History of Pediatric Sleep Medicine
Development of Pediatrics as a Unique Discipline
Prior to the beginning of the twentieth century, health care for children and adolescents was virtually non-existent. Health care for children was principally provided by family members. Mortality rates for infants were high. More than one-third of infants died before their fifth birthday.1 Despite this significant incidence of infant mortality, little was done to improve health care for children and few took particular notice of the lack of professional services. Health care for children by the medical profession was provided using adult criteria, adult standards of care, adult definitions of diseases/disorders, and utilization of therapeutic techniques developed for adult patients.2
Medical practitioners who limited their practice to children were few and considered ‘baby feeders’ since little was known of the cause of illness in children. Infectious diseases prevailed and diarrheal diseases resulting in dehydration affected many. It has been estimated that at the turn of the century there were not more than 50 medical practitioners in the United States who were particularly interested in the health care of children, and less than a dozen limited their practice exclusively to children.3 Health care facilities for clinical evaluation and management of childhood disease, specifically designed for children’s needs were non-existent.2 Being considered the property of their parents, neither earning a living, paying taxes, nor voting, children then and now possess neither an economic nor political influence.
Childhood diseases were widespread. Prevention was the only underlying principle. Approaches to treatment of illness during childhood included tea, barley water, and protein milk. Floating hospitals and country sanatoria were occasionally utilized for management of childhood illness since sun, fresh air, and isolation were treatments of choice and standard of care. Nonetheless, because of the lack of children’s clinics for diagnosis and management of pediatric diseases/disorders, care of children remained in the home.4 Because of the lack of diagnostic methods, evaluation of childhood illness was based primarily on anecdotes, and clinical signs and symptoms. Even congenital malformations were thought by many child health care practitioners to be due to maternal influences.5 Treatment was principally based on either adult medical interventions or was purely empiric. Climate therapy was common. Exposure to sunlight was prescribed for various illnesses including but not limited to tuberculosis, cutaneous abnormalities, anemia, and rickets. Some treatments were effective, but most were relatively ineffective. For example, treatment of pneumonia often included administration of digitalis, camphor, strichnia, and alcohol.
Development of Sleep Medicine as a Unique Discipline
Although there has been a fascination with sleep since antiquity, the scientific investigation of sleep and its disorders can be traced back to 1930 when Berger first described spontaneous EEG activity in the brains of sleeping subjects;6 differentiation of sleep into specific and distinct states by Harvey, Loomis, and Hobart in 1937.7 Eye movements in sleep were previously described in sleeping infants8 and the first description of rapid eye movement (REM) sleep by Aserinsky and Kleitman at the University of Chicago in 1953.9 Five years later, Dement and Kleitman reported the cycling of REM sleep and non-rapid eye movement (NREM) sleep throughout the sleep period, proposed a classification system of NREM sleep into four distinct stages, and the association of eye movements in REM sleep with dream mentation.10–11
It had become clear that these discoveries ushered in the realization that it was not enough to evaluate health and disease during only waking hours, but throughout the 24-hour continuum. A new era of medical and scientific research emerged focusing on physiology, pharmacology, pathophysiology, and even anatomy that are different during sleep than during the waking state.12 Sleep research provided the groundwork and basis for the realization that clinical evaluation and management of patients might differ during sleep when compared to wake, resulting in the emergence of clinical sleep medicine.13
At first, clinical sleep medicine evolved from patient self-referrals. Most sleep complaints were related to problem insomnia. However, it became clear that the common belief that the majority of etiologies of insomnia were not purely psychiatric in origin.13 Obstructive sleep apnea had been identified in Europe, but there had been little notice in the United States. In 1970, Lugaresi and colleagues published remarkable success of tracheostomy in the treatment of obstructive sleep apnea.14 Nonetheless, similar evaluation and management of obstructive sleep apnea was not yet accepted. In 1972, Guilleminault demonstrated remarkable results in managing uncontrollable hypertension in a -year-old boy with tracheostomy.15 It is stunning that demonstration of the first successful treatment of sequelae of obstructive sleep apnea in the United States was in a pediatric patient.
Physiological evaluation of sleep had also progressed with adaptation of polygraphy used in monitoring EEG to evaluation of other physiological variables during sleep. Termed polysomnography, Holland et al.16 changed the face of clinical assessment of sleep in adult patients. Now there were methods for both basic evaluation by history and physical examination as well as physiological assessment of sleep-related complaints in a clinical laboratory setting.
By the end of the 1970s, clinical sleep disorders medicine became an accepted area of medical inquiry, although practice of sleep disorders medicine was still couched in other disciplines of pulmonology, psychiatry, neurology, and internal medicine. In 1968, the Manual of Standardized Terminology, Techniques, and Scoring System for Sleep Stages of Human Subjects was published.17 This was a significant step forward in standardizing sleep stage scoring in adults and to eliminate unreliability and inconsistencies in laboratory evaluation of sleep both between laboratories and within laboratories. It was clear at that time this standardization was not appropriate for identification of stages of sleep and evaluation of sleep in newborns, infants, and children. Anatomical and physiological variables differed markedly from the adult. Similar standardization of sleep stage identification was a daunting task due to the rapid and constantly changing biology of the maturing and developing child. Therefore, the newborn infant became a starting point for a similar process that was started by Drs. Rechtschaffen and Kales in 1968. Drs. Anders, Emdee, and Parmelee co-chaired an ad-hoc committee to provide similar standards and the result was the publication in 1971 of A Manual for Standardized Techniques and Criteria for Scoring of States of Sleep and Wakefulness in Newborn Infants.18 Strikingly, between publication of this manual and today, 42 years later, there has been no similar effort for infants and children older than 2 months of age and the beginning of puberty. Many problems precluded this task. Standardization in the pediatric age group is a formidable endeavor. First, there are rapid and dynamic changes that occur during the first two decades of life. The nervous system is constantly changing structurally and functionally during this period of life. Attempting to define cross-sectional criteria for evaluation of children both within same-age subjects and between subjects is extraordinarily difficult because of normal internal and external variability. Normal ranges can be extensive. Limitations include number of evaluations required for appropriate power. External reliability and validity can also be quite difficult to establish. Several longitudinal points are often required for appropriate comparison of polysomnographic variables. This has been suggested to be termed developmental polysomnography.19 This would then take into account normal progression of maturation, rather than evaluating a single polygraphic study at a single point in time. Because of these immense difficulties, little evidence-based standardized information has been available to provide accurate and reproducible normative data, despite evidence that sleep and its normal structure and maturation has far-reaching implications on growth, development, and learning.20,21
Beginning in 1978, the American Board of Sleep Medicine (ABSM) provided an examination in clinical polysomnography to assure quality of practitioners practicing sleep disorders medicine and interpreting polysomnograms. The first examination certified 21 candidates. During the next 28 years, the ABSM certified more than 3400 individuals.22 This examination was not specialty-specific and was taken by internists, psychiatrists, psychologists, neurologists, family practitioners, and pediatricians. Successful applicants became diplomates of the ABSM. Indeed, sleep disorders medicine as a new and unique discipline became the focus of more clinical practitioners.
Pediatric and adolescent sleep medicine has become an outgrowth of this sleep disorders medicine practice. Inspiration has come from several directions: scientific and clinical interest in sudden infant death syndrome (SIDS); identification of obstructive sleep apnea and other sleep-related breathing disorders occurring with significant prevalence in the pediatric population; identification of the importance of sleep in the origin of daytime behavioral difficulties; and the influence of sleep disorders on children’s daytime performance and learning.
In the early 1980s the practice of pediatrics was a highly respected medical discipline. One of the principal textbooks utilized by most students and practitioners of health care for children was entitled Nelson’s Textbook of Pediatrics.23 Nevertheless, the fourteenth edition of this text, published in 1992, had a total of eleven paragraphs uniquely devoted to sleep disorders in children.
In 1985, two seminal works were published: one for parents and the other for sleep scientists. The first was publication of Dr. Richard Ferber’s book for parents entitled Solve Your Child’s Sleep Problems.24 Based on Dr. Ferber’s work at Boston Children’s Hospital, this book reviewed all aspects of sleep in childhood and provided practical information in management of many sleep-related difficulties that occur during infancy and childhood. The second publication was entitled Sleep and Its Disorders in Children edited by Dr. Christian Guilleminault.25 This book was a compilation of ground-breaking scientific papers on normative data providing a basis for future direction in the scientific study of sleep and sleep–wake cycles during infancy, childhood, and adolescence.
Clinical pediatric sleep medicine has had to rely on nosology developed for adults.26 Adaptations have been attempted,27 but it is clearly apparent that adapting adult criteria to infants and children can lead to many false starts and wrong turns. Most sleep-related problems in children might carry similar nomenclature, but children are different and it would be no less inappropriate to apply adult sleep medicine anatomical, physiological, and pathological criteria to veterinary medicine.
In 2002, the American Academy of Sleep Medicine (AASM)applied to the Accreditation Council on Graduate Medical Education (ACGME) for establishment of sleep medicine training programs under the auspices of the ACGME as part of a comprehensive plan along with the American Board of Medical Specialists (ABMS) to accept sleep medicine as an independent medical specialty. In 2003, this was approved and a consensus plan was developed for establishment of a new multi-disciplinary specialty examination in sleep medicine to be jointly offered by the ABIM, ABPN, and the ABP, ABFM, and ABO.22 The first examination was administered in 2007. Considerations and disorders unique to childhood comprised 2% of the first examination. Although pediatrics is a required portion of a sleep medicine fellowship curriculum, it is unclear how much pediatric medicine and sleep disorders in children are afforded to internists, otolaryngologists, psychiatrists, and neurologists studying general sleep medicine in these programs. It is also unclear whether training in developmental medicine and children’s health care can be translated into the practice of sleep medicine without a comprehensive underpinning of pediatric medicine.