A 16-year-old boy diagnosed with delayed sleep phase syndrome benefited from a combination of controlled sleep deprivation and phase advance.

A 16-year-old white male presented to the sleep disorders clinic at Children’s Medical Center, Dayton, Ohio, in February 2003 for evaluation of sleep onset insomnia that had been going on for years. His mother made the appointment because she was tired of struggling to get him up in the morning. She had read about narcolepsy and sleep apnea on the Internet and convinced herself that her son had one of these disorders.

The teenager went to bed at midnight on school nights but did not feel sleepy for “hours.” He would not fall asleep until 3:00 am, even if he turned off the lights and lay quietly in bed. Once he fell asleep, he slept through the night. If allowed to, he woke on his own at noon and felt refreshed. Unfortunately, he needed to get up at 7:00 am for school. His mother would get him up in the morning since he would sleep through two alarm clocks and could not get up on his own. Once out of bed, he found it was hard to get moving, and he would doze during breakfast, in the car to school, and during morning classes.

His weekend schedule was more erratic. He played Cosmic Bowling until 2:00 am on Fridays and would stay up on Saturday nights listening to music or surfing the Web. He would feel sleepy around 3:00 am and went right to sleep if he put off bedtime until then. His mother allowed him to sleep as late as he wanted on weekends, and he would get up on his own around lunchtime. He did not nap.

There was no history of snoring, parasomnias, sleep paralysis, or cataplexy. He denied hypnagogic hallucinations. When he slept until noon, he was alert for the rest of the day.

The family turned their finished basement into the boy’s bedroom to allow him privacy and to distance themselves from his pet collection, which included a tarantula, a snake, and rats. He had an active social life with a close-knit group of friends and a steady girlfriend. He was a high school sophomore, planned a technical trade career, and was an average student. His grades fell that year because he fell asleep in class. He smoked one pack of cigarettes per day but did not drink caffeine or use recreational drugs.

Patient History
Medical history was noteworthy for migraines and for mononucleosis when he was in grade school. His headaches would occur three or four times a year and improved after he gave up caffeine. He used antihistamines on a PRN basis for environmental allergies. The family took him to see a psychologist for mood swings and depression when he first started high school, but these symptoms resolved. He did not use any routine medications.

The family history was negative for narcolepsy, sleep apnea, restless legs syndrome, chronic insomnia, and parasomnias. A maternal uncle had depression, and a grandmother had hypertension. The parents were healthy, and the patient had no siblings.

On examination, he was well nourished, had long dyed hair, and dressed in “gothic” apparel. He was cooperative, talkative, and articulate. He weighed 173 pounds and was 5’8” tall. His vital signs included: temperature, 36.20; pulse, 86; respirations, 16; and blood pressure, 115/49. He had a long soft palate, but the posterior pharynx was not crowded. He had a trace of tonsil tissue and a tiny uvula. The nares were patent without septal deviation or mucosal congestion. He had mild acne, multiple ear and eyebrow piercings, and black fingernail polish. The neurological, cardiovascular, and abdominal examinations were unremarkable.

The patient’s mother appeared to be more distressed about sleep issues than her son. The patient was nonplussed by his sleep schedule, though he admitted it was causing difficulty in school and was the source of family conflict. He always considered himself to be a “night owl.” He tried taking melatonin and valerian pills from a health food store, but these did not work for him. A primary care physician once prescribed tizanidine, but the family did not recall if this was for sleep or for some other problem. Forcing the boy to go to bed early and removing distractions from his bedroom (television, stereo, and computer) did not work. He tried reading “boring magazines” at bedtime to help him fall asleep and used a “white noise machine” for a while, but these interventions also failed.

Diagnosis
A diagnosis of delayed sleep phase syndrome (DSPS) was made on the basis of the history and normal physical examination. The child’s sleep diary proved to be a convincing piece of evidence used in discussions with the patient and his mother. The family was relieved to learn that their son did not have narcolepsy or sleep apnea, though they were uneasy at first with the decision not to perform a sleep study.

The physician presented therapy options, and the family implemented a treatment plan. The patient understood that DSPS could be corrected but that success hinged on his willingness to follow through with the regimen. The sleep center could provide guidance, but no one could fix the problem for him. A written action plan with specific instructions was given to the family, and the center made a series of phone calls over the next several weeks to monitor the teen’s progress.

The International Classification of Sleep Disorders¹ describes DSPS as a disorder in which the major sleep episode is delayed in relation to the desired clock time, resulting in complaints of sleep onset insomnia and/or difficulty in awakening at the desired time. Symptoms must be present for at least 1 month, and other explanations to account for excessive daytime sleepiness must be excluded. Classic features of DSPS were evident in our teenager’s history and sleep log. Specifically, when he was allowed to sleep on his own schedule, sleep quality and duration were normal and he spontaneously woke feeling refreshed. Also, he consistently felt sleepy at the same time every night and would rise at about the same time if allowed to. His sleep requirement of 9 hours is within a normal range for adolescents.

Polysomnography, if performed on patients with DSPS, characteristically shows normal sleep architecture apart from slight delays in sleep latency when carried out at the patient’s usual sleep time.² It is not unusual for adolescents with DSPS to be in stage 3 or 4 sleep in the morning hours at the time that they are expected to get up for school. This accounts for parental reports that they are very difficult to wake. We elected not to perform polysomnography on this patient given the strength of the history and sleep diary. A sleep study could always be performed at a future point if he failed to respond to therapy or if we later questioned the diagnosis.

DSPS may be classified as “extrinsic” if the root cause appears to be due to social factors and “intrinsic” when due to malfunctioning of the internal circadian pacemaker.¹ A distinction is not always apparent in clinical situations, particularly in adolescents.

The differential diagnosis of DSPS includes non-24-hour sleep-wake syndrome in which the phase delay is not static and successively lengthens each night. Individuals with “free running” circadian clocks would also have periods of very late sleep onset and late rise times, but their night-to-night schedule varies in an irregular fashion.³ Either of these disorders, if present, could be identified from a sleep log. Shy individuals or those with phobias may utilize a delayed sleep phase to avoid contact with people or stressful situations,¹ but it was clear that this was not the case in our patient. The boy was outgoing, led an active social life, and enjoyed school even though he had difficulty staying awake in class.

Evaluations of adolescents with drastic phase delay should consider the possibility of substance abuse, and it may be helpful to have a few minutes to interview the child alone, without the parents. Depression is always among the differential diagnoses and may mimic features of DSPS or coexist with it.⁴ Indeed, our patient had a history of mild depression when he started high school and underwent counseling for a period though he appeared to recover completely. The manic phase of bipolar disorder may also present with phase delay,1,4 but other symptoms of the disorder were lacking in our patient. We considered that some teenagers cultivate a delayed phase to avoid contact/conflict with family members3 but ruled out the possibility. Though the patient chose a distinctive “Goth” mode of dress, it was evident that he had a loving family who accepted him. They appeared to have open lines of communication and mutual respect.

Therapy
DSPS would not be problematic in a world without appointments and schedules. It becomes an issue when it results in conflict at school or work, or with family. Some adolescents are content with their DSPS and have no real desire to change. This perspective presents a serious barrier to therapy. Ultimately, it falls on patients to decide whether they are willing to make the effort and sacrifices required. We stress to families up front that DSPS can be alleviated in a motivated teenager, but therapy is “active” and not “passive.” It cannot be imposed on someone who is not willing to make substantial changes in lifestyle and habits.

We reviewed the sleep logs the family brought with them and determined that the boy needed about 9 hours of sleep a night. During the initial interview, the teen confirmed that he needed about 8 to 9 hours to feel as if he were “running on all cylinders.” The therapy plan was devised around a 9-hour sleep requirement. Sleep hygiene issues were reviewed as an essential component of treatment.

Most human beings have an internal circadian clock whose period is a little longer than a 24-hour cycle.⁵ Accordingly, most people find it easier to stay up later than to fall asleep early. This principle serves as a core element in the chronotherapy approach to DSPS. The strategy may be implemented in a number of ways, and the choice of how it is undertaken depends on the patient’s particular situation.

One effective chronotherapy regimen requires delaying the phase shift 3 hours further on a daily basis. An adolescent who starts to feel sleepy around 3 am is asked to delay bedtime until 6 am on the first night, 9 am on the second night, and so on until he arrives at a more desirable bedtime goal that is agreed on ahead of time. He must rise from bed 9 hours after bedtime, which is the amount of sleep he needs based on review of his sleep log. In this way, his sleep period is “walked around the clock” until he is on a target schedule. Typically, this is achieved in about 1 week. During this time, the patient must refrain from naps. Once he is on schedule, he needs to keep to that regular pattern of bedtime and rise-time on both weekdays and weekends to maintain correction.^3,6,7

This regimen is effective but often impractical for patients trying to function in the context of going to work or school. A useful modification devised by Thorpy² combines controlled sleep deprivation with phase advance (SDPA). This regimen is much less disruptive to families and was the treatment plan our patient and his family elected to follow.

SDPA is a five-step process. In step one, the patient implements a regular bedtime and rise-time for at least 6 days, even if these times are very late. Step two entails a night of total sleep deprivation, followed by advancing the bedtime by 90 minutes. Step three is to continue this modified sleep schedule for an additional 6 days to consolidate the pattern. In step four, the process of one night of total sleep deprivation followed by advancing the bedtime by 90 minutes is repeated. The process is continued every week until a target bedtime and rise-time are attained. Step 5 calls for maintaining the regular bedtime and rise-time once reached to prevent relapse of DSPS. This regimen is less threatening than “walking around the clock,” is less disruptive to school, and still takes advantage of the fact that most people find it easier to stay up late than to fall asleep early.

Adolescents with developmental delays present special challenges. At times, a strategy combining earlier rise-times, nap elimination, and judicious use of short-term zolpidem or zaleplon is useful. Some centers utilize melatonin or other sleep aids in the adjustment process. In general, we avoid using pharmacological agents and stress the importance of lifestyle changes. First and foremost, success depends on patient motivation and perseverance. Pharmaceuticals are valuable as an adjunct measure but should not be the linchpins of therapy. Many patients find that phototherapy is another valuable measure, using sunlight or light boxes in the early morning hours to help reset the individual’s internal circadian pacemaker.^9,10

Younger children may develop DSPS as well as adolescents. Overnight sleep deprivation is more difficult to perform in school-aged children but is rarely necessary. Mild cases of DSPS, or DSPS in young schoolchildren, frequently respond to eliminating naps, implementing sleep hygiene measures, and gradually moving the rise-time to an earlier hour.⁸ We explain that parents cannot control when their children fall asleep, but they can control when they wake up. Families can use this strategy during the last couple of weeks of summer vacation to help their child adapt to the schedule they will need when school resumes in the fall.

Conclusion
Our patient responded to a trial of SDPA, though success required three attempts. Social events and failing to refrain from naps during the day interfered with the first trial. During the next attempt, he developed some performance anxiety around falling asleep even after being sleep deprived. He would lie in bed and worry about not being able to fall asleep. In this circumstance, we felt justified in adding a short course of zolpidem at bedtime for 1 week in combination with SDPA, and he successfully reset his schedule to a 10:30 pm bedtime and 7:00 am rise-time. The family enlisted help from his friends to reschedule their social activities, and this helped the therapy plan move ahead as well.

It is likely that we have not heard the last from our young man. DSPS patients have a high rate of recidivism, particularly when opportunities to fall off schedule (vacation, travel, college, final examinations, social events, and jobs) present themselves. It is our hope that we succeeded at least in giving our patient the tools he needed to correct his sleep troubles and that he will remember them and know what he needs to do the next time problems arise.

Michael E. Steffan, MD, DABSM, is associate professor of pediatrics at Wright State University School of Medicine and medical director of the Sleep Disorders Program at Children’s Medical Center, Dayton, Ohio.

References
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