Home-based SSP on Individuals With PWS

Evaluating the Effectiveness of Home-based SSP on Individuals With PWS

The Polyvagal Theory focuses on how function and structure changed in the vertebrate autonomic nervous system during evolution. The theory is named for the vagus, a major cranial nerve that regulates bodily state. As a function of evolution, humans and other mammals have a "new" vagal pathway that links the regulation of bodily state to the control of the muscles of the face and head including the middle ear muscles. These pathways regulating body state, facial gesture, listening (i.e., middle ear muscles), and vocal communication collectively function as a Social Engagement System (SES). Because the Social Engagement System is an integrated system, interventions influencing one component of this system (e.g., middle ear muscles) may impact on the other components.

Individuals with Prader-Willi Syndrome (PWS) exhibit many behaviors that are consistent with a compromised Social Engagement System. Atypical function of the Social Engagement System results in problems associated with state regulation (e.g., impulsivity, tantrums, and difficulty with change in routine), ingestion (e.g., difficulties in sucking at birth, hyperphagia), coordination of suck/swallow/breathe, intonation of vocalizations, auditory processing and hypersensitivity, and socialization. The investigatiors propose to confirm that several features of the behavioral phenotype of PWS may be explained within the context of a dysfunctional SES, which may be partially rehabilitated via an intervention designed as a 'neural exercise' of the SES (i.e., the Safe and Sound Protocol, "SSP").

Specific Aims:

Aim I: To demonstrate the effectiveness of the Safe and Sound Protocol (SSP) on improvement of social and regulation behaviors in individuals with PWS.

Aim II: To evaluate a new methodology for collecting and evaluating vocal samples for analyses of prosody, one of the indices of the functioning of the SES.

Study Overview

• Status: Terminated
• Conditions: Prader-Willi Syndrome
• Intervention / Treatment: Behavioral: Safe and Sound Protocol

• Study Type: Interventional
• Enrollment (Actual): 10
• Phase: Not Applicable

Contacts and Locations

Study Locations

• United States
  • Colorado
    • Aurora, Colorado, United States, 80014
      • Integrated Listening Systems

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 8 months to 13 years (Child)
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

Description

Inclusion Criteria:

1. Child participants must meet criteria for Prader-Willi Syndrome (per parent membership in Prader-Willi Parent Support Group).
2. Child participants must be between ages 3-17 years. Parent must be 18 years or older.
3. Child participants must have normal hearing (confirmed via parental report on Qualtrics questionnaire)

Exclusion Criteria:

1) Child participants who are hearing-impaired (without correction)

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Basic Science
• Allocation: N/A
• Interventional Model: Single Group Assignment
• Masking: None (Open Label)

Number of Arms

1

Arms and Interventions

Participant Group / Arm

Intervention / Treatment

Experimental: Safe and Sound Protocol; All child participants will participate in 1 pre-intervention assessment and 1 post-intervention assessment. The auditory intervention (i.e., Safe and Sound Protocol, SSP) will last for 1 hour per day, for 5 consecutive days.

Behavioral: Safe and Sound Protocol; The auditory intervention will consist of listening to computer-altered acoustic stimulation, designed to modulate the frequency band of vocal music passed to the participant. The frequency characteristics of the acoustic stimulation are selected to emphasize the relative importance of specific frequencies in conveying the information embedded in human speech. Modulation of the acoustic energy within the frequencies of human voice, similar to an exaggerated vocal prosody, are hypothesized to recruit and modulate the neural regulation of the middle ear muscles and to functionally reduce sound hypersensitivities and improve auditory processing.; Other Names: Listening Project Protocol

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Change from baseline in sensory sensitivities at 1 week	Brain-Body Center Sensory Scales (BBC Sensory Scales) (parental questionnaire)	pre-intervention (within 1 week before intervention), post-intervention (within 1 week after intervention)

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Change from baseline in disruptive behavior at 1 week	Developmental Behavior Checklist (DBC2) (questionnaire)	pre-intervention (within 1 week before intervention), post-intervention (within 1 week after intervention)
Change from baseline in social behavior at 1 week	Safe and Sound Protocol (SSP) Parent Questionnaire	post-intervention (within 1 week after intervention)
Change from baseline in Foundational Abilities (balance, gross & fine motor control, sensory, social/emotional, auditory/language and attention/organization and sleep) at 1 week	Measure of Foundational Abilities (MFA) questionnaire	pre-intervention (within 1 week before intervention), post-intervention (within 1 week after intervention)
Change from baseline in prosody at 1 week	Prosody assessment of recorded speech	pre-intervention (within 1 week before intervention), post-intervention (within 1 week after intervention)

Collaborators and Investigators

• Sponsor: Indiana University
• Collaborators: Integrated Listening Systems
• Investigators: Principal Investigator: Jacek Kolacz, PhD, Indiana University/Kinsey Institute

Publications and helpful links

General Publications

• Porges SW. The polyvagal theory: phylogenetic substrates of a social nervous system. Int J Psychophysiol. 2001 Oct;42(2):123-46. doi: 10.1016/s0167-8760(01)00162-3.
• Porges SW, Bazhenova OV, Bal E, Carlson N, Sorokin Y, Heilman KJ, Cook EH, Lewis GF. Reducing auditory hypersensitivities in autistic spectrum disorder: preliminary findings evaluating the listening project protocol. Front Pediatr. 2014 Aug 1;2:80. doi: 10.3389/fped.2014.00080. eCollection 2014.

Study record dates

Study Major Dates

• Study Start (Actual): April 8, 2019
• Primary Completion (Actual): December 16, 2019
• Study Completion (Actual): December 16, 2019

Study Registration Dates

• First Submitted: February 25, 2019
• First Submitted That Met QC Criteria: March 11, 2019
• First Posted (Actual): March 12, 2019

Study Record Updates

• Last Update Posted (Actual): March 7, 2022
• Last Update Submitted That Met QC Criteria: February 18, 2022
• Last Verified: February 1, 2022

More Information

Terms related to this study

• Keywords: Prosody; Polyvagal Theory; Social Behavior; Hyperacusis; Auditory intervention
• Additional Relevant MeSH Terms: Nervous System Diseases; Neurologic Manifestations; Neurobehavioral Manifestations; Congenital Abnormalities; Overnutrition; Nutrition Disorders; Genetic Diseases, Inborn; Intellectual Disability; Abnormalities, Multiple; Chromosome Disorders; Obesity; Prader-Willi Syndrome
• Other Study ID Numbers: 1807300643

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