Development of a core outcome set for studies of interventions designed to assess and manage voice, swallowing, mucus, and cough in adults with laryngotracheal stenosis

Background

Adult acquired laryngotracheal stenosis (LTS) is a rare, chronic condition with a heterogenous range of causes and treatment options. LTS has a significant symptom burden, with research so far focusing on breathing outcomes. There is an understanding that LTS and its treatment can impact voice, swallowing, mucus, and cough, but there are no evidence-based guidelines available for clinicians working with LTS patients. Interventions for these outcomes are guided by clinician experience and are based on therapeutic strategies used with other conditions, for example use of instrumental assessment and tailored exercise programmes for voice and swallowing difficulties, or recommending nebuliser protocols for mucus and cough.

A qualitative study focusing on the lived experience of adults with LTS showed that whilst they will always prioritise breathing, they experience significant challenges and limitation to their quality of life because of voice and swallowing difficulties. These symptoms had a functional impact on all aspects of their lives whether emotional, physical, or medical. These impacts were present irrespective of the stage of their LTS and patients stressed the need for individualised treatment and information to support them. A follow up survey study focused on mucus and cough indicated that these are hugely problematic symptoms for patients with no consistent management or treatment.

A 2018 systematic review of voice and swallowing outcomes in adults with LTS who underwent airway reconstruction surgery identified 20 observational studies with heterogenous outcomes and measures relating to voice and swallowing. We are currently repeating the searches of this review to capture newer studies including those involving mucus and cough outcomes.

There are no registered core outcome set studies for LTS in the COMET database.

Aim
To establish a core outcome measurement set (COMS) for studies of interventions designed to assess and manage voice and swallowing in adults with laryngotracheal stenosis.

Objectives
1. To characterise selection and definition of outcomes and measures used in existing studies of interventions designed to assess and manage voice and swallowing in adults with laryngotracheal stenosis; and
2. To obtain consensus on a COMS for future studies of interventions designed to assess and manage voice and swallowing in adults with laryngotracheal stenosis

Methods
Item generation
1. A systematic review has already been undertaken to extract data on primary and secondary outcomes reported, measures used and measurement time-points (1). Since this review took place in 2018, we will re-run searches to capture new studies that were not included at this time.
2. Focus groups with patients and family members: we will conduct focus groups with patients, family members and other relevant stakeholders as to outcomes they consider important to measure in future studies of interventions designed to assess and manage voice, swallowing, mucus, and cough in adults with laryngotracheal stenosis

Consensus Building
1. We will conduct a modified Delphi study to establish consensus on outcomes for inclusion in a core outcome set. We will recruit an international expert panel representing key stakeholders and end users including former patients and family members.
2. If consensus on a core outcome set is not achieved via the Delphi process, we will host a virtual consensus meeting using nominal group technique and voting to establish the core outcomes.
3. We will hold a second virtual consensus meeting using nominal group technique and voting to establish appropriate measures for the core outcomes.

Contributors

Principal Investigators:
Dr Gemma Clunie MRCSLT, PhD
Dr Amy Freeman-Sanderson CPSP, PhD
Professor Louise Rose RN, PhD

Collaborators:
Professor Guri Sandhu MD
Professor Caroline Alexander PhD
Catherine Anderson, patient advisor
Dr Justin Roe PhD
Dr Chadwan Al Yaghchi PhD
Dr Andrew Kinshuck PhD
Professor Reza Nouraei PhD
Dr Emilia Dronkers PhD

Further Study Information

Current Stage: Ongoing
Date: June 2023 - July 2024
Funding source(s): Kings College London, Faculty of Nursing, Midwifery & Palliative Care, Seedcorn funding


Health Area

Disease Category: Ear, nose, & throat

Disease Name: Laryngotracheal stenosis

Target Population

Age Range: 18 - 90

Sex: Either

Nature of Intervention: Any

Stakeholders Involved

- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Patient/ support group representatives
- Researchers

Study Type

- COS for clinical trials or clinical research

Method(s)

- Consensus meeting
- Delphi process
- Focus group(s)
- Interview
- Literature review

Methods
Item generation
1. A systematic review has already been undertaken to extract data on primary and secondary outcomes reported, measures used and measurement time-points (1). Since this review took place in 2018, we will re-run searches to capture new studies that were not included at this time.
2. Focus groups with patients and family members: we will conduct focus groups with patients, family members and other relevant stakeholders as to outcomes they consider important to measure in future studies of interventions designed to assess and manage voice, swallowing, mucus, and cough in adults with laryngotracheal stenosis

Consensus Building
1. We will conduct a modified Delphi study to establish consensus on outcomes for inclusion in a core outcome set. We will recruit an international expert panel representing key stakeholders and end users including former patients and family members.
2. If consensus on a core outcome set is not achieved via the Delphi process, we will host a virtual consensus meeting using nominal group technique and voting to establish the core outcomes.
3. We will hold a second virtual consensus meeting using nominal group technique and voting to establish appropriate measures for the core outcomes.

Linked Studies

    No related studies


Related Links

    No related links