Down Syndrome Speech Intelligibility Diagnostic Treatment Study

NCT04059354 | Terminated Results

• 4 other identifiers: 2019-0706A348700VCRGE/WAISMAN/WAISMANProtocol Version 12/10/2019
• Study Type: interventional
• Target: 3
• Locations: 1 country
• Sites: 1

Brief Summary

The 'Down syndrome speech intelligibility diagnostic treatment study' is a pilot clinical intervention study assessing the efficacy of an integrative speech treatment approach for individuals with Down syndrome (DS). This study will use a single-subject design with 10 male participants with DS, aged 7 to 16 years. The proposed intervention, combines two treatment approaches that are documented to be efficacious with other speech disorders: 1) The Cycles Phonological Remediation Approach, designed for highly unintelligible speech, as guided by the Hodson Assessment of Phonological Patterns (HAPP-3); and 2) Script training, a functional speech motor intervention that promotes phrase-level productions. During the twelve-week intensive intervention period, both approaches will be adapted to prioritize the production of words with low vowels to incorporate increased practice of the low-front and low-back vowels. This modification is based on the investigator's perceptual, acoustic and anatomic research findings. Outcome measures will include perceptual, intelligibility, and acoustic analyses obtained from acoustic recordings. These measures will be used to assess treatment efficacy, and to gain insight into the motoric versus anatomic limitations in speech production commonly present in individuals with DS.

Conditions

Down Syndrome

Keywords

Speech intelligibility

Outcome Measures

Primary Outcomes (4)

• Hodson Assessment of Phonological Patterns-3 (HAPP-3) Occurrence Scores

  The HAPP-3 test is designed to assess the phonological disorder severity in children, guide intervention and document progress. This 50-word test categorizes phonological deviations into two patterns to guide intervention: 1) Word/Syllable Structure Omissions (129 total instances): including omission of syllables, consonant clusters, and single consonants; 2) Consonant Category Deficiencies (144 instances): including consonant substitutions and assimilations. The range of the combined sum of errors from both patterns, or the Total Occurrence of Major Phonological Deviations (TOMPD) score, is 0-273 with a severity of phonological disorder rating of 0-150 (0: No errors; 1-50: Mild; 51-100: Moderate; 101-150: Severe; \>150: Profound). Lower TOMPD scores imply fewer errors and better speech intelligibility, and higher scores indicate more errors and reduced intelligibility. A decrease in TOMPD score post intervention implicates improvement in speech production.

  Baseline (pre-treatment), post-treatment (12 weeks), 1-month post-treatment (16weeks), and 6 months post-treatment (36 weeks)

• Percentages of the Top 3 Major Phonological Deviations of HAPP-3 Addressed in Intervention

  The HAPP-3 Comprehensive Phonological Evaluation Record Form specifies, for each phonological deviation pattern, the number of instances the 50-words contain the various deviation types. For example, 1) Word/Syllable Structure Omissions include 39 Consonant Clusters and 16 Syllables; 2) Consonant Category Deficiencies include 19 Liquids /r, l/ and 42 Stridents /s, f/. This enables the calculation of 'Percent of Occurrence' for each error type, allowing personalized intervention where each speaker's deficiencies with higher percentages are targeted first, using the Cycles phonological approach. Here, the top three occurrences of major phonological deviations and deficiencies that were addressed during intervention are reported: Deficiencies of Liquids and Stridents (for all participants), omission of Consonant Clusters for participants 1 and 3, and omission of Syllables for participant 2. Decrease in percent occurrence post-treatment signifies improvement in phonological skills.

  Baseline (pre-treatment), post-treatment (12 weeks), 1 month post-treatment (16 weeks), and 6 months post-treatment (36 weeks)

• Pre- and Post-Intervention Percentage of Words and Vowels Correctly Identified by Naïve Listeners in Perceptual Ratings of Vocal Tract Development Lab (VTLab) Stimuli

  Two adult naïve listeners will orthographically transcribe participants' speech recordings of VTLab stimuli to assess word and vowel intelligibility. These stimuli, designed for lifespan studies of vowel acoustics, were chosen due to the lack of standardized speech samples for clinical assessment and the predominant focus of most formal tests on consonants, despite vowels' contribution to intelligibility. VTLab stimuli consist of 25 monosyllabic words, including five words each for four extreme vowels (/i, u, æ, ɑ/) and the central vowel (/ə/) as in 'eat', 'hoot', 'hat', 'hot', and 'hug'. Intelligibility is measured by comparing transcriptions to intended target words and calculating the percentage correct for total words, all aforementioned vowels, and the extreme front-back low vowels (/æ/ and /ɑ/), which are reportedly more difficult to produce in speakers with Down syndrome. An increase in correct percentages indicates improved speech intelligibility.

  Baseline (pre-treatment), post-treatment (12 weeks), 1 month post-treatment (16 weeks), and 6 months post-treatment (36 weeks)

• Pre- and Post-Intervention Vowel Formant Frequencies F1 and F2 for /ɑ/ and /æ/ in Recorded Productions of VTLab Stimuli

  The vowel portion of the VTLab word stimuli was analyzed acoustically. Pre- and post-intervention formant frequencies F1 and F2 of seven words per vowel /æ/ and /ɑ/ are reported here, as the intervention provided practice for their distinct front-back productions. F1 and F2 are critical for determining vowel type and specify acoustic space that is related to speech intelligibility, serving as a proxy for articulatory working space (i.e. reflecting dimensionality of tongue movement/position). See Detailed Description section for additional information. Reported here are formant frequencies averaged across all pre- and post-conditions, a feasible comparison given similarity in the F1 and F2 increase/decrease trend across conditions. The F2-F1 difference correlates with tongue advancement, increasing as the tongue moves forward and decreasing as it retracts.

  Baseline (pre-treatment), post-treatment (12 weeks)

Secondary Outcomes (3)

• Percentage of the HAPP-3 50-Word Stimuli With Correct Phonetic Transcriptions by Trained Listeners Using the International Phonetic Alphabet (IPA)

  Baseline (pre-treatment), post-treatment (12 weeks), 1 month post-treatment (16 weeks), and 6 months post-treatment (36 weeks)

• Number of Scripts Produced

  during treatment, up to12 weeks

• Scores for the Intelligibility in Context Scale as Rated by Parent

  Baseline (pre-treatment), post-treatment (12 weeks), 1 month post-treatment (16 weeks), and 6 months post-treatment (36 weeks)

Study Arms (1)

Speech therapy

EXPERIMENTAL

Treatment: Direct speech therapy will be provided for a 12 week period with up to three one-hour sessions per week (36 sessions per participant) as tolerated and 30 minutes weekly home practice.

Behavioral: Speech therapy addressing phonology and motor learning

Interventions

Speech therapy addressing phonology and motor learning BEHAVIORAL

Determine the efficacy of a newly devised intensive speech treatment plan, to use with individuals with Down syndrome, that combines two treatment approaches: Cycles Phonological Remediation Approach and Script training, while adapting each to prioritize use of practice words with low vowels given research findings that the low vowels /ɑ/ as in 'hot' and /æ/as in 'hat' are less intelligible than the high vowels /i/ as in 'heat' and /u/ as in 'boot'; also, vowel acoustic analyses that revealed word-intelligibility to improve when the production of the low vowels /ɑ/ and /æ/ had more distinctive second formants/vocal tract resonances (a measure that is associated with tongue front-back movement). Each intervention session will include 40-50 minutes of the adapted 'Cycles Phonological Remediation Approach' focusing on articulatory/phonological training at the syllable/word level, and 10-20 minutes of the adapted Script program for motor training at the sentence level.

Also known as: Speech motor learning

Speech therapy

Eligibility Criteria

• Age: 7 Years - 16 Years
• Gender Eligibility Details: At this time, only males are eligible to participate.
• Healthy Volunteers: No
• Age Groups: Child (0-17)

You may qualify if:

• Males with a diagnosis of Down syndrome between the ages of 7 and 16 years
• Primary language is English
• Interested in improving their speech intelligibility.
• All participants must consent/assent to participate. As this is a multiple single subject design, services are available to all motivated participants who are willing to commit to the time/durations described in the diagnostic treatment.

You may not qualify if:

• Primary language is not American English
• Do not have a diagnosis of Down syndrome
• Female
• Ages birth-6 and 17 years or older
• Individuals who have a co-occurring diagnosis that affects communication abilities \[e.g., diagnosed as deaf with cochlear implants, Autism, only use an alternative/augmentative communication (AAC) device to communicate\],
• Severe hearing loss which will limit their ability to participate in the treatment
• Individuals with a known mild or moderate hearing loss or current speech- language therapy services will not be excluded, but this information will be requested in the Parent Questionnaire. In addition, access to their current individualized education program (IEP) will be requested.

Sponsors & Collaborators

• University of Wisconsin, Madison: lead
• Office of the Vice Chancellor for Research and Graduate Education: collaborator
• Wisconsin Alumni Research Foundation: collaborator

Study Sites (1)

Waisman Center, 1500 Highland Avenue

Madison, Wisconsin, 53705, United States

Location

Related Publications (5)

• Wild A, Vorperian HK, Kent RD, Bolt DM, Austin D. Single-Word Speech Intelligibility in Children and Adults With Down Syndrome. Am J Speech Lang Pathol. 2018 Feb 6;27(1):222-236. doi: 10.1044/2017_AJSLP-17-0002.

  PMID: 29214307 BACKGROUND

• Vorperian HK, Kent RD, Lee Y, Bolt DM. Corner vowels in males and females ages 4 to 20 years: Fundamental and F1-F4 formant frequencies. J Acoust Soc Am. 2019 Nov;146(5):3255. doi: 10.1121/1.5131271.

  PMID: 31795713 BACKGROUND

• Kent RD, Eichhorn J, Wilson EM, Suk Y, Bolt DM, Vorperian HK. Auditory-Perceptual Features of Speech in Children and Adults With Down Syndrome: A Speech Profile Analysis. J Speech Lang Hear Res. 2021 Apr 14;64(4):1157-1175. doi: 10.1044/2021_JSLHR-20-00617. Epub 2021 Mar 31.

  PMID: 33789057 BACKGROUND

• Vorperian HK, Kent RD, Lee Y, Buhr KA. Vowel Production in Children and Adults With Down Syndrome: Fundamental and Formant Frequencies of the Corner Vowels. J Speech Lang Hear Res. 2023 Apr 12;66(4):1208-1239. doi: 10.1044/2022_JSLHR-22-00510. Epub 2023 Apr 4.

  PMID: 37015000 BACKGROUND

• Eichhorn JA, Pearl-Soloman N, Vorperian HK. Treatment for Speech Intelligibility in Children with Down Syndrome: A Pilot Clinical Trial. Poster presented at the 2022 Annual Convention of the American Speech-Language Hearing Association in New Orleans, LA.

  RESULT

Related Links

• University of Wisconsin, Madison, Waisman Center, Vocal Tract Development Lab

MeSH Terms

Conditions

Down Syndrome; Speech Intelligibility

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; Speech; Verbal Behavior; Communication; Behavior

Limitations and Caveats

COVID-19 pandemic-related study termination limited enrollment and hindered our ability to address initial study challenges of obtaining multiple baseline measures for two participants, 1-month post-intervention assessment for another, and 6-month post for all participants. Also, rescheduling the final assessment for the third participant was not possible given fatigue-related behavioral issues. Individualized single-subject design intervention is optimal given individual differences in DS.

Results Point of Contact

• Title: Houri K. Vorperian, PhD, Senior Scientist Emerita
• Organization: University of Wisconsin - Madison, Waisman Center

vorperian@waisman.wisc.edu

608-213-3257

Study Officials

• Houri K Vorperian, PhD, CCC-SLP

  University of Wisconsin, Madison - Waisman Center

  PRINCIPAL INVESTIGATOR

Publication Agreements

• PI is Sponsor Employee: Yes

Study Design

• Study Type: interventional
• Phase: not applicable
• Allocation: NA
• Masking: NONE
• Purpose: TREATMENT
• Intervention Model: SINGLE GROUP
• Sponsor Type: OTHER
• Responsible Party: SPONSOR

Model Details: Multiple single-subject design

Study Record Dates

• First Submitted: August 14, 2019
• First Posted: August 16, 2019
• Study Start: September 5, 2019
• Primary Completion: February 28, 2020
• Study Completion: February 28, 2020
• Last Updated: February 7, 2025
• Results First Posted: February 7, 2025

Record last verified: 2025-02

Data Sharing

• IPD Sharing: Will not share

Locations

US (1)