This document summarises the conclusions of a European Respiratory Society Task Force on the diagnosis and management of obstructive sleep disordered breathing (SDB) in childhood and refers to children aged 2-18 years. Prospective cohort studies describing the natural history of SDB or randomised, double-blind, placebo-controlled trials regarding its management are scarce. Selected evidence (362 articles) can be consolidated into seven management steps. SDB is suspected when symptoms or abnormalities related to upper airway obstruction are present (step 1). Central nervous or cardiovascular system morbidity, growth failure or enuresis and predictors of SDB persistence in the long-term are recognised (steps 2 and 3), and SDB severity is determined objectively preferably using polysomnography (step 4). Children with an apnoea-hypopnoea index (AHI) >5 episodes·h(-1), those with an AHI of 1-5 episodes·h(-1) and the presence of morbidity or factors predicting SDB persistence, and children with complex conditions (e.g. Down syndrome and Prader-Willi syndrome) all appear to benefit from treatment (step 5). Treatment interventions are usually implemented in a stepwise fashion addressing all abnormalities that predispose to SDB (step 6) with re-evaluation after each intervention to detect residual disease and to determine the need for additional treatment (step 7).

https://erj.ersjournals.com/content/erj/47/1/69.full.pdf

Obstructive sleep disordered breathing in 2- to 18-year-old children: diagnosis and management.

Attention deficit hyperactivity disorder and sleep disordered breathing in pediatric populations: A meta-analysis

Background 
Sleep disordered breathing (SDB) can lead to daytime sleepiness, growth failure and developmental delay in children. Polysomnography (PSG), the gold standard to diagnose SDB, is a highly resource-intensive test, confined to the sleep laboratory.


Aim 
To combine the blood oxygen saturation (SpO2) characterization and cardiac modulation, quantified by pulse rate variability (PRV), to identify children with SDB using the Phone Oximeter, a device integrating a pulse oximeter with a smartphone.


Methods 
Following ethics approval and informed consent, 160 children referred to British Columbia Children's Hospital for overnight PSG were recruited. A second pulse oximeter sensor applied to the finger adjacent to the one used for standard PSG was attached to the Phone Oximeter to record overnight pulse oximetry (SpO2 and photoplethysmogram (PPG)) alongside the PSG.


Results 
We studied 146 children through the analysis of the SpO2 pattern, and PRV as an estimate of heart rate variability calculated from the PPG. SpO2 variability and SpO2 spectral power at low frequency, was significantly higher in children with SDB due to the modulation provoked by airway obstruction during sleep (p-value ). PRV analysis reflected a significant augmentation of sympathetic activity provoked by intermittent hypoxia in SDB children. A linear classifier was trained with the most discriminating features to identify children with SDB. The classifier was validated with internal and external cross-validation, providing a high negative predictive value (92.6%) and a good balance between sensitivity (88.4%) and specificity (83.6%). Combining SpO2 and PRV analysis improved the classification performance, providing an area under the receiver operating characteristic curve of 88%, beyond the 82% achieved using SpO2 analysis alone.


Conclusions 
These results demonstrate that the implementation of this algorithm in the Phone Oximeter will provide an improved portable, at-home screening tool, with the capability of monitoring patients over multiple nights.

/pdf/development-of-a-screening-tool-for-sleep-disordered-252d0okrpd.pdf

Development of a screening tool for sleep disordered breathing in children using the phone Oximeter

Overnight polysomnography (PSG) is an expensive procedure which can only be used in a minority of cases, although it remains the gold standard for the diagnosis of sleep disordered breathing (SDB). The objective of this study was to develop a simple, PSG-validated tool to screen SDB, thus reducing the use of PSG. For every participant we performed PSG and a sleep clinical record was completed. The sleep clinical record consists of three items: physical examination, subjective symptoms and clinical history. The clinical history analyses behavioural and cognitive problems. All three items were used to create a sleep clinical score (SCS). We studied 279 children, mean ± SD age 6.1 ± 3.1 years, 63.8% male; 27.2% with primary snoring and 72.8% with obstructive sleep apnoea (OSA) syndrome. The SCS was higher in the OSA syndrome group compared to the primary snoring group (8.1 ± 9.6 versus 0.4 ± 0.3, p<0.005), correlated with apnoea/hypopnoea index (p=0.001) and had a sensitivity of 96.05%. Positive and negative likelihood ratios were 2.91 and 0.06, respectively. SCS may effectively be used to screen patients as candidates for PSG study for suspected OSA syndrome, and to enable those with a mild form of SDB to receive early treatment.

https://erj.ersjournals.com/content/erj/41/6/1355.full.pdf

Sleep clinical record: an aid to rapid and accurate diagnosis of paediatric sleep disordered breathing

Objective To determine the prevalence of sleep-disordered breathing (SDB) symptoms and respiratory events during sleep in infants with cleft lip and/or palate (CL/P). Design Prospective observational study. Setting Cleft palate clinic, tertiary care paediatric hospital, before palate surgery. Patients Consecutive newborn infants with CL/P. Main outcome measures Demographics, clinical history, sleep symptoms, facial measurement and polysomnography (PSG; sleep study) data. Results Fifty infants completed PSG at 2.7±2.3 months; 56% were male, and 30% had a clinical diagnosis of Pierre Robin sequence (PRS) or a syndrome. The majority of infants (75%) were reported to snore frequently or constantly, while 74% were reported to have heavy or loud breathing during sleep. The frequency of parent-reported difficulty with breathing during sleep was 10% for infants with isolated CL/P, 33% for those with syndrome, and 43% for PRS (χ 2 16.1, p 1 event/h, and 75% had an OMAHI >3 events/h. Infants with PRS had higher OMAHI (34.3±5.1) than infants with isolated CL/P (7.6±1.2) or infants with syndromes (15.6±5.7, F stat, p Conclusions The results highlight that infants across the spectrum of CL/P have a high risk of SDB symptoms and obstructive respiratory events before palate surgery. Clinicians should enquire about symptoms of SDB and consider investigation with polysomnography in all infants with CL/P.

https://adc.bmj.com/content/archdischild/97/12/1058.full.pdf

The spectrum of sleep-disordered breathing symptoms and respiratory events in infants with cleft lip and/or palate

Neurocognitive assessment and sleep analysis in children with sleep-disordered breathing.

CHRONIC CARDIOVASCULAR AUTONOMIC ABNORMALITIES DURING BOTH SLEEP AND WAKEFULNESS HAVE BEEN DEMONSTRATED IN ADULTS WITH obstructive sleep apnea syndrome (OSAS), caused by sympathovagal balance alterations.1,–3 Persistent muscle sympathetic nerve activation and high levels of circulating norepinephrine increase the sympathetic drive in adults with OSAS.4, 5 At the same time, heart rate variability (HRV) during sleep is markedly reduced, which suggests impaired parasympathetic control.6 The pathogenesis of this persistent sympathetic activation has yet to be fully understood. Repetitive episodes of apnea cause hypoxia and sleep fragmentation, inducing tonic activation of chemoreflex activity, and an increase in sympathetic nerve activity, as, demonstrated by muscle sympathetic microneurography.7,–11 Studies in children with OSAS have reported increased blood pressure,12 changes in cardiac structure and function,13,14 increased fasting insulin and lipid levels,15,16 and endothelial dysfunction, as signs of cardiovascular damage.17,18 However, the few studies that have evaluated autonomic dysfunction reported an increase in diastolic blood pressure, both during wakefulness and sleep,19,20 as well as an increase in sympathetic activity, demonstrated by peripheral arterial tonometry,21,22 and catecholamine concentration measurements in plasma and urine.23 Yet other studies have documented an increase in HRV,24 and a drop in blood pressure during wakefulness, as assessed by the head-up tilt test, in children with OSAS.25 The head-up tilt test is part of the Ewing battery, which is a physiological test for the assessment of autonomic function.26 It studies cardiovascular autonomic reflexes by analyzing changes in heart rate and arterial pressure induced by a range of physiological stimuli.

The aim of our study was to investigate the activity of the autonomic nervous system during wakefulness using the cardiovascular tests in a cohort of children with OSAS, in order to detect not only sympathetic, but also parasympathetic activity.

/pdf/autonomic-cardiovascular-tests-in-children-with-obstructive-51104jtmgj.pdf

Autonomic Cardiovascular Tests in Children with Obstructive Sleep Apnea Syndrome

There is growing recognition that the heart is a key contributor to the pathophysiology of congenital diaphragmatic hernia (CDH), in conjunction with developmental abnormalities of the lung and pulmonary vasculature. Investigations to date have demonstrated altered fetal cardiac morphology, notably relative hypoplasia of the fetal left heart, as well as early postnatal right and left ventricular dysfunction which appears to be independently associated with adverse outcomes. However, many more unknowns remain, not least an understanding of the genetic and cellular basis for cardiac dysplasia and dysfunction in CDH, the relationship between fetal, postnatal and long-term cardiac function, and the impact on other parts of the body especially the developing brain. Consensus on how to measure and classify cardiac function and pulmonary hypertension in CDH is also required, potentially using both non-invasive imaging and biomarkers. This may allow routine assessment of the relative contribution of cardiac dysfunction to individual patient pathophysiological phenotype and enable better, individualized therapeutic strategies incorporating targeted use of fetal therapies, cardiac pharmacotherapies, and extra-corporeal membrane oxygenation (ECMO). Collaborative, multi-model approaches are now required to explore these unknowns and fully appreciate the role of the heart in CDH.

The heart in congenital diaphragmatic hernia: Knowns, unknowns, and future priorities

(1) Objective: This review aims to identify the clinical and practical barriers to optimizing nutrition in newborn infants with congenital heart disease (CHD) and to describe updated evidence-based recommendations for clinical and nutritional management of these patients in a narrative review. (2) Research Methods and Procedures: We conducted a search of the relevant literature published from 2000 to December 2021. (3) Results: CHD patients undergo several nutritional challenges related to the underlying cardiac disease anomaly, the potential increased risk of NEC, and delayed enteral feeding, resulting in inadequate energy intake and sub-optimal growth, increased morbidity and mortality. (4) Conclusions: To optimize nutrition and growth in newborn infants with CHD, standardized protocols should be implemented. Regular nutritional and growth assessment with a multi-disciplinary team is essential. We propose a decisional algorithm that may represent a potentially useful tool to guide clinicians to optimize growth and nutrition.

/pdf/current-strategies-to-optimize-nutrition-and-growth-in-d5jidx22.pdf

Current Strategies to Optimize Nutrition and Growth in Newborns and Infants with Congenital Heart Disease: A Narrative Review

A patient with comorbid intellectual disability, catatonic schizophrenia, and recurrent oneiroid state of consciousness improved on long-acting risperidone and remains well at the three-year follow-up. We report a case treated with 50 mg long-acting risperidone administered every 14 days, who has been followed-up for three years. We studied his regional cerebral blood flow through technetium-99 m hexamethylpropyleneamine oxime single-photon emission computed tomography after two years of treatment. Symptoms of catatonic schizophrenia improved after two months of treatment, followed suit by oneiroid syndrome remission. Two years later, his brain perfusion was normal. No side effect has occurred since the patient was started on long-acting risperidone. Long-acting risperidone proved to be safe and effective in treating symptoms of catatonia and oneiroid syndrome.

Anna Claudia Massolo

Papers

Neurocognitive assessment and sleep analysis in children with sleep-disordered breathing.

Autonomic Cardiovascular Tests in Children with Obstructive Sleep Apnea Syndrome

The heart in congenital diaphragmatic hernia: Knowns, unknowns, and future priorities

Current Strategies to Optimize Nutrition and Growth in Newborns and Infants with Congenital Heart Disease: A Narrative Review

Effectiveness of long-acting risperidone in a patient with comorbid intellectual disability, catatonic schizophrenia, and oneiroid syndrome: