This is a multi-centric study that will be carried out in Child and Adolescent Mental Health Services from three Hospitals in Spain: Hospital Materno-Infantil in Málaga (HMIM), Hospital General Universitario Gregorio Marañón in Madrid (HGUGM) and Hospital Sant Joan de Déu in Barcelona (HSJD). Eligible participants will be parents of children diagnosed with ASD and parents of preterm children with communication and/or socialization difficulties.
Sample
The IY-ASLD® program will be implemented in Child and Adolescent Mental Health Services in each site. In two of them (HMIM and HGUGM) parents of children with ASD will be recruited (n=48) and in one site (HSJD) parents of preterm children with communication and/or socialization difficulties will be included (n=24). Participating families will be randomly allocated to the TAU condition or to the intervention group within each site.
For the sample size calculation, we used the outcome Parental Stress, measured through the questionnaire Parental Stress Index Short Form (PSI-SF) (36). This is a 36-item scale, with a range of 180 points. There is data showing that a decrease of 16.5 points in the total scale stress score can be seen after attending an IY program (37). Given that this study will be conducted with parents of children presenting neurodevelopmental difficulties, we anticipated that the decrease in the PSI-SF score would be lower. We estimated the necessary sample size considering a power of 80%, α = 0.05, a difference between pre and post-test of 10 points, and a standard deviation of 20 (38). We estimated 34 participants needed per arm of the study. Thus, we aim to recruit approximately 70 participants. Also, a sample of approximately 70 participants has been recommended in similar feasibility studies (39).
Eligibility criteria
Inclusion criteria:
1. a. HMlM and HUGM sites: parents/caregivers of children diagnosed with ASD (clinical diagnosis performed by psychiatrists or clinical psychologists in the service, based on DSM-V diagnostic criteria).
b. HSJD site: parents/caregivers of preterm children (<37 weeks of gestational age) with communication and/or socialization difficulties (defined as Vineland-III scores below 1SD in any of the communication or socialization subdomains).
2. Children aged 2-5 years.3.
3. Parents/caregivers showing good understanding of Spanish language.
4. Parents/caregivers consenting to take part in the study and signing the informed consent.
Exclusion criteria:
- Attending another structured parenting program (focused on improving parental strategies to help their children with their developmental or regulation difficulties) during the intervention phase of the study.
- Looked-after children.
Recruitment
Parents/caregivers will be recruited between January and February 2020 within the Child and Adolescent Mental Health Services of each hospital site, where patients have previously been assisted for their neurodevelopmental difficulties (Figure 1). A leaflet with inclusion and exclusion criteria will be handed out to clinicians working in the services. Clinicians will be asked to discuss the study with eligible families and will ask the families permission to be contacted by study researchers. If they agree to be contacted about the project, a research assistant will call the family to discuss the study further. If the family is interested in participating in the study and meets inclusion criteria, an appointment will be made with parents to sign the informed consent and to complete the pre-intervention assessment. During this visit, researchers will make sure that participants receive all the necessary information and have the opportunity to ask any questions. Participants will be able to discontinue the treatment sessions or drop out from the control group at any point at their request.
Randomization
After completing baseline measures, families taking part in the study will be randomly allocated to the intervention group or to the TAU condition within each center. Randomization will be conducted using the Program R 3.6.2 (R Foundation for Statistical Computing), generating a replicable process fixing a random seed. Allocation will be stratified by developmental level.
An independent researcher will carry out the randomization process, and researchers who are responsible for patient recruitment or for intervention delivery will not be involved. Due to the pragmatic and clinically focused nature of the study, further blinding procedures will not be possible.
Intervention
The IY-ASLD® program is a 14-session group-based intervention for parents of children presenting neurodevelopmental difficulties. One of the main principles of the intervention is the collaborative approach, promoting parents’ discussion around the topics of interest and facilitating a solution-based perspective. The intervention includes video modeling and emphasizes the importance of practice-based learning through role-play. The IY-ASLD® program takes into consideration the different developmental level of each children and pairs parents according to this variable in role-play and other one-to-one discussions. Weekly home tasks will be assigned to parents and families will be phoned each week to encourage home-based practice (32).
Fidelity to the treatment manual will be ensured in different ways. Group interventions will be conducted by experimented clinicians (Child and Adolescent Psychiatrists and Clinical Psychologists), officially trained in the IY-ASLD® model by an accredited trainer. Group leaders will complete weekly checklists regarding fidelity to the intervention and participants will also complete weekly parent evaluations. Group leaders will be supervised by certified supervisors and will pursue accreditation according to the IY-ASLD® program standards.
Measures (Table 1).
Baseline and sample descriptors
At baseline, the following sociodemographic and child clinical variables will be assessed: family structure, child's age, gender, medical conditions, type and date of psychiatric diagnosis, psychotropic medication, educational status, current and past mental health interventions for parents and for the child and neurodevelopmental problems in siblings. Socioeconomic status will be determined using the Hollingdale’s Index of Social Position (40).
At baseline, the following questionnaires will be administered:
The Modified Checklist for Autism in Toddlers-Revised with Follow-up (M-CHAT-R/F). (41). This tool is a parent-reported 20-item questionnaire screening ASD symptoms in children aged 24-30 months. This instrument will be administered before the intervention to describe children’s social communication difficulties. The Spanish translation has shown valid and reliable results.
Social Communication Questionnaire (SCQ). (42). It is a 40-item parent report measure, yes/no format, based on the Autism Diagnostic Interview-Revised (ADI-R) (43). The Lifetime version of this questionnaire will be administered before the intervention with the same aim as the M-CHAT-R, in children aged between 30 months and 5 years. It is a robust tool that has shown good validity, and it has been widely adopted by both the research and clinical community.
Vineland Adaptive Behavior Scale-III (VABS-III, parent/caregiver report form) (44). It assesses adaptive functioning in different areas: communication (receptive, expressive, written), socialization (interpersonal/play and leisure time/coping), daily living skills (personal, domestic, community) and motor skills (fine and gross). It also generates a final adaptive composite score. This instrument will be used before the intervention to collect the level of functioning in different developmental areas. It has been considered a very efficient tool to measure the adaptive behavior profile of preschool children with developmental problems and shown an excellent test-retest reliability ) (45).
Developmental Profile-3 (DP-3) (46). This is a 180-item parental questionnaire assessing developmental delays in different domains: motor, adaptative, socio-emotional, cognitive, and communication. It also computes an overall general development score. It will be used before the intervention to collect the child developmental level. It has shown good internal consistency.
Primary outcomes (feasibility)
Parents’ engagement with the program and participant retention will be monitored throughout, expecting they will attend at least 8/14 sessions with a minimum of 6 parents out of 12 (>50%) finishing the program.
Autism Program Parent Weekly Evaluation (47). This instrument is part of the IY-ASLD® program materials and will be used to collect information regarding compliance and satisfaction throughout the study.
Autism Program Parent Final Satisfaction Questionnaire (47). This questionnaire is included within the IY-ASLD® program. It will be used to measure the acceptability and satisfaction with the intervention after the last session. It covers five areas: (1) the overall program; (2) usefulness of teaching format; (3) usefulness of specific teaching strategies; (4) evaluation of the group facilitators and (5) the parent group.
Implementation fidelity will be ensured and monitored throughout the intervention process, following the strategies described above.
Secondary outcomes (preliminary effectiveness for parental outcomes)
ASD-P Parent Strategies Questionnaire (47). This is a 60-item questionnaire included within the IY-ASLD® program. It is divided into 5 subdomains regarding different strategies that will be learned throughout the program to promote social, emotional, language and academic development, and how often parents use these strategies. This tool will be administered before and after the program implementation.
Parent Stress Inventory-Short Form (PSI-SF) (36). This is a 36-item questionnaire that specifically focuses on assessing parental stress associated with the care of their offspring. It has three domains: Parental Distress, Parent-Child Dysfunctional Interaction, and Difficult Child, which combine to form a Total Stress scale. This tool will be administered before and after the intervention. It has shown good internal consistency.
Beck Depression Inventory (BDI) (48). This is a 21-item screening tool assessing the severity of depressive symptoms. It is a standardized and validated questionnaire, often used in mood disorder assessments. It will be collected before and after the intervention. It has good reliability.
Alabama Parenting Questionnaire-Preschool revision (APQ-Pr). (49). This is a 32-item parent-reported questionnaire measuring parenting practices that are consistently associated with disruptive child behaviors. This version has 3 dimensions: Positive Parenting, Inconsistent Parenting and Punitive Parenting. It will be collected before and after the intervention. This measure has shown good internal consistency and validity.
Autism-Specific Five Minute Speech Sample (AS-FMFSS). (50) This is a narrative 5-minute interview used to measure parental expressed emotions for children with ASD and related disorders. Parents are asked to speak about their child and the parent-child relationship (51) - “I’d like you to speak for 5 minutes, telling me what kind of person (child name) is and how the two of you have got along together over the past 6 months.” Speech samples are audiotaped, transcribed, and coded following four global categories (a) Initial Statement, (b) Warmth, (c) Relationship, (d) Emotional Over-Involvement and critical comments (e) and positive comments (f). Expressed emotions will be measured before and after the intervention. Benson et al. (51), assessed 30 randomly selected speech sample by three different raters. Inter-rater reliability and code-recode reliability on two separate occasions, for all six AS-FMSS components and for total EE score were both in the good to excellent range.
Other outcomes (preliminary effectiveness for child outcomes)
Child Behavior Checklist (CBCL 1.5-5) (52). This is a parent-reported 99-item inventory that addresses specific externalizing and internalizing behaviors. The sum of scores form a total problems score and it also includes scores for externalizing and internalizing difficulties subdomains. It will be collected before and after the intervention. It has shown good internal consistency, also in children diagnosed with ASD (53).
Data collection
Data will be collected at baseline (before performing randomization), and after finishing the IY-ASLD® intervention. Parents consenting to participate (mother, father or both) will be offered a hospital appointment before and after the intervention with a research assistant to complete the assessment, including self-report measures, children outcome measures, and a voice recording for the ASFMSS tool. Those parents who discontinue or deviate from the intervention protocols will receive a telephone call by the research assistant to arrange further hospital appointments or to check if they are willing to complete the questionnaires for the main outcomes on the phone.
The timeline for participants' enrollment, data collection and the conduction of the intervention are scheduled in Table 1.
Data analysis
Descriptive analysis will be used for primary outcomes (e.g. recruitment rates, number of sessions attended by participants, proportion attending at least 8 out of 14 sessions, and proportion finishing the program). Attrition and satisfaction rates with the program will be also described.
Differences in baseline and descriptive variables between intervention and TAU conditions will be explored. Differences between-site will also be analyzed. These variables will be controlled for in the main analyses.
For secondary outcomes, analysis will follow an intention-to-treat basis. Differences between treatment groups in parental levels of stress, expressed emotions and parenting practices will be analyzed using general linear models, controlling for important confounding variables, such as socio-demographic variables, severity of symptoms at baseline or treatment site. Further secondary analysis will use similar methods to compare groups for additional child outcomes (i.e. CBCL externalizing problems).
Missing data (i.e. missing item responses in questionnaires) will be treated following the instructions stipulated in the questionnaire's manual. Imputation methods will be considered for full-case missing data. Analysis will be conducted using SPSS/STATA statistical packages.
Trial Status
The protocol for the current pilot study was registered in ClinicalTrials.gov (ID number: NCT04358484. Unique Protocol ID: PIC-220-19) and it was released in April 2020. The study began on January 2020, and the anticipated finish date was September 2020. The recruitment process was carried out during the same period in the 3 sites, from 3rd of February to 2nd of March.
ETHICS
The study has received ethical approval from the Ethics Committee at each site. The trial data set will be stored in institutional encrypted computers and will itself be encrypted with a password. Only staff involved in the study will be authorized to access the data. Participants will be informed about all protection data procedures and will be able to disengage from the study at any point, without affecting their usual clinical care. Written information regarding these aspects will be handed, and contact details from research assistants will be offered in order to clarify any doubts or worries participants might have.
Participants will be informed of potential benefits and risks of participating in the study. Due to the nature of the study, it is unlikely that taking part in the research will result in any damage. After families are provided with sufficient information about the project, they will be asked to sign the written informed consent.
The study will be conducted following the principles developed by the World Medical Association, outlined in the Declaration of Helsinki.