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Article ; Online: Broncholithiasis and Primary Ciliary Dyskinesia: An Association Not Identified in Other Chronic Airway Inflammatory Disorders.

Kennedy, Marcus P / Noone, Peadar G / Knowles, Michael R

2020 Volume 157, Issue 3, Page(s) 737

MeSH term(s)	Bronchial Diseases ; Humans ; Kartagener Syndrome ; Pulmonary Disease, Chronic Obstructive
Language	English
Publishing date	2020-02-29
Publishing country	United States
Document type	Letter ; Comment
ZDB-ID	1032552-9
ISSN	1931-3543 ; 0012-3692
ISSN (online)	1931-3543
ISSN	0012-3692
DOI	10.1016/j.chest.2019.10.055
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Article ; Online: Respiratory infections in patients with cystic fibrosis undergoing lung transplantation.

Lobo, Leonard J / Noone, Peadar G

The Lancet. Respiratory medicine

2014 Volume 2, Issue 1, Page(s) 73–82

Abstract: Cystic fibrosis is an inherited disease characterised by chronic respiratory infections associated with bronchiectasis. Lung transplantation has helped to extend the lives of patients with cystic fibrosis who have advanced lung disease. However, ... ...

Abstract	Cystic fibrosis is an inherited disease characterised by chronic respiratory infections associated with bronchiectasis. Lung transplantation has helped to extend the lives of patients with cystic fibrosis who have advanced lung disease. However, persistent, recurrent, and newly acquired infections can be problematic. Classic cystic fibrosis-associated organisms, such as Staphylococcus aureus and Pseudomonas aeruginosa, are generally manageable post-transplantation, and are associated with favourable outcomes. Burkholderia cenocepacia poses particular challenges, although other Burkholderia species are less problematic. Despite concerns about non-tuberculous mycobacteria, especially Mycobacterium abscessus, post-transplantation survival has not been definitively shown to be less than average in patients with these infections. Fungal species can be prevalent before and after transplantation and are associated with high morbidity, so should be treated aggressively. Appropriate viral screening and antiviral prophylaxis are necessary to prevent infection with and reactivation of Epstein-Barr virus and cytomegalovirus and their associated complications. Awareness of drug pharmacokinetics and interactions in cystic fibrosis is crucial to prevent toxic effects and subtherapeutic or supratherapeutic drug dosing. With the large range of potential infectious organisms in patients with cystic fibrosis, infection control in hospital and outpatient settings is important. Despite its complexity, lung transplantation in the cystic fibrosis population is safe, with good outcomes if the clinician is aware of all the potential pathogens and remains vigilant by means of surveillance and proactive treatment.
MeSH term(s)	Cystic Fibrosis/microbiology ; Cystic Fibrosis/surgery ; Gram-Negative Bacterial Infections/prevention & control ; Humans ; Lung/microbiology ; Lung Transplantation ; Microbiota/physiology ; Mycobacterium Infections, Nontuberculous/prevention & control ; Mycoses/prevention & control ; Postoperative Complications/prevention & control ; Respiratory Tract Infections/prevention & control ; Staphylococcal Infections/prevention & control ; Virus Diseases/prevention & control
Language	English
Publishing date	2014-01
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	2686754-0
ISSN	2213-2619 ; 2213-2600
ISSN (online)	2213-2619
ISSN	2213-2600
DOI	10.1016/S2213-2600(13)70162-0
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Article ; Online: Non-invasive ventilation for the treatment of hypercapnic respiratory failure in cystic fibrosis.

Noone, Peadar G

Thorax

2008 Volume 63, Issue 1, Page(s) 5–7

MeSH term(s)	Cystic Fibrosis/complications ; Humans ; Hypercapnia/therapy ; Respiration, Artificial/methods ; Respiratory Insufficiency/therapy
Language	English
Publishing date	2008-01
Publishing country	England
Document type	Comment ; Editorial
ZDB-ID	204353-1
ISSN	1468-3296 ; 0040-6376
ISSN (online)	1468-3296
ISSN	0040-6376
DOI	10.1136/thx.2007.086710
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Article ; Online: Lung transplant and cystic fibrosis: what's new from the UK and France?

Noone, Peadar G

Thorax

2008 Volume 63, Issue 8, Page(s) 668–670

MeSH term(s)	Adolescent ; Burkholderia Infections/complications ; Burkholderia Infections/mortality ; Burkholderia cepacia complex ; Child ; Child, Preschool ; Cystic Fibrosis/mortality ; Cystic Fibrosis/surgery ; France ; Humans ; Infant ; Lung Transplantation/mortality ; Lung Transplantation/trends ; Postoperative Complications/microbiology ; Postoperative Complications/mortality ; United Kingdom
Language	English
Publishing date	2008-08
Publishing country	England
Document type	Editorial ; Comment
ZDB-ID	204353-1
ISSN	1468-3296 ; 0040-6376
ISSN (online)	1468-3296
ISSN	0040-6376
DOI	10.1136/thx.2008.099622
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Article ; Online: Primary ciliary dyskinesia.

Lobo, Jason / Zariwala, Maimoona A / Noone, Peadar G

Seminars in respiratory and critical care medicine

2015 Volume 36, Issue 2, Page(s) 169–179

Abstract: Primary ciliary dyskinesia (PCD) is an autosomal recessive disorder of cilia structure, function, and biogenesis leading to chronic infections of the respiratory tract, fertility problems, and disorders of organ laterality. The diagnosis can be ... ...

Abstract	Primary ciliary dyskinesia (PCD) is an autosomal recessive disorder of cilia structure, function, and biogenesis leading to chronic infections of the respiratory tract, fertility problems, and disorders of organ laterality. The diagnosis can be challenging, using traditional tools such as characteristic clinical features, ciliary function, and ultrastructural defects and newer screening tools such as nasal nitric oxide levels and genetic testing add to the diagnostic algorithm. There are 32 known PCD-causing genes, and in the future, comprehensive genetic testing may screen young infants before developing symptoms, thus improving survival. Therapies include surveillance of pulmonary function and microbiology, in addition to airway clearance, antibiotics, and early referral to bronchiectasis centers. As with cystic fibrosis (CF), standardized care at specialized centers using a multidisciplinary approach likely improves outcomes. In conjunction with the CF foundation, the PCD foundation, with experienced investigators and clinicians, is developing a network of PCD clinical centers to coordinate the effort in North America and Europe. As the network grows, clinical care and knowledge will improve.
MeSH term(s)	Anti-Bacterial Agents/therapeutic use ; Anti-Inflammatory Agents/therapeutic use ; Cilia/ultrastructure ; Genetic Testing ; Humans ; Kartagener Syndrome/diagnosis ; Kartagener Syndrome/genetics ; Kartagener Syndrome/therapy ; Microscopy, Electron, Transmission ; Mutation ; Nitric Oxide/analysis ; Phenotype ; Prognosis ; Respiratory Function Tests
Chemical Substances	Anti-Bacterial Agents ; Anti-Inflammatory Agents ; Nitric Oxide (31C4KY9ESH)
Language	English
Publishing date	2015-04
Publishing country	United States
Document type	Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't ; Review
ZDB-ID	1183617-9
ISSN	1098-9048 ; 1069-3424
ISSN (online)	1098-9048
ISSN	1069-3424
DOI	10.1055/s-0035-1546748
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Article: Genetics, diagnosis, and future treatment strategies for primary ciliary dyskinesia.

Daniels, M Leigh Anne / Noone, Peadar G

Expert opinion on orphan drugs

2014 Volume 3, Issue 1, Page(s) 31–44

Abstract: Introduction: Primary ciliary dyskinesia (PCD) is a genetically heterogeneous recessive disorder resulting in chronic oto-sino-pulmonary disease. While PCD is estimated to occur in 1 in 20,000 individuals, fewer than 1,000 patients in the US have a well- ...

Abstract	Introduction: Primary ciliary dyskinesia (PCD) is a genetically heterogeneous recessive disorder resulting in chronic oto-sino-pulmonary disease. While PCD is estimated to occur in 1 in 20,000 individuals, fewer than 1,000 patients in the US have a well-established diagnosis. Areas covered: We provide an overview of the clinical manifestations of PCD, describe the evolution of diagnostic methods, and critique the literature on management of PCD. Expert opinion: Although interest in clinical studies in non-CF bronchiectasis has increased in recent years, some of whom enroll patients with PCD, the literature regarding therapy for PCD as a distinct entity is lacking, as the numbers are small, and there have been no sub-analyses published. However, with improved screening and diagnostic methods, the development of clinical and research consortiums, and actively enrolling registries of PCD patients, the environment is conducive to perform longitudinal studies of disease course and therapeutic studies to alter that course.
Language	English
Publishing date	2014-11-29
Publishing country	England
Document type	Journal Article
ISSN	2167-8707
ISSN	2167-8707
DOI	10.1517/21678707.2015.989212
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Article ; Online: Sodium channel blockers for cystic fibrosis.

Burrows, Elinor F / Southern, Kevin W / Noone, Peadar G

The Cochrane database of systematic reviews

2014 , Issue 4, Page(s) CD005087

Abstract: Background: People with cystic fibrosis (CF) have increased transport of the salt, sodium across their airway lining. Over-absorption of sodium results in the dehydration of the liquid that lines the airway surface and (along with defective chloride ... ...

Abstract	Background: People with cystic fibrosis (CF) have increased transport of the salt, sodium across their airway lining. Over-absorption of sodium results in the dehydration of the liquid that lines the airway surface and (along with defective chloride secretion) is a primary defect in people with CF. Objectives: To determine whether the topical administration of drugs that block sodium transport improves the respiratory condition of people with CF. Search methods: We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register comprising references identified from comprehensive electronic database searches, handsearching relevant journals and abstract books of conference proceedings. We contacted principal investigators known to work in the field, previous authors and pharmaceutical companies who manufacture ion transport agents for unpublished or follow-up data.Most recent search of the Group's register: 19 December 2013. Selection criteria: Published or unpublished randomised controlled trials (RCTs) or quasi-randomised controlled trials of sodium channel blockers compared to placebo or another sodium channel blocker or the same sodium channel blocker at a different dosing regimen. Data collection and analysis: Two authors independently extracted data. Meta-analysis was limited due to differing study designs. Main results: Five RCTs, with a total of 226 participants, examining the topical administration of the short-acting sodium channel blocker, amiloride, compared to placebo were identified as eligible for inclusion in the review. In three studies over six months, there was a significant difference found in the difference in relative change in FVC in favour of placebo (weighted mean difference 1.51% (95% confidence interval -2.77 to -0.25), although heterogeneity was evident. A two-week study demonstrated that hypertonic saline with amiloride pre-treatment did not result in a significant improvement in respiratory function or mucus clearance, in contrast to pre-treatment with placebo. There were no significant differences identified in other clinically relevant outcomes. Authors' conclusions: We found no evidence that the topical administration of a short-acting sodium channel blocker improves respiratory condition in people with cystic fibrosis and some limited evidence of deterioration in lung function.
MeSH term(s)	Amiloride/administration & dosage ; Amiloride/therapeutic use ; Anti-Bacterial Agents/administration & dosage ; Cystic Fibrosis/drug therapy ; Cystic Fibrosis/physiopathology ; Forced Expiratory Volume/drug effects ; Forced Expiratory Volume/physiology ; Humans ; Mucus/metabolism ; Randomized Controlled Trials as Topic ; Respiration/drug effects ; Saline Solution, Hypertonic/administration & dosage ; Sodium Channel Blockers/administration & dosage ; Sodium Channel Blockers/therapeutic use ; Vital Capacity/drug effects ; Vital Capacity/physiology
Chemical Substances	Anti-Bacterial Agents ; Saline Solution, Hypertonic ; Sodium Channel Blockers ; Amiloride (7DZO8EB0Z3)
Language	English
Publishing date	2014-04-09
Publishing country	England
Document type	Journal Article ; Meta-Analysis ; Review ; Systematic Review
ISSN	1469-493X
ISSN (online)	1469-493X
DOI	10.1002/14651858.CD005087.pub4
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Article ; Online: Medical student imaging case files in the cloud.

Lilly, John F / Verma, Neha / Jordan, Sheryl G / Oldan, Jorge D / Fordham, Lynn A / Noone, Peadar G / Beck Dallaghan, Gary L

The clinical teacher

2019 Volume 17, Issue 3, Page(s) 275–279

Abstract: Background: Millennial digital learners value meaningful work, immediate feedback, collaborative communication and technology implementation. A student-produced digital imaging teaching case file-centric flipped curriculum offers these benefits. Our ... ...

Abstract	Background: Millennial digital learners value meaningful work, immediate feedback, collaborative communication and technology implementation. A student-produced digital imaging teaching case file-centric flipped curriculum offers these benefits. Our questions included: (i) is a cloud-based website platform supporting the online publication of student-selected and student-submitted teaching cases feasible; and (ii) what were the impressions of students of this educational intervention? Methods: An open-source medical student-centric radiology website was created with limited-access cloud upload capability, with site analytics continuously recorded. Medical students submitted de-identified radiology cases on a topic of their choosing, for peer review and publication. By making the host site publicly accessible, we empowered students to list their publication(s) on resumes. Following six blocks of the 2018/19 academic year after implementation, an electronic survey was sent to the eligible medical student cohort who had were enrolled in a radiology elective in order to assess the effectiveness of the intervention (n = 107). Results: The survey response rate was 52% (n = 56), of which 98% participated (n = 55) and 75% completed a teaching file (n = 42). The students assessed their ability to systematically review imaging, communicate pertinent clinical information, appropriately order imaging, correctly use the ACR Appropriateness Criteria Conclusions: Our successful student-developed teaching file takes advantage of digital radiology and the educational tools favoured by millennials. This activity meets core competencies in self-directed and lifelong learning.
MeSH term(s)	Communication ; Curriculum ; Educational Measurement ; Humans ; Learning ; Radiology/education ; Students, Medical ; Teaching
Language	English
Publishing date	2019-08-26
Publishing country	England
Document type	Journal Article
ZDB-ID	2151518-9
ISSN	1743-498X ; 1743-4971
ISSN (online)	1743-498X
ISSN	1743-4971
DOI	10.1111/tct.13059
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Article ; Online: Pan-Resistant Achromobacter xylosoxidans and Stenotrophomonas maltophilia Infection in Cystic Fibrosis Does Not Reduce Survival After Lung Transplantation.

Lobo, Leonard Jason / Tulu, Zeynep / Aris, Robert M / Noone, Peadar G

Transplantation

2015 Volume 99, Issue 10, Page(s) 2196–2202

Abstract: Background: The number of cystic fibrosis (CF) patients undergoing lung transplantation continues to grow, as does the prevalence of multidrug-resistant (MDR) gram-negative rods. However, the posttransplant survival of patients with MDR pathogens, ... ...

Abstract	Background: The number of cystic fibrosis (CF) patients undergoing lung transplantation continues to grow, as does the prevalence of multidrug-resistant (MDR) gram-negative rods. However, the posttransplant survival of patients with MDR pathogens, specifically pan-resistant Achromobacter xylosoxidans and Stenotrophomonas maltophilia, is poorly characterized. Methods: This was a retrospective review of CF patients (n = 186; all age, > 16 years) transplanted at the University of North Carolina from 1990 through 2013. Respiratory cultures before transplantation were reviewed for Achromobacter xylosoxidans and Stenotrophomonas maltophilia and their antibiotic susceptibility patterns. Bacteria were defined as pan-resistant if they were resistant or intermediate to all antibiotics tested; otherwise, organisms were defined as MDR. Patients were divided into 5 groups: pan-resistant Achromobacter xylosoxidans (n = 9), MDR Achromobacter xylosoxidans (n = 15), pan-resistant Stenotrophomonas maltophilia (n = 5), MDR Stenotrophomonas maltophilia (n = 26), and CF patients without Achromobacter xylosoxidans, Stenotrophomonas maltophilia or Bulkholderia cenocepacia (n = 131). Survival was compared, and cause of death was described. Results: The survival was similar between all cohorts (P = 0.29). Recurrence of the primary pathogen was the most common with pan-resistant Achromobacter xylosoxidans (100%) followed by MDR Stenotrophomonas maltophilia (46%), MDR Achromobacter xylosoxidans (33%), and finally, pan-resistant Stenotrophomonas maltophilia (20%). Death attributable to the primary pathogen was uncommon, occurring in 2 patients with MDR Stenotrophomonas maltophilia and 2 patients with MDR Achromobacter xylosoxidans. Conclusions: The CF patients with Achromobacter xylosoxidans and Stenotrophomonas maltophilia have similar posttransplant survival as compared to other CF patients, irrespective of their antibiotic susceptibility patterns. The presence of these organisms should not preclude lung transplantation.
MeSH term(s)	Achromobacter denitrificans/drug effects ; Adolescent ; Adult ; Anti-Bacterial Agents/therapeutic use ; Cystic Fibrosis/microbiology ; Cystic Fibrosis/mortality ; Cystic Fibrosis/surgery ; Drug Resistance, Bacterial ; Drug Resistance, Multiple ; Female ; Gram-Negative Bacterial Infections/complications ; Gram-Negative Bacterial Infections/drug therapy ; Gram-Negative Bacterial Infections/mortality ; Humans ; Lung Transplantation/adverse effects ; Male ; Prevalence ; Recurrence ; Retrospective Studies ; Stenotrophomonas maltophilia/drug effects ; Treatment Outcome ; Young Adult
Chemical Substances	Anti-Bacterial Agents
Language	English
Publishing date	2015-10
Publishing country	United States
Document type	Journal Article
ZDB-ID	208424-7
ISSN	1534-6080 ; 0041-1337
ISSN (online)	1534-6080
ISSN	0041-1337
DOI	10.1097/TP.0000000000000709
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Article ; Online: Primary ciliary dyskinesia

Noone Peadar G

Respiratory Research, Vol 2, Iss 5, p E

a report from ATS 2001, May 18–23, San Francisco

2001 Volume 001

Abstract: Abstract Primary ciliary dyskinesia (PCD) is a genetic disorder of abnormal ciliary structure and function that leads to defective mucociliary clearance, resulting in oto-sino-pulmonary disease, and infertility. The disease is currently under intense ... ...

Abstract	Abstract Primary ciliary dyskinesia (PCD) is a genetic disorder of abnormal ciliary structure and function that leads to defective mucociliary clearance, resulting in oto-sino-pulmonary disease, and infertility. The disease is currently under intense investigation by a number of research groups worldwide. At the recent American Thoracic Society meeting in San Francisco in May 2001, two sessions focused on PCD; a symposium session on May 21 with several featured expert speakers was followed by a mini-symposium on Tuesday May 22, with one featured speaker and presentation of nine abstracts covering a range of research topics. Mattias Salathe (University of Miami, USA) and Stephen Brody (Washington University, St Louis, USA) chaired the symposium session. Presentations focused on the clinical spectrum of PCD, the genetics of PCD, a proteomics approach to detail the structure of cilia, the role of cilia in the embryology of situs laterality, and airway epithelial cell biology. The mini-symposium was chaired by Peadar Noone (University of North Carolina, USA) and Malcolm King (University of Alberta, USA) and included presentations on the use of PCD as a human disease model, accurate definition of the phenotype using clinical and cell biologic markers, and molecular studies. The latter reports ranged from isolation of a protein involved in ciliary structure and function to genetic studies using linkage analysis and the candidate gene approach. Clinicians and scientists alike displayed considerable interest at both sessions, and there were several lively question–answer sessions.
Keywords	cilia ; dynein ; mutations ; nitric oxide ; primary ciliary dyskinesia ; Diseases of the respiratory system ; RC705-779 ; Specialties of internal medicine ; RC581-951 ; Internal medicine ; RC31-1245 ; Medicine ; R ; DOAJ:Internal medicine ; DOAJ:Medicine (General) ; DOAJ:Health Sciences
Subject code	610
Language	English
Publishing date	2001-06-01T00:00:00Z
Publisher	BioMed Central
Document type	Article ; Online
Database	BASE - Bielefeld Academic Search Engine (life sciences selection)

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