Functional analysis of cilia and ciliated epithelial ultrastructure in healthy children and young adults

Chilvers, Mark; Rutman, Andrew; O’Callaghan, Christopher

doi:10.1136/thorax.58.4.333

Cited by 126 publications

(143 citation statements)

References 28 publications

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“…The nasal brush biopsy sample from each of these 42 children contained at least seven normal epithelial edges for ciliary function studies. PCD was excluded in all 42 children by ciliary function studies and analysis of ultrastructure, using previously published methodology and normative data [5,7]. The images recorded from these 42 children also included a variable number of disrupted epithelial edges that belong to the five different categories.…”

Section: Methodsmentioning

confidence: 99%

“…Also, CBF and beat pattern are commonly used outcome measures in studies that assess the effect of pathogenic organisms or pharmacological agents on the ciliated respiratory epithelium. The recent advent of high speed digital video microscopy allows us to precisely assess the ciliary beat pattern, in addition to the measurement of CBF [5] and its use is being adopted internationally for the diagnostic testing of PCD [6].…”

mentioning

confidence: 99%

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Disrupted ciliated epithelium shows slower ciliary beat frequency and increased dyskinesia

Thomas¹,

Rutman²,

O’Callaghan³

2009

European Respiratory Journal

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Ciliary function studies for the diagnosis of primary ciliary dyskinesia (PCD) are usually performed on nasal brush biopsy samples. It is not uncommon to find disrupted epithelial strips of tissue in these samples, and occasionally throughout a sample. The aim of the present study was to determine if cilia on disrupted ciliated epithelial edges beat with a normal pattern and frequency similar to that of cilia on undisrupted edges.Nasal brush biopsy samples from 42 children in whom the diagnosis of PCD was excluded were assessed. The epithelial strips were categorised into five groups: intact undisrupted ciliated epithelial edge, ciliated epithelial edge with minor projections, ciliated epithelial edge with major projections, an isolated ciliated cell on an epithelial edge and single unattached ciliated cells. Ciliary beat frequency and beat pattern of 50 samples from each group were determined using high speed digital video microscopy.The cilia on epithelial edges with varying degrees of disruption showed significantly reduced beat frequency and significantly increased dyskinesia compared with those on intact, undisrupted ciliated epithelial edges.Ideally, the assessment of ciliary beat pattern and frequency for PCD diagnosis should only be performed on undisrupted ciliated edges.

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Section: Methodsmentioning

confidence: 99%

mentioning

confidence: 99%

Disrupted ciliated epithelium shows slower ciliary beat frequency and increased dyskinesia

Thomas¹,

Rutman²,

O’Callaghan³

2009

European Respiratory Journal

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“…Analysis also permits measurement of ciliary beat frequency. Specific beat patterns have been shown to be related to particular ultrastructural defects [79][80][81]. This analysis is particularly useful in identifying patients who have ciliary dyskinesia due to an ultrastructural defect for which beat frequency is normal, e.g.…”

Section: Ciliary Beat Pattern and Frequency Analysismentioning

confidence: 99%

Primary ciliary dyskinesia: a consensus statement on diagnostic and treatment approaches in children

Barbato¹,

Frischer²,

Kuehni³

et al. 2009

European Respiratory Journal

458

587

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Primary ciliary dyskinesia (PCD) is associated with abnormal ciliary structure and function, which results in retention of mucus and bacteria in the respiratory tract, leading to chronic oto-sino-pulmonary disease, situs abnormalities and abnormal sperm motility.The diagnosis of PCD requires the presence of the characteristic clinical phenotype and either specific ultrastructural ciliary defects identified by transmission electron microscopy or evidence of abnormal ciliary function.Although the management of children affected with PCD remains uncertain and evidence is limited, it remains important to follow-up these patients with an adequate and shared care system in order to prevent future lung damage.This European Respiratory Society consensus statement on the management of children with PCD formulates recommendations regarding diagnostic and therapeutic approaches in order to permit a more accurate approach in these patients. Large well-designed randomised controlled trials, with clear description of patients, are required in order to improve these recommendations on diagnostic and treatment approaches in this disease.

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“…Samples of cilia were obtained during routine flexible bronchoscopy by brushing the trachea with a 2‐mm cytology brush (Olympus Endotherapy, Olympus, Skinjuku‐ku, Tokyo) 10. Samples were analyzed using a digital high‐speed video camera (MotionPro X4, IDT, CA) at a frame rate of 500 frames s–1 to allow assessment of ciliary beat frequency and pattern.…”

Section: Methodsmentioning

confidence: 99%

Tissue-Engineered Tracheal Replacement in a Child: A 4-Year Follow-Up Study

Hamilton

Kanani

Roebuck

et al. 2015

American Journal of Transplantation

161

145

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In 2010, a tissue‐engineered trachea was transplanted into a 10‐year‐old child using a decellularized deceased donor trachea repopulated with the recipient's respiratory epithelium and mesenchymal stromal cells. We report the child's clinical progress, tracheal epithelialization and costs over the 4 years. A chronology of events was derived from clinical notes and costs determined using reference costs per procedure. Serial tracheoscopy images, lung function tests and anti‐HLA blood samples were compared. Epithelial morphology and T cell, Ki67 and cleaved caspase 3 activity were examined. Computational fluid dynamic simulations determined flow, velocity and airway pressure drops. After the first year following transplantation, the number of interventions fell and the child is currently clinically well and continues in education. Endoscopy demonstrated a complete mucosal lining at 15 months, despite retention of a stent. Histocytology indicates a differentiated respiratory layer and no abnormal immune activity. Computational fluid dynamic analysis demonstrated increased velocity and pressure drops around a distal tracheal narrowing. Cross‐sectional area analysis showed restriction of growth within an area of in‐stent stenosis. This report demonstrates the long‐term viability of a decellularized tissue‐engineered trachea within a child. Further research is needed to develop bioengineered pediatric tracheal replacements with lower morbidity, better biomechanics and lower costs.

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Functional analysis of cilia and ciliated epithelial ultrastructure in healthy children and young adults

Cited by 126 publications

References 28 publications

Disrupted ciliated epithelium shows slower ciliary beat frequency and increased dyskinesia

Disrupted ciliated epithelium shows slower ciliary beat frequency and increased dyskinesia

Primary ciliary dyskinesia: a consensus statement on diagnostic and treatment approaches in children

Tissue-Engineered Tracheal Replacement in a Child: A 4-Year Follow-Up Study

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