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ResumenLas malformaciones craneofaciales (secuencia de Pierre Robin, síndrome de Threacher- Collins, síndrome de Nager, etc.) con frecuencia van asociadas a hipoplasia mandibular grave, que puede causar obstrucción de la vía aérea superior por retroposición de la base de la lengua dentro del espacio faríngeo posterior.La mayoría de los pacientes responden al tratamiento postural, en decúbito prono, puede ser necesario controlar la saturación de oxígeno, insertar un tubo nasofaríngeo e incluso intratraqueal. En casos más graves con pausas prolongadas y frecuentes de apnea, la traqueostomía puede ser necesaria, pero se asocia a una alta morbilidad y, ocasionalmente, mortalidad.En los últimos 2 años, en la Unidad Multidisciplinaria de Labio y Fisura Palatina del Hospital Virgen de las Nieves de Granada, se ha tratado a 4 niños con apnea obstructiva grave secundaria a hipoplasia mandibular grave mediante distracción mandibular osteogénica, y este procedimiento se ha mostrado eficaz en la resolución del problema. Ha evitado la traqueostomía y se ha elongado la mandíbula en el plazo de 3–4 semanas. En este tiempo han desaparecido los problemas respiratorios obstructivos, así como también de la deglución, y los resultados estéticos obtenidos han resultado excelentes y las complicaciones, por el momento, mínimas.AbstractCraniofacial malformations (Pierre-Robin sequence, Treacher-Collins syndrome, Nager syndrome, etc.) are frequently accompanied by severe mandibular hypoplasia, which can cause upper airway obstruction due to retroposition of the base of the tongue in the posterior pharyngeal space.The majority of patients respond to postural treatment in decubitus prono. It may be necessary to monitor oxygen saturation and insert a nasopharyngeal or even an endotracheal tube. Tracheostomy may be necessary in more serious cases with long and frequent apnea pauses, but it is associated with high morbidity and occasional mortality. In the last two years, four children with severe obstructive apnea secondary to mandibular hypoplasia were treated by means of “osteogenic mandibular distraction” in the multidisciplinary Cleft Palate Department of Virgin de las Nieves Hospital (Granada, Spain). This procedure effectively resolved the problem, making tracheostomy unnecessary and lengthening the jaw within 3–4 weeks; in this period, obstructive respiratory problems and swallowing difficulties disappeared. The aesthetic results were excellent and the complications so far have been minimal.ObjectiveTo present the results of a patient series with several types of POP treated using the same approach and operation.

We describe a child with sickle cell anemia and multiple ischemic infarctions who was found to have severe obstructive sleep apnea and hypoxemia, secondary to adenotonsillar enlargement. The apnea-associated hypoxemia likely contributed to the development of the strokes in this child. Moreover, because stroke in patients with sickle cell anemia, and maximal tonsillar enlargement (the most common cause of obstructive apnea in children) both have peak incidence at the same age (6-7 years), obstructive sleep apnea may be an important factor in the development of stroke in other children with sickle cell anemia.

The objective of this paper is to provide some clinical and technical information about electronic monitoring of various physiologic parameters in infants at risk for the sudden infant death syndrome. Although they are very important home monitoring and transducers problems will not be discussed.

A portable, easily assembled system that allows quantitative monitoring of ventilation in the sleeping human subject is described. It is a modification of the system used by Kinney et al. (Metab. Clin. Exp. 13: 205-211, 1964) and Spencer et al. (J. Appl. Physiol. 33: 523-528, 1972). The system directly measures gas flow without reliance on indirect CO2 or temperature sensors or on chest wall motion. It is comfortable and reflects the tidal volume with an accuracy of 92%. It also allows measurement of oxygen consumption and CO2 production. The gas breathed by the subject can be varied in composition. Two illustrative examples of the use of the system are given: 1) a case of obstructive apnea; and 2) a case in which a drop in arterial oxygen saturation is explained by quantified hypoventilation undetected by a nasal CO2 probe.

Evaluation of 32 individuals with achondroplasia revealed that 28% had a history of apnea and 22% had respiratory abnormalities on polysomnography. In those patients requiring posterior fossa Decemberompressive surgery, improvement was noted in follow-up polysomnograms. Multimodality studies suggested that brainstem compression was common in achondroplasia and could account in part for the abnormal respiratory function in this disorder, including obstructive apnea, central apnea, and hypoxemia.

A 7-week-old infant with severe sleep apnea underwent polysomnography that revealed as many as 455 obstructive apneas per night; the apneic episodes had a mean duration of 34 s. A growing tumor in the neck, a parapharyngeal cystic hygroma, was discovered and surgically removed. The infant's condition improved dramatically, and a follow-up polygraphic recording was normal. During the following 10 months the child's condition remained stable. The case reported illustrates a rare cause of severe sleep obstructive apnea. It also reinforces the need for a complete medical approach to the diagnosis and treatment of obstructive sleep apnea in infants.

Publisher Summary This chapter explores interactive effects of central and obstructive apnea. Irregularly recurring apneic periods lasting for more than ten seconds appear during sleep in one-third of normal individuals. The apneas are of two types: (1) central and (2) obstructive. Central apnea is characterized by an absence of airflow past the nose and mouth because of a cessation of respiratory efforts. In obstructive apnea, despite persistent respiratory effects, airflow ceases because of total upper airway obstruction. This study presents a mathematical model of the chemical control of breathing, which examines the hypotheses that instability in the negative feedback chemical control system, which regulates breathing, can result in central apnea during sleep. The results suggest that the model reasonably reproduces many of the features of the pattern of breathing seen in patients with recurrent sleep apnea and support the contention that obstructive and central apneas are interrelated and depend on variations in the effect of chemical drive on chest wall and upper airway muscles.

From inspection of breathing patterns recorded for 12 hours at monthly intervals on 100 infants, we determined that the longest episode of normal apnea, almost invariably following a sigh, was 14 sec. Therefore, we define abnormal apnea as an episode of 15 sec. or greater.

The site of upper airway obstruction during mixed and obstructive apnea was investigated in nine preterm infants with clinical apnea. The level of obstruction was determined using a saline-filled pharyngeal catheter that detects respiratory pressure changes. High pharyngeal obstruction was found in 87 of 94 spells. These data suggest that the mechanism of upper airway obstruction in infants with apnea may be similar to that seen in older children and adults with obstructive sleep apnea.

Twenty-nine near miss for sudden infant death syndrome and thirty normal infants between the ages of 3 weeks and 6 months were monitored polygraphically for 24 hours. The distribution of sleep and abnormal respiratory events were analyzed for both groups. On the basis of mixed and obstructive apnea, 12-hour nocturnal segments (8 pm to 8 am) consistently distinguished near miss from normal infant groups between the ages of 3 weeks and 4.5 months. Daytime naps do not provide statistical differences sufficient to differentiate between the two groups. During sleep, abnormal respiratory events are more likely to occur between 1 am and 6 am, at least 40 minutes after sleep onset. Respiratory pauses show a significant increase just prior to waking (a strong respiratory stimulus). Any impairment of the arousal threshold during sleep will place near miss infants at increased risk.

Eight children with obstructive apnea were evaluated by transnasal fiberoptic endoscopy. Three children had obstructive adenoids and tonsils, 2 had pharyngeal collapse, 1 had an obstructive tongue, and 1 had a subglottic cyst. The remaining patient had no abnormality. The results of transnasal endoscopy influenced therapeutic interventions.

Brief end-expiratory airway occlusions were performed in 22 preterm babies, 17 with and 5 without clinical apnea, and 4 full-term babies, 1 with Pierre-Robin syndrome. Airway stability was evaluated by comparing pressures measured simultaneously in the chest and nasal passages during occluded inspiratory efforts. The airway remained patent throughout all 301 trials in 20 babies during rapid-eye-movement (REM) and quiet sleep. Airway closure occurred during 31/102 trials in 6 babies (5 preterm and 1 term with Pierre-Robin syndrome), more commonly in quiet than in REM sleep. Overall and within individuals, mean closing pressures were significantly lower than the mean maximum falls in airway pressure recorded during occlusions without closure. Mixed-obstructive and obstructive apnea was significantly more frequent in babies with airway closure than in those without (5.3 +/- 4.0 vs. 0.4 +/- 0.8 episodes/h). Pauses in breathing greater than or equal to 3 s occurred during 28% of occlusions in preterm infants and 2% of occlusions in full-term babies. There was no significant difference between the mean frequency of pauses during occlusion and during the preceding control period or in the incidence of pauses in occlusions with vs. those without closure. It is concluded that the airway of most preterm and full-term babies is remarkably stable under load. Intermittent closure occurs in certain infants and may be related to airway muscle dysfunction.

Gastroesophageal reflux is common in infants and children. In most cases it causes little more than inconvenience and remits spontaneously with time and maturation. A small and select group of refluxing children, however, will develop complications of reflux severe enough to justify operative control when medical treatment fails. Recurrent pulmonary infections, obstructive apnea, nutritional wasting, and progressive inflammatory injury to the esophagus all qualify as surgical indications, provided a reasonable cause-effect relationship with reflux can be established. The procedure of choice depends very much upon the skill and experience of the surgeon. The complete fundoplication seems to offer more complete control of reflux, but it has the potential for more frequent and more complicated side-effects.

We examined the rate of fall of arterial O2 saturation (dSao2/dt) after apnea onset in four spontaneously breathing adult male baboons. We postulated that a lower mixed venous O2 saturation (Svo2) would steepen dSao2/dt by more rapid depletion of alveolar O2. Single isolated (NREP) and five or more sequential repetitive apneas (REP) were created by clamping an indwelling cuffed endotracheal tube at end expiration. Fiberoptic catheters were used for continuous monitoring of Sao2, Svo2, and cardiac output. The mean dSao2/dt for all duration NREP apneas was 0.60%/s. Mean dSao2/dt increased above base line for each consecutive REP apnea and was higher in 60 s than in 45 and 30 s REP apnea series. The increase in dSao2/dt corresponded closely with the fall in preapneic Svo2. Preapneic arterial O2 content fell during successive REP apneas but the maximal decrement from base line (1.3 ml/dl) was much less than the maximal decrement in preapneic mixed venous O2 content of 5.1 ml/dl. Preapneic cardiac output for NREP apneas and nadir cardiac output for REP apneas remained constant. Nadir cardiac output for NREP apneas showed higher values for longer duration apneas. We concluded that dSao2/dt is inversely related to preapneic Svo2.

The sleep apnea syndrome (SAS) is characterised by transient respiratory arrest occurring as soon as the patient falls asleep or as his sleep deepens. Three types of sleep apnea are usually distinguished: central apnea, characterised by an absence of respiratory gas-flow and a complete cessation of the activity of the respiratory muscles; obstructive apnea, i.e. absence of respiratory gas-flow despite the persistence of thoraco-abdominal respiratory movements; mixed apnea, which is a combination of central and obstructive apneas, the former always preceding the latter (12, 8, 11, 17, 31, 24). It is now recognised that these sleep related respiratory arrests may occur in patients with or without diurnal hypersomnolence, alveolar hypo-ventilation, pulmonary and systemic arterial hypertension or polycythemia. This clearly shows the relationships between the sleep apnea syndrome and the Pickwickian syndrome (5).

• A case of life-threatening sleep apnea syndrome secondary to irradiation-induced fibrosis of the neck occurred. The diagnosis was made with the use of relatively simple monitoring techniques. Obstructive apnea of this origin is potentially fatal yet remediable. (Arch Otolaryngol1981;107:629-630)

Central neural drive to the respiratory muscles can be assessed by a variety of methods. These include measurement of respiratory nerve and muscle electrical activity, measurement of the pressures generated by the contraction of respiratory muscles, and measurement of the final output of the respiratory system––ventilation. We have assessed these output parameters in normal subjects during CO2 rebreathing with and without externally applied loads and during sleep in patients with sleep disorders.

Concordance for Goldenhar anomaly has been described in monozygotic (MZ) twins on 2 occasions but never in dizygotic (DZ) twins. In both cases the twins were similarly, although not identically, affected. We report on a pair of probably monozygotic twins (8% probability of DZ) who presented with extremely diverse manifestations of this anomaly complex. One of them required a tracheostomy because of obstructive apnea due to severe micrognathia and subsequently died. This twin had a midline lower lip cleft which has not previously been described in the Goldenhar anomaly.

Obstructive apnea during sleep is accompanied by intermittent hypoxia (IH) leading to hypertension and other cardiovascular disturbances. A comparative evaluation of long-term effects of the neonatal IH on the cardiovascular functions was performed in normotensive Sprague-Dawley and spontaneously hypertensive rats (SHR). The newborn rats were placed for 30 days to conditions of IH (8% and 21% O2, alternating every 90 s for 12 h/day). Control groups of rats were constantly kept in normoxia. By 6 months, in the spontaneously hypertensive rats exposed to IH at the period of wakefulness there was a statistically significant increase (as compared with control) of the systolic (185.8 ± 1.7 and 169.9 ± 1.4 mm Hg, correspondingly, p < 0.010 and the diastolic pressure (96.2 ± 4.9 and 86.0 ± 2.6 mm Hg, correspondingly, p < 0.01). During sleep, the systolic and diastolic pressure in these rats was higher than in control animals by 10 mm Hg (p < 0.01) and 12 mm Hg (p < 0.01), its decrease during sleep being absent. In SHR submitted to IH there was an increase in the power ratio of the heart rate variability from 0.9 ± 0.15 to 1.5 ± 0.17, which indicates a shift of the sympathico-parasympathetic balance in this group towards predominance of the sympathetic component. In the Sprague-Dawley rats exposed to neonatal hypoxia, the above-indicated changes were not prominent. These peculiarities of the hypertensive rats allow establishing connection of the genetic factor with the sympathetic mechanism providing long-term consequences of the neonatal IH for the cardiovascular control in the SHR.

MIKI, H., HIDA, W., INOUE, H. and TAKISHIMA, T. A New Treatment for Obstructive Sleep Apnea Syndrome by Electrical Stimulation of Submental Region. Tohoku J. exp. Med., 1988, 154 (1), 91-92 - Loss of upper airway muscle tone during sleep has been thought to be responsible for obstructive apnea episodes. To examine the effect of electrical stimulation of the submental region on sleep apnea episodes, patients with obstructive sleep apnea syndrome were polysomnographically studied during all-night session with and without stimulation.

To the Editor.— It was with interest that we read the article on obstructive apnea in human infants by van Someren and Stothers.1 We would, however, like to correct some of the misconceptions concerning our work. We did not find that "the only difference between `near miss SIDS infants' and control groups was an increased frequency of short 3- to 6-second periods of mixed and obstructive apnea in the high risk group." As written in Pediatrics,2 we found that "the total number of mixed and obstructive apnea greater than 3 seconds during total sleep time, between 3 weeks and 4½ months statistically differentiated the two groups."

In Reply. —Shore and Millman, together with Sharp et al, 1 have observed three patients with central sleep apnea that converted to obstructive apnea with acetazolamide administration. This phenomenon has several possible explanations. First, as suggested by Sharp et al, 1 the subclassification of apneas into obstructive and central may be inappropriate, as a patient's condition may convert from one form of apnea to another with changes in central respiratory drive. This, however, is not always the case, as our six patients demonstrated. A second explanation might be that the three patients of Shore and Millman and Sharp et al had a predisposition to obstructive apnea, with decreased upper-airway caliber. 2 As a result, with the increased respiratory drive produced by acetazolamide, greater inspiratory pressure developed, causing upper-airway collapse and obstruction. Finally, these three patients may originally have had obstructive sleep apnea. Over time, the recurrent hypoxia and hypercapnia resulting

Sleep apnea syndrome (SAS) often presents a difficult therapeutic problem to the clinician since many of the accepted modes of therapy are associated with only partial success or with a number of long-term complications. We present three patients with obstructive SAS in whom dramatic clinical improvement occurred following repair of a deviated nasal septum. The subjective improvement was associated with a diminution in the number and duration of obstructive apnea episodes as observed during a standard sleep study. Because of its simplicity and low rate of complication, we propose that repair of a deviated nasal septum be further evaluated as a mode of therapy for this condition.

In Reply.— While we agree with Shaw that nasal obstruction is one possible cause of sudden infant death syndrome (SIDS), the purpose of our article was to highlight the pitfalls in diagnosing obstructive apnea. Implicit in his letter is a criticism of the direction of SIDS research over the past few years: countless hours of "respiratory" monitoring of questionable physiologic quality and analysis of dubious meaning, usually followed by complex statistical comparisons of little clinical import. We share this view and hope that research along such lines will cease shortly to make demands on already strained financial resources.

To evaluate the effects of inspiratory flow resistance (RI) on ventilatory response, 7 infants recovering from RDS were studied: Mean wt. 1.6 kg, age 21 days, gest. age 32 wks. Control tidal vol. (TV), min. vent. (MV), peak insp. flow (VI), insp. time/tot. resp. time (TI/TTot), resp. freq. (f), dynamic compliance (CL) and PACO2 were obtained before applying a 250 or 450 cm H20/L/sec. RI. Studies were repeated immediately, 5 and 10 minutes following application of RI. Infants were breathing room air throughout the study. Mean ± SEM control values were: TV=14.4±6.5 ml, MV=804.8±313.8 ml/min, VI=2.13±1.08 L/min, TI/TTot=.44±.03, f=53.66±10.91, CL=3.46±1.48 (n=5), PACO2=41.1±4.5 mmHg. Following application of a RI of 450 cm H2O/L/sec there was a decrease immediately and at 5 and 10 minutes for: TV(ml)=7.0±3.8 (p

• Fetal alcohol syndrome is a well-known entity, but its association with upper airway anomalies has not been established. We describe three patients, all of whom had fetal alcohol syndrome and upper airway obstruction. Recognition of this problem is important, as it may help to prevent serious complications such as obstructive apnea, sudden infant death syndrome, or pulmonary hypertension in these already compromised infants. (AJDC1986;140:1039-1041)

We performed diagnostic monitoring (heart rate, abdominal excursion, nasal and oral airflow) on infants referred because of apnea and bradycardia (HR≤100) spells. By classifying apnea (absent airflow ≥20sec or 2sec central plus obstructive), we identified 5 preterm infants (GA:26-32wks; postnatal age: 6-34days) with predominantly OA and MA. We used bedside monitoring for bradycardia to determine frequency of apnea pre and post THEO since 99% of all apneic spells were associated with bradycardia and 99% of bradycardias were preceded by apnea by our diagnostic monitoring. All infants received THEO (6mg/kg then 2mg/kgTID; serum levels: 6-13 μg%). All infants had a decrease in the frequency of bradycardia spells in the 48hr period post vs. pre THEO (p

In Reply.— We were interested in the comments of Guilleminault et al and were pleased to see that they agree that efforts should be made to recognize obstructive events appropriately. As indicated in our paper, when we began monitoring infants we had difficulty in distinguishing between movement and obstructive apnea. We turned to the "Methods" section of papers published by experienced investigators, including Guilleminault, for enlightenment. After careful reading, it was not clear to us that Guilleminault et al used endoesophageal pressure analysis, the presence of bradycardia, oxygen saturation, and behavioral observation to define obstructive apnea.

Mixed apnea of infancy is a respiratory pattern characterized by both central and obstructive apnea. In order to characterize this form of apnea more completely, 67 patients were evaluated in the Apnea Screening Program of The Children's Hospital of Philadelphia. These infants had predominantly mixed apnea and represented 8.1% (67/838) of the children evaluated for apnea during a two year period. Mean BW was 1700 ± 88.8 SEM gms, mean GA was 31.6 ± 0.4 SEM wks. The average age at the time of study was 5.8 ± 0.5 SEM wks. Fifty-nine (88%) infants were premature, eight (12%) were term babies with infantile apnea or siblings of SIDS victims. Infants were evaluated for a minimum of 6 hours by thermistor-pneumocardiogram study. 279 episodes of mixed apnea were detected. 217 (77.8%) episodes began as central apnea and progressed to obstructive apnea. 62 episodes (22.2%) initially demonstrated obstructive apnea that subsequently became central. 155 (55.6%) episodes of mixed apnea were accompanied by bradycardia (decrease in heart rate >40 bpm below baseline level). The duration of mixed apneic episodes tended to be prolonged with 85 (30.5%) 10-15 seconds duration, 120 (43%) 15-20 seconds duration, and 74 (26.6%) > 20 seconds duration. Children with predominantly mixed apnea had associated other apnea: 33 (49.3%) had central apnea, 11 (16.4%) obstructive apnea, and 24 (35.8%) had excessive periodic breathing (

Background Obstructive apnea is a sleep disorder characterized by pauses in breathing during sleep: breathing is interrupted by a physical block to airflow despite effort. The purpose of this study was to test if osteopathy could influence the incidence of obstructive apnea during sleep in infants. Methods Thirty-four healthy infants (age: 1.5–4.0 months) were recruited and randomized in two groups; six infants dropped out. The osteopathy treatment group (n = 15 infants) received 2 osteopathic treatments in a period of 2 weeks and a control group (n = 13 infants) received 2 non-specific treatments in the same period of time. The main outcome measure was the change in the number of obstructive apneas measured during an 8-hour polysomnographic recording before and after the two treatment sessions. Results The results of the second polysomnographic recordings showed a significant decrease in the number of obstructive apneas in the osteopathy group (p = 0.01, Wilcoxon test), in comparison to the control group showing only a trend suggesting a gradual physiologic decrease of obstructive apneas. However, the difference in the decline of obstructive apneas between the groups after treatment was not significant (p = 0.43). Conclusion Osteopathy may have a positive influence on the incidence of obstructive apneas during sleep in infants with a previous history of obstructive apneas as measured by polysomnography. Additional research in this area appears warranted.