Malavi Srikar, Reny Raju, Divya Swaminathan, Rachel Elizabeth Johnson, [Linda R. Watson](https://onlinelibrary.wiley.com/authored-by/Watson/Linda+R.), Deepa Bhat Nair, John Vijay Sagar Kommu, Jo Chopra-McGowan, [Prathyusha P. Vasuki](https://onlinelibrary.wiley.com/authored-by/Vasuki/Prathyusha+P.)
Shoba S. Meera and Malavi Srikar contributed equally to this work.
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Preliminary evidence indicates potential benefit of providing caregiver-mediated intervention, prior to diagnosis, for infants at elevated familial likelihood for autism and related developmental delays including language delay (EL-A). However, delivering such interventions online and in low-resource settings like India has not been reported. This study aimed to evaluate the feasibility and acceptability of delivering a novel manualized caregiver-mediated early support program, the “LiL' STEPS,” online in India, for EL-A infants. LiL' STEPS stands for Language development & Intervention Lab's (LiL') Supporting Early social-communication and language by Promoting caregiver Sensitive responsiveness (STEPS). The program comprised 14 sessions with a focus on social-communication and language, conducted over 12-weeks using demonstration and video feedback. Families of 36 EL-A infants aged 9 to 15-months participated in this feasibility randomized controlled trial (RCT). Families were randomized in a 2:1 ratio (n = 24 LiL' STEPS and n = 12 care as usual groups). Information on feasibility and acceptability was collated following a mixed methods approach from caregiver interviews, fidelity forms, session notes, and study register. Findings indicated the LiL' STEPS study trial as feasible and acceptable with recruitment rate of 4 per month, 100% willingness for randomization, 8.3% attrition, and 3.03% loss of blinding. Interventionist and caregiver fidelity was maintained above 80%. Despite challenges like interruptions during sessions, 100% families found the program acceptable and satisfactory, 86% said they would recommend the program to others, and 71% preferred online modality. Caregivers' perspectives on beneficial components and experience attending the program have been described. Accordingly, recommendations for future definitive RCTs have been presented.
Early intervention is known to have positive long-term outcomes in autistic children (e.g., Landa & Kalb, 2012). Although infants can be identified as autistic by 18-months (American Psychological Association, 2017), globally an autism diagnosis is being made anywhere between the ages of 30 and 74 months for children less than 10-years of age (van't Hof et al., 2020). This delay in diagnosis is more in some parts of the world than others. For instance, a study conducted in India reported the average age of first consultation to be 32.5 months (Kandaswamy et al., 2017). One of the reasons for this delay may be attributed to lack of routine screening protocols for autism in India especially for infant-toddlers less than 18-months of age. Hence, many children miss out on the crucial window for early intervention. However, globally, there has recently been a shift toward providing prodromal intervention very early in life, that is, before a “formal diagnosis” is made (Green, 2022; Grzadzinski et al., 2021; Whitehouse, 2023). One group of infants who may benefit from prodromal intervention are infants at elevated familial likelihood for autism and related developmental delays (EL-A infants), that is, younger siblings of autistic children. About 20% of EL-A infants are at an elevated familial likelihood for autism (Ozonoff et al., 2015) and another 20%–30% may demonstrate related developmental delays (Constantino et al., 2010; Georgiades et al., 2013) including language delay (Swanson et al., 2017). Indeed, preliminary evidence suggests that providing intervention prior to formal diagnosis could benefit EL-A infants (Hampton & Rodriguez, 2021; Landa, 2018). In this paper, we refer to such interventions as “early support programs.”
The role of these early support programs is supported by several theories that view infant development dynamically within the context of their social environment. For instance, the “development as adaptation” framework considers early behavioral phenotypes of autism to be a combination of genetic predisposition and the individual's interactions with the environment (Grzadzinski et al., 2021). The “interactive specialization” theory, for instance, postulates that early experiences may influence the development of the “social brain” by sharpening neural connections in activity-dependent interactions between brain regions (Johnson, 2001). In his social interactionist theory, Vygotsky (1978) emphasized the role of social interaction (to-and-fro) between a child and an adult for language development. Similarly, the “social feedback loop” indicates that caregivers' contingent responses to their infants' vocalizations support their infants' language development and communication (Warlaumont et al., 2014). In addition, these theories indicate caregivers' important role in infants' early social environment, making caregivers integral to early support programs.
Hence, most early support programs tend to be caregiver-mediated (Hampton & Rodriguez, 2021). Here, caregivers are taught strategies to support their infant-toddlers' development by making specific changes in how they interact with them. Strategies include modifying aspects of infant-toddlers' social environment such as caregiver-infant dyadic interaction (Green et al., 2015; Whitehouse et al., 2019), caregiver responsiveness (Davis et al., 2022; Kasari et al., 2014; Watson et al., 2017), or setting up joint action routines (Yoder et al., 2021). These programs were found to promote infant-toddlers' proximal skills such as attentiveness (Green et al., 2015), communication intent and motor imitation (Yoder et al., 2021), vocal responsiveness (Brian et al., 2017), or receptive and expressive language (Kasari et al., 2014). Various methods for delivering early support programs include video feedback (Green et al., 2015; Whitehouse et al., 2019), parent coaching (Watson et al., 2017; Yoder et al., 2021), or live coaching (Brian et al., 2017). For instance, during video feedback, videos of caregiver-infant play interactions are jointly viewed by caregivers and a professional. Caregivers then receive feedback while observing their behavior and their infant's behavior in a reflective context (Aldred et al., 2018; Poslawsky et al., 2014). Some programs use these methods in combination with demonstration of strategies (Kasari et al., 2014; Tanner & Dounavi, 2020) for more hands-on training. However, majority of the literature for the EL-A group stems from English-speaking, high-resource settings. Hence, there is a need for early support programs suitable for EL-A infants in a culturally diverse, low-resource setting like India.
Most of these early support programs have been conducted in-person. However, there is growing evidence for caregiver-mediated interventions delivered online for older autistic children, with studies reporting caregiver acceptability and self-efficacy (Pan et al., 2023; Sengupta et al., 2023). Employing an online modality can help overcome geographical barriers (Parsons et al., 2017), cut costs, and increase families' access to services, particularly in underserved or rural areas (Little et al., 2018). Further, long-term positive outcomes resulting from early support programs could potentially reduce net costs and strain on resources in the long run (Segal et al., 2023). This could particularly be beneficial for a country like India which is densely populated (The World Bank, 2022) with limited resources leading to a treatment-gap (Patel et al., 2013). With a rise in smartphone users and access to cellular internet across the country, India too seems to be equipped for an online modality of service delivery (Mohan et al., 2017). However, to the best of our knowledge, in India or globally, there are no published studies on early support programs delivered online for EL-A infants. Hence, there is merit in describing and evaluating an early support program delivered online for EL-A infants, in the context of India.
Though a definitive randomized controlled trial (RCT) is considered the gold standard to evaluate the efficacy of an intervention/early support program, it is resource demanding with a stringent protocol to promote scientific rigor. Feasibility trials, on the other hand, provide information regarding the practicality of conducting a program in terms of the process including willingness to randomization, resources such as equipment, management involving documentation, and scientific basis of the trial while offering some flexibility (Tickle-Degnen, 2013). Additionally, there is merit in systematically examining caregivers' perspectives on the program being delivered such as specific strategies covered and structure of the program. Hence, prior to conducting a large-scale definitive RCT, assessing the feasibility of conducting a novel program and/or in a novel setting forms the first step (Eldridge et al., 2016).
In this study, we have aimed to design a novel, socio-culturally informed, manualized caregiver-mediated early support program for EL-A infants in India– the LiL' STEPS1 with a focus on social communication and language. Designing our own program, provided flexibility to shape it based on input/feedback from professionals and stakeholders from our socio-cultural background in terms of (a) core content based on theoretical support, previous work done (e.g., Green et al., 2015; Yoder et al., 2021), and clinical experience of the research team (e.g., components of social communication and language), (b) method (e.g., demonstration with culturally relevant toys/activities and video feedback), (c) structure (e.g., intensity/pace suitable to EL-A families in India), and (d) mode of delivery (e.g., online modality to overcome geographical barriers and travel costs to increase reach across the country). Thus, in the present study, we evaluated the feasibility and acceptability of delivering the LiL' STEPS program online in India. The objectives of this study were to systematically evaluate the (a) feasibility and acceptability of the LiL' STEPS study trial in terms of achievable recruitment rate, willingness for randomization, attrition, and loss of blinding, (b) feasibility of conducting the LiL' STEPS program online in terms of interventionist and caregiver fidelity, and (c) acceptability of the LiL' STEPS program in terms of overall caregiver satisfaction, recommendation to other families, and preference for online modality.
The study was an assessor-blinded, parallel, feasibility randomized controlled trial conducted at the National Institute of Mental Health and Neurosciences, a tertiary care hospital in India. Families were invited to participate if the following inclusion criteria were met: infants aged 9 to 15-months having an older sibling with a community diagnosis of autism; participating caregiver(s) comfortable communicating and reading in at least one of the following languages: English, Kannada, and/or Hindi; access to basic technology that supported video conferencing including a device (e.g., laptop, smartphone) and adequate internet connectivity (at least 3G cellular internet). Families were required to identify at least one caregiver who would consistently attend the program. Since there are no robust screening tools for autism that have been validated for infants less than 18-months in the Indian context, all infants at familial likelihood for autism were included based on caregivers' willingness to participate rather than identification of behavioral indicators for autism. Exclusion criteria included: infants with any significant pre-peri-post-natal medical history, birth weight <2000 g, gestational age <37 weeks, infants who were twins, adopted or half-siblings, infants having first-degree relatives diagnosed with schizophrenia, psychosis, or bipolar affective disorder, and participating caregivers with uncorrected sensory impairments (e.g., vision, hearing). Families residing pan India were recruited as this was an online study. Participants were reimbursed for their participation. Families who did not have access to Wi-Fi were reimbursed for getting additional cell phone internet packs.