Sleep Disordered Breathing in Neurodevelopmental Disorders

Imran, Sehyr; Arganbright, Jill M.; Ingram, David G.

doi:10.1007/s40675-021-00203-4

Cited by 9 publications

(3 citation statements)

References 85 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Similarly, almost one third of individuals with CS were estimated to have laryngeal anomalies. These presentations are worthy of consideration in clinical contexts as laryngeal anomalies and micrognathia are known to increase the burden of respiratory and therefore sleep disordered breathing for example in 22q11.2 deletion [79], Treacher Collins and Nager syndromes [80].…”

Section: Physical Characteristics and Conditionsmentioning

confidence: 99%

Phenotypic characteristics and variability in CHARGE syndrome: a PRISMA compliant systematic review and meta-analysis

Thomas

Waite

Williams

et al. 2022

J Neurodevelop Disord

View full text Add to dashboard Cite

Background CHARGE syndrome (OMIM #214800) is a phenotypically complex genetic condition characterised by multi-system, multi-sensory impairments. Behavioural, psychological, cognitive and sleep difficulties are not well delineated and are likely associated with biopsychosocial factors. Methods This meta-analysis investigated the prevalence of clinical features, physical characteristics and conditions, behavioural, psychological, cognitive and sleep characteristics in CHARGE syndrome, and statistically evaluated directional associations between these characteristics. Pooled prevalence estimates were calculated using reliable, prespecified quality weighting criteria, and meta-regression was conducted to identify associations between characteristics. Results Of the 42 eligible studies, data could be extracted for 1675 participants. Prevalence estimates were highest for developmental delay (84%), intellectual disability (64%), aggressive behaviour (48%), self-injurious behaviour (44%) and sleep difficulties (45%). Meta-regression indicated significant associations between intellectual disability and choanal atresia, intellectual disability and inner ear anomalies, sleep difficulties and growth deficiency, and sleep difficulties and gross motor difficulties. Conclusions Our comprehensive review of clinical features, behavioural, psychological, cognitive and physical characteristics, conditions and comorbidities in CHARGE syndrome provides an empirically based foundation to further research and practice.

show abstract

Section: Physical Characteristics and Conditionsmentioning

confidence: 99%

Phenotypic characteristics and variability in CHARGE syndrome: a PRISMA compliant systematic review and meta-analysis

Thomas

Waite

Williams

et al. 2022

J Neurodevelop Disord

View full text Add to dashboard Cite

show abstract

“…Many features of PWS predispose these children to snoring and obstructive sleep apnea, including obesity, narrow upper airway and micrognathia, low tone of the upper airway, and weak respiratory muscles related to generalized hypotonia. 6 All these features likely contribute to the increased sleep apnea prevalence of around 80%, 7 compared to 2% to 3% in the general population. 8 In addition to the multifactorial etiology of their OSA, children with PWS tend to have disproportionately more associated hypoventilation compared to neurotypical children.…”

Section: Sleep-related Breathing Disordersmentioning

confidence: 99%

Sleep Disorders in Children with Prader Willi Syndrome: Current Perspectives

Ingram¹,

Arganbright²,

Paprocki³

et al. 2022

NSS

View full text Add to dashboard Cite

Children with Prader-Willi syndrome (PWS) face a multitude of potential health challenges including life-threatening obesity, endocrinopathies, behavioral and emotional dysregulation, developmental delays, and sleep disorders. In the current perspective piece, we provide a focused review of the condition's etiology and clinical findings, as well as a more in-depth discussion of sleep disorders frequently associated with PWS. In particular, we highlight and discuss difficult clinical scenarios frequently encountered by the pediatric sleep physician caring for this patient population, including diagnosis and treatment of complex sleep-related breathing disorders, considerations for sleep apnea surgery, the interplay between growth hormone and sleep apnea, diagnostic challenges in hypersomnia/narcolepsy, and current and emerging therapies for hypersomnia/narcolepsy. Overall, although there are many areas that need further research, sleep disorders remain a fruitful target for improving quality of life of children with PWS and their families.

show abstract

“…It is worth noting that the initial weeks of rhGH treatment may lead to worsened sleep-related breathing disorders (SRBDs) and adenoid hypertrophy, possibly due to high levels of insulin-like growth factor 1 (IGF-1) after starting rhGH [ 8 ]. SRBDs are common symptoms of PWS, and they can contribute to poor physical health, neurocognitive function, and prognosis due to obesity, narrowed upper airways, reduced saliva excretion, adenoid/tonsillar hypertrophy, hypotonic breathing muscles, or scoliosis [ 9 ].…”

Section: Introductionmentioning

confidence: 99%

Evaluating the effect of recombinant human growth hormone treatment on sleep-related breathing disorders in toddlers with Prader–Willi syndrome: a one-year retrospective cohort study

Guo,

Fu,

Zhou

et al. 2024

BMC Pediatr

View full text Add to dashboard Cite

Background Recombinant human growth hormone (rhGH) therapy is beneficial for children with Prader–Willi syndrome (PWS) in improving short stature and metabolism, but the effect of early rhGH treatment on respiratory and sleep parameters for PWS children under three years old remains elusive. Thus, this study aimed to investigate the impact of rhGH treatment on sleep-related breathing disorders (SRBDs) for toddlers with PWS. Methods A total of 17 age-matched PWS patients receiving rhGH treatment (rhGH group) and 17 control individuals not receiving rhGH treatment (non-rhGH group) were recruited for this study between October 2018 and January 2023. Data related to polysomnography-polygraphy (PSG) and serum levels of insulin-like growth factor (IGF-1) and insulin-like growth factor binding protein 3 (IGFBP-3) were collected. Results The mean age in the rhGH group was 20.76 ± 9.22 months, which was comparable to that of the non-rhGH group (25.23 ± 13.81 months). The demographic and anthropometric parameters were similar across the two groups after 52 weeks of treatment. Administration of rhGH to toddlers did not exert adverse effects on the obstructive apnea–hypopnea index (OAHI), central apnea index (CAI), oxygen desaturation index (ODI), mean percutaneous oxygen saturation (SpO2), lowest SpO2, duration when SpO2 is lower than 90%, or proportion of the patients with SpO2 lower than 90%. Furthermore, the increased IGF-1 z-score and IGFBP-3 level did not worsen SRBDs. Conclusion Treatment with rhGH for 52 weeks on young toddlers with PWS showed no deleterious effects on SRBDs. This shed more light on the importance of initiating rhGH therapy early in PWS patients.

show abstract

Sleep Disordered Breathing in Neurodevelopmental Disorders

Cited by 9 publications

References 85 publications

Phenotypic characteristics and variability in CHARGE syndrome: a PRISMA compliant systematic review and meta-analysis

Phenotypic characteristics and variability in CHARGE syndrome: a PRISMA compliant systematic review and meta-analysis

Sleep Disorders in Children with Prader Willi Syndrome: Current Perspectives

Evaluating the effect of recombinant human growth hormone treatment on sleep-related breathing disorders in toddlers with Prader–Willi syndrome: a one-year retrospective cohort study

Contact Info

Product

Resources

About