Peadar G. Noone

University of North Carolina Health Care

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Michael R. Knowles

University of North Carolina at Chapel Hill

Maimoona A. Zariwala

University of North Carolina at Chapel Hill

Kenneth N. Olivier

University of North Carolina at Chapel Hill

Margaret W. Leigh

University of North Carolina at Chapel Hill

M. Leigh Anne Daniels

University of North Carolina at Chapel Hill

Marcus P. Kennedy

Cork University Hospital

Charles L. Daley

National Jewish Health

Heymut Omran

Klinik und Poliklinik für Kinder- und Jugendmedizin

Johnny L. Carson

University of North Carolina at Chapel Hill

Kevin Winthrop

Oregon Health & Science University

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University of North Carolina at Chapel Hill

135

Lung Institute

University of North Carolina Health Care

Harvard University

Indiana University School of Medicine

University College London

Pulmonary and Critical Care Associates

Boston Children's Hospital

University of Washington

Communities In Schools of Orange County

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Benign neonatal sleep myoclonus.

PubMed (1995)

Peadar G. Noone Michelle A. King B. G. Loftus

Benign neonatal sleep myoclonus is a disorder characterised by abnormal episodic jerky movements, which occur exclusively during sleep. When the history is suggestive of the diagnosis, and physical examination and basic investigations are normal, a confident diagnosis can usually be reached, and other potentially more serious neonatal seizure disorders excluded. This allows the doctor to reassure the parents as to the prognosis--the condition resolves spontaneously within the first year, most often within the first three months, with no sequelae. We describe four patients who presented to one unit with this condition.

Sleep

Neurological examination

Medical History

Seizure Disorders

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Citations (8)

Pseudomonas aeruginosa associated with severity of non-cystic fibrosis bronchiectasis measured by the modified bronchiectasis severity score (BSI) and the FACED: The US bronchiectasis and NTM Research Registry (BRR) study

Respiratory Medicine (2020)

Radmila Choate Timothy R. Aksamit David M. Mannino Doreen Addrizzo‐Harris Alan R. Barker

Nontuberculous Mycobacteria

10.1016/j.rmed.2020.106285

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Citations (13)

Pharmacotherapy for Non-Cystic Fibrosis Bronchiectasis

CHEST Journal (2017)

Emily Henkle Timothy R. Aksamit Alan F. Barker Jeffrey R. Curtis Charles L. Daley

10.1016/j.chest.2017.04.167

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Citations (43)

Cystic fibrosis gene mutations and pancreatitis risk: Relation to epithelial ion transport and trypsin inhibitor gene mutations

Gastroenterology (2001)

Peadar G. Noone Zhaoqing Zhou Lawrence M. Silverman Paul S. Jowell Michael R. Knowles

PstI

Hereditary pancreatitis

Pancreatic Disease

10.1053/gast.2001.29673

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Citations (309)

US Cystic Fibrosis Foundation and European Cystic Fibrosis Society consensus recommendations for the management of non-tuberculous mycobacteria in individuals with cystic fibrosis

Carolina Digital Repository (University of North Carolina at Chapel Hill) (2016)

R Andres Floto Kenneth N. Olivier Lisa Saiman Charles L. Daley Jean‐Louis Herrmann

Foundation (evidence)

10.17615/0e9w-2s88

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Citations (0)

Emergence and spread of a human-transmissible multidrug-resistant nontuberculous mycobacterium

Carolina Digital Repository (University of North Carolina at Chapel Hill) (2016)

Josephine M. Bryant Dorothy Grogono Daniela Rodriguez-Rincon Isobel Everall Karen Brown

Nontuberculous Mycobacteria

10.17615/434w-g420

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Citations (3)

Trends in therapy of abnormal airway epithelial ion and liquid transport.

PubMed (1993)

Peadar G. Noone Michael R. Knowles

Airway epithelial ion transport, particularly that of sodium and chloride, influences the volume and composition of airway surface liquid. Defects in ion transport play an important role in the pathogenesis of some chronic airway diseases, for example cystic fibrosis. New aerosolised therapies that modulate defects in ion transport, and hence influence the natural history of cystic fibrosis are currently undergoing scrutiny. Agents such as sodium channel inhibitors and chloride channel activators are included in this class.

Pathogenesis

Chloride channel

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Citations (4)

Sodium channel blockers for cystic fibrosis

Cochrane database of systematic reviews (2006)

E. Burrows Kevin W Southern Peadar G. Noone

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 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: 26th August 2009. 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.

Amiloride

Sodium channel blocker

10.1002/14651858.cd005087.pub2

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Citations (9)

Enlarged Dural Sac in Idiopathic Bronchiectasis Implicates Heritable Connective Tissue Gene Variants

Annals of the American Thoracic Society (2016)

M. Leigh Anne Daniels Katherine R. Birchard Jared R. Lowe Michael V. Patrone Peadar G. Noone

Rationale: Patients with idiopathic bronchiectasis are predominantly female and have an asthenic body morphotype and frequent nontuberculous mycobacterial respiratory infections. They also demonstrate phenotypic features (scoliosis, pectus deformity, mitral valve prolapse) that are commonly seen in individuals with heritable connective tissue disorders.Objectives: To determine whether lumbar dural sac size is increased in patients with idiopathic bronchiectasis as compared with control subjects, and to assess whether dural sac size is correlated with phenotypic characteristics seen in individuals with heritable connective tissue disorders.Methods: Two readers blinded to diagnosis measured anterior–posterior and transverse dural sac diameter using L1–L5 magnetic resonance images of 71 patients with idiopathic bronchiectasis, 72 control subjects without lung disease, 29 patients with cystic fibrosis, and 24 patients with Marfan syndrome. We compared groups by pairwise analysis of means, using Tukey’s method to adjust for multiple comparisons. Dural sac diameter association with phenotypic and clinical features was also tested.Measurements and Main Results: The L1–L5 (average) anterior–posterior dural sac diameter of the idiopathic bronchiectasis group was larger than those of the control group (P < 0.001) and the cystic fibrosis group (P = 0.002). There was a strong correlation between increased dural sac size and the presence of pulmonary nontuberculous mycobacterial infection (P = 0.007) and long fingers (P = 0.003). A trend toward larger dural sac diameter was seen in those with scoliosis (P = 0.130) and those with a family history of idiopathic bronchiectasis (P = 0.149).Conclusions: Individuals with idiopathic bronchiectasis have an enlarged dural sac diameter, which is associated with pulmonary nontuberculous mycobacterial infection, long fingers, and family history of idiopathic bronchiectasis. These findings support our hypothesis that “idiopathic” bronchiectasis development reflects complex genetic variation in heritable connective tissue and associated transforming growth factor-β–related pathway genes.

10.1513/annalsats.201603-161oc

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Citations (9)

Lung transplant outcomes in cystic fibrosis patients with pre‐operative Mycobacterium abscessus respiratory infections

Clinical Transplantation (2013)

Leonard J. Lobo Lydia C. Chang Charles R. Esther Peter H. Gilligan Zeynep Tulu

Abstract Background Mycobacterium abscessus in cystic fibrosis ( CF ) patients is considered a contraindication to lung transplantation. We examine the post‐transplant outcomes of CF patients with M . abscessus pre‐transplant. Methods CF patients transplanted at the U niversity of N orth C arolina from 1992 to 2012 were retrospectively examined. Patients with at least one respiratory sample positive for M . abscessus prior to transplantation were included. Data collected included age, FEV 1 , body mass index ( BMI ), systemic steroid use, diabetes mellitus, ventilatory assistance, co‐existent CF pathogens, imaging, post‐transplant complications, and survival. Results (n = 13) At transplant, mean age was 24.6 yr, mean BMI was 18.1 kg/m 2 , six had 3+ positive smears for M . abscessus , and three were ventilator dependent. All met A merican T horacic S ociety microbiological criteria for disease pre‐transplant. Three patients developed M . abscessus ‐related complications, with clearance of the organism following treatment. Survival post‐transplant shows 77% alive at one yr, 64% at three yr, and 50% at five yr; none died of M . abscessus . The survival data showed no statistically significant difference (p = 0.8) compared with a contemporaneously transplanted population of CF patients without M . abscessus (n = 154). Conclusion Lung transplantation, with favorable survival, is possible in CF patients with M . abscessus . Even if M . abscessus recurs, local control and clearance is possible.

Mycobacterium abscessus

Contraindication

10.1111/ctr.12140

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Citations (104)