followed a chronological sequence of the RCT events (see Figure 1). For example, the interview questions asked about the parent’s choice to fill out the FYI, decision to participate in CS, and intervention experience. In line with the qualitative nature of the study, the interview process remained flexible, allowing for deviation from the chronological sequence of questions, so that a natural conversational interview could ensue. The principal investigator (PI), Grace Baranek, consulted with a qualitative methods expert, Virginia Dickie, on the interview methods, design, and question development. Baranek and Dickie reviewed the data obtained following the first few interviews in order to refine interview questions as needed to ensure that the interview guide was obtaining sufficient data.

Figure 1.

Sample interview questions at each RCT time point.

Interviews were conducted by one of the first two authors in participant’s homes, or in one case in the child’s school, and lasted anywhere from 1 to 2 h. The PI trained the first author in interview procedures, accompanied her on the first several interviews, and provided feedback on interview techniques. The PI reviewed all of the transcripts of the interviews conducted by the first author following the interviews. In one form of member checking, participants were provided with opportunities to confirm and expand upon their responses throughout the interview by the interviewer using the strategy of asking clarifying follow-up questions. The first two authors engaged in ongoing conversations surrounding interview questions, experiences, and data.

Neither of the interviewers had participated in the evaluation or intervention process, which allowed for the families to talk about their experiences more openly. Interviews were audio-recorded and transcribed. Families received a US$25 check for their participation at the time of interview.

Analysis

ATLAS.ti (Version 5.0; Scientific Software Development, Berlin, Germany) was used for coding and analysis. Analysis involved a systematic process of coding and theme identification that was informed by a thematic analysis approach (Braun and Clarke, 2006). In general, the process of analysis involves a series of steps, beginning with a review of transcripts and ending with identification of broader themes that extend throughout an entire set of interviews (Morse and Field, 1995). This process can be described as iterative in that an ongoing review of transcripts takes place as new codes are identified, calling for a rereview of all transcripts from the beginning to ensure data are comprehensively reviewed and coded.

First, a priori codes were generated based on anticipated categories of responses to interview questions. These codes included both “Decision-making” time points (e.g. Decision to fill out FYI, Decision to participate in CS) and “Experience” time points (e.g. Experience-evaluation, Experience-intervention). These a priori generated codes were used to identify “chunks” of data that were related to specific phases of the research process or particular interview questions (Zhang and Wildemuth, 2009). Coding these particular experience contexts allowed for identification of codes that emerged within particular phases of the research process. In other words, a priori codes were expected to co-occur with emergent codes and allowed for a process of filtering the data by particular decision-making points or experiences that occurred for all families throughout the RCT process.

The second step of analysis involved a review of transcripts in their entirety by the first two authors, which included informal note taking of initial coding ideas. These initial notes also included a list of both positive and negative experiences reported by parents throughout their participation in the RCT.

Third, transcripts were coded line by line using both a priori and emergent coding for the entirety of individual cases across groups. Emergent coding included the identification of features of the data that the first author identified as salient, reoccurring, or interesting, which revealed themselves directly from the text data through the process of line-by-line coding (Hsieh and Shannon, 2005).

The fourth step involved the identification of initial themes both across and within treatment and control groups. More specifically, coded data were analyzed across groups in order to identify broader themes that emerged surrounding parent experiences. Coded data were then collated by group (treatment and control), and common themes within groups were identified.

Next, visual maps were generated to describe the relationships between codes and to identify broader themes both across and within groups. Thematic maps consisted of visually mapping codes to identify how they grouped together or fell out into distinct themes that represented the data (Braun and Clarke, 2006). These visual maps also helped to determine what aspect of the data each theme captured, to ensure that the data were well represented across themes.

The final step involved the refinement and naming of themes, as well as a discussion of their relevance to the current study and the broader literature (Braun and Clarke, 2006). This naming of themes and the application to the current literature took place primarily between the first two authors, on an ongoing basis throughout the analysis process. The findings were also presented to the entire team for discussion and further refinement of ideas following the initial phase of analysis in order to validate the findings.

Results

Themes across groups

Four themes represented the data across both groups (treatment and control). These themes were named Working against all odds, Getting the ball rolling, Value of personal relationships, and Getting dad on board. In addition, parents in both groups revealed both positive and negative experiences associated with participation in the RCT.

Working against all odds was one theme that surfaced across both groups. This theme describes the primary caregivers’ (mostly mothers in our sample) ongoing struggles to be heard about their early concerns surrounding their child’s development and a search for validation for these concerns. Mothers often reported finding themselves in a situation early in their child’s life where the pediatrician, extended family members, and even the EI system professionals were not hearing or validating their concerns. These mothers often reported a “wait-and-see” attitude from the father and a lack of validation of concerns from extended family members such as grandparents. One mother described the validation she felt from being in the study stating, “… my family says ‘oh he’s fine, he’ll grow out of it,’ and I know, yeah there is actually a problem … you all saw there is something too.” One mother described the early barriers she faced in trying to get validation for her early concerns stating, “… if we had not gotten into the study, by now we would have heard ‘no’ so many times that we would have given up trying to get anybody to sort of see what we were seeing.” Another mother similarly described the role that the study played in validating her concerns and subsequent choice to pursue services, “You all (the study) saw something too. And I think without that I would have put it off, and now I am pursuing it” (referring to CS).

Parents reported that their child’s pediatrician either had no concern or appeared dismissive of parent’s concerns about the child’s development and would “get involved” only when medical issues accompanied the concerns, such as gastrointestinal issues. One mother said, “I feel like they just thought I was a nervous first-time mother.” Another mother said, “He’s (pediatrician) more relying on his observations during exams than talking with the parents about their concerns.” Another mother stated, “He (pediatrician) thought that (child) seemed perfectly normal …” Only one family mentioned their pediatrician validating early concerns and referring them for diagnostic testing.

A second theme that emerged across all groups was that of Getting the ball rolling. Although most parents noted having early concerns or noticing, as one parent put it, that something was “not quite right,” some reported feeling “surprised” that their child qualified as being at risk on the FYI. One parent stated, “If it (FYI) hadn’t have come, I don’t think I would have been looking for any kind of assessment or anything.” Enrolling in the study, even filling out the screening tool and participating in evaluation, appeared to have an effect of initiating the beginning of either EI (be it ART or CS) or, at a minimum, education about available services. In general, parents across both groups reported that participating in the study served to educate them about what resources were available to them in the community. One parent described, “The study did a lot to educate me on what was possible, why it would be useful, and who might provide that.” Parents often reported thinking that CS are only for children who are “severe” and that participation in the study expanded their perceptions of available options through education about CS. One mother stated, “We thought that it would be good for him to have the opportunity to work with someone … but we still weren’t convinced enough that he had problems that would qualify him for (community) programs.” Other caregivers who reported having early concerns described feeling that they did not know where to “start” as far as seeking out CS as well as having feelings of uncertainty around whether their child would qualify for CS.

Another aspect of this theme included the idea of “setting the stage” for future intervention and therapy experiences. Two parents specifically reported that participation in the ART group, as well as EI through the community, set the stage for knowing what to expect from future therapists. One mother reported, “It got us both prepared for working with people in our home … so, it’s like old hat now, but, yeah, it was a little strange at first.” This early exposure to intervention facilitated parent’s level of comfort with therapists coming into the home and eased any ambiguity surrounding the process and purpose of various interventions.

A third theme that surfaced across groups was named the Value of the personal relationship. Parents overwhelmingly reported the personal relationships with professionals, be it through the project or in the community, as being the key to their buy-in to intervention as well as their evaluation of their overall experience. Furthermore, parent’s feelings of positive personal relationships with treatment team members appeared to validate parent concerns, facilitate feelings of support, and contribute to their overall experiences throughout the EI process. One parent stated, “It is much nicer to talk with someone instead of just reading the report … the one-on-one tends to be a lot more reassuring.” Another parent highlighted the unique experiences of parents who have a child with a disability and the need for supportive personal relationships with professionals who work with them. She said,

… it’s overwhelming for parents. It’s very emotional. I mean, you want your child to be typically developing and when they’re not, you know, I just think you need … the same person to talk to, calling … she got to know us.

Parents in the ART group most often described their relationship with the interventionist as contributing to overall positive experiences in the intervention process. This relationship was based on parents both feeling a connection with the interventionist and observing a positive relationship between their child and the interventionist. One parent described, “She held our hands through the whole thing.” Another family described their initial uncertainty surrounding someone coming into the home and a subsequent evolving relationship with the interventionist as being “like family” over time. Another parent described the supporting role that the interventionist played in the context of the mother feeling “overwhelmed a lot of the time” by stating that the interventionist “was like a breath of fresh air.”

Having a consistent point of contact in both the ART and the CS groups was one aspect of the intervention process that parents reported valuing. Caregivers also reported a positive experience when they felt they had a rapport with the assessment team, psychologists, and case managers. For those families whose child received a diagnosis of autism, parents described those professionals with whom they had a rapport as serving as a first line of support.

A final theme that represented the data across groups was that of Getting dad on board. Mothers in our sample often reported that the fathers either did not share early concerns or had a “wait-and-see” attitude. Some mothers described a sense of stigma that impacted a father’s decision not to share diagnostic information with family members. One mother described this experience stating, “His first reaction was to say ‘well, don’t tell anybody’ … which is a hard thing … I would like to have a few people that I can have to talk to …” Another father described the way that he first responded to his son being “at risk” of autism as well as a “wait-and-see attitude” stating, “I just thought everything will be okay. Everything works out.” The mother in this same interview said that this attitude is a way that fathers typically approach “negative issues” stating, “If there is a negative issue that needs to be dealt with … he will just brush it off and say, ‘I don’t want to hear about it.’”

Fathers often expressed early hesitation to participating in EI. This hesitation appeared to have to do with uncertainty about the intervention process itself as well as questions about whether their child “needed” intervention. One father said, “I didn’t want to do it at first … I didn’t want to have to deal with people coming into the house and having to cater to somebody else.” He went on to say, “I really just didn’t understand why we had to do this anyway.” Another father described a change in his perception of his son’s need for interventions stating, “I was like he’s eventually, it’s just gonna pop, it’s just gonna break. I was more delusional than she was …”

In many cases, it took both parents being “on board” with the desire to seek out services before pursuing services in the community or participating in the treatment phase of the study. Mothers often described being in the role of advocating for their child by having to convince the fathers that the intervention or study could only benefit the child. One father said, “As soon as she (mother) explained to me that it couldn’t hurt, and its free help, then I thought, Why not?”

The personal relationship with the interventionists in the ART group in particular appeared to play a key role and contributed specifically to fathers’ buy-in to intervention. One father who reported feeling “hesitant” early in the intervention process later described his observation of the relationship between his son and the interventionist. He stated, “… you could tell like she just truly and honestly cared about him, like he was her own … she could just read him like it was nobody’s business … we hated when she left.” Another father who said that in the beginning he did not want to have to deal with people coming into the house went on to say, “Once we started, it was awesome … ’cause just watching him interact with (interventionist) and seeing how good he was …” When asked about his intervention experience later in the interview, this father said, “I think for me it just felt normal. Like, that he enjoyed somebody else’s company.”

Positive experiences and burdens

Parents in both groups revealed both positive and negative experiences within each of the interview topics. Parents reported such positive aspects of participation as developing relationships with EI professionals, feelings of parenting support (e.g. feelings that the team was available to the family to respond to any concerns or needs that arose), as well as increased knowledge of and access to resources. Parents also described some negative aspects of participation, which included the burden of travel to evaluations, anxiety surrounding evaluation outcomes, and making time for intervention. Parents offered some insights into ways in which these burdens could be lessened, to include conducting evaluations in the families’ homes as well as connecting parents with each other in order to “talk to other families going through it.” Parents also verbalized that some of the burdens they experienced were mitigated by strategies used by the study. For example, intervention times were offered that would optimally fit in with the busy lives of the families to include evenings and weekends. Parents also mentioned that their anxiety surrounding evaluation outcomes was tempered by availability and sensitivity of the team during and following the evaluation experience.

Unique theme within the ART treatment group

When asked about the randomization experience, parents in both groups unanimously expressed hope for the ART treatment group. Therefore, those who were randomized into the ART treatment group expressed a feeling of Win-win when describing their experience of being randomized and participating in the intervention. The essence of this theme was that parents described their view of treatment and intervention as overwhelmingly positive and felt that there was no harm in getting help. Parents also often expressed a sense of relief that came with decreased parent responsibility in seeking out services for their child. In other words, since the child was randomized into the “treatment” group and the family was participating in intervention as provided by the study, parents described feeling relieved that they were not in a position of having to navigate CS or left to determine the types of services that their child needed. Even those families who expressed minimal concerns at the time of the initial evaluation described the intervention experience as positive. Parents described a range of reasons why they viewed treatment as a “Win-win.” For example, one mother stated, “There is nothing but a positive to be involved with something like this.”

Families randomized into the ART group often reported a sense of hope knowing that their child would receive intervention. One mother described feeling hopeful about the intervention stating, “I thought, this might just work for (child).” The father agreed and said, “Yeah, it felt like the cards fell in line …” Other parents described a sense of hope that came from being randomized into the treatment group. For example, one mother said, “I just knew she would make progress, and it would help us too.”

Another mother who had minimal concerns at the time of the initial assessment reported seeing the intervention as helping to close a gap as well as giving her son an edge as he prepares for school. This mother stated, “Whether he has an issue or not, they’re offering us a chance to try to get early intervention.”

Unique theme within the CS group

One theme represented the data in the CS group and was named Navigating amidst ambiguity. Parents who were randomized into the CS group reported challenges in having an increased responsibility to seek out services or “do the legwork.” Along with this challenge came reported stress and strain as well as ambiguity in determining what services their child qualified for and then coordinating these services.

Parents reported having to research, advocate, and basically become service coordinators based on their child’s needs. One mother stated, “I did some research. I knew we were entitled to it. And as a parent you can fight. You’re going to fight- be your child’s biggest advocate.” Another mother described her struggle with coordinating and having consistent therapists for her son. She said, “He would have to switch around therapists … and at that point, I couldn’t quite tell how he would do with new people.” Another mother described her concern surrounding having multiple therapists in the home saying, “We were concerned about overloading him with three areas and three people.” Another mother described her first encounter with CS that she pursued following a referral from a specialty clinic. She said, “They referred us to a facility that does in-home therapy. We used them for a while, but I didn’t care for them too much. And they were unreliable and hard to schedule, so I switched.” Another mother described her lack of awareness surrounding EI services prior to participating in the study stating, “I didn’t know there were services for that young … I didn’t know there was anything up until he started school.”

Parents also described their motivation to navigate and participate in CS as impacted by their level of concern about the child’s delays. In addition, some differences in experiences were reported from family to family, which often resulted from variations in “severity” of their child’s symptoms, level of parent concern, the county in which they resided, and subsequent availability and quality of services.

One mother described the benefit of participating in the RCT, even though the mothers were randomized into the “control” group, stating, “Even though we were randomized into the no treatment group, I would have probably not realized what was available to us.” Parents also reported that having the diagnostic information in particular from the EDP study accelerated the process of getting services started for their child.

Discussion

This novel study illuminates the personal experiences and contextual influences affecting families who participated in a specific RCT and begins to explicate the myriad of benefits and burdens associated with early identification of “risk” status and subsequent intervention for ASD in very young infants. Four themes represented the data across groups, suggesting some continuity of experience between the ART and the CS groups. There was also a unique theme that best represented the data within each of the randomized groups.

Caregivers described retrospectively that participation in the RCT as a means for Getting the ball rolling or seeking out services for their child during a time when they felt there was a delay or something “not quite right.” Interestingly, at the time of screening, some parents described feeling “surprised” that their child was determined to be “at risk” of ASD, which calls into question whether these families would have chosen to seek services early if they had not participated in the RCT. For those parents who did express concerns prior to participating in the study, however, they often questioned the accuracy of their concerns based on a lack of validation from their partners, pediatricians, or extended family members. This finding highlights the need for practitioners to attend and respond to early parent concerns (Johnson et al., 2007).

In many cases, it took both parents being “on board” with the desire to seek out services before pursuing services or agreeing to participate in the treatment phase of the study. This is particularly disconcerting as mothers in this study illuminated their challenges in Getting dad on board. These findings highlight the importance of communicating with and understanding the perspective of both mothers and fathers. While research has identified elevated stress in fathers who have a child with ASD as compared to father’s of children with other DDs (Baker-Ericzen et al., 2005), little is known about the early experiences of fathers and their perceptions surrounding early screening, intervention, or diagnosis.

Personal relationships appeared to be vital across both groups as they contributed to uptake of services, getting the father’s buy-in, overall intervention experience, as well as encounters with the community. The quality of the relationship, as described by the parents, appeared to have a tremendous impact on the families’ experiences, regardless of the team member’s role. Parents in the ART group talked about the assessment team, the project coordinator, and the interventionist, while the parents in the CS group mentioned the case managers, the therapists, and the project coordinator. Overall, the importance of developing personal relationships with families early in the research or intervention process should not be underestimated. This relationship appears to be grounded in both having consistent points of contact, as well as evaluators and therapists who show empathy toward parents’ experiences and a genuine interest in and connection with the child. This finding supports Bishop et al.’s (2007) finding that a lack of adequate professional support is one of the most significant causes of stress for parents of a child with autism.

Although there were themes that emerged across groups, the unique themes that emerged within groups suggest that the experiences of ART versus CS families can be differentiated. All families expressed hope for the ART treatment group, and those randomized to this group viewed ART intervention as a positive experience regardless of level of early concerns. Participation in the ART group appeared to come with a perceived decreased parent responsibility as far as coordination of services and navigation of the EI system. Furthermore, participation in the ART group took out the “decision” of whether or not to pursue services, and parents did not face the same dilemma in evaluating their level of concerns as a motivator for seeking out services. Despite their level of concern, parents described being invested in the ART treatment and described it as a positive overall experience.

Parents in the CS group described being in a position of navigating CS in the context of ambiguity and uncertainty. Compared to the ART group, the CS group was not provided with a “treatment” consisting of the ART intervention. Although they received resource and referral guidance as participants in the EDP study, parents still described feeling “uncertain” about the process of establishing services and reported burden surrounding having to initiate and coordinate these services. This finding supports Wachtel and Carter’s (2008) study that found that despite exposure to service options, parents often feel that it is ultimately their job to make treatment decisions and coordinate services for their child.

Parents described both level of concern and knowledge about CS as impacting their experience and decision-making process. This finding highlights a potential implication for EI services for those parents who have not yet had anyone identify their child as needing services or who have limited knowledge surrounding CS (e.g. whether their child would qualify and what types of services would be most appropriate for their child). Experiences described by the CS group may be generalizable to parents who do have early concerns about their child but are uncertain about the resources that are available to their child or even how to access these services. This is problematic considering the evidence for beginning EI services early on and the assumption that ambiguity and uncertainty might lead parents to shy away from initial screenings or, in more extreme cases, to not seek out any type of CS at all. This is further disconcerting in light of the finding that parents in this study did not feel validated for their concerns by their pediatricians who may otherwise serve to connect them to these resources.

These findings highlight previously identified parent perceptions of the lack of adequate training of professionals (Osborne and Reed, 2008) as well as reported negative experiences with primary care providers on several aspects of care (Liptak et al., 2006) for those parents who have children with autism as well as children with other DDs. This information also supports prior findings that parents often report being frustrated by the delayed and disjointed diagnostic process, particularly in the early stages of diagnosis (Mansell and Morris, 2004; Osborne and Reed, 2008).

Parents in both groups revealed both positive and negative experiences surrounding various phases of the RCT. Parents reported such positive aspects of participation as developing relationships with EI professionals, feelings of parenting support, as well as increased knowledge of and access to resources. Parents also described some negative aspects of participation, which included the burden of travel to evaluations, anxiety surrounding assessment outcomes, and making time for intervention.

Overall, this study identified that families behaved differently following randomization in an RCT. For example, parents may or may not choose to participate in CS, and their perceptions of what is available and beneficial for their child regarding EI may vary based on their exposure to resources as a result of participating in an RCT. Furthermore, stigma surrounding “risk” status or diagnosis might be ameliorated with increased knowledge about “risk” and ASD as well as education surrounding child outcomes following intervention. This finding has implications for other RCTs of interventions for children at risk of or diagnosed with disabilities as well. There are a number of decision-making time points that occur through such a trial, and at each point, caregivers must make choices that are based on their own beliefs, level of concern for their child’s development, knowledge of the potential diagnosis and outcomes, and other contextual influences, such as both parents being on board and life circumstances like knowledge about services and feasibility of accessing these services.

Limitations

Participants in this study consisted of a small convenience sample. Furthermore, participants were recruited based on their self-selection to initially complete the FYI and were largely White/non-Hispanic and middle class, thus limiting the diversity in this analysis. Therefore, additional or different themes may have emerged from a more diverse sample. The retrospective nature of the interviews should be noted, as parent recall bias may impact “accuracy” of factual details and changes in perceptions in hindsight. In addition, we are uncertain of how comfortable all families were with sharing negative experiences, given that they may have been concerned about negative comments being linked back to them by members of the EDP team they had interacted with during the study. Finally, the current study could have employed such qualitative strategies as triangulation of data or developing individual impressions prior to group meetings in order to further strengthen the trustworthiness of our findings.

Future directions

Qualitative research provides a unique perspective that may help researchers and practitioners to better incorporate the voices of parents who make treatment decisions and who play a vital role in intervention quality and efficacy. There remains a need for longitudinal prospective studies of family experiences and decision-making processes in the realm of EI and autism. Future studies could expand these interviews to prospective methods having the potential to identify more specific moderators of child outcomes in the context of EI models. In addition, consideration of other contextual variables that may impact experiences is needed, as well as further analysis of variation in family behaviors as a consequence of randomization and subsequent effects on outcomes.

Future studies should also consider the differences in experiences for those families whose children actually go on to receive a diagnosis of autism versus those who do not. The focus of the current study was to explore the retrospective experience of families as they participated in an RCT; therefore, we did not specifically ask parents about how having an ASD diagnosis impacted their experience. The clinical data from the larger EDP study revealed that three of the ART group families (3/10) who consented to interviews had children who were given a diagnosis of ASD. In contrast, one of the CS group families (1/4) interviewed had a child who received an ASD diagnosis. Therefore, we were unable to systematically analyze similarities and differences in experiences between these two subgroups. Larger longitudinal, prospective studies could further illuminate the impact of the diagnosis experience on the decision-making process surrounding uptake of services, the evolving perception of “risk” status over time, as well as overall experience of participation in an RCT.

Incorporating qualitative methods into EI studies that target quantitatively measured child outcomes may also create opportunities for mixed methods, which could link families’ experiences with child/family outcomes in RCTs. Information gained from these interviews may serve to refine EI models, enhance families’ intervention experiences, and improve child outcomes. Overall, research is needed that considers the various perceptions and resources with which parents respond to the pressures and opportunities associated with participation in EI, particularly for infants who are at risk of ASD but do not (yet) have a diagnosed condition.

Footnotes

Acknowledgements

We would like to thank Dr Virginia Dickie for her consultation with regard to qualitative methods and construction of interview questions.

Declaration of conflicting interests

The authors declare that there is no conflict of interest.

Funding

This study was partially funded by Autism Speaks and The Ireland Family Foundation/ Program in Early Autism Research Leadership and Service (PEARLS) Advancement Fund.

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