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Obstructive sleep apnea syndrome (OSAS) in children: diagnostic challenges.

Obstructive sleep apnea syndrome (OSAS) is increasingly recognized in the pediatric population. It is characterized by a combination of partial upper airway obstruction and intermittent obstructive apnea that disrupts normal ventilation and sleep. It is estimated to occur in 1-3% of children with a peak age of 2 to 5 years. Common symptoms include habitual snoring, difficulty breathing during sleep, restlessness, and witnessed apnea. Adenotonsillar hypertrophy is the most common associated condition in otherwise normal children, but cranialfacial abnormalities, neuromuscular diseases, and obesity are also predisposing factors. Severe OSAS can have serious neurobehavioral and cardiorespiratory consequences including excessive daytime sleepiness, growth failure, school failure, behavioral problems, cor pulmonale, or even death. Diagnosis is based on data from the history, physical exam, and laboratory studies that confirm the presence and severity of the upper airway obstruction. Polysomnography has been the diagnostic tool of choice. Treatment depends on the severity of symptoms and the underlying anatomic and physiologic abnormalities. Since childhood OSAS is usually associated with adenotonsillar hypertrophy, the majority of cases are amenable to surgical treatment. However, there is increasing pediatric experience with CPAP therapy when tonsillectomy and adenoidectomy are either unsuccessful or inappropriate.

Adenoidectomy↗

Depressed baroreceptor reflex in patients with obstructive sleep apnea (OSA).

In this study we evaluated the cardiovascular autonomic function in twenty-five OSA patients and in twenty-five control healthy subjects, by computerized spectral analysis of R-R interval variation at rest and during orthostatism by head-up tilt maneuver to up-right position (80 degrees) as a sympathetic provocation. The results of our study show that most patients affected by OSA have a sympathetic overactivity and a decreased baroreflex response in comparison with normal subjects. The method here described is simple, objective and very sensitive and may be utilized to discover early signs of an autonomic dysfunction consequent to OSA leading to cardiovascular complications of the late stage of the disease.

Baroreflex↗

Michels syndrome, Carnevale syndrome, OSA syndrome, and Malpuech syndrome: variable expression of a single disorder (3MC syndrome)?

We report on a 3-year-old girl with Michels syndrome, a rare condition characterized by craniosynostosis, blepharophimosis, ptosis, epicanthus inversus, cleft lip/palate, abnormal supra-umbilical abdominal wall, and mental deficiency. The phenotypic findings are compared with the six previously reported Michels cases, and with patients referred to as Carnevale, OSA, and Malpuech syndromes. Michels syndrome is characterized by cleft lip and palate, anterior chamber anomalies, blepharophimosis, epicanthus inversus, and craniosynostosis. Carnevale syndrome shows hypertelorism, downslanting palpebral fissures, ptosis, strabismus synophrys, large and fleshy ears, and lozenge-shaped diastasis around the umbilicus. OSA syndrome resembles Carnevale, with humeroradial synostoses, and spinal anomalies as extra features. Malpuech syndrome shows IUGR, hypertelorism, cleft lip and palate, micropenis, hypospadias, renal anomalies, and caudal appendage. All are autosomal recessive. Despite the presence of apparently distinctive key features, it appears that these four entities share multiple similarities in the facial Gestalt and the pattern of MCA. Those similarities lead us to postulate that they belong to the same spectrum, which could be referred to as "3MC syndrome" (Malpuech-Michels-Mingarelli-Carnevale syndrome).

Abnormalities, Multiple↗

STEALTH liposome-encapsulated cisplatin (SPI-77) versus carboplatin as adjuvant therapy for spontaneously arising osteosarcoma (OSA) in the dog: a randomized multicenter clinical trial.

PURPOSE: This trial was designed to compare the efficacy of adjuvant STEALH liposome-encapsulated cisplatin (SPI-77) to "standard-of-care" carboplatin therapy in dogs with osteosarcoma (OSA) in the context of a randomized study design. METHODS: The study included 40 pet dogs with spontaneously arising OSA which were randomized to receive SPI-77 (350 mg/m(2) i.v. every 3 weeks for four treatments) or carboplatin (300 mg/m(2) i.v. every 3 weeks for four treatments) along with amputation of the affected limb. Median disease-free (DFS) and overall survival (OS) were compared using standard life-table analysis. RESULTS: The median follow-up was 693 days (range 321-730 days). Of 38 dogs eligible for follow-up, 25 were dead of their disease, 9 were alive and disease-free (8 receiving SPI-77, 1 receiving carboplatin; P=0.02), 2 were free of disease when they were lost to follow-up at 321 and 395 days, and 2 had died of an unrelated disease. The median DFS times for dogs treated with SPI-77 and carboplatin were 156 and 123 days, respectively ( P=0.19). The median OS times for dogs treated with SPI-77 and carboplatin were 333 and 207 days, respectively ( P=0.18). CONCLUSIONS: While STEALTH liposome encapsulation of cisplatin allowed the safe administration of five times the maximally tolerated dose of free cisplatin to dogs without concurrent hydration protocols, this did not translate into significantly prolonged DFS or OS. However, a larger proportion of dogs receiving SPI-77 enjoyed long-term DFS when compared with dogs receiving carboplatin.

Amputation, Surgical↗

[Why is obstructive sleep apnea (OSA) a cardiovascular risk factor?].

Patients with obstructive sleep apnea (OSA) frequently suffer from cardiovascular diseases. Mechanisms like intrathoracic pressure variations, changes in blood gases (hypoxia), arousals and neurohumeral adaptation mechanisms, combined with breathing disorders are causing these cardiovascular sequelae. In particular repetitive hypoxemia and activation of the sympathetic nervous system have to be considered as stressors for the cardiovascular system. Special clinical findings should take OSA into consideration as a differential diagnosis. A systematic anamnesis with questions to daytime conditions (hypersomnia, decrease of performance), snoring and apneas while sleeping is easy to ascertain, and will lead to the correct diagnosis in more than 90% of cases. The extent and need for therapy should be assessed by three criteria: 1) daytime symptoms, 2) the extent of breathing disorder and 3) cardiovascular comorbidity.

Arousal↗

Parallel changes in resting muscle sympathetic nerve activity and blood pressure in a hypertensive OSAS patient demonstrate treatment efficacy.

Studies comparing the efficacy of continuous positive airway pressure (CPAP) versus surgery in correcting cardiovascular abnormalities in OSAS are lacking. We describe an OSAS patient with hypertension who responded favorably to CPAP treatment, whereas subsequent uvulopalatopharyngoplasty (UPPP) was less successful. While CPAP markedly lowered daytime muscle sympathetic nerve activity (MSNA) and blood pressure (BP), findings after UPPP were comparable to pre-treatment baseline. Thus, parallel changes in MSNA and BP demonstrated treatment efficacy.

Blood Pressure↗

Nasal breathing and continuous positive airway pressure (CPAP) in patients with obstructive sleep apnea (OSA).

Although nasal continuous positive airway pressure (nCPAP) treatment is the most efficient therapy for obstructive sleep apnea (OSA), compliance with therapy is poor because of several side effects. Among these adverse effects some are related to the reactions of the nose to nCPAP which are briefly described. In a long-term survey of 109 OSA patients, 80.6% continued nCPAP for at least 3 years until the last follow-up. Follow-up ranged between 0.8 and 109 months with a mean time of 43 (SD +/- 24.7) months. Among all patients treated with nCPAP, only 46.6% met our criteria for long-term compliance, defined as a mean use of the CPAP machine for at least 5 hours per night. Within the objective measures such as age, body mass index, neck circumference, nasal airflow, apnea-hypopnea index, minimal oxygen saturation, and level of nCPAP pressure, no predictive factor for daily use of nCPAP could be found. However, pretherapeutic daytime somnolence (measured by visual analogue scale and Epworth Sleepiness Scale) and its improvement obtained from the therapy showed a significant effect on the daily use of the CPAP machine.

Body Mass Index↗

Possible cause of excess sludge reduction in an oxic-settling-anaerobic activated sludge process (OSA process).

Modification of a conventional activated sludge process by inserting a sludge holding tank in a sludge return line forms an oxic-settling-anaerobic (OSA) process that may provide a cost-effective way to reduce excess sludge production in activated sludge processes. In this paper we systematically evaluate the following possible scenarios that may explain the reduction of excess sludge in the OSA process: (i). energy uncoupling, (ii). domination of slow growers, (iii). soluble microbial products (SMPs) effect and (iv). sludge decay in the sludge holding tank under a low oxidation-reduction potential (ORP) condition. Results show that only the final scenario may reasonably explain this reduction. It has also been found that the sludge decay process in the sludge holding tank may involve the reduction of the cell mass.

Bacteria, Anaerobic↗

The effect of adenotonsillectomy in children with OSA.

Adenotonsillar hypertrophy and abnormal facial morphology are thought to be important for the occurrence of obstructive sleep apnea syndrome (OSA). We evaluated the effects of adenidectomy and/or tonsillectomy and the relationship between the treatment results and facial morphology in 134 children with OSA. Significant improvements in apnea-hypopnea indes (AHI) and lowest blood oxygen saturation (SaO2) were noted and 78.5% of the patients improved after adenoidectomy and/or tonsillectomy. Additional operations were needed in two out of 13 cases of the adenoidectomy group and two out of four cases of the adeno-monotonsillectomy group. In the adenotonsillectomy group, the unimproved children tended to have smaller tonsils, narrower epipharyngeal airspace, and more poorly-developed maxillary and mandibular protrusion than the improved children.

Adenoidectomy↗

nCPAP treatment interruption in OSA patients.

This study has investigated the effect of interrupting nasal continuous positive airway pressure (nCPAP) therapy on 10 obstructive sleep apnoea (OSA) patients (nine male, one female) (53.6 +/- 7.3 years) treated over 2 years. The effect of nCPAP interruption was determined by variations in sleep counts and gasometric values during five consecutive nights. On the first night, the patient used his habitual nCPAP. On the remaining nights, nasal nCPAP was not applied. The apnoea-hypopnoea index (AHI) was found to increase significantly in the second night, attaining a similar level to that of the basal study (2 years ago). SaO2 minimum decreased and PaCO2 increased in the second night with respect to the first night. The interruption of nCPAP therapy in OSA patients treated over a long period of time increases the sleep counts and impairs the gasometric parameters. Consequently, any change in nCPAP time therapy must be checked to avoid negative effects.

Carbon Dioxide↗

OSAS in children: clinical and polysomnographic respiratory profile.

UNLABELLED: Obstructive sleep apnea and hypopnea syndrome in children (osas) has an estimated prevalence of up to 3% and can be associated with neurocognitive and behavioural abnormalities, and also cardiovascular complications. This study may help pediatricians, who are unaware of the problem, to recognize osas. STUDY DESIGN: series of cases. AIM: to describe the clinical characteristics and polysomnographic respiratory findings in a population of children with obstructive sleep apnea and hypopnea syndrome referred to the sleep laboratory from january 2002 up to july 2003. METHODS: we studied 93 patients between 2 and 10 years of age with polysomnographic diagnosis of obstructive sleep apnea and hypopnea syndrome. Age, gender, racial group and questions about the childrens health and sleep related disorders were evaluated. Apnea-hypopnea index, oxyhemoglobin desaturation, and arousal index were evaluated too. RESULTS: males represented 61.3%, With a mean age of 5.2+/-2.1 (Years-old). The complaints that most commonly lead to the exams were snoring in 24.7% And restless sleep in 24.7%. Associated medical conditions frequently reported were allergic rhinitis (98.9%) And adenoid hypertrophy (50.6%). Mild apnea was found in 66%. The mean and sd of spo2 nadir was 89.1+/-3.5% And the mean and sd of the number of arousals was 8.4+/-3.5/Hour of sleep. CONCLUSION: the results suggest the possibility that obstructive sleep apnea and hypopnea syndrome should be suspected in children with allergic diseases and adenoid and tonsil hypertrophy with snoring and restless sleep complaints.

Adenoids↗

Proposed cephalometric diagnosis for osteogenic obstructive sleep apnea (OSA): the mandibular/pharyngeal ratio.

The purpose of this laboratory study is to devise a simple, diagnostic test to assess possible osteological deficiency as a probable cause of OSA. Modern day skulls of fifty males and forty eight females of Tuscan origin and an additional seventy-five skulls, from India, with their cephalograms were used for this study. Mandibular length and antero-posterior dimension of the nasopharynx--pharyngeal tubercle (PhT) to posterior nasal spine (PNS)--were measured on the Tuscan skulls. The nasopharynx was similarly measured on the Indian skulls and readings multiplied by 1.14, the magnification factor of the cephalometric apparatus used. The PhT-PNS distance was then plotted on the cephalogram of Indian skulls with point PhT at the basiocciput. The possible presence of an osteogenic etiology of OSA can be determined by comparison of the mandibular/pharyngeal ratio obtained from the skull cephalograms to that of the individual patient.

Adolescent↗

Vascular function in OSA.

A number of pathophysiological phenomena linked to sleep disordered breathing are likely to affect vascular function. This report briefly reviews current knowledge regarding neurogenic vascular tone and a number of circulating hormones with vascular actions in obstructive sleep apnea (OSA). New evidence suggesting a role of the vascular endothelium in the development of vascular disease in OSA is also presented.

Aldosterone↗

Using self-reported questionnaire data to prioritize OSA patients for polysomnography.

Many laboratories have large numbers of patients with suspected obstructive sleep apnea (OSA) waiting to be tested. We assessed the use of simple clinical data to detect those patients with an apnea index <20 (low AI) who could be studied less emergently. Using questionnaires completed by patients prior to evaluation, we collected data on 354 consecutive patients (281 males, 73 females; mean age 48.6 years) referred for OSA and assessed with polysomnography (PSG). The questionnaires included the Epworth sleepiness scale (ESS), height, weight, age, and a history of observed apnea. Analysis of receiver operating characteristics curves revealed that both body mass index (BMI) [area under curve = 0.7258, standard error (SE) = 0.03, p < 0.01] and ESS (area under curve = 0.5581, SE = 0.03, p = 0.03) were significantly better than chance alone in detecting people with AI < 20. ESS < or =12 was found in 37.9% of the subjects but 39.6% of those expected to have a low AI using ESS had an AI > or =20. A BMI < or =28 was found in 24.9% of the subjects; 14.8% of those expected to have a low AI using BMI had an AI > or =20. Combining these variables improved accuracy but resulted in smaller groups; a cut-off of ESS < or =12 and BMI < or =28 resulted in a group of 33 (9.3% of subjects), only two (6%) of whom were falsely called low AI. Adding to this the fact that apnea had not been observed resulted in a group of nine patients (2.5% of subjects), none of whom had an AI > or =20. Thus there is a tradeoff; the more variables used, the greater the accuracy but the smaller the percent of cases selected to have low AI. However, in laboratories with hundreds of patients waiting to be tested, any procedure better than chance to help prioritize patients seems worthwhile.

Appointments and Schedules↗

OSa, a 'new' low-frequency red cell antigen.

A new low-frequency red cell antigen, OSa, in a Japanese blood donor is inherited as a Mendelian dominant. A family study showed that OSa is not controlled by ABO, P1, Rh, Kidd, or Diego loci, nor is the antigen X- or Y-borne.

Antigen-Antibody Reactions↗

Expression of tumour markers CA125, CASA and OSA in minimal/mild endometriosis.

Ovarian cancer associated antigens CA125, CASA and OSA were measured in serum from 23 patients with mild endometriosis before, during and after medical therapy. Pretreatment CA125 levels were elevated above 35 mu/ml in 4(17%), and above 25 mu/ml in 7 (30%) patients. Mean CA125 levels decreased during treatment, but in only 10 patients did levels reflect disease response. There was no correlation between CA125 levels and disease severity as measured by modified American Fertility Society Scoring. Neither CASA nor OSA were detected in these women.

Antigens, Neoplasm↗

Reduced genioglossal activity with upper airway anesthesia in awake patients with OSA.

We examined whether topical upper airway anesthesia leads to a reduction in genioglossal (GG) electromyogram (EMG) in patients with obstructive sleep apnea (OSA). Airway mechanics were also evaluated. In 13 patients with OSA, we monitored GG EMG during tidal breathing and during the application of pulses of negative airway pressure (-10 to -12 cmH(2)O). Airflow resistance and airway collapsibility were determined. All measurements were performed with and without topical anesthesia (lidocaine). Anesthesia led to a significant fall in the peak GG EMG response to negative pressure from 36.1 +/- 4.7 to 24.8 +/- 5.3% (SE) of maximum (P < 0.01). This was associated with a fall in phasic and tonic EMG during tidal breathing (phasic from 24.4 +/- 4.1 to 16.4 +/- 3.4% of maximum and tonic from 10.9 +/- 1.6 to 8.0 +/- 1.3% of maximum, P < 0.01). A significant rise in pharyngeal airflow resistance was also observed. Our results demonstrate that topical receptor mechanisms in the nasopharynx importantly influence dilator muscle activity and are likely important in driving the augmented dilator muscle activity seen in the apnea patient.

Airway Resistance↗

Initiation of CPAP therapy for OSA: does prophylactic humidification during CPAP pressure titration improve initial patient acceptance and comfort?

BACKGROUND: Heated humidifiers (HH) enable effective treatment of upper airway dryness during nasal continuous positive airway pressure (nCPAP) therapy for obstructive sleep apnoea (OSA), but the role of prophylactic use of HH during the initiation of nCPAP treatment has not been studied so far. OBJECTIVES: The aim of the present study was to investigate whether prophylactic HH during the initiation of CPAP would result in improved initial patient comfort and acceptance. METHODS: In 44 consecutive, previously untreated OSA patients with no history of upper airway dryness, CPAP titration with and without HH was performed on two consecutive nights in a randomised order. The patients were interviewed after each treatment night in order to establish the comfort of the treatment, and, after the second treatment, they were asked which of the two nights they considered more pleasant, and which treatment they would prefer for long-term use. RESULTS: Following CPAP titration with HH, 32 patients (73%) claimed to have had a better night's sleep than usual (i.e. without CPAP treatment) compared with 33 patients (75%) saying the same following CPAP treatment without HH. For 21 patients (47.7%) treatment with HH was more pleasant, 23 (52.3%) saw no difference or said that treatment without HH was more pleasant. Nineteen patients (43.2%) gave preference to treatment with HH for long-term use, while 25 patients (56.8%) had no preference or said they would prefer treatment without HH. CONCLUSIONS: The use of HH during the initiation phase of CPAP treatment was associated neither with an initial improvement in comfort nor with greater initial treatment acceptance.

Adult↗