James E. Dillon

Pediatric sleep questionnaire (PSQ): validity and reliability of scales for sleep-disordered breathing, snoring, sleepiness, and behavioral problems

Objective: To develop and validate questionnaire scales that can be used in research to investigate the presence of childhood SRBDs and prominent symptom complexes, including snoring, daytime sleepiness, and related behavioral disturbances.Background: Obstructive sleep-related breathing disorders (SRBDs) are common but usually undiagnosed among children. Methods to help identify SRBDs without the expense of polysomnography could greatly facilitate clinical and epidemiological research.Methods: Subjects were children aged 2-18 years who had polysomnographically-confirmed SRBDs (n=54) or appointments at either of two general pediatrics clinics (n=108). Parents completed a Pediatric Sleep Questionnaire which contained items under consideration for inclusion in desired scales.Results: Item reduction, based on data from a randomly selected 50% of the subjects (group A), produced a 22-item SRBD score that was strongly associated with diagnosis of an SRBD (P<0.0001) in a logistic regression model that accounted for age and gender. Diagnosis was also strongly associated with subscores for snoring (four items, P<0.0001), sleepiness (four items, P=0.0003), and behavior (six items, P<0.0001) among group A subjects. The scales performed similarly well among group B subjects, and among subjects of different ages and gender. In group A and B subjects, respectively, a selected criterion SRBD score produced a sensitivity of 0.85 and 0.81; a specificity of 0.87 and 0.87; and a correct classification for 86 and 85% of subjects. The scales showed good internal consistency and, in a separate sample (n=21), good test-retest stability.Conclusions: These scales for childhood SRBDs, snoring, sleepiness, and behavior are valid and reliable instruments that can be used to identify SRBDs or associated symptom-constructs in clinical research when polysomnography is not feasible.

Sleep-Disordered Breathing, Behavior, and Cognition in Children Before and After Adenotonsillectomy

OBJECTIVES. Most children with sleep-disordered breathing (SDB) have mild-to-moderate forms, for which neurobehavioral complications are believed to be the most important adverse outcomes. To improve understanding of this morbidity, its long-term response to adenotonsillectomy, and its relationship to polysomnographic measures, we studied a series of children before and after clinically indicated adenotonsillectomy or unrelated surgical care. METHODS. We recorded sleep and assessed behavioral, cognitive, and psychiatric morbidity in 105 children 5.0 to 12.9 years old: 78 were scheduled for clinically indicated adenotonsillectomy, usually for suspected SDB, and 27 for unrelated surgical care. One year later, we repeated all assessments in 100 of these children. RESULTS. Subjects who had an adenotonsillectomy, in comparison to controls, were more hyperactive on well-validated parent rating scales, inattentive on cognitive testing, sleepy on the Multiple Sleep Latency Test, and likely to have attention-deficit/hyperactivity disorder (as defined by the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition) as judged by a child psychiatrist. In contrast, 1 year later, the 2 groups showed no significant differences in the same measures. Subjects who had an adenotonsillectomy had improved substantially in all measures, and control subjects improved in none. However, polysomnographic assessment of baseline SDB and its subsequent amelioration did not clearly predict either baseline neurobehavioral morbidity or improvement in any area other than sleepiness. CONCLUSIONS. Children scheduled for adenotonsillectomy often have mild-to-moderate SDB and significant neurobehavioral morbidity, including hyperactivity, inattention, attention-deficit/hyperactivity disorder, and excessive daytime sleepiness, all of which tend to improve by 1 year after surgery. However, the lack of better correspondence between SDB measures and neurobehavioral outcomes suggests the need for better measures or improved understanding of underlying causal mechanisms.

Conduct problems and symptoms of sleep disorders in children.

OBJECTIVE Conduct problems and hyperactivity are frequent among children referred for sleep-disordered breathing (SDB), restless legs syndrome, or periodic leg movements during sleep (PLMS), but children not referred to sleep centers have received little study. METHOD Parents of children aged 2 to 14 years were surveyed at two general clinics between 1998 and 2000. A Pediatric Sleep Questionnaire generated validated scores for SDB and PLMS. The Conners Parent Rating Scale (CPRS-48) produced an age- and sex-adjusted Conduct Problem Index (CPI) and Hyperactivity Index. RESULTS Parents of about 1,400 children were approached; those of 872 (62%) completed the surveys. Bullying and other specific aggressive behaviors were generally two to three times more frequent among 114 children at high risk for SDB than among the remaining children. An association between high CPI and SDB scores (p <.0001) retained significance after adjustment for sleepiness, high Hyperactivity Index, stimulant use, or PLMS scores. Analogous results were obtained for the association between high CPI and PLMS scores. CONCLUSIONS Conduct problems were associated with symptoms of SDB, restless legs syndrome, and PLMS. Although these results cannot prove a cause-and-effect relationship, assessment for sleep disorders may provide a new treatment opportunity for some aggressive children.

Neuropsychological and behavioral functioning in children with and without obstructive sleep apnea referred for tonsillectomy.

Adenotonsillectomy (AT) is among the most common pediatric surgical procedures and is performed as often for obstructive sleep apnea (OSA) as for recurrent tonsillitis. This study compared behavioral, cognitive, and sleep measures in 27 healthy control children recruited from a university hospital-based pediatric general surgery clinic with 40 children who had OSA (AT/OSA+) and 27 children who did not have OSA (AT/OSA-) scheduled for AT. Parental ratings of behavior, sleep problems, and snoring, along with specific cognitive measures (i.e., short-term attention, visuospatial problem solving, memory, arithmetic) reflected greater difficulties for AT children compared with controls. Differences between the AT/OSA- and control groups were larger and more consistent across test measures than were those between the AT/OSA+ and control groups. The fact that worse outcomes were not clearly demonstrated for the AT/OSA+ group compared with the other groups was not expected based on existing literature. This counterintuitive finding may reflect a combination of factors, including age, daytime sleepiness, features of sleep-disordered breathing too subtle to show on standard polysomnography, and academic or environmental factors not collected in this study. These results underscore the importance of applying more sophisticated methodologies to better understand the salient pathophysiology of childhood sleep-disordered breathing.

Changes in neuropsychological and behavioral functioning in children with and without obstructive sleep apnea following Tonsillectomy.

The most common treatment for sleep disordered breathing (SDB) is adenotonsillectomy (AT). Following AT, SDB resolves in most cases, and gains in cognitive and behavior scores are consistently reported, although persistent neuropsychological deficits or further declines also have been noted. This study presents results of the comprehensive 1-year follow-up neuropsychological examinations for children in the Washtenaw County Adenotonsillectomy Cohort I (95% return rate). After adjusting for normal developmental and practice-effect related changes in control children, significant improvements 1 year following AT were noted in polysomnography and sleepiness, as well as parental reports of behavior, although cognitive outcomes were mixed. Children undergoing AT with and without polysomnography-confirmed obstructive sleep apnea improved across a range of academic achievement measures, a measure of delayed visual recall, short-term attention/working memory, and executive functioning, along with parental ratings of behavior. On the other hand, measures of verbal abstraction ability, arithmetic calculations, visual and verbal learning, verbal delayed recall, sustained attention, and another measure of visual delayed recall demonstrated declines in ability, while other measures did not improve over time. These findings call into question the expectation that AT resolves most or all behavioral and cognitive difficulties in children with clinical, office-based diagnoses of SDB.

Esophageal Pressures, Polysomnography, and Neurobehavioral Outcomes of Adenotonsillectomy in Children

BACKGROUND Esophageal pressure monitoring during polysomnography in children offers a gold-standard, “preferred” assessment for work of breathing, but is not commonly used in part because prospective data on incremental clinical utility are scarce. We compared a standard pediatric apnea/hypopnea index to quantitative esophageal pressures as predictors of apnea-related neurobehavioral morbidity and treatment response. METHODS Eighty-one children aged 7.8 ± 2.8 (SD) years, including 44 boys, had traditional laboratory-based pediatric polysomnography, esophageal pressure monitoring, multiple sleep latency tests, psychiatric evaluations, parental behavior rating scales, and cognitive testing, all just before clinically indicated adenotonsillectomy, and again 7.2 ± 0.8 months later. Esophageal pressures were used, along with nasal pressure monitoring and oronasal thermocouples, not only to identify respiratory events but also more quantitatively to determine the most negative esophageal pressure recorded and the percentage of sleep time spent with pressures lower than -10 cm H(2)O. RESULTS Both sleep-disordered breathing and neurobehavioral measures improved after surgery. At baseline, one or both quantitative esophageal pressure measures predicted a disruptive behavior disorder (Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition-defined attention-deficit/hyperactivity disorder, conduct disorder, or oppositional defiant disorder) and more sleepiness and their future improvement after adenotonsillectomy (each P < .05). The pediatric apnea/hypopnea index did not predict these morbidities or treatment outcomes (each P > .10). The addition of respiratory effort-related arousals to the apnea/hypopnea index did not improve its predictive value. Neither the preoperative apnea/hypopnea index nor esophageal pressures predicted baseline hyperactive behavior, cognitive performance, or their improvement after surgery. CONCLUSIONS Quantitative esophageal pressure monitoring may add predictive value for some, if not all, neurobehavioral outcomes of sleep-disordered breathing.

Plasma catecholamines and social behavior in male vervet monkeys (Cercopithecus aethiops sabaeus).

Many investigations in humans indicate that epinephrine, norepinephrine and their ratio may correlate with such traits as social competence, academic achievement, and aggression. However, the socioeconomic, dietary, and environmental confounds accompanying most human studies complicate their interpretation. Social status, aggression, and other social behaviors can be reliably assessed in nonhuman primates under conditions controlling for crucial environmental factors. If interpretation of human studies is correct, dominant and subordinate male vervet monkeys should exhibit distinctive patterns of catecholamine secretion. To test this possibility, seventeen adult male monkeys living in six stable social groups were observed for 6 months. Based on their success in agonistic events, subjects were categorized as dominant or subordinate. Alpha scores were calculated from empirically derived factors to provide a noncategorical measure of dominant behavioral style. Plasma epinephrine and norepinephrine samples obtained from anesthetized subjects did not differ between dominant and subordinate males. Alpha scores, however, distinguished high from low norepinephrine/epinephrine ratio groups. These findings are consistent with studies in humans linking high epinephrine, low norepinephrine, and social competence.

Self-Injurious Behavior Associated With Clonidine Withdrawal in a Child With Tourette's Disorder

A 7-year-old boy with Tourettes disorder, atypical pervasive developmental disorder, borderline mental retardation, and a history of self-injurious behavior was treated for 21 months with clonidine transdermal patches at doses ranging from 0.1 to 0.5 mg weekly. When withdrawn from clonidine over 4 weeks to assess the need for continued therapy, the patient developed multiple self-destructive behaviors involving the theme of suffocation. The importance of careful clinical monitoring of the behavior of patients undergoing withdrawal from prolonged treatment with high doses of clonidine is emphasized. (J Child Neurol 1990;5:308-310).

Pediatric Sleep Questionnaire Scores Predict PostOp Improvement on PSG and Behavior

Problem: Children scheduled for adenotonsillectomy frequently also have reversible disruptive behavior disorders (DBD). We examined whether preoperative scores on a sleep questionnaire could assist in predicting postoperative improvement of both polysomnographic variables and disruptive behavior disorders. Methods: Sixty children, (mean age, 8.3 years) were studied. Behavior assessment, a sleep questionnaire, and nocturnal polysomnography were completed shortly before and 1 year after adenotonsillectomy. Psychiatric evaluation was performed by a child psychiatrist. The Sleep Disordered Breathing Subscale Questionnaire included questions pertaining to symptoms of nocturnal obstruction and behavior items. Obstructive apneas and hypopneas were defined as 2 breaths or longer in duration. Rates of respiratory event-related arousals plus apneas and hypopneas defined the respiratory disturbance index (RDI). Results: Severity of SDB generally improved after adeno-tonsillectomy. DBDs were initially diagnosed in 25 (42%) of subjects and resolved in 12 (48%) of the diagnosed children postoperatively. There were no significant differences noted in the preoperative obstructive apnea index, apnea/hypopnea index, RDI or minimum oxygen saturation (t tests, all P > 0.05) in children whose DBD resolved as compared to those whose DBD did not. There were, however, significant correlations between high scores on the preoperative SDB subscale and postoperative improvement in apnea/hypopnea index (Pearson correlation coefficient r = .28, P < 0.03), obstructive apnea index (r = 0.32, P < 0.02), and RDI (r = 0.28, P < 0.04). There was also a strong correlation seen between high preoperative questionnaire scores and resolution of DBD (r = 0.28, P < 0.02). Conclusion: Questionnaires about preoperative symptoms of SDB are predictive of improvement in polysomnographic abnormalities and DBD after adenotonsillectomy. The use of a Pediatric Sleep Questionnaire may be useful in determining which patients have behavioral disorders potentially treatable with surgical intervention. Significance: Questionnaires may provide a simple, inexpensive method of predicting clinical improvement after adenotonsillectomy in children with SDB and DBDs. Support: None reported.