Study of an Early Parenting Intervention for Children With Genetic Abnormalities and Mental Health Problems (The GAP)

Early Treatment for Children With Mental Health Problems and Genetic Abnormalities Through a Parenting Intervention (The GAP): A Pragmatic Randomized Controlled Trial

The GAP study is a randomized controlled trial that aims to determine the feasibility and efficacy of the "Incredible Years Autism Spectrum and Language Delays" (IY-ASLD®) intervention for families of children with developmental problems from a genetic basis. It is a multicentric trial where families will randomly be assigned to the intervention group or to a control group (they will follow their usual treatment). The intervention will be carried out in an online format, and it will involve 22 weekly group sessions. The results of The GAP study will help clinicians and policy makers in guiding towards evidence-based treatment options for these particularly vulnerable group of infants.

Study Overview

• Status: Recruiting
• Conditions: Parenting; Genetic Syndrome; Language Development; Autism or Autistic Traits
• Intervention / Treatment: Behavioral: Incredible Years Autism Spectrum and Language Delays Parent Program (IY-ASLD®)

Detailed Description

Children born with genetic abnormalities have higher risk of presenting developmental and mental health problems. Their parents tend to express higher anxiety levels, as parenting children with developmental problems can be a stressful challenge for many families. These infants can present important behaviour, emotion regulation and social interaction problems. These problems usually have a complex clinical presentation determined by their genetic abnormality. Despite many of their impairing symptoms can overlap with the characteristics of the Autism Spectrum Disorder (ASD), in many cases they do not meet criteria for a full-blown ASD diagnosis. These patients are placed in a double disadvantaged situation, as on top of suffering a genetic disease, they do not usually enter the ASD care-pathways and do not have access to effective therapies for their impairing symptoms. This problem should be addressed, as there is wide scientific evidence on early parenting programs that improve infants' developmental problems and parents' mental health. These parenting programs are structured interventions that provide parenting skills to improve the acquisition of children's social and emotion regulation abilities. Such interventions have been widely implemented internationally, but unfortunately, most families treated within our mental health system still do not have access to them. A good example is the parenting program "Incredible Years Autism Spectrum and Language Delays (IY-ASLD®)", focused on improving young children's development and parental stress levels. Recently, the feasibility and acceptability of implementing this program in different areas of the national territory has been studied, with high satisfaction levels expressed by participant families (parents of children with language delays or ASD). However, the scientific evidence is very limited for children with developmental problems from a genetic basis, particularly for those with dysfunctional symptoms' that do not meet diagnostic threshold for an ASD diagnosis.

The GAP study aims to determine, for the first time, the feasibility and efficacy of the IY-ASLD® intervention for families of children with developmental problems from a genetic basis. It is a multicentric randomized controlled trial where families will randomly be assigned to the intervention group or to a control group (they will follow their usual treatment). As genetic abnormalities are rare, clinicians will carry out the intervention in an online format (22 weekly sessions), reaching out all affected families that live scattered throughout the national territory. The results of The GAP study will help clinicians and policy makers in guiding towards evidence-based treatment options for these particularly vulnerable group of infants.

• Study Type: Interventional
• Enrollment (Estimated): 68
• Phase: Not Applicable

Contacts and Locations

• Study Contact: Name: Laia Villalta, MD, PhD; Phone Number: +34936009751; Email: laia.villalta@sjd.es

Study Locations

• Spain
  • Barcelona, Spain
    • Recruiting
    • Hospital Universitari Vall d'Hebron
    • Contact: Anna Maria Cueto-González
  • Sabadell, Spain
    • Recruiting
    • Hospital Universitari Parc Tauli
    • Contact: Carmen Manso, MD
  • Barcelona
    • Esplugues De Llobregat, Barcelona, Spain, 08950
      • Recruiting
      • Sant Joan de Deu
      • Contact: Laia Villalta, MD, PhD; Phone Number: +34936009751; Email: laia.villalta@sjd.es

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: Child
• Accepts Healthy Volunteers: No

Description

Inclusion Criteria:

1. Children aged 3.0 - 7.11 years at recruitment
2. Children with a diagnosis or in diagnostic process for high suspicion of a genetic abnormality
3. For children up to 5.11 years with withdrawn (defined as CBCL/1.5-5 scores above the borderline clinical range, T-score> 65) AND/OR pervasive developmental problems (defined as CBCL/1.5-5 scores above the borderline clinical range, T-score> 65) AND/OR socialization difficulties (defined as Vineland-III scores below 1SD in the socialization subdomains).
4. For children over 6 years with social problems (defined as CBCL/6-18 scores above the borderline clinical range, T-score> 65) AND/OR thought problems (defined as CBCL/6-18 scores above the borderline clinical range, T-score> 65) AND/OR socialization difficulties (defined as Vineland-III scores below 1SD in the socialization subdomains).
5. Parents/caregivers showing good understanding of the Spanish or Catalan language
6. Parents/caregivers consenting to take part in the study and signing the informed consent

Exclusion Criteria:

1. Children diagnosed with an Autism Spectrum Disorder
2. Children scoring above diagnostic cut-off for Autism or Autism Spectrum Disorder in the ADOS-2.
3. Attending another structured parenting program
4. Children in the care of their local authority

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Treatment
• Allocation: Randomized
• Interventional Model: Parallel Assignment
• Masking: None (Open Label)

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Experimental: Intervention group; Those in the intervention group will participate in a weekly group-based online intervention for approximately 6 months (Incredible Years-Autism Spectrum and Language Delays Parent Program, IY-ASLD®). Families allocated to the intervention group will also receive treatment as usual (TAU).	Behavioral: Incredible Years Autism Spectrum and Language Delays Parent Program (IY-ASLD®); The IY-ASLD® program is a weekly group-based intervention for parents of children presenting neurodevelopmental problems (ASD symptoms, communication or language difficulties). The group is formed by 6-10 parents, and it is led by a group leader and a co-therapist, trained in the model. The intervention is manualized. It includes video modelling and emphasizes the importance of practice-based learning through role-playing. The IY-ASLD® program takes into consideration the different developmental levels of each child 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. The intervention will be conducted online. Even though the IY-ASLD® original intervention comprises 14 sessions, the online format requires 22 weekly sessions. Fidelity to the intervention will be assessed in accordance with the regulations of the program.; Other Names: Parenting intervention
No Intervention: Treatment as usual (TAU) group; The TAU condition involves outpatient appointments with different paediatric specialists in the hospitals of the 3 sites of the study. Depending on the patients' needs, some cases will be assisted in early years centers or child mental health centers based in the community.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Percentage of parents engaging with and finishing the program	Parents' attendance at the sessions will be regeristered weekly with a sheet of attendance, with the aim to determine levels of compliance and engagement with the intervention. An attendance of at least 15/22 sessions is expected, with a minimum of 50% of parents finishing the program.	Attendance sheet completed weekly throughout treatment (22 weeks total)
Compliance and satisfaction throughout the study (Autism Program Parent Weekly Evaluation)	Parents' compliance and satisfaction with the intervention will be assessed weekly after each session with the questionnaire "Autism Program Parent Weekly Evaluation", which is part of the IY-ASLD® program materials.	Questionnaire administered to the intervention group weekly throughout treatment (22 weeks total)
Parents' acceptability and satisfaction with the program (evaluated with the Autism Program Parent Final Satisfaction Questionnaire)	Parents' acceptability and satisfaction with the program will be assessed with the Autism Program Parent Final Satisfaction Questionnaire (included within the IY-ASLD® program). Data will be collected after the last session of the intervention.	Administered to the intervention group at treatment completion (approximately 22 weeks after baseline)
Parents' overall experiences with the program (evaluated with individual interviews)	Individual interviews will be conducted after the last session of the intervention to explore from a qualitative perspective: (1) parents' acceptability, satisfaction and overall experience with the intervention, and (2) parents' perceived changes in their parenting skills and parental distress after the intervention. Outcome measurements 3 and 4 will be combined to determine parents' experiences, acceptability and satisfaction with the program.	Interviews to the participants of the intervention group at treatment completion (approximately 22 weeks after baseline)

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Changes from baseline to post-intervention in Parent Stress Inventory-Short Form (PSI-SF)	The PSI-SF 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.	Administered to the intervention and control group at baseline (prior to programme initiation) and at immediately after the intervention (approximately 22 weeks after baseline)
Changes from baseline to post-intervention in Alabama Parenting Questionnaire-Preschool revision (APQ-Pr)	The APQ-Pr 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.	Administered to the intervention and control group at baseline (prior to programme initiation) and at immediately after the intervention (approximately 22 weeks after baseline)
Changes from baseline to post-intervention in Beck Depression Inventory (BDI)	The BDI 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.	Administered to the intervention and control group at baseline (prior to programme initiation) and at immediately after the intervention (approximately 22 weeks after baseline)
Changes from baseline to post-intervention in Autism-Specific Five Minute Speech Sample (ASFMFSS)	ASFMFSS is a narrative 5-min 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. Speech samples are audiotaped, transcribed, and coded following four global categories: (a) initial statement, (b) warmth, (c) relationship, (d) emotional over-involvement, (e) critical comments, and (f) positive comments. Expressed emotions will be measured before and after the intervention. Inter-rater reliability and code-recode reliability of this measure have been shown to be good to excellent.	Administered to the intervention and control group at baseline (prior to programme initiation) and at immediately after the intervention (approximately 22 weeks after baseline)
Clinicians' experiences with the intervention delivered	Individual interviews will be conducted after the last session with clinicians who delivered the intervention.	Interviews to clinicians at treatment completion (approximately 22 weeks after baseline)

Collaborators and Investigators

• Sponsor: Fundació Sant Joan de Déu
• Collaborators: Hospital Universitari Vall d'Hebron Research Institute; Fundació La Marató de TV3; Parc Taulí Hospital Universitari
• Investigators: Principal Investigator: Laia Villalta, MD, PhD, Hospital Sant Joan de Déu. Fundació Privada per a la Recerca i la Docència Sant Joan de Déu; Study Chair: Anna Maria Cueto-González, MD, Hospital Universitari Vall d'Hebron. Vall d'Hebron Institut de Recerca; Study Chair: Carmen Manso, MD, Parc Tauli Hospital Universitari; Study Chair: Mercedes Serrano, MD, PhD, Hospital Sant Joan de Déu

Publications and helpful links

General Publications

• Williams ME, Hastings RP, Hutchings J. The Incredible Years Autism Spectrum and Language Delays Parent Program: A Pragmatic, Feasibility Randomized Controlled Trial. Autism Res. 2020 Jun;13(6):1011-1022. doi: 10.1002/aur.2265. Epub 2020 Jan 21.
• Valencia F, Urbiola E, Romero-Gonzalez M, Navas I, Elias M, Garriz A, Ramirez A, Villalta L. Protocol for a randomized pilot study (FIRST STEPS): implementation of the Incredible Years-ASLD(R) program in Spanish children with autism and preterm children with communication and/or socialization difficulties. Trials. 2021 Apr 20;22(1):291. doi: 10.1186/s13063-021-05229-1.
• Landa RJ. Efficacy of early interventions for infants and young children with, and at risk for, autism spectrum disorders. Int Rev Psychiatry. 2018 Feb;30(1):25-39. doi: 10.1080/09540261.2018.1432574. Epub 2018 Mar 14.
• Morel A, Peyroux E, Leleu A, Favre E, Franck N, Demily C. Overview of Social Cognitive Dysfunctions in Rare Developmental Syndromes With Psychiatric Phenotype. Front Pediatr. 2018 May 3;6:102. doi: 10.3389/fped.2018.00102. eCollection 2018.
• Richards C, Jones C, Groves L, Moss J, Oliver C. Prevalence of autism spectrum disorder phenomenology in genetic disorders: a systematic review and meta-analysis. Lancet Psychiatry. 2015 Oct;2(10):909-16. doi: 10.1016/S2215-0366(15)00376-4. Epub 2015 Sep 1.
• Webster-Stratton C, McCoy KP. Bringing The Incredible Years(R) Programs to Scale. New Dir Child Adolesc Dev. 2015 Fall;2015(149):81-95. doi: 10.1002/cad.20115.

Study record dates

Study Major Dates

• Study Start (Actual): March 14, 2023
• Primary Completion (Estimated): November 1, 2024
• Study Completion (Estimated): December 1, 2024

Study Registration Dates

• First Submitted: October 23, 2023
• First Submitted That Met QC Criteria: November 3, 2023
• First Posted (Estimated): November 9, 2023

Study Record Updates

• Last Update Posted (Estimated): November 9, 2023
• Last Update Submitted That Met QC Criteria: November 3, 2023
• Last Verified: November 1, 2023

More Information

Terms related to this study

• Keywords: randomized controlled trial; mental health; early intervention; parenting; development; pragmatic; genetic syndromes
• Additional Relevant MeSH Terms: Congenital Abnormalities
• Other Study ID Numbers: PIC-168-22; 434/U/2022 (Other Grant/Funding Number: Fundació La MaratóTV3)

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: NO

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No