Clinical trial NCT03101826: Optimizing the Social Engagement System in Prader-Willi Syndrome: Insights From the Polyvagal Theory
Official title: Optimizing the Social Engagement System in Prader-Willi Syndrome: Insights From the Polyvagal Theory
Study type: INTERVENTIONAL
Overall status: TERMINATED
Phase: NA
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
Lead sponsor: Indiana University
Sponsor class: OTHER
Collaborators: name: Latham Centers
First posted: 2017-04-05
Enrollment: 5
Sex: ALL
Minimum age (years): 13
Maximum age (years): 25
Standard ages: CHILD
ADULT
Accepts healthy volunteers: no
Allocation: NA
Intervention model: SINGLE_GROUP
Intervention model description: All participants will receive the active (filtered music) intervention.
Primary purpose: BASIC_SCIENCE

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.


Eligibility criteria: 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

Primary outcomes:
measure: Effect of Intervention on Autonomic regulation
timeFrame: Change from 1 week pre-intervention RSA to 1 month post-intervention
Secondary outcomes:
measure: Effect of Intervention on Heart period
timeFrame: Change from 1 week pre-intervention heart period to 1 month post-intervention
measure: Effect of Intervention on Auditory processing
timeFrame: Change from 1 week pre-intervention Filtered Words to 1 month post-intervention
measure: Effect of Intervention on Auditory processing
timeFrame: Change from 1 week pre-intervention Competing Words to 1 month post-intervention
measure: Effect of Intervention on Sensory Sensitivities
timeFrame: Change from 1 week pre-intervention BBC Sensory Scales to 1 month post-intervention
measure: Effect of Intervention on Latency to Affect recognition
timeFrame: Change from 1 week pre-intervention DARE (latency) to 1 month post-intervention
measure: Effect of Intervention on Accuracy of Affect recognition
timeFrame: Change from 1 week pre-intervention DARE (accuracy) to 1 month post-intervention
measure: Effect of Intervention on Prosody
timeFrame: Change from 1 week pre-intervention Prosody to 1 month post-intervention
measure: Effect of Intervention on Disruptive Behavior
timeFrame: Change from 1 week pre-intervention DBC to 1 month post-intervention
measure: Effect of Intervention on Social behavior
timeFrame: Change from 1 week post-intervention LPP to 1 month post-intervention
measure: Effect of Intervention on Oxytocin
timeFrame: Change from 1 week pre-intervention oxytocin to 1 month post-intervention
measure: Effect of Intervention on Vasopressin
timeFrame: Change from 1 week pre-intervention vasopressin to 1 month post-intervention
Other outcomes:
measure: Stability of Autonomic regulation
timeFrame: Change from 6 months pre-intervention RSA to 1 week pre-intervention
measure: Immediate effects of intervention on Autonomic regulation
timeFrame: Change from 1 week pre-intervention RSA to 1 week post-intervention
measure: Short-term effects of intervention on Autonomic regulation
timeFrame: Change from 1 week post-intervention RSA to 1 month post-intervention
measure: Stability of Heart period
timeFrame: Change from 6 months pre-intervention heart period to 1 week pre-intervention
measure: Immediate effects of intervention on Heart period
timeFrame: Change from 1 week pre-intervention heart period to 1 week post-intervention
measure: Short-term effects of intervention on Heart period
timeFrame: Change from 1 week post-intervention heart period to 1 month post-intervention
measure: Stability of Auditory processing
timeFrame: Change from 6 months pre-intervention Filtered Words to 1 week pre-intervention
measure: Immediate effects of intervention on Auditory processing
timeFrame: Change from 1 week pre-intervention Filtered Words to 1 week post-intervention
measure: Short-term effects of intervention on Auditory processing
timeFrame: Change from 1 week post-intervention Filtered Words to 1 month post-intervention
measure: Stability of Auditory processing
timeFrame: Change from 6 months pre-intervention Competing Words to 1 week pre-intervention
measure: Immediate effects of intervention on Auditory processing
timeFrame: Change from 1 week pre-intervention Competing Words to 1 week post-intervention
measure: Short-term effects of intervention on Auditory processing
timeFrame: Change from 1 week post-intervention Competing Words to 1 month post-intervention
measure: Stability of Sensory Sensitivities
timeFrame: Change from 6 months pre-intervention BBC Sensory Scales to 1 week pre-intervention
measure: Stability of Latency to Affect recognition
timeFrame: Change from 6 months pre-intervention DARE latency to 1 week pre-intervention
measure: Immediate effects of intervention on Latency to Affect recognition
timeFrame: Change from 1 week pre-intervention DARE latency to 1 week post-intervention
measure: Short-term effects of intervention on Latency to Affect recognition
timeFrame: Change from 1 week post-intervention DARE latency to 1 month post-intervention
measure: Stability of Accuracy of Affect recognition
timeFrame: Change from 6 months pre-intervention DARE accuracy to 1 week pre-intervention
measure: Immediate effects of intervention on Accuracy of Affect recognition
timeFrame: Change from 1 week pre-intervention DARE accuracy to 1 week post-intervention
measure: Short-term effects of intervention on Accuracy of Affect recognition
timeFrame: Change from 1 week post-intervention DARE accuracy to 1 month post-intervention
measure: Stability of Prosody
timeFrame: Change from 6 months pre-intervention prosody to 1 week pre-intervention
measure: Immediate effects of intervention on Prosody
timeFrame: Change from 1 week pre-intervention prosody to 1 week post-intervention
measure: Short-term effects of intervention on Prosody
timeFrame: Change from 1 week post-intervention prosody to 1 month post-intervention
measure: Stability of Disruptive behavior
timeFrame: Change from 6 months pre-intervention DBC to 1 week pre-intervention
measure: Immediate effects of intervention on Disruptive behavior
timeFrame: Change from 1 week pre-intervention DBC to 1 week post-intervention
measure: Short-term effects of intervention on Disruptive behavior
timeFrame: Change from 1 week post-intervention DBC to 1 month post-intervention
measure: Stability of Oxytocin
timeFrame: Change from 6 months pre-intervention oxytocin to 1 week pre-intervention
measure: Stability of Vasopressin
timeFrame: Change from 6 months pre-intervention vasopressin to 1 week pre-intervention
Locations:
city: Brewster
country: United States
facility: Latham Centers School
state: Massachusetts
Study officials:
affiliation: Indiana University/Kinsey Institute
name: Stephen W Porges, PhD
role: PRINCIPAL_INVESTIGATOR
References:
citation: 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.
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.
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.
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.
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.
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.