Optimizing the Social Engagement System in Prader-Willi Syndrome: Insights From the Polyvagal Theory

NCT03101826 | Terminated

• 1 other identifier: 1609337519
• Study Type: interventional
• Target: 5
• Locations: 1 country
• Sites: 1

Brief Summary

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. We propose to confirm that several features of the behavioral phenotype of PWS may be explained within the context of a dysfunctional SES (Specific Aim I), which may be partially rehabilitated via an intervention designed as a 'neural exercise' of the SES (Specific Aim II). Specific Aims: Aim I: To demonstrate that children with PWS have atypical regulation of the SES. We hypothesize these effects will be manifested by dampened vagal regulation of the heart (low parasympathetic tone); poor middle ear muscle regulation resulting in auditory hypersensitivities and poor auditory processing; lack of voice intonation (prosody), and difficulties in accurately detecting the emotions of others. Aim II: To demonstrate the effectiveness of the Listening Project Protocol (LPP) in decreasing the atypical features of the SES in adolescents with PWS. We hypothesize that individuals who complete the LPP will have improved vagal regulation of the heart, improved middle ear muscle regulation, increased voice intonation and improved ability to accurately detect the emotions of others.

Conditions

Prader-Willi Syndrome

Keywords

Polyvagal Theory; Respiratory Sinus Arrhythmia; Heart rate; Social Behavior; Arginine Vasopressin; Dichotic Listening Tests; Oxytocin; Hyperacusis; Affect; Auditory perception; Sound spectrography

Outcome Measures

Primary Outcomes (1)

• Effect of Intervention on Autonomic regulation

  Respiratory sinus arrhythmia (RSA)

  Change from 1 week pre-intervention RSA to 1 month post-intervention

Secondary Outcomes (11)

• Effect of Intervention on Heart period

  Change from 1 week pre-intervention heart period to 1 month post-intervention

• Effect of Intervention on Auditory processing

  Change from 1 week pre-intervention Filtered Words to 1 month post-intervention

• Effect of Intervention on Auditory processing

  Change from 1 week pre-intervention Competing Words to 1 month post-intervention

• Effect of Intervention on Sensory Sensitivities

  Change from 1 week pre-intervention BBC Sensory Scales to 1 month post-intervention

• Effect of Intervention on Latency to Affect recognition

  Change from 1 week pre-intervention DARE (latency) to 1 month post-intervention

Other Outcomes (27)

• Stability of Autonomic regulation

  Change from 6 months pre-intervention RSA to 1 week pre-intervention

• Immediate effects of intervention on Autonomic regulation

  Change from 1 week pre-intervention RSA to 1 week post-intervention

• Short-term effects of intervention on Autonomic regulation

  Change from 1 week post-intervention RSA to 1 month post-intervention

Study Arms (1)

Filtered Music Intervention

EXPERIMENTAL

All participants will participate in pre-intervention assessments (6 months, 1 week prior) and post-intervention assessments (1 week, 1 month post). The Filtered Music Intervention (i.e., Listening Project Protocol) will last for 1 hour per day, for 5 consecutive days.

Behavioral: Listening Project Protocol

Interventions

Listening Project Protocol BEHAVIORAL

The filtered music 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.

Also known as: Safe and Sound Protocol

Filtered Music Intervention

Eligibility Criteria

• Age: 13 Years - 25 Years
• Sex: all
• Healthy Volunteers: No
• Age Groups: Child (0-17), Adult (18-64)

You may qualify if:

• Participants must be current residents at Latham Centers (LC) School in Brewster MA
• Participants must meet criteria for Prader-Willi Syndrome
• Participants must be between ages 13 - 25 years
• Participants must have normal hearing
• Participants must have normal vision (or corrected vision)

You may not qualify if:

• Individuals with current (or a history of) heart disease
• Individuals who are hearing-impaired
• Individuals who are being treated for seizure disorder
• Individuals who do not read/speak English
• Individuals who are sight-impaired without correction

Sponsors & Collaborators

• Indiana University: lead
• Latham Centers: collaborator

Study Sites (1)

Latham Centers School

Brewster, Massachusetts, 02631, United States

Location

Related Publications (6)

• Bal E, Harden E, Lamb D, Van Hecke AV, Denver JW, Porges SW. Emotion recognition in children with autism spectrum disorders: relations to eye gaze and autonomic state. J Autism Dev Disord. 2010 Mar;40(3):358-70. doi: 10.1007/s10803-009-0884-3.

  PMID: 19885725 BACKGROUND

• Borg E, Counter SA. The middle-ear muscles. Sci Am. 1989 Aug;261(2):74-80. doi: 10.1038/scientificamerican0889-74. No abstract available.

  PMID: 2667133 BACKGROUND

• Lewis GF, Furman SA, McCool MF, Porges SW. Statistical strategies to quantify respiratory sinus arrhythmia: are commonly used metrics equivalent? Biol Psychol. 2012 Feb;89(2):349-64. doi: 10.1016/j.biopsycho.2011.11.009. Epub 2011 Dec 3.

  PMID: 22138367 BACKGROUND

• 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.

  PMID: 11587772 BACKGROUND

• Porges SW, Macellaio M, Stanfill SD, McCue K, Lewis GF, Harden ER, Handelman M, Denver J, Bazhenova OV, Heilman KJ. Respiratory sinus arrhythmia and auditory processing in autism: modifiable deficits of an integrated social engagement system? Int J Psychophysiol. 2013 Jun;88(3):261-70. doi: 10.1016/j.ijpsycho.2012.11.009. Epub 2012 Nov 29.

  PMID: 23201146 BACKGROUND

• 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.

  PMID: 25136545 BACKGROUND

MeSH Terms

Conditions

Prader-Willi Syndrome; Arrhythmia, Sinus; Social Behavior; Diabetes Insipidus; Hyperacusis

Condition Hierarchy (Ancestors)

Intellectual Disability; Neurobehavioral Manifestations; Neurologic Manifestations; Nervous System Diseases; Abnormalities, Multiple; Congenital Abnormalities; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Chromosome Disorders; Genetic Diseases, Inborn; Imprinting Disorders; Obesity; Overweight; Overnutrition; Nutrition Disorders; Nutritional and Metabolic Diseases; Arrhythmias, Cardiac; Heart Diseases; Cardiovascular Diseases; Pathologic Processes; Pathological Conditions, Signs and Symptoms; Behavior; Kidney Diseases; Urologic Diseases; Female Urogenital Diseases; Female Urogenital Diseases and Pregnancy Complications; Urogenital Diseases; Male Urogenital Diseases; Pituitary Diseases; Endocrine System Diseases; Hearing Disorders; Ear Diseases; Otorhinolaryngologic Diseases; Sensation Disorders; Signs and Symptoms

Study Officials

• Stephen W Porges, PhD

  Indiana University/Kinsey Institute

  PRINCIPAL INVESTIGATOR

Study Design

• Study Type: interventional
• Phase: not applicable
• Allocation: NA
• Masking: NONE
• Masking Details: No masking.
• Purpose: BASIC SCIENCE
• Intervention Model: SINGLE GROUP
• Sponsor Type: OTHER
• Responsible Party: PRINCIPAL INVESTIGATOR
• PI Title: Distinguished University Scientist

Model Details: All participants will receive the active (filtered music) intervention.

Study Record Dates

• First Submitted: March 23, 2017
• First Posted: April 5, 2017
• Study Start: November 1, 2017
• Primary Completion: March 8, 2023
• Study Completion: March 8, 2023
• Last Updated: April 3, 2023

Record last verified: 2023-03

Data Sharing

• IPD Sharing: Will not share

Locations

US (1)