The changing face of cystic fibrosis research: challenges of multi-centre microbiology cohort studies

Abstract

INTRODUCTION: Infection is a primary cause of progressive pulmonary disease in people with cystic fibrosis (pwCF). Researchers frequently utilize data registries or centre-based cohort studies to study infections in CF. We examine challenges encountered in conducting two multi-centre non-interventional investigator-led cohort studies undertaken a decade apart. METHODS: The Australian Clonal Pseudomonas Study (ACPinCF, 1294 participants) and the National NTM in CF study (1314 participants) leveraged clinical practice, with data was supplied by clinics and the Australian CF Data Registry. Diagnostic laboratories performed cultures according to local protocols. RESULTS: Site approval times were lengthy; 16 months for the ACPinCF study and between 21-44 months for the National NTM in CF study. The latter required individual contract negotiation with each clinical site as a requirement of governance approval. Changing clinical demands and unforeseen challenges such as the SARS-CoV-2 pandemic significantly impacted the NTM study. Despite these challenges, participant retention remained robust, with minimal loss to follow-up. Changes in CF centre leadership and health service policy on cost-recovery for research participation further exacerbates the challenges of maintaining longitudinal cohort CF studies. CONCLUSIONS: Conducting and sustaining longitudinal non-interventional studies present significant challenges. Even after costs for centre participation, sample processing, data curation, data analysis and results output are covered by grant funding, these studies will still heavily depend on significant support provided by the centres without substantial funding.