Ciliary Disorientation in Patients with Chronic Upper Respiratory Tract Inflammation

Rayner, Charlotte F. J.; Rutman, Andrew; Dewar, A.; Cole, Peter; Wilson, Robert

doi:10.1164/ajrccm/151.3_pt_1.800

Cited by 61 publications

(31 citation statements)

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“…Consistent with these functional defects, we identified ultrastructural changes (Fig. 1), which are commonly encountered in SCD such as axonemal membrane alterations (13)(14)(15)(16). Thus, our findings confirm that indeed IL-13 negatively affects ciliogenesis and produces a phenotype in suspension cultures of primary human respiratory epithelial cells that resembles SCD.…”

Section: Discussionsupporting

confidence: 84%

Axonemal Localization of the Dynein Component DNAH5 Is Not Altered in Secondary Ciliary Dyskinesia

et al. 2006

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Primary ciliary dyskinesia (PCD) is a heterogeneous genetic disorder characterized by recurrent airway infections and situs inversus in half of affected individuals. Diagnosis currently relies on demonstration of abnormal ciliary ultrastructure or altered ciliary beat. Alterations encountered in secondary ciliary dyskinesia (SCD) caused by inflammation often complicate the diagnostic workup. We have recently shown that in respiratory epithelial cells from PCD patients with outer dynein arm defects the dynein protein DNAH5 is mislocalized and either completely or partially absent from the ciliary axoneme. In this study, we addressed the question whether SCD might affect axonemal DNAH5 localization in respiratory cells. To induce SCD in vitro, we treated primary human respiratory epithelial cell cultures with interleukin-13 (IL-13). Ciliary function and ultrastructure were assessed by high-speed videomicroscopy and transmission electron microscopy, respectively. For in vivo localization of DNAH5, we performed nasal brushing biopsies in patients with evidence of SCD. Expression of DNAH5 was analyzed by immunofluorescence microscopy. IL-13-treated cells showed evidence of SCD. Ciliary beat frequency was significantly reduced and ultrastructural analyses showed axonemal disorganization compared with control cells. High-resolution immunofluorescence studies of respiratory epithelial cells with SCD identified in vitro and in vivo normal axonemal DNAH5 localization. DNAH5 localization is not altered by SCD, indicating a high potential for immunofluorescence analysis as a novel diagnostic tool in PCD. (Pediatr Res 59: [418][419][420][421][422] 2006)

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Section: Discussionsupporting

confidence: 84%

Axonemal Localization of the Dynein Component DNAH5 Is Not Altered in Secondary Ciliary Dyskinesia

et al. 2006

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“…Disorientation results in a larger SD. Typical normal values are SD 10-15% and for PCD with disorientation, 20-25% [30,37]. However, ciliary disorientation can be transient, secondary to infection, so a second sample is taken after treatment to reduce inflammation and eradicate infection before a diagnosis of PCD secondary to disorientation is made.…”

Section: -Diagnostic Algorithm For Primary Ciliary Dyskinesia (Pcd)mentioning

confidence: 99%

Primary ciliary dyskinesia: diagnosis and standards of care

Bush¹,

Cole²,

Hariri³

et al. 1998

Eur Respir J

347

285

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Primary ciliary dyskinesia (PCD) is characterized by disease of the upper and lower respiratory tract, in association with visceral mirror image arrangement in 50% of cases, due to abnormal structure and/or function of cilia. The purpose of this paper is to review the clinical features, diagnosis and management of PCD. Presentations include neonatal respiratory distress, recurrent lower respiratory tract infection, chronic rhinosinusitis and male infertility. PCD enters the differential diagnosis of bronchiectasis, atypical asthma, and unusually severe upper airway disease. Diagnosis is by a cascade of investigations, starting with the saccharin test in patients older than 10 yrs; ciliary beat frequency and pattern on light microscopy; and electron microscopy to assess ciliary morphology and orientation. It is important not to confuse primary and secondary ciliary abnormalities. Nasal nitric oxide is low in PCD, and this measurement shows promise as a screening test for PCD. Diagnosis is important, in order to prevent the development of bronchiectasis and to avoid any unnecessary otorhinolaryngological procedures. Regular follow-up is essential, and management should be multidisciplinary, with input from centres with a special interest in PCD, having access to paediatric and adult chest physicians, otolaryngologists and audiological physicians, physiotherapists, counselling services and fertility clinics. The prognosis is good, but morbidity can be considerable if PCD is incorrectly managed.

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“…The orientation of cilia in normal subjects varied with a disorientation of 14.6+/-3.3° in a study by De Iongh and Rutland [8] and 10.47+/-0.53° by Rayner et al [6]. Our finding is similar to the result of Rautiainen, in which the disorientation was 23.8+/-6.3° in nasal samples and 29.9+/-7.3° in sphenoid sinuses [7].…”

Section: Discussionsupporting

confidence: 88%

“…To determine the coordination of ciliary beating, variations in the orientation of cilia have been previously measured by examining crosssections of the epithelium using an electron microscope [5]. It has been found that ciliary disorientation may occur secondary to inflammation caused by infection [6] and that the measured orientation of cilia may vary in normal subjects [6]-[8]. Ciliary disorientation alone can lead to the clinical syndrome of primary ciliary dyskinesia (PCD) [9].…”

Section: Introductionmentioning

confidence: 99%

Direct measurement of directional disorder for ciliary metachronal wave

Park²,

Lee

et al.

2001 Conference Proceedings of the 23rd Annual International Conference of the IEEE Engineering in Medicine and Biology Society

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Abstract-The interrelationship of cilia and the directional disorder of ciliary metachronal wave were analyzed using digital microscopic images. The degree of synchronization between ciliary beats was determined by the correlation factor between two different spots. Principal axes of inertia were applied to find out the uniphase directions of beating cilia. The standard deviation of wave directions in an ROI was defined as a measure of ciliary wave disorder (CWD). The pooled mean of CWD was 22.9+/-8.73° in ROIs of 8 µ m× 8µ m and 25.4+/-6.46° in 32µ m× 24µ m from the sphenoid sinus mucosa of 5 normal subjects. Our result shows that there is a considerable variation in metachronal wave directions of cilia beating on the epithelium.Keywords -Digital microscopic images, correlation map, uniphase directions of cilia, principal axis of inertia, ciliary wave disorder (CWD). I. INTRODUCTIONThe main task of mucociliary systems in the respiratory tract is to eliminate inhaled particles by the propulsion of mucus. The o verall propulsive effect depends upon the arrangement of the cilia, their metachronal relationships, as well as on the ciliary beat frequency (CBF) [1]. Some methods based on image analysis allow the determination of the CBF of a single cell or multiple ciliated cells simultaneously [2]-[4]. Disorientation of cilia leads to an impairment of mucociliary transport. To determine the coordination of ciliary beating, variations in the orientation of cilia have been previously measured by examining crosssections of the epithelium using an electron microscope [5]. It has been found that ciliary disorientation may occur secondary to inflammation caused by infection [6] and that the measured orientation of cilia may vary in normal subjects [6]-[8]. Ciliary disorientation alone can lead to the clinical syndrome of primary ciliary dyskinesia (PCD) [9]. The directions based on observations of ciliary cross-sections may differ from the metachronal wave directions of cilia beating in media. We have applied principal axis of inertia to the 2-dimensional correlation map calculated from sequential ciliary images. This method directly measures the phase relations and wave directions of multiple cilia beating in culture media. II. MATERIALS AND METHODSThe mucosa in the sphenoid sinus were collected from subjects of no respiratory diseases with informed consent. The ciliary movements were recorded using a CCD camera from an inverted microscope. A personal computer equipped with an image-grabber [IC-Comp ; Imaging Technology , Inc.] converted VCR signals into digital images with a depth of 8 bits. Under the magnification used, one pixel size equated to 0.1µm. The digitized images for each processing were acquired at 30Hz for about 4.27 seconds (128 frames). The whole field of 640 by 480 pixels was divided into equal sized blocks (spots) of 2 by 2 pixels. Gray-level values of all pixels in each spot were summed sequentially from the first frame to the last.Movements of cilia are coordinated in time and space by phase differen...

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Ciliary Disorientation in Patients with Chronic Upper Respiratory Tract Inflammation

Cited by 61 publications

References 0 publications

Axonemal Localization of the Dynein Component DNAH5 Is Not Altered in Secondary Ciliary Dyskinesia

Axonemal Localization of the Dynein Component DNAH5 Is Not Altered in Secondary Ciliary Dyskinesia

Primary ciliary dyskinesia: diagnosis and standards of care

Direct measurement of directional disorder for ciliary metachronal wave

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