Valerie G. Kirk

Diagnostic approach to obstructive sleep apnea in children

Obstructive sleep apnea syndrome (OSAS) in childhood is a disorder of breathing during sleep characterized by prolonged partial upper airway obstruction and/or intermittent complete obstruction that disrupts normal ventilation during sleep and normal sleep patterns. A spectrum of severity related to the degree of upper airway resistance, to the duration of the disease, to the presence or absence of hypoxemia episodes, and to certain clinical features can be described. Symptomatic children may not fit the criteria for diagnosis established for OSAS in adults; age-specific standards are needed. Both anatomical factors that increase upper airway resistance, e.g. adenotonsillar hypertrophy, and functional processes that decrease upper airway tone, e.g. REM sleep, contribute to the pathogenesis of pediatric OSAS. Sequelae of OSAS in children include neurobehavioural abnormalities, stunting of growth, and cor pulmonale. Both the history and physical examination should target the sleeping child; parents often report loud snoring, difficulty breathing, and obstructive apneas. The gold standard investigation to establish the diagnosis and to quantitate disease severity is overnight polysomnography. Home cardiopulmonary sleep studies have been shown to be an accurate and practical alternative to overnight laboratory polysomnography for routine evaluation of non-complex children with adenotonsillar hypertrophy. Children with documented severe OSAS are at increased post-operative risk for airway compromise and should be observed and monitored carefully. Adenotonsiliectomy is the most common therapy for OSAS in children; as a second-line treatment, the use of nasal CPAP in children with OSAS has been very successful in experienced hands.

SLEEP-DISORDERED BREATHING IN PATIENTS WITH MYELOMENINGOCELE : THE MISSED DIAGNOSIS

Moderate to severe sleep‐disordered breathing (SDB) was identified in 20% (17 of 83) of children with spina bifida/myelomeningocele (SB/MM) at the Montreal Childrens Hospital. The prevalence of SDB in patients with SB/MM elsewhere has not been determined. To establish current practices for identifying SDB in patients with SB/MM, questionnaires were sent to the coordinators of the 212 spina‐bifida clinics in Canada and in the United States. Eighty‐six (41%) questionnaires were returned, representing data on 13 349 patients. Although 67% of the responding centers reported availability of cardiorespiratory sleep studies, only 996 (7.5%) patients with SB/MM had been tested and only 418 (3.1%) patients had been diagnosed with SDB. Across clinics, the prevalence of SDB was directly related to the frequency of testing. Of 380 deaths over the past 10 years, SDB and sudden unexplained death during sleep were identified as the cause of death in 49 (12.8%) and 34 (8.9%) patients, respectively. Moderate to severe SDB may not have been identified in a significant number of patients with SB/MM because they have not been tested.

Non-respiratory indications for polysomnography and related procedures in children: An evidence-based review

OBJECTIVEnThis evidence-based review provides a systematic and comprehensive review of the literature regarding the utility of polysomnography for the evaluation of non-respiratory sleep disorders in children including hypersomnias, parasomnias, sleep-related movement disorders, and sleep in other special populations.nnnMETHODSnA task force of pediatric sleep medicine experts performed a systematic review of the literature regarding the use of polysomnography for non-respiratory sleep disorders in children. They identified and graded 76 papers as evidence.nnnRESULTSnThe main results include (1) polysomnography combined with the multiple sleep latency test is useful for evaluating disorders of excessive somnolence to objectively quantify sleepiness. The results have to be interpreted with consideration of the pubertal stage and regularity of the sleep patterns of the child; (2) polysomnography is indicated in children with parasomnias or sleep related movement disorders who have a high likelihood of having obstructive sleep apnea (OSA); (3) polysomnography is not routinely indicated in children with enuresis unless there is a high likelihood of OSA; (4) polysomnography can be helpful in evaluating children with restless legs syndrome (RLS) and when periodic limb movement disorder (PLMD) is suspected.nnnCONCLUSIONSnThese findings suggest that, in children with non-respiratory sleep disorders, polysomnography should be a part of a comprehensive sleep evaluation in selected circumstances to determine the nature of the events in more detail or when the suspicion of OSA is relatively high.

A double-blind, placebo-controlled intervention trial of 3 and 10 mg sublingual melatonin for post-concussion syndrome in youths (PLAYGAME): study protocol for a randomized controlled trial

BackgroundBy the age of sixteen, one in five children will sustain a mild traumatic brain injury also known as concussion. Our research found that one in seven school children with mild traumatic brain injury suffer post-concussion syndrome symptoms for three months or longer. Post-concussion syndrome is associated with significant disability in the child and his/her family and yet there are no evidence-based medical treatments available. Melatonin has several potential mechanisms of action that could be useful following mild traumatic brain injury, including neuroprotective effects. The aim of this study is to determine if treatment with melatonin improves post-concussion syndrome in youths following mild traumatic brain injury. Our hypothesis is that treatment of post-concussion syndrome following mild traumatic brain injury with 3 or 10 mg of sublingual melatonin for 28 days will result in a decrease in post-concussion syndrome symptoms compared with placebo.Methods/DesignNinety-nine youths with mild traumatic brain injury, aged between 13 and 18 years, who are symptomatic at 30 days post-injury will be recruited. This study will be conducted as a randomized, double blind, placebo-controlled superiority trial of melatonin. Three parallel treatment groups will be examined with a 1:1:1 allocation: sublingual melatonin 3 mg, sublingual melatonin 10 mg, and sublingual placebo. Participants will receive treatment for 28 days. The primary outcome is a change on the Post-Concussion Symptom Inventory (Parent and Youth). The secondary outcomes will include neurobehavioral function, health-related quality of life and sleep. Neurophysiological and structural markers of change, using magnetic resonance imaging techniques and transcranial magnetic stimulation, will also be investigated.DiscussionMelatonin is a safe and well-tolerated agent that has many biological properties that may be useful following a traumatic brain injury. This study will determine whether it is a useful treatment for children with post-concussion syndrome. Recruitment commenced on 4 December 2014.Trial registrationThis trial was registered on 6 June 2013 at ClinicalTrials.gov. Registration number: NCT01874847.

Munchausen's syndrome presenting as hemoptysis in a 12-year-old girl.

The case of Munchausens syndrome presenting as hemoptysis in a 12-year-old girl is presented. The features of Munchausens syndrome are reviewed. Munchausens syndrome should be included in the differential diagnosis of hemoptysis in a child, especially when accompanied by a dramatic presentation, changing symptoms and negative diagnostic investigations.

American Academy of Sleep Medicine Position Paper for the Use of a Home Sleep Apnea Test for the Diagnosis of OSA in Children

INTRODUCTIONnThe purpose of this position paper is to establish the American Academy of Sleep Medicines (AASM) position on the use of a home sleep apnea test (HSAT) for the diagnosis of obstructive sleep apnea (OSA) in children (birth to 18 years of age).nnnMETHODSnThe AASM commissioned a task force of 8 experts in sleep medicine to review the available literature on the use of an HSAT to diagnose OSA in children. The task force developed the position statement based on a thorough review of these studies and their clinical expertise. The AASM Board of Directors approved the final position statement.nnnPOSITION STATEMENTnUse of a home sleep apnea test is not recommended for the diagnosis of obstructive sleep apnea in children. The ultimate judgment regarding propriety of any specific care must be made by the clinician, in light of the individual circumstances presented by the patient, available diagnostic tools, accessible treatment options, and resources.

Infancy onset of symptoms of narcolepsy in a child.

In 1880, Gelineaul first coined the term narcolepsy, a pathological condition characterized by recurrent attacks of irresistible sleepiness. It was not until 80 years later that Yoss and Daly2 described narcolepsy as a tetrad of clinical features including the following: excessive daytime sleepiness, cataplexy (sudden loss of muscle tone without losing consciousness; brought on by emotional triggers), sleep paralysis (muscle atonia in rapid eye movement (REM) sleep continuing while awake), and hypnogogic hallucinations (vivid dream-like visual images before falling asleep). These 4 symptoms are variants of inappropriate REM sleep intrusion during wakefulness. In children, the diagnosis remains underrecognized despite reported cases in the literature.3-5 The potential delays in diagnosis are related to the frequent absence of 1 or more of the associated features of narcolepsy, compounded by differences in expression of symptoms in children. The significance of recognizing and treating this disease in children is to prevent potentially serious psychological and academic consequences.

Noninvasive Positive Airway Pressure Treatment in Children Less Than 12 Months of Age

Study Objectives. We identified the associated conditions of patients less than 12 months of age who were referred for polysomnogram (PSG) studies. We collated PSG findings and physician interpretation. We determined the correlation between the recommended treatment by the PSG interpreting physician and actual prescribed treatment by the referring or subjects physician. We determined adherence with noninvasive positive airway pressure (PAP) treatment. Methods. This was a retrospective cohort study. Participants included children less than 12 months of age referred for PSG studies between 2007 and 2012. Results. 92 patients under the age of 12 months were included in the study analysis. Mean (standard deviation, SD) age in days at time of the PSG study was 208.5 (101.2). 35 (38%) patients had a diagnosis of Trisomy 21. Seven (8%) patients had no prior diagnosis. Median (Q1, Q3) apnea hypopnea index (AHI) was 22.5 (11.3–37.0). Agreement between the PSG interpreting physicians recommendation and actual prescribed treatment by the referring or subjects physician was 85.9% [95% CI 77.1–91.6]. Mean (SD) percentage days with PAP therapy usage more than 4 hours was 25.2% (32). Conclusions. In our experience, despite consistent physician messaging to families, adherence with noninvasive PAP treatment is low.

The scope of sleep problems in Canadian children and adolescents with obesity

OBJECTIVEnTo determine the scope of sleep concerns, clinical features, and polysomnography (PSG) results and to identify factors that predict obstructive sleep apnea (OSA) in a cohort of children with obesity.nnnMETHODSnThe study was a multicenter retrospective chart review. Data were collected from three pediatric sleep laboratories over a two year period for all children of age 8-16 years with a body mass index [BMI] ≥95th centile who were undergoing PSG. Data sources included clinical charts and PSG results. Clinical and PSG factors were examined as predictors of OSA.nnnRESULTSnA total of 210 children met inclusion criteria, and 205 had sufficient data for analysis. The mean age was 12.5xa0±xa02.7 years; and 65% were male. Multiple sleep concerns and comorbidities were reported in most children (90% and 91%, respectively). OSA was identified by PSG in 44% of children; and 28% of children demonstrated moderate/severe OSA. Mouth breathing/nasal congestion (odds ratio [OR]xa0=xa00.33, 95% confidence interval [CI]xa0=xa00.18-0.61), syndrome/multiple anomalies (ORxa0=xa02.4, 95% CIxa0=xa01.22-4.93), and family history of OSA (ORxa0=xa02.7, 95% CIxa0=xa01.2-5.8) or sleep problems (ORxa0=xa012.4, 95% CIxa0=xa01.5, 99.6) were the only factors predictive of OSA. Oxygen desaturation index <6 events/h measured by PSG showed an OR of 4.96 (95% CIxa0=xa02.27-10.86) for the absence of OSA.nnnCONCLUSIONSnChildren with obesity who undergo PSG are medically complex with multiple sleep concerns including a high burden of daytime symptoms; slightly less than half of children demonstrate polysomnographic features of OSA. Earlier identification of OSA, recognition of non-OSA sleep concerns, and treatment strategies to improve sleep may contribute to overall health outcomes for children with obesity.

Insulin Resistance and Hypertension in Obese Youth With Sleep-Disordered Breathing Treated With Positive Airway Pressure: A Prospective Multicenter Study

STUDY OBJECTIVESnThere is evidence that cardiometabolic disease associated with obesity and sleep-disordered breathing (SDB) in adults is present in youth. SDB is often treated with positive airway pressure (PAP) in youth with obesity. Our aims were to determine: (1) the prevalence of cardiometabolic disease and (2) whether PAP improves markers of cardiometabolic disease, in youth with obesity and newly diagnosed moderate-severe SDB.nnnMETHODSnA prospective multicenter cohort study was conducted in youth (8 to 16 years old) with obesity, prescribed PAP therapy for newly diagnosed moderate-severe SDB. Assessments occurred at baseline and at 6 and 12 months. Outcomes included markers of insulin resistance (change in homeostasis model assessment of insulin resistance (HOMA-IR) at 6 months = primary outcome), hypertension (24-hour ambulatory/blood pressure) and inflammation (high-sensitivity C-reactive protein: hs-CRP).nnnRESULTSnTwenty-seven participants were enrolled. Of those with baseline testing available, 10/25 (40%) had HOMA-IR above the 97th percentile, 10/23 (44%) were hypertensive, 16/23 (70%) had loss of nocturnal blood pressure dip and hs-CRP was elevated in 16/27 (64%). There were no significant changes over time in markers of metabolic dysfunction or blood pressure, nor between PAP-adherent and non-adherent subgroups.nnnCONCLUSIONSnIn youth with obesity and SDB, metabolic dysfunction and hypertension were highly prevalent. There were no statistically significant improvements in cardiometabolic markers 1 year after the prescription of PAP therapy, although clinically relevant improvements were seen in insulin resistance and systolic blood pressure load, important predictors of future risk of cardiovascular disease. Larger, longer-term studies are needed to determine whether PAP improves cardiometabolic outcomes in obese youth.nnnCOMMENTARYnA commentary on this article appears in this issue on page 1025.