Sleep
Apnea
in Eight
Children
Christian Guilleminault, M.D., Frederic L. Eldridge, M.D., F. Blair Simmons, M.D., and
William C. Dement, M.D.
From tl,e Sleep Disorders Clinic and the Dicisions of Respiratory Medicine and Otolaryngology, Stanford University School of Medicine, Stanford, California
ABSTRACT. Eight children, 5 to 14 years of age, were diagnosed by means of nocturnal polygraphic monitoring with a sleep apnea syndrome similar to that seen in adults. Excessive daytime sleepiness, decrease in school perfor-mance, abnormal daytime behavior, recent enuresis, morning headache, abnormal weight, and progressive devel-opment of hypertension should suggest the possibility of a sleep apnea syndrome when any of these symptoms is associated with loud snoring interrupted by pauses during sleep. Surgery may eliminate the clinical symptomatology. Pediatrics, 58:23-31, 1976, APNEA, HYPERTENSION, AIRWAY OBSTRUCTION, TRACHEOSTOMY.
tonsils and adenoids who also have sleep-induced
apneic episodes may be particularly “at risk” of
developing cardiac problems.
This report focuses on the clinical
symptoma-tology of eight children diagnosed with a sleep
apnea syndrome. The children include seven boys
and one girl, aged 5 to 14 years. Diagnostic
techniques and treatment will also be briefly
described along with two representative case
reports. The latter will serve to illustrate the
difficult diagnostic problems that may be posed
by these children.
Con pulmonale, pulmonary hypertension, and
systemic hypertension have been found in adults
in association with an upper airway problem that
is present only during sleep.’ This upper airway
obstruction, which appears to be entirely
func-tional and specifically induced by sleep, has been
referred to as a “sleep apnea syndrome.”
Allevia-tion of the cardiac and hemodynamic
abnormali-ties and complete resolution of the apneic periods
during sleep have been reported after chronic
tracheostomy that is patent only during sleep and
closed when the patient is awake.7”
In children, partial airway obstruction due to
enlarged tonsils and adenoids is a recognized
clinical entity, and several reports have dealt with
the problems of cardiomegaly and cor pulmonale
in chronic nasopharyngeal obstruction. ‘#{176} ‘ Our
findings in adults have led us to postulate that
sleep apnea syndromes may also exist in children
and infants’5 and that children with enlarged
CASE REPORTS Case 1
Patient 1 is a Caucasian boy who was 8 years old when first brought to the Stanford University Sleep Disorders Clinic (Sleep Clinic) with the complaint of “abnormal daytime sleepiness and disturbed sleep with snoring and hallucina-tions.” The last of four children in his family, he had a normal delivery following an uneventful full-term pregnancy. His development was normal until age 3, when his mother noted intermittent attacks of “wheezing” at night, particularly
(Received May 27; revision accepted for publication December 15, 1975.)
Supported by grant HD 08339 from the National Institute of Child Health and Human Development and research grant R.R.-70 from the General Clinical Research Centers, Divi-sion of Research Resources, Public Health Service.
ADDRESS FOR REPRINTS: (CC.) Sleep Disorders Clinic and Laboratory, Stanford University School of Medicine, Stanford, California 94305.
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TABLE I
CLINICAL SYMPTOMS AND NUMBER OF APNEIC EPISODES PER NIGHT OF CHILDREN WITH SLEEP APNEA
Day- Decreased
time School Mood & Total
Somno- Recent Perform- Morning Behaeioral Weight 1-lyper- Apneic
Patient Sex Age lence Snoring Enuresis ance Headache Disturbance Change tension Periods0
1 M 8 + + + + + + + + 78
2 F 14 + + + + + + + + 674
3 M 12 + + + + + + + + 816
4 M 9 0 + Intermittent 0 0 0 + 0 418
5 M 6 0 + 0 0 0 0 0 + 103
6 M 5 + + + + + + + 0 129
7 M 11 + + + + + + + + 696
8 M 7 0 + + 0 0 + + 0 122
#{176}Totalapneic periods: number of apneic episodes occurring during one nocturnal sleep period.
when the boy had a cold. In the next two years his respiration at night became worse, and in addition to wheezing he began to emit loud, gurgling sounds. At this time his physical status also began to deteriorate, and his weight (which was 12.6 kg
at age 2) was only 12.2 kg at age 5. Although he had been toilet trained at age 2, nocturnal enuresis reappeared. When
he was 5 years old, the patient was hospitalized for evalua-tion of these symptoms. The discharge diagnoses included: failure to thrive, trichinosis (eosinophilia-hepatomegaly), nocturnal “asthma,” and family problems.
The patient received medical attention at irregular inter-vals for the next two years, but the problems persisted. The parents separated when the patient was 7 years of age, and he was placed in a special residential center for treatment. At this time, he was not only physically underdeveloped, but also began to show abnormal behavior. He seemed particu-larly frightened of going to bed. In the evening, he postponed this as long as possible and often absolutely refused to get into his bed. He became very hyperactive at these times, obviously fighting against sleep. No matter how long he slept, he was very difficult to awaken and when finally aroused he was often disoriented. He seemed to be tired and sleepy during much of the day and was observed to fall asleep while eating and even when taking a shower. Finally, he frequently complained that “monsters” surrounded him while falling asleep and when waking up.
When seen at the Sleep Clinic at age 8, the patient was underweight (17.55 kg [normal range, 18.45 to 22.05 kg]), hyperactive, and talkative. He was able to cooperate only for short periods of time. Physical examination was normal,
except for hepatomegaly. Eosinophil total count was 17%,
but all Trichinella tests were negative. Radiologic examina-tion gave a bone age of 5 years. Laryngoscopic examination during wakefulness showed enlarged tonsils and adenoids. All-night diagnostic polygraphic monitoring (see below)
revealed 90 apneic episodes and more than 100 hypopneic
episodes during eight hours of sleep.
Case 2
Patient 2 is a Caucasian girl who was 14 years old when first seen at the Sleep Clinic with the chief complaint of “disabling daytime sleepiness.” The elder of two children, the patient had a normal birth following term pregnancy, and normal development until age 7. Tonsillectomy and adenoidectomy had been performed at 3 years of age. During
her eighth year, the patient began to complain of progres-sively worsening headaches associated with daytime sleepi-ness. The headaches appeared when the patient awoke in the morning and tended to dissipate during the afternoon. Sleepiness and sleep spells also occurred unrelated to head-aches approximately eight to ten times per month, usually at school or at home during quiet, boring situations.
When she was 8#{189}years old, the patient was hospitalized on a neurological ward for evaluation of these symptoms. Discharge diagnosis was migraine headache, apparently based on a family history of migraine.
During the following year, headaches and daytime sleep spells became more frequent, and the mother noted that snoring, which had existed at night for at least two years, increased in intensity and was also present during daytime sleep. The sleep episodes could be terminated only with great difficulty, and the patient seemed disoriented for several minutes after awakening. At age 9, although toilet trained at age 2, the patient began to have nightly enuretic episodes. Because the enuresis and the exacerbation of the daytime sleepiness began shortly after parental separation, a diagnosis of migraine associated with an emotional disorder was again considered. Ergotamine was prescribed and psy-chotherapy was initiated. At about the same time, her performance in school began to deteriorate noticeably. A clinical EEC was performed which was considered “slightly abnormal,” but neither the EEC nor the patient’s behavior during sleep episodes indicated an epileptic disorder. At age 10 she underwent psychological testing, the results of which were reported to show “intelligence in the borderline retardation range.” The mother was also told, however, that the low scores might be the result of an “emotional block.”
In the years that followed, a variety of medications
(including imipramine, dextroamphetamine sulfate, and methylphenidate) were tried without significantly improving the patient’s condition. Another neurological examination, performed at age 12, was within normal limits, although the clinical EEC was again interpreted as “mildly abnormal.” Psychometric tests were repeated and the results indicated a further decline in intelligence. School psychologists and teachers attributed the child’s learning problems to “mm-imal brain dysfunction” or an “emotional block.”
At the time of her initial visit to the Sleep Clinic, the patient was taking a daily dosage of 75 mg of
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date and 300 mg of diphenylhydantomn and was attending individual and family psychotherapy. She still complained of daytime sleep spells, morning headaches, and nightly enure-sis. During the examination, she was fully alert and well-oriented, although her language was slightly childish for her age. Her weight was 46.3 kg (within the normal range), and she appeared to be a normally developed girl of 14 (men-arche at age 12, but menses were still irregular with cycles of 25 to 40 days). Neurological examination was entirely normal. Blood pressure was abnormally elevated at 150/99. (Previous blood pressure readings obtained from her pedia-trician were: age 12.5, 110/75; age 13.5, 127/80.)
All-night diagnostic polygraphic recording showed 729 apneic pauses (mean duration, 32 seconds) during eight hours of sleep.
COMMON CLINICAL SYMPTOMATOLOGY
Excessive Daytime Sleepiness
Five of the eight children were referred to the
Sleep Clinic for evaluation of excessive daytime
sleepiness and inappropriate sleep episodes
(
Table I). The other three were referred forevaluation of abnormal breathing during sleep.
The daytime sleepiness in the five children was
very persistent. The children, particularly at
school, felt embarrassed by their drowsy behavior
and sleep spells and tried desperately to fight
them off, usually without success. To avoid falling
asleep, the children tended to move about and
gave the appearance of hyperactivity. If seated
for a long period, as in a classroom, they
some-times appeared to be awake, but would
exper-ience repeated lapses. In three children who were
monitored for 24 hours, these lapses were
ac-companied by microsleep episodes which have
been reported to impair learning and to lead to an
automatic behavior syndrome.16
Snoring
All eight children snored loudly every night.
Typically, the snoring had first been noticed
several years earlier, and had become increasingly
louder in the ensuing interval. Initially, snoring
was intermittent, but eventually became
contin-uous. In one case, snoring was noticed at 6 months
of age. Four of the mothers reported that the
snoring was peculiar: a loud snort frequently
followed a respiratory silence, which might last as
long as 40 to 60 seconds. This problem raised
sufficient anxiety in three of the families for them
to consult a pediatrician.
Nocturnal Enuresis
All eight children had been completely toilet
trained at night. However, in seven cases,
bed-wetting reappeared. These episodes occurred
nightly in six children and intermittently in one.
Three children had extensive urologic
examina-tions for nocturnal enuresis, with normal findings.
The enuretic episodes were not confined to the
first third of the night (as is usually seen in
“essential” enuresis), but occurred at any time
during the night, and several children had more
than one enuretic episode during a single night.
Decreased School Performance
Five of the seven children who were attending
school had learning difficulties. Teachers
re-ported lack of attention, hyperactivity
inter-rupted by sleep spells, and a general decrease in
intellectual performance, particularly in older
children.
Morning Headaches
Five children complained of headaches that
were present when they woke up in the morning.
These headaches were not localized nor were
they associated with other symptoms (fever,
nausea, vomiting, etc.). A typical feature of the
headaches was their tendency to dissipate
corn-pletely by the late morning.
Mood and Personality Changes
Four of the children had received counseling or
family psychotherapy for “emotional problems.”
Three were classified as hyperactive, despite their
daytime sleep spells. Three children were
parti-cularly disturbed at bedtime: they consistently
avoided going to bed, fighting desperately against
sleepiness. They refused to be left alone in their
rooms while falling asleep and, if allowed, would
go to sleep on the floor in the living room. Two of
the children also told of having terrifying
hypna-gogic hallucinations.
Weight Problems
Body weight was abnormal (compared to
normative values corrected for age and height) in
seven of the cases; five children were
under-weight and two overweight.
Hypertension
Five children presented abnormal diastolic and
systolic blood pressure values compared to
age-matched controls. The hypertension was
gener-ally a more recent development and was
discov-ered at the Sleep Clinic in four of the cases.
DIAGNOSTIC PROCEDURES
All-Night Polygraphic Recording
When seen at the Sleep Clinic, the association
of excessive daytime sleepiness and loud
FIG. 1. Example of sleep-induced upper airway apnea in case 1. The endoesophageal pressure increased when the upper airway obstruction developed. The oxygen saturation, measured by an ear oximeter, dropped during the apneic episode. The carbon dioxide expired tracing demonstrates the interruption of airflow. During the apneic period, the diaphragmatic (endoesophageal pressure) swings cause some exhalation, as shown by the small variations in
percentage of carbon dioxide expired (between the arrows).
The diagnosis was conclusively established by
all-night polygraphic monitoring of sleep,
respira-tion, and cardiac rhythm.
The polygraphic variables monitoring states of
sleep included standard EEG, electro-oculogram
(EOG), and digastric electromyogram (EMG).’T
Each successive 30 seconds of polygraphic
re-cording was assigned to a specific stage of sleep or
to wakefulness according to rules and definitions
set forth in the standard manual by Rechtschaffen
and Kales.’T
Respiration was recorded using thoracic and
abdominal mercury-filled capillary strain gauges
and buccal and nasal thermistors; an ear oximeter
continuously measured arterial oxygen saturation.
An apneic episode was defined as cessation of
airflow at the nose and mouth enduring for at
least ten seconds. Used in this way, the term
“apnea” (or repetitive episodes of apnea) is
differ-entiated from the terms “periodic breathing”
and “respiratory pause.”
During at least two nights, an endoesophageal
catheter was also used to differentiate the types
of apneic episodes (central, upper airway, or
mixed). Central apnea was indicated if no change
in endoesophageal pressure occurred, reflecting
an absence of respiratory movement. On the
other hand, a cessation of airflow associated with
a progressive increase in endoesophageal pressure
swings indicated upper airway apnea. Mixed
apneas occurred as a central apnea giving way to
an upper airway apnea.
Cardiac rhythm was monitored from I
electro-cardiographic derivation. In two cases this
moni-toring was performed using n Avionic tape
recorder (Holter recording). The ECG tapes were
processed by computer, providing accurate
deter-mination and quantification of arrhythmias.’
Pulmonary studies performed during
wakeful-ness included lung volumes and spirometry,
arterial blood gas, and carbon dioxide response
curves. Daytime sleepiness and
uncooperative-ness of the children presented problems with this
testing. Accordingly, concomitant polygraphic
monitoring was performed to verify the alertness
of the child during the pulmonary test series.
A thorough upper airway examination,
includ-ing indirect laryngoscopy to evaluate the cord
movement, the immediate subglottal airway, and
the subglottal contour, was done on every child.
Direct laryngoscopy was performed under
decision to perform tracheostomy was made.
When hypertension was present, the following
evaluation was performed: urinary
vanillylman-delic acid (VMA), 17 hydroxysteroid, 17
ketoste-roid, 24-hour metanephron, catecholamine,
plas-ma cortisol, bleeding plasma renin, and, finally,
intravenous pyelogram.
RESULTS OF DIAGNOSTIC TESTING
All-Night Polygraphic Recordings
The nocturnal polygraphic monitoring showed
that all eight children presented not only
abnormal sleep patterns, but also abnormal
respi-ration and cardiac rhythm during sleep.
Abnornwl Respiration During Sleep-All
chil-dren showed profound respiratory disturbances
associated uniquely with the sleeping state (Fig. 1
and 2). These disturbances most typically took the
form of repetitive apneic episodes. The number of
apneic episodes ranged from a single-night high of
824 to a low of 78. Hypopneic episodes without
complete apnea were also noted. The duration of
apneic episodes varied widely, although, in each
patient, the majority were clustered about an
average value, which ranged from 15 to 32.5
seconds.
Upper airway apneas accounted for 83% of all
apneic episodes observed during sleep. The
remaining 23% were about equally distributed
among central and mixed episodes. Tracings from
the ear oximeter indicated that each apneic
episode was associated with oxygen desaturation.
Values as low as 40 mm Hg were frequently
observed depending mainly on duration of the
episode although oxygen desaturation was always
greater during upper airway apneas than during
central or mixed apneas of similar durations.
Thus, a sleep-related alveolar hypoventilation
syndrome was present in each child.
Relationship Between Sleep Stages and
Respi-ration-Hypopneas and apneas were always
predominant in stages 1 and 2 non-REM sleep,
and the most severe episodes occurred in these
sleep stages. In contrast, respiration was nearly
normal in stages 3 and 4 non-REM sleep: apneas
and hypopneic episodes were less frequent, and
snoring was less noticeable. Respiratory
abnor-mnalities reappeared in REM sleep. Central
ap-fleas, which rarely occurred in non-REM sleep,
predominated in REM, and upper airway apneas
were virtually never observed in this state.
Resumption of respiration following apneic
episodes was accompanied by carphologic
move-ments and a loud, gurgling snort. Frequently, an
“alpha arousal” or a lightening of sleep was also
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FIG. 2. The continuous recording of the endoesophageal pressure and ear oximetric curve obtained in case 3. When the patient is awake (top tracing), oxygen saturation is approximately 95% and endoesophageal pressure is in a normal range. Sleep appears at the beginning of the second tracing. Endoesophageal pressure increases secondary to the sleep-induced upper airway apnea, and oxygen saturation falls. At the end (concomitant with or just prior to) of the apnea, the child presented an electroencephalographic
arousal or very light sleep. One interest of this figure is to show the interrelationship of diaphragmatic and upper airway pauses-mixed apnea. This pattern is evidence for the “central” origin (i.e., involving the central control of respi-ration) of the sleep-induced apnea syndrome in both
dia-phragmatic and upper airway pauses.
observed just prior to or concomitant with the
end of the apnea.
Abnormal Sleep-Each child presented sleep
stage abnormalities. In severe cases of sleep
apnea, only a semblance of the normal non-REM
sleep stages 1 (relatively low amplitude, mixed
frequency activity) and 2 (sleep spindles and K
complexes) could be seen. Within seconds after
During the apnea, sleep spindles were rare and
poorly defined. Most of the records showed a low
amplitude theta activity. Slow waves of high
amplitude, resembling repetitive K complexes,
were usually noted after 15 to 20 seconds of
apnea. When patients resumed breathing, the
EEG usually changed from a theta rhythm to slow
alpha, mimicking stage 1 non-REM sleep. The
disruption of sleep that resulted from the
repeti-tive apneic episodes was less severe during
non-REM stages 3 and 4 (delta sleep). However, it
appeared that the normal progression to these
stages was delayed or precluded when repetitive
apneas persisted. Stages 3 and 4 patterns seemed
entirely normal when they appeared. There was
also no difficulty recognizing REM sleep. (A
complete description of the sleep data will be
presented in another 20
Abnormal Cardiac Rhythm During
Sleep-Electrocardiographic recordings from
wakeful-ness were completely normal. However, marked
sinus arrhythmias were consistently observed in
association with sleep apneic episodes.
Waking Evaluations
Daytime pulmonary function tests were normal
in all eight children. The hypertensive work-up
was negative in the five patients who had
presented abnormal diastolic and systolic pressure
values. In five of the cases hypertrophic tonsils or
adenoidal tissue were found with laryngologic
examination.
TREATMENT
One child had no follow-up after diagnostic
procedures. Six of the children underwent
tonsil-lectomy and adenoidectomy. Three and six
months after surgery five of these children were
reevaluated with all-night polygraphic
moni-toring and clinical tests. The sixth child had
surgery less than three months ago. Four children
were clinically improved. Daytime sleepiness,
loud snoring, nocturnal enuresis, and morning
headaches disappeared. School performance
im-proved, but remained low in two cases (patients 1
and 2). Blood pressure was within normal limits.
Polygraphic monitoring revealed a return of
normal sleep structure, with normal proportions
of non-REM sleep stages and REM sleep.
Respi-ratory irregularities continued to occur during
sleep, but the number was reduced to an average
of only ten apneic episodes per night. These
episodes were equally distributed in stage
non-REM sleep and REM sleep and were chiefly
central apneas that caused few hemodynamic
changes. In several cases, however, marked sinus
arrhythmia was seen with an occasional apneic
episode.
One 12-year-old child (patient 3) had no
improvement after tonsillectomy and
adenoidec-tomy. At his six-month follow-up, resting blood
pressure was 180/120 mm Hg. Nocturnal
poly-graphic recording showed 674 apneic episodes in
eight hours of sleep, compared to 816 prior to
tonsillectomy and adenoidectomy. A
tracheos-tomy was performed and a valve was
posi-tioned.
The remaining child (patient 2) who showed no
ENT findings in the daytime was given several
more extensive ENT and allergy work-ups.
Because these results were negative and
hyper-tension was worsening, tracheostomy was
per-formed. In both children who underwent
tracheostomy, there was a dramatic reversal of
clinical symptoms and hemodynamic
abnormali-ties within 48 hours after surgery. Three months
after tracheostomy, blood pressure readings were
within normal limits, and respiration during sleep
was essentially normal with an open
tracheos-tomy valve. A few brief isolated central apneas
(mean, 11 per night) were seen during REM
periods.
LONG-TERM FOLLOW-UP OF CHILDREN
WITH TRACHEOSTOMY AND VALVE
The two children (patients 2 and 3) who had
tracheostomies have been followed for 22 and 28
months after surgery. The children are able to
maintain the valve themselves, closing it during
the day and opening it at night. No complications
have arisen from the tracheostomy procedures.
One male patient (case 3), now 14#{189}years old,
plays football on his school team. The second
patient (case 2) is a skillful equestrian. If the
tracheostomy valve is closed at night, however,
upper airway apneic episodes are immediately
seen (in one case 400 apnea episodes were
recorded when the valve was closed) along with
nocturnal enuresis and complaints of fatigue and
sleepiness the following day.
COMMENTS
Nocturnal polygraphic monitoring of sleep,
respiratory, and hemodynamic variables
per-formed in adults has shown that profound and
puzzling daytime abnormalities can be associated
with a sleep apnea syndrome.5-2’22 In typical
cases, respiratory or upper airway abnormalities
are completely undetectable when patients are
awake. It has become clear that the daytime
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problems, typically hypersomnolence and
cardio-vascular changes, are caused by the apneic and
hypoxemic episodes during sleep.
Similar daytime abnormalities can be observed
in children. Excessive daytime sleepiness is
parti-cularly damaging in this age group. It interferes
greatly with normal intellectual development,
learning, and performance in school. It further
appears that excessive sleepiness may be less
easily recognized in children than adults and that
the associated disabilities may be erroneously
attributed to other problems, chiefly emotional.
The continual sleep disruption induced by the
repetitive apneic episodes must play a role in the
development of daytime sleepiness. However, it is
doubtful that this is the only factor because
apneas and snoring appear to antedate excessive
sleepiness. One possibility is that the continuous
rapid changes of Po,, Pco2, and pH levels caused
by the apneas finally reach a threshold for
affec-ting neurotransmitter synthesis in the brain
(par-ticularly ‘y-aminobutyric acid and serotonin)
which in turn leads to increased daytime
sleepi-ness.2’24
Morning headaches are more readily explained.
Upper airway apneas are associated with
tremen-dous swings in intrathoracic pressure. These
fluc-tuations lead to marked changes in blood flow and
modification of thoracic and abdominal pressure.
A progressive increase of intracranial pressure
secondary to such changes during sleep may also
result, leading to the morning headaches.
Nocturnal enuresis may be related to the
abnormal level of consciousness during sleep in
these patients in which arousal threshold appears
to be extremely high.”25
The cardiac and hemodynamic changes
asso-ciated with sleep-related apneas are a major issue.
These changes have been well documented in
adults.52’2226 It also has been recently
demon-strated that loud snoring alone-a clinical
substrata of hypopnea-is associated with
noctur-nal hemodynamic changes.27 Coccagna et al.”
have reported the case of a 12-year-old child with
sleep apnea who had continuous nocturnal
moni-toring of pulmonary arterial and femoral arterial
pressure before and after tracheostomy. The
findings were similar to those found in adults and
in the cases presented here. The possibility that
sleep-associated hemodynamic changes may lead
to more permanent pathology (hypertension, cor
pulmonale) appears to be a valid hypothesis. The
strongest evidence for this is the progressive
normalization of blood pressure after alleviation
of the sleep-induced upper airway apnea. The
apneic-related marked sinus arrhythmias
record-ed in our children were similar to those seen in
adults with sleep apnea.2” However, the two
children who had 24-hour Holter monitoring
never had heart block or runs of ventricular
tachycardia as have been seen in adults.
An unresolved but critical question concerns
the polygraphic results obtained after
tonsillec-tomy and adenoidectomy. Significant reduction
of the sleep-related respiratory pauses has been
shown. Nonetheless, a small number of central
apneic episodes were observed during sleep.
Several reports suggest that a limited amount of
central apnea is a normal feature in adults.27”
There is not sufficient data to determine exactly
where normality ends and pathology begins. Does
the occurrence of ten apneic episodes per night
indicate that a discrete dysfunction of the central
control of respiration exists in these children? Are
these children “at risk” of redeveloping a sleep
apnea syndrome in adulthood? A careful,
long-term follow-up of these and other similar cases
may answer these questions.
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