Suppression of sleepiness and melatonin by bright light exposure during breaks in night work

Night work is non-optimal for performance and recuperation because of a lack of circadian influence that fully promote a night orientation. Our study assessed, in an industrial setting, the effects of bright light exposure (BL) on sleepiness, sleep and melatonin, during night work and during the following readaptation to day work. In a crossover design, 18 workers at a truck production plant were exposed to either BL (2500 lx) during breaks or normal light during four consecutive weeks. Twenty minute breaks were initiated by 67% of the workers between 03:00 and 04:00 hours. Sleep/wake patterns were assessed through actigraphs and ratings were given in a sleep/wake diary. Saliva melatonin was measured at 2-h intervals before, during and after night shift weeks. A significant interaction demonstrated a reduction of sleepiness in the BL condition particularly on the first two nights at 04:00 and 06:00 hours. Day sleep in the BL condition was significantly lengthened. Bright light administration significantly suppressed melatonin levels during night work and most strongly at 02:00 hours. Daytime melatonin during the readaptation after night work remained unaffected. The present findings demonstrate the feasibility and benefits of photic stimulation in industrial settings to increase adaptation to night work.     

Sleep tendency during extended wakefulness: insights into adolescent sleep regulation and behavior

Sleep tendency (latency to sleep onset) was examined during extended waking in prepubertal and mature adolescents to determine whether sleep pressure is lower near bedtime in the latter group. Participants were nine prepubertal (pubertal stage Tanner 1, mean age 11.1 years, SD+/-1.3 years, five males) and 11 pubertally mature adolescents (Tanner 5, 13.9+/-1.2 years, three males). They spent 10 nights at home on an identical fixed 10-h sleep schedule followed by a 36-h constant routine with sleep latency tests at 2-h intervals using standard polysomnography. Saliva was collected to assess dim-light melatonin onset (DLMO) phase. DLMO was earlier in the Tanner 1 (mean clock time=20:33 hours, SD=49 min) than Tanner 5 group (21:29 hours+/-42 min). Sleep latency compared at a 'critical period' spanning 12.5 (20:30 hours clock time) to 18.5 h (02:30 hours) after waking did not differ at 20:30 hours, but was shorter for the Tanner 1 group at 22:30 hours (Tanner 1=9.2+/-6.3 min; Tanner 5=15.7+/-5.8 min), 00:30 hours (Tanner 1=3.6+/-1.7 min; Tanner 5=9.0+/-6.4 min), and 02:30 hours (Tanner 1=2.0+/-1.7 min; Tanner 5=4.3+/-3.2 min; trend). These differences were apparent controlling for circadian phase by partial correlation. Sleep tendency after 14.5, 16.5, and 18.5 h awake was lower in mature versus prepubertal adolescents, supporting our hypothesis that a developmental change of intrinsic sleep-wake regulation may provide physiologically mediated 'permission' for later bedtimes in older adolescents.     

Quantitative description of EEG periodicities during stationary sleep stages

SUMMARY  A quantitative method was applied in order to assess variations in EEG activities during sleep. Three classes of variations were distinguished: variations connected with sleep-stage changes (class 1), higher-frequency variations described by the envelope of frequency-band activities (class 2), intermediate-frequency variations, corresponding to periods from 4 to 120 s (class 3). For each class, parameters characterizing the frequency spectra were computed. These parameters were mathematically simple and clear in their meaning, since they measured power, modulation index and mean squared frequency. A statistical comparison of the mean values of the parameters during different sleep stages evidenced a certain number of significant shifts in each of the three classes. The most important class-1 and class-2 variations were described by our parameters with high levels of significance. The results obtained for class 3 were in agreement with visual observations reported in the literature, such as the progressive increase in the interval between successive arousals from sleep onset to Stage 4 and the frequent occurrence of cyclic alternating patterns during Stage 2 epochs immediately preceding REM sleep.     

Topographic mapping of EEG during sleep

Summary Topographic aspects of all night sleep EEG were investigated in 10 healthy volunteers (age 20–35 years). EEG brain maps showed an increase of delta power from stage 1 to 4, a decrease of alpha power most pronounced parieto-occipitally and a slowing of the dominant alpha frequency. Differences of EEG power in different sleep stages (as compared to wakefulness) are displayed topographically. Analysis of the course of stage 2 showed an increase of delta power and a decrease of theta power in the first sections of the night, and an increase of beta power later in the night.     

Sleep propensity and sleep architecture after bright light exposure at three different times of day

SUMMARY  The aim of this work was to study the effects of bright light-induced circadian phase shifts on sleep propensity and sleep architecture while the timing of the sleep/wake cycle is kept constant. Twenty-three normal subjects underwent an 11-day study including: (i) baseline sleep and vigilance evaluation; (ii) baseline evaluation of the circadian temperature rhythm with a 40-h constant routine; (iii) five hours of bright light exposure on each of three days; (iv) post-treatment sleep and vigilance evaluation; (v) post-treatment circadian rhythm evaluation with a second 40-h constant routine. Subjects were divided into three groups: eight subjects were exposed to bright light in the morning ('Morning group'), eight subjects were exposed in the evening ('Evening group'), and seven subjects were exposed in the afternoon ('Afternoon group'). After light exposure, the Morning group showed an advance of 1.23 h in the phase of the temperature rhythm, the Evening group showed a delay of 1.62 h, and the Afternoon group showed a non-significant advance of 0.5 h. In support of expectations, early-night sleep propensity was decreased by evening bright light, was increased in almost all subjects exposed to morning bright light, and was not changed by afternoon bright light exposure. The phase shift created by bright light exposure did not seem to be large enough to have a systematic effect on sleep consolidation or on REM sleep parameters in any of the three groups, suggesting that these variables are less sensitive to alterations in phase of the circadian oscillator than early-night sleep propensity.     

Effects of melatonin administration on daytime sleep after simulated night shift work

Disturbed sleep and on-the-job sleepiness are widespread problems among night shift workers. The pineal hormone melatonin may prove to be a useful treatment because it has both sleep-promoting and circadian phase-shifting effects. This study was designed to isolate melatonin's sleep-promoting effects, and to determine whether melatonin could improve daytime sleep and thus improve night time alertness and performance during the night shift. The study utilized a placebo-controlled, double-blind, cross-over design. Subjects (n=21, mean age=27.0 +/- 5.0 years) participated in two 6-day laboratory sessions. Each session included one adaptation night, two baseline nights, two consecutive 8-h night shifts followed by 8-h daytime sleep episodes and one recovery night. Subjects took 1.8 mg sustained-release melatonin 0.5 h before the two daytime sleep episodes during one session, and placebo before the daytime sleep episodes during the other session. Sleep was recorded using polysomnography. Sleepiness, performance, and mood during the night shifts were evaluated using the multiple sleep latency test (MSLT) and a computerized neurobehavioral testing battery. Melatonin prevented the decrease in sleep time during daytime sleep relative to baseline, but only on the first day of melatonin administration. Melatonin increased sleep time more in subjects who demonstrated difficulty in sleeping during the day. Melatonin had no effect on alertness on the MSLT, or performance and mood during the night shift. There were no hangover effects from melatonin administration. These findings suggest that although melatonin can help night workers obtain more sleep during the day, they are still likely to face difficulties working at night because of circadian rhythm misalignment. The possibility of tolerance to the sleep-promoting effects of melatonin across more than 1 day needs further investigation.     

Effects of light exposure and sleep displacement on dim light melatonin onset

The purpose of the study was to induce in two different ways, a phase-angle difference between the circadian pacemaker and the imposed sleep-wake cycle in humans, we intended to: (i) shift the circadian pacemaker by exposure to bright light and keep the timing of the sleep-wake cycle fixed; and (ii) keep the timing of the circadian pacemaker fixed by a constant light-dark cycle and displace sleep. We monitored dim light melatonin onset (DLMO), core body temperature and sleep. DLMO was delayed significantly after 3 days of a 3-h delayed sleep-phase when compared with 3 days of sleep at a normal or 3-h advanced sleep-phase. The shifts in DLMO were not accompanied by shifts in body temperature, changes in waking-up time or by a change in the duration of the first rapid eye movement (REM) sleep episode. Three days of light exposure in the morning or evening resulted in shifts in DLMO of similar magnitude, but this was accompanied by shifts in the rhythm of body temperature, changes in waking-up time and in the duration of the first REM sleep episode. We conclude that the changes observed after light exposure reflect shifts in the circadian pacemaker. In contrast, we propose that the changes observed in DLMO after sleep displacement are not mediated by the circadian pacemaker. These results raise some doubts about the reliability of DLMO as a marker of circadian phase in cases of sleep disturbances. Finally, we initiate a search for changes in sleep that might be responsible for the unexpected effects on DLMO.     

Muscle artifacts in the sleep EEG: Automated detection and effect on all-night EEG power spectra

Owing to the use of scalp electrodes in human sleep recordings, cortical EEG signals are inevitably intermingled with the electrical activity of the muscle tissue on the skull. Muscle artifacts are characterized by surges in high frequency activity and are readily identified because of their outlying high values relative to the local background activity. To detect bursts of myogenic activity a simple algorithm is introduced that compares high frequency activity (26.25–32.0 Hz) in each 4-s epoch with the activity level in a local 3-min window. A 4-s value was considered artifactual if it exceeded the local background activity by a certain factor. Sensitivity and specificity of the artifact detection algorithm were empirically adjusted by applying different factors as artifact thresholds. In an analysis of sleep EEG signals recorded from 25 healthy young adults 2.3% (SEM: 0.16) of all 4-s epochs during sleep were identified as artifacts when a threshold factor of four was applied. Contamination of the EEG by muscle activity was more frequent towards the end of non-REM sleep episodes when EEG slow wave activity declined. Within and across REM sleep episodes muscle artifacts were evenly distributed. When the EEG signal was cleared of muscle artifacts, the all-night EEG power spectrum showed significant reductions in power density for all frequencies from 0.25–32.0 Hz. Between 15 and 32 Hz, muscle artifacts made up a substantial part (20–70%) of all-night EEG power density. It is concluded that elimination of short-lasting muscle artifacts reduces the confound between cortical and myogenic activity and is important in interpreting quantitative EEG data. Quantitative approaches in defining and detecting transient events in the EEG signal may help to determine which EEG phenomena constitute clinically significant arousals.     

Circadian and homeostatic sleep regulation in morningness-eveningness

Morningness-eveningness has been associated with the entrained circadian phase. However, we recently identified morning and evening types having similar circadian phases. In this paper, we compared parameters of slow-wave activity (SWA) decay in non-rapid-eye-movement (NREM) sleep between these two subgroups to test the hypothesis that differences in the dynamics of nocturnal homeostatic sleep pressure could explain differences in sleep timing preference. Twelve morning-type subjects and 12 evening-type subjects with evening types (aged 19-34 years) selected using the Morningness-Eveningness Questionnaire were further classified according to the phase of their dim light melatonin onset (DLMO). The six morning types with the earliest DLMO were compared to the six evening types with the latest DLMO ('extreme' phases), and the six morning types with the latest DLMO were compared to the six evening types with the earliest DLMO ('intermediate' phases). Subjects slept according to their preferred sleep schedule. Spectral activity in four midline derivations (Fz, Cz, Pz, Oz) was calculated in NREM sleep and an exponential decay function was applied on SWA data averaged per sleep cycle. In the subjects with intermediate circadian phases, both initial level and decay rate of SWA in Fz were significantly higher in morning than in evening types. No difference appeared between chronotypes of extreme circadian phases. There was no correlation between individual estimates of SWA decay and DLMO. These results support the hypothesis that chronotype can originate from differences in the dissipation of sleep pressure and that homeostatic and circadian processes influence the sleep schedule preference independently.     

Unilateral vibrissae stimulation during waking induces interhemispheric EEG asymmetry during subsequent sleep in the rat

To test the theory that sleep is a regional, use-dependent process, rats were subjected to unilateral sensory stimulation during waking. This was achieved by cutting the whiskers on one side, in order to reduce the sensory input to the contralateral cortex. The animals were kept awake for 6 h in an enriched environment to activate the cortex contralateral to the intact side. Whiskers are known to be represented in the barrel field of the contralateral somatosensory cortex and their stimulation during exploratory behavior results in a specific activation of the projection area. In the 6 h recovery period following sleep deprivation, spectral power of the nonrapid eye-movement (NREM) sleep EEG in the 0.75-6.0 Hz range exhibited an interhemispheric shift towards the cortex that was contralateral to the intact whiskers. The results support the theory that sleep has a regional, use-dependent facet.     

The dim light melatonin onset following fixed and free sleep schedules

The time at which the dim light melatonin onset (DLMO) occurs can be used to ensure the correct timing of light and/or melatonin administration in order to produce desired circadian phase shifts. Sometimes however, measuring the DLMO is not feasible. Here we determined if the DLMO was best estimated from fixed sleep times (based on habitual sleep times) or free (ad libitum) sleep times. Young healthy sleepers on fixed (n=60) or free (n=60) sleep schedules slept at home for 6 days. Sleep times were recorded with sleep logs verified with wrist actigraphy. Half-hourly saliva samples were then collected during a dim light phase assessment and were later assayed to determine the DLMO. We found that the DLMO was more highly correlated with sleep times in the free sleepers than in the fixed sleepers (DLMO versus wake time, r=0.70 and r=0.44, both P<0.05). The regression equation between wake time and the DLMO in the free sleepers predicted the DLMO in an independent sample of free sleepers (n=23) to within 1.5 h of the actual DLMO in 96% of cases. These results indicate that the DLMO can be readily estimated in people whose sleep times are minimally affected by work, class and family commitments. Further work is necessary to determine if the DLMO can be accurately estimated in people with greater work and family responsibilities that affect their sleep times, perhaps by using weekend wake times, and if this method will apply to the elderly and patients with circadian rhythm disorders.     

Shine light on sleep: Morning bright light improves nocturnal sleep and next morning alertness among college students

The relationship between daytime light, especially morning light and sleep, has not been well documented. People who work in an office spend most of their time indoors and thus have less access to high-level daylight. The current study employed a field intervention approach to investigate whether exposure to 1.5 h of bright electric light in the early morning for 1 workweek would benefit sleep among students who spent most of their time in an office at the university. Twelve students (24.92 ± 1.78 years) underwent a 2 workday baseline measurement and two inconsecutive 5 workday interventions (with 1 week washout) with morning bright light and regular office light (1000 lx, 6500 K vs. 300 lx, 4000 K, at eye level). The sleep outcomes were recorded with actigraphy and a sleep diary. In addition, self-ratings of daytime sleepiness, mood, mental fatigue, perceived effort, and next morning sleepiness were measured each workday. The results showed that exposure to morning bright light versus regular office light yielded a higher sleep efficiency (83.82% ± 1.60 vs. 80.35% ± 1.57, p = 0.02), a smaller fragmentation index (15.26% ± 1.31 vs. 17.18% ± 1.28, p = 0.05), and a shorter time in bed (7.12 ± 0.13 vs. 7.51 ± 0.12, p = 0.03). Meanwhile, an earlier sleep onset time, shorter sleep latency, and lower morning sleepiness were observed after a 5 workday morning bright light intervention compared with the baseline (ps <0.05), no such benefit was found for self-ratings (ps >0.05). These findings support existing evidence that morning bright light could function as an enhancer of sleep and alertness for office occupants.     

The circadian and homeostatic modulation of sleep pressure during wakefulness differs between morning and evening chronotypes

The purpose of this study was to evaluate homeostatic and circadian sleep process in 'larks' and 'owls' under daily life conditions. Core body temperature, subjective sleepiness and waking electroencephalogram (EEG) theta-alpha activity (6.25-9 Hz) were assessed in 18 healthy men (nine morning and nine evening chronotypes, 21.4 +/- 1.9 years) during a 36-h constant routine that followed a week of a normal 'working' sleep-wake schedule (bedtime: 23.30 h, wake time: 07.30 h). The phase of the circadian rhythm of temperature and sleepiness occurred respectively, 1.5 h (P = 0.01) and 2 h (P = 0.009) later in evening- than in morning-type subjects. Only morning-type subjects showed a bimodal rhythm of sleep-wake propensity. The buildup of subjective sleepiness, as quantified by linear regression, was slower in evening than in morning types (P = 0.04). The time course of EEG theta-alpha activity of both chronotypes could be closely fitted by an exponential curve. The time constant of evening types was longer than that of morning types (P = 0.03), indicating a slower increase in sleep pressure during extended wakefulness. These results suggest that both the circadian signal and the kinetics of sleep pressure buildup differ between the two chronotypes even under prior naturalistic conditions mimicking the usual working day.     

Enhanced performance in elderly subjects following bright light treatment of sleep maintenance insomnia

Sixteen older individuals with sleep maintenance insomnia were treated with night-time bright-light exposure (BL) while living at home. Twelve consecutive days of acute light treatment were followed by a 3-mo maintenance light-treatment period. Subjects completed laboratory evaluation sessions on five separate occasions (prior to and following the acute light-treatment period, and once per month during the maintenance period). During each laboratory session, performance levels, sleep, and core body temperature were measured. The performance battery consisted of four computerized tasks (Logical Reasoning, Stroop Congruency, Two Letter Visual Search, and Wilkinson Four-Choice Reaction Time) and was administered every 2 h between 10.00 and 18.00 hours. Subjects improved significantly on three of the four tasks from pre-BL to post-BL. During the maintenance period, subjects who received active BL treatment maintained significantly higher performance levels than a control BL group. Light treatment improved sleep efficiency and delayed the phase of the body temperature rhythm. Performance improvements were significantly related only to sleep and not to circadian phase. The implications for non-circadian treatments of sleep maintenance insomnia and cognitive functioning in the elderly are discussed.     

Effect of prolonged wakefulness on electroencephalographic oscillatory activity during sleep

The human sleep electroencephalogram (EEG) is characterized by the occurrence of distinct oscillatory events such as delta waves, sleep spindles and alpha activity. We applied a previously proposed algorithm for the detection of such events and investigated their incidence and frequency in baseline and recovery sleep after 40 h of sustained wakefulness in 27 healthy young subjects. The changes in oscillatory events induced by sleep deprivation were compared to the corresponding spectral changes. Both approaches revealed, on average, an increase in low frequency activity and a decrease in spindle activity after sleep deprivation. However, the increase of oscillatory events in the delta range and decrease in the sigma range occurred in a more restricted frequency range compared to spectral changes. The mean relative power spectra showed a significant increase in theta and alpha activity after sleep deprivation while, on average, the event analysis showed only a weak effect in the theta band. The reason for this discrepancy is that the spectral analysis does not distinguish between diffuse activity and clearly visible temporally localized oscillations, while the event analysis would detect only the latter. Additionally, only a few individuals clearly showed activity in the theta or alpha frequency bands. Conversely, event analysis revealed that some individuals showed an increased rate of sleep spindles after sleep deprivation, a fact that was not evident in the relative power spectra due to a decrease in background activity. The two methods complement each other and facilitate the interpretation of distinct changes induced by prolonged wakefulness in sleep EEG.     

Correlation of EEG activities between slow-wave sleep and wakefulness in patients with supra-tentorial stroke

Summary Using topographic EEG mapping, we studied the relationships between delta activity during slow-wave sleep (SWS) and the background EEG activity during wakefulness, in 11 normal subjects and 35 stroke patients with unilateral supra-tentorial lesions. Delta-1 power during SWS showed a significant positive correlation with alpha-1 power during wakefulness, in both hemispheres. Delta-1 and delta-2 power during SWS correlated positively not only with alpha-2 power, but also with delta-1 and delta-2 power during wakefulness in the affected hemisphere. These figures indicate that the amount of delta activity during SWS can be associated with that of alpha activity during wakefulness. A close negative correlation was observed between delta power during SWS and the age of the subjects in the patient group. The Barthel index showed no significant correlation with delta-1 or delta-2 power in either hemisphere in patient group. Our results suggest that delta activity during SWS may be associated with dysfunction of the cerebral cortex in stroke patients as well as in normal aged subjects.     

The alteration of human sleep and circadian rhythms during spaceflight

Numerous anecdotes in the past suggest the concept that sleep disturbances in astronauts occur more frequently during spaceflight than on ground. Such disturbances may be caused in part by exogenous factors, but also an altered physiological state under microgravity may add to reducing sleep quality in a spacecraft. The present investigation aims at a better understanding of possible sleep disturbances under microgravity. For the first time, experiments were conducted in which sleep and circadian regulation could be simultaneously assessed in space. Four astronauts took part in this study aboard the Russian MIR station. Sleep was recorded polygraphically on tape together with body temperature. For a comparison, the same parameters were measured during baseline periods preceding the flights. The circadian phase of body temperature was found to be delayed by about 2 h in space compared with baseline data. A free-run was not observed during the first 30 d in space. Sleep was shorter and more disturbed than on earth. In addition, the structure of sleep was significantly altered. In space, the latency to the first REM episode was shorter, and slow-wave sleep was redistributed from the first to the second sleep cycle. Several mechanisms may be responsible for these alterations in sleep regulation and circadian phase. Most likely, altered circadian zeitgebers on MIR and a deficiency in the process S of Borbély's sleep model cause the observed findings. The change in process S may be related to changes in physical activity as a result of weightlessness.     

Interaction of chronic sleep restriction and circadian system in humans

Nocturnal sleep restriction and compensation with daytime naps is common in today’s society. In a between‐participants design, we examined the effects of chronic (10 nights) sleep restriction on 24 h plasma melatonin profiles in humans. Following a baseline period with 8.2 h time in bed (TIB) for sleep, participants were randomized to a control (8.2 h TIB) or sleep‐restriction condition (4.2 h TIB), with and without diurnal naps. Sleep restriction was achieved via delaying bedtime and advancing wake time by 2 h each relative to the baseline sleep period. Participants were maintained in a controlled, time isolated laboratory environment throughout the protocol, with light levels below 40 lx at all times. Twenty‐four hour plasma melatonin profiles were assessed at baseline and at the end of the sleep‐restriction period, with subjects maintained in a constant posture protocol. Compared with the baseline assessment and the 8.2 h TIB control group, a significant phase delay in melatonin onset (1.2 ± 0.9 h) occurred in all sleep‐restriction (4.2 h TIB) groups (P < 0.05). There was no evidence of a phase advance or shortening of the period of melatonin secretion associated with the advanced waking time. These results suggest that nocturnal light and dark exposure may be more potent in effecting circadian phase shifts than exposure to morning light, at least in conditions of controlled, dim lighting in the laboratory.     

Evolution of sleep and sleep EEG after hemispheric stroke

The evolution of subjective sleep and sleep electroencephalogram (EEG) after hemispheric stroke have been rarely studied and the relationship of sleep variables to stroke outcome is essentially unknown. We studied 27 patients with first hemispheric ischaemic stroke and no sleep apnoea in the acute (1-8 days), subacute (9-35 days), and chronic phase (5-24 months) after stroke. Clinical assessment included estimated sleep time per 24 h (EST) and Epworth sleepiness score (ESS) before stroke, as well as EST, ESS and clinical outcome after stroke. Sleep EEG data from stroke patients were compared with data from 11 hospitalized controls and published norms. Changes in EST (>2 h, 38% of patients) and ESS (>3 points, 26%) were frequent but correlated poorly with sleep EEG changes. In the chronic phase no significant differences in sleep EEG between controls and patients were found. High sleep efficiency and low wakefulness after sleep onset in the acute phase were associated with a good long-term outcome. These two sleep EEG variables improved significantly from the acute to the subacute and chronic phase. In conclusion, hemispheric strokes can cause insomnia, hypersomnia or changes in sleep needs but only rarely persisting sleep EEG abnormalities. High sleep EEG continuity in the acute phase of stroke heralds a good clinical outcome.     

The prevalence of delayed and advanced sleep phase syndromes

To determine the prevalence of the delayed sleep phase syndrome (DSPS) and the contrasting advanced sleep phase syndrome (ASPS), a cross-sectional nationwide epidemiological study was performed in Norway. Screening questionnaires were sent to a random sample of 10,000 adult individuals (18-67 y), of both sexes, taken from the National register of Norway. The response rate was 77%. Diagnoses of DSPS and ASPS were based on International Classification of Sleep Disorders (ICSD) criteria. All individuals suspected of having DSPS or ASPS were requested to fill out a second questionnaire, and a sleep log for four weeks. Subjects for whom the suspicion of DSPS or ASPS could be upheld were contacted by telephone for a final confirmation. Of the 129 possible DSPS cases identified from the screening questionnaires, 17 (9 f; 8 m) remained with the confirmed diagnosis of DSPS. The prevalence was calculated to be 0.17% (95% Confidence Intervals: 0.10-0.28). Thirteen individuals had a mild to moderate DSPS and four had a severe DSPS. The mean age of onset was 15.4 y, and mean duration was 19.2 y. There was no significant correlation between prevalence and age. A sleep phase delay (MSPD) induced by social/environmental or psychological factors was found in 55 subjects (prevalence = 0.72%). Using strict ICSD criteria, no case of ASPS was detected, confirming earlier assumptions of the extreme rarity of this condition.