Respiratory polygraphy (RP) underestimates the obstructive apnea-hypopnea index (OAHI) compared with polysomnography (PSG). To minimize this bias, a built-in algorithm integrated into the RP software was used to classify wake, NREM and REM sleep stages (RP-Algorithm). The aim was to determine the accuracy of the algorithm in detecting the three-sleep stages and to assess the agreement between OAHI obtained by RP-Algorithm or RP and OAHI by PSG. PSG recordings from 88 children (median age [1st; 3rd quartile]: 10.5 [7.5; 13]years) were included. Several weeks after the PSG analysis, the scoring was deleted, and recordings blindly reanalyzed considering only respiratory signals and the built-in algorithm (Nox BodySleep™ 1.0, ResMed; RP-Algorithm). Few weeks later, a final analysis was performed after deleting the previous, and recordings were blindly reanalyzed as RP. The RP-Algorithm showed an accuracy of 90% and a Cohen's kappa of 0.68 for the three-stage sleep classification. OAHI was underestimated by RP-Algorithm (3.5 [2.2; 6.2/h) or RP (3.0 [1.9; 5.5]/h) compared to PSG (4.8 [3.3; 6.6]/h; p < 0.0001 for both), confirmed by Bland-Altman analysis (mean difference -1.39/h (95%CI: -3.62-0.84) for RP-Algorithm and -1.92 events/h (95%CI: -4.41-0.57) for RP). Severity category changed in 13 children (15%) when diagnosed by RP-Algorithm and in 23 children (26%) when diagnosed by RP. The built-in algorithm incorporated into RP interpretation demonstrated high accuracy for three-stage sleep classification and reduced the underestimation of the OAHI compared with standard RP, when PSG was used as gold standard, with only a few children changing the severity category.

Objectif La polygraphie ventilatoire (PV) sous-estime habituellement l’index d’apnée-hypopnée obstructive (IAHO) étant donné l’absence de prise en compte des événements respiratoires liés aux microéveils corticaux. Afin de minimiser ce biais, l’algorithme BodySleep a été rajouté à la PV (PV-BodySleep), un algorithme d’apprentissage automatique qui utilise un réseau neuronal artificiel pour classer des périodes de 30 secondes en états de sommeil ou veille, inclus dans le logiciel Noxturnal. L’objectif de cette étude a été de comparer les paramètres respiratoires obtenus par PV-BodySleep à ceux rapportés par la polysomnographie (PSG) et par la PV. Méthodes Des enregistrements de PSG de 52 enfants (âge médian [1er ; 3e quartile] : 10 [7 ; 12] ans) ont été inclus. Plusieurs semaines après l’analyse de la PSG, l’analyse a été supprimée et les enregistrements ont été réanalysés en aveugle comme PV-Bodysleep et quelques semaines après comme PV. Résultats Le temps total de sommeil n’a pas été significativement différent entre PSG et PV-BodySleep (440 [363 ; 505]min en PSG vs 452 [373 ; 502]min en PV-BodySleep], mais il a été significativement plus élevé en PV (46 [385 ; 530]min) qu’en PSG ou PV-BodySleep (p<0,0001 pour les deux). L’IAHO médian [1er ; 3e quartile] a été sous-estimé par la PV-BodySleep (3 [2,1 ; 5,4]/h) et par la PV (2,8 [1,6 ; 4,1]/h) par rapport à la PSG (4,4 [3 ; 5,9]/h (p<0,0001 pour les deux). L’analyse de Bland-Altman a montré que la sous-estimation de l’IAHO par rapport à la PSG a été moindre par la PV-BodySleep que par la PV (ampleur de la différence de −0,95±1,29 pour la PV-Bodysleep et de −1,50±1,70 pour la PV). Les 4 enfants classés SAOS sévère en PSG l’ont aussi été en PV-BodySleep, et 1 a été classé modéré en PV. Parmi les 17 enfants SAOS modérés en PSG, 6 ont été légers en PV-BodySleep et 9 en PV. Conclusion L’algorithme BodySleep sous-estime l’IAHO par rapport à l’IAHO de la PSG, mais cette sous-estimation est moindre que la sous-estimation de l’IAHO par la PV seule. L’ajout de l’algorithme a amélioré la capacité diagnostique de la PV pour le diagnostic du SAOS.

OBJECTIVES:To assess the effectiveness in children of long-term ventilatory support (VS) (continuous positive airway pressure [CPAP], non-invasive ventilation [NIV]), on symptoms, sleep, respiratory parameters using polysomnography (PSG), evaluate the prevalence of residual sleepiness and/or asthenia (RSA) under VS and identify parameters associated with RSA. METHODS:Multicenter retrospective study including children with SDB aged between 1 month and 18 years, under long-term VS, evaluated by PSG, both at diagnosis in room air and under VS. Clinical, sleep, and respiratory characteristics were compared between diagnostic PSG and PSG under VS for the entire cohort and between patients with and without RSA. RESULTS:A total of 61 children were included (median [IQR] age at diagnostic PSG and at PSG under VS, 7 [0-13] and 8 [0-13] years respectively). While the SDB night-time symptoms significantly improved with VS, the SDB daytime symptoms were not all well controlled; RSA under VS was present in 22/53 (42%) children. At PSG under VS compared to diagnostic PSG, non-respiratory arousal index remained abnormal despite significant improvement (median n = 15/hr [8-23] versus 21/h [12-29], p = 0.009) and OSA became mild (median OAHI 2/hr [1-4] versus 13/h [6-27], p < 0.001). Children with RSA, compared to children without RSA, more frequently used NIV than CPAP (82% versus 18%, p < 0.01) and had a lower proportion of rapid eye movement under VS (18% [14-25] versus 24% [20-32], p = 0.038). CONCLUSION:While SDB symptoms improved under VS concurrent with the respiratory parameters, a significant proportion of children remained symptomatic with RSA, underlying the importance of carefully monitoring these patients during follow-up.

BACKGROUND:The 2019 French national survey showed that 1447 children were treated with long-term continuous positive airway pressure (CPAP) or noninvasive ventilation (NIV). Data about the pediatric disorders that require CPAP or NIV are scarce. The aim of the study was to report, for CPAP and NIV, the detail of the different disorders, according to age categories, based on the 2019 national survey. METHODS:Age at CPAP/NIV initiation was categorized as follow: 0-2, 3-5, 6-10 and ≥ 11 years old. The number of children was also reported for each age category. RESULTS:Children <3 years old were the most frequently treated by CPAP/NIV. Down syndrome and Robin sequence were the most common disorders requiring CPAP in this age group, obstructive sleep apnea syndrome (OSAS) and Down syndrome between 3 and 5 years old, and obesity and OSAS from ≥6 years old. Spinal muscular atrophy (SMA) types 1 and 2 were the most common disorders requiring NIV at < 3 years old, SMA type 2 between 3 and 10 years old, and Duchenne muscular dystrophy (DMD) from ≥11 years old. Overall, Down syndrome represented one of the most common disorders for which CPAP was started at any age. SMA type 2 was one of the most common disorders for which NIV was started at any age. CONCLUSIONS:Obesity, Down syndrome, OSAS, SMA type 2, and DMD represented the most common disorders treated with home CPAP/NIV in France. These data may change with the emergence of innovative therapies for rare diseases.

BACKGROUND:Data are lacking for lung evaluation after hematopoietic cell stem transplantation (HSCT) in children under the age of six, as this population cannot be included in respiratory function monitoring protocols. METHODS AND SETTINGS:The RESPPEDHEM cohort included individuals under the age of 18 who underwent HSCT between January 2014 and November 2017. The eligible population for this study consisted of children from the RESPEDHEM cohort, who underwent HSCT before the age of 6, were still alive in October 2023 and had pulmonary function tests (PFTs) performed more than 3 years after HSCT. The primary objective of our multicenter study was to describe long-term PFT outcomes in children who received HSCT before the age of six, as included in the RESPEDDHEM cohort. The secondary objective was to identify clinical, radiological and transplant-related factors associated with abnormal lung function. Pulmonary abnormalities were defined according to the guidelines of the American Thoracic Society (ATS), the European Respiratory Society (ERS), and Global Lung Initiative (GLI). RESULTS:Among the 40 children, the mean (SD) age at transplantation was 3.7 ± 1.4 years; 50 % had undergone PFT before HSCT. The last follow-up lung function test was performed at 10.2 ± 2.2 years. Twelve individuals (30 %; 95 %CI: 17-47 %) had abnormal lung function at the end of the study, based on the recent ERS/ATS technical standard on interpretive strategies for routine lung function tests. The main anomalies were obstructive defect (n=4) and restrictive defect (n=4). CONCLUSION:This study is the first lung function analysis of children undergoing HSCT before the age of six. Abnormalities persist in about one-third of the population, and 42 % of these children were asymptomatic. Therefore, systematic and long-term respiratory monitoring is needed even if the absence of clinical symptoms. However, half of the cohort did not undergo pre-HSCT pulmonary function testing, which limits the ability to attribute abnormalities solely to the transplant.

Introduction Real-world data on children with severe asthma is scarce. We report characteristics of children with severe asthma already on biologics, enrolled in the Severe Paediatric Asthma Collaborative in Europe, a clinical research collaboration of the European Respiratory Society. Methods We describe patient's characteristics including asthma control assessed with Global Initiative for Asthma (GINA) criteria, composite asthma severity index (CASI), exacerbations, unscheduled medical attendances, lung function and quality of life in children on biologic treatment because of severe asthma. We also assessed previous biologics use. Forced expiratory volume in 1 s, CASI, GINA, Paediatric Asthma Quality of Life Questionnaire score, exacerbations, unscheduled medical attendance and hospital admission comparisons in patients treated with different biologics were adjusted by age, sex and biologic therapy duration. Results Among the 250 children (median age 13.2 years) recruited, 56.8% used omalizumab, 21.6% mepolizumab and 21.6% dupilumab. At enrolment, the dupilumab group was older (median 15.0 years), while the omalizumab group had been on biologic treatment the longest (median 622 days). Overall, 27% and 8% had partly controlled and uncontrolled asthma respectively, according to GINA. In the last 12 months, 52% and 29% had at least one and two exacerbations, respectively; airflow obstruction was found in 33%. 10% were admitted to hospital due to exacerbation. A previous switch from another biologic was recorded in 16%, predominantly due to nonresponse. Conclusions Most children on biologics obtained good symptom control, but many still experienced asthma attacks. Switching between biologics was substantial. There is still an unmet need in severe paediatric asthma.

Purpose: The aim of the study was to analyze the characteristics of otherwise healthy children with obstructive sleep apnea (OSA; OSA-I) and children with OSA and non-syndromic obesity (OSA-II) treated with long term continuous positive airway pressure (CPAP) or noninvasive ventilation (NIV) in 2019 in France. Methods: Data were collected from a national survey on paediatric home noninvasive ventilatory support. CPAP/ NIV initiation criteria and duration, age at CPAP/NIV initiation, equipment used and CPAP/NIV settings, and objective compliance were analyzed. Results: Patients with OSA-I and OSA-II represented 6 % (n = 84, 71 % males) and 10 % (n = 144, 72 % males) of the national cohort, respectively. The apnea-hypopnea index (63 % vs 76 %), alone or combined with nocturnal gas exchange (25 % vs 21 %, for OSA-II and OSA-I patients respectively) were used as initiation criteria of CPAP/ NIV. OSA-II patients were older at CPAP/NIV initiation (mean age 11.0 f 4.0 vs 6.8 f 4.5 years, p < 0.001) and were treated for a longer time (2.3 f 2.6 vs 1.3 f 1.5 years, p = 0.008) than OSA-I patients. NIV was used in 6 % of OSA-I patients and 13 % of OSA-II patients (p = 0.142). Both groups used preferentially a nasal mask. Mean CPAP level was higher in OSA-II patients as compared to OSA-I patients (8.7 f 2.0 vs 7.7 f 2.4 cmH2O, p = 0.02). Objective compliance was comparable (mean use 6.8 f 2.6 vs 5.9 f 3.0 h/night in OSA-I and OSA-II, respectively, p = 0.054). Conclusion: Six and 10 % of children treated with long term CPAP/NIV in France in 2019 had OSA-I and OSA-II, respectively. Both groups were preferentially treated with CPAP and were comparable except for age, with OSAII patients being older at CPAP/NIV initiation.

Background:Pediatric sleep disorders are underreported and underdiagnosed in primary care settings. This study aimed to compare the screening and management of sleep in young children during routine follow-up consultations by general practitioners (GPs) and physicians from Maternal and Child Protection services (MCPPs) in a region of France. Methods:A cross-sectional survey was conducted using a questionnaire with 20 closed questions designed to characterize knowledge and practices and to evaluate the perceived need for additional training in pediatric sleep. The survey was administered to 343 GPs and 28 MCPPs. Results:A total of 211 (62%) GPs and 17 (61%) MCPPs participated in the survey. The respondents indicated a strong focus on sleep, GPs reporting significantly lower scores compared to MCPPs (4 [3, 4] vs 4 [4, 5], p < 0.008). A significantly higher proportion of physicians addressed the issue of sleep in the MCPPs group, 16 (94%), in contrast to an equivalent proportion of parents and physicians in the GPs group (p = 0.0007). The difficulties falling asleep were significantly less frequently addressed by GPs than MCPPs (p = 0.042), with no difference for the nighttime awakenings. A limited number of physicians, 24 (11%), have received training in sleep medicine, with significantly fewer GPs than MCPPs, while over half, 148 (65%), expressed a need for additional education. Discussion:GPs demonstrated lower awareness and training in young children's sleep issues compared to MCPPs. These findings emphasize the need for improved training and awareness, highlighting the importance of evaluating children's sleep issues during pediatric consultations by GPs.

BACKGROUND:A French national survey showed that 1447 children were treated with long-term continuous positive airway pressure (CPAP) or noninvasive ventilation (NIV) in 2019. Data about the ventilatory settings for children are scarce. The aim of the study was to report the CPAP/NIV settings from the survey according to the patients' age and disorders. METHODS:CPAP and NIV settings were compared between 5 age groups (<1, 1-5, 6-11, 12-17 and ≥ 18 years), and 6 disease categories (upper airway disorders; neuromuscular disease, NMD; disorder of the central nervous system; cardiorespiratory disorder; congenital bone disease, CBD; and other). RESULTS:Age correlated positively with constant CPAP pressure (r = 0.364, p < 0.0001), and negatively with CPAP adherence (r = -0.173, p < 0.0001). Mean age at CPAP initiation, CPAP pressures and adherence did not differ between disorders. Regarding NIV, mean inspiratory positive airway pressure (IPAP) increased with age (r = 0.152, p = 0.0001), whereas respiratory rate (RR; r = -0.593, p < 0.0001) and adherence to NIV decreased with age (r = -0.154, p = 0.0002). NIV settings were quite similar between disease categories, with the CBD group having the highest IPAP, and NMD group having the lowest expiratory positive airway pressure and RR. Adherence tended to be higher with NIV than CPAP. CONCLUSIONS:CPAP pressure and IPAP increase with age, while settings seem quite similar between diseases. Even if our study provides some information about CPAP/NIV settings, they should always be individually adapted according to the severity of the disease.

Respiratory polygraphy (RP) underestimates the obstructive apnea-hypopnea index (OAHI) compared to polysomnography (PSG) because it does not account for respiratory events associated with cortical arousals. To address this issue, RP can incorporate respiratory events associated with autonomic arousals, identified by drops in pulse wave amplitude (PWAD), obtained from the oximetry signal. This study aimed to compare the OAHI measured by RP alone, RP incorporating respiratory events associated with PWAD, and PSG. PSG recordings from 91 children (median age [1st; 3rd quartile]: 10 [8; 14]years) were analyzed. A few weeks later, the initial analysis was deleted, and the recordings were reanalyzed as RP alone and as RP incorporating respiratory events associated with PWAD. Three PWAD thresholds were tested: drops greater than 30 % (RP-PWAD30), 40 % (RP-PWAD40), and 50 % (RP-PWAD50) from baseline. The median [1st; 3rd quartile] OAHI measured by RP alone was 2.8 [1.7; 5.7]/h, underestimating OAHI measured by PSG (4.8 [3.0; 6.8]/h; Bland-Altman analysis: mean f SD =-2.11 f 2.51). In contrast, incorporating respiratory events associated with PWAD led to OAHI overestimation: 10.2 [7.0; 13.3]/h (Bland-Altman = 4.96 f 4.18) for RP-PWAD30; 8.7 [6.0; 12.7]/h (Bland-Altman = 3.73 f 3.76) for RP-PWAD40; 7.9 [5.1; 11.3]/ h (Bland-Altman = 2.89 f 3.69) for RP-PWAD50), in comparison to PSG. Since cortical arousals cannot be measured during RP, incorporating respiratory events associated with PWAD can improve OAHI estimation. Among the tested thresholds, the 50 % PWAD threshold resulted in the least overestimation in comparison to PSG.

Objectif Il n’existe pas de définition consensuelle de l’échec de support ventilatoire (SV) à domicile ni chez l’adulte ni chez l’enfant. L’objectif de l’étude était de définir l’échec de SV via la méthode Delphi et de tester cette définition sur une cohorte d’enfants. Méthodes Trente et un experts ont participé à 6 tours DELPHI. Ils ont défini l’échec d’observance, l’échec clinique et l’échec paraclinique. Cette définition 1 a été testée rétrospectivement sur les enfants sous SV de Lyon et Grenoble entre novembre 2022 et décembre 2023. Résultats Échec d’observance si<4h/nuit et/ou < 3 nuits/semaine, échec clinique si symptômes de syndrome d’apnées obstructives (SAOS) non corrigés ou décompensations respiratoires hospitalisées en réanimation/soins continus, échec paraclinique si clusters de désaturation persistant et/ou ≥ 5 % du temps avec SpO2≤90 % ; TcPCO2 moyenne≥50mmHg et/ou TcPCO2≥50mmHg pendant≥5 % du temps et/ou gaz du sang au réveil sous machine avec pCO2≥50mmHg ou bicarbonates≥30mmol/L ; poly(somnographie) retrouvant un index d’apnées-hypopnées obstructives restant≥10/h. Parmi les 209 patients inclus (âge médian [min–max] 11 ans [4–15], SAOS type 3 [93 %]), dont 167 encore sous SV, 65 (39 %) avaient eu un échec. Parmi eux, 38 (58 %) avaient eu un échec d’observance, 15 (23 %) un échec clinique, 26 (40 %) un échec paraclinique. Aucun facteur associé à l’échec n’était significativement identifié. Comparé à l’échec noté par le médecin référent du patient, la définition avait une valeur prédictive positive de 66 %. Après ce test en population, une définition 2 a été retenue avec assouplissement de la définition de l’échec paraclinique. Conclusion L’échec de support ventilatoire peut se définir selon 3 versants (observance, clinique, paraclinique). Cette définition est à modérer en fonction de l’indication.

La dyspnée d’effort est un symptôme fréquent chez l’enfant. Les consultations qui en découlent ont souvent pour motif d’établir un diagnostic d’asthme induit par l’exercice. Si celui-ci est fréquent dans la population pédiatrique, toute gêne respiratoire à l’exercice ne doit pas se résumer à un asthme, ni même à une bronchoconstriction à l’exercice. La dyspnée d’effort peut survenir à la suite d’un ensemble de troubles de l’adaptation cardiorespiratoire qui fait suite au stress que constitue un exercice physique, alors que la bronchoconstriction à l’exercice s’exprime elle par des symptômes assez caractéristiques qu’il faut rechercher spécifiquement à l’interrogatoire, à l’examen clinique, et aux EFR avant et après un test de provocation bronchique. Un certain nombre de diagnostics différentiels sont à rechercher devant une dyspnée à l’exercice, constituant parfois des comorbités d’un asthme sous-jacent, comme l’hyperventilation à l’exercice, les dyskinésies des cordes vocales ou un déconditionnement physique.

Oximetry was proposed as an abbreviated exam, easily accepted by the child, for the diagnosis of obstructive sleep apnea (OSA) for children located in regions where access to pediatric sleep labs is limited. The objective of this study was to determine the diagnostic value of the oxygen desaturation index (ODI), the number of ≥ 3

Objective The aim of the study was to describe the characteristics of otherwise healthy children with obstructive sleep apnea (OSA; OSA-I) and children with OSA and obesity (OSA-II) treated with long term continuous positive airway pressure (CPAP) or noninvasive ventilation (NIV) in 2019 in France. Design National cross-sectional survey. Patients Children with OSA-I and OSA-II. Main outcome measures Initiation criteria, age, adherence, equipment and settings Results Patients with OSA-I and OSA-II represented 6% (n=84, 71% males) and 10% (n=144, 72% males) of the national cohort, respectively. The apnea-hypopnea index (63% vs 76%), alone or combined with nocturnal gas exchange (25% vs 21%, for OSA-II and OSA-I patients respectively) were used as initiation criteria of CPAP/NIV. OSA-II patients were older at CPAP/NIV initiation (mean age 11.0±4.0 vs 6.8±4.5 years, p<0.001) and were treated for a longer time (2.3±2.6 vs 1.3±1.5 years, p=0.008) than OSA-I patients. NIV was used in 6% of OSA-I patients and 13% of OSA-II patients (p=0.142). Nasal mask was the most used interface in both groups. Mean CPAP level was higher in OSA-II patients as compared to OSA-I patients (8.7±2.0 vs 7.7±2.4 cmH O, p=0.02). Objective compliance was comparable (mean use 6.8±2.6 vs 5.9±3.0 hours/night in OSA-I and OSA-II, respectively, p=0.054). Conclusion Six and 10% of children treated with long term CPAP/NIV in France in 2019 had OSA-I and OSA-II, respectively. Both groups were preferentially treated with CPAP and were comparable except for age, with OSA-II patients being older.

Objective Dyspnoea and sleep-disordered breathing (SDB) are common in children with life-limiting conditions but studies on treatment with non-invasive ventilation (NIV) or continuous positive airway pressure (CPAP) are scarce. The aim of the study was to describe children treated with long-term NIV/CPAP within a paediatric palliative care programme in France.Methods Cross-sectional survey on children and young adults with complex medical conditions treated within the French paediatric NIV network with long-term NIV/CPAP. Characteristics of the patients were analysed and patient-related outcome measures of NIV/CPAP benefit were reported.Results The data of 50 patients (68% boys), median age 12 (0.4-21) years were analysed. Twenty-three (46%) patients had a disorder of the central nervous system and 5 (10%) a chromosomal anomaly. Thirty-two (64%) patients were treated with NIV and 18 (36%) with CPAP. NIV/CPAP was initiated on an abnormal Apnoea-Hypopnoea Index in 18 (36%) of the patients, an abnormal nocturnal gas exchange alone in 28 (56%), and after an acute respiratory failure in 11 (22%) of the patients. Mean objective NIV/CPAP adherence was 9.3 +/- 3.7 hours/night. NIV/CPAP was associated with a decrease in dyspnoea in 60% of patients, an increase in sleep duration in 60% and in sleep quality in 74%, and an improvement in parents' sleep in 40%.Conclusions In children with life-limiting conditions, long-term NIV/CPAP may be associated with relief of dyspnoea, an improvement of SDB and an improvement in parents' sleep.