Association between Reported Sleep Disorders and Behavioral Issues in Children with Myotonic Dystrophy Type 1—Results from a Retrospective Analysis in Italy Article Swipe
Federica Trucco
,
Andrea Lizio
,
Elisabetta Roma
,
Alessandra Di Bari
,
Francesca Salmin
,
Emilio Albamonte
,
Jacopo Casiraghi
,
Susanna Pozzi
,
Stefano Becchiati
,
Laura Antonaci
,
Anna Salvalaggio
,
Michela Catteruccia
,
Michele Tosi
,
Gemma Marinella
,
Federica Rachele Danti
,
F. Bruschi
,
Marco Veneruso
,
Stefano Parravicini
,
Chiara Fiorillo
,
Angela Berardinelli
,
Antonella Pini
,
Isabella Moroni
,
Guja Astrea
,
Roberta Battini
,
Adele D’Amico
,
Federica Ricci
,
Marika Pane
,
Eugenio Mercuri
,
Nicholas E. Johnson
,
Valeria Sansone
·
YOU?
·
· 2024
· Open Access
·
· DOI: https://doi.org/10.3390/jcm13185459
YOU?
·
· 2024
· Open Access
·
· DOI: https://doi.org/10.3390/jcm13185459
Background: Sleep disorders have been poorly described in congenital (CDM) and childhood (ChDM) myotonic dystrophy despite being highly burdensome. The aims of this study were to explore sleep disorders in a cohort of Italian CDM and ChDM and to assess their association with motor and respiratory function and disease-specific cognitive and behavioral assessments. Methods: This was an observational multicenter study. Reported sleep quality was assessed using the Pediatric Daytime Sleepiness Scale (PDSS) and Pediatric Sleep Questionnaire (PSQ). Sleep quality was correlated to motor function (6 min walk test, 6MWT and grip strength; pulmonary function (predicted Forced Vital Capacity%, FVC% pred.); executive function assessed by BRIEF-2; autism traits assessed by Autism Spectrum Screening Questionnaire (ASSQ) and Repetitive Behavior Scale-revised (RBS-R); Quality of life (PedsQL) and disease burden (Congenital Childhood Myotonic Dystrophy Health Index, CCMDHI). Results: Forty-six patients were included, 33 CDM and 13 ChDM, at a median age of 10.4 and 15.1 years. Daytime sleepiness and disrupted sleep were reported by 30% children, in both subgroups of CDM and ChDM. Daytime sleepiness correlated with autism traits in CDM (p < 0.05). Disrupted sleep correlated with poorer executive function (p = 0.04) and higher disease burden (p = 0.03). Conclusions: Sleep issues are a feature of both CDM and ChDM. They correlate with behavioral issues and impact on disease burden.
Related Topics
Concepts
Medicine
Myotonic dystrophy
Autism spectrum disorder
Excessive daytime sleepiness
Autism
Pediatrics
Sleep disorder
Physical therapy
Psychiatry
Cognition
Internal medicine
Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.3390/jcm13185459
- OA Status
- gold
- Cited By
- 6
- References
- 62
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4402774373
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
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https://openalex.org/W4402774373Canonical identifier for this work in OpenAlex
- DOI
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https://doi.org/10.3390/jcm13185459Digital Object Identifier
- Title
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Association between Reported Sleep Disorders and Behavioral Issues in Children with Myotonic Dystrophy Type 1—Results from a Retrospective Analysis in ItalyWork title
- Type
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articleOpenAlex work type
- Language
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enPrimary language
- Publication year
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2024Year of publication
- Publication date
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2024-09-14Full publication date if available
- Authors
-
Federica Trucco, Andrea Lizio, Elisabetta Roma, Alessandra Di Bari, Francesca Salmin, Emilio Albamonte, Jacopo Casiraghi, Susanna Pozzi, Stefano Becchiati, Laura Antonaci, Anna Salvalaggio, Michela Catteruccia, Michele Tosi, Gemma Marinella, Federica Rachele Danti, F. Bruschi, Marco Veneruso, Stefano Parravicini, Chiara Fiorillo, Angela Berardinelli, Antonella Pini, Isabella Moroni, Guja Astrea, Roberta Battini, Adele D’Amico, Federica Ricci, Marika Pane, Eugenio Mercuri, Nicholas E. Johnson, Valeria SansoneList of authors in order
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https://doi.org/10.3390/jcm13185459Publisher landing page
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YesWhether a free full text is available
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goldOpen access status per OpenAlex
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https://doi.org/10.3390/jcm13185459Direct OA link when available
- Concepts
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Medicine, Myotonic dystrophy, Autism spectrum disorder, Excessive daytime sleepiness, Autism, Pediatrics, Sleep disorder, Physical therapy, Psychiatry, Cognition, Internal medicineTop concepts (fields/topics) attached by OpenAlex
- Cited by
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6Total citation count in OpenAlex
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2025: 6Per-year citation counts (last 5 years)
- References (count)
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62Number of works referenced by this work
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10Other works algorithmically related by OpenAlex
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| cited_by_percentile_year.min | 98 |
| countries_distinct_count | 2 |
| institutions_distinct_count | 30 |
| sustainable_development_goals[0].id | https://metadata.un.org/sdg/1 |
| sustainable_development_goals[0].score | 0.7799999713897705 |
| sustainable_development_goals[0].display_name | No poverty |
| citation_normalized_percentile.value | 0.88024818 |
| citation_normalized_percentile.is_in_top_1_percent | False |
| citation_normalized_percentile.is_in_top_10_percent | True |