The Role of Functional Respiratory Imaging in Treatment Selection of Children With Obstructive Sleep Apnea and Down Syndrome

Slaats, M.; Loterman, Dieter; Holsbeke, Cedric Van; Vos, Wim; Hoorenbeeck, Kim Van; Backer, Jan De; Backer, Wilfried De; Wojciechowski, Marek; Boudewyns, An; Verhulst, Stijn

doi:10.5664/jcsm.7064

Cited by 12 publications

(11 citation statements)

References 35 publications

(51 reference statements)

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“…CFD simulation has been used experimentally to describe pulmonary airflow dynamics and cardiovascular hemodynamics 26 . Some groups endeavor to apply CFD analysis to virtually predict and assess surgical treatment outcomes in OSA patients or DS patients with OSA 14,[27][28][29] . Although this technique is still far from clinical practice due to large individual variability, the use of CFD analysis in well-validated Dp16 mice provides valuable insights in the airway structure and the site of obstruction by computing 3D modeling and flow parameters such as velocity and airflow pressure.…”

Section: Discussionmentioning

confidence: 99%

“…Previous reports have demonstrated its advantages over anatomical evaluation alone 8 – 10 . CFD has also been utilized not only to predict the effect of OSA treatments in general populations 11 – 13 , but also to tailor treatment strategies for DS individuals with OSA 14 . Although CFD has the potential to visualize and stratify multilevel narrowing of the upper airway, CFD has not been used for this purpose in mice.…”

Section: Introductionmentioning

confidence: 99%

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Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome

Takahashi

Sakai

Iwasaki

et al. 2020

Sci Rep

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A high prevalence of obstructive sleep apnea (OSA) has been reported in Down syndrome (DS) owing to the coexistence of multiple predisposing factors related to its genetic abnormality, posing a challenge for the management of OSA. We hypothesized that DS mice recapitulate craniofacial abnormalities and upper airway obstruction of human DS and can serve as an experimental platform for OSA research. This study, thus, aimed to quantitatively characterize the upper airway as well as craniofacial abnormalities in Dp(16)1Yey (Dp16) mice. Dp16 mice demonstrated craniofacial hypoplasia, especially in the ventral part of the skull and the mandible, and rostrally positioned hyoid. These changes were accompanied with a shorter length and smaller cross-sectional area of the upper airway, resulting in a significantly reduced upper airway volume in Dp16 mice. Our non-invasive approach, a combination of computational fluid dynamics and high-resolution micro-CT imaging, revealed a higher negative pressure inside the airway of Dp16 mice compared to wild-type littermates, showing the potential risk of upper airway collapse. Our study indicated that Dp16 mice can be a useful model to examine the pathophysiology of increased upper airway collapsibility of DS and to evaluate the efficacy of therapeutic interventions for breathing and sleep anomalies.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome

Takahashi

Sakai

Iwasaki

et al. 2020

Sci Rep

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“…Zaffanello M. et al [61] reported other genetic syndromes that could be cause of OSAS: Achondroplasia [62,63,64,65], Ehlers–Danlos Syndrome [66,67], Pierre Robin sequence/complex [68], Ellis–van Creveld Syndrome [69], sickle cell disease [70] and Noonan Syndrome [71]. Any congenital or acquired conditions that involve the respiratory control center may potentially lead to the development of OSAS.…”

Section: Risk Factorsmentioning

confidence: 99%

Risk Factors for Obstructive Sleep Apnea Syndrome in Children: State of the Art

Gulotta

Iannella

Vicini

et al. 2019

IJERPH

142

135

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The obstructive sleep apnea syndrome (OSAS) represents only part of a large group of pathologies of variable entity called respiratory sleep disorders (RSD) which include simple snoring and increased upper airway resistance syndrome (UARS). Although the etiopathogenesis of adult OSAS is well known, many aspects of this syndrome in children are still debated. Its prevalence is about 2% in children from 2 to 8 years of age, mostly related to the size of the upper airways adenoid tissue. Several risk factors linked to the development of OSAS are typical of the pediatric age. The object of this paper is to analyze the state of the art on this specific topic, discussing its implications in terms of diagnosis and management.

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“…The minimal upper airway cross-sectional area, measured with CT scan, was smaller in DS children with severe OSAS (aged 4.3±2.3 years). Children with a less favourable response to A&T had a smaller volume of regions below the tonsils, due to enlargement of the lingual tonsils, glossoptosis, or macroglossia (15).…”

Section: Down Syndromementioning

confidence: 99%

Sleep-disordered breathing in paediatric setting: existing and upcoming of the genetic disorders

Zaffanello¹,

Antoniazzi²,

Tenero³

et al. 2018

Ann. Transl. Med.

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Childhood obstructive sleep apnea syndrome (OSAS) is characterized by anatomical and functional upper airway abnormalities as pathophysiological determinants, and clinical symptoms are frequently clear. OSAS is widely described in rare genetic disorders, such as achondroplasia, Down syndrome, Prader-Willi syndrome, Pierre Robin sequence, and mucopolysaccharidosis. Craniofacial and upper airway involvement is frequently morbid conditions. In children with genetic diseases, the clinical symptoms of OSAS are often slight or absent, and related morbidities are usually more severe and can be observed at any age. The present review is aimed to updating the discoveries regarding OSAS on Achondroplasia, Down syndrome, Prader-Willi syndrome, Pierre Robin sequence, Sickle cell disease, or encountered in our clinical practice (Ehlers-Danlos syndrome, Ellis-van Creveld syndrome, Noonan syndrome). Two additional groups of genetic disorders will be focused (mucopolysaccharidoses and osteogenesis imperfecta). The flowing items are covered for each disease: (I) what is the pathophysiology of OSAS? (II) What is the incidence/prevalence of OSAS? (III) What result from the management and prognosis? (IV) What are the recommendations? Considering the worries of OSAS, such as inattention and behavioural problems, daytime sleepiness, failure to thrive, cardiological and metabolic complications, the benefit of a widespread screening and the treatment in children with genetic diseases is undoubtful. The goals of the further efforts can be the inclusion of various genetic diseases into guidelines for the screening of OSAS, updating the shreds of evidence based on the research progression.

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The Role of Functional Respiratory Imaging in Treatment Selection of Children With Obstructive Sleep Apnea and Down Syndrome

Abstract: A commentary on this article appears in this issue on page 501.

Cited by 12 publications

References 35 publications

Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome

Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome

Risk Factors for Obstructive Sleep Apnea Syndrome in Children: State of the Art

Sleep-disordered breathing in paediatric setting: existing and upcoming of the genetic disorders

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