Craniofacial morphology in pediatric patients with persistent obstructive sleep apnea with or without positive airway pressure therapy: a cross-sectional cephalometric comparison with controls.

INTRODUCTION: Compression on the midface with nasal mask-delivered positive airway pressure (PAP) therapy in growing patients might contribute to midface retrusion. The objective of this study was to investigate the association between long-term PAP use and craniofacial morphologic pattern in children with persistent obstructive sleep apnea. METHODS: Images generated with cone-beam volumetric imaging were used to complete lateral cephalometric analyses of anteroposterior projection of the midface region. The study group included 12 subjects (10 boys, 2 girls; mean age, 9.0 years) who used PAP therapy for at least 6 months and at least 6 hours per night. Measurements from this group were compared with those of a control group of 11 subjects (5 boys, 6 girls; mean age, 9.6 years) with obstructive sleep apnea who did not have PAP. Measurements were taken at 1 time point. RESULTS: No significant differences were identified between the groups for any cephalometric variable. Multivariate linear regression analysis also did not identify a significant association between the number of months of PAP therapy and the cephalometric variables. Cephalometric data for both groups were pooled for comparison with appropriate published normative values for age and sex. Anterior cranial base length, overall anteroposterior length of the maxillary base, and mandibular body length were significantly shorter than normal in the subjects compared with published normative values. CONCLUSIONS: No association was demonstrated between midface projection and PAP use in growing patients. When compared with normative data for anterior cranial base, children with obstructive sleep apnea had shorter maxillary and mandibular lengths.

Characterization of craniofacial-based clinical phenotypes in children with suspected obstructive sleep apnea.

STUDY OBJECTIVES: We conducted this study to identify phenotypes of obstructive sleep apnea (OSA) in children based on lifestyle, sleep habits, age, obesity, sex, soft tissue facial features, and specific craniofacial abnormalities. METHODS: Seventy-three children with symptoms of pediatric OSA who underwent overnight observed polysomnography participated in this study. Soft tissue facial features were assessed using a 3-dimensional stereophotogrammetric system. Craniofacial abnormalities were evaluated based on the most common facial features associated with orthodontic treatment needs. Data regarding lifestyle, sleep habits, age, obesity, and sex were also collected. To identify phenotypes of OSA, a sequential analysis was then performed on categories of variables using fuzzy clustering with medoids. RESULTS: Craniofacial abnormalities and soft tissue facial features defined clusters. Three clusters were identified. Cluster 1 comprised a group of younger children (5.9 ± 3.8 years) without obesity, without craniofacial abnormalities, and with smaller soft tissue facial features dimensions. Cluster 2 comprised a group of older children (9.6 ± 3.9 years) without obesity and with larger mandibular dimensions and mildly arched palates (71.4%). Cluster 3 comprised a group of older children (9.2 ± 3.9 years) with obesity and a history of health issues (68.4%), excessive lower facial height (63.2%), and midface deficiency (73.7%). No differences were observed across clusters regarding sleep features. A moderate severity of obstructive and mixed respiratory events was observed in all 3 clusters. CONCLUSIONS: The study results did not identify distinct phenotypes of pediatric OSA based on soft tissue facial features or craniofacial abnormalities alone. Age and body mass index likely modify the effect of soft tissue facial features and craniofacial abnormalities as risk factors for OSA in children. CITATION: Fernandes Fagundes NC, Loliencar P, MacLean JE, Flores-Mir C, Heo G. Characterization of craniofacial-based clinical phenotypes in children with suspected obstructive sleep apnea. J Clin Sleep Med. 2023;19(11):1857-1865.

The scope of sleep problems in Canadian children and adolescents with obesity.

OBJECTIVE: To determine the scope of sleep concerns, clinical features, and polysomnography (PSG) results and to identify factors that predict obstructive sleep apnea (OSA) in a cohort of children with obesity. METHODS: The study was a multicenter retrospective chart review. Data were collected from three pediatric sleep laboratories over a two year period for all children of age 8-16 years with a body mass index [BMI] ≥95th centile who were undergoing PSG. Data sources included clinical charts and PSG results. Clinical and PSG factors were examined as predictors of OSA. RESULTS: A total of 210 children met inclusion criteria, and 205 had sufficient data for analysis. The mean age was 12.5 ± 2.7 years; and 65% were male. Multiple sleep concerns and comorbidities were reported in most children (90% and 91%, respectively). OSA was identified by PSG in 44% of children; and 28% of children demonstrated moderate/severe OSA. Mouth breathing/nasal congestion (odds ratio [OR] = 0.33, 95% confidence interval [CI] = 0.18-0.61), syndrome/multiple anomalies (OR = 2.4, 95% CI = 1.22-4.93), and family history of OSA (OR = 2.7, 95% CI = 1.2-5.8) or sleep problems (OR = 12.4, 95% CI = 1.5, 99.6) were the only factors predictive of OSA. Oxygen desaturation index <6 events/h measured by PSG showed an OR of 4.96 (95% CI = 2.27-10.86) for the absence of OSA. CONCLUSIONS: Children with obesity who undergo PSG are medically complex with multiple sleep concerns including a high burden of daytime symptoms; slightly less than half of children demonstrate polysomnographic features of OSA. Earlier identification of OSA, recognition of non-OSA sleep concerns, and treatment strategies to improve sleep may contribute to overall health outcomes for children with obesity.

Surgical versus nonsurgical interventions to relieve upper airway obstruction in children with Pierre Robin sequence.

BACKGROUND: Newborns with Pierre Robin sequence (PRS) often experience chronic intermittent hypoxemia/hypoventilation associated with airway obstruction. The heterogeneity of the severity of upper airway obstruction makes management a challenge; the optimal intervention in individual cases is not clear. OBJECTIVE: To investigate the prevalence of surgical/nonsurgical interventions for PRS at two children's hospitals. Patient characteristics and outcomes were examined. METHODS: The present retrospective chart review identified 139 patients with PRS born between 2000 and 2010. Demographic information, mode of airway management, associated anomalies and syndromes, polysomnography results, length of intensive care unit and hospital stay, complications and deaths were extracted. RESULTS: Interventions included prone positioning (alone [61%]), tongue-lip adhesion (45%), nasopharyngeal intubation (28%), continuous positive airway pressure (20%), tracheostomy (19%) and mandibular distraction osteogenesis (5%). Tracheostomies were more prevalent in syndromic patients (P=0.03). Patients who underwent tracheostomy had a lower birth weight (P=0.03) compared with newborns with other interventions. Patients who underwent surgical interventions had longer intensive care unit stays (P<0.001). No intervention was associated with a statistically significant likelihood of requiring a subsequent intervention. Thirty percent of patients underwent polysomnography, with a higher proportion of these using continuous positive airway pressure (n=15) (P<0.01). CONCLUSIONS: In the present descriptive study, patients with syndromic PRS or low birth weight underwent early intervention, which included a tracheostomy. Objective measures of airway obstruction were underutilized. Decision making regarding evaluation and management of upper airway obstruction in this population remains clinician and resource dependent. Reporting data obtained from a large cohort of PRS patients is important to compare experiences and motivate future studies investigating this complex condition.

Developing an index for the orthodontic treatment need in paediatric patients with obstructive sleep apnoea: a protocol for a novel communication tool between physicians and orthodontists.

INTRODUCTION: Sleep disordered breathing in the paediatric population can manifest as an array of different systemic symptoms; among them is a distinct malocclusion and craniofacial phenotype. Emerging research suggests that the treatment of this malocclusion and/or craniofacial phenotype through orthodontic intervention may help with the symptoms of these patients. Selecting the patients who would benefit from orthodontic treatment can be a difficult task for the physician with minimal dental training. Therefore the aim of this study is to develop a simple index to be used by medical professionals to identify those paediatric patients with orthodontic treatment needs who may benefit their obstructive sleep apnoea (OSA) symptoms. METHODS AND ANALYSIS: The methodology in this project has been devised through the WHO's recommendations on developing an index, with modifications based on the specific needs of this study. Based on the available literature, a draft index will be produced and subjected to multiple iterative revisions based on the feedback from: the Index Development Group, a group of multidisciplinary and internationally acclaimed experts in the field; the External Review Group, a group of potential end users and interested parties and the Steering Committee. Once the index has been formalised, it will be subjected to a pair of reliability tests using physicians and orthodontists scored 2 weeks apart. Subsequently, the index will be validated using dichotomous responses from orthodontists on whether they would treat a patient for OSA symptoms, and comparing the responses to the score of the index on the same patient. ETHICS AND DISSEMINATION: The index will be translated into French and will be presented in orthodontic and medical conferences, workshops, seminars, round table discussions, and free copies for download will be made available on the website of the University of Alberta Interdisciplinary Airway Research Clinic (iarc.ualberta.ca). Furthermore, the index will be published in a peer-reviewed medical journal to further increase the exposure of the index.