Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders

Henriette H.M. Dreyer; Eleonora Sofie van Tuyll van Serooskerken; Lisa W. Rodenburg; Arnold J.N. Bittermann; Hubertus G.M. Arets; Ellen M.B.P. Reuling; Johannes W. Verweij; Eric G. Haarman; David C. van der Zee; Stefaan H.A.J. Tytgat; Cornelis K. van der Ent; Jeffrey M. Beekman; Gimano D. Amatngalim; Maud Y.A. Lindeboom

doi:10.3390/children10061020

Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders

Henriette H.M. Dreyer^*, Eleonora Sofie van Tuyll van Serooskerken, Lisa W. Rodenburg, Arnold J.N. Bittermann, Hubertus G.M. Arets, Ellen M.B.P. Reuling, Johannes W. Verweij, Eric G. Haarman, David C. van der Zee, Stefaan H.A.J. Tytgat, Cornelis K. van der Ent, Jeffrey M. Beekman, Gimano D. Amatngalim, Maud Y.A. Lindeboom

^*Corresponding author for this work

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Abstract

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.

Original language	English
Article number	1020
Pages (from-to)	1-9
Number of pages	9
Journal	Children (Basel, Switzerland)
Volume	10
Issue number	6
DOIs	https://doi.org/10.3390/children10061020
Publication status	Published - 5 Jun 2023

Keywords

airway epithelium
esophageal atresia
organoids
primary ciliary dyskinesia
tracheal anomaly

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children-10-01020-v2Final published version, 1.92 MBLicence: CC BY

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Dreyer, H. H. M., van Tuyll van Serooskerken, E. S., Rodenburg, L. W., Bittermann, A. J. N., Arets, H. G. M., Reuling, E. M. B. P., Verweij, J. W., Haarman, E. G., van der Zee, D. C., Tytgat, S. H. A. J., van der Ent, C. K., Beekman, J. M., Amatngalim, G. D., & Lindeboom, M. Y. A. (2023). Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders. Children (Basel, Switzerland), 10(6), 1-9. Article 1020. https://doi.org/10.3390/children10061020

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title = "Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders",

abstract = "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.",

keywords = "airway epithelium, esophageal atresia, organoids, primary ciliary dyskinesia, tracheal anomaly",

author = "Dreyer, {Henriette H.M.} and {van Tuyll van Serooskerken}, {Eleonora Sofie} and Rodenburg, {Lisa W.} and Bittermann, {Arnold J.N.} and Arets, {Hubertus G.M.} and Reuling, {Ellen M.B.P.} and Verweij, {Johannes W.} and Haarman, {Eric G.} and {van der Zee}, {David C.} and Tytgat, {Stefaan H.A.J.} and {van der Ent}, {Cornelis K.} and Beekman, {Jeffrey M.} and Amatngalim, {Gimano D.} and Lindeboom, {Maud Y.A.}",

note = "Publisher Copyright: {\textcopyright} 2023 by the authors.",

year = "2023",

month = jun,

day = "5",

doi = "10.3390/children10061020",

language = "English",

volume = "10",

pages = "1--9",

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Dreyer, HHM, van Tuyll van Serooskerken, ES, Rodenburg, LW, Bittermann, AJN, Arets, HGM, Reuling, EMBP, Verweij, JW, Haarman, EG, van der Zee, DC , Tytgat, SHAJ , van der Ent, CK , Beekman, JM , Amatngalim, GD & Lindeboom, MYA 2023, 'Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders', Children (Basel, Switzerland), vol. 10, no. 6, 1020, pp. 1-9. https://doi.org/10.3390/children10061020

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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.

PY - 2023/6/5

Y1 - 2023/6/5

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

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DO - 10.3390/children10061020

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SN - 2227-9067

VL - 10

SP - 1

EP - 9

JO - Children (Basel, Switzerland)

JF - Children (Basel, Switzerland)

IS - 6

M1 - 1020

ER -

Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders

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