Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders
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 u...
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| Format: | Article |
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MDPI AG
2023-06-01
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| Series: | Children |
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| Online Access: | https://www.mdpi.com/2227-9067/10/6/1020 |
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| author | 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 |
| author_facet | 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 |
| author_sort | Henriette H. M. Dreyer |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-03-11T02:37:44Z |
| format | Article |
| id | doaj.art-49357c20bf05479cb1c93c1c52b71d08 |
| institution | Directory Open Access Journal |
| issn | 2227-9067 |
| language | English |
| last_indexed | 2024-03-11T02:37:44Z |
| publishDate | 2023-06-01 |
| publisher | MDPI AG |
| record_format | Article |
| series | Children |
| spelling | doaj.art-49357c20bf05479cb1c93c1c52b71d082023-11-18T09:50:27ZengMDPI AGChildren2227-90672023-06-01106102010.3390/children10061020Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract DisordersHenriette H. M. Dreyer0Eleonora Sofie van Tuyll van Serooskerken1Lisa W. Rodenburg2Arnold J. N. Bittermann3Hubertus G. M. Arets4Ellen M. B. P. Reuling5Johannes W. Verweij6Eric G. Haarman7David C. van der Zee8Stefaan H. A. J. Tytgat9Cornelis K. van der Ent10Jeffrey M. Beekman11Gimano D. Amatngalim12Maud Y. A. Lindeboom13Department of Pediatric Pulmonology, Wilhelmina Children’s Hospital, University Medical Center, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Pulmonology, Wilhelmina Children’s Hospital, University Medical Center, 3508 AB Utrecht, The NetherlandsPediatric Upper Gastrointestinal and Airway Treatment Center, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Pulmonology, Wilhelmina Children’s Hospital, University Medical Center, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsDepartment of Paediatric Pulmonology, Emma Children’s Hospital, Amsterdam UMC, 1105 AZ Amsterdam, The NetherlandsDepartment of Pediatric Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Pulmonology, Wilhelmina Children’s Hospital, University Medical Center, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Pulmonology, Wilhelmina Children’s Hospital, University Medical Center, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Pulmonology, Wilhelmina Children’s Hospital, University Medical Center, 3508 AB Utrecht, The NetherlandsDepartment of Pediatric Surgery, Wilhelmina Children’s Hospital, University Medical Center Utrecht, 3508 AB Utrecht, The NetherlandsEsophageal 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.https://www.mdpi.com/2227-9067/10/6/1020esophageal atresiatracheal anomalyairway epitheliumorganoidsprimary ciliary dyskinesia |
| spellingShingle | 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 Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders Children esophageal atresia tracheal anomaly airway epithelium organoids primary ciliary dyskinesia |
| title | Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders |
| title_full | Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders |
| title_fullStr | Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders |
| title_full_unstemmed | Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders |
| title_short | Airway Epithelial Cultures of Children with Esophageal Atresia as a Model to Study Respiratory Tract Disorders |
| title_sort | airway epithelial cultures of children with esophageal atresia as a model to study respiratory tract disorders |
| topic | esophageal atresia tracheal anomaly airway epithelium organoids primary ciliary dyskinesia |
| url | https://www.mdpi.com/2227-9067/10/6/1020 |
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