Hypoglossal Nerve Stimulation for Obstructive Sleep Apnea in a Young Child With Down Syndrome.

Obstructive sleep apnea (OSA) is common in children with Down syndrome (DS). Adenoidectomy and/or tonsillectomy are the usual first interventions employed to treat OSA in children with DS but sometimes do not achieve adequate resolution of clinical signs. Positive airway pressure treatment is often used next, but this treatment is poorly tolerated by this population. Persistent OSA can adversely affect a child's health and cognitive development. Hypoglossal nerve stimulation (HGNS), previously shown to be safe and effective in adults with OSA, has been used in children as young as 10 years old with DS and has achieved measurable neurocognitive benefits. The US Food and Drug Administration recently lowered the age for HGNS implantation to 13 years for children with DS. However, questions remain regarding treatment of refractory OSA in younger children. Here, we report the case of a 4-year-old boy with DS and treatment-refractory OSA who underwent successful HGNS implantation. The decision to proceed with HGNS implantation in such a young child involved discussions about anatomic feasibility and potential neurocognitive benefits. The device was implanted without complication and with minimal postoperative bulk. This case suggests a possible treatment option that can be discussed in the course of shared decision-making between clinicians and families of young children with DS and treatment-refractory OSA.

Paediatric sleep apnea event prediction using nasal air pressure and machine learning.

Sleep-disordered breathing is an important health issue for children. The objective of this study was to develop a machine learning classifier model for the identification of sleep apnea events taken exclusively from nasal air pressure measurements acquired during overnight polysomnography for paediatric patients. A secondary objective of this study was to differentiate site of obstruction exclusively from hypopnea event data using the model. Computer vision classifiers were developed via transfer learning to either normal breathing while asleep, obstructive hypopnea, obstructive apnea or central apnea. A separate model was trained to identify site of obstruction as either adeno-tonsillar or tongue base. In addition, a survey of board-certified and board-eligible sleep physicians was completed to compare clinician versus model classification performance of sleep events, and indicated very good performance of our model relative to human raters. The nasal air pressure sample database available for modelling comprised 417 normal, 266 obstructive hypopnea, 122 obstructive apnea and 131 central apnea events derived from 28 paediatric patients. The four-way classifier achieved a mean prediction accuracy of 70.0% (95% confidence interval [67.1-72.9]). Clinician raters correctly identified sleep events from nasal air pressure tracings 53.8% of the time, whereas the local model was 77.5% accurate. The site of obstruction classifier achieved a mean prediction accuracy of 75.0% (95% confidence interval [68.7-81.3]). Machine learning applied to nasal air pressure tracings is feasible and may exceed the diagnostic performance of expert clinicians. Nasal air pressure tracings of obstructive hypopneas may "encode" information regarding the site of obstruction, which may only be discernable by machine learning.

The Great ESKAPE: Exploring the Crossroads of Bile and Antibiotic Resistance in Bacterial Pathogens.

Throughout the course of infection, many pathogens encounter bactericidal conditions that threaten the viability of the bacteria and impede the establishment of infection. Bile is one of the most innately bactericidal compounds present in humans, functioning to reduce the bacterial burden in the gastrointestinal tract while also aiding in digestion. It is becoming increasingly apparent that pathogens successfully resist the bactericidal conditions of bile, including bacteria that do not normally cause gastrointestinal infections. This review highlights the ability of Enterococcus, Staphylococcus, Klebsiella, Acinetobacter, Pseudomonas, Enterobacter (ESKAPE), and other enteric pathogens to resist bile and how these interactions can impact the sensitivity of bacteria to various antimicrobial agents. Given that pathogen exposure to bile is an essential component to gastrointestinal transit that cannot be avoided, understanding how bile resistance mechanisms align with antimicrobial resistance is vital to our ability to develop new, successful therapeutics in an age of widespread and increasing antimicrobial resistance.

Bronchiectasis in Primary Antibody Deficiencies: A Multidisciplinary Approach.

Bronchiectasis, the presence of bronchial wall thickening with airway dilatation, is a particularly challenging complication of primary antibody deficiencies. While susceptibility to infections may be the primary factor leading to the development of bronchiectasis in these patients, the condition may develop in the absence of known infections. Once bronchiectasis is present, the lungs are subject to a progressive cycle involving both infectious and non-infectious factors. If bronchiectasis is not identified or not managed appropriately, the cycle proceeds unchecked and yields advanced and permanent lung damage. Severe symptoms may limit exercise tolerance, require frequent hospitalizations, profoundly impair quality of life (QOL), and lead to early death. This review article focuses on the appropriate identification and management of bronchiectasis in patients with primary antibody deficiencies. The underlying immune deficiency and the bronchiectasis need to be treated from combined immunology and pulmonary perspectives, reflected in this review by experts from both fields. An aggressive multidisciplinary approach may reduce exacerbations and slow the progression of permanent lung damage.

Preclinical assessment of resorbable silk splints for the treatment of pediatric tracheomalacia.

OBJECTIVE: Tracheomalacia is characterized by weakness of the tracheal wall resulting in dynamic airway collapse during respiration; severe cases often require surgical intervention. Off-label external splinting with degradable implants has been reported in humans; however, there remains a need to develop splints with tunable mechanical properties and degradation profiles for the pediatric population. The objective of this pilot study is to assess the safety and efficacy of silk fibroin-based splints in a clinically relevant preclinical model of tracheomalacia. METHODS: Silk splints were evaluated in a surgically induced model of severe tracheomalacia in N = 3 New Zealand white rabbits for 17, 24, and 31 days. An image-based assay was developed to quantify the dynamic change in airway area during spontaneous respiration, and histopathology was used to study the surrounding tissue response. RESULTS: The average change in area in the native trachea was 23% during spontaneous respiration; surgically induced tracheomalacia resulted in a significant increase to 86% (P < 0.001). The average change in airway area after splint placement was reduced at all terminal time points (17, 24, and 31 days postimplantation), indicating a clinical improvement, and was not statistically different than the native trachea. Histopathology showed a localized inflammatory reaction characterized by neutrophils, eosinophils, and mononuclear cells, with early signs suggestive of fibrosis at the splint and tissue interface. CONCLUSION: This pilot study indicates that silk fibroin splints are well tolerated and efficacious in a rabbit model of severe tracheomalacia, with marked reduction in airway collapse following implantation and good tolerability over the studied time course. LEVEL OF EVIDENCE: NA Laryngoscope, 129:2189-2194, 2019.

Molecular Confirmation of Ascaris suum: Further Investigation into the Zoonotic Origin of Infection in an 8-Year-Old Boy with Loeffler Syndrome.

An 8-year-old male from south Louisiana was diagnosed with Loeffler syndrome of suspected Ascaris origin. Further investigation of the farm recovered larvated, infective Ascaris eggs from the soil in drains surrounding pens on the family's small hog farm. Molecular analysis of the recovered eggs, in conjunction with Ascaris-specific IgE, inadequate farm management and sanitation, and behavioral risk factors indicate the patient had an Ascaris suum soil-transmitted infection.