Cooperative Parent Mediated Therapy in Children With Fragile X Syndrome and Williams Syndrome

Summary

Fragile X Syndrome (FXS) and Williams-Beuren Syndrome (WBS) are relatively rare disorders characterized by developmental delay associated to socio-communicative deficit and autistic-like behaviours.

WBS has been considered for a long time as the "polar opposite" of ASD, given their hypersociable phenotype. Nonetheless, recent researches have emphasized similarities between ASD and WBS phenotypes. By following some authors "social abnormalities in ASD and WS can be characterized in terms of analogous difficulties in social cognition), and distinct patterns of social motivation which appears to be reduced in ASD and enhanced in WBS". More than opposite condition, these authors suggests that WBS and ASD could share the same difficult in comprehension of social relationship, with opposite pattern of social engagement (enhanced in WBS and weakened ASD). Given, these similarities authors suggest testing the feasibility and validity of therapy for ASD in children with WBS. Parent Mediated Therapy (PMT) is a group of "technique-focused interventions where the parent is the agent of change and the child is the direct beneficiary of treatment". PMT demonstrated evidence of effectiveness in socio-communicational improvement for children with ASD in a randomized controlled trial (RCT).

Some recent researchers have extended the use of PMT to children with genetic disorders and autistic features, such as FXS. While showing encouraging results, the samples of research were limited.

They main aim of this research is to to verify effectiveness of Cooperative PMT (CMPT) for socio-communicative deficit in children with FXS and WBS. Our hypothesis is that CPMT, in addition to conventional rehabilitation therapies (mainly speech therapy and occupational therapies), could contribute to the enhancement of socio-communicative skills and the reduction of behavioural problems. We also expected also an improvement in family quality of life and a reduction of parental stress.

Conditions

• Fragile X Syndrome
• Williams Syndrome

Interventions

BEHAVIORAL

Cooperative Parent Mediated Therapy" (CPMT)

CMPT usually last 6 months, for a total amount of 15 sessions of 90 min; twelve core sessions (one session per week) are delivered in the first 3 months, followed by 3 monthly booster sessions. Each weekly core session had a specific focus and specific intervention strategies based on active parent coaching during parent-child interaction, and included the parent-child dyad with the parent being actively coached by a trained therapist. Live active coaching increases parents' competence in implementing strategies to enhance child development, and at the same time increases their confidence. This intervention has demonstrated evidence of effectiveness in socio-communicational improvement as measured by ADOS-G (Valeri, 2019) in a randomized controlled trial (RCT).

BEHAVIORAL

As usual

Speech Language Therapy and Occuapational Therapy provided as usual by National Health Services

Sponsors & Collaborators

• Autour Des Williams

  collaborator UNKNOWN

• Acea

  collaborator UNKNOWN

• Bambino Gesù Hospital and Research Institute

  lead OTHER

Principal Investigators

• Paolo Alfieri, PhD, MD · Ospedale Pediatrico Bambino Gesù

Study Design

Allocation

RANDOMIZED

Purpose

TREATMENT

Masking

TRIPLE

Model

PARALLEL

Eligibility

Min Age

1 Year

Max Age

7 Years

Sex

ALL

Healthy Volunteers

No

Timeline & Regulatory

Start

2017-05-17

Primary Completion

2021-06-30

Completion

2021-06-30

Countries

• Italy

Study Locations