Treatment of a Case of Advanced Sleep Phase Syndrome by Phase Advance Chronotherapy

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1 Sleep. 9(l):61---{j Raven Press, New York Treatment of a Case of Advanced Sleep Phase Syndrome by Phase Advance Chronotherapy H. Moldofsky, S. Musisi, and *E. A. Phillipson Departments of Psychiatry and *Medicine, University of Toronto, Toronto, Ontario, Canada Summary: We describe here the case of a 62-year-old man with advanced sleep phase syndrome who was treated successfully by phase advance chronotherapy. Key Words: Advanced sleep phase syndrome-chronotherapy. Advanced sleep phase syndrome (ASPS) is an unusual sleep-wake schedule disorder, characterized by habitual sleep onset and wake times that are several hours earlier than desired. No difficulty is reported in maintaining sleep. The person is unable to delay sleep onset and to follow a conventional sleep-wake schedule (1). Only one case has been suggested (2), and no treatment has been described. It is uncertain whether the disorder constitutes a pathologic syndrome (1). We describe a 62-year-old man with symptoms of ASPS who was treated successfully by phase advance chronotherapy. CASE REPORT Mr. A is a 62-year-old married man with an 8-year history of intractable irresistable afternoon and evening sleepiness. At those times he would fall asleep in quiet passive situations, e.g., while reading, watching TV, or eating his supper. Sometimes he fell asleep during conversations and once he fell asleep while riding his bicycle. He and his family were especially concerned after he had fallen asleep while driving his car and had been involved in a motor vehicle accident. Usually he felt obliged to go to bed at 1830 hand would fall asleep immediately. He slept continuously, except for a few brief awakenings to urinate, after which he would fall asleep readily upon returnidg to bed. He snored, but did not complain of any sleep-related breathing difficulty. Typically, he slept until 0300 h when he would awake feeling refreshed and would jog for an hour. He remained active and alert until early afternoon. He did not describe any auxilliary symptoms of narcolepsy. Until 8 years ago, he operated a restaurant business for 30 years. His work required alternating 2-week shifts from 1700 to 0200 h and from 0500 to 1700 h. He was in good physical health except for mild benign prostatic hypertrophy and Peyronie's disease. At that time he was receiving treatment for hypothyroidism with L-thyroxine 0.1 mg/day. He had been treated for depression 4 years earlier, but clinical examination and psychologic testing (using the SCL-90 questionnaire) revealed no current evidence for any psychiatric disorder. There was no family history of sleep-wake or psychiatric disorders. Accepted for publication September Address correspondence and reprint requests to Dr. H. Moldofsky at Department of Psychiatry, Toronto Western Hospital, 399 Bathurst Street, Toronto, Ontario M5T 2S8, Canada. 61

2 62 H. MOLDOFSKY ET AL. TABLE 1. Pre- and posttreatment sleep measures Baseline Posttreatment Wake after sleep onset (%) Stage 1 (%) Stage 2 (%) Stages 3 and 4 (%) REM (%) Sleep efficiency (%) Hours of sleep Time in bed (min) Latency to stage 2 (min) Latency to REM (min) Wakings/h of sleep Movement arousals/h of sleep Stage changes/h of sleep Initial polysomnography carried out by one of us (E.P.) using conventional techniques showed that the patient went to bed at 2330 h, slept for 4.5 h and awoke at 0400 h feeling alert and refreshed. He was observed to doze while electrodes were being attached in preparation for the sleep study. His sleep physiology is shown in Table 1. During the night he snored and had 74 obstructive apneas (sleep apnea index = 17) as measured by respiratory induction plethysmography. There were no accompanying cardiac arrhythmias. Arterial oxygen saturation (measured by ear oximetry) ranged from 92 to 96% (mean 94%) in wakefulness, 84 to 96% (mean 93%) during stages 1 and 2 NREM sleep, and 82 to 96% (mean 89%) in REM sleep. The multiple sleep latency test (3) carried out at 0900, 1100, 1300, and J500 h showed an abbreviated onset to sleep at 3.3, 2.0, 0.7, and 3.3 min, respectively. A single episode of REM appeared during the second nap after 16 min. He described relatively more sleepiness in the afternoon versus the morning naps. While his sleep study suggested a mild obstructive sleep apnea disorder, this sleep-related respiratory disorder was not considered to be entirely accountable for his excessive daytime somnolence. Home diary observations during the baseline initial week showed that he slept from 1830 to 0230 h, and he often napped for 1-2 h in the early afternoon (see Fig. 1). Throughout ~his subsequent time in hospitals and at home, the patient continued to maintain a sleepwake diary for a total of 100 days. He also kept an hourly record of his oral temperature throughout his waking period, while in hospital and for 1 week, 5 months later. During the second week in hospital, the nursing staff attempted to have him delay his bedtime to =2230 h. However, he awoke between 0315 and 0400 h and continued to have uncontrollable afternoon and evening sleepiness. This phase delay procedure was not found to be practical and confirmed his failure on previous occasions to delay his sleep time. Over the next 2 weeks his bedtime was phase-advanced sequentially by 3 h every 2 days until his bedtime was set at 2300 h (see Fig. 1). After 1 week's stabilization of bedtime from 2300 to 0700 h, overnight polysomnography was carried out without respiratory measures (see Table O. The study showed that he was quite fatigued and sleepy when he retired at 2300 h. Although he slept longer, he had more wakefulness after sleep onset, and less REM and slow wave sleep. These changes may be adaptation effects during the stabilization period (see Fig. O. However, he continued to show an abbreviated onset to REM sleep. Nevertheless, in the morning he felt alert and energetic, and went on his usual I-h morning run. He returned home, free of symptoms. Sleep, Vol. 9, No, 1, 1986

3 ADVANCED SLEEP PHASE SYNDROME 63 Bolne T,"ofen, b: doysi! - phose delay! :::: 3 days r 20 T reotment by phose advance 1 T Stobilization (Hosp;'ol)...:L i Stabilization (Home) Day Number 1 Time wee~s T Follow up f 3 we ~;4 Follow up..l. 135 lr--=:: "~... ~;~~;-~31r_~ --- ':.1~~ FIG. 1. Double 24-h time plot of chronotherapy and follow-up of patient with Advanced Sleep Phase Syndrome (ASPS). Thin horizontal lines indicate time in bed, thick horizontal bars indicate self-estimated time asleep. Follow-up 5 months later showed that he was able to maintain a sleep time from 2300 to 0600 h with no prolonged wakefulness after sleep onset and he had no daytime symptoms. The pattern of oral temperature during the 7 days before phase advance chronotherapy showed low oral temperature after awakening at 0400 h. At the 5-month follow-up, his postawakening oral temperature at 0700 h was similar to the pretreatment recording at that time, but his 2200 h recording was lower (see Fig. 2). Peak mean pretreatment daytime oral temperatures and those at the 5-month follow-up were similar at ~ C at 1300 h. a G 37 :;: 36.5 E ~ 36 Pre-treatment Oral Temperatures ~ 35.5 [,,, Time I 2400 b j 5-month Post-treatment Oral Temperatures G 37 ci 36.5 E ~ Time FIG. 2. Pretreatment (a) and posttreatment (b) hourly oral temperatures. Each point represents the hourly mean and SD for 7 days. No oral temperatures were obtained during consolidated sleep. Sleep, Vol. 9, No.1, 1986

4 64 H. MOWOFSKY ET AL. DISCUSSION The patient showed clinical features that were consistent with ASPS. He was chronically unable to remain awake during the later afternoon until a socially desirable evening bedtime. After awakening from his sleep in the early hours of the morning he was alert and active until early or midafternoon. There was no evidence for a depressive disorder or other psychopathology to account for his behavior. While sleep apnea was uncovered on polysomnography, he awoke each day feeling refreshed and had no daytime cardiorespiratory problem; his late afternoon and evening somnolence could not be easily related to his sleep apnea. Furthermore, with rescheduling of his sleep-wake behavior to a socially conventional time, i.e., 2230 to 0600 h, his daytime and evening somnolence completely disappeared. No posttreatment multiple sleep latency test was carried out, but he no longer felt obliged to nap during the day and evenings, and his sleep cycle and wakefulness after sleep onset were unremarkable (see Fig. 1). The only other previous case report (2) differs from our patient. That report described a 63-year-old woman who went to bed at 2100 h and arose at 0600 hours. She complained of insomnia and did not have daytime somnolence. Investigation in a time isolation facility showed that her sleep-wake cycle and body temperature circadian rhythm were both 24.5 h. Her sleep was fragmented and she spent considerable time lying in bed awake. Although this woman tended to go to bed early and arise early, her sleep schedule was not unduly phase-shifted from social convention. Unlike this woman, our patient conforms to the expected clinical features of ASPS that would be the counterpart of the delayed sleep phase syndrome (DSPS). Patients with DSPS complain of an inability to fall asleep at a desired time and to be easily awakened; typically, they can~ot fall asleep until some time between 0200 and 0600 h. DSPS patients complain of daytime sleepiness when they attempt to conform to work or social schedule expectations, but are not sleepy at conventional bedtimes. When allowed to sleep free of social demands, their sleep is of normal length. They awaken spontaneously in the late morning or early afternoon and feel refreshed. DSPS patients function best and are most alert during the late evening and night hours. In DSPS only the phase of the sleep period within the scheduled day is abnormal. Sleep is not abnormal, unless there is an attempt to overcome the symptoms (4). The observation in this case of an abnormal timing (advanced phase) of sleep is consistent with ASPS. However, the patient's altered sleep physiology with abbreviated onset to REM sleep and mild obstructive sleep apnea differ from the expected description of a primary ASPS. The abbreviated onset to REM sleep has been observed in patients with narcolepsy (1) and in major affective disorders (5). While there was no evidence for the narcolepsy syndrome, our patient had experienced a major depressive illness 4 years earlier during the time of his sleep schedule disorder. His abbreviated onset to REM sleep, observed before and after phase advance chronotherapy, might relate to an inherent predisposition to major depressive illness. Patients with a major depressive disorder have been considered to have a phase advance of their sleep-wake cycle (6). Furthermore, phase advance chronotherapy has been successfully employed in the treatment of a patient with bipolar depressive illness (6). Patients with DSPS are successfully treated with progressive phase delay of their sleep schedule and then are maintained in a socially convenient sleep-wake time period (4). That such patients have a small "advance" portion of the circadian phase response curve is suggested to be the theoretical rationale for this form of chronotherapy (4). On the other hand, our patient was successfully treated by progressive phase advance of his sleep sched- Sleep, Vol. 9, No.1, 1986

5 ADVANCED SLEEP PHASE SYNDROME 65 ul~. There lir~ two possibl~ explmlltiops for his successful response to this tre1ltment. The patient may have an unusual short sleep-wake circ1ldian rhythm, i.e., <24 h. Ideally, it would be preferable to investigate such patients in a time isolation facility to evaluate their various sleep-wake and temperature rhythms, If he were Jiving in s\lcb a facilityi he would be expected to show progressively earl-ier bedtimes or sleepiness across days. l-iowever, to adapt to daily enviromp~ntal demands, he mily b~ require<!. constantly t9 res~t his sleepwake schedule with sleep fragmentation and r(!sultant inappropriate daytime sleepiness. The observation of no difference in the time for the peak oral temperliture betwe~n baseline and posttreatment measures is consistent with the possibility of a 20-h synchronous sleepwake and temperature rhythms. This would allow for the r~lative ease of the phase advance treatment and the resetting of the sleep onset tjme 4 h later apd retaining the Slime peak time for oral temperature. Another possibility might be thlit the sleep-wike sche<!.ule was not phase-locked with the circadian temperature rhythm during the symptomatic state. As a result of his previous shift work schedule, his sleep-wake cycle may have become dissociated from the temperature cycle, and his sleep apnea mlly hlive compoundeq his problem. With phase a.<!.v!!.nc~chronotbeqpy, the two ~ycles Pecame ;:\.ligne~f l-iowever, given the possibility th.!j.t these biologic rhythms may Pecome desyncbronl~e<!., such p!j.tients should be followed for a lengthy time following trelltment to determine recurn,mce of symptoms. Acknowl~~gll)!,!nt; We; than)< the staff of tlw Sleep Rese;!.rch LabQf1Mry at the Queen Blizabeth Hospital and the Sleep Laboratory at ~he Toronto Western Hospital and the nursing staff of the Psychobiologic Medicine Unit at the Toromo Weste;Tfi f{ospitill for their ilssist1lpce as well as Ms. Kim Tepper for typing the lllilnl,ls<;ript. - REFERENCES 1. AssociatioQ of Slei:!p Disoniers Centres, Diagnostic!,lassifiq~tioQ of sl~ep aqq ousal disorders, first editioi) p~p1jfeg by me Sle~P O~soniers qassifica!ioq Committe.e, W Roffwaig, C/1i!irman. Sleep 1979;2: ~. KaI)1ei ~, J-!ugl)es J-" Miles L-, l)emeqt we. J\dva)l!'eg-s~e,ep phase syndrome stl,l(~kd in a time isolation facility. ChroilObiologia 1979;6: 11_ Hoddes E, Zarcone V, SlIJytpe H, Phillips R, D,eme)JJ we, Q)IaQtificatiop pf s.leepiness: a )lew approach. Psychophysiology 1973;10; WeitzI)1aQ EO,.C;zeis.ler J\, CP\elIJaQ RM, Spielman Al, ZiJ:nlIJeqn!ll). Je, Dement we. Delayed sleep phase SY)ldrol)le. A cijropobiologicaldisorqer wit:)1 sleep-onse! jpso)llpi.a. Arch Gen fsychiatry 1981 ;38: Kupfer PJ. REM latel1,.cy: a psychobiolpgic I)1arker for primary depr~ssive djs~ase. Biol Psychiatry 1976;IJ:1!i Wehr TA., Wir;ZcJustjc.eA, DoopW~)l fk, Duncan W, Omi!! Ie. Ph;lS~ <J.dY!l)lc~ gf the sleep-wake cycle.asan antjd!!pressant. Science 1979;206: Sleep. Vol. 9, No. I. 1986

Facts about Sleep. Circadian rhythms are important in determining human sleep patterns/ sleep-waking cycle

Facts about Sleep. Circadian rhythms are important in determining human sleep patterns/ sleep-waking cycle Sleep Sleep is described as a state of unconsciousness or partial consciousness from which a person can be roused by stimulation Period of rest and recovery People spend about a third of their lives sleeping