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

Prader-Willi Syndrome Clinical Trial

Official title:

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

Clinical Trial Details — Status: Terminated

Administrative data

• NCT number: NCT03101826
• Other study ID #: 1609337519
• Status: Terminated
• Phase: N/A
• Start date: November 1, 2017
• Est. completion date: March 8, 2023

Study information

• Verified date: March 2023
• Source: Indiana University
• Contact: n/a
• Is FDA regulated: No
• Study type: Interventional

Clinical Trial 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.

Recruitment information / eligibility

• Status: Terminated
• Enrollment: 5
• Est. completion date: March 8, 2023
• Est. primary completion date: March 8, 2023
• Accepts healthy volunteers: No
• Gender: All
• Age group: 13 Years to 25 Years

Eligibility

Inclusion Criteria:

1. Participants must be current residents at Latham Centers (LC) School in Brewster MA

2. Participants must meet criteria for Prader-Willi Syndrome

3. Participants must be between ages 13 - 25 years

4. Participants must have normal hearing

5. Participants must have normal vision (or corrected vision)

Exclusion Criteria:

1. Individuals with current (or a history of) heart disease

2. Individuals who are hearing-impaired

3. Individuals who are being treated for seizure disorder

4. Individuals who do not read/speak English

5. Individuals who are sight-impaired without correction

Related Conditions & MeSH terms

• Prader-Willi Syndrome
• Syndrome

Intervention

Behavioral:
• Listening Project Protocol
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.

Locations

Country	Name	City	State
United States	Latham Centers School	Brewster	Massachusetts

Sponsors (2)

Lead Sponsor	Collaborator
Indiana University	Latham Centers

Country where clinical trial is conducted

United States, 

References & 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. — View Citation

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

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. — View Citation

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. — View Citation

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. — View Citation

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. — View Citation

Outcome

Type	Measure	Description	Time frame	Safety issue
Other	Stability of Autonomic regulation	Respiratory sinus arrhythmia (RSA)	Change from 6 months pre-intervention RSA to 1 week pre-intervention
Other	Immediate effects of intervention on Autonomic regulation	Respiratory sinus arrhythmia (RSA)	Change from 1 week pre-intervention RSA to 1 week post-intervention
Other	Short-term effects of intervention on Autonomic regulation	Respiratory sinus arrhythmia (RSA)	Change from 1 week post-intervention RSA to 1 month post-intervention
Other	Stability of Heart period	heart period	Change from 6 months pre-intervention heart period to 1 week pre-intervention
Other	Immediate effects of intervention on Heart period	heart period	Change from 1 week pre-intervention heart period to 1 week post-intervention
Other	Short-term effects of intervention on Heart period	heart period	Change from 1 week post-intervention heart period to 1 month post-intervention
Other	Stability of Auditory processing	Filtered Words subtest of SCAN	Change from 6 months pre-intervention Filtered Words to 1 week pre-intervention
Other	Immediate effects of intervention on Auditory processing	Filtered Words subtest of SCAN	Change from 1 week pre-intervention Filtered Words to 1 week post-intervention
Other	Short-term effects of intervention on Auditory processing	Filtered Words subtest of SCAN	Change from 1 week post-intervention Filtered Words to 1 month post-intervention
Other	Stability of Auditory processing	Competing Words subtest of SCAN	Change from 6 months pre-intervention Competing Words to 1 week pre-intervention
Other	Immediate effects of intervention on Auditory processing	Competing Words subtest of SCAN	Change from 1 week pre-intervention Competing Words to 1 week post-intervention
Other	Short-term effects of intervention on Auditory processing	Competing Words subtest of SCAN	Change from 1 week post-intervention Competing Words to 1 month post-intervention
Other	Stability of Sensory Sensitivities	BBC Sensory Scales (questionnaire)	Change from 6 months pre-intervention BBC Sensory Scales to 1 week pre-intervention
Other	Stability of Latency to Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - latency	Change from 6 months pre-intervention DARE latency to 1 week pre-intervention
Other	Immediate effects of intervention on Latency to Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - latency	Change from 1 week pre-intervention DARE latency to 1 week post-intervention
Other	Short-term effects of intervention on Latency to Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - latency	Change from 1 week post-intervention DARE latency to 1 month post-intervention
Other	Stability of Accuracy of Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - accuracy	Change from 6 months pre-intervention DARE accuracy to 1 week pre-intervention
Other	Immediate effects of intervention on Accuracy of Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - accuracy	Change from 1 week pre-intervention DARE accuracy to 1 week post-intervention
Other	Short-term effects of intervention on Accuracy of Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - accuracy	Change from 1 week post-intervention DARE accuracy to 1 month post-intervention
Other	Stability of Prosody	Analyses of vocal recordings	Change from 6 months pre-intervention prosody to 1 week pre-intervention
Other	Immediate effects of intervention on Prosody	Analyses of vocal recordings	Change from 1 week pre-intervention prosody to 1 week post-intervention
Other	Short-term effects of intervention on Prosody	Analyses of vocal recordings	Change from 1 week post-intervention prosody to 1 month post-intervention
Other	Stability of Disruptive behavior	Developmental Behavior Checklist (DBC) (questionnaire)	Change from 6 months pre-intervention DBC to 1 week pre-intervention
Other	Immediate effects of intervention on Disruptive behavior	Developmental Behavior Checklist (DBC) (questionnaire)	Change from 1 week pre-intervention DBC to 1 week post-intervention
Other	Short-term effects of intervention on Disruptive behavior	Developmental Behavior Checklist (DBC) (questionnaire)	Change from 1 week post-intervention DBC to 1 month post-intervention
Other	Stability of Oxytocin	oxytocin levels derived from salivary sample	Change from 6 months pre-intervention oxytocin to 1 week pre-intervention
Other	Stability of Vasopressin	vasopressin levels derived from salivary sample	Change from 6 months pre-intervention vasopressin to 1 week pre-intervention
Primary	Effect of Intervention on Autonomic regulation	Respiratory sinus arrhythmia (RSA)	Change from 1 week pre-intervention RSA to 1 month post-intervention
Secondary	Effect of Intervention on Heart period	heart period	Change from 1 week pre-intervention heart period to 1 month post-intervention
Secondary	Effect of Intervention on Auditory processing	Filtered Words subtest of SCAN	Change from 1 week pre-intervention Filtered Words to 1 month post-intervention
Secondary	Effect of Intervention on Auditory processing	Competing Words subtest of SCAN	Change from 1 week pre-intervention Competing Words to 1 month post-intervention
Secondary	Effect of Intervention on Sensory Sensitivities	BBC Sensory Scales (questionnaire)	Change from 1 week pre-intervention BBC Sensory Scales to 1 month post-intervention
Secondary	Effect of Intervention on Latency to Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - latency	Change from 1 week pre-intervention DARE (latency) to 1 month post-intervention
Secondary	Effect of Intervention on Accuracy of Affect recognition	Dynamic Affect Recognition Evaluation (DARE) - accuracy	Change from 1 week pre-intervention DARE (accuracy) to 1 month post-intervention
Secondary	Effect of Intervention on Prosody	Analyses of vocal recordings	Change from 1 week pre-intervention Prosody to 1 month post-intervention
Secondary	Effect of Intervention on Disruptive Behavior	Developmental Behavior Checklist (DBC) (questionnaire)	Change from 1 week pre-intervention DBC to 1 month post-intervention
Secondary	Effect of Intervention on Social behavior	Listening Project Parent (LPP) Questionnaire	Change from 1 week post-intervention LPP to 1 month post-intervention
Secondary	Effect of Intervention on Oxytocin	oxytocin levels derived from salivary sample	Change from 1 week pre-intervention oxytocin to 1 month post-intervention
Secondary	Effect of Intervention on Vasopressin	vasopressin levels derived from salivary sample	Change from 1 week pre-intervention vasopressin to 1 month post-intervention