Introduction: Insufficient sleep negatively impacts the cardiovascular system. Studies have also ... more Introduction: Insufficient sleep negatively impacts the cardiovascular system. Studies have also shown associations between sleep duration and several CVD risk factors. No study has examined the association between sleep duration and heart age (predicted age of a person's vascular system based on their cardiovascular risk factor profile), a simplified way to express CVD risk. This study examines the association between sleep duration and excess heart age (EHA) (difference between heart age and chronological age) among U.S. adults. Methods: Pooled 2007-2014 National Health and Nutrition Examination Survey data were used. Self-reported sleep duration was classified into 5 categories (≤5, 6, 7, 8, and ≥9 hours of sleep per night). We used the sex-specific Framingham heart age algorithm to calculate each individual's heart age and used multivariable linear or logistic regression to examine the association between sleep duration and EHA or risk of EHA ≥10 years.
STUDY OBJECTIVES In this study we investigated the clinical correlates of restless legs syndrome ... more STUDY OBJECTIVES In this study we investigated the clinical correlates of restless legs syndrome in children with autism and report on our experiences with response to treatment. METHODS A retrospective chart review of children seen in our sleep center from 2016-2019 was performed to identify children with autism and chronic insomnia. Patients underwent clinical assessments for restless legs symptomatology. Overnight polysomnogram, serum ferritin testing, and response to clinical treatment data were collected. RESULTS A total of 103 children with autism and chronic insomnia were identified (age range 2 - 19 years). Of these, 41 children (39%) were diagnosed with restless legs syndrome. The diagnosis of restless legs syndrome was associated with significantly lower serum ferritin levels (mean 29 ±18.62 ng/mL versus non-RLS 56.7 ± 17.59, P<0.001) and higher PLMS on PSG (8.12 ± 6.6 versus non-RLS 0.06 ±0.17). The presence of leg kicking, body rocking, or any symptoms involving the legs, highly correlated with the diagnosis of RLS. Positive treatment response was noted in nearly all treated patients, including those treated with oral iron supplementation alone (25 children, 23 responders), gabapentin alone (12 children, all responders), and combination therapy (3 children, all responders). CONCLUSIONS Our findings suggest restless legs syndrome may represent an underrecognized cause of insomnia in children with autism. Initial assessment should include a thorough query of behaviors related to nocturnal motor complaints, because restless legs syndrome may be a treatable cause of sleep disruption.
Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is asso... more Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is associated with reduced functional recovery and an increased risk for recurrent vascular events. Aims and/or hypothesis The Sleep for Stroke Management and Recovery Trial (Sleep SMART) aims to determine whether automatically adjusting continuous positive airway pressure (aCPAP) treatment for obstructive sleep apnea improves clinical outcomes after acute ischemic stroke or high-risk transient ischemic attack. Sample size estimate A total of 3062 randomized subjects for the prevention of recurrent serious vascular events, and among these, 1362 stroke survivors for the recovery outcome. Methods and design Sleep SMART is a phase III, multicenter, prospective randomized, open, blinded outcome event assessed controlled trial. Adults with recent acute ischemic stroke/transient ischemic attack and no contraindication to aCPAP are screened for obstructive sleep apnea with a portable sleep apnea test....
Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is asso... more Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is associated with reduced functional recovery and an increased risk for recurrent vascular events. Aims and/or hypothesis The Sleep for Stroke Management and Recovery Trial (Sleep SMART) aims to determine whether automatically adjusting continuous positive airway pressure (aCPAP) treatment for obstructive sleep apnea improves clinical outcomes after acute ischemic stroke or high-risk transient ischemic attack. Sample size estimate A total of 3062 randomized subjects for the prevention of recurrent serious vascular events, and among these, 1362 stroke survivors for the recovery outcome. Methods and design Sleep SMART is a phase III, multicenter, prospective randomized, open, blinded outcome event assessed controlled trial. Adults with recent acute ischemic stroke/transient ischemic attack and no contraindication to aCPAP are screened for obstructive sleep apnea with a portable sleep apnea test....
Cats rendered hemianopic by a unilateral visual cortical ablation can recover the visual orientin... more Cats rendered hemianopic by a unilateral visual cortical ablation can recover the visual orienting response in the hemianopic visual field following disruption of the caudal non-tectotectal containing half of the commissure of the superior colliculus. Ibotenic acid lesions of a small &#39;critical zone&#39; in the contralateral substantia nigra result in a similar recovery effect. A conceptual framework developed by Wallace et al. (1990) [J. Comp. Neurol. 296, 222-252] proposed that elimination of contralateral substantia nigra &#39;critical zone&#39; inhibition on the superior colliculus ipsilateral to a visual cortical lesion is responsible for the recovery. This model is insufficient, however, to explain the observation that hemi-decorticate cats with contralateral substantia nigra &#39;critical zone&#39; lesions which include but extend beyond the &#39;critical zone&#39; do not demonstrate the recovery. In these cats, subsequent transection of the commissure of the superior colliculus does lead to the recovery. We hypothesize that another projection through the caudal commissure of the superior colliculus, from the pedunculopontine nucleus, is involved in the recovery effect. Visual orienting behavior was recorded before and after ibotenic acid lesions made in the pedunculopontine nucleus region contralateral to a visual cortical ablation in 16 cats. Four cats with lesions in a small rostral region of the contralateral pedunculopontine nucleus recovered the visual orienting response in the previously hemianopic visual field. Contralateral tectal projections from the pedunculopontine nucleus are thought to be cholinergic and terminate as distinct patches in the intermediate gray layers of the superior colliculus. Since this region of the pedunculopontine nucleus also receives GABA-ergic afferents from the substantia nigra, we propose that a subcortical neural circuit including the substantia nigra, pedunculopontine nucleus, and superior colliculus is involved in the recovery of visual orienting.
Background: Specific diagnostic criteria for pediatric restless legs syndrome (RLS) were publishe... more Background: Specific diagnostic criteria for pediatric restless legs syndrome (RLS) were published in 2003 following a workshop at the National Institutes of Health. Due to substantial new research and revision of the adult RLS diagnostic criteria, a task force was chosen by the International Restless Legs Syndrome Study Group (IRLSSG) to consider updates to the pediatric diagnostic criteria. Methods: A committee of seven pediatric RLS experts developed a set of 15 consensus questions to review, conducted a comprehensive literature search, and extensively discussed potential revisions. The committee recommendations were approved by the IRLSSG executive committee and reviewed by the IRLSSG membership. Results: The pediatric RLS diagnostic criteria were simplified and integrated with the newly revised adult RLS criteria. Specific recommendations were developed for pediatric application of the criteria, including consideration of typical words used by children to describe their symptoms. Pediatric aspects of differential diagnosis, comorbidity, and clinical significance were then defined. In addition, the research criteria for probable and possible pediatric RLS were updated and criteria for a related condition, periodic limb movement disorder (PLMD), were clarified. Conclusions: Revised diagnostic criteria for pediatric RLS have been developed, which are intended to improve clinical practice and promote further research.
We compared the effects of unilateral surgical aspiration and ibotenic acid produced lesions of t... more We compared the effects of unilateral surgical aspiration and ibotenic acid produced lesions of the superior colliculus (SC) on visual orienting behavior in 20 cats. Four animals with aspiration lesions initially showed an hemianopia in the contralateral hemifield which recovered fully in 4.5 weeks or less. These lesions also destroyed axons in the commissure of the superior colliculus (CS). In 9 animals we produced complete loss of cells in one SC, with preservation of axons in the CSC, by injections of ibotenic acid. In these animals the contralateral hemianopia persisted for an average of 16.6 weeks, but may have persisted longer had we not intervened by either sacrificing the animal or ablating the visual cortex contralateral to the SC lesion. The cortical lesion produced an immediate hemianopia in the contralateral hemifield and a recovery in the previously hemianopic (&#39;collicular&#39;) hemifield. In the remaining 7 animals with attempted ibotenic acid lesions, 5 had incomplete lesions and 2 others sustained major damage to the SC as well as the CSC. These 7 animals recovered visual orienting on an average of 3.0 weeks postoperatively. We conclude that unilateral loss of collicular cell function and the presence of fibers coursing through the commissure of the superior colliculus are both necessary for the prolonged deficit in visual orienting behavior. We suggest that competition between the two hemifields may play a role in the hemianopia caused by collicular manipulations and that the cholinergic pathway from the pedunculopontine nucleus to the contralateral SC via the CSC may be involved.
Deficits in daytime performance due to sleep loss are experienced universally and associated with... more Deficits in daytime performance due to sleep loss are experienced universally and associated with a significant social, financial, and human cost. Microsleeps, sleep attacks, and lapses in cognition increase with sleep loss as a function of state instability. Sleep deprivation studies repeatedly show a variable (negative) impact on mood, cognitive performance, and motor function due to an increasing sleep propensity and destabilization of the wake state. Specific neurocognitive domains including executive attention, working memory, and divergent higher cognitive functions are particularly vulnerable to sleep loss. In humans, functional metabolic and neurophysiological studies demonstrate that neural systems involved in executive function (i.e., prefrontal cortex) are more susceptible to sleep deprivation in some individuals than others. Recent chronic partial sleep deprivation experiments, which more closely replicate sleep loss in society, demonstrate that profound neurocognitive d...
Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine, 2014
Current emphasis on patient outcomes within sleep medicine, with a particular focus on quality im... more Current emphasis on patient outcomes within sleep medicine, with a particular focus on quality improvement and contained costs, calls for sleep specialists to develop innovative models for long-term care and management of sleep disorders patients. Multidisciplinary sleep centers can facilitate highest-quality care that is timely and cost-effective. Effective resource use in a multidisciplinary sleep center can help minimize fragmentation of care, reduce effort duplication, and control costs. Proposed strategies to help achieve a balance between quality of care and cost-effectiveness include: (1) multidisciplinary specialty clinics, (2) optimized use of information technology, and (3) adoption of reliable performance measures.
Objective: To develop a questionnaire to measure Pediatric Restless Legs Syndrome (P-RLS) symptom... more Objective: To develop a questionnaire to measure Pediatric Restless Legs Syndrome (P-RLS) symptoms and impact for use in clinical research. Methods: Questionnaire items were developed based on open-ended, qualitative interviews of 33 children and adolescents diagnosed with definite RLS (ages 6-17 years) and their parents. The draft questionnaire was then tested through cognitive debriefing interviews with 21 of the same children/adolescents and 15 of their parents. This involved the children and parents answering the draft items and then interviewing them about the child's ability to understand and interpret the questionnaire. Expert clinicians provided clinical guidance throughout. Results: Draft severity questions were generated to measure the four-symptom and four-impact domains identified from the concept elicitation interviews: RLS sensations, move/rub due to RLS, relief from move/ rub, pain, and impact of RLS on sleep, awake activities, emotions, and tiredness. RLS descriptions, symptoms, and impact were compared between those who had comorbid attention-deficit/hyperactivity disorder and those who did not. Revisions to several questions were made based on the cognitive debriefing interviews and expert clinician review, resulting in a severity scale with 17 morning and 24 evening items. Caution regarding self-administration in children ages 6-8 years is recommended. To complement the child/adolescent measures, a separate parent questionnaire was also developed. Conclusions: The P-RLS-SS was constructed based on detailed input from children and adolescents with RLS, their parents, and clinical experts, thus providing a scale with strong content validity that is intended to be comprehensive, clinically relevant, and important to patients. Validation of this scale is recommended.
Deficits in daytime performance due to sleep loss are experienced universally and associated with... more Deficits in daytime performance due to sleep loss are experienced universally and associated with a significant social, financial, and human cost. Microsleeps, sleep attacks, and lapses in cognition increase with sleep loss as a function of state instability. Sleep deprivation studies repeatedly show a variable (negative) impact on mood, cognitive performance, and motor function due to an increasing sleep propensity and destabilization of the wake state. Specific neurocognitive domains including executive attention, working memory, and divergent higher cognitive functions are particularly vulnerable to sleep loss. In humans, functional metabolic and neurophysiological studies demonstrate that neural systems involved in executive function (i.e., prefrontal cortex) are more susceptible to sleep deprivation in some individuals than others. Recent chronic partial sleep deprivation experiments, which more closely replicate sleep loss in society, demonstrate that profound neurocognitive deficits accumulate over time in the face of subjective adaptation to the sensation of sleepiness. Sleep deprivation associated with disease-related sleep fragmentation (i.e., sleep apnea and restless legs syndrome) also results in neurocognitive performance decrements similar to those seen in sleep restriction studies. Performance deficits associated with sleep disorders are often viewed as a simple function of disease severity; however, recent experiments suggest that individual vulnerability to sleep loss may play a more critical role than previously thought.
The characteristic symptoms of restless legs syndrome (RLS) have been known for hundreds of years... more The characteristic symptoms of restless legs syndrome (RLS) have been known for hundreds of years and were first reported in medicine in the 1600s. Clinicians must consider potential mimics, comorbid, and associated conditions when evaluating children with RLS symptoms. The traditional differentiation of RLS from periodic limb movement disorder (PLMD) is noted in children as well as adults. Because current pediatric RLS research is sparse, this article provides the most up-to-date evidence-based as well as consensus opinion-based information on the subject of childhood RLS and PLMD. Prevalence, pathophysiology, diagnosis, treatment, and clinical associations are discussed.
The specific aims of this study were to collect and analyze detailed symptom descriptions from pa... more The specific aims of this study were to collect and analyze detailed symptom descriptions from patients with pediatric restless legs syndrome, ages 6 to 17 years, as well as assess symptom impact and the usefulness of drawings. Trained qualitative interviewers conducted face-to-face audio-recorded interviews of children and adolescents who met criteria for definite restless legs syndrome. Thirty-three patients in 3 age groups used 16 different categories of descriptors for restless legs sensations, with a mean of 3 or more categories used per patient in each age group. ''Need to move/kick,'' ''pain/hurts,'' ''uncomfortable/ cannot get comfortable,'' and ''like bugs or ants/crawling'' were the most common descriptors. Two-thirds reported daytime sensations, and nearly half had arm involvement. They described impact on sleep, cognitive function, and affect. Drawings provided useful diagnostic information. These detailed empirical data will be useful in clinical practice, as well as in the development of formal diagnostic tools and severity measures.
Introduction: Insufficient sleep negatively impacts the cardiovascular system. Studies have also ... more Introduction: Insufficient sleep negatively impacts the cardiovascular system. Studies have also shown associations between sleep duration and several CVD risk factors. No study has examined the association between sleep duration and heart age (predicted age of a person's vascular system based on their cardiovascular risk factor profile), a simplified way to express CVD risk. This study examines the association between sleep duration and excess heart age (EHA) (difference between heart age and chronological age) among U.S. adults. Methods: Pooled 2007-2014 National Health and Nutrition Examination Survey data were used. Self-reported sleep duration was classified into 5 categories (≤5, 6, 7, 8, and ≥9 hours of sleep per night). We used the sex-specific Framingham heart age algorithm to calculate each individual's heart age and used multivariable linear or logistic regression to examine the association between sleep duration and EHA or risk of EHA ≥10 years.
STUDY OBJECTIVES In this study we investigated the clinical correlates of restless legs syndrome ... more STUDY OBJECTIVES In this study we investigated the clinical correlates of restless legs syndrome in children with autism and report on our experiences with response to treatment. METHODS A retrospective chart review of children seen in our sleep center from 2016-2019 was performed to identify children with autism and chronic insomnia. Patients underwent clinical assessments for restless legs symptomatology. Overnight polysomnogram, serum ferritin testing, and response to clinical treatment data were collected. RESULTS A total of 103 children with autism and chronic insomnia were identified (age range 2 - 19 years). Of these, 41 children (39%) were diagnosed with restless legs syndrome. The diagnosis of restless legs syndrome was associated with significantly lower serum ferritin levels (mean 29 ±18.62 ng/mL versus non-RLS 56.7 ± 17.59, P<0.001) and higher PLMS on PSG (8.12 ± 6.6 versus non-RLS 0.06 ±0.17). The presence of leg kicking, body rocking, or any symptoms involving the legs, highly correlated with the diagnosis of RLS. Positive treatment response was noted in nearly all treated patients, including those treated with oral iron supplementation alone (25 children, 23 responders), gabapentin alone (12 children, all responders), and combination therapy (3 children, all responders). CONCLUSIONS Our findings suggest restless legs syndrome may represent an underrecognized cause of insomnia in children with autism. Initial assessment should include a thorough query of behaviors related to nocturnal motor complaints, because restless legs syndrome may be a treatable cause of sleep disruption.
Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is asso... more Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is associated with reduced functional recovery and an increased risk for recurrent vascular events. Aims and/or hypothesis The Sleep for Stroke Management and Recovery Trial (Sleep SMART) aims to determine whether automatically adjusting continuous positive airway pressure (aCPAP) treatment for obstructive sleep apnea improves clinical outcomes after acute ischemic stroke or high-risk transient ischemic attack. Sample size estimate A total of 3062 randomized subjects for the prevention of recurrent serious vascular events, and among these, 1362 stroke survivors for the recovery outcome. Methods and design Sleep SMART is a phase III, multicenter, prospective randomized, open, blinded outcome event assessed controlled trial. Adults with recent acute ischemic stroke/transient ischemic attack and no contraindication to aCPAP are screened for obstructive sleep apnea with a portable sleep apnea test....
Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is asso... more Rationale Obstructive sleep apnea is common among patients with acute ischemic stroke and is associated with reduced functional recovery and an increased risk for recurrent vascular events. Aims and/or hypothesis The Sleep for Stroke Management and Recovery Trial (Sleep SMART) aims to determine whether automatically adjusting continuous positive airway pressure (aCPAP) treatment for obstructive sleep apnea improves clinical outcomes after acute ischemic stroke or high-risk transient ischemic attack. Sample size estimate A total of 3062 randomized subjects for the prevention of recurrent serious vascular events, and among these, 1362 stroke survivors for the recovery outcome. Methods and design Sleep SMART is a phase III, multicenter, prospective randomized, open, blinded outcome event assessed controlled trial. Adults with recent acute ischemic stroke/transient ischemic attack and no contraindication to aCPAP are screened for obstructive sleep apnea with a portable sleep apnea test....
Cats rendered hemianopic by a unilateral visual cortical ablation can recover the visual orientin... more Cats rendered hemianopic by a unilateral visual cortical ablation can recover the visual orienting response in the hemianopic visual field following disruption of the caudal non-tectotectal containing half of the commissure of the superior colliculus. Ibotenic acid lesions of a small &#39;critical zone&#39; in the contralateral substantia nigra result in a similar recovery effect. A conceptual framework developed by Wallace et al. (1990) [J. Comp. Neurol. 296, 222-252] proposed that elimination of contralateral substantia nigra &#39;critical zone&#39; inhibition on the superior colliculus ipsilateral to a visual cortical lesion is responsible for the recovery. This model is insufficient, however, to explain the observation that hemi-decorticate cats with contralateral substantia nigra &#39;critical zone&#39; lesions which include but extend beyond the &#39;critical zone&#39; do not demonstrate the recovery. In these cats, subsequent transection of the commissure of the superior colliculus does lead to the recovery. We hypothesize that another projection through the caudal commissure of the superior colliculus, from the pedunculopontine nucleus, is involved in the recovery effect. Visual orienting behavior was recorded before and after ibotenic acid lesions made in the pedunculopontine nucleus region contralateral to a visual cortical ablation in 16 cats. Four cats with lesions in a small rostral region of the contralateral pedunculopontine nucleus recovered the visual orienting response in the previously hemianopic visual field. Contralateral tectal projections from the pedunculopontine nucleus are thought to be cholinergic and terminate as distinct patches in the intermediate gray layers of the superior colliculus. Since this region of the pedunculopontine nucleus also receives GABA-ergic afferents from the substantia nigra, we propose that a subcortical neural circuit including the substantia nigra, pedunculopontine nucleus, and superior colliculus is involved in the recovery of visual orienting.
Background: Specific diagnostic criteria for pediatric restless legs syndrome (RLS) were publishe... more Background: Specific diagnostic criteria for pediatric restless legs syndrome (RLS) were published in 2003 following a workshop at the National Institutes of Health. Due to substantial new research and revision of the adult RLS diagnostic criteria, a task force was chosen by the International Restless Legs Syndrome Study Group (IRLSSG) to consider updates to the pediatric diagnostic criteria. Methods: A committee of seven pediatric RLS experts developed a set of 15 consensus questions to review, conducted a comprehensive literature search, and extensively discussed potential revisions. The committee recommendations were approved by the IRLSSG executive committee and reviewed by the IRLSSG membership. Results: The pediatric RLS diagnostic criteria were simplified and integrated with the newly revised adult RLS criteria. Specific recommendations were developed for pediatric application of the criteria, including consideration of typical words used by children to describe their symptoms. Pediatric aspects of differential diagnosis, comorbidity, and clinical significance were then defined. In addition, the research criteria for probable and possible pediatric RLS were updated and criteria for a related condition, periodic limb movement disorder (PLMD), were clarified. Conclusions: Revised diagnostic criteria for pediatric RLS have been developed, which are intended to improve clinical practice and promote further research.
We compared the effects of unilateral surgical aspiration and ibotenic acid produced lesions of t... more We compared the effects of unilateral surgical aspiration and ibotenic acid produced lesions of the superior colliculus (SC) on visual orienting behavior in 20 cats. Four animals with aspiration lesions initially showed an hemianopia in the contralateral hemifield which recovered fully in 4.5 weeks or less. These lesions also destroyed axons in the commissure of the superior colliculus (CS). In 9 animals we produced complete loss of cells in one SC, with preservation of axons in the CSC, by injections of ibotenic acid. In these animals the contralateral hemianopia persisted for an average of 16.6 weeks, but may have persisted longer had we not intervened by either sacrificing the animal or ablating the visual cortex contralateral to the SC lesion. The cortical lesion produced an immediate hemianopia in the contralateral hemifield and a recovery in the previously hemianopic (&#39;collicular&#39;) hemifield. In the remaining 7 animals with attempted ibotenic acid lesions, 5 had incomplete lesions and 2 others sustained major damage to the SC as well as the CSC. These 7 animals recovered visual orienting on an average of 3.0 weeks postoperatively. We conclude that unilateral loss of collicular cell function and the presence of fibers coursing through the commissure of the superior colliculus are both necessary for the prolonged deficit in visual orienting behavior. We suggest that competition between the two hemifields may play a role in the hemianopia caused by collicular manipulations and that the cholinergic pathway from the pedunculopontine nucleus to the contralateral SC via the CSC may be involved.
Deficits in daytime performance due to sleep loss are experienced universally and associated with... more Deficits in daytime performance due to sleep loss are experienced universally and associated with a significant social, financial, and human cost. Microsleeps, sleep attacks, and lapses in cognition increase with sleep loss as a function of state instability. Sleep deprivation studies repeatedly show a variable (negative) impact on mood, cognitive performance, and motor function due to an increasing sleep propensity and destabilization of the wake state. Specific neurocognitive domains including executive attention, working memory, and divergent higher cognitive functions are particularly vulnerable to sleep loss. In humans, functional metabolic and neurophysiological studies demonstrate that neural systems involved in executive function (i.e., prefrontal cortex) are more susceptible to sleep deprivation in some individuals than others. Recent chronic partial sleep deprivation experiments, which more closely replicate sleep loss in society, demonstrate that profound neurocognitive d...
Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine, 2014
Current emphasis on patient outcomes within sleep medicine, with a particular focus on quality im... more Current emphasis on patient outcomes within sleep medicine, with a particular focus on quality improvement and contained costs, calls for sleep specialists to develop innovative models for long-term care and management of sleep disorders patients. Multidisciplinary sleep centers can facilitate highest-quality care that is timely and cost-effective. Effective resource use in a multidisciplinary sleep center can help minimize fragmentation of care, reduce effort duplication, and control costs. Proposed strategies to help achieve a balance between quality of care and cost-effectiveness include: (1) multidisciplinary specialty clinics, (2) optimized use of information technology, and (3) adoption of reliable performance measures.
Objective: To develop a questionnaire to measure Pediatric Restless Legs Syndrome (P-RLS) symptom... more Objective: To develop a questionnaire to measure Pediatric Restless Legs Syndrome (P-RLS) symptoms and impact for use in clinical research. Methods: Questionnaire items were developed based on open-ended, qualitative interviews of 33 children and adolescents diagnosed with definite RLS (ages 6-17 years) and their parents. The draft questionnaire was then tested through cognitive debriefing interviews with 21 of the same children/adolescents and 15 of their parents. This involved the children and parents answering the draft items and then interviewing them about the child's ability to understand and interpret the questionnaire. Expert clinicians provided clinical guidance throughout. Results: Draft severity questions were generated to measure the four-symptom and four-impact domains identified from the concept elicitation interviews: RLS sensations, move/rub due to RLS, relief from move/ rub, pain, and impact of RLS on sleep, awake activities, emotions, and tiredness. RLS descriptions, symptoms, and impact were compared between those who had comorbid attention-deficit/hyperactivity disorder and those who did not. Revisions to several questions were made based on the cognitive debriefing interviews and expert clinician review, resulting in a severity scale with 17 morning and 24 evening items. Caution regarding self-administration in children ages 6-8 years is recommended. To complement the child/adolescent measures, a separate parent questionnaire was also developed. Conclusions: The P-RLS-SS was constructed based on detailed input from children and adolescents with RLS, their parents, and clinical experts, thus providing a scale with strong content validity that is intended to be comprehensive, clinically relevant, and important to patients. Validation of this scale is recommended.
Deficits in daytime performance due to sleep loss are experienced universally and associated with... more Deficits in daytime performance due to sleep loss are experienced universally and associated with a significant social, financial, and human cost. Microsleeps, sleep attacks, and lapses in cognition increase with sleep loss as a function of state instability. Sleep deprivation studies repeatedly show a variable (negative) impact on mood, cognitive performance, and motor function due to an increasing sleep propensity and destabilization of the wake state. Specific neurocognitive domains including executive attention, working memory, and divergent higher cognitive functions are particularly vulnerable to sleep loss. In humans, functional metabolic and neurophysiological studies demonstrate that neural systems involved in executive function (i.e., prefrontal cortex) are more susceptible to sleep deprivation in some individuals than others. Recent chronic partial sleep deprivation experiments, which more closely replicate sleep loss in society, demonstrate that profound neurocognitive deficits accumulate over time in the face of subjective adaptation to the sensation of sleepiness. Sleep deprivation associated with disease-related sleep fragmentation (i.e., sleep apnea and restless legs syndrome) also results in neurocognitive performance decrements similar to those seen in sleep restriction studies. Performance deficits associated with sleep disorders are often viewed as a simple function of disease severity; however, recent experiments suggest that individual vulnerability to sleep loss may play a more critical role than previously thought.
The characteristic symptoms of restless legs syndrome (RLS) have been known for hundreds of years... more The characteristic symptoms of restless legs syndrome (RLS) have been known for hundreds of years and were first reported in medicine in the 1600s. Clinicians must consider potential mimics, comorbid, and associated conditions when evaluating children with RLS symptoms. The traditional differentiation of RLS from periodic limb movement disorder (PLMD) is noted in children as well as adults. Because current pediatric RLS research is sparse, this article provides the most up-to-date evidence-based as well as consensus opinion-based information on the subject of childhood RLS and PLMD. Prevalence, pathophysiology, diagnosis, treatment, and clinical associations are discussed.
The specific aims of this study were to collect and analyze detailed symptom descriptions from pa... more The specific aims of this study were to collect and analyze detailed symptom descriptions from patients with pediatric restless legs syndrome, ages 6 to 17 years, as well as assess symptom impact and the usefulness of drawings. Trained qualitative interviewers conducted face-to-face audio-recorded interviews of children and adolescents who met criteria for definite restless legs syndrome. Thirty-three patients in 3 age groups used 16 different categories of descriptors for restless legs sensations, with a mean of 3 or more categories used per patient in each age group. ''Need to move/kick,'' ''pain/hurts,'' ''uncomfortable/ cannot get comfortable,'' and ''like bugs or ants/crawling'' were the most common descriptors. Two-thirds reported daytime sensations, and nearly half had arm involvement. They described impact on sleep, cognitive function, and affect. Drawings provided useful diagnostic information. These detailed empirical data will be useful in clinical practice, as well as in the development of formal diagnostic tools and severity measures.
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Papers by Jeffrey Durmer