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3. Crowdsourcing Airway Annotations in Chest Computed Tomography Images

Author

Cheplygina, Veronika, Perez-Rovira, Adria, Kuo, Wieying, Tiddens, Harm A. W. M., and de Bruijne, Marleen

Subjects
Computer Science - Computer Vision and Pattern Recognition, Computer Science - Human-Computer Interaction
Abstract

Measuring airways in chest computed tomography (CT) scans is important for characterizing diseases such as cystic fibrosis, yet very time-consuming to perform manually. Machine learning algorithms offer an alternative, but need large sets of annotated scans for good performance. We investigate whether crowdsourcing can be used to gather airway annotations. We generate image slices at known locations of airways in 24 subjects and request the crowd workers to outline the airway lumen and airway wall. After combining multiple crowd workers, we compare the measurements to those made by the experts in the original scans. Similar to our preliminary study, a large portion of the annotations were excluded, possibly due to workers misunderstanding the instructions. After excluding such annotations, moderate to strong correlations with the expert can be observed, although these correlations are slightly lower than inter-expert correlations. Furthermore, the results across subjects in this study are quite variable. Although the crowd has potential in annotating airways, further development is needed for it to be robust enough for gathering annotations in practice. For reproducibility, data and code are available online: \url{http://github.com/adriapr/crowdairway.git}.

Published
2020
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4. Spectral Data Augmentation Techniques to quantify Lung Pathology from CT-images

Author

Kayal, Subhradeep, Dubost, Florian, Tiddens, Harm A. W. M., and de Bruijne, Marleen

Subjects
Electrical Engineering and Systems Science - Image and Video Processing, Computer Science - Computer Vision and Pattern Recognition
Abstract

Data augmentation is of paramount importance in biomedical image processing tasks, characterized by inadequate amounts of labelled data, to best use all of the data that is present. In-use techniques range from intensity transformations and elastic deformations, to linearly combining existing data points to make new ones. In this work, we propose the use of spectral techniques for data augmentation, using the discrete cosine and wavelet transforms. We empirically evaluate our approaches on a CT texture analysis task to detect abnormal lung-tissue in patients with cystic fibrosis. Empirical experiments show that the proposed spectral methods perform favourably as compared to the existing methods. When used in combination with existing methods, our proposed approach can increase the relative minor class segmentation performance by 44.1% over a simple replication baseline., Comment: 5 pages including references, accepted as Oral presentation at IEEE ISBI 2020

Published
2020
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6. The effect of azithromycin on structural lung disease in infants with cystic fibrosis (COMBAT CF): a phase 3, randomised, double-blind, placebo-controlled clinical trial

Author

Stick, Stephen M, Foti, Alexia, Ware, Robert S, Tiddens, Harm A W M, Clements, Barry S, Armstrong, David S, Selvadurai, Hiran, Tai, Andrew, Cooper, Peter J, Byrnes, Catherine A, Belessis, Yvonne, Wainwright, Claire, Jaffe, Adam, Robinson, Philip, Saiman, Lisa, and Sly, Peter D

Published
2022
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7. The effect of inhaled hypertonic saline on lung structure in children aged 3–6 years with cystic fibrosis (SHIP-CT): a multicentre, randomised, double-blind, controlled trial

Author

Andrinopoulou, Eleni-Rosalina, Anthony, Margaret M., Au, Jacky, Belessis, Yvonne, Bonte, Merlijn, Chen, Yuxin, Cheney, Joyce, Clem, Charles, Clements, Barry, Cooper, Peter, Dasiewicz, Alison, Davis, Stephanie D., Davis, Miriam, de Boeck, Kris, de Marchis, Matteo, De Wachter, Elke, Delaisi, Bertrand, Delaup, Véronique, DeRicco, Adrienne, Foti, Alexia, Gan, Richard, Garriga, Laura, Gartner, Silvia, Genatossio, Alan, Grogan, Sam, Hilton, Jodi, Hinckley Stukovsky, Karen D, Hoppe, Jordana E., Janssens, Hettie M., Jensen, Renee, Johnson, Robin, Kemner-van de Corput, Mariette P.C., Klein, Brendan, Kronmal, Richard A., Lucca, Francesca, Lucidi, Vincencina, Montemitro, Enza, Nahidi, Lily, Nielsen, Kim G., Pearce, Kasey, Pittman, Jessica E, Powers, Michael, Prentice, Carley, Pressler, Tania, Ratjen, Felix, Rayment, Jonathan H, Reix, Philippe, Retsch-Bogart, George, Riera, Luis, Robinson, Phil, Robinson, Paul, Rosenfeld, Margaret, Sanders, Don B., Sandoval, Rodrigo A., Sandvik, Rikke Mulvad, Saunders, Clare, Siegel, Molly, Smith, Julie, Solomon, Melinda, Stanojevic, Sanja, Stick, Stephen Michael, Tai, Andrew, Tiddens, Harm A.W.M., van de Puttelaar, Jorien, Van den Brande, Christel, van Straten, Marcel, Vermeulen, Francois, Volpi, Sonia, Wainwright, Claire E., Weiner, Daniel J., Yuan, Yi, Zaimeddine, Sarah, Tiddens, Harm A W M, Davis, Stephanie D, and Kronmal, Richard A

Published
2022
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8. Early Experiences with Crowdsourcing Airway Annotations in Chest CT

Author

Cheplygina, Veronika, Perez-Rovira, Adria, Kuo, Wieying, Tiddens, Harm A. W. M., and de Bruijne, Marleen

Subjects
Computer Science - Computer Vision and Pattern Recognition
Abstract

Measuring airways in chest computed tomography (CT) images is important for characterizing diseases such as cystic fibrosis, yet very time-consuming to perform manually. Machine learning algorithms offer an alternative, but need large sets of annotated data to perform well. We investigate whether crowdsourcing can be used to gather airway annotations which can serve directly for measuring the airways, or as training data for the algorithms. We generate image slices at known locations of airways and request untrained crowd workers to outline the airway lumen and airway wall. Our results show that the workers are able to interpret the images, but that the instructions are too complex, leading to many unusable annotations. After excluding unusable annotations, quantitative results show medium to high correlations with expert measurements of the airways. Based on this positive experience, we describe a number of further research directions and provide insight into the challenges of crowdsourcing in medical images from the perspective of first-time users.

Published
2017
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9. Structural Lung Disease and Clinical Phenotype in Bronchiectasis Patients: The EMBARC CT Study.

Author

Pieters, Angelina L. P., van der Veer, Tjeerd, Meerburg, Jennifer J., Andrinopoulou, Eleni-Rosalina, van der Eerden, Menno M., Ciet, Pierluigi, Aliberti, Stefano, Burgel, Pierre-Regis, Crichton, Megan L., Shoemark, Amelia, Goeminne, Pieter C., Shteinberg, Michal, Loebinger, Michael R., Haworth, Charles S., Blasi, Francesco, Tiddens, Harm A. W. M., Caudri, Daan, and Chalmers, James D.

Subjects
CILIARY motility disorders, LUNG diseases, ATELECTASIS, BRONCHIECTASIS, DYSKINESIAS, CHRONIC obstructive pulmonary disease, MYCOBACTERIAL diseases
Abstract

Rationale: Chest computed tomography (CT) scans are essential to diagnose and monitor bronchiectasis (BE). To date, few quantitative data are available about the nature and extent of structural lung abnormalities (SLAs) on CT scans of patients with BE. Objectives: To investigate SLAs on CT scans of patients with BE and the relationship of SLAs to clinical features using the EMBARC (European Multicenter Bronchiectasis Audit and Research Collaboration) registry. Methods: CT scans from patients with BE included in the EMBARC registry were analyzed using the validated Bronchiectasis Scoring Technique for CT (BEST-CT). The subscores of this instrument are expressed as percentages of total lung volume. The items scored are atelectasis/consolidation, BE with and without mucus plugging (MP), airway wall thickening, MP, ground-glass opacities, bullae, airways, and parenchyma. Four composite scores were calculated: total BE (i.e., BE with and without MP), total MP (i.e., BE with MP plus MP alone), total inflammatory changes (i.e., atelectasis/consolidation plus total MP plus ground-glass opacities), and total disease (i.e., all items but airways and parenchyma). Measurements and Main Results: CT scans of 524 patients with BE were analyzed. Mean subscores were 4.6 (range, 2.3–7.7) for total BE, 4.2 (1.2–8.1) for total MP, 8.3 (3.5–16.7) for total inflammatory changes, and 14.9 (9.1–25.9) for total disease. BE associated with primary ciliary dyskinesia was associated with more SLAs, whereas chronic obstructive pulmonary disease was associated with fewer SLAs. Lower FEV1, longer disease duration, Pseudomonas aeruginosa and nontuberculous mycobacterial infections, and severe exacerbations were all independently associated with worse SLAs. Conclusions: The type and extent of SLAs in patients with BE are highly heterogeneous. Strong relationships between radiological disease and clinical features suggest that CT analysis may be a useful tool for clinical phenotyping. [ABSTRACT FROM AUTHOR]

Published
2024
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13. CT-quantified pulmonary artery volume associates independently with higher mortality in smokers with and without COPD

Author

van der Veer, Tjeerd, primary, Kwee, Anastasia Kimberley Ay-Lan, additional, Gallardo Estrella, Leticia, additional, Andrinopoulou, Elrozy R., additional, Charbonnier, Jean Paul, additional, Humphries, Stephen M., additional, De Jong, Pim A., additional, Lynch, David A., additional, Tiddens, Harm A. W. M., additional, and Pompe, Esther, additional

Published
2023
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15. Higher pulmonary artery and vein volumes are associated with disease severity and mortality in smokers with and without COPD

Author

Kwee, A. K. A. L., primary, Van Der Veer, T., additional, Gallardo Estrella, L., additional, Andrinopoulou, Eleni Rosalina, additional, Charbonnier, Jean-Paul, additional, Tiddens, Harm A. W. M., additional, Humphries, Stephen M., additional, Lynch, David A., additional, De Jong, Pim A., additional, and Pompe, E., additional

Published
2023
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16. Increased volume of pulmonary veins on CT associates independently with higher mortality in smokers with and without COPD

Author

van der Veer, Tjeerd, primary, Kwee, Anastasia Kimberley Ay-Lan, additional, Gallardo Estrella, Leticia, additional, Andrinopoulou, Elrozy R., additional, Charbonnier, Jean Paul, additional, Humphries, Stephen M., additional, De Jong, Pim A., additional, Lynch, David A., additional, Tiddens, Harm A. W. M., additional, and Pompe, Esther, additional

Published
2023
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18. Early Experiences with Crowdsourcing Airway Annotations in Chest CT

Author

Cheplygina, Veronika, Perez-Rovira, Adria, Kuo, Wieying, Tiddens, Harm A. W. M., de Bruijne, Marleen, Hutchison, David, Series editor, Kanade, Takeo, Series editor, Kittler, Josef, Series editor, Kleinberg, Jon M., Series editor, Mattern, Friedemann, Series editor, Mitchell, John C., Series editor, Naor, Moni, Series editor, Pandu Rangan, C., Series editor, Steffen, Bernhard, Series editor, Terzopoulos, Demetri, Series editor, Tygar, Doug, Series editor, Weikum, Gerhard, Series editor, Carneiro, Gustavo, editor, Mateus, Diana, editor, Peter, Loïc, editor, Bradley, Andrew, editor, Tavares, João Manuel R. S., editor, Belagiannis, Vasileios, editor, Papa, João Paulo, editor, Nascimento, Jacinto C., editor, Loog, Marco, editor, Lu, Zhi, editor, Cardoso, Jaime S., editor, and Cornebise, Julien, editor

Published
2016
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21. Airway Disease in Children with Primary Ciliary Dyskinesia: Impact of Ciliary Ultrastructure Defect and Genotype

Author

Kinghorn, BreAnna, primary, Rosenfeld, Margaret, additional, Sullivan, Erin, additional, Onchiri, Frankline, additional, Ferkol, Thomas W., additional, Sagel, Scott D., additional, Dell, Sharon D., additional, Milla, Carlos, additional, Shapiro, Adam J., additional, Sullivan, Kelli M., additional, Zariwala, Maimoona A., additional, Pittman, Jessica E., additional, Mollica, Federico, additional, Tiddens, Harm A. W. M., additional, Kemner-van de Corput, Mariette, additional, Knowles, Michael R., additional, Davis, Stephanie D., additional, and Leigh, Margaret W., additional

Published
2023
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22. COllaborative Neonatal Network for the first European CPAM Trial (CONNECT): a study protocol for a randomised controlled trial

Author

Kersten, Casper M, primary, Hermelijn, Sergei M, additional, Dossche, Louis W J, additional, Muthialu, Nagarajan, additional, Losty, Paul D, additional, Schurink, Maarten, additional, Rietman, André B, additional, Poley, Marten J, additional, van Rosmalen, Joost, additional, Zanen - van den Adel, Tabitha P L, additional, Ciet, Pierluigi, additional, von der Thüsen, Jan, additional, Brosens, Erwin, additional, Ijsselstijn, Hanneke, additional, Tiddens, Harm A W M, additional, Wijnen, Rene M H, additional, and Schnater, J Marco, additional

Published
2023
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23. The effect of CFTR modulators on structural lung disease in cystic fibrosis

Author

Mok, L Clara, Garcia-Uceda, Antonio, Cooper, Matthew N, Kemner-Van De Corput, Mariette, De Bruijne, Marleen, Feyaerts, Nathalie, Rosenow, Tim, De Boeck, Kris, Stick, Stephen, Tiddens, Harm A W M, Mok, L Clara, Garcia-Uceda, Antonio, Cooper, Matthew N, Kemner-Van De Corput, Mariette, De Bruijne, Marleen, Feyaerts, Nathalie, Rosenow, Tim, De Boeck, Kris, Stick, Stephen, and Tiddens, Harm A W M

Abstract

Background: Newly developed quantitative chest computed tomography (CT) outcomes designed specifically to assess structural abnormalities related to cystic fibrosis (CF) lung disease are now available. CFTR modulators potentially can reduce some structural lung abnormalities. We aimed to investigate the effect of CFTR modulators on structural lung disease progression using different quantitative CT analysis methods specific for people with CF (PwCF). Methods: PwCF with a gating mutation (Ivacaftor) or two Phe508del alleles (lumacaftor-ivacaftor) provided clinical data and underwent chest CT scans. Chest CTs were performed before and after initiation of CFTR modulator treatment. Structural lung abnormalities on CT were assessed using the Perth Rotterdam Annotated Grid Morphometric Analysis for CF (PRAGMA-CF), airway-artery dimensions (AA), and CF-CT methods. Lung disease progression (0-3 years) in exposed and matched unexposed subjects was compared using analysis of covariance. To investigate the effect of treatment in early lung disease, subgroup analyses were performed on data of children and adolescents aged <18 years. Results: We included 16 modulator exposed PwCF and 25 unexposed PwCF. Median (range) age at the baseline visit was 12.55 (4.25-36.49) years and 8.34 (3.47-38.29) years, respectively. The change in PRAGMA-CF %Airway disease (-2.88 (-4.46, -1.30), p = 0.001) and %Bronchiectasis extent (-2.07 (-3.13, -1.02), p < 0.001) improved in exposed PwCF compared to unexposed. Subgroup analysis of paediatric data showed that only PRAGMA-CF %Bronchiectasis (-0.88 (-1.70, -0.07), p = 0.035) improved in exposed PwCF compared to unexposed. Conclusion: In this preliminary real-life retrospective study CFTR modulators improve several quantitative CT outcomes. A follow-up study with a large cohort and standardization of CT scanning is needed to confirm our findings.

Published
2023

24. Improvement in Lung Clearance Index and Chest Computed Tomography Scores with Elexacaftor/Tezacaftor/Ivacaftor Treatment in People with Cystic Fibrosis Aged 12 Years and Older -- The RECOVER Trial.

Author

McNally, Paul, Lester, Karen, Stone, Gavin, Elnazir, Basil, Williamson, Michael, Cox, Des, Linnane, Barry, Kirwan, Laura, Rea, David, O'Regan, Paul, Semple, Tom, Saunders, Clare, Tiddens, Harm A. W. M., McKone, Edward, and Davies, Jane C.

Abstract

Rationale: Clinical trials have shown that use of elexacaftor/ tezacaftor/ivacaftor (ETI) is associated with improvements in sweat chloride, pulmonary function, nutrition, and quality of life in people with cystic fibrosis (CF). Little is known about the impact of ETI on ventilation inhomogeneity and lung structure. Objectives: RECOVER is a real-world study designed to measure the impact of ETI in people with CF. The primary endpoints were lung clearance (lung clearance index; LCI2.5) and FEV1. Secondary endpoints included spirometry-controlled chest computed tomography (CT) scores. Methods: The study was conducted in seven sites in Ireland and the United Kingdom. Participants ages 12 years and older who were homozygous for the F508del mutation (F508del/F508del) or heterozygous for F508del and a minimum-function mutation (F508del/MF) were recruited before starting ETI and were followed up over 12 months. LCI2.5 was measured using nitrogen multiple breath washout (MBW) at baseline and at 6 and 12 months. Spirometry was performed as per the criteria of the American Thoracic Society and the European Respiratory Society. Spirometry-controlled chest CT scans were performed at baseline and at 12 months. CT scans were scored using the Perth Rotterdam Annotated Grid Morphometric Analysis (PRAGMA) system. Other outcome measures include weight, height, Cystic Fibrosis Quality of Life Questionnaire--Revised (CFQ-R), and sweat chloride. Measurements and Main Results: One hundred seventeen people with CF ages 12 and older were recruited to the study. Significant improvements were seen in LCI scores (-2.5; 95% confidence interval [CI], -3.0, 22.0) and in the percents predicted for FEV1 (8.9; 95% CI, 7.0, 10.9), FVC (6.6; 95% CI, 4.9, 8.3), and forced expiratory flow between 25% and 75% of expired volume (12.4; 95% CI, 7.8, 17.0). Overall PRAGMA-CF scores reflecting airway disease improved significantly (-3.46; 95% CI, -5.23, -1.69). Scores for trapped air, mucus plugging, and bronchial wall thickening improved significantly, but bronchiectasis scores did not. Sweat chloride levels decreased in both F508del/F508del (-43.1; 95% CI, -47.4, 238.9) and F508del/MF (-42.8; 95% CI, -48.5, -37.2) groups. Scores on the Respiratory Domain of the CFQ-R improved by 14.2 points (95% CI, 11.3, 17.2). At 1 year, sweat chloride levels were significantly lower for the F508del/F508del group compared with scores for the F508del/MF group (33.93 vs. 53.36, P,0.001). Conclusions: ETI is associated with substantial improvements in LCI2.5, spirometry, and PRAGMA-CF CT scores in people with CF ages 12 years and older. ETI led to improved nutrition and quality of life. People in the F508del/F508del group had significantly lower sweat chloride on ETI treatment compared with the F508del/MF group. [ABSTRACT FROM AUTHOR]

Published
2023
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25. Management of respiratory tract exacerbations in people with cystic fibrosis: Focus on imaging

Author

Landini, Nicholas, primary, Ciet, Pierluigi, additional, Janssens, Hettie M., additional, Bertolo, Silvia, additional, Ros, Mirco, additional, Mattone, Monica, additional, Catalano, Carlo, additional, Majo, Fabio, additional, Costa, Stefano, additional, Gramegna, Andrea, additional, Lucca, Francesca, additional, Parisi, Giuseppe Fabio, additional, Saba, Luca, additional, Tiddens, Harm A. W. M., additional, and Morana, Giovanni, additional

Published
2023
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26. Airway Inflammation in Children with Primary Ciliary Dyskinesia

Author

Sagel, Scott D., primary, Kupfer, Oren, additional, Wagner, Brandie D., additional, Davis, Stephanie D., additional, Dell, Sharon D., additional, Ferkol, Thomas W., additional, Hoppe, Jordana E., additional, Rosenfeld, Margaret, additional, Sullivan, Kelli M., additional, Tiddens, Harm A. W. M., additional, Knowles, Michael R., additional, and Leigh, Margaret W., additional

Published
2023
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30. 129Xe and Free‐Breathing 1H Ventilation MRI in Patients With Cystic Fibrosis: A Dual‐Center Study

Author

Marshall, Helen, primary, Voskrebenzev, Andreas, additional, Smith, Laurie J., additional, Biancardi, Alberto M., additional, Kern, Agilo L., additional, Collier, Guilhem J., additional, Wielopolski, Piotr A., additional, Ciet, Pierluigi, additional, Tiddens, Harm A. W. M., additional, Vogel‐Claussen, Jens, additional, and Wild, Jim M., additional

Published
2022
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32. 129Xe and Free‐Breathing 1H Ventilation MRI in Patients With Cystic Fibrosis: A Dual‐Center Study.

Author

Marshall, Helen, Voskrebenzev, Andreas, Smith, Laurie J., Biancardi, Alberto M., Kern, Agilo L., Collier, Guilhem J., Wielopolski, Piotr A., Ciet, Pierluigi, Tiddens, Harm A. W. M., Vogel‐Claussen, Jens, and Wild, Jim M.

Subjects
CYSTIC fibrosis, VENTILATION, PULMONARY function tests, MAGNETIC resonance imaging, STATISTICAL correlation
Abstract

Background: Free‐breathing 1H ventilation MRI shows promise but only single‐center validation has yet been performed against methods which directly image lung ventilation in patients with cystic fibrosis (CF). Purpose: To investigate the relationship between 129Xe and 1H ventilation images using data acquired at two centers. Study type: Sequence comparison. Population: Center 1; 24 patients with CF (12 female) aged 9–47 years. Center 2; 7 patients with CF (6 female) aged 13–18 years, and 6 healthy controls (6 female) aged 21–31 years. Data were acquired in different patients at each center. Field Strength/Sequence: 1.5 T, 3D steady‐state free precession and 2D spoiled gradient echo. Assessment: Subjects were scanned with 129Xe ventilation and 1H free‐breathing MRI and performed pulmonary function tests. Ventilation defect percent (VDP) was calculated using linear binning and images were visually assessed by H.M., L.J.S., and G.J.C. (10, 5, and 8 years' experience). Statistical Tests: Correlations and linear regression analyses were performed between 129Xe VDP, 1H VDP, FEV1, and LCI. Bland–Altman analysis of 129Xe VDP and 1H VDP was carried out. Differences in metrics were assessed using one‐way ANOVA or Kruskal–Wallis tests. Results: 129Xe VDP and 1H VDP correlated strongly with; each other (r = 0.84), FEV1 z‐score (129Xe VDP r = −0.83, 1H VDP r = −0.80), and LCI (129Xe VDP r = 0.91, 1H VDP r = 0.82). Bland–Altman analysis of 129Xe VDP and 1H VDP from both centers had a bias of 0.07% and limits of agreement of −16.1% and 16.2%. Linear regression relationships of VDP with FEV1 were not significantly different between 129Xe and 1H VDP (P = 0.08), while 129Xe VDP had a stronger relationship with LCI than 1H VDP. Data Conclusion: 1H ventilation MRI shows large‐scale agreement with 129Xe ventilation MRI in CF patients with established lung disease but may be less sensitive to subtle ventilation changes in patients with early‐stage lung disease. Evidence Level: 2 Technical Efficacy: Stage 2 [ABSTRACT FROM AUTHOR]

Published
2023
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36. The Management of Asymptomatic Congenital Pulmonary Airway Malformation: Results of a European Delphi Survey

Author

Kersten, Casper M., primary, Hermelijn, Sergei M., additional, Mullassery, Dhanya, additional, Muthialu, Nagarajan, additional, Cobanoglu, Nazan, additional, Gartner, Silvia, additional, Bagolan, Pietro, additional, Mesas Burgos, Carmen, additional, Sgrò, Alberto, additional, Heyman, Stijn, additional, Till, Holger, additional, Suominen, Janne, additional, Schurink, Maarten, additional, Desender, Liesbeth, additional, Losty, Paul, additional, Steyaert, Henri, additional, Terheggen-Lagro, Suzanne, additional, Metzelder, Martin, additional, Bonnard, Arnaud, additional, Sfeir, Rony, additional, Singh, Michael, additional, Yardley, Iain, additional, Rikkers-Mutsaerts, Noor R. V. M., additional, van der Ent, Cornelis K., additional, Qvist, Niels, additional, Cox, Des W., additional, Peters, Robert, additional, Bannier, Michiel A. G. E., additional, Wessel, Lucas, additional, Proesmans, Marijke, additional, Stanton, Michael, additional, Hannon, Edward, additional, Zampoli, Marco, additional, Morini, Francesco, additional, Tiddens, Harm A. W. M., additional, Wijnen, René M. H., additional, and Schnater, Johannes M., additional

Published
2022
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37. The effect of inhaled hypertonic saline on lung structure in children aged 3–6 years with cystic fibrosis (SHIP-CT): a multicentre, randomised, double-blind, controlled trial

Author

Tiddens, Harm A W M, primary, Chen, Yuxin, additional, Andrinopoulou, Eleni-Rosalina, additional, Davis, Stephanie D, additional, Rosenfeld, Margaret, additional, Ratjen, Felix, additional, Kronmal, Richard A, additional, Hinckley Stukovsky, Karen D, additional, Dasiewicz, Alison, additional, Stick, Stephen Michael, additional, Anthony, Margaret M., additional, Au, Jacky, additional, Belessis, Yvonne, additional, Bonte, Merlijn, additional, Cheney, Joyce, additional, Clem, Charles, additional, Clements, Barry, additional, Cooper, Peter, additional, Davis, Stephanie D., additional, Davis, Miriam, additional, de Boeck, Kris, additional, de Marchis, Matteo, additional, De Wachter, Elke, additional, Delaisi, Bertrand, additional, Delaup, Véronique, additional, DeRicco, Adrienne, additional, Foti, Alexia, additional, Gan, Richard, additional, Garriga, Laura, additional, Gartner, Silvia, additional, Genatossio, Alan, additional, Grogan, Sam, additional, Hilton, Jodi, additional, Hoppe, Jordana E., additional, Janssens, Hettie M., additional, Jensen, Renee, additional, Johnson, Robin, additional, Kemner-van de Corput, Mariette P.C., additional, Klein, Brendan, additional, Kronmal, Richard A., additional, Lucca, Francesca, additional, Lucidi, Vincencina, additional, Montemitro, Enza, additional, Nahidi, Lily, additional, Nielsen, Kim G., additional, Pearce, Kasey, additional, Pittman, Jessica E, additional, Powers, Michael, additional, Prentice, Carley, additional, Pressler, Tania, additional, Rayment, Jonathan H, additional, Reix, Philippe, additional, Retsch-Bogart, George, additional, Riera, Luis, additional, Robinson, Phil, additional, Robinson, Paul, additional, Sanders, Don B., additional, Sandoval, Rodrigo A., additional, Sandvik, Rikke Mulvad, additional, Saunders, Clare, additional, Siegel, Molly, additional, Smith, Julie, additional, Solomon, Melinda, additional, Stanojevic, Sanja, additional, Tai, Andrew, additional, Tiddens, Harm A.W.M., additional, van de Puttelaar, Jorien, additional, Van den Brande, Christel, additional, van Straten, Marcel, additional, Vermeulen, Francois, additional, Volpi, Sonia, additional, Wainwright, Claire E., additional, Weiner, Daniel J., additional, Yuan, Yi, additional, and Zaimeddine, Sarah, additional

Published
2022
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38. Lung structure and function on MRI in preterm born school children with and without BPD:A feasibility study

Author

Elders, Bernadette B. L. J., Tiddens, Harm A. W. M., Pijnenburg, Marielle W. H., Reiss, Irwin K. M., Wielopolski, Piotr A., Ciet, Pierluigi, Elders, Bernadette B. L. J., Tiddens, Harm A. W. M., Pijnenburg, Marielle W. H., Reiss, Irwin K. M., Wielopolski, Piotr A., and Ciet, Pierluigi

Abstract

Background and Objective The most common respiratory complication of prematurity is bronchopulmonary dysplasia (BPD), leading to structural lung changes and impaired respiratory outcomes. However, also preterm children without BPD may show similar adverse respiratory outcomes. There is a need for a safe imaging modality for preterm children with and without BPD for disease severity assessment and risk stratification. Our objective was to develop a magnetic resonance imaging (MRI) protocol in preterm children with and without BPD at school age. Methods Nine healthy volunteers (median age 11.6 [range: 8.8–12.8] years), 11 preterm children with BPD (11.0 [7.2–15.6] years), and 9 without BPD (11.1 [10.7–12.6] years) underwent MRI. Images were scored on hypo- and hyperintense abnormalities, bronchopathy, and architectural distortion. MRI data were correlated to spirometry. Ventilation and perfusion defects were analyzed using Fourier Decomposition (FD) MRI. Results On MRI, children with BPD had higher %diseased lung (9.1 (interquartile range [IQR] 5.9–11.6)%) compared to preterm children without BPD (3.4 (IQR 2.5–5.4)%, p < 0.001) and healthy volunteers (0.4 (IQR 0.1–0.8)%, p < 0.001). %Diseased lung correlated negatively with %predicted FEV1 (r = −0.40, p = 0.04), FEV1/FVC (r = −0.49, p = 0.009) and FEF75 (r = −0.63, p < 0.001). Ventilation and perfusion defects on FD sequence corresponded to hypointense regions on expiratory MRI. Conclusion Chest MRI can identify structural and functional lung damage at school age in preterm children with and without BPD, showing a good correlation with spirometry. We propose MRI as a sensitive and safe imaging method (without ionizing radiation, contrast agents, or the use of anesthesia) for the long-term follow-up of preterm children.

Published
2022

39. Diaphragmatic dysfunction in neuromuscular disease, an MRI study

Author

Harlaar, Laurike, Ciet, Pierluigi, van Tulder, Gijs, Brusse, Esther, Timmermans, Remco G M, Janssen, Wim G M, de Bruijne, Marleen, van der Ploeg, Ans T, Tiddens, Harm A W M, van Doorn, Pieter A, van der Beek, Nadine A M E, Harlaar, Laurike, Ciet, Pierluigi, van Tulder, Gijs, Brusse, Esther, Timmermans, Remco G M, Janssen, Wim G M, de Bruijne, Marleen, van der Ploeg, Ans T, Tiddens, Harm A W M, van Doorn, Pieter A, and van der Beek, Nadine A M E

Abstract

The aim of this exploratory study was to evaluate diaphragmatic function across various neuromuscular diseases using spirometry-controlled MRI. We measured motion of the diaphragm relative to that of the thoracic wall (cranial-caudal ratio vs. anterior posterior ratio; CC-AP ratio), and changes in the diaphragmatic curvature (diaphragm height and area ratio) during inspiration in 12 adults with a neuromuscular disease having signs of respiratory muscle weakness, 18 healthy controls, and 35 adult Pompe patients - a group with prominent diaphragmatic weakness. CC-AP ratio was lower in patients with myopathies (n=7, 1.25±0.30) and motor neuron diseases (n=5, 1.30±0.10) than in healthy controls (1.37±0.14; p=0.001 and p=0.008), but not as abnormal as in Pompe patients (1.12±0.18; p=0.011 and p=0.024). The mean diaphragm height ratio was 1.17±0.33 in patients with myopathies, pointing at an insufficient diaphragmatic contraction. This was also seen in patients with Pompe disease (1.28±0.36), but not in healthy controls (0.82±0.33) or patients with motor neuron disease (0.82±0.24). We conclude that spirometry-controlled MRI enables us to investigate respiratory dysfunction across neuromuscular diseases, suggesting that the diaphragm is affected in a different way in myopathies and motor neuron diseases. Whether MRI can also be used to evaluate progression of diaphragmatic dysfunction requires additional studies.

Published
2022

40. Small airways targeted treatment with smart nebulizer technology could improve severe asthma in children: a retrospective analysis

Author

van den Bosch, Wytse B., Kloosterman, Sanne F., Andrinopoulou, Eleni-Rosalina, Greidanus, Rients, Pijnenburg, Marielle W. H., Tiddens, Harm A. W. M., Janssens, Hettie M., van den Bosch, Wytse B., Kloosterman, Sanne F., Andrinopoulou, Eleni-Rosalina, Greidanus, Rients, Pijnenburg, Marielle W. H., Tiddens, Harm A. W. M., and Janssens, Hettie M.

Abstract

Objective: Conventional inhaler devices have a low efficacy in targeting small airways. Smart nebulizers can be used to increase deposition to small airways by adjusting the flow and depth of each inhalation based on patients ‘individual inspiratory capacity. We investigated whether targeting of high dose inhaled corticosteroids (ICS) to small airways with a smart nebulizer could reduce exacerbation rate in children with severe asthma (SA). Methods: We conducted a retrospective study in children with SA using a smart nebulizer (Akita® Jet nebulizer) for the administration of high dose ICS in our outpatient clinic at the Erasmus MC — Sophia Children’s Hospital. Clinical data before and after start of treatment were collected. The primary outcome was exacerbation rate, defined as: number of asthma exacerbations for which oral corticosteroid courses (OCS) were prescribed. The exacerbation rate 1 year before treatment was compared with the exacerbation rate 1 year after start of treatment. Secondary outcomes were changes in spirometry parameters, hospital admissions and medication use. Results: Data on OCS use was available for 28/31 patients. Median number of asthma exacerbations requiring OCS courses 1 year before decreased from 2 (interquartile range(IQR) 2) to 0.5 (IQR 3) 1 year after treatment (p = 0.021). Hospital admission decreased from 1 (IQR 3) to 0 (IQR 1)(p = 0.028). FEV1, FEF25-75 and FEF75 were not significantly improved after one year of treatment with the smart nebulizer (p = 0.191; p = 0.248; p = 0.572). Conclusion: Targeting small airways with high dose ICS using a smart nebulizer resulted in a significant reduction in exacerbations requiring OCS after one year of treatment.

Published
2022

41. A dual center and dual vendor comparison study of automated perfusion‐weighted phase‐resolved functional lung magnetic resonance imaging with dynamic contrast‐enhanced magnetic resonance imaging in patients with cystic fibrosis

Author

Behrendt, Lea, primary, Smith, Laurie J., additional, Voskrebenzev, Andreas, additional, Klimeš, Filip, additional, Kaireit, Till F., additional, Pöhler, Gesa H., additional, Kern, Agilo L., additional, Gonzalez, Cristian Crisosto, additional, Dittrich, Anna‐Maria, additional, Marshall, Helen, additional, Schütz, Katharina, additional, Hughes, Paul J. C., additional, Ciet, Pierluigi, additional, Tiddens, Harm A. W. M., additional, Wild, Jim M., additional, and Vogel‐Claussen, Jens, additional

Published
2022
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