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1. Amikacin liposome inhalation suspension for chronic Pseudomonas aeruginosa infection in cystic fibrosis

Author

Renner, Sabine, Knoop, Christiane, Malfroot, Anne, Dupont, Lieven, Desager, Kristine, De Baets, Frans, Bosheva, Miroslava, Nedkova, Vania, Galabov, Ivan, Galeva, Ivanka, Freitag, Andreas, Morrison, Nancy, Wilcox, Pearce, Pressler, Tanja, Martinet, Yves, Chiron, Raphael, Fajac, Isabelle, Dominique, Stephan, Reix, Philippe, Prevotat, Anne, Sermet, Isabelle, Durieu, Isabelle, Fischer, Rainald, Huber, Rudolf, Staab, Doris, Mellies, Uwe, Sextro, Wolfgang, Welte, Tobias, Wilkens, Heinrike, Sommerwerk, Urte, Bewig, Burkhard, Inglezos, Ilias, Doudounakis, Stavros-Eleftherios, Bede, Olga, Gönczi, Ferenc, Újhelyi, Rita, McKone, Edward, McNally, Paul, Lucidi, Vincenzina, Cipolli, Marco, La Rosa, Mario, Minicucci, Laura, Padoan, Rita, Pisi, Giovanna, Gagliardini, Rolando, Colombo, Carla, Bronsveld, Inez, Sapiejka, Ewa, Mazurek, Henryk, Sands, Dorota, Górnicka, Grażyna, Stelmach, Iwona, Batura-Gabryel, Halina, Rachel, Marta, Minic, Predrag, Orosova, Jaroslava, Takac, Branko, Feketova, Anna, Martinez, Carmen, Hernandez, Gloria Garcia, Villa-Asensi, Jose Ramon, Gartner, Silvia, Sole, Amparo, Lindblad, Anders, Ledson, Martin, Bilton, Diana, Whitehouse, Joanna, Smyth, Alan, Ketchell, Ian, Lee, Timothy, MacGregor, Gordon, Pressler, Tacjana, Clancy, John Paul, Solé, Amparo, Quittner, Alexandra L., Liu, Keith, McGinnis, John P., II, Eagle, Gina, Gupta, Renu, and Konstan, Michael W.

Published
2020
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6. Allogeneic Hematopoietic Stem Cell Transplantation After Prior Lung Transplantation for Hereditary Pulmonary Alveolar Proteinosis: A Case Report

Author

Beeckmans, Hanne, Ambrocio, Gene PL, Bos, Saskia, Vermaut, Astrid, Geudens, Vincent, Vanstapel, Arno, Vanaudenaerde, Bart M, De Baets, Frans, Malfait, Thomas LA, Emonds, Marie-Paule, Van Raemdonck, Dirk E, Schoemans, Helene M, Vos, Robin, and Leuven, Lung Transplant Grp

Subjects
bone marrow transplantation, pulmonary alveolar proteinosis, Pulmonary Fibrosis, Immunology, Hematopoietic Stem Cell Transplantation, Pulmonary Surfactants, Surface-Active Agents, Receptors, Granulocyte-Macrophage Colony-Stimulating Factor, lung transplantation, Quality of Life, Humans, Immunology and Allergy, allogeneic hematopoietic stem cell transplantation, rejection
Abstract

Pulmonary alveolar proteinosis (PAP) is a rare, diffuse lung disorder characterized by surfactant accumulation in the small airways due to defective clearance by alveolar macrophages, resulting in impaired gas exchange. Whole lung lavage is the current standard of care treatment for PAP. Lung transplantation is an accepted treatment option when whole lung lavage or other experimental treatment options are ineffective, or in case of extensive pulmonary fibrosis secondary to PAP. A disadvantage of lung transplantation is recurrence of PAP in the transplanted lungs, especially in hereditary PAP. The hereditary form of PAP is an ultra-rare condition caused by genetic mutations in genes encoding for the granulocyte macrophage-colony stimulating factor (GM-CSF) receptor, and intrinsically affects bone marrow derived-monocytes, which differentiate into macrophages in the lung. Consequently, these macrophages typically display disrupted GM-CSF receptor-signaling, causing defective surfactant clearance. Bone marrow/hematopoietic stem cell transplantation may potentially reverse the lung disease in hereditary PAP. In patients with hereditary PAP undergoing lung transplantation, post-lung transplant recurrence of PAP may theoretically be averted by subsequent hematopoietic stem cell transplantation, which results in a graft-versus-disease (PAP) effect, and thus could improve long-term outcome. We describe the successful long-term post-transplant outcome of a unique case of end-stage respiratory failure due to hereditary PAP-induced pulmonary fibrosis, successfully treated by bilateral lung transplantation and subsequent allogeneic hematopoietic stem cell transplantation. Our report supports treatment with serial lung and hematopoietic stem cell transplantation to improve quality of life and prolong survival, without PAP recurrence, in selected patients with end-stage hereditary PAP. ispartof: FRONTIERS IN IMMUNOLOGY vol:13 ispartof: location:Switzerland status: published

Published
2022

19. Amikacin liposome inhalation suspension for chronic Pseudomonas aeruginosa infection in cystic fibrosis

Author

Bilton, Diana, primary, Pressler, Tacjana, additional, Fajac, Isabelle, additional, Clancy, John Paul, additional, Sands, Dorota, additional, Minic, Predrag, additional, Cipolli, Marco, additional, Galeva, Ivanka, additional, Solé, Amparo, additional, Quittner, Alexandra L., additional, Liu, Keith, additional, McGinnis, John P., additional, Eagle, Gina, additional, Gupta, Renu, additional, Konstan, Michael W., additional, Renner, Sabine, additional, Knoop, Christiane, additional, Malfroot, Anne, additional, Dupont, Lieven, additional, Desager, Kristine, additional, De Baets, Frans, additional, Bosheva, Miroslava, additional, Nedkova, Vania, additional, Galabov, Ivan, additional, Freitag, Andreas, additional, Morrison, Nancy, additional, Wilcox, Pearce, additional, Pressler, Tanja, additional, Martinet, Yves, additional, Chiron, Raphael, additional, Dominique, Stephan, additional, Reix, Philippe, additional, Prevotat, Anne, additional, Sermet, Isabelle, additional, Durieu, Isabelle, additional, Fischer, Rainald, additional, Huber, Rudolf, additional, Staab, Doris, additional, Mellies, Uwe, additional, Sextro, Wolfgang, additional, Welte, Tobias, additional, Wilkens, Heinrike, additional, Sommerwerk, Urte, additional, Bewig, Burkhard, additional, Inglezos, Ilias, additional, Doudounakis, Stavros-Eleftherios, additional, Bede, Olga, additional, Gönczi, Ferenc, additional, Újhelyi, Rita, additional, McKone, Edward, additional, McNally, Paul, additional, Lucidi, Vincenzina, additional, La Rosa, Mario, additional, Minicucci, Laura, additional, Padoan, Rita, additional, Pisi, Giovanna, additional, Gagliardini, Rolando, additional, Colombo, Carla, additional, Bronsveld, Inez, additional, Sapiejka, Ewa, additional, Mazurek, Henryk, additional, Górnicka, Grażyna, additional, Stelmach, Iwona, additional, Batura-Gabryel, Halina, additional, Rachel, Marta, additional, Orosova, Jaroslava, additional, Takac, Branko, additional, Feketova, Anna, additional, Martinez, Carmen, additional, Hernandez, Gloria Garcia, additional, Villa-Asensi, Jose Ramon, additional, Gartner, Silvia, additional, Sole, Amparo, additional, Lindblad, Anders, additional, Ledson, Martin, additional, Bilton, Diana, additional, Whitehouse, Joanna, additional, Smyth, Alan, additional, Ketchell, Ian, additional, Lee, Timothy, additional, and MacGregor, Gordon, additional

Published
2020
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21. Revisiting human IL-12R(beta)1 deficiency: a survey of 141 patients from 30 countries

Author

de Beaucoudrey, Ludovic, Samarina, Arina, Bustamante, Jacinta, Cobat, Aurelie, Boisson-Dupuis, Stephanie, Feinberg, Jacqueline, Al-Muhsen, Saleh, Janniere, Lucile, Rose, Yoann, de Suremain, Maylis, Kong, Xiao-Fei, Filipe-Santos, Orchidee, Chapgier, Ariane, Picard, Capucine, Fischer, Alain, Dogu, Figen, Ikinciogullari, Aydan, Tanir, Gonul, Al-Hajjar, Sami, Al-Jumaah, Suliman, Frayha, Husn H., AlSum, Zobaida, Al-Ajaji, Sulaiman, Alangari, Abdullah, Al-Ghonaium, Abdulaziz, Adimi, Parisa, Mansouri, Davood, Ben-Mustapha, Imen, Yancoski, Judith, Garty, Ben-Zion, Rodriguez-Gallego, Carlos, Caragol, Isabel, Kutukculer, Necil, Kumararatne, Dinakantha S., Patel, Smita, Doffinger, Rainer, Exley, Andrew, Jeppsson, Olle, Reichenbach, Janine, Nadal, David, Boyko, Yaryna, Pietrucha, Barbara, Anderson, Suzanne, Levin, Michael, Schandene, Liliane, Schepers, Kinda, Efira, Andre, Mascart, Francoise, Matsuoka, Masao, Sakai, Tatsunori, Siegrist, Claire-Anne, Frecerova, Klara, Bluetters-Sawatzki, Renate, Bernhoft, Jutta, Freihorst, Joachim, Baumann, Ulrich, Richter, Darko, Haerynck, Filomeen, De Baets, Frans, Novelli, Vas, Lammas, David, Vermylen, Christiane, Tuerlinckx, David, Nieuwhof, Chris, Pac, Malgorzata, Haas, Walther H., Muller-Fleckenstein, Ingrid, Fleckenstein, Bernhard, Levy, Jacob, Raj, Revathi, Cohen, Aileen Cleary, Lewis, David B., Holland, Steven M., Yang, Kuender D., Wang, Xiaochuan, Wang, Xiaohong, Jiang, Liping, Yang, Xiqiang, Zhu, Chaomin, Xie, Yuanyuan, Lee, Pamela Pui Wah, Chan, Koon Wing, Chen, Tong-Xin, Castro, Gabriela, Natera, Ivelisse, Codoceo, Ana, King, Alejandra, Bezrodnik, Liliana, Di Giovani, Daniela, Gaillard, Maria Isabel, de Moraes-Vasconcelos, Dewton, Grumach, Anete Sevciovic, da Silva Duarte, Alberto Jose, Aldana, Ruth, Espinosa-Rosales, Francisco Javier, Bejaoui, Mohammed, Bousfiha, Ahmed Aziz, El Baghdadi, Jamila, Ozbek, Namik, Aksu, Guzide, Keser, Melike, Somer, Ayper, Hatipoglu, Nevin, Aydogmus, Cigdem, Asilsoy, Suna, Camcioglu, Yildiz, Gulle, Saniye, Ozgur, Tuba T., Ozen, Meteran, Oleastro, Matias, Bernasconi, Andrea, Mamishi, Setareh, Parvaneh, Nima, Rosenzweig, Sergio, Barbouche, Ridha, Pedraza, Sigifredo, Lau, Yu Lung, Ehlayel, Mohammad S., Fieschi, Claire, Abel, Laurent, Sanal, Ozden, and Casanova, Jean-Laurent

Subjects
Interleukin-12 -- Physiological aspects, Interleukin-12 -- Abnormalities, Mycobacterial infections -- Risk factors, Mycobacterial infections -- Distribution, Mycobacterial infections -- Surveys, Disease susceptibility -- Surveys, Mendel's law -- Analysis, Company distribution practices
Published
2010

31. Pulmonary function in children after lobectomy

Author

Schaballie, Heidi, primary, Rigolle, Hanne, additional, Schelstraete, Petra, additional, Van Daele, Sabine, additional, Willekens, Julie, additional, Proesmans, Marijke, additional, Malfroot, Anne, additional, Smets, Dirk, additional, and De Baets, Frans, additional

Published
2019
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33. Comparison of culture and qPCR for the detection of Pseudomonas aeruginosa in not chronically infected cystic fibrosis patients

Author

Boboli Hedwige, Casimir Georges, Knoop Christiane, Lebecque Patrick, Malfroot Anne, De Wachter Elke, Van daele Sabine, Haerynck Filomeen, Van Simaey Leen, Lopes dos Santos Santiago Guido, Schelstraete Petra, Deschaght Pieter, Pierart Frédéric, Desager Kristine, Vaneechoutte Mario, and De Baets Frans

Subjects
Microbiology, QR1-502
Abstract

Abstract Background Pseudomonas aeruginosa is the major respiratory pathogen causing severe lung infections among CF patients, leading to high morbidity and mortality. Once infection is established, early antibiotic treatment is able to postpone the transition to chronic lung infection. In order to optimize the early detection, we compared the sensitivity of microbiological culture and quantitative PCR (qPCR) for the detection of P. aeruginosa in respiratory samples of not chronically infected CF patients. Results In this national study, we followed CF patients during periods between 1 to 15 months. For a total of 852 samples, 729 (86%) remained P. aeruginosa negative by both culture and qPCR, whereas 89 samples (10%) were positive by both culture and qPCR. Twenty-six samples were negative by culture but positive by qPCR, and 10 samples were positive by culture but remained negative by qPCR. Five of the 26 patients with a culture negative, qPCR positive sample became later P. aeruginosa positive both by culture and qPCR. Conclusion Based on the results of this study, it can be concluded that qPCR may have a predictive value for impending P. aeruginosa infection for only a limited number of patients.

Published
2010
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34. Comparison of the sensitivity of culture, PCR and quantitative real-time PCR for the detection of Pseudomonas aeruginosa in sputum of cystic fibrosis patients

Author

De Vos Daniel, De Baets Frans, Van daele Sabine, Van Simaey Leen, De Baere Thierry, Deschaght Pieter, Pirnay Jean-Paul, and Vaneechoutte Mario

Subjects
Microbiology, QR1-502
Abstract

Abstract Background Pseudomonas aeruginosa is the major pathogen involved in the decline of lung function in cystic fibrosis (CF) patients. Early aggressive antibiotic therapy has been shown to be effective in preventing chronic colonization. Therefore, early detection is important and sensitive detection methods are warranted. In this study, we used a dilution series of P. aeruginosa positive sputa, diluted in a pool of P. aeruginosa negative sputa, all from CF patients - to mimick as closely as possible the sputa sent to routine laboratories - to compare the sensitivity of three culture techniques versus that of two conventional PCR formats and four real-time PCR formats, each targeting the P. aeruginosa oprL gene. In addition, we compared five DNA-extraction protocols. Results In our hands, all three culture methods and the bioMérieux easyMAG Nuclisens protocol Generic 2.0.1, preceded by proteinase K pretreatment and followed by any of the 3 real-time PCR formats with probes were most sensitive and able to detect P. aeruginosa up to 50 cfu/ml, i.e. the theoretical minimum of one cell per PCR mixture, when taking into account the volumes used in this study of sample for DNA-extraction, of DNA-elution and of DNA-extract in the PCR mixture. Conclusion In this study, no difference in sensitivity could be found for the detection of P. aeruginosa from sputum between microbiological culture and optimized DNA-extraction and real-time PCR. The results also indicate the importance of the optimization of the DNA-extraction protocol and the PCR format.

Published
2009
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40. Development and validation of an LC tandem MS assay for the quantification of β-lactam antibiotics in the sputum of cystic fibrosis patients

Author

Forier, Katrien, primary, Van Heck, Virginie, additional, Carlier, Mieke, additional, Van Braeckel, Eva, additional, Van Daele, Sabine, additional, De Baets, Frans, additional, Schelstraete, Petra, additional, Haerynck, Filomeen, additional, Stove, Veronique, additional, Van Simaey, Leen, additional, Vaneechoutte, Mario, additional, and Verstraete, Alain G, additional

Published
2017
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43. Early-onset primary antibody deficiency resembling common variable immunodeficiency challenges the diagnosis of Wiedeman-Steiner and Roifman syndromes

Author

Bogaert, Delfien J., primary, Dullaers, Melissa, additional, Kuehn, Hye Sun, additional, Leroy, Bart P., additional, Niemela, Julie E., additional, De Wilde, Hans, additional, De Schryver, Sarah, additional, De Bruyne, Marieke, additional, Coppieters, Frauke, additional, Lambrecht, Bart N., additional, De Baets, Frans, additional, Rosenzweig, Sergio D., additional, De Baere, Elfride, additional, and Haerynck, Filomeen, additional

Published
2017
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45. Epidemic Achromobacter xylosoxidans strain among Belgian cystic fibrosis patients and review of literature

Author

Cools, Piet, primary, Ho, Erwin, additional, Vranckx, Katleen, additional, Schelstraete, Petra, additional, Wurth, Bettina, additional, Franckx, Hilde, additional, Ieven, Greet, additional, Van Simaey, Leen, additional, Van daele, Sabine, additional, Verhulst, Stijn, additional, De Baets, Frans, additional, and Vaneechoutte, Mario, additional

Published
2016
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46. When One Rare Disease Hides Another: Kartagener Syndrome Masking FMF.

Author

Hoste, Levi, De Baets, Frans, Van Daele, Sabine, Schelstraete, Petra, Boon, Mieke, De Bruyne, Marieke, Dullaers, Melissa, Coppieters, Frauke, and Haerynck, Filomeen

Subjects
*GENETIC disorder diagnosis, *INFLAMMATION, *CELL physiology, *CONSANGUINITY, *DIFFERENTIAL diagnosis, *GENEALOGY, *GENETIC disorders, *GENETIC techniques, *PEDIATRICS, *CILIARY motility disorders, *DIAGNOSIS
Abstract

The article presents a case study of a 16-year-old boy with recurrent episodes of fever, without any respiratory problems but with concomitant abdominal and sometimes thoracic pain and arthralgia. It mentions that genetic evaluation was performed to identify the molecular cause of Primary ciliary dyskinesia (PCD) as well as Familial Mediterranean fever (FMF). It informs that the patient was treated with colchicine.

Published
2018
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47. Development and validation of an LC tandem MS assay for the quantification of β-lactam antibiotics in the sputum of cystic fibrosis patients.

Author

Forier, Katrien, Van Heck, Virginie, Carlier, Mieke, Van Braeckel, Eva, Van Daele, Sabine, De Baets, Frans, Schelstraete, Petra, Haerynck, Filomeen, Stove, Veronique, Van Simaey, Leen, Vaneechoutte, Mario, and Verstraete, Alain G.

Subjects
CYSTIC fibrosis, BETA lactam antibiotics, SPUTUM examination, LIQUID chromatography-mass spectrometry, PIPERACILLIN, AZTREONAM, PATIENTS, THERAPEUTICS, ANTIBIOTICS assay, COMPARATIVE studies, HIGH performance liquid chromatography, MASS spectrometry, RESEARCH methodology, MEDICAL cooperation, RESEARCH, SPUTUM, EVALUATION research, CARBAPENEMS, AMPICILLIN, CEFTAZIDIME
Abstract

Objectives: Antibiotic therapy is of vital importance for the control of infectious exacerbations in cystic fibrosis (CF) patients. However, very little is known regarding the fraction of systemically administered antibiotics reaching the lower respiratory tract secretions. We developed and validated a method to measure the concentrations of piperacillin, ceftazidime, meropenem and aztreonam in CF sputum, and present the validation data.Methods: Ultra-performance LC coupled to tandem MS was used. A single sample can be measured in 2.5 min with multiple reaction monitoring in positive electrospray ionization mode. Deuterated internal standards were used and a concentration range of 0.7-160 mg/L was covered. The method was validated according to the EMA guideline on analytical method validation.Results: The boundaries within which a reliable measurement in CF sputum can be performed were determined. A few constraints are linked to the instability of the antibiotics in sputum. Piperacillin showed limited stability at room temperature and during freeze-thaw cycles. Autosampler instability was observed after 15 h for aztreonam at low concentrations.Conclusions: The method allows a reliable measurement of the selected antibiotics, if precautions are taken regarding the limited stability of piperacillin at room temperature. Due to freeze-thaw instability, piperacillin should always be analysed on the day of sampling. Quick review of the analytical data and reanalysis are needed as low concentrations of aztreonam are not stable in the autosampler. [ABSTRACT FROM AUTHOR]

Published
2018
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50. Early referral to a specialised centre is associated with a better respiratory outcome

Author

Lebecque, Patrick, Casimir, Georges, De Baets, Frans, De Boeck, Christiane, Desager, Kristine, Godding, Véronique, Malfroot, Anne, Reychler, Gregory, Leal, Teresinha, 29th European Cystic Fibrosis Conference, UCL - MD/BICL - Département de biochimie et de biologie cellulaire, UCL - MD/MINT - Département de médecine interne, UCL - MD/GYPE - Département de gynécologie, d'obstétrique et de pédiatrie, UCL - MD/IEPR - Institut d'éducation physique et de réadaptation, UCL - (SLuc) Service de pneumologie, UCL - (SLuc) Service de biochimie médicale, and UCL - (SLuc) Service de pédiatrie générale

Subjects
and Child Health, Pulmonary and Respiratory Medicine, Pediatrics, Perinatology
Published
2006

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