The 'Lombard Effect' in Patients Affected by Adductor Laryngeal Dystonia (LoQVAdSD)

The 'Lombard Effect' in Patients Affected by Adductor Laryngeal Dystonia: a Pilot Study

Adductory spasmodic dysphonia (AdSD) is a rare condition characterised by irregular and uncontrolled voice interruptions, most commonly affecting women around the age of 45. The diagnosis is clinical and usually requires evaluation by several specialists. The exact cause is not known, but a disturbance of the motor system is hypothesised, probably related to various causes such as loss of cortical inhibition or problems with sensory input.

Neuroimaging studies have shown hyperactivity in various brain regions during speech production in patients with AdSD, but it is still unclear whether this hyperactivity is due to a malfunction of auditory and somatosensory feedback or an impairment of motor programming.

Recent research indicates that patients with AdSD show excessive muscle activation during phonation, probably due to abnormal processing of auditory feedback. This suggests that intervention in the auditory system may offer new treatment opportunities.

The proposed study aims to describe the acoustic, auditory-perceptual and subjective voice and speech changes in AdSD subjects during the Quick-Lombard Test (LT), a test that assesses vocal response under noisy conditions.

Study Overview

• Status: Not yet recruiting
• Conditions: Adductor Spasmodic Dysphonia
• Intervention / Treatment: Other: Lombard test

• Study Type: Interventional
• Enrollment (Estimated): 18
• Phase: Not Applicable

Contacts and Locations

• Study Contact: Name: Maria Raffaella Marchese, MD, PhD; Phone Number: +39-3391144556; Email: raffaellamarchese@gmail.com
• Study Contact Backup: Name: Lucia DAlatri, MD, PhD; Phone Number: +39-630154244; Email: lucia.dalatri@policlinicogemelli.it

Study Locations

• Italy
  • Roma, Italy, 00168
    • Fondazione Policlinico Universitario A. Gemelli IRCCS
    • Contact: Maria Raffaella Marchese; Phone Number: +390630154439; Email: mariaraffaella.marchese@policlinicogemelli.it
    • Contact: Maria Raffaella Marchese; Phone Number: +390630155701; Email: direzione.scientifica@policlinicogemelli.it
    • Principal Investigator: Maria Raffaella Marchese

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: Adult; Older Adult
• Accepts Healthy Volunteers: Yes

Description

Inclusion Criteria:

• Native Italian-speaking patients;
• Normal hearing, hearing threshold < 20 dB HL for frequencies from 0.5 to 4 KHz;
• Age >18 years and <65 years;
• Written informed consent.

Exclusion Criteria:

• Non-Italian-speaking patients.
• Patients undergoing treatment for dystonia.
• Previous laryngeal surgery.
• Patient undergoing speech therapy.
• Patient undergoing dopaminergic therapy.
• Inability to sustain phonation of sufficient duration >3 seconds or to perform sufficient tests to assess vocal outcomes.
• Lack of written informed consent.

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Other
• Allocation: Non-Randomized
• Interventional Model: Single Group Assignment
• Masking: None (Open Label)

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Experimental: Case group; patients affected by adductor laryngeal dystonia	Other: Lombard test; In this study a single-subject enrolment session will be set up, with the presence of a dedicated physician for informed consent, the performance of a tonal audiometric examination and a clinical practice phoniatric examination for all enrolled subjects, the collection of voice recordings both before and during the Lombard Test (LT) and the administration of the rating scales both before and after the LT performance. For each subject, voice recordings will be made of the vowel /a/ sustained for at least four seconds with a comfortable tone and volume and of the speech obtained from the reading aloud and volume of normal conversation of a phonetically balanced text (The Desert) lasting about 1 minute, first in a silent condition and then in the presence of noise. Specifically, for the LT, each subject will be exposed to a free-fieldwhite noise of speech(SWN) administered through headphones. The intensity of the noise will be 70 dB SPL. Voice recordings will be made in a quiet room.
Active Comparator: Control group; healthy patients	Other: Lombard test; In this study a single-subject enrolment session will be set up, with the presence of a dedicated physician for informed consent, the performance of a tonal audiometric examination and a clinical practice phoniatric examination for all enrolled subjects, the collection of voice recordings both before and during the Lombard Test (LT) and the administration of the rating scales both before and after the LT performance. For each subject, voice recordings will be made of the vowel /a/ sustained for at least four seconds with a comfortable tone and volume and of the speech obtained from the reading aloud and volume of normal conversation of a phonetically balanced text (The Desert) lasting about 1 minute, first in a silent condition and then in the presence of noise. Specifically, for the LT, each subject will be exposed to a free-fieldwhite noise of speech(SWN) administered through headphones. The intensity of the noise will be 70 dB SPL. Voice recordings will be made in a quiet room.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
OMNI Vocal Effort Scale	It is a validated instrument for the assessment of vocal effort perceived by the patient with AdSD. The OMNI-VES is a visual analogue scale 0-10 with graphic representation of vocal effort. The OMNI-VES will be administered under silent conditions and after the LT	At the time of enrollement
Auditory-perceptual assessment	Blinded perceptual assessment, using the Global Grading Scale - Hoarseness - Straining - Asthenia - Straining (GRBAS) 0-3 (0 = normal; 1 = mild; 2 = moderate; 3 = severe) will be performed on the recorded voice samples by two speech therapists who will not be involved in the patients' care. Each voice sample will be anonymised and randomly distributed to the assessors.	At the time of enrollement
Maximum Phonation Time	The maximum phonatory time (TMF) will be obtained by asking the subject to sustain the vowel /a/ as long as possible in one breath. The longest of the three attempts will be calculated as TMF in seconds. The TMF will be obtained before and during LT.	at the time of enrollement
Average Frequency (Hz)	The Average Frequency is the fundamental frequency of the voice (pitch) usually ranges from approximately 30-300 Hz, but this varies according to different speakers: typically males' pitch ranges from 50-180Hz and females from 80-250Hz, so we usually set the pitch range to a reasonable range of 50-400Hz for general usage	At the time of enrollement
Maximum Frequency (Hz)	The Maximum Frequency (Hz) is the highest frequency recorded within the voice signal	At the time of enrollement
Maximum Intensity (dB SPL)	The Maximum Intensity (dB SPL) parameter refers to the highest intensity of the recorded voice signal	At the time of enrollement
Degree of Voice Interruptions (%)	Degree of Voice Interruptions (%) is useful for the evaluation of temporary vocal interruption	At the time of enrollement
Cepstral Peak Prominence-Smoothed (CPPS)	Cepstral Peak Prominence-Smoothed (CPPS) has been defined as a spectrum of a spectrum, whereby an inverse Fast Fourier Transform of the natural log of a frequency-domain spectrum is applied, transforming the spectrum to the time domain. The cepstral peak (CP) is a representation of the most dominant peak in the cepstrum - normally the fundamental frequency (fo) in a type I signal. After its first applications to voice was established the method of normalizing the amplitude of the overall cepstrum using a linear regression line. When applied to the cepstrum, the level difference (dB) between the cepstral peak and the regression line at the same quefrency provides the cepstral peak prominence (CPP). The CPPS values obtained from speech and vowel tasks proved reliable in predicting overall grade of dysphonia as well as breathiness.	At the time of enrollement
Number of Voice Interruptions	Number of Voice Interruptions is useful for the evaluation of temporary vocal interruption	At the time of enrollement

Collaborators and Investigators

• Sponsor: Fondazione Policlinico Universitario Agostino Gemelli IRCCS
• Investigators: Principal Investigator: Maria Raffaella Marchese, Fondazione Policlinico Universitario A. Gemelli, IRCCS

Study record dates

Study Major Dates

• Study Start (Estimated): May 1, 2024
• Primary Completion (Estimated): April 30, 2025
• Study Completion (Estimated): May 1, 2025

Study Registration Dates

• First Submitted: April 19, 2024
• First Submitted That Met QC Criteria: May 2, 2024
• First Posted (Actual): May 7, 2024

Study Record Updates

• Last Update Posted (Actual): May 7, 2024
• Last Update Submitted That Met QC Criteria: May 2, 2024
• Last Verified: April 1, 2024

More Information

Terms related to this study

• Keywords: spasmodic dysphonia; laryngeal dystonia; Lombard effect; auditory system
• Additional Relevant MeSH Terms: Central Nervous System Diseases; Nervous System Diseases; Respiratory Tract Diseases; Respiration Disorders; Neurologic Manifestations; Otorhinolaryngologic Diseases; Movement Disorders; Dyskinesias; Signs and Symptoms, Respiratory; Laryngeal Diseases; Voice Disorders; Dystonia; Dystonic Disorders; Dysphonia; Hoarseness
• Other Study ID Numbers: ID 6117

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: NO
• IPD Plan Description: The data are paper-based

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No