Can intensive practice of articulation exercises improve speech in people with Friedreich’s ataxia?
| ISRCTN | ISRCTN84887840 |
|---|---|
| DOI | https://doi.org/10.1186/ISRCTN84887840 |
| Secondary identifying numbers | UEC22/24 |
- Submission date
- 01/08/2022
- Registration date
- 04/08/2022
- Last edited
- 04/08/2022
- Recruitment status
- No longer recruiting
- Overall study status
- Completed
- Condition category
- Nervous System Diseases
Plain English Summary
Background and study aims
Friedreich's ataxia is an inherited disorder that affects some of the body's nerves and causes difficulty walking, a loss of sensation in the arms and legs, and impaired speech. Evidence for the effectiveness of speech treatment to improve communication for people with Friedreich's ataxia is steadily increasing. A few different treatment approaches have recently been reported, each of them showing some benefits for different aspects important to communication, including the quality of the voice, intelligibility of speech, and communication confidence. However, whilst they each showed promise, no study has been able to show conclusively so far that it can achieve the desired communication benefits for people with progressive ataxia across the board. The critical next step to achieve this is to run a larger scale trial that tells us whether speech therapy is indeed effective across people with ataxia and also, which approach works best. Before this, researchers want to carry out one more pilot study of a newly developed model to help them decide whether this should be considered in the larger trial in future. They need to find out how acceptable this is for people with Friedreich's ataxia, and whether there is any indication it results in better communication.
Who can participate?
Patients aged 18 years and over with Friedreich’s ataxia who experience mild to moderate speech or voice problems, which means people can still understand most of what they say although they might need to listen more carefully
What does the study involve?
The treatment is called LSVT Artic. It includes intensive articulation practice in four 1 hour sessions per week over 4 weeks. It has had positive results for intelligibility in people with Parkinson’s disease. The treatment will be offered online. Participants will be assessed twice before treatment at 4-week intervals and twice after treatment, immediately and 6 months later.
What are the possible benefits and risks of participating?
This therapy has not been done with people with ataxia before and it is not known whether it works. It is hoped that participants' communication will improve as part of the treatment. If it does not, the researchers might be able to suggest other methods that could have more success depending on their speech symptoms. Whatever the outcomes, it is hoped that the study will have wider benefits for people with ataxia and lead to improvements in care in the long term.
The treatment is relatively intensive and participants might experience some fatigue following sessions. The researchers will closely monitor their response to treatment and make adjustments during its course where necessary. There are no known negative side effects of the treatment or any of the assessment tasks.
Where is the study run from?
Strathclyde University (UK)
When is the study starting and how long is it expected to run for?
January 2022 to October 2023
Who is funding the study?
Ataxia UK
Who is the main contact?
Prof Anja Lowit, a.lowit@strath.ac.uk
Contact information
Principal Investigator
School of Psychological Sciences and Health
Strathclyde University
40 George St
Glasgow
G1 1QE
United Kingdom
 ORCID ID |
0000-0003-0842-584X |
|---|---|
| Phone | +44 (0)7986080537 |
| a.lowit@strath.ac.uk |
Study information
| Study design | Single-centre interventional study |
|---|---|
| Primary study design | Interventional |
| Secondary study design | Non randomised study |
| Study setting(s) | Home |
| Study type | Treatment |
| Participant information sheet | 42164_PIS_v2.0_22July2022.pdf |
| Scientific title | A feasibility study of LSVT Artic to improve speech performance in people with Friedreich's ataxia |
| Study hypothesis | It is hypothesised that LSVT Artic will improve participants' intelligibility immediately after treatment and 6 months later. |
| Ethics approval(s) | Approved 25/03/2022, Strathclyde University Ethics Committee (University of Strathclyde, 16 Richmond St, Glasgow, G1 1XQ, UK; +44 (0)141 548 3707; ethics@strath.ac.uk), ref UEC22/24 |
| Condition | Friedreich's ataxia |
| Intervention | Participants are offered a course of 16 sessions of LSVT Artic over the course of 4 weeks. Prior to this they will have completed a no treatment phrase of the same duration. LSVT Artic consists of four consecutive sessions per week lasting 60 minutes focusing on clear speech production. This will be practised on individual sound segments initially, followed by single words, short then longer phrases, passage reading and free conversation. In addition to the weekly sessions, participants will be asked to complete homework exercises. |
| Intervention type | Behavioural |
| Primary outcome measure | Speech outcome measures consisting of: 1. Speed of syllable repetitions (p, t, k, p-t, n-g, oo-ee & oo-ah) is expressed as the average number of repetitions in 5 seconds over 5 attempts at baseline (weeks 1 & 4) and post-treatment (weeks 8 and 32); 2. Clarity of syllable repetitions is judged by trained listeners over 5 attempts at baseline (weeks 1 & 4) and post-treatment (weeks 8 and 32); 3. Intelligibility and naturalness of the reading passage are rated with a Direct Magnitude Evaluation method by trained listeners at weeks 1, 4, 8 and 32; 4. Intelligibility and naturalness of 1 minute monologue samples are rated on a visual analogue scale by trained listeners at weeks 1, 4, 8 and 32. |
| Secondary outcome measures | 1. Vowel space is measured through LPC analysis of word repetitions at weeks 1, 4, 8 and 32; 2. Communication confidence is rated on a 1-10 scale by participants at weeks 1, 8 and 32; 3. Communication participation is measured using the Communication Participation Item Bank (Baylor et al. 2013) at weeks 1, 8 and 32. |
| Overall study start date | 10/01/2022 |
| Overall study end date | 31/10/2023 |
Eligibility
| Participant type(s) | Patient |
|---|---|
| Age group | Adult |
| Lower age limit | 18 Years |
| Sex | Both |
| Target number of participants | 5 |
| Participant inclusion criteria | 1. Confirmed diagnosis of Friedreich's ataxia 2. Mild to moderate speech impairment 3. No previous SLT input 4. No history of other neurological or developmental problems affecting speech or language 5. Fluent English speaker 6. Enough hearing and visual ability to follow the therapy programme over Zoom and read large print therapy materials 7. Access to a computer, tablet or smartphone and a good enough internet connection to take part in online therapy. 8. Above 18 years of age 9. Not in receipt of other speech treatment from the NHS or private sector during the duration of this study, or have received such treatment in the past |
| Participant exclusion criteria | 1. Diagnosis of another type of ataxia 2. Previously in receipt of SLT 3. Presence of other neurological or speech and language problems 4. English language skills too poor to allow reliable evaluation of speech patterns 5. Below 18 years of age 6. No access to necessary technology 7. Vision and hearing too impaired to allow participation in assessment and treatment sessions |
| Recruitment start date | 04/04/2022 |
| Recruitment end date | 31/03/2023 |
Locations
Countries of recruitment
- Scotland
- United Kingdom
Study participating centre
40 George St
Glasgow
G1 1QE
United Kingdom
Sponsor information
University/education
16 Richmond St
Glasgow
G1 1XQ
Scotland
United Kingdom
| Phone | +44 (0)141 5524400 |
|---|---|
| ethics@strath.ac.uk | |
| Website | http://www.strath.ac.uk/ |
"ROR" |
https://ror.org/00n3w3b69 |
Funders
Funder type
Charity
Private sector organisation / Other non-profit organizations
- Alternative name(s)
- Ataxia
- Location
- United Kingdom
Results and Publications
| Intention to publish date | 31/12/2023 |
|---|---|
| Individual participant data (IPD) Intention to share | Yes |
| IPD sharing plan summary | Stored in publicly available repository |
| Publication and dissemination plan | Results will be published in a peer-reviewed journal |
| IPD sharing plan | Data will be shared openly through the university repository. Data will include an excel file with acoustic analyses and listener ratings that will be stored on the Strathclyde University data repository. Where participants have consented to share their speech recordings, acoustic data files will also be available. Data will be accessible by contacting the repository administrator (researchdataproject@strath.ac.uk) or the principal investigator. All data will be fully anonymised and only gender, date of birth and medical diagnosis information will be shared. Data will be available indefinitely from the date of publication of results. |
Study outputs
| Output type | Details | Date created | Date added | Peer reviewed? | Patient-facing? |
|---|---|---|---|---|---|
| Participant information sheet | version 2.0 | 22/07/2022 | 04/08/2022 | No | Yes |
| Participant information sheet | Poster for recruitment version 2.0 |
22/07/2022 | 04/08/2022 | No | Yes |
Additional files
- 42164_PIS_v2.0_22July2022.pdf
- 42164_PIS (poster)_v2.0_22July2022.pdf
- Poster for recruitment
Editorial Notes
04/08/2022: Trial's existence confirmed by Strathclyde University Ethics Committee
