CohortAutism research : official journal of the International Society for Autism Research2022

Evaluation of electroencephalography biomarkers for Angelman syndrome during overnight sleep.

Yuval Levin, Nishitha S Hosamane, Taylor E McNair and 4 others

PMID 35304979

WHAT IT FOUND

Delta brain wave abnormalities in Angelman syndrome are reliably detected during both wake and sleep.

However, the difference is actually greater during wake, so overnight sleep studies are not necessary for tracking this biomarker in clinical trials.

Key findings

01Delta power was increased in Angelman syndrome during both wake and all stages of NREM sleep compared to controls.

02The relative increase in delta power in Angelman syndrome was significantly higher during wake than during sleep, suggesting wake EEGs are sufficient for biomarker detection.

03Sleep spindle impairments, previously seen in short daytime recordings, were difficult to detect reliably during overnight sleep using standard automated methods.

STILL TO COME

How it was doneWhat they found

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What it does not show

Small sample size (12 per group), with only 2 participants per group older than 18 years, limiting conclusions about adult biomarkers. Sleep staging may have been imperfect in AS subjects due to high background delta rhythms confusing technicians. Clinical severity of Angelman syndrome was not assessed, so it is unclear if wake or sleep delta better correlates with symptom severity. All subjects had obstructive sleep apnea, which can affect sleep architecture and spindle detection, though apnea severity did not correlate with spindle findings.

Declared interests

Michael S. Sidorov received consulting fees from Medpace, Inc. for EEG analysis. The study was funded by the Eunice Kennedy Shriver National Institute of Child Health and Human Development.

The easy way to misread this

Do not assume overnight sleep studies are required to monitor Angelman syndrome treatment response via EEG. The study found that the biomarker signal (delta power) is actually stronger and more distinct during wakefulness, making standard wake EEGs sufficient and potentially preferable for clinical trials.

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The study

Participants
36 EEGs (12 per group: Angelman syndrome, neurotypical, Down syndrome)
Certainty of evidence
Low

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    Cite

    Yuval Levin, Nishitha S Hosamane, Taylor E McNair, et al. Evaluation of electroencephalography biomarkers for Angelman syndrome during overnight sleep. Autism research : official journal of the International Society for Autism Research. 2022.

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