First name
Stacey
Middle name
L
Last name
Ishman

Title

Associations of actigraphy measures of sleep duration and continuity with executive function, vigilance, and fine motor control in children with snoring and mild sleep-disordered breathing.

Year of Publication

2023

Number of Pages

Date Published

04/2023

ISSN Number

1550-9397

Abstract

STUDY OBJECTIVES: Children with snoring and mild sleep-disordered breathing may be at increased risk for neurocognitive deficits despite few obstructive events. We hypothesized that actigraphy-based sleep duration and continuity associate with neurobehavioral functioning and explored whether these associations vary by demographic and socioeconomic factors.

METHODS: 298 children enrolled in the Pediatric Adenotonsillectomy Trial, aged 3 to 12 years, 47.3% from racial or ethnic minority groups, with habitual snoring and an apnea-hypopnea index <3 were studied with actigraphy (mean 7.5 ± 1.4 days) and completed a computerized vigilance test (Go-No-Go) and a test of fine motor control (9-Hole Pegboard). Caregivers completed the Behavior Rating Inventory of Executive Function (BRIEF). Regression analyses evaluated associations between sleep exposures (24-hour and nocturnal sleep duration, sleep fragmentation index, sleep efficiency) with the BRIEF Global Executive Composite index, pegboard completion time (fine motor control), and vigilance (d prime on the Go-No-Go), adjusting for demographic factors and study design measures.

RESULTS: Longer sleep duration, higher sleep efficiency and lower sleep fragmentation were associated with better executive function; each additional hour of sleep over 24 hours associated with more than a 3-point improvement in executive function (p=0.002). Longer nocturnal sleep (p=0.02) and less sleep fragmentation (p=0.001) were associated with better fine motor control. Stronger associations were observed for boys and children less than six years old.

CONCLUSIONS: Sleep quantity and continuity are associated with neurocognitive functioning in children with mild sleep-disordered breathing, supporting efforts to target these sleep health parameters as part of interventions for reducing neurobehavioral morbidity.

CLINICAL TRIAL: Pediatric Adenotonsillectomy for Snoring (PATS), clinicaltrials.gov.: NCT02562040.

DOI

10.5664/jcsm.10620

Alternate Title

J Clin Sleep Med

PMID

37185231
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Title

Diagnosis and management of Cornelia de Lange syndrome: first international consensus statement.

Year of Publication

2018

Number of Pages

649-666

Date Published

2018 10

ISSN Number

1471-0064

Abstract

<p>Cornelia de Lange syndrome (CdLS) is an archetypical genetic syndrome that is characterized by intellectual disability, well-defined facial features, upper limb anomalies and atypical growth, among numerous other signs and symptoms. It is caused by variants in any one of seven genes, all of which have a structural or regulatory function in the cohesin complex. Although recent advances in next-generation sequencing have improved molecular diagnostics, marked heterogeneity exists in clinical and molecular diagnostic approaches and care practices worldwide. Here, we outline a series of recommendations that document the consensus of a group of international experts on clinical diagnostic criteria, both for classic CdLS and non-classic CdLS phenotypes, molecular investigations, long-term management and care planning.</p>

DOI

10.1038/s41576-018-0031-0

Alternate Title

Nat. Rev. Genet.

PMID

29995837
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