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Non-invasive sources of cells with primary cilia from pediatric and adult patients
BACKGROUND: Ciliopathies give rise to a multitude of organ-specific pathologies; obtaining relevant primary patient material is useful for both diagnostics and research. However, acquisition of primary ciliated cells from patients, particularly pediatric patients, presents multiple difficulties. Bio...
Autores principales: | , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2015
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4450497/ https://www.ncbi.nlm.nih.gov/pubmed/26034581 http://dx.doi.org/10.1186/s13630-015-0017-x |
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author | Ajzenberg, Henry Slaats, Gisela G. Stokman, Marijn F. Arts, Heleen H. Logister, Ive Kroes, Hester Y. Renkema, Kirsten Y. van Haelst, Mieke M. Terhal, Paulien A. van Rooij, Iris A. Keijzer-Veen, Mandy G. Knoers, Nine V. Lilien, Marc R. Jewett, Michael A. Giles, Rachel H. |
author_facet | Ajzenberg, Henry Slaats, Gisela G. Stokman, Marijn F. Arts, Heleen H. Logister, Ive Kroes, Hester Y. Renkema, Kirsten Y. van Haelst, Mieke M. Terhal, Paulien A. van Rooij, Iris A. Keijzer-Veen, Mandy G. Knoers, Nine V. Lilien, Marc R. Jewett, Michael A. Giles, Rachel H. |
author_sort | Ajzenberg, Henry |
collection | PubMed |
description | BACKGROUND: Ciliopathies give rise to a multitude of organ-specific pathologies; obtaining relevant primary patient material is useful for both diagnostics and research. However, acquisition of primary ciliated cells from patients, particularly pediatric patients, presents multiple difficulties. Biopsies and blood samples are invasive, and patients (and their parents) may be reluctant to travel to medical centers, especially for research purposes. We sought to develop non-invasive methods of obtaining viable and ciliated primary cells from ciliopathy patients which could be obtained in the home environment. FINDINGS: We introduce two methods for the non-invasive acquisition of primary ciliated cells. In one approach, we collected spontaneously shed deciduous (milk) teeth from children. Fibroblast-like cells were observed after approximately 2 weeks of culture of fragmented teeth. Secondly, urine samples were collected from children or adults. Cellular content was isolated and after approximately 1 week, renal epithelial cells were observed. Both urine and tooth-derived cells ciliate and express ciliary proteins visible with immunofluorescence. Urine-derived renal epithelial cells (URECs) are amenable to 3D culturing, siRNA knockdown, and ex vivo drug testing. CONCLUSIONS: As evidence continues to accumulate showing that the primary cilium has a central role in development and disease, the need for readily available and ciliated patient cells will increase. Here, we introduce two methods for the non-invasive acquisition of cells with primary cilia. We believe that these cells can be used for further ex vivo study of ciliopathies and in the future, for personalized medicine. |
format | Online Article Text |
id | pubmed-4450497 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2015 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-44504972015-06-02 Non-invasive sources of cells with primary cilia from pediatric and adult patients Ajzenberg, Henry Slaats, Gisela G. Stokman, Marijn F. Arts, Heleen H. Logister, Ive Kroes, Hester Y. Renkema, Kirsten Y. van Haelst, Mieke M. Terhal, Paulien A. van Rooij, Iris A. Keijzer-Veen, Mandy G. Knoers, Nine V. Lilien, Marc R. Jewett, Michael A. Giles, Rachel H. Cilia Short Report BACKGROUND: Ciliopathies give rise to a multitude of organ-specific pathologies; obtaining relevant primary patient material is useful for both diagnostics and research. However, acquisition of primary ciliated cells from patients, particularly pediatric patients, presents multiple difficulties. Biopsies and blood samples are invasive, and patients (and their parents) may be reluctant to travel to medical centers, especially for research purposes. We sought to develop non-invasive methods of obtaining viable and ciliated primary cells from ciliopathy patients which could be obtained in the home environment. FINDINGS: We introduce two methods for the non-invasive acquisition of primary ciliated cells. In one approach, we collected spontaneously shed deciduous (milk) teeth from children. Fibroblast-like cells were observed after approximately 2 weeks of culture of fragmented teeth. Secondly, urine samples were collected from children or adults. Cellular content was isolated and after approximately 1 week, renal epithelial cells were observed. Both urine and tooth-derived cells ciliate and express ciliary proteins visible with immunofluorescence. Urine-derived renal epithelial cells (URECs) are amenable to 3D culturing, siRNA knockdown, and ex vivo drug testing. CONCLUSIONS: As evidence continues to accumulate showing that the primary cilium has a central role in development and disease, the need for readily available and ciliated patient cells will increase. Here, we introduce two methods for the non-invasive acquisition of cells with primary cilia. We believe that these cells can be used for further ex vivo study of ciliopathies and in the future, for personalized medicine. BioMed Central 2015-06-01 /pmc/articles/PMC4450497/ /pubmed/26034581 http://dx.doi.org/10.1186/s13630-015-0017-x Text en © Ajzenberg et al.; licensee BioMed Central. 2015 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. |
spellingShingle | Short Report Ajzenberg, Henry Slaats, Gisela G. Stokman, Marijn F. Arts, Heleen H. Logister, Ive Kroes, Hester Y. Renkema, Kirsten Y. van Haelst, Mieke M. Terhal, Paulien A. van Rooij, Iris A. Keijzer-Veen, Mandy G. Knoers, Nine V. Lilien, Marc R. Jewett, Michael A. Giles, Rachel H. Non-invasive sources of cells with primary cilia from pediatric and adult patients |
title | Non-invasive sources of cells with primary cilia from pediatric and adult patients |
title_full | Non-invasive sources of cells with primary cilia from pediatric and adult patients |
title_fullStr | Non-invasive sources of cells with primary cilia from pediatric and adult patients |
title_full_unstemmed | Non-invasive sources of cells with primary cilia from pediatric and adult patients |
title_short | Non-invasive sources of cells with primary cilia from pediatric and adult patients |
title_sort | non-invasive sources of cells with primary cilia from pediatric and adult patients |
topic | Short Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4450497/ https://www.ncbi.nlm.nih.gov/pubmed/26034581 http://dx.doi.org/10.1186/s13630-015-0017-x |
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