Abstract
Background: Cystic fibrosis-related diabetes (CFRD) has become more common due to higher life expectancy with cystic fibrosis. Early recognition and prompt treatment of CFRD leads to improved outcomes. Methods: We performed a network meta-analysis (NMA) in order to identify the most valuable diagnostic metrics for diagnosing CFRD out of available screening tools (index test), using the oral glucose tolerance test as a reference standard. Pooled sensitivity (Se), specificity (Sp), and superiority indices were calculated and used to rank the index tests. Results: A total of 31 articles with 25 index tests were eligible for inclusion. Two-day, continuous glucose monitoring (CGM) ranked the highest (Se: 86% Sp: 76%), followed by glucose measurement from blood capillary samples (Se: 70%, Sp: 82%) and three-day CGM (Se: 96%, Sp: 56%). When we compared the CGM of different durations, two-day CGM performed best (Se: 88%, Sp: 80%), followed by three-day (Se: 96%, Sp: 59%) and six-day CGM (Se: 66%, Sp: 79%). Conclusions: Considering its overall performance ranking, as well as the high sensitivity, two-day CGM appears to be a promising screening test for CFRD.
Highlights
Cystic fibrosis (CF) is an autosomal recessive genetic disease caused by mutations of the cystic fibrosis transmembrane conductance regulator (CFTR) gene
Even though oral glucose tolerance test (OGTT) serves as a basis for defining Cystic fibrosis-related diabetes (CFRD), its thresholds were established in diabetes without CF, and were not designed to detect hyperglycaemiaassociated risk of deterioration in lung function or under-nutrition [39,45]
Our results indicate that continuous glucose monitoring (CGM) performs well in diagnosing CFRD, which raises the idea of using CGM as an alternative reference standard instead of OGTT in further studies
Summary
Cystic fibrosis (CF) is an autosomal recessive genetic disease caused by mutations of the cystic fibrosis transmembrane conductance regulator (CFTR) gene. The pathogenic mutations cause abnormal chloride transport across secretory epithelial cells, leading to thick, sticky mucus production, mainly affecting the lungs and the digestive system [1]. Due to advances in treatment and diagnostics, the life expectancy of CF patients has increased over the last decade. Previously rare extra-pulmonary complications are highly prevalent. Risk factors for developing cystic fibrosis-related diabetes (CFRD) include pancreatic insufficiency, severe genotype (deltaF508 homozygotes), and increased age; it affects approximately 20% of adolescent and 40–50% of adult individuals with CF [2,3]. In the initial stage of the disease, insulin and glucagon
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