OBJECTIVE:To present reference data for nocturnal oximetry (NOx) in infants without respiratory disorders and evaluate their diagnostic accuracy for distinguishing moderate-to-severe upper airway obstruction (UAO) from mild/no UAO. MATERIALS AND METHODS:Infants (aged 1-12 months) without respiratory disease hospitalized for common disorders (e.g. gastroenteritis) underwent NOx (reference). Infants with and without UAO who had NOx were included as validation group. Abnormal basal SpO2 (<10th percentile for age), oxygen desaturation (≥3 %) index (ODI3) (>90th percentile), cumulative nocturnal hypoxemia score (>90th percentile) and McGill oximetry score (MOS) (>1) were assessed as predictors of UAO requiring intervention (moderate-to-severe UAO). RESULTS:Of 187 infants without respiratory disorder and with NOx, 138 (73.8 %) had acceptable tracings. Basal SpO2 was not age-related (P = .877; 10th percentile >95 %), while ODI3 and cumulative nocturnal hypoxemia score decreased with age (P < .001). MOS>1 likelihood diminished with age (OR 0.72 [0.59-0.86]; P < .001). For reference validation, 20 infants without respiratory disorder underwent NOx, and NOx of 77 infants with UAO were analyzed retrospectively (32.5 % required intervention post-NOx). Although NOx parameters values in UAO and reference groups partially overlapped, abnormal ODI3, cumulative nocturnal hypoxemia score and MOS were associated with increased odds of moderate-to-severe UAO (OR [95 %CI]: 5.33 [1.93-14.71] for ODI3-10s (≥10s desaturation duration); 3.57 [1.27-9.99]; and 5.98 [1.82-19.69], respectively). MOS>1 outweighed ODI3-10s in sensitivity for moderate-to-severe UAO detection (80.0 % [95 %CI: 60.9 %-91.1 %) vs. 52.0 % [95 %CI: 32.4 %-71.6 %]; P = .040). CONCLUSION:Frequent desaturations are common in infants with UAO, but also in those without respiratory disorder. Clusters of desaturations (MOS>1) represent an acceptable index of moderate-to-severe UAO.
OBJECTIVE:Nocturnal intermittent hypoxemia may enhance sympathetic nervous system activity. We aimed to assess the potential relationship of age-adjusted nocturnal pulse rate (% distance from 95th percentile for age-pulse rate index) with nocturnal oximetry parameters and their interaction with upper airway obstruction (UAO) presence. METHODS:Nocturnal oximetry data of 1-12-month-old infants without UAO/lung disease who were hospitalized for common pediatric problems (e.g. urinary tract infection) and of infants with clinically apparent UAO were analyzed. General linear models were applied to evaluate associations of nocturnal pulse rate index with group (infants with vs. without UAO) or nocturnal hypoxemia severity (oxygen desaturation [≥3 %] index for events lasting ≥2s, ≥5s, ≥10s or ≥20s (ODI3-2s, ODI3-5s, ODI3-10s or ODI3-20s, respectively). RESULTS:Oximetry data of 158 infants without UAO (age 1-12.7 months) and 23 with UAO (age 1-11.1 months) were analyzed. There were no significant associations of pulse rate index with group or ODI3 (P > .05). However, significant interactions were identified between ODI3-10s or ODI3-20s and group: in infants with UAO, pulse rate increased with raising ODI3-10s or ODI3-20s compared to participants without UAO (beta .612 [95 %CI .083-1.140; P = .023 and 1.140 [.145-2.134]; P = .025, respectively). For every 10 episodes/h increase in ODI3-20s among infants with UAO, average pulse rate approached or exceeded the 95th percentile for age by 11.4 % compared to infants without UAO. CONCLUSIONS:Despite overlap of desaturation indices values in infants without UAO and those with UAO, only in the latter, frequency of intermittent hypoxemia predicts an enhanced heart rate response.
Background Asthma exacerbation is a cause of morbidity and mortality in the pediatric population. In guidelines there is no consensus regarding discharge criteria post exacerbation. Moreover, sleep has a known impact on gas exchange, which might be clinically relevant in patients with obstructive lung disease. Therefore, the aim of this study was to investigate the nocturnal oximetry profile in hospitalized children with asthma exacerbation, pre-discharge. Materials and methods Nocturnal oximetry recordings of children with asthma admitted in Aghia Sofia Children's Hospital between December 2020 and April 2024 were reviewed. In this cross-sectional study we compared them with recordings of children with Sleep Disordered Breathing (SDB) and controls. Night-time basal SpO2, Oxygen Desaturation Index3 (ODI3), as well as % of time spent <95 % were compared. The Kruskal-Wallis test was employed to evaluate heterogeneity, followed by Mann-Whitney-Wilcoxon test for pairwise comparisons between the groups. Results Asthmatic patients (n = 13) had lower basal SpO2 in comparison with control (n = 21) and SDB group (N = 37) (SpO2 93.7 % versus 96.4 % and 96.7 % respectively, p < 0.0001). They spent more time in SpO2< 95 % and had similar ODI3 with the two other groups (p < 0.001 and p < 0.001 for asthma versus SDB and asthma versus control group respectively). Moreover, in patients with asthma a higher spot SpO2 was documented during the day, versus night-time basal SpO2. Conclusion This study highlights that clinical improvement precedes SpO2 improvement in asthmatic patients. A clinical review 48 h post-discharge is advisable. Nocturnal basal SpO2 is lower than daytime spot SpO2, which should be taken under consideration in implementation of guidelines.
Background Interstitial lung disease is rarer in children than adults, but, with increasing diagnostic awareness, more cases are being discovered. The prognosis of childhood interstitial lung disease is often poor, but increasing numbers are now surviving into adulthood. Aim To characterise childhood interstitial lung disease survivors and identify their impact on adult interstitial lung disease centres. Methods This was a European study (34 adult and childhood interstitial lung disease centres) reporting incident/prevalent cases of childhood interstitial lung disease survivors from January to July 2023. Epidemiological, clinical, physiological and genetic data were collected. Results 244 patients were identified with a median (interquartile range) age at diagnosis of 12.5 years (6-16 years) and age at study inclusion of 25 years (22-33 years), with 51% male, 86% nonsmokers and a median (interquartile range) % predicted forced vital capacity of 70% (47-89%) and diffusing capacity of the lungs for carbon monoxide of 48% (32-75%). 32% were prescribed long-term oxygen and 227 (93%) were followed up in adult centres whereas 17 (7%) never transitioned. The commonest diagnoses (82%) were childhood interstitial lung disease category B1 (sarcoidosis, hemosiderosis, connective tissue disorders, vasculitis) at 35%, A4 (surfactant-related) at 21%, B2 (bronchiolitis obliterans, hypersensitivity pneumonitis) at 14% and Bz (unclassified interstitial lung disease) at 13%. Bz patients had the worst functional status. 60% of all patients were still being prescribed corticosteroids. Re-specification of diagnosis and treatment were made after transition for 9.8% and 16% of patients, respectively. Not all childhood interstitial lung disease diagnoses were recognised in adult interstitial lung disease classifications. Conclusion Childhood interstitial lung disease survivors are seen in most adult interstitial lung disease centres and only a minority continue follow-up in paediatric centres. Survivors have a significant loss of lung function. The heterogeneity of their aetiologies and therapeutic requirements has a real impact on adult interstitial lung disease centres. Re-specification of diagnosis and treatment may contribute to precision and personalisation of management.
Introduction Pseudohypoaldosteronism type 1b (PHA1B) is a rare autosomal recessive disease caused by dysfunction of amiloride-sensitive epithelial sodium channels (ENaC), that might present with a wide variety of pulmonary symptoms. Methods We provide a systematic review of published cases with PHA1B and respiratory symptoms, adding a relevant case from our clinic. Results Thirty-seven publications presenting 61 cases were identified apart from our case. Parental consanguinity was reported in 24/62 patients. In 39 patients the onset of pulmonary manifestations was early in infancy. Lower respiratory tract infections caused by Pseudomonas aeruginosa and Staphylococcus aureus were reported in 3 cases each, while 2 patients developed bronchiectasis. Pathological sweat test results were recorded in all 34 patients with available data. In 36/47 patients the underlying pathogenic variant was identified in SCNN1A gene. Conclusion High clinical suspicion is required when treating patients with PHA1B for the potential need for early treatment of respiratory symptoms to avert any permanent pulmonary damage.
The aim of this review is to summarise evidence that became available after publication of the 2017 European Respiratory Society statement on the diagnosis and management of obstructive sleep apnoea syndrome (OSAS) in 1- to 23-month-old children. The definition of OSAS in the first 2 years of life should probably differ from that applied in children older than 2 years. An obstructive apnoea–hypopnoea index >5 events·h −1 may be normal in neonates, as obstructive and central sleep apnoeas decline in frequency during infancy in otherwise healthy children and those with symptoms of upper airway obstruction. A combination of dynamic and fixed upper airway obstruction is commonly observed in this age group, and drug-induced sleep endoscopy may be useful in selecting the most appropriate surgical intervention. Adenotonsillectomy can improve nocturnal breathing in infants and young toddlers with OSAS, and isolated adenoidectomy can be efficacious particularly in children under 12 months of age. Laryngomalacia is a common cause of OSAS in young children and supraglottoplasty can provide improvement in children with moderate-to-severe upper airway obstruction. Children who are not candidates for surgery or have persistent OSAS post-operatively can be treated with positive airway pressure (PAP). High-flow nasal cannula may be offered to young children with persistent OSAS following surgery, as a bridge until definitive therapy or if they are PAP intolerant. In conclusion, management of OSAS in the first 2 years of life is unique and requires consideration of comorbidities and clinical presentation along with PSG results for treatment decisions, and a multidisciplinary approach to treatment with medical and otolaryngology teams.
Background: Obstructive sleep apnea (OSA) is the most common sleep-related breathing disorder. Although adenotonsillectomy is first-line management for pediatric OSA, up to 40% of children may have persistent OSA. This document provides an evidence-based clinical practice guideline on the management of children with persistent OSA. The target audience is clinicians, including physicians, dentists, and allied health professionals, caring for children with OSA. Methods: A multidisciplinary international panel of experts was convened to determine key unanswered questions regarding the management of persistent pediatric OSA. We conducted a systematic review of the relevant literature. The Grading of Recommendations, Assessment, Development, and Evaluation approach was used to rate the quality of evidence and the strength of the clinical recommendations. The panel members considered the strength of each recommendation and evaluated the benefits and risks of applying the intervention. In formulating the recommendations, the panel considered patient and caregiver values, the cost of care, and feasibility. Results: Recommendations were developed for six management options for persistent OSA. Conclusions: The panel developed recommendations for the management of persistent pediatric OSA based on limited evidence and expert opinion. Important areas for future research were identified for each recommendation.
Over the past few years, there has been a surge in interest regarding the connection between sleep duration and quality, sleep disorders, mainly Obstructive sleep apnoea (OSA) and vitamin D. There is growing evidence to support a new role of vitamin D in the maintenance and regulation of optimal sleep. Furthermore, a notable link has been identified between OSA and a decrease in serum Vitamin D levels, which appears to intensify as the severity of sleep apnea worsens. Vitamin D status could also potentially serve as a mediator or provide an explanation for the association between OSA and cardiometabolic morbidity, but the current state of research in this area is inadequate. Studies have indicated that the supplementation of Vitamin D can optimize sleep quality, presenting more proof of the connection between insufficient vitamin D levels and sleep disorders. However, it is unclear whether low serum vitamin D levels are a contributing factor to OSA development or if OSA predisposes individuals to Vitamin D deficiency. As a result, various studies have endeavored to examine the complex relationship between OSA and Vitamin D deficiency. In children and adolescents, while data is limited, there seems also to be a link between sleep disorders and Vitamin D levels. Therefore, the objective of this review is to provide a comprehensive overview of the current evidence on the association between Vitamin D and sleep disorders in both adults and children.
SummaryNocturnal oximetry is an alternative modality for evaluating obstructive sleep apnea syndrome (OSAS) severity when polysomnography is not available. The Oxygen Desaturation (≥3%) Index (ODI3) and McGill Oximetry Score (MOS) are used as predictors of moderate‐to‐severe OSAS (apnea‐hypopnea index‐AHI >5 episodes/h), an indication for adenotonsillectomy. We hypothesised that ODI3 is a better predictive parameter for AHI >5 episodes/h than the MOS. All polysomnograms performed in otherwise healthy, snoring children with tonsillar hypertrophy in a tertiary hospital (November 2014 to May 2019) were analysed. The ODI3 and MOS were derived from the oximetry channel of each polysomnogram. Logistic regression was applied to assess associations of ODI3 or MOS (predictors) with an AHI >5 episodes/h (primary outcome). Receiver operating characteristic (ROC) curves and areas under ROC curves were used to compare the ODI3 and MOS as predictors of moderate‐to‐severe OSAS. The optimal cut‐off value for each oximetry parameter was determined using Youden's index. Polysomnograms of 112 children (median [interquartile range] age 6.1 [3.9–9.1] years; 35.7% overweight) were analysed. Moderate‐to‐severe OSAS prevalence was 49.1%. The ODI3 and MOS were significant predictors of moderate‐to‐severe OSAS after adjustment for overweight, sex, and age (odds ratio [OR] 1.34, 95% confidence interval [CI] 1.19–1.51); and OR 4.10, 95% CI 2.06–8.15, respectively; p < 0.001 for both). Area under the ROC curve was higher for the ODI3 than for MOS (0.903 [95% CI 0.842–0.964] versus 0.745 [95% CI 0.668–0.821]; p < 0.001). Optimal cut‐off values for the ODI3 and MOS were ≥4.3 episodes/h and ≥2, respectively. The ODI3 emerges as preferable or at least a complementary oximetry parameter to MOS for detecting moderate‐to‐severe OSAS in snoring children when polysomnography is not available.
We have read with great interest, the article by Tapia et al1Tapia I.E. Shults J. Cielo C.M. et al.A trial of intranasal corticosteroids to treat childhood OSA syndrome.Chest. 2022; 162: 899-919Abstract Full Text Full Text PDF PubMed Scopus (5) Google Scholar in CHEST (October 2022) that demonstrated that the administration of intranasal corticosteroids (INCS) to children with mild-to-severe OSA syndrome does not result in a significant decrease in obstructive apnea-hypopnea index (OAHI) or in neurobehavioral, symptom, or polysomnography changes at 3 and 12 months of treatment compared with placebo. The authors propose that their findings do not favor the use of INCS in children with OSA syndrome. We appreciate the important findings presented in the paper by Tapia et al1Tapia I.E. Shults J. Cielo C.M. et al.A trial of intranasal corticosteroids to treat childhood OSA syndrome.Chest. 2022; 162: 899-919Abstract Full Text Full Text PDF PubMed Scopus (5) Google Scholar and wish to comment on some study characteristics that could possibly modify interpretation of the results. The use of INCS has been suggested for children with mild-to-moderate OSA syndrome, and the beneficial effect on upper airway obstruction has been attributed to a reduction in the size of the adenoids, which increases rapidly in children with snoring early in life and does not change thereafter.2Papaioannou G. Kambas I. Tsaoussoglou M. Panaghiotopoulou-Gartagani P. Chrousos G. Kaditis A.G. Age-dependent changes in the size of adenotonsillar tissue in childhood: implications for sleep-disordered breathing.J Pediatr. 2013; 162: 269-274.e264Abstract Full Text Full Text PDF PubMed Scopus (66) Google Scholar, 3Kaditis A. Alonso Alvarez M.L. Boudewyns A. et al.Obstructive sleep disordered breathing in 2- to 18-year-old children: diagnosis and management.Eur Respir J. 2016; 47: 69-94Crossref PubMed Scopus (454) Google Scholar, 4Demain J.G. Goetz D.W. Pediatric adenoidal hypertrophy and nasal airway obstruction: reduction with aqueous nasal beclomethasone.Pediatrics. 1995; 95: 355-364Crossref PubMed Google Scholar, 5Alexopoulos E.I. Kaditis A.G. Kalampouka E. et al.Nasal corticosteroids for children with snoring.Pediatr Pulmonol. 2004; 38: 161-167Crossref PubMed Scopus (104) Google Scholar In the current study, it is not specified whether participants had adenoidal hypertrophy or at least chronic nasal obstruction. In a multicenter, retrospective study, less than 7 years of age and normal weight were favorable predictors of mild OSA syndrome resolution after a 12-week regimen of INCS and oral montelukast.6Kheirandish-Gozal L. Bhattacharjee R. Bandla H.P.R. Gozal D. Antiinflammatory therapy outcomes for mild OSA in children.Chest. 2014; 146: 88-95Abstract Full Text Full Text PDF PubMed Scopus (89) Google Scholar Of note, in the clinical trial by Tapia et al,1Tapia I.E. Shults J. Cielo C.M. et al.A trial of intranasal corticosteroids to treat childhood OSA syndrome.Chest. 2022; 162: 899-919Abstract Full Text Full Text PDF PubMed Scopus (5) Google Scholar one-third of the subjects were obese, and more than one-half of the subjects were overweight or obese. Moreover, most children were more than 7 years old (mean age was 8.2 years in the INCS arm and 8.1 years in the placebo arm); approximately one-quarter of them had more than 10 episodes/h at baseline on the OAHI. These three participants' characteristics may have modified the effect of INCS adversely on the OAHI. Another point that needs to be clarified is the percentage of patients with OAHI score of more than 5 episodes/h who had mild OSA syndrome at 3 months after treatment with INCS or placebo. Children with OAHI scores of less than 5 episodes/h, but without OSA syndrome-associated morbidity or impaired quality of life, do not necessarily need treatment; thus, adenotonsillectomy can be avoided. Although results of the clinical trial by Tapia et al1Tapia I.E. Shults J. Cielo C.M. et al.A trial of intranasal corticosteroids to treat childhood OSA syndrome.Chest. 2022; 162: 899-919Abstract Full Text Full Text PDF PubMed Scopus (5) Google Scholar do not favor INCS use for pediatric OSA syndrome and given that they are prescribed widely in clinical practice to avoid adenotonsillectomy, we propose that targeted treatment of nonobese 2- to 6-year-old children with mild-to-moderate OSA syndrome and adenoidal hypertrophy should be further explored. The jury for INCS in pediatric OSA syndrome is still out. None declared. A Trial of Intranasal Corticosteroids to Treat Childhood OSA SyndromeCHESTVol. 162Issue 4PreviewIn children with OSAS, treatment with INCS did not result in significant polysomnography, neurobehavioral, or symptom changes at 3 and 12 months of treatment. Twelve months of INCS treatment resulted in a statistically significant but not clinically relevant OAHI reduction. Full-Text PDF
Although nocturnal oximetry has been used for OSAS management in resource-limited settings, little evidence is available regarding reference values in infancy. Our aim was to summarize reference values for oximetry parameters in healthy infants aged 1-6 months. Methods: Prospectively collected oximetry data from full-term infants without history of apnea, upper airway obstruction (UAO), craniofacial abnormalities, neuromuscular disorders, genetic syndromes or lung disease were analyzed. Summary statistics of oxyhemoglobin desaturation (≥3%) index (ODI3),baseline SpO2, and cumulative nocturnal hypoxemia score ([%recording time-%RT with SpO2 90–94%]X1 + [%RT with SpO2 85–89%]X2 + [%RT with SpO2 80–84%]X3 + [%RT with SpO2 ≤79%]X4) as well as frequency of abnormal McGill Oximetry Score (MOS>1) were calculated. Results: Data from 51 infants (median age 2.8 months; range 1-6.6) were analyzed (Table). ODI3 decreased linearly with age (P=.01) and MOS was abnormal in an appreciable proportion of participants. Conclusions: In the first 1-6 months of life, nocturnal baseline SpO2 is above 95% and-in contrast to older children-ODI3 decreases with age while MOS is frequently abnormal in the absence of UAO.
For more than three decades, type III devices have been used in the diagnosis of sleep disordered breathing in supervised as well as unsupervised settings. They have satisfactory positive and negative predictive values for detecting obstructive and central sleep apnoea in populations with moderately high pre-test probability of symptoms associated with these events. However, standardisation of commercially available type III devices has never been undertaken and the technical specifications can vary widely. None have been subjected to the same rigorous processes as most other diagnostic modalities in the medical field. Although type III devices do not include acquisition of electroencephalographic signals overnight, the minimum number of physical sensors required to allow for respiratory event scoring using standards outlined by the American Academy of Sleep Medicine remains debatable. This technical standard summarises data on type III studies published since 2007 from multiple perspectives in both adult and paediatric sleep practice. Most importantly, it aims to provide a framework for considering current type III device limitations in the diagnosis of sleep disordered breathing while raising research- and practice-related questions aimed at improving our use of these devices in the present and future.