Nasal Brushing Sampling and Processing using Digital High Speed Ciliary Videomicroscopy &amp;ndash; Adaptation for the COVID-19 Pandemic

Bricmont, Noémie; Benchimol, Lionel; Poirrier, Anne‐Lise; Grignet, Christine; Seaton, Claire; Chilvers, Mark; Seghaye, Marie-Christine; Louis, Renaud; Lefèbvre, Philippe; Kempeneers, Céline

doi:10.3791/61949

Cited by 6 publications

(7 citation statements)

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“…To establish patient‐specific PCD organoids, we obtained airway organoid (AO) cultures from nasal inferior turbinate brush (NITB) samples collected from healthy and affected individuals with known mutations in PCD‐related genes. Several studies have shown the use of nasal epithelium as model for airway diseases like PCD (Barlocco et al , 1991 ; Hirst et al , 2010 ; Lucas et al , 2014 ; Adil et al , 2017 ; Marthin et al , 2017 ; Shoemark et al , 2017 ; Bricmont et al , 2020 ; Coles et al , 2020 ). The non‐invasively received NITB samples were obtained from 4 patients and 2 healthy controls.…”

Section: Resultsmentioning

confidence: 99%

Modelling of primary ciliary dyskinesia using patient‐derived airway organoids

et al. 2021

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Patient‐derived human organoids can be used to model a variety of diseases. Recently, we described conditions for long‐term expansion of human airway organoids (AOs) directly from healthy individuals and patients. Here, we first optimize differentiation of AOs towards ciliated cells. After differentiation of the AOs towards ciliated cells, these can be studied for weeks. When returned to expansion conditions, the organoids readily resume their growth. We apply this condition to AOs established from nasal inferior turbinate brush samples of patients suffering from primary ciliary dyskinesia (PCD), a pulmonary disease caused by dysfunction of the motile cilia in the airways. Patient‐specific differences in ciliary beating are observed and are in agreement with the patients' genetic mutations. More detailed organoid ciliary phenotypes can thus be documented in addition to the standard diagnostic procedure. Additionally, using genetic editing tools, we show that a patient‐specific mutation can be repaired. This study demonstrates the utility of organoid technology for investigating hereditary airway diseases such as PCD.

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Section: Resultsmentioning

confidence: 99%

Modelling of primary ciliary dyskinesia using patient‐derived airway organoids

et al. 2021

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“…Indeed, the respiratory epithelium can be sampled using a brush, a curette or forceps, usually from the nose [ 34 ]. Moreover, ciliary function varies under differing environmental and physiological conditions, such as pH [ 35 , 36 , 37 ], osmolarity [ 36 , 37 ], oxygen [ 38 ], the presence of drugs [ 39 ], vitamins [ 40 ] or ions [ 41 ] within the medium, the temperature [ 35 , 42 ] (with some centers measuring at 37 °C [ 26 , 43 , 44 , 45 ] and others at lower temperatures [ 7 , 9 , 46 ]) and the time between sampling and video recording [ 47 ]. These conditions need to be strictly regulated, since minor variations may affect ciliary function.…”

Section: Update Of Literature Concerning Ciliary Videomicroscopy Since the 2017 European Respiratory Society Recommendation Concerning CImentioning

confidence: 99%

Ciliary Videomicroscopy: A Long Beat from the European Respiratory Society Guidelines to the Recognition as a Confirmatory Test for Primary Ciliary Dyskinesia

et al. 2021

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Primary ciliary dyskinesia (PCD) is a rare inherited ciliopathy in which respiratory cilia are stationary or dyskinetic. The clinical presentation of PCD is highly non-specific since it includes infections and disorders of the upper (otitis and rhinosinusitis) and lower (neonatal respiratory distress, bronchitis, pneumonia and bronchiectasis) airways, starting in early life. Clinical examination alone does not allow a PCD diagnosis, which relies on several concordant tests, since none are sensitive or specific enough alone. Despite being the most sensitive and specific test to diagnose PCD, digital high-speed videomicroscopy (DHSV) is not sufficiently standardized, preventing its use with complete confidence as a confirmatory diagnostic test for PCD, or its inclusion in a diagnostic algorithm. Since the 2017 ERS recommendations for PCD diagnosis, three main issues remain to be solved in order to optimize DHSV ciliary beating evaluation: the problem in defining an accurate sensitivity and specificity as there is no gold standard method to diagnose all PCD cases, a lack of standardization in the operating procedure for processing respiratory samples, and in the choice of measured parameters (self-operating or not). The development of new automated analysis approaches is promising and will require full clinical validation.

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“…Nasal nitric oxide gas is decreased in most patients with PCD. In addition, defects of the ciliary beat pattern and frequency can be identified by high-speed video microscopy analysis (HSVMA) [26]. HSVMA harbors tremendous rates of sensitivity and specificity for PCD.…”

Section: Pcd Under the Lensmentioning

confidence: 99%

Vitamin D and Primary Ciliary Dyskinesia: A Topic to Be Further Explored

Sergi

2021

Applied Sciences

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Primary ciliary dyskinesia (PCD) is a genetic disease characterized by abnormalities in ciliary structure/function. The diagnosis of PCD relies on a combination of clinical evaluation and ultrastructural (electron microscopic) analysis of the ciliary architecture. This diagnosis may be challenging due to clinical and genetic heterogeneity and artifacts during the ciliary ultrastructure preparation and assessment. Recently, vitamin D supplementation has been proposed for several groups probably suffering from D-hypovitaminosis. Some patients with inflammatory bowel disease may have significant malabsorption, and vitamin D supplementation in these patients is recommended. Two recent reports suggest that a low plasmatic level of this vitamin is present in the PCD population. The utility of vitamin D supplementation may be essential in this group of individuals, and further investigations are warranted. Still, in examining the literature papers, it seems relevant that the authors concentrate solely on lung function in both studies. Future studies should probably target the intestinal function in patients with PCD independently from the vitamin D supplementation to fully evaluate its role.

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Nasal Brushing Sampling and Processing using Digital High Speed Ciliary Videomicroscopy – Adaptation for the COVID-19 Pandemic

Cited by 6 publications

References 0 publications

Modelling of primary ciliary dyskinesia using patient‐derived airway organoids

Modelling of primary ciliary dyskinesia using patient‐derived airway organoids

Ciliary Videomicroscopy: A Long Beat from the European Respiratory Society Guidelines to the Recognition as a Confirmatory Test for Primary Ciliary Dyskinesia

Vitamin D and Primary Ciliary Dyskinesia: A Topic to Be Further Explored

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