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1.
Pediatr Pulmonol ; 2024 Jun 05.
Article in English | MEDLINE | ID: mdl-38837889

ABSTRACT

INTRODUCTION: Obstructive sleep apnea (OSA) in infants is treated with low flow oxygen via nasal cannula (NC), CPAP (continous positive airway pressure), or surgery. Literature supports the use of high flow NC (HFNC) in children in the outpatient setting, however there is limited data on the use of HFNC in infants. OBJECTIVE: The purpose of this study was to compare HFNC and low-flow oxygen as treatments for OSA in infants. METHODS: A prospective pilot study was performed at two institutions. Infants with primarily OSA underwent a 3-4 h sleep study with HFNC titration at 6-14 lpm for OSA, followed by clinical polysomnography (PSG) for oxygen titration (1/8-1 lpm). Infants with primarily central apnea were excluded. RESULTS: Nine infants were enrolled, with a mean age of 1.3 ± 1.7 months. Average apnea hypopnea index (AHI), average obstructive apnea hypopnea index (OAHI) and average central apnea index during the diagnostic PSG was 17.2 ± 7/h, 13.4 ± 5.4/h and 3.7 ± 4.8/h respectively. OSA improved in 44.4% of subjects with HFNC; the mean AHI and OAHI decreased from 15.6 ± 5.65/h and 12.8 ± 4.4/h on diagnostic PSG to 5.12 ± 2.5/h and 4.25 ± 2.5/h on titration PSG. OSA improved universally with low flow oxygen; the mean AHI decreased from 17.2 ± 7/h on diagnostic PSG to 4.44 ± 3.6/h on titration PSG. CONCLUSION: HFNC reduced OSA in some infants, though low flow oxygen reduced OSA in all subjects. Respiratory instability (high loop gain) in infants may explain why infants responded to low flow oxygen. More studies are needed to determine if HFNC is beneficial in selected groups of infants with OSA.

2.
J Clin Sleep Med ; 20(1): 9-16, 2024 Jan 01.
Article in English | MEDLINE | ID: mdl-37584453

ABSTRACT

STUDY OBJECTIVES: Postoperative respiratory complications (PORCs) can occur following supraglottoplasty (SGP) for obstructive sleep apnea. However, there are very limited data on risk factors associated with these complications. This study aims to evaluate the occurrence of PORC in children undergoing SGP and to assess clinical factors and polysomnographic parameters predicting these complications. METHODS: A retrospective study was performed in children with laryngomalacia who underwent SGP with preoperative polysomnography. RESULTS: 400 children who underwent SGP met the criteria for entry into the analysis with a total of 416 surgeries with corresponding preoperative polysomnography. The median age (interquartile range) at the time of polysomnography was 0.4 (0.2, 1.5) years. A total of 96 (23.1%) PORCs were noted. Compared with those without complications, children with PORCs had a higher proportion of congenital heart disease (P < .05), higher median obstructive apnea-hypopnea index (obstructive AHI; median 16.0 vs 11.4 events/h; P < .01), and lower median oxygen saturation (SpO2) (P < .001). The unadjusted odd ratio indicated an increased risk of PORCs in children with congenital heart disease (odds ratio 1.66; P < .05) and those with an obstructive AHI > 10 events/h (odds ratio 2.06; P < .01). Multiple regression analysis demonstrated that an obstructive AHI > 10 events/h was the only independent risk factor for PORCs (P < .05). CONCLUSIONS: In our cohort of children with laryngomalacia undergoing SGP, those with underlying congenital heart disease, higher obstructive AHI, and lower SpO2 were more likely to develop PORCs. Only children with an obstructive AHI > 10 events/h were at significantly increased risk for PORCs following SGP. Preoperative polysomnography is useful in preoperative planning in children undergoing SGP. CITATION: Kanavitoon S, Ngamprasertwong P, Gurbani N, et al. Polysomnographic parameters and clinical risk factors predicting postoperative respiratory complications in children undergoing supraglottoplasty. J Clin Sleep Med. 2024;20(1):9-16.


Subject(s)
Heart Defects, Congenital , Laryngomalacia , Sleep Apnea, Obstructive , Child , Humans , Infant , Laryngomalacia/complications , Laryngomalacia/surgery , Retrospective Studies , Sleep Apnea, Obstructive/complications , Sleep Apnea, Obstructive/surgery , Postoperative Complications/etiology , Risk Factors
3.
Semin Pediatr Neurol ; 48: 101092, 2023 12.
Article in English | MEDLINE | ID: mdl-38065635

ABSTRACT

Sleep disordered breathing (SDB) is prevalent among children with neuromuscular disorders (NMD). The combination of respiratory muscle weakness, altered drive, and chest wall distortion due to scoliosis make sleep a stressful state in this population. Symptomatology can range from absent to snoring, nocturnal awakenings, morning headaches, and excessive daytime sleepiness. Sequelae of untreated SDB includes cardiovascular effects, metabolic derangements, and neurocognitive concerns which can be compounded by those innate to the NMD. The clinician should have a low threshold for obtaining polysomnography and recognize the nuances of individual disorders due to disproportionately impacted muscle groups such as hypoventilation in ambulating patients from diaphragm weakness. Non-invasive or invasive ventilation are the mainstay of treatment. In this review we explore the diagnosis and treatment of SDB in children with various NMD.


Subject(s)
Neuromuscular Diseases , Sleep Apnea Syndromes , Humans , Child , Sleep , Sleep Apnea Syndromes/complications , Sleep Apnea Syndromes/diagnosis , Sleep Apnea Syndromes/therapy , Neuromuscular Diseases/complications , Neuromuscular Diseases/diagnosis , Neuromuscular Diseases/therapy , Hypoventilation/complications , Hypoventilation/therapy , Polysomnography
4.
Pediatr Pulmonol ; 58(12): 3458-3465, 2023 Dec.
Article in English | MEDLINE | ID: mdl-37701984

ABSTRACT

BACKGROUND: Sleep disordered breathing (SDB) may exacerbate asthma and is a treatable comorbidity. OBJECTIVE: To design and implement a screening process for SDB in patients hospitalized for asthma exacerbation using quality improvement (QI) methods. We sought to improve screening for SDB from zero to 60% from July 2019 to December 2020. DESIGN/METHODS: A multidisciplinary team used QI methods to screen for SDB using the Michigan pediatric sleep questionnaire (PSQ) in patients 2-18 years hospitalized for asthma exacerbation. Key interventions included: pairing the PSQ screen with another element of routine care (the asthma risk factor screen), educating staff and physicians, engaging respiratory therapists to complete the PSQ and document scores, and modifying the electronic medical record (asthma order set and flowsheet for PSQ score documentation). A run chart tracked progress and descriptive statistics were generated. RESULTS: There were 2067 patients admitted for asthma exacerbation during this project. The PSQ was completed for 1531 patients (74%) overall. Of screened patients, 360 (24%) had a positive PSQ; the mean age was 8.6 years. Approximately 14 months after the project began, ~90% of children admitted for asthma were being screened; subsequently, >80% of patients were being screened until May 2022. Screening with the PSQ occurred approximately 90% of the time when routine asthma risk screens were completed. CONCLUSION: A screening process for SDB was successfully implemented and appeared feasible and sustainable. The high proportion of positive screens reinforces the importance of evaluating for SDB in the high-risk population of children requiring hospitalization for asthma exacerbation.


Subject(s)
Asthma , Sleep Apnea Syndromes , Child , Humans , Sleep Apnea Syndromes/complications , Sleep Apnea Syndromes/diagnosis , Sleep Apnea Syndromes/epidemiology , Sleep , Comorbidity , Asthma/complications , Asthma/diagnosis , Asthma/epidemiology , Surveys and Questionnaires
5.
Pediatr Pulmonol ; 58(12): 3566-3573, 2023 Dec.
Article in English | MEDLINE | ID: mdl-37728219

ABSTRACT

BACKGROUND: Obstructive sleep apnea (OSA) has been associated with pulmonary hypertension (PH) through a complex and bidirectional relationship. The prevalence of OSA and other forms of sleep-disordered breathing (SDB) among children with PH is not well understood. A better understanding could help elucidate contributing factors for PH, guide diagnostic testing, and inform therapies. We therefore determined the proportion of SDB codiagnosis and OSA risk factors among a cohort of children with PH. METHODS: Retrospective chart review was performed for children 0-21 years old with PH who underwent polysomnography (PSG) between 1 January 2010 and 31 August 2020. The primary outcome was OSA occurrence and risk based on demographics, diagnoses, and PH classification. The secondary outcome was occurrence and risk of other SDB diagnoses based on PH classification. RESULTS: Among 89 children identified, diagnoses included OSA (N = 79, 89%), central sleep apnea (N = 11, 12%), sleep-related hypoventilation (N = 6, 7%), and non-apneic hypoxemia (N = 28, 31%). Trisomy 21 diagnosis (N = 38, 43%) was associated with increased OSA risk (RR: 1.24, 95% CI: [1.09-1.42]) and age >12 months at PSG was associated with decreased OSA risk (N = 52, 58%, RR: 0.60, 95% CI: [0.45-0.81]). There was no difference in SDB risk based on Group 1 (N = 56, 63%) or Group 3 (N = 33, 37%) PH and no difference in OSA severity based on demographics and diagnoses. CONCLUSION: OSA was diagnosed among a majority of children with PH, and other SDB diagnoses were identified in a significant proportion of children. These findings support routine screening for SDB with PSG in children with PH.


Subject(s)
Hypertension, Pulmonary , Sleep Apnea Syndromes , Sleep Apnea, Central , Sleep Apnea, Obstructive , Child , Humans , Infant , Infant, Newborn , Child, Preschool , Adolescent , Young Adult , Adult , Retrospective Studies , Hypertension, Pulmonary/epidemiology , Hypertension, Pulmonary/complications , Sleep Apnea Syndromes/diagnosis , Sleep Apnea, Obstructive/complications , Sleep Apnea, Obstructive/epidemiology , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Central/diagnosis
6.
Sleep Med ; 107: 179-186, 2023 07.
Article in English | MEDLINE | ID: mdl-37196396

ABSTRACT

BACKGROUND/OBJECTIVE: While positive airway pressure (PAP) is an efficacious intervention for the treatment of obstructive sleep apnea syndrome (OSAS) in children with Down syndrome (DS), implementation and consistent use can be difficult. Caregiver perspectives and experiences using PAP are described with the aim of informing clinical practice. METHODS: Qualitative semi-structured phone interviews were conducted with 40 caregivers (i.e., mothers) of children with DS and OSAS treated with PAP for at least 6 months. Content analysis was used to identify themes associated with adherence and non-adherence. RESULTS: Respondents indicated variability in caregiver experience with the adoption of PAP and observed benefits of PAP. Varied experiences were attributed to several themes including accessing supplies, interactions with the medical team and equipment company, and patients' unique needs and behaviors, including the child's willingness and ability to adapt to PAP, sensory sensitivities, keeping the mask on all night, and differences in daytime behavior. Many families reported that desensitization with a reward system and trust within the caregiver-patient relationship were helpful. Caregiver suggestions for improving PAP adherence for families of children with DS included improving communication with the medical team and medical equipment company, emphasizing patience, using visual supports, and social support and education for extended family. CONCLUSIONS: Although family experiences varied, several actionable strategies by both the medical team and families emerged for improving the experience of and adherence to PAP in children with DS.


Subject(s)
Down Syndrome , Sleep Apnea, Obstructive , Humans , Child , Caregivers , Down Syndrome/complications , Continuous Positive Airway Pressure , Patient Compliance
7.
Pediatr Pulmonol ; 57(2): 560-566, 2022 02.
Article in English | MEDLINE | ID: mdl-34816625

ABSTRACT

OBJECTIVE: With advances in technology, pediatric patients are sent home with chronic invasive mechanical ventilation (CIMV). Ventilation optimization and preparation for weaning are essential processes, however, there is no standard approach to weaning and titrating ventilator settings in children. There is little information in the literature on the utility of polysomnography (PSG) in weaning and titration of CIMV. Our objective is to review the role of PSG on titration and weaning of CIMV in children. METHODS: A retrospective review of medical records and polysomnograms was performed on CIMV dependent patients at Cincinnati Children's Hospital Medical Center from January 2011 to October 2017. Patients underwent overnight ventilator weaning or titration PSG. Subjects with less than 3 h of total sleep time were excluded. RESULTS: About 163 PSGs were divided into 97 PSGs for ventilator titration and 66 PSGs for ventilator weaning. Of the 97 ventilator titration PSGs, 59 (60.8%) had inadequate ventilation, 10 (10.3%) had inadequate oxygenation, 22 (22.7%) had significant tracheostomy leak, 13 (13.4%) had autocycling, 3 (3.1%) had failure to trigger, 4 (4.1%) had central sleep apnea, and 24 (25.8%) had adequate ventilation on current ventilator settings. Of the 66 ventilator weaning PSGs, 48 (72.7%) weaned to lower ventilator settings or off ventilator support and 18 (27.3%) did not wean. DISCUSSION: Our results indicate that PSG is useful in titration and weaning of ventilator settings in children with CIMV. Larger studies are needed to evaluate the subgroup of this population who would benefit most from PSG for assessment of adequate ventilator support.


Subject(s)
Respiration, Artificial , Ventilator Weaning , Child , Humans , Polysomnography , Tracheostomy , Ventilator Weaning/methods , Ventilators, Mechanical
8.
Pediatr Pulmonol ; 56(6): 1635-1643, 2021 06.
Article in English | MEDLINE | ID: mdl-33647193

ABSTRACT

BACKGROUND: The care of mechanically ventilated pediatric patients is complex and burdensome. It is essential to adequately support the family caregivers of these children to optimize outcomes; however, there is no literature describing interventions for caregivers in this population. RESEARCH QUESTION: This study described a novel behavioral health program and examined its impact on family caregiver engagement and psychological distress on a pediatric inpatient chronic ventilator unit. STUDY DESIGN AND METHODS: Electronic chart review was completed with retrospective and prospective participant enrollment for the purposes of longitudinal evaluation of caregiver engagement. For analytic purposes, participants were grouped into three categories: (1) preprogram, (2) postprogram, and (3) postprogram with completion of psychotherapy. RESULTS: The behavioral health program was associated with increased caregiver participation in rounds, t = 7.76, p = < .001. Parents who completed a course of psychotherapy within the behavioral health program demonstrated reduced time to training completion (F = 5.89; p < .01), higher staff-rated caregiver engagement (F = 3.69; p < .05), and significantly reduced levels of caregiver distress (t = 2.09; p < .05).


Subject(s)
Caregivers , Home Care Services , Child , Humans , Prospective Studies , Retrospective Studies , Ventilators, Mechanical
9.
Pediatr Pulmonol ; 56(4): 700-709, 2021 04.
Article in English | MEDLINE | ID: mdl-32720756

ABSTRACT

The term neuromuscular disease (NMD) encompasses a large variety of disorders that result in abnormal muscle function. Although it may be conventional to relate the use of this term to the most common muscular diseases (Duchenne muscular dystrophy [DMD], spinal muscular atrophy [SMA], and amyotrophic lateral sclerosis, etc), it is important to extend the term to pathologies manifested by severe neurologic (brain and spinal cord) malformations and injuries. In many of these scenarios, there are common mechanisms that contribute to sleep disordered breathing (SDB) and respiratory insufficiency although comorbidities may be somewhat different. Advances in the understanding of these diseases and their natural history, and increasing availability of mechanical ventilation to these patients have improved survival. The development of novel genetic and molecular therapies (as in the cases of DMD, SMA, and X-linked myotubular myopathy) provides an opportunity to use SDB as a reasonable outcome measure while also allowing the use of polysomnography as a validation tool in the assessments of effectiveness of therapies. We seek to provide an understanding of SDB in NMDs, and in the same light, would like to begin the conversation of thinking about weaning respiratory support when possible.


Subject(s)
Neuromuscular Diseases , Sleep Apnea Syndromes , Humans , Muscular Atrophy, Spinal/diagnosis , Muscular Atrophy, Spinal/therapy , Muscular Dystrophy, Duchenne , Neuromuscular Diseases/complications , Neuromuscular Diseases/therapy , Polysomnography , Sleep Apnea Syndromes/diagnosis , Sleep Apnea Syndromes/etiology , Sleep Apnea Syndromes/therapy
10.
ACG Case Rep J ; 7(4): e00365, 2020 Apr.
Article in English | MEDLINE | ID: mdl-32548193

ABSTRACT

We report a unique case of ulcerative colitis-associated bronchiectasis in a pediatric patient 6 years after colectomy. The patient presented with a chronic cough and had a computed tomography demonstrating bronchiectasis. She was treated with sputum expectoration (airway clearance) via chest physiotherapy and pulse-dose steroids with a prolonged oral taper. Her initial response was excellent; however, she experienced a recurrence of symptoms with de-escalation of airway clearance. Pulmonary extraintestinal manifestations of inflammatory bowel disease are most often diagnosed later in life. Both the severity of this patient's presentation and her age are unique to this case.

11.
Sleep Med ; 68: 160-166, 2020 04.
Article in English | MEDLINE | ID: mdl-32044552

ABSTRACT

STUDY OBJECTIVES: To describe sleep manifestations, polysomnographic (PSG) findings, and specific sleep disorders in children with Eosinophilic Esophagitis (EoE). METHODS: This retrospective study included children with EoE who were referred to sleep clinics. Clinical manifestations, PSG variables, and diagnosis of sleep disorders were analyzed. Sleep architecture of patients with EoE was compared to control subjects. RESULTS: In sum, 81 children with EoE met the criteria for entry into the analysis with a mean age of 10.1 ± 4.4 years. Of those, 46 children (57%) presented in the sleep clinic with active EoE symptoms, while 35 (43%) children did not have active EoE symptoms at presentation. Several sleep complaints were common in children with EoE, including snoring (62, 76.5%), restless sleep (54, 66.6%), legs jerking or leg discomfort (35, 43.2%) and daytime sleepiness (47, 58.0%). Comparing sleep architecture with controls, children with EoE had significantly higher NREM2 (P= < 0.001), lower NREM3 (P= < 0.001), lower rapid eye movement (REM) (P = 0.017), increased periodic leg movements (PLM) index (P= < 0.001) and increased arousal index (P = 0.007). There were no significant differences in the sleep efficiency between the EoE and control subjects. Common sleep diagnoses included obstructive sleep apnea (OSA, 30, 37.0%) and periodic limb movements disorder (PLMD, 20, 24.6%). Of note, we found a much higher percentage of PLMD in active EoE compared to inactive EoE (P = 0.004). CONCLUSIONS: Children with EoE have frequent sleep complaints and several sleep disorders identified from the sleep study, including sleep-disordered breathing and PLMD. Analysis of sleep architecture demonstrates significant sleep fragmentation as evidenced by decreased slow-wave sleep and REM sleep and increased arousal index.


Subject(s)
Eosinophilic Esophagitis , Nocturnal Myoclonus Syndrome , Adolescent , Child , Child, Preschool , Eosinophilic Esophagitis/complications , Humans , Polysomnography , Retrospective Studies , Sleep
13.
J Clin Sleep Med ; 15(5): 743-748, 2019 05 15.
Article in English | MEDLINE | ID: mdl-31053208

ABSTRACT

STUDY OBJECTIVES: Previous studies have shown that non-rapid eye movement (NREM) sleep parasomnias commonly coexist with restless legs syndrome (RLS) and periodic limb movement disorder (PLMD) in children, leading to speculation that RLS/PLMD may precipitate or worsen parasomnias. However, there are limited data about the effect of the treatment of RLS/PLMD on parasomnias in children. Hence, we performed this study to determine whether the treatment of RLS/PLMD with oral iron therapy is associated with improvement of parasomnias in children. METHODS: A retrospective database was created for children with RLS/PLMD who were treated with iron therapy. These participants were followed for at least 1 year at Cincinnati Children's Hospital Medical Center. All participants had ferritin level testing and were treated with iron therapy. In addition, all participants underwent polysomnography before starting iron therapy for RLS/PLMD except for one participant who was already on iron but required a higher dose. Most participants underwent polysomnography after iron therapy. RESULTS: A total of 226 participants were identified with the diagnosis of RLS/PLMD. Of these, 50 had parasomnias and 30 of them were treated with iron therapy. Of the 30 participants, RLS symptoms improved in 15 participants (50%) and resolution of parasomnias was noted in 12 participants (40%) participants after iron therapy. Repeat polysomnography after iron therapy was performed in 21 participants (70%). After iron therapy, there was a significant decrease in periodic limb movement index (17.2 ± 8.8 [before] versus 6.7 ± 7.3 [after] events/h, P < .001). In addition, there were significant decreases in PLMS (24.52 ± 9.42 [before] versus 7.50 ± 7.18 [after] events/h, P < .0001), PLMS-related arousals (4.71 ± 1.81 [before] versus 1.35 ± 1.43 [after] events/h, P < .0001), and total arousals (11.65 ± 5.49 [before] versus 8.94 ± 3.65 [after] events/h, P < .01) after iron therapy. CONCLUSIONS: Parasomnias are common in our cohort of children with RLS/PLMD. Iron therapy was associated with a significant improvement in periodic limb movement index, RLS symptoms, and resolution of a significant proportion of NREM sleep parasomnias, suggesting that RLS/PLMD may precipitate NREM sleep parasomnia.


Subject(s)
Iron/therapeutic use , Nocturnal Myoclonus Syndrome/complications , Nocturnal Myoclonus Syndrome/drug therapy , Restless Legs Syndrome/complications , Restless Legs Syndrome/drug therapy , Child , Cohort Studies , Female , Humans , Male , Nocturnal Myoclonus Syndrome/physiopathology , Parasomnias/complications , Parasomnias/drug therapy , Parasomnias/physiopathology , Polysomnography/methods , Restless Legs Syndrome/physiopathology , Retrospective Studies , Treatment Outcome
14.
J Pediatr ; 210: 194-200.e2, 2019 07.
Article in English | MEDLINE | ID: mdl-30955791

ABSTRACT

OBJECTIVE: To describe and compare the lung function decline in patients with Duchenne muscular dystrophy on glucocorticoid therapy in contrast with glucocorticoid-naïve patients, and to define the deciles of pulmonary decline in glucocorticoid-treated patients. STUDY DESIGN: This retrospective study examined lung function of patients with Duchenne muscular dystrophy over 6 years of age followed between 2001 and 2015 at 2 centers-glucocorticoid-treated patients in Cincinnati, Ohio, and glucocorticoid-naïve patients in Paris, France. Forced vital capacity (FVC, FVC%), forced expiratory volume in 1 second, maximal inspiratory pressure, maximal expiratory pressure, and peak expiratory flow data were analyzed. Only FVC data were available for the French cohort. RESULTS: There were 170 glucocorticoid-treated patients (92%), 5 patients (2.7%) with past glucocorticoid use, and 50 French glucocorticoid-naïve patients. The peak absolute FVC was higher and was achieved at earlier ages in glucocorticoid-treated compared with glucocorticoid-naïve patients (peak FVC, 2.4 ± 0.6 L vs 1.9 ± 0.7 L; P < .0001; ages 13.5 ± 3.0 years vs 14.3 ± 2.8 years; P = .03). The peak FVC% was also higher and was achieved at earlier ages in glucocorticoid-treated patients (peak FVC%, 105.1 ± 25.1% vs 56 ± 20.9%; P < .0001; ages 11.9 ± 2.9 years vs 13.6 ± 3.2 years; P = .002). Rates of decline for both groups varied with age. Maximal rates of decline were 5.0 ± 0.26% per year (12-20 years) for glucocorticoid-treated and 5.1 ± 0.39% per year for glucocorticoid-naïve patients (11-20 years; P = .2). Deciles of FVC% decline in glucocorticoid-treated patients show that patients experience accelerated decline at variable ages. CONCLUSIONS: These data describe nonlinear rates of decline of pulmonary function in patients with Duchenne muscular dystrophy, with improved function in glucocorticoid-treated patients. FVC% deciles may be a useful tool for clinical and research use.


Subject(s)
Muscular Dystrophy, Duchenne/drug therapy , Respiratory Function Tests , Adolescent , Child , Disease Progression , Glucocorticoids/therapeutic use , Humans , Male , Muscular Dystrophy, Duchenne/physiopathology , Prednisone/therapeutic use , Pregnenediones/therapeutic use , Retrospective Studies
15.
Paediatr Respir Rev ; 25: 14-18, 2018 Jan.
Article in English | MEDLINE | ID: mdl-28108192

ABSTRACT

It is now recognized that there are two types of narcolepsy. Narcolepsy type I or Narcolepsy with cataplexy is caused by the loss of hypocretin or orexin neurons. Narcolepsy type II or narcolepsy without cataplexy has normal hypocretin and the etiology is unknown. Hypocretin is a neuropeptide produced by neurons in the lateral hypothalamus. Both genetic and environmental factors play a crucial role in the pathogenesis of narcolepsy. Most patients with narcolepsy type I and half of patients with narcolepsy type II carry HLA-DQB1*0602. HLA-DQB1*0602 forms a heterodimer with HLA-DQA1*0102 and may act as an antigen presenter to the T cell receptors, resulting in narcolepsy susceptibility. In addition, narcolepsy has been shown to be linked to polymorphisms in other non-HLA genes that may affect immune regulatory function, leading to speculation that autoimmune processes may play a crucial role in the loss of hypocretin neurons. Infections have been proposed as a potential trigger for the autoimmune-mediated mechanism. Several recent studies have shown increased cases of narcolepsy, especially in children and adolescents in relation with H1N1 influenza. The increased cases in Europe seems to be related to a specific type of H1N1 influenza vaccination (Pandemrix), while the increased cases in China are related to influenza infection. The data from the Pediatric Working Group of the Sleep Research Network have shown similar increases of early onset narcolepsy in the United States.


Subject(s)
Autoimmunity , HLA-DQ beta-Chains/genetics , Influenza, Human/complications , Narcolepsy , Streptococcal Infections/complications , Gene-Environment Interaction , Humans , Influenza A Virus, H1N1 Subtype , Influenza Vaccines/pharmacology , Narcolepsy/diagnosis , Narcolepsy/etiology , Narcolepsy/genetics , Narcolepsy/immunology
16.
J Clin Sleep Med ; 13(6): 777-783, 2017 Jun 15.
Article in English | MEDLINE | ID: mdl-28212689

ABSTRACT

STUDY OBJECTIVES: Idiopathic central sleep apnea (ICSA) is categorized as a type of nonhypercapnic central sleep apnea (CSA). Recurrent cessation and resumption of respiration leads to sleep fragmentation, which causes excessive daytime sleepiness, frequent nocturnal awakenings, or both. ICSA has been described in the adult population but there is limited information in children. The purpose of this study was to describe clinical manifestations and polysomnographic findings in children with ICSA. METHODS: A retrospective review of medical records and polysomnograms was performed for 14 pediatric patients with ICSA, 9 from Cincinnati Children's Hospital Medical Center and 5 from Antwerp University Hospital. Polysomnographic features of patients with ICSA were compared with those of nine age-matched control group subjects. Patients with CSA caused by medical or neurological disorders, medication use, or substance use were excluded. RESULTS: Sleep complaints were common in the 14 children with ICSA, including those with sleep-onset insomnia (7 children), frequent nighttime awakening (3 children), restless sleep (7 children), and daytime sleepiness (5 children). Symptoms of sleep-disordered breathing were noted in 11 of 14 subjects. Compared to that of the control group, sleep latency in the ICSA group was significantly prolonged (P < .05). The percentage of stage 2 sleep was significantly higher (P < .05), and slow wave sleep was significantly lower in patients with ICSA (P < .05). CONCLUSIONS: Similar to adult patients, children with ICSA present with complaints of insomnia, daytime sleepiness, and symptoms of obstructive sleep apnea. Analysis of polysomnograms reveals prolonged sleep latency, increased stage 2 sleep, and decreased slow wave sleep. Further studies are needed to assess mechanisms and the role of hypercapnic response in the pathogenesis of children with ICSA.


Subject(s)
Sleep Apnea, Central/complications , Sleep Apnea, Central/physiopathology , Adolescent , Child , Child, Preschool , Female , Humans , Male , Polysomnography/methods , Retrospective Studies , Sleep Stages/physiology , Sleep Wake Disorders/complications , Sleep Wake Disorders/physiopathology
17.
Otolaryngol Head Neck Surg ; 153(4): 649-55, 2015 Oct.
Article in English | MEDLINE | ID: mdl-26124266

ABSTRACT

OBJECTIVE: Decannulation readiness is approached in several ways and is tailored to the individual patient. While microlaryngoscopy and bronchoscopy evaluate airway patency at all levels, polysomnography assesses sleep-related upper airway physiology. There are limited data in the literature on the utility of these tools. Thus, the main objectives of this study were (1) to identify sleep and respiratory parameters associated with successful decannulation and (2) to determine the agreement between microlaryngoscopy and bronchoscopy and polysomnography. STUDY DESIGN: Case series with chart review. SETTING: Quaternary care pediatric hospital. SUBJECTS AND METHODS: A retrospective review of medical records, microlaryngoscopy and bronchoscopy, and polysomnographs was performed on subjects preparing for decannulation from 2005 to 2010. Fifty-nine subjects who underwent overnight polysomnography with a tracheostomy capping trial and microlaryngoscopy and bronchoscopy were included. Prediction of successful decannulation from polysomnography parameters was determined using logistic regression analysis. RESULTS: Fifty-nine subjects with a total of 78 polysomnographs were subdivided into 2 groups: 42 polysomnographs were classified as successfully decannulated, and 36 polysomnographs belonged to the group that did not decannulate. Logistic regression analysis determined that the Apnea Hypopnea Index (P = .0423) and maximal end-tidal CO2 (P = .046) were significant predictors for successful decannulation. CONCLUSION: Airway evaluation by microlaryngoscopy and bronchoscopy is an essential tool in the assessment of decannulation readiness. Polysomnography is an important additional tool for children with complex airway problems. Our results indicate that certain polysomnographic parameters such as Apnea Hypopnea Index and maximal end-tidal CO2 are valuable in predicting successful tracheostomy decannulation.


Subject(s)
Bronchoscopy , Catheters , Device Removal/methods , Laryngoscopy , Polysomnography , Tracheostomy/instrumentation , Child , Child, Preschool , Humans , Infant , Predictive Value of Tests , Retrospective Studies , Treatment Outcome
18.
Clin EEG Neurosci ; 37(1): 30-3, 2006 Jan.
Article in English | MEDLINE | ID: mdl-16475482

ABSTRACT

The objective of this study was to determine if screening by a neurologist of all non-neurologist electroencephalogram (EEG) referrals prior to approval reduces the number of inappropriate requests. This retrospective survey included 600 consecutive EEG requisitions referred to the Anaheim Kaiser Permanente Neurodiagnostic Laboratory to rule out epilepsy. Patients with established epilepsy referred for a repeat EEG for management issues were excluded. Three groups of EEG referrals were analyzed. Each group consisted of 200 EEGs (100 pediatric and 100 adult EEGs). The first group was referred directly by non-neurologists, the second group was referred by non-neurologists with scrutiny by a neurologist, and the third group was referred by a neurologist directly. In the pediatric group, the ratio of abnormal EEG vs normal EEG was 1:3.35 in the first group, 1:0.69 in the second group and 1:0.33 in the third group. In the adult group, the ratio of abnormal EEGs vs normal EEGs was 1:2.23 in the first group, 1:0.82 in the second group and 1:0.45 in the third group. In the combined pediatric and adult groups, the ratio of abnormal EEG vs normal EEG was 1:2.70 in the first group, 1:0.75 in the second group and 1:0.39 in the third group. There was a significant difference between the results of the EEGs ordered by non-neurologists directly versus non-neurologists with scrutiny (p=.334, chi-square test). Scrutiny by a neurologist of EEG referrals from non-neurologists led to a reduction in the number of normal EEG results. This suggests that inappropriate EEG requests for non-epileptic patients that yield normal EEG results are significantly reduced with scrutiny. This can help reduce the indiscriminate overuse of EEGs by non-neurologists thereby leading to better utilization of healthcare resources.


Subject(s)
Electroencephalography/statistics & numerical data , Epilepsy/diagnosis , Epilepsy/epidemiology , Health Services/statistics & numerical data , Mass Screening/statistics & numerical data , Neurology/statistics & numerical data , Referral and Consultation/statistics & numerical data , California/epidemiology , Health Care Surveys , Humans , Prevalence , Quality Assurance, Health Care/methods , Reproducibility of Results , Sensitivity and Specificity
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