Sleep related rhythmic movement disorder: phenotypic characteristics and treatment response in a paediatric cohort
Sleep related rhythmic movement disorder: phenotypic characteristics and treatment response in a paediatric cohort
Objective: to describe phenotypic, polysomnographic characteristics, impact, and treatment response in children with sleep related rhythmic movement disorder (SR-RMD).
Background: there is limited research on SR-RMD. We have developed a systematic clinical evaluation of children with SR-RMD to improve understanding and treatment.
Methods: a retrospective chart review of 66 children at a UK tertiary hospital. Baseline assessment included validated screening questionnaires to study autism spectrum characteristics, general behaviour and sensory profile. A standardised questionnaire assessed impact on sleep quality and daytime wellbeing of child and family. Polysomnography data were collated.
Results: children were aged 0.9–16.3 years (78.8% male). 51.5% had a neurodevelopmental disorder, most commonly autism spectrum disorder. High rates of behavioural disturbance and sensory processing differences were reported, not confined to children with neurodevelopmental disorders. Parents reported concerns about risk of injury, loss of sleep and persistence into adulthood. Daytime wellbeing was affected in 72% of children and 75% of other family members. Only 31/48 children demonstrated rhythmic movements during video-polysomnography, occupying on average 6.1% of time in bed. Most clusters occurred in the settling period but also arose from N1, N2 and REM sleep and wake after sleep onset. Melatonin was prescribed to 52 children, all but one were extended-release preparations. 24/27 children with available data were reported to improve with melatonin.
Conclusions: SR-RMD places a significant burden on child and family wellbeing. Our novel findings of sensory processing differences in this population and parent reported therapeutic response to extended-release melatonin offer potential avenues for future research.
Autism spectrum disorder, Jactatio capitis nocturna, Melatonin, Neurodevelopmental disorder, Sensory processing, Sleep-related rhythmic movement disorder, Melatonin/therapeutic use, Humans, Autism Spectrum Disorder, Male, Sleep Wake Disorders/diagnosis, Movement Disorders, Parasomnias/drug therapy, Sleep, Female, Retrospective Studies, Child
21-29
Joels, H.
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Benny, A.
0748b59c-808e-4d43-b790-6e10ac456cd1
Sharpe, A.
5af2d9d0-265b-4b05-83f8-e34c8297606b
Postigo, B.
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Joseph, B.
ede92259-7a24-4f8d-b2b2-964675db9e5b
Piantino, C.
e6d278b0-ad2a-44f3-9d8b-a0e9313bb523
Marshall, A
5014c604-6673-4691-adc6-68b5ffc94e47
Hewertson, V.
7852f13b-5e38-4b36-8a96-080523c6ab75
Hill, C.M.
867cd0a0-dabc-4152-b4bf-8e9fbc0edf8d
5 October 2023
Joels, H.
2a00040e-65af-4ef9-a89a-774af9cd15d4
Benny, A.
0748b59c-808e-4d43-b790-6e10ac456cd1
Sharpe, A.
5af2d9d0-265b-4b05-83f8-e34c8297606b
Postigo, B.
19e6b780-601e-4bfc-b2cc-493f8d3572c9
Joseph, B.
ede92259-7a24-4f8d-b2b2-964675db9e5b
Piantino, C.
e6d278b0-ad2a-44f3-9d8b-a0e9313bb523
Marshall, A
5014c604-6673-4691-adc6-68b5ffc94e47
Hewertson, V.
7852f13b-5e38-4b36-8a96-080523c6ab75
Hill, C.M.
867cd0a0-dabc-4152-b4bf-8e9fbc0edf8d
Joels, H., Benny, A., Sharpe, A., Postigo, B., Joseph, B., Piantino, C., Marshall, A, Hewertson, V. and Hill, C.M.
(2023)
Sleep related rhythmic movement disorder: phenotypic characteristics and treatment response in a paediatric cohort.
Sleep Medicine, 112, .
(doi:10.1016/j.sleep.2023.09.020).
Abstract
Objective: to describe phenotypic, polysomnographic characteristics, impact, and treatment response in children with sleep related rhythmic movement disorder (SR-RMD).
Background: there is limited research on SR-RMD. We have developed a systematic clinical evaluation of children with SR-RMD to improve understanding and treatment.
Methods: a retrospective chart review of 66 children at a UK tertiary hospital. Baseline assessment included validated screening questionnaires to study autism spectrum characteristics, general behaviour and sensory profile. A standardised questionnaire assessed impact on sleep quality and daytime wellbeing of child and family. Polysomnography data were collated.
Results: children were aged 0.9–16.3 years (78.8% male). 51.5% had a neurodevelopmental disorder, most commonly autism spectrum disorder. High rates of behavioural disturbance and sensory processing differences were reported, not confined to children with neurodevelopmental disorders. Parents reported concerns about risk of injury, loss of sleep and persistence into adulthood. Daytime wellbeing was affected in 72% of children and 75% of other family members. Only 31/48 children demonstrated rhythmic movements during video-polysomnography, occupying on average 6.1% of time in bed. Most clusters occurred in the settling period but also arose from N1, N2 and REM sleep and wake after sleep onset. Melatonin was prescribed to 52 children, all but one were extended-release preparations. 24/27 children with available data were reported to improve with melatonin.
Conclusions: SR-RMD places a significant burden on child and family wellbeing. Our novel findings of sensory processing differences in this population and parent reported therapeutic response to extended-release melatonin offer potential avenues for future research.
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Accepted/In Press date: 17 September 2023
e-pub ahead of print date: 20 September 2023
Published date: 5 October 2023
Keywords:
Autism spectrum disorder, Jactatio capitis nocturna, Melatonin, Neurodevelopmental disorder, Sensory processing, Sleep-related rhythmic movement disorder, Melatonin/therapeutic use, Humans, Autism Spectrum Disorder, Male, Sleep Wake Disorders/diagnosis, Movement Disorders, Parasomnias/drug therapy, Sleep, Female, Retrospective Studies, Child
Identifiers
Local EPrints ID: 485641
URI: http://eprints.soton.ac.uk/id/eprint/485641
ISSN: 1389-9457
PURE UUID: 50605a90-b281-45b2-a732-77e1f8419271
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Date deposited: 13 Dec 2023 17:33
Last modified: 18 Mar 2024 02:50
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Contributors
Author:
H. Joels
Author:
A. Benny
Author:
A. Sharpe
Author:
B. Postigo
Author:
B. Joseph
Author:
C. Piantino
Author:
A Marshall
Author:
V. Hewertson
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