Home Respiratory Polygraphy and Spirometry in Normal Weight and Children with Obesity Suspected for Obstructive Sleep Apnea Syndrome: Are There Any Associations?

Aim: It is known that children and adolescents with obesity are more prone to obstructive sleep apnea syndrome (OSAS) and that their lung function may show some disturbance. Literature is scarce about potential associations; therefore, we aimed to study the relationship between OSAS, lung function, and adiposity in a population of children suspected of OSAS. Material and Methods. We performed home respiratory polygraphy and spirometry in all subjects. The relationships between body mass index z-score (zBMI), polygraphy, and spirometry data were analyzed. Results: We recruited 81 subjects aged between 5 and 16 years, 63% being obese. 43.2% of subjects were diagnosed with OSAS (32.1% mild, 4.9% moderate, and 6.2% severe). We found no correlation between respiratory polygraphy and the zBMI. The mean spirometric value FEV1, FVC, and FEV1/FVC ratio z's were normal in all subjects, whereas FVC z's and FEV1/FVC ratio z's were significantly positively related for obesity and negatively for normal weight (p < 0.05). FEV1 z's was inversely correlated to the percentage of analyzed time passed below 90% of SpO2 (r = -0.224, p = 0.044). All subjects with FEV1 (n = 8) and/or FVC (n = 9) z's below the lower limit for normal (LLN) had an AHI ≥ 1 (FEV1: p = 0.001; FVC: p < 0.001), especially subjects with normal weight (FEV1: p = 0.003; FVC: p = 0.010). Conclusion: When comparing normal-weight children and adolescents with obesity, the prevalence of OSAS but not spirometric values was strongly related to BMI z-score, probably because obesity engenders advanced puberty and an accelerated growth spurt. FEV1 was more frequently

Cardiopulmonary function in adolescent patients with pectus excavatum or carinatum.

BACKGROUND: Pectus excavatum (PE) and pectus carinatum (PC) have generally been considered an aesthetic issue, although there is growing evidence of associated cardiopulmonary function (CPF) impairment, especially in PE patients. The study goal was to determine any correlation between pectus malformations and cardiopulmonary symptoms and function based on systematic assessment of CPF and thoracic measurements, such as Haller Index (HI) and sternal torsion angle (STA). METHODS: Data from 76 adolescent patients with PE (n=30) or PC (n=46) were retrospectively collected referred between January 2015 and April 2018. CPF measurements and thoracic imaging were performed in all patients. HI and STA correction indexes were measured in all patients. FINDINGS: Medical records from 76 patients (PE n=30; PC n=46) were analysed. Patients were predominantly male (>93.3%), and aged between 13 and 14½ old. PE was associated with airway obstruction, with a forced expiratory volume in 1 s value under the lower limit of normal in 13% of cases (p<0.001). Restrictive syndrome was observed in 23% of cases (p<0.001), with a Z score for total lung capacity under the lower limit of normal. In PC, pulmonary function was not affected. All patients showed slightly decreased values of left and right ejection fraction and cardiac index at rest, although values were within normal range. There were no significant correlations between pulmonary and cardiac functions or between low CPF and thoracic measurements. INTERPRETATION: Our results confirm the modest impact of pectus malformations on CPF at rest, without correlation with anamnestic dyspnoea on exertion, nor with chest pain or anatomical measurements. Validation of new correction indexes could be helping characterise these malformations and choose optimal therapeutic management.

Home respiratory polygraphy in obstructive sleep apnea syndrome in children: Comparison with a screening questionnaire.

OBJECTIVES: The prevalence of obstructive sleep apnea syndrome (OSAS) in children referred for sleep-disordered breathing reaches up to 59%. We aimed to test the adequacy of a questionnaire compared to home respiratory polygraphy (HRP), in 45 subjects (5-16 years-old), without maxillofacial malformations nor other comorbidities, presenting with symptoms compatible with OSAS. METHODS: All children passed a 12-items questionnaire (Obstructive Airway Child test: OACT) and the HRP. OSAS was classified in severity according to the apnea-hypopnea index (AHI). RESULTS: With HRP, 60% and 15% children were detected to have at least mild (AHI ≥1) and moderate (AHI >5) OSAS, respectively. The sensitivity of the questionnaire to detect mild and moderate OSAS was good (93% and 71%, respectively) but the specificity was very low (11% and 34%). However, an OACT score under 61 showed a very good negative predictive value for moderate and severe OSAS (87%). With the questionnaire, we could have avoided a complementary PSG or HRP in 25/45 (56%) of our subjects as in children with mild OSAS and without comorbidities only clinical observation is usually advised. CONCLUSIONS: The OACT questionnaire has shown to be a good and quick instrument to exclude moderate and severe OSAS in our population of children without maxillofacial malformations. Indeed children scoring under 61 could avoid a constraining and expensive sleep exam. However, if the score is above this cut-off, the performance to recognize OSAS is low and the child's evaluation must be completed by a HRP or PSG.

Respiratory polygraphy data of children investigated for sleep-disordered breathing with different congenital or respiratory diseases.

This short report describes respiratory indices of polygraphies (PG) performed to investigate several sleep-related disorders of breathing in children. It refers to the work of Michelet et al., Successful home respiratory polygraphy to investigate sleep-disordered breathing in children, Sleep Medicine [1]. Indications for PGs were grouped according to 6 categories: craniofacial malformation, neuromuscular disease, obesity, suspected obstructive sleep apnea (OSA), prematurity, and other. The reported data concern the initial interpretable PGs (N = 289); initial was defined as performed for the first time in any subject. Non-interpretability was defined as absent or unreliable oxygen saturation by pulse oximetry (SpO2), and/or airflow and respiratory inductance plethysmography (RIP) flow trace signals during time analyzed. Analyzed time is reported. In a subset of patients, transcutaneous carbon dioxide partial pressure (ptcCO2) was also measured. Data may be re-used for comparison in future validating research for PGs in children [2].

Successful home respiratory polygraphy to investigate sleep-disordered breathing in children.

OBJECTIVE: Sleep-disordered breathing (SDB) in children is common. Interest in sleep tests, such as polygraphy (PG), which can be performed in a non-attended setting, are gaining is increasing. PG has, however, been little studied in children with co-morbidities other than obstructive sleep apnea (OSA), and in particular, if performed in a non-attended setting. We report on the feasibility and interpretability of implementing PGs at home versus in hospital. METHODS: PGs were analyzed according to the setting (hospital or home) and sequence (initial or subsequent) in which they were performed. Non-interpretability was defined as absent or unreliable oxygen saturation by pulse oximetry (SpO2), or airflow and respiratory inductance plethysmography flow trace signals during the time analyzed. RESULTS: We retrospectively analyzed 400 PGs; 332/400 were initial PGs. Indications were: suspected OSA (65%), obesity (13%), craniofacial malformations (5%), neuromuscular disease (4%), and other (13%) which included prematurity. 16% were recorded in hospitals and 84% at home. The mean age was 5.7 ± 5.8 years and 7.3 ± 4.5 years for the hospital and home groups, respectively. Interpretability was similar in both settings (87%). In the 68 subsequent PGs, interpretability was 84% when performed for follow-up and 96% when repeated for non-interpretability. Non-interpretability was predominantly due to a failure of the SpO2 channel. CONCLUSIONS: PG performed at home is both feasible and interpretable for a variety of indications. Non-interpretability was not predictable in association with the setting, anthropometric data, or indication, independently of the sequence (initial or subsequent PG) in which the parameters were analyzed.

Safely Decreasing Rigid Bronchoscopies for Foreign-Body Aspiration in Children: An Algorithm for the Emergency Department.

INTRODUCTION: Rigid bronchoscopy was traditionally performed in the management of foreign-body aspiration (FBA). More recently, since development of a less invasive method, flexible bronchoscopy has been proposed in some centers for the management of FBA. For the past few years, we have applied a decisional algorithm, privileging flexible bronchoscopy for diagnosis and, in some cases, for extraction of foreign body (FB). Our aims are first to analyze our current management of FBA and second to examine the bronchoscopic findings and complications. MATERIALS AND METHODS: Retrospective medical chart review of all patients with clinical suspicion of FBA who underwent bronchoscopy (flexible and/or rigid) from 2009 through 2014. RESULTS: An FB was found in 23 (33%) of the 70 patients included in the study (45 boys, 25 girls; median age: 21.5 months). Diagnosis of FBA was made on first intention in 22/23 (96%) and extraction was performed in 7/23 (30%) by flexible bronchoscopy. Rigid bronchoscopy was necessary for the extraction of the 16/23 (70%) remaining FBs. The rigid procedure was performed as first intention in only two (3%) patients, and one of the two was negative. Among the clinical signs of FBA, none were > 90% specific except for apnea (100%), but which was poorly sensitive (22%). Seven clinical and radiologic signs were found to be significantly different between FB+ and FB- groups: sudden choking, cyanosis, apnea, decreased breath sounds, atelectasis, mediastinal shift, and air trapping. Conversely, when none of these symptoms or signs and no clear history of sudden choking were present (in 15/70 patients), no FB was found. No life-threatening complications or death were observed. CONCLUSION: Our current management of FBA allows us to avoid almost all negative rigid bronchoscopies. In addition, we identified some symptoms and clinical and radiologic signs whose absence was highly predictive of negative bronchoscopy. We propose a novel algorithm for management of FBA that will help decrease the number of negative bronchoscopies.

Airway compression management in late-presenting absent pulmonary valve syndrome.

INTRODUCTION: Patients with absent pulmonary valve syndrome often present early with airway compression from aneurysmal pulmonary arteries. This study reviews our experience in managing absent pulmonary valve syndrome in later presenting children, and techniques used for managing airway compression. METHODS: This study is a retrospective chart review of all patients who underwent repair of absent pulmonary valve syndrome from 2000 to 2012 at our institution. The primary endpoints were post-operative bronchoscopic and clinical evidence of persistent airway compression and need for reinterventions on the pulmonary arteries. RESULTS: A total of 19 patients were included during the study period. The mean age at repair was 4.1±3.0 years (range 10 months-11 years). In all, seven patients had pre-operative bronchoscopic evidence of airway compression, which was managed by pulmonary artery reduction plasty in four patients and Lecompte manoeuvre in three patients. There were no peri-operative deaths. In patients with pulmonary artery plasty, two had no post-operative airway compression, one patient had improved compression, and one patient had unchanged compression. In patients managed with a Lecompte manoeuvre, two patients had no or trivial airway compression and one had improved compression. There were six late reinterventions or reoperations on the pulmonary arteries - two out of four in the pulmonary artery plasty group and one out of three in the Lecompte group. CONCLUSIONS: Most late-presenting patients with absent pulmonary valve syndrome do not have airway compression. Either pulmonary artery reduction plasty or the Lecompte manoeuvre can relieve proximal airway compression, without a significantly different risk of pulmonary artery reintervention between techniques.

Impact of clinical decision support guidelines on therapeutic drug monitoring of gentamicin in newborns.

BACKGROUND: Our institution's gentamicin dosing and therapeutic drug monitoring (TDM) practices for newborns were suspected to be very heterogeneous. Once-daily dosing (ODD) or extended-interval dosing (EID) and trough concentration measurement were recommended. Clinical decision support guidelines were developed and implemented as clinical decision support in the computerized prescriber order entry system. Impact on dosing, TDM practices, and blood sampling were evaluated. METHODS: A 1-year retrospective historically controlled study before (April 2008-March 2009) and after the implementation of guidelines (January 2010-December 2010) for newborns (<30 days of life) receiving gentamicin. Blood concentrations (% of peak concentrations sampled, % of patients with zero or one concentration sampled, % of trough concentrations ≤1 mg/L) and dose regimen (ODD/EID) were compared between groups. Factors potentially associated with gentamicin concentration were analyzed (multivariate analysis). RESULTS: One hundred thirty-two (postguidelines) versus 102 (preguidelines) patients were included (median gestational age: 34.3 versus 35.8 weeks, P > 0.05). After implementation of the guidelines, an ODD/EID regimen was almost exclusively used (97.7% versus 61.6%, P < 0.001), the percentage of peak concentrations (0.9% versus 17.2%, P < 0.001) and the number of blood samples per patient (87.1% having 0 or 1 concentration measured versus 48.0, P < 0.001) sharply reduced. A significantly higher percentage of trough concentrations were ≤1 mg/L (68.5% versus 33.0%, P < 0.001). The probability of a trough concentration ≤1 mg/L increased with an ODD/EID regimen (odds ratio, 7.23; 95% confidence interval: 3.48-15.0, P < 0.001) and in the postguidelines group (odds ratio, 2.02; 95% confidence interval: 1.01-4.02, P = 0.045). CONCLUSIONS: Guideline implementation generated a sharp reduction in blood sampling. Clinical benefits of better gentamicin dosing and TDM practices were evident. Cost-effectiveness and clinical benefit of reduced blood sampling should be evaluated.

Early blood exchange transfusion in malignant pertussis: a case report.

OBJECTIVE: To report early blood exchange transfusion in malignant pertussis and a favorable clinical outcome. SETTING: A pediatric intensive care unit in a tertiary hospital in Geneva, Switzerland. DESIGN: A descriptive case report. PATIENT: An 8-wk-old girl was diagnosed with malignant pertussis (extreme leukocytosis, seizures, pneumonia, and secondary severe hypoxic respiratory failure associated with pulmonary hypertension). After administration of a one-volume blood exchange transfusion, a rapid decrease in white blood cell count (from 119,000/mm(3) to 36,500/mm(3)) was observed and followed by clinical improvement and favorable outcome despite the initial presence of all described risk factors associated with a high mortality. CONCLUSION: The use of exchange blood transfusion early in the course of the disease might help to prevent a fatal outcome of malignant pertussis.

Bronchoscopic diagnosis of asymptomatic unilateral pulmonary vein atresia in an infant.

An eight-month-old boy with findings of persistent left pulmonary basal infiltrate was diagnosed with congenital unilateral pulmonary vein atresia by bronchoscopy. Cardiac catheterization documented slow left pulmonary venous return to atretic pulmonary veins. Conservative treatment was chosen because the child was asymptomatic and corrective surgery or percutaneous intervention was not technically possible. After a 3-year follow-up, the child still has no documented pulmonary hypertension. Early diagnosis of unilateral pulmonary vein atresia is important to anticipate potential threatening complications like pulmonary hypertension and hemoptysis. Surgical treatment of this entity might be drastic and complex and should be weighed against a conservative alternative and careful follow-up.

Measurement of nasal nitric oxide.

Nasal nitric oxide (nNO) is produced in high quantity in the upper airways. It is thought to be involved in host defence functions and regulation of mucociliary function, and to serve as a biochemical airborne transmitter. The measurement of nNO is easy and non-invasive. It has evolved as a screening test to exclude primary ciliary dyskinesia (PCD) in patients with suggestive symptoms, because nNO is extremely low in this condition. Nasal NO is also altered in other nasal, sinus and pulmonary pathologies, but is without diagnostic value outside of PCD.

Multiple pulmonary abscesses caused by Legionella pneumophila infection in an infant with croup.

Legionella infections are uncommon in childhood. We report the case of an infant who developed multiple pulmonary abscesses caused by Legionella pneumophila after receiving prolonged corticosteroid treatment of severe croup. Diagnosis was not suspected because immunosuppression was not initially considered. Caution should be used when prolonging high-dose corticosteroids in children with respiratory diseases.

Nasal nitric oxide measurements to screen children for primary ciliary dyskinesia.

STUDY OBJECTIVE: To examine the usefulness of exhaled and nasal nitric oxide (NO) measurements to detect primary ciliary dyskinesia (PCD) in children. DESIGN AND METHODS: The study population consisted of 34 children with symptoms suggestive of PCD who were previously referred to our pediatric university respiratory disease clinic for a diagnostic workup including analysis of ciliary structure and function by respiratory mucosal biopsy. PCD was diagnosed in 17 of the 34 children according to the ciliary biopsy results. Measurements of nasal and exhaled NO were performed according to European Respiratory Society and American Thoracic Society guidelines in the patients with and without biopsy-proven PCD, and also in 24 healthy age-matched subjects. RESULTS: Nasal NO was significantly lower in those children with proven PCD (geometric mean; 13.7 parts per billion [ppb]), compared to those who had negative biopsy results (132.7 ppb) and healthy control subjects (223.7 ppb). The measurement of nasal NO in our study population showed, below a cut-off level of < 105 ppb, a specificity of 88% for PCD, and positive predictive value of 89%. Nasal NO above a cut-off level of 105 ppb excluded PCD with a 100% certainty. The lower levels of exhaled NO in patients with PCD did not reach statistical significance. CONCLUSION: The measurement of nasal NO appears to be a useful tool to screen children for PCD and to exclude this disease in those with high nasal NO levels.