Cystic fibrosis (CF), a most deadly genetic disorder, is caused by mutations of CF transmembrane receptor (CFTR) - a chloride channel present at the surface of epithelial cells. In general, two steps have to be involved in treatment of the disease: correction of cellular defects and potentiation to further increase channel opening. Consequently, a combinatorial simultaneous treatment with two drugs with different mechanisms of action, lumacaftor and ivacaftor, has been recently proposed. While lumacaftor is used to correct p.Phe508del mutation (the loss of phenylalanine at position 508) and increase the amount of cell surface-localized CFTR protein, ivacaftor serves as a CFTR potentiator that increases the open probability of CFTR channels. Since the main organ that is affected by cystic fibrosis is the lung, the delivery of drugs directly to the lungs by inhalation has a potential to enhance the efficacy of the treatment of CF and limit adverse side effects upon healthy tissues and organs. Based on our extensive experience in inhalation delivering of drugs by different nanocarriers, we selected nanostructured lipid carriers (NLC) for the delivery both drugs directly to the lungs by inhalation and tested NLC loaded with drugs in vitro (normal and CF human bronchial epithelial cells) and in vivo (homozygote/homozygote bi-transgenic mice with CF). The results show that the designed NLCs demonstrated a high drug loading efficiency and were internalized in the cytoplasm of CF cells. It was found that NLC-loaded drugs were able to restore the expression and function of CFTR protein. As a result, the combination of lumacaftor and ivacaftor delivered by lipid nanoparticles directly into the lungs was highly effective in treating lung manifestations of cystic fibrosis.
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http://dx.doi.org/10.1016/j.jconrel.2019.01.025 | DOI Listing |
Am J Physiol Lung Cell Mol Physiol
January 2025
Division of Pulmonology, Asthma, Cystic Fibrosis, and Sleep, Emory University School of Medicine, Atlanta, GA, USA.
Secondhand smoke exposure (SHSe) is a public health threat for people with cystic fibrosis (CF) and other lung diseases. Primary smoking reduces CFTR channel function, the causative defect in CF. We reported that SHSe worsens respiratory and nutritional outcomes in CF by disrupting immune responses and metabolic signaling.
View Article and Find Full Text PDFPediatr Pulmonol
January 2025
University Hospitals Cleveland Medical Center, Division of Pulmonary, Critical Care and Sleep Medicine, Cleveland, Ohio, USA.
Objective: Although studies have examined changes in C-reactive protein (CRP) during pulmonary exacerbations (PEX) in people with cystic fibrosis (PwCF), few have evaluated CRP profiles across age groups. Here, we characterize age-related CRP responses to PEX treatment.
Methods: We measured CRP concentrations at the beginning and end of intravenous (IV) antibiotic therapy for PEX in 100 pediatric and 147 adult PwCF at 10 US CF Centers.
Pediatr Pulmonol
January 2025
Centre de Ressources et de Compétences pour la Mucoviscidose, CHU Timone-Enfants, Marseille, France.
Introduction: Cellular characteristics of induced sputum (IS) are not investigated in cystic fibrosis (CF) patients.
Objectives: This pilot study, conducted on 17 expectorating CF adolescents, compared sputa obtained the same day, in a stable period, by autogenic drainage (expectorating sputum, ES) and 4 h later after inhaling hypertonic saline (IS).
Results: No difference was noted concerning weight, volume, and percentage of dead cells between the two collection methods.
Front Immunol
January 2025
Inflammation and Immunity in Global Health Program, Research Institute of the McGill University Health Centre, Montreal, QC, Canada.
Background: Factors leading to severe COVID-19 remain partially known. New biomarkers predicting COVID-19 severity that are also causally involved in disease pathogenesis could improve patient management and contribute to the development of innovative therapies. Autophagy, a cytosolic structure degradation pathway is involved in the maintenance of cellular homeostasis, degradation of intracellular pathogens and generation of energy for immune responses.
View Article and Find Full Text PDFCureus
December 2024
Internal Medicine, Kettering Health Network, Kettering, USA.
Aquagenic syringeal acrokeradermatoma (ASA) is a dermatological condition characterized by the transient appearance of edematous, white, translucent papules on the palms, typically triggered by water exposure. While ASA is most commonly associated with cystic fibrosis (CF) and predominantly affects young females, there has been a significant increase in ASA cases since the most recent update in 2015. The COVID-19 pandemic increased the number of patients diagnosed with ASA following exposure to the viral infection.
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