Near miss sudden infant death infants: a summary of findings (1972-1981).
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Biomedical subjects
Publications and source records attributed to C Guilleminault.
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Employment which requires frequent shift rotation may lead to the development of specific sleep disorders. Delayed sleep phase (DSP) insomnia, a syndrome identified recently, can greatly impair an individual's circadian rhythm-dependent functions; it can occur when shift work disrupts normal sleep-waking schedules. Disorders of excessive daytime sleepiness, such as narcolepsy, occur in some subjects after they have been subjected to frequently rotating shifts. Understanding the problems associated with circadian rhythm disturbances and their interaction with sleep disorders is particularly important in industrial medicine; any clinician whose patients are subjected to frequent shift rotations should consider the effects of disrupted sleep-waking schedules.
Thirty-one normal infants were selected for 24-hour polygraphic monitoring at different ages during the first six months of life. The development of sleep-wake distribution patterns during day and night was observed. Qualitative changes in non-rapid eye movement (NREM) sleep as it becomes differentiated in stages 1, 2, and 3-4 were measured. Sustained periods of wake are present by 6 weeks of age. After 3 months of age, wake is predictably distributed in late afternoon and early evening. REM sleep is disproportionately distributed within sleep in 24 hours, presenting a higher percent of total sleep during the night. At 4.5 and 6 months of age, stages 2 and 3-4 NREM are coincident during the nocturnal hours and 3-4 NREM sleep peaks in the early period of the night. The decreasing proportion of REM sleep, particularly in its daytime distribution, suggests a reciprocal relationship to the developing of wakefulness.
Six infants with congenital alveolar hypoventilation syndrome (CCHS) were seen and observed over several years. Two had an association of CCHS with Hirchsprung's disease. All infants were treated by tracheostomy and mechanical ventilation. Three infants have survived (including one with CCHS and Hirchsprung's disease). However, all survivors have required frequent rehospitalization. The infant with the longest survival (now 4 years of age) has developed significant daytime problems involving the "behavioral control" of ventilation. One infant was considered as a "near miss for sudden infant death syndrome" and became significantly symptomatic after establishment of delta (stage 3-4 non-rapid eye movement) sleep, which normally develops between 2 and 4 months of age. CCHS involves autonomic nervous system dysfunction, and the question of a defect involving the integration of chemoreceptor information more than a direct defect of the central chemoreceptor is discussed.
The effects of pharmacological alteration of the monoamine systems were investigated in a canine model of narcolepsy. Cataplexy was quantified in eight severely affected dogs by means of the food-elicited cataplexy test. The specific norepinephrine (NE) uptake blocker nisoxetine, and (to a much lesser extent) the specific serotonin (5-HT) uptake blocker fluoxetine, significantly suppressed cataplexy, as did the tricyclic antidepressants protriptyline, amitriptyline, and chlorimipramine. Thus, experimental cataplexy is suppressed more by inhibition of the uptake of NE than of 5-HT. Methylphenidate, the alpha-adrenoreceptor blocker clonidine, and the dopamine receptor blocker pimozide also suppressed cataplexy in dogs. The beta-adrenergic blocker propranolol, the fatty acid gamma-hydroxybutyrate, and the monoamine oxidase inhibitors clorgyline and pargyline had little or no effect. With one exception (pimozide), all the drugs that suppressed cataplexy are known to be potent suppressors of REM sleep. The suppression of cataplexy induced by nisoxetine or protriptyline was reversed by the anticholinesterase physostigmine, further supporting a postulated aminergic-cholinergic interaction in the mechanisms for cataplexy.
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Six patients with sleep apnea syndrome were studied with continuous hemodynamic monitoring during sleep. Sleep apnea had been previously documented with an average number of apneas per hour of sleep ranging from 23 to 93 ((mean 63). There was significant decrease in heart rate during sleep (82 +/- 5 to 69 +/- 6, P less than 0.01). There was a significant rise in systemic blood pressure (103 +/- 2 mn Hg to 116 +/- 6 mm Hg, P less than 0.05) and pulmonary artery pressure (20 +/- 1 mm Hg to 32 +/- 5 mm Hg) during sleep. In addition, pulmonary artery wedge pressure increased (12 +/- 2 mm Hg to 20 +/- 3 mm Hg, P less than 0.05) during sleep and 5 of the 6 patients developed an abnormal pulmonary wedge pressure. There was a significant decrease in PO2 during sleep (71 +/- 3 mm Hg to 49 +/- 2 mm Hg, P less than 0.005). These findings suggest that increases in pulmonary wedge pressures may be contributing to increase in pulmonary artery pressures in these patients during sleep.
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A consecutive series of 100 sleep apnea free patients with the complaint of excessive daytime somnolence (EDS) were evaluated; data from medical histories, physical examination, personality inventories, and polysomnography [nocturnal polysomnography (NPSG) and daytime multiple sleep latency testing (MSLT)] were tabulated. Significant differences were found between narcoleptic and non-narcoleptic patients in a number of parameters, including EDS severity, mean sleep latency on MSLT, sleep latency on NPSG, latency to REM sleep at night, number of REM sleep at night, number of REM sleep segments throughout the night, the total number of nocturnal myoclonic jerks (as well as the number occurring per hour of NREM and REM sleep), and the number of arousals and wake periods preceded by a myoclonic jerk. Significant differences in sleep latency during MSLT and NPSG testing were found between different EDS diagnostic groups of non-narcoleptic patients. The majority of patients in the MSLT group with long sleep latencies were in the diagnostic groups of EDS associated with psychophysiological and/or psychiatric problems or with drug abuse; patients with a diagnosis of idiopathic central nervous system hypersomnia or EDS associated with disturbed nocturnal sleep formed the majority of the MSLT group with short sleep latencies. The non-narcoleptic patients in a MSLT group with short sleep latencies had significantly shorter sleep latencies at night, more sleep cycles, higher sleep efficiency, and earlier REM sleep than patients with long sleep latencies.
Ten patients with autonomic nervous system dysfunction (familial dysautonomia, juvenile diabetes, or Shy-Drager syndrome) were studied to assess the impact of their impairment on breathing during sleep. Several types of breathing dysfunction during sleep were identified independent of the patients' primary complaints. Obstructive sleep apnea syndrome was the most common; central sleep apnea and disturbances of te respiratory oscillator also were seen. Esophageal reflux was found to be the cause of some sleep-related problems. The observed respiratory irregularities were not associated with the usual cardiac response; a "decoupling" of heart rate from the respiratory cycle was noted during sleep in these patients.
The study was designed to investigate the possible role of endogenous opioids in the fall in blood pressure (BP) seen during initial sleep. Seven normal men, ages 20-30 years, were studied for three consecutive nights. Each night, electroencephalogram, chin electromyogram, electrooculogram, heart rate (all continuously), and blood pressure (every 15 minutes) were recorded. Night 1 was used for orientation. On nights 2 and 3, subjects received, in randomized order, an infusion of naloxone 0.2 mg/kg over 1 minute or volume-matched saline. Blood pressure data from the first 4 hours of non-rapid eye movement sleep were combined. On the placebo night, systolic BP fell from 114.6 +/- 6 mm Hg to 103.7 +/- 8 mm Hg (+/- SD) (p < 0.05, Wilcoxon rank-sum test). On the naloxone night, systolic BP did not change. Neither diastolic BP nor heart rate were influenced by naloxone. these data suggest that endogenous opioids could be involved in the fall in systolic BP seen during initial sleep.
To determine the diurnal rhythm of plasma beta-endorphin (beta-End), 10 healthy male volunteers between the ages of 20 and 32 yr were studied in a sleep laboratory setting for a 24-h period. Blood samples were taken through an indwelling catheter at 0800, 1000, 1400, 1800, 2200, 2300, and 2400 h and then half-hourly until 0730 h, and they were then assayed for total beta-End-like immunoreactivity (ir beta-End), PRL, and cortisol. Subjects were habituated by spending the night before the study in the sleep laboratory with an indwelling catheter and electrodes for sleep recording in place. There was a clear diurnal variation of ir beta-End, with lowest levels between 2200 and 0330 h and highest levels between 0400 and 1000 h. There was no evidence for direct entrainment with sleep stage. There was a close correlation with cortisol levels, suggesting a similar secretory pattern for beta-End and PRL in only 5 of the subjects. Because the beta-End antiserum used has a cross-reactivity of 30% with beta-lipotropin gel permeation chromatography was done on extracts of plasma taken at 1400, 2400, 0400, and 0730 h for each subject. The peak in the beta-End elution position showed a clear diurnal variation similar to that of ir beta-End.
Five adult subjects with severe kyphoscoliosis were monitored during sleep. Several types of breathing abnormalities, including obstructive apnea and hypopnea, were noted. The lowest oxygen desaturations occurred during rapid eye movement (REM) sleep. Arterial pressure, continuously measured in one subject, progressively increased throughout the night in association with abnormal breathing. The use of a cuirass ventilator did not improve the nocturnal problem.
Obstructive sleep apnea syndrome (OSAS), a disabling disorder that leads to life-threatening cardiorespiratory events during sleep, has been treated by tracheostomy. This article reports long-term follow-up data of 50 patients who have undergone this procedure, and the indications for surgery are summarized. Surgery may result in secondary local and general acute and subacute complications, but, on a long-term basis, patients were completely relieved of clinical symptoms, returned to full activity, and adapted normally to social and familial life. Temporary closure of the tracheostomy during sleep led to recurrence of obstructive sleep apnea.
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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.
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