TY - JOUR
T1 - Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders
AU - Dreyer, Henriette H. M.
AU - van Tuyll van Serooskerken, Eleonora Sofie
AU - Rodenburg, Lisa W.
AU - Bittermann, Arnold J. N.
AU - Arets, Hubertus G. M.
AU - Reuling, Ellen M. B. P.
AU - Verweij, Johannes W.
AU - Haarman, Eric G.
AU - van der Zee, David C.
AU - Tytgat, Stefaan H. A. J.
AU - van der Ent, Cornelis K.
AU - Beekman, Jeffrey M.
AU - Amatngalim, Gimano D.
AU - Lindeboom, Maud Y. A.
N1 - Publisher Copyright: © 2023 by the authors.
PY - 2023/6/1
Y1 - 2023/6/1
N2 - Esophageal atresia (EA) is a rare birth defect in which respiratory tract disorders are a major cause of morbidity. It remains unclear whether respiratory tract disorders are in part caused by alterations in airway epithelial cell functions such as the activity of motile cilia. This can be studied using airway epithelial cell culture models of patients with EA. Therefore, the aim of this study was to evaluate the feasibility to culture and functionally characterize motile cilia function in the differentiated air–liquid interface cultured airway epithelial cells and 3D organoids derived from nasal brushings and bronchoalveolar lavage (BAL) fluid from children with EA. We demonstrate the feasibility of culturing differentiated airway epithelia and organoids of nasal brushings and BAL fluid of children with EA, which display normal motile cilia function. EA patient-derived airway epithelial cultures can be further used to examine whether alterations in epithelial functions contribute to respiratory disorders in EA.
AB - Esophageal atresia (EA) is a rare birth defect in which respiratory tract disorders are a major cause of morbidity. It remains unclear whether respiratory tract disorders are in part caused by alterations in airway epithelial cell functions such as the activity of motile cilia. This can be studied using airway epithelial cell culture models of patients with EA. Therefore, the aim of this study was to evaluate the feasibility to culture and functionally characterize motile cilia function in the differentiated air–liquid interface cultured airway epithelial cells and 3D organoids derived from nasal brushings and bronchoalveolar lavage (BAL) fluid from children with EA. We demonstrate the feasibility of culturing differentiated airway epithelia and organoids of nasal brushings and BAL fluid of children with EA, which display normal motile cilia function. EA patient-derived airway epithelial cultures can be further used to examine whether alterations in epithelial functions contribute to respiratory disorders in EA.
KW - airway epithelium
KW - esophageal atresia
KW - organoids
KW - primary ciliary dyskinesia
KW - tracheal anomaly
UR - http://www.scopus.com/inward/record.url?scp=85163651662&partnerID=8YFLogxK
U2 - https://doi.org/10.3390/children10061020
DO - https://doi.org/10.3390/children10061020
M3 - Article
C2 - 37371252
SN - 2227-9067
VL - 10
JO - Children
JF - Children
IS - 6
M1 - 1020
ER -