Comparison of Frequency and Severity of Sleep-Related Breathing Disorders in Children with Simple Obesity and Paediatric Patients with Prader–Willi Syndrome

Lecka-Ambroziak, Agnieszka; Wysocka‐Mincewicz, Marta; Świercz, Anna; Jedrzejczak, M; Szalecki, Mieczysław

doi:10.3390/jpm11020141

Cited by 7 publications

(5 citation statements)

References 31 publications

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“…The higher risk of central sleep apnea in individuals with PWS is associated with hypothalamic dysfunction [ 21 ]. However, several other factors, such as obesity, hypotonia, scoliosis, narrowing of the upper airways, reduction in saliva excretion, and adenoid/tonsillar hypertrophy, can also lead to obstructive sleep apnea (OSA) [ 22 ]. Sleep apnea is associated with increased sympathetic activation, vagal withdrawal, altered hemodynamic loading conditions, and hypoxemia.…”

Section: Discussionmentioning

confidence: 99%

Endocrine and Metabolic Illnesses in Young Adults with Prader–Willi Syndrome

Noh

Kim

et al. 2022

JPM

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Prader–Willi syndrome (PWS) is a rare genetic disorder characterized by an insatiable appetite that leads to morbid obesity. Previous studies reported health problems in adults with PWS. However, studies on younger adults are lacking, and there are no specific studies of endocrine and metabolic illness in this age group. We performed a retrospective cohort study of 68 individuals with PWS aged 19 to 34 years at Samsung Medical Center. The prevalence of endocrine and metabolic illnesses were compared with those in an age-, sex-, and BMI-matched healthy control group. Young adults with PWS had a higher prevalence of metabolic syndrome (35.3% vs. 4.4%), type 2 diabetes mellitus (50.0% vs. 5.4%), hypertension (30.8% vs. 16.1%), dyslipidemia (38.2% vs. 14.7%), decreased bone density (26.4% vs. 0.9%), and sleep apnea (32.3% vs. 4.4%) than controls (all p < 0.05). The PWS group that maintained recombinant human growth (rhGH) treatment in adulthood had a lower probability of having a BMI ≥ 30 at the last follow-up (odds ratio = 0.106 (0.012–0.948), p = 0.045). Endocrine and metabolic illnesses in individuals with PWS may have already started in the early teens; therefore, appropriate screening and early intervention are important. Better understanding of the natural history of PWS and age-related complications will lead to better-quality medical care for individuals with PWS.

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Section: Discussionmentioning

confidence: 99%

Endocrine and Metabolic Illnesses in Young Adults with Prader–Willi Syndrome

Noh

Kim

et al. 2022

JPM

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“…Meinhardt et al [39] recorded three cases of sleep apnea during long-term GH treatment, with no other serious adverse events reported. Lecka-Ambroziak et al [40] stated that rhGH treatment can influence the severity of SDB in children with simple obesity and PWS. The mortality risk appears to be higher in children with PWS on GH treatment, especially in the first 9 months of therapy [41].…”

Section: Resultsmentioning

confidence: 99%

The Impact of Growth Hormone Therapy on Sleep-Related Health Outcomes in Children with Prader–Willi Syndrome: A Review and Clinical Analysis

Zaffanello,

Pietrobelli,

Piacentini

et al. 2023

JCM

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This literature review of growth hormone (GH) therapy and sleep-related health outcomes in children diagnosed with Prader–Willi syndrome (PWS) assembles evidence for the consequences of sleep deprivation and poor sleep quality: difficulty concentrating and learning at school, behavioral problems, diminished quality of life, and growth impairment. Sleep-disordered breathing (SDB) is another factor that impacts a child’s well-being. We searched the electronic databases Medline PubMed Advanced Search Builder, Scopus, and Web of Science using MeSH terms and text words to retrieve articles on GH deficiency, recombinant human growth hormone (rhGH) therapy, sleep quality, SDB, and PWS in children. The censor date was April 2023. The initial search yielded 351 articles, 23 of which were analyzed for this review. The study findings suggest that while GH may have a role in regulating sleep, the relationship between GH treatment and sleep in patients with PWS is complex and influenced by GH dosage, patient age, and type and severity of respiratory disorders, among other factors. GH therapy can improve lung function, linear growth, and body composition in children with PWS; however, it can also trigger or worsen obstructive sleep apnea or hypoventilation in some. Long-term GH therapy may contribute to adenotonsillar hypertrophy and exacerbate sleep apnea in children with PWS. Finally, GH therapy can improve sleep quality in some patients but it can also cause or worsen SDB in others, leading to diminished sleep quality and overall quality of life. The current evidence suggests that the initial risk of worsening SDB may improve with long-term therapy. In conclusion, rhGH is the standard for managing patients with PWS. Nonetheless, its impact on respiratory function during sleep needs to be thoroughly evaluated. Polysomnography is advisable to assess the need for adenotonsillectomy before initiating rhGH therapy. Close monitoring of sleep disorders in patients with PWS receiving GH therapy is essential to ensure effective and safe treatment.

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“…showed a significant increase in the occurrence of OAHI for up to 4 years after rhGH onset, and the proportion of patients with an OAHI > 1 increased from 3 to 22, 36 and 38 at the 6 weeks, 2 years, and 4 years after the rhGH treatment, respectively [ 12 ], but the authors did explore the effect of hypopnea on the OAHI. A recent study has provided evidence indicating that there is a correlation between the aggravation of SRBDs and the progressive rise in insulin resistance among children with simple obesity and patients with PWS treated with rhGH [ 26 ].…”

Section: Discussionmentioning

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

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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.

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Comparison of Frequency and Severity of Sleep-Related Breathing Disorders in Children with Simple Obesity and Paediatric Patients with Prader–Willi Syndrome

Cited by 7 publications

References 31 publications

Endocrine and Metabolic Illnesses in Young Adults with Prader–Willi Syndrome

Endocrine and Metabolic Illnesses in Young Adults with Prader–Willi Syndrome

The Impact of Growth Hormone Therapy on Sleep-Related Health Outcomes in Children with Prader–Willi Syndrome: A Review and Clinical Analysis

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

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