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1.
Sleep ; 45(9)2022 09 08.
Article in English | MEDLINE | ID: mdl-35695235

ABSTRACT

This study was aimed to evaluate the yearly incidence of pediatric narcolepsy prior to and following the 2009 H1N1 pandemic and to evaluate seasonal patterns of narcolepsy onset and associations with H1N1 influenza infection in the United States. This was a multicenter retrospective study with prospective follow-up. Participants were recruited from members of the Pediatric Working Group of the Sleep Research Network including 22 sites across the United States. The main outcomes were monthly and yearly incident cases of childhood narcolepsy in the United States, and its relationship to historical H1N1 influenza data. A total of 950 participants were included in the analysis; 487 participants were male (51.3%). The mean age at onset of excessive daytime sleepiness (EDS) was 9.6  ±â€… 3.9 years. Significant trend changes in pediatric narcolepsy incidence based on EDS onset (p  <  .0001) occurred over the 1998-2016 period, peaking in 2010, reflecting a 1.6-fold increase in narcolepsy incidence. In addition, there was significant seasonal variation in narcolepsy incident cases, with increased cases in spring (p  <  .05). Cross-correlation analysis demonstrated a significant correlation between monthly H1N1 infection and monthly narcolepsy incident cases (p  =  .397, p  <  .0001) with a lag time of 8 months. We conclude that there is a significant increase in pediatric narcolepsy incidence after the 2009 H1N1 pandemic in the United States. However, the magnitude of increase is lower than reported in European countries and in China. The temporal correlation between monthly H1N1 infection and monthly narcolepsy incidence, suggests that H1N1 infection may be a contributing factor to the increased pediatric narcolepsy incidence after the 2009 H1N1 pandemics.


Subject(s)
Disorders of Excessive Somnolence , Influenza A Virus, H1N1 Subtype , Influenza Vaccines , Influenza, Human , Narcolepsy , Child , Disorders of Excessive Somnolence/complications , Female , Humans , Incidence , Influenza, Human/complications , Influenza, Human/epidemiology , Male , Narcolepsy/epidemiology , Narcolepsy/etiology , Prospective Studies , Retrospective Studies , Sleep , Vaccination/adverse effects
3.
Cleft Palate Craniofac J ; 57(7): 808-818, 2020 07.
Article in English | MEDLINE | ID: mdl-31973553

ABSTRACT

OBJECTIVE: To determine pre- and postoperative prevalence of obstructive sleep apnea (OSA) in patients with 22q11.2 deletion syndrome (DS) undergoing wide posterior pharyngeal flap (PPF) surgery for velopharyngeal dysfunction (VPD). DESIGN: Retrospective study using pre- and postoperative polysomnography (PSG) to determine prevalence of OSA. Medical records were reviewed for patients' medical comorbidities. Parents were surveyed about snoring. SETTING: Academic tertiary care pediatric hospital. PATIENTS: Forty patients with laboratory confirmed 22q11.2DS followed over a 6-year period. INTERVENTIONS: Pre- and postoperative PSG, speech evaluation, and parent surveys. MAIN OUTCOME MEASURE: Severity and prevalence of OSA, defined by obstructive apnea hypopnea index (OAHI), before and after PPF surgery to determine whether PPF is associated with increased risk of OSA. RESULTS: Mean OAHI did not change significantly after PPF surgery (1.1/h vs 2.1/h, P = .330). Prevalence of clinically significant OSA (OAHI ≥ 5) was identical pre- and postoperatively (2 of 40), with both cases having severe-range OSA requiring positive airway pressure therapy. All other patients had mild-range OSA. Nasal resonance was graded as severe preoperatively in 85% of patients. None were graded as severe postoperatively. No single patient factor or parent-reported concern predicted risk of OSA (OAHI ≥ 1.5). CONCLUSIONS: Patients with 22q11.2DS are medically complex and are at increased risk of OSA at baseline. Wide PPF surgery for severe VPD does not significantly increase risk of OSA. Careful perioperative planning is essential to optimize both speech and sleep outcomes.


Subject(s)
DiGeorge Syndrome , Sleep Apnea, Obstructive , Child , Humans , Pharynx , Retrospective Studies
4.
J Atten Disord ; 23(4): 333-340, 2019 02.
Article in English | MEDLINE | ID: mdl-25646024

ABSTRACT

OBJECTIVE: The aim of this study is to understand the impact of a 5-day period of nap restriction on sleep patterns and cognitive function in typically developing preschoolers, aged 3 to 4 years. METHOD: Following 1 week of baseline assessment, 28 children were randomly assigned to either a "napping as usual" group ( n = 15) or a 5-day period of nap restriction ( n = 13). Sleep was assessed with sleep logs and actigraphy; cognition was assessed at baseline and at the end of the intervention week. RESULTS: No group differences in sleep or cognitive function were observed at baseline. For the no-nap group, the 5-day period of daytime nap restriction resulted in increased nighttime sleep. Children in the no-nap group also showed a significant improvement in attentional control compared with baseline, whereas no such changes were observed among children in the napping-as-usual group. CONCLUSION: In preschool children who typically take naps, short-term nap restriction is associated with increased nighttime sleep and may contribute to improved attentional function.


Subject(s)
Attention Deficit Disorder with Hyperactivity/physiopathology , Attention/physiology , Sleep/physiology , Actigraphy , Analysis of Variance , Child, Preschool , Circadian Rhythm/physiology , Cognition/physiology , Female , Humans , Male , Pilot Projects , Sleep Deprivation/physiopathology
5.
Paediatr Respir Rev ; 25: 9-13, 2018 Jan.
Article in English | MEDLINE | ID: mdl-28216256

ABSTRACT

Kleine-Levin syndrome (KLS) is a rare disorder characterized by discrete episodes of hypersomnia associated with cognitive and behavioural abnormalities, as well as normal alertness and function between episodes. The prevalence of KLS may be underestimated as it is often misdiagnosed and managed as another sleep disorder, neurological disorder or psychiatric condition. KLS is more typically seen in adolescence than at other ages, and is more common in males than in females. There are currently neither standard biomarkers nor specific imaging study findings, making the diagnosis of KLS a challenge. Furthermore, there are no consistently effective therapies. The prognosis, however, is felt to be overall favorable, as episodes become progressively milder and less frequent before resolving entirely in most patients.


Subject(s)
Kleine-Levin Syndrome , Adolescent , Age Factors , Behavioral Symptoms , Cognition , Diagnosis, Differential , Female , Humans , Kleine-Levin Syndrome/diagnosis , Kleine-Levin Syndrome/psychology , Male , Prognosis
6.
Sleep ; 37(8): 1349-52, 2014 Aug 01.
Article in English | MEDLINE | ID: mdl-25083015

ABSTRACT

STUDY OBJECTIVES: Although the American Academy of Sleep Medicine (AASM) mandates that periodic limb movements during sleep (PLMS) be scored on every polysomnogram, and considers a periodic limb movement index (PLMI) > 5/h abnormal in children, there is a lack of community-derived data regarding the prevalence of PLMS in children, and no data to support this cutoff value. Therefore, the aim of this study was to determine the prevalence of PLMS in a sample of normal children. DESIGN: Retrospective study. PARTICIPANTS: 195 healthy, non-snoring children aged 5-17 years, recruited from the community, who underwent polysomnography for research purposes. METHODS: PLMS were scored using the AASM 2007 criteria. MEASUREMENTS AND RESULTS: The group age (median [IQR]) was 12.9 [10-15] years, and 58% were male. Sleep architecture was normal, and the obstructive apnea hypopnea index was 0.1 [0-0.3]/h. The median PLMI was 0/h, ranging from 0 to 35.5/h. Fifteen (7.7%) subjects had a PLMI > 5/h, and only 3 (1.5%) met the adult pathologic criterion of more than 15/h. Use of the 95th percentile PLMI cutoff of 7.2/h produced little difference in categorization between groups. Children with a PLMI > 5/h had a higher arousal index than those with a lower PLMI (11.6 [8.8-14.6] vs 8.1 [6.1-9.9]/h, respectively, P = 0.003). CONCLUSIONS: This study provides normative data to the field and supports the clinical periodic limb movement index cutoff of > 5/h based on both prevalence and the correlate of increased sleep fragmentation. Periodic limb movements during sleep are infrequent in normal children recruited from the community. CITATION: Marcus CL, Traylor J, Gallagher PR, Brooks LJ, Huang J, Koren D, Katz L, Mason TB, Tapia IE. Prevalence of periodic limb movements during sleep in normal children.


Subject(s)
Extremities/physiology , Movement , Sleep/physiology , Adolescent , Child , Female , Healthy Volunteers , Humans , Male , Nocturnal Myoclonus Syndrome/epidemiology , Nocturnal Myoclonus Syndrome/physiopathology , Polysomnography , Prevalence , Reference Values , Retrospective Studies , Sleep Deprivation/physiopathology
7.
Int J Pediatr Otorhinolaryngol ; 78(8): 1360-4, 2014 Aug.
Article in English | MEDLINE | ID: mdl-24958162

ABSTRACT

UNLABELLED: Otolaryngologic problems are common in the 22q11.2 deletion syndrome (DS) population. Structural anomalies and retrognathia may predispose these patients to obstructive sleep apnea (OSA). The current association of OSA in this population is not defined. OBJECTIVE: (1) Define the frequency of OSA in 22q11.2 DS patients referred for polysomnography (PSG). (2) Determine if OSA is present before and/or after surgery to correct velopharyngeal insufficiency (VPI). (3) Determine effect of prior adenotonsillectomy on OSA following VPI surgery. METHODS: Retrospective review of children treated from 2006 to 2013 in a tertiary care setting identified by ICD-9 758.32 (velocardiofacial syndrome) and 279.11 (DiGeorge syndrome). Surgical history and PSG data were abstracted from the identified records. RESULTS: We identified 323 patients with 22q11.2 DS; 57 (18%) were screened at any point in care using PSG and 15 patients had PSG at multiple time points in care. In most cases, indication for PSG was sleep disordered breathing or pre-operative planning. Overall, 33 patients met criteria for OSA on PSG, accounting for 10.2% of our study population; however, the percentage of patients with OSA was significantly higher within the group of 57 patients (58%) who were screened with PSG. Twenty-one of the screened patients (54%) had PSG prior to any pharyngeal surgery and had mild to severe OSA (obstructive apnea/hypopnea index (AHI): median 5.1/h, range 1.9-25.6). Eighteen patients had PSG after adenotonsillectomy; 8 of these patients (44%) had mild to moderate OSA (median AHI 2.95/h, range 1.9-5.4). Seventeen patients had PSG after VPI surgery (palatopharyngeal flap (PPF) n=16, sphincteroplasty n=1). Nine of these patients (53%) had mild to severe OSA (median AHI 3/h, range 1.9-15). Patients who underwent adenotonsillectomy prior to VPI surgery had similar prevalence of OSA (50%, n=12) than those who did not (OSA: 60%, n=5, p=0.70). Most children had mild OSA. CONCLUSION: Prevalence of OSA in this population of 22q11.2 DS patients is higher than expected in the general population. OSA risk is highest after VPI surgery, and may be decreased by adenotonsillectomy. Providers should have awareness of increased prevalence of OSA in patients with 22q11.2 DS. Close monitoring for OSA is warranted given the likelihood of subsequent surgical intervention that can worsen OSA.


Subject(s)
DiGeorge Syndrome/complications , Sleep Apnea, Obstructive/diagnosis , Adenoidectomy , Adolescent , Child , Child, Preschool , DiGeorge Syndrome/surgery , Female , Humans , Infant , Male , Polysomnography , Retrospective Studies , Severity of Illness Index , Tonsillectomy , Velopharyngeal Insufficiency/surgery
8.
Pediatr Neurol ; 49(3): 209-12, 2013 Sep.
Article in English | MEDLINE | ID: mdl-23835274

ABSTRACT

BACKGROUND: Sleepwalking is typically a benign and self-limited non-rapid eye movement parasomnia of childhood. PATIENT: We describe an unusual 15-year-old boy referred to our sleep center for new-onset sleepwalking. RESULTS: An overnight polysomnogram was normal from a respiratory standpoint, but a concurrent extended electroencephalogram montage showed frequent epileptiform discharges from the right parietal-temporal region and two electroclinical seizures arising from the right-frontal-central-temporal region during sleep. Magnetic resonance imaging scan revealed a right parasagittal parietal region lesion consistent with a low-grade neoplasm, and surgical resection of the lesion demonstrated a right parietal dysembryoplastic neuroepithelial tumor. Complex partial seizures and sleepwalking remitted completely with anticonvulsant therapy following surgery. CONCLUSIONS: This patient highlights the differential diagnosis of nocturnal events appearing to be typical parasomnias, especially when they arise abruptly at an older age.


Subject(s)
Brain Neoplasms/complications , Somnambulism/etiology , Adolescent , Electroencephalography , Humans , Magnetic Resonance Imaging , Male , Polysomnography
9.
Br J Haematol ; 163(1): 112-7, 2013 Oct.
Article in English | MEDLINE | ID: mdl-23869561

ABSTRACT

Sickle cell disease (SCD) is a disorder known to impact the respiratory system. We sought to identify respiratory muscle force and lung volume relationships in a paediatric SCD population. Thirty-four SCD-SS subjects underwent pulmonary function testing. Height, weight, age, and gender-adjusted percent predicted maximal inspiratory pressure (MIP) and maximal expiratory pressure (MEP) values were compared to spirometry and lung volumes. Statistical analyses were performed using Pearson's correlation coefficient and paired two-tailed t-test. The mean ± standard deviation (SD) MIP and MEP was 69·6 ± 31·6 cm H2 O and 66·9 ± 22·9 cm H2 O, respectively, and mean ± SD percent predicted MIP (101·3 ± 45·9) exceeded MEP (72·1 ± 26·0) (P = 0·002). MIP correlated with forced vital capacity (FVC; r = 0·51, P = 0·001) and TLC (r = 0·54, P < 0·0001). MEP also correlated with FVC (r = 0·43, P = 0·011) and total lung capacity (TLC; r = 0·42, P = 0·013). Pearson's correlation coefficient testing yielded relationships between MIP and MEP (r = 0·64, P < 0·0001). SCD-SS patients showed correlations between respiratory muscle force and lung volume, and reduced percent predicted expiratory muscle force compared to inspiratory muscle force. Respiratory muscle strength may affect lung volumes in these patients, and expiratory muscles may be more susceptible than the diaphragm to SCD-induced vaso-occlusive damage.


Subject(s)
Anemia, Sickle Cell/physiopathology , Muscle Strength , Respiratory Muscles/physiopathology , Total Lung Capacity , Adolescent , Child , Female , Humans , Male , Prospective Studies , Respiratory Function Tests , Spirometry
10.
Am J Med Genet A ; 158A(9): 2152-61, 2012 Sep.
Article in English | MEDLINE | ID: mdl-22847950

ABSTRACT

We describe 19 unrelated individuals with submicroscopic deletions involving 10p15.3 characterized by chromosomal microarray (CMA). Interestingly, to our knowledge, only two individuals with isolated, submicroscopic 10p15.3 deletion have been reported to date; however, only limited clinical information is available for these probands and the deleted region has not been molecularly mapped. Comprehensive clinical history was obtained for 12 of the 19 individuals described in this study. Common features among these 12 individuals include: cognitive/behavioral/developmental differences (11/11), speech delay/language disorder (10/10), motor delay (10/10), craniofacial dysmorphism (9/12), hypotonia (7/11), brain anomalies (4/6) and seizures (3/7). Parental studies were performed for nine of the 19 individuals; the 10p15.3 deletion was de novo in seven of the probands, not maternally inherited in one proband and inherited from an apparently affected mother in one proband. Molecular mapping of the 19 individuals reported in this study has identified two genes, ZMYND11 (OMIM 608668) and DIP2C (OMIM 611380; UCSC Genome Browser), mapping within 10p15.3 which are most commonly deleted. Although no single gene has been identified which is deleted in all 19 individuals studied, the deleted region in all but one individual includes ZMYND11 and the deleted region in all but one other individual includes DIP2C. There is not a clearly identifiable phenotypic difference between these two individuals and the size of the deleted region does not generally predict clinical features. Little is currently known about these genes complicating a direct genotype/phenotype correlation at this time. These data however, suggest that ZMYND11 and/or DIP2C haploinsufficiency contributes to the clinical features associated with 10p15 deletions in probands described in this study.


Subject(s)
Chromosome Deletion , Chromosomes, Human, Pair 10 , Telomere , Child , Female , Humans , Infant , Infant, Newborn , Male
11.
Sleep Med ; 13(8): 1013-20, 2012 Sep.
Article in English | MEDLINE | ID: mdl-22841030

ABSTRACT

OBJECTIVES: To test agreement and define differences in periodic limb movements in sleep (PLMS) measured by polysomnography and an ankle activity monitor, and to describe PLMS variability across nights, feasibility of home monitoring, and correlates of PLMS in children with sickle cell disease (SCD). METHODS: Twenty children with SCD and restless legs syndrome (RLS) symptoms or polysomnography-documented PLMS underwent concurrent attended polysomnography and ankle activity monitoring over one to two nights and home activity monitoring for three nights. Serum iron and ferritin were measured pre- and post-polysomnography. RESULTS: Adequate sensitivity (1.00), specificity (0.69), and mean bias (5.0±7.4 PLMS/h) for identifying elevated PLMS by activity monitor were obtained when scoring the period from sleep onset to offset rather than time in bed per manufacturer recommendation, and using a cut-point of 10 PLMS/h. Compared to activity monitor, only polysomnographic PLMS demonstrated periodicity, at inter-movement intervals (IMI) 20-35 s; the activity monitor overscored PLMS at the beginning and end of sleep and at shorter IMI (5-15s; p≤0.003), suggesting misclassification of nonperiodic leg movements as PLMS by activity monitor. PLMS varied across four nights by 16.1±13.4 PLMS/h. Post-polysomnography ferritin was associated (positively) with PLMS (p=0.034); RLS symptoms were not. CONCLUSIONS: Ankle activity monitoring is a valid screening measure for PLMS in children with SCD and can readily be performed at home. Interpretation should incorporate a threshold for elevated PLMS of 10/h and scoring from sleep onset to offset, which could be identified with concurrent wrist actigraphy, to better account for true PLMS.


Subject(s)
Actigraphy/standards , Anemia, Sickle Cell/complications , Monitoring, Physiologic/standards , Polysomnography/standards , Restless Legs Syndrome , Actigraphy/methods , Adolescent , Ankle Joint/physiology , Child , Child, Preschool , Feasibility Studies , Female , Ferritins/blood , Humans , Iron/blood , Male , Monitoring, Physiologic/methods , Prospective Studies , Reproducibility of Results , Restless Legs Syndrome/complications , Restless Legs Syndrome/diagnosis , Restless Legs Syndrome/physiopathology , Sensitivity and Specificity
12.
Pediatr Pulmonol ; 47(8): 808-15, 2012 Aug.
Article in English | MEDLINE | ID: mdl-22328189

ABSTRACT

Assessment of oxyhemoglobin saturation in patients with sickle cell disease (SCD) is vital for prompt recognition of hypoxemia. The accuracy of pulse oximeter measurements of blood oxygenation in SCD patients is variable, partially due to carboxyhemoglobin (COHb) and methemoglobin (MetHb), which decrease the oxygen content of blood. This study evaluated the accuracy and reliability of a non-invasive pulse co-oximeter in measuring COHb and MetHb percentages (SpCO and SpMet) in children with SCD. We hypothesized that measurements of COHb and MetHb by non-invasive pulse co-oximetry agree within acceptable clinical accuracy with those made by invasive whole blood co-oximetry. Fifty children with SCD-SS underwent pulse co-oximetry and blood co-oximetry while breathing room air. Non-invasive COHb and MetHb readings were compared to the corresponding blood measurements. The pulse co-oximeter bias was 0.1% for COHb and -0.22% for MetHb. The precision of the measured SpCO was ± 2.1% within a COHb range of 0.4-6.1%, and the precision of the measured SpMet was ± 0.33% within a MetHb range of 0.1-1.1%. Non-invasive pulse co-oximetry was useful in measuring COHb and MetHb levels in children with SCD. Although the non-invasive technique slightly overestimated the invasive COHb measurements and slightly underestimated the invasive MetHb measurements, there was close agreement between the two methods.


Subject(s)
Anemia, Sickle Cell/blood , Carboxyhemoglobin/analysis , Methemoglobin/analysis , Oximetry/methods , Adolescent , Blood Gas Analysis , Child , Child, Preschool , Female , Humans , Male , Reproducibility of Results , Spectrophotometry
13.
Chest ; 142(1): 94-100, 2012 Jul.
Article in English | MEDLINE | ID: mdl-22241762

ABSTRACT

BACKGROUND: The prevalence of obstructive sleep apnea syndrome (OSAS) is higher in children with sickle cell disease (SCD) as compared with the general pediatric population. It has been speculated that overgrowth of the adenoid and tonsils is an important contributor. METHODS: The current study used MRI to evaluate such an association. We studied 36 subjects with SCD (aged 6.9 ± 4.3 years) and 36 control subjects (aged 6.6 ± 3.4 years). RESULTS: Compared with control subjects, children with SCD had a significantly smaller upper airway (2.8 ± 1.2 cm(3) vs 3.7 ± 1.6 cm(3), P < .01), and significantly larger adenoid (8.4 ± 4.1 cm(3) vs 6.0 ± 2.2 cm(3), P < .01), tonsils (7.0 ± 4.3 cm(3) vs 5.1 ± 1.9 cm(3), P < .01), retropharyngeal nodes (3.0 ± 1.9 cm(3) vs 2.2 ± 0.9 cm(3), P < .05), and deep cervical nodes (15.7 ± 5.7 cm(3) vs 12.7 ± 4.0 cm(3), P < .05). Polysomnography showed that 19.4% (seven of 36) of children with SCD had OSAS compared with 0% (zero of 20) of control subjects (P < .05) and that in children with SCD the apnea-hypopnea index correlated positively with upper airway lymphoid tissues size (r = 0.57, P < 001). In addition, children with SCD had lower arterial oxygen saturation nadir (84.3% ± 12.3% vs 91.2% ± 4.2%, P < .05), increased peak end-tidal CO(2) (53.4 ± 8.5 mm Hg vs 42.3 ± 5.3 mm Hg, P < .001), and increased arousals (13.7 ± 4.7 events/h vs 10.8 ± 3.8 events/h, P < .05). CONCLUSIONS: Children with SCD have reduced upper airway size due to overgrowth of the surrounding lymphoid tissues, which may explain their predisposition to OSAS.


Subject(s)
Adenoids/pathology , Anemia, Sickle Cell/pathology , Lymphoid Tissue/pathology , Palatine Tonsil/pathology , Adolescent , Anemia, Sickle Cell/complications , Case-Control Studies , Child , Child, Preschool , Female , Humans , Magnetic Resonance Imaging , Male , Polysomnography , Prevalence , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/epidemiology
14.
Sleep Med ; 12(9): 892-7, 2011 Oct.
Article in English | MEDLINE | ID: mdl-21940205

ABSTRACT

OBJECTIVE: To analyze sleep in children with Williams Syndrome (WS) compared to normal healthy controls in order to determine whether particular sleep features are characteristic of WS, and to explore associations between disturbed sleep and behavior. METHODS: Thirty-five children with genetically-confirmed WS and 35 matched controls underwent overnight polysomnography and performance testing in the Sleep Center at the Children's Hospital of Philadelphia. Parents completed questionnaires regarding the subjects' sleep and behavior. RESULTS: WS subjects had significantly different sleep than controls, with decreased sleep efficiency, increased respiratory-related arousals and increased slow wave sleep on overnight polysomnography. WS subjects were also noted to have more difficulty falling asleep, with greater restlessness and more arousals from sleep than controls. Fifty-two percent of WS subjects had features of attention deficit-hyperactivity disorder. CONCLUSION: Children with WS had significantly different sleep than controls in our sample. These differences demonstrated in our study may reflect genetic influences on sleep.


Subject(s)
Sleep Wake Disorders/genetics , Sleep Wake Disorders/physiopathology , Sleep, REM/physiology , Williams Syndrome/genetics , Williams Syndrome/physiopathology , Adolescent , Attention Deficit Disorder with Hyperactivity/genetics , Attention Deficit Disorder with Hyperactivity/physiopathology , Child , Child Behavior , Child, Preschool , Female , Humans , Male , Parents/psychology , Polysomnography , Sleep Wake Disorders/diagnosis , Surveys and Questionnaires
15.
Sleep ; 34(7): 899-908, 2011 Jul 01.
Article in English | MEDLINE | ID: mdl-21731140

ABSTRACT

STUDY OBJECTIVES: To describe the rate, distribution and correlates of periodic limb movements in sleep (PLMS) in children with sickle cell disease (SCD). DESIGN: Prospective, cross-sectional. SETTING: Hospital-based sleep laboratory. PARTICIPANTS: Sixty-four children aged 2-18 years with SCD, hemoglobin SS-type who had an overnight polysomnogram and a parent-completed Pediatric Sleep Questionnaire. Mean age was 8.4 years (SD 4.8); 50% were male. INTERVENTIONS: N/A. MEASUREMENTS AND RESULTS: The mean PLMS index was 3.7 (6.6) and ranged from 0 to 31.8, with 23.4% of the sample having PLMS ≥ 5/h. Sleep efficiency was decreased (P = 0.03), and the total arousal index (P = 0.003) and PLMS arousal index (P < 0.001) were increased in children with PLMS ≥ 5/h compared to those with PLMS < 5/h. PLMS were most frequent in NREM stage 2 sleep and during the fourth hour of sleep. Inter-movement interval duration peaked at 25-30 s. "Growing pains worst in bed" or "restlessness of the legs", suggesting restless legs syndrome (RLS), were reported in 12.5% of the total sample and were more common in children with elevated PLMS. A PLMS score for identifying elevated PLMS in children, based on items from the Pediatric Sleep Questionnaire, did not significantly predict PLMS ≥ 5/h. CONCLUSIONS: Elevated PLMS are common in children with SCD and are associated with sleep disruption and symptoms of RLS. Future research into the time structure of PLMS, their causes and consequences, and development of a disease-specific sleep disorders screening questionnaire, is needed in children with SCD.


Subject(s)
Anemia, Sickle Cell/complications , Restless Legs Syndrome/etiology , Sleep Stages/physiology , Adolescent , Anemia, Sickle Cell/physiopathology , Child , Child, Preschool , Cross-Sectional Studies , Extremities/physiopathology , Female , Humans , Infant , Male , Movement , Polysomnography , Prospective Studies , Restless Legs Syndrome/epidemiology
16.
Behav Sleep Med ; 9(3): 184-93, 2011.
Article in English | MEDLINE | ID: mdl-21722013

ABSTRACT

Actigraphy provides a non-invasive objective means to assess sleep-wake cycles. In young children, parent logs can also be useful for obtaining sleep-wake information. The authors hypothesized that actigraphy and parent logs were both equally valid instruments in healthy preschool-aged children. The authors studied 59 children aged 3 to 5 years in full-time day care. Each child was screened for medical problems and developmental delays before being fitted with an actigraphy watch, which was worn for 1 week. Parents maintained logs of sleep and wakefulness during the same period, with input from day care workers. In general, parents overestimated the amount of nighttime sleep measured by actigraphy by 13% to 22% (all significant). Although there was no difference in sleep onset times, parents reported later rise times on the weekend and fewer nighttime awakenings. There was no significant difference between parent logs and actigraphy with regard to daytime napping. The authors conclude that parent logs are best utilized in assessing daytime sleep and sleep onset, whereas actigraphy should be used to assess nighttime sleep and sleep offset time.


Subject(s)
Actigraphy/methods , Medical Records/statistics & numerical data , Adult , Child, Preschool , Female , Humans , Male , Parents , Sleep , Wakefulness
17.
Sleep ; 34(6): 773-8, 2011 Jun 01.
Article in English | MEDLINE | ID: mdl-21629365

ABSTRACT

STUDY OBJECTIVES: The prevalence of obstructive sleep apnea syndrome (OSAS) in sickle cell disease (SCD) has been reported to be higher than that in the general pediatric population. However, not all subjects with SCD develop OSAS. We hypothesized that SCD patients with OSAS have a blunted neuromuscular response to subatmospheric pressure loads during sleep, making them more likely to develop upper airway collapse. DESIGN: Subjects with SCD with and without OSAS underwent pressure-flow measurements during sleep using intraoral surface electrodes to measure genioglossal EMG (EMGgg). Two techniques were applied to decrease the nasal pressure (P(N)) to subatmospheric levels, resulting in an activated and relatively hypotonic upper airway. The area under the curve of the inspiratory EMGgg moving time average was analyzed. EMGgg activity was expressed as a percentage of baseline. Changes in EMGgg in response to decrements in nasal pressure were expressed as the slope of the EMGgg vs. nasal pressure (slope of EMGgg-P(N)). SETTING: Sleep laboratory. PARTICIPANTS: 4 children with SCD and OSAS and 18 children with SCD but without OSAS. RESULTS: THE MAJOR FINDINGS OF THIS STUDY WERE: (1) using the activated but not the hypotonic technique, the slope of EMGgg-P(N) was more negative in SCD controls than SCD OSAS; (2) the slope of EMGgg-P(N) was significantly lower using the activated technique compared to the hypotonic technique in SCD controls only; (3) similarly, the critical closing pressure, Pcrit, was more negative using the activated technique than the hypotonic technique in SCD controls but not in SCD OSAS. CONCLUSION: This preliminary study has shown that children with SCD but without OSAS have more prominent upper airway reflexes than children with SCD and OSAS.


Subject(s)
Anemia, Sickle Cell/complications , Anemia, Sickle Cell/physiopathology , Sleep Apnea, Obstructive/epidemiology , Adolescent , Case-Control Studies , Child , Electromyography , Female , Humans , Male , Polysomnography , Respiratory Mechanics/physiology , Respiratory Muscles/physiopathology , Respiratory System/physiopathology , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/physiopathology , Tongue/physiopathology
18.
J Clin Sleep Med ; 7(1): 81-7, 2011 Feb 15.
Article in English | MEDLINE | ID: mdl-21344042

ABSTRACT

STUDY OBJECTIVES: The gold-standard test used to diagnose childhood obstructive sleep apnea is polysomnography. However, this test requires an overnight stay at a sleep laboratory and the attachment of multiple sensors to the patient. The long-term impact of this testing on the child and family are not known. We hypothesized that polysomnography does not precipitate acute or chronic psychological effects in children. METHODS: A consecutive cohort of children who had undergone sleep studies 2 to 4 months prior to the interview were administered a standardized questionnaire via telephone. RESULTS: Of the 118 families that were eligible to participate, 67% could be contacted and agreed to participate; 87% of respondents reported the experience to have been satisfactory (mean Likert score of 8.6 ± 2.0 [SD] on a scale of 1-10). Similar levels of satisfaction were reported by parents of children with developmental delay or those who were younger than 3 years. The night's sleep was considered typical in 68% of cases. Sleep was less likely to be typical in children younger than 3 years (47%, p = 0.043). Eight percent of children experienced pain during the study. By caregiver report, of those children who remembered the sleep study, memories were positive in 84%. No child had evidence of serious long-term psychological issues. CONCLUSIONS: The vast majority of children and families found the polysomnography experience to be satisfactory, with no psychological sequelae. However, many children, especially those younger than 3 years, demonstrated sleep patterns different from their usual sleep. The clinical relevance of this finding merits further study. Further research evaluating the generalizability of this study is also needed.


Subject(s)
Monitoring, Physiologic/methods , Polysomnography/methods , Professional-Family Relations , Sleep Apnea Syndromes/diagnosis , Adolescent , Child , Child, Preschool , Female , Hospitalization , Humans , Infant , Interviews as Topic , Linear Models , Male , Parent-Child Relations , Patient Satisfaction , Pediatrics , Sleep Apnea Syndromes/psychology , Statistics, Nonparametric , Surveys and Questionnaires , Young Adult
19.
J Dev Behav Pediatr ; 32(2): 90-7, 2011.
Article in English | MEDLINE | ID: mdl-21217402

ABSTRACT

OBJECTIVE: To determine the relationship between napping and cognitive function in preschool-aged children. METHODS: Daytime napping, nighttime sleep, and cognitive function were assessed in 59 typically developing children aged 3 to 5 years, who were enrolled in full-time childcare. Participants wore an actigraphy watch for 7 days to measure sleep and napping patterns and completed neuropsychological testing emphasizing attention, response control, and vocabulary. Parents of participants completed behavior ratings and sleep logs during the study. Sleep/wake cycles were scored with the Sadeh algorithm. RESULTS: Children who napped more on weekdays were also more likely to nap during weekends. Weekday napping and nighttime sleep were inversely correlated, such that those who napped more slept less at night, although total weekday sleep remained relatively constant. Weekday napping was significantly (negatively) correlated with vocabulary and auditory attention span, and weekday nighttime sleep was positively correlated with vocabulary. Nighttime sleep was also significantly negatively correlated with performance, such that those who slept less at night made more impulsive errors on a computerized go/no-go test. CONCLUSIONS: Daytime napping is actually negatively correlated with neurocognitive function in preschoolers. Nighttime sleep seems to be more critical for development of cognitive performance. Cessation of napping may serve as a developmental milestone of brain maturation. Children who nap less do not appear to be sleep deprived, especially if they compensate with increased nighttime sleep. An alternative explanation is that children who sleep less at night are sleep deprived and require a nap. A randomized trial of nap restriction would be the next step in understanding the relationship between napping and neurocognitive performance.


Subject(s)
Activity Cycles , Cognition , Sleep , Actigraphy , Child Development , Child, Preschool , Female , Humans , Male , Maryland
20.
J Sleep Res ; 19(4): 559-66, 2010 Dec.
Article in English | MEDLINE | ID: mdl-20545836

ABSTRACT

Obesity continues to be a major public health issue. In adolescents, there are limited studies on the relationship between obesity and sleep duration. We found hypothesized that an average sleep duration of <6 h in adolescents was associated with obesity. Data were from the National Longitudinal Study of Adolescent Health (ADD Health); a survey of 90,000 youths, aged 12-18 years; surveyed in several waves. The sample population for our study was 13,568. Weighted multiple logistic regression was used to identify the relationship between obesity at Wave II and sleep duration, having adjusted for skipping breakfast ≥ 2/week; race, gender, parental income, TV ≥ 2 h per day, depression, and obesity at Wave I. At Wave I, the mean age was 15.96 ± 0.11 years; mean sleep hours were 7.91 ± 0.04. At Waves I and II, respectively, 10.6 and 11.2% of adolescents were obese. Adjusted analyses suggest that the effect of shortened sleep duration in Wave I was not significantly predictive of obesity in Wave II (P < 0.218). Longitudinally, depression and TV ≥ 2 h per day at Wave I was associated with a higher risk of obesity at Wave II in adjusted analyses. Depressed adolescents were almost twice as likely to be obese (OR = 1.84, 95% CI = 1.25-2.72); adolescents who watched TV ≥ 2 h per day were 37% more likely to be obese (OR = 1.37, 95% CI = 1.09-1.72). Environmental factors including TV ≥ 2 h per day and depression were significantly associated with obesity; shortened sleep duration was not. Future longitudinal studies in adolescents are needed to determine whether timing of television watching directly influences sleep patterns and, ultimately, obesity.


Subject(s)
Obesity/etiology , Sleep Deprivation/complications , Adolescent , Age Factors , Feeding Behavior/physiology , Female , Humans , Logistic Models , Longitudinal Studies , Male , Motor Activity/physiology , Nutritional Status/physiology , Obesity/physiopathology , Racial Groups , Sex Factors , Sleep/physiology , Sleep Deprivation/physiopathology , United States
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