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1.
Arch Cardiovasc Dis ; 117(6-7): 409-416, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38811278

RESUMO

BACKGROUND: Sacubitril/valsartan has been demonstrated to significantly improve left ventricular performance and remodelling in patients with heart failure. However, its effects on the right ventricle in patients with chronic heart failure and sleep-disordered breathing (SDB) have not been studied. AIM: To investigate the impact of sacubitril/valsartan treatment on right ventricular function in patients with SDB. METHODS: This was a subanalysis of an observational prospective multicentre study involving 101 patients. At inclusion, patients were evaluated by echocardiography and nocturnal ventilatory polygraphy, which allowed patients to be divided into three groups: "central-SDB"; "obstructive-SDB"; and "no-SDB". RESULTS: After 3 months of sacubitril/valsartan therapy, a positive impact on right ventricular function was observed. In the general population, tricuspid annular plane systolic excursion increased by +1.32±4.74mm (P=0.024) and systolic pulmonary artery pressure decreased by -3.1±10.91mmHg (P=0.048). The central-SDB group experienced the greatest echocardiographic improvement, with a significant increase in tricuspid annular plane systolic excursion of +2.1±4.9mm (P=0.045) and a significant reduction in systolic pulmonary artery pressure of -8.4±9.7mmHg (P=0.001). CONCLUSIONS: Sacubitril/valsartan improved right ventricular function in patients with heart failure and SDB after only 3 months of treatment. The greatest improvement in right ventricular function was observed in the central-SDB group.


Assuntos
Aminobutiratos , Compostos de Bifenilo , Combinação de Medicamentos , Insuficiência Cardíaca , Recuperação de Função Fisiológica , Valsartana , Função Ventricular Direita , Humanos , Valsartana/uso terapêutico , Masculino , Feminino , Aminobutiratos/uso terapêutico , Aminobutiratos/efeitos adversos , Função Ventricular Direita/efeitos dos fármacos , Estudos Prospectivos , Pessoa de Meia-Idade , Resultado do Tratamento , Idoso , Insuficiência Cardíaca/fisiopatologia , Insuficiência Cardíaca/tratamento farmacológico , Insuficiência Cardíaca/diagnóstico , Fatores de Tempo , Apneia do Sono Tipo Central/fisiopatologia , Apneia do Sono Tipo Central/diagnóstico , Apneia do Sono Tipo Central/tratamento farmacológico , Bloqueadores do Receptor Tipo 1 de Angiotensina II/uso terapêutico , Bloqueadores do Receptor Tipo 1 de Angiotensina II/efeitos adversos , Tetrazóis/uso terapêutico , Tetrazóis/efeitos adversos , Inibidores de Proteases/uso terapêutico , Inibidores de Proteases/efeitos adversos , Polissonografia , Neprilisina/antagonistas & inibidores , Doença Crônica
2.
J Clin Pathol ; 77(6): 378-382, 2024 May 17.
Artigo em Inglês | MEDLINE | ID: mdl-38458747

RESUMO

Paired-like homeobox 2B (PHOX2B) is a gene essential in the development of the autonomic nervous system. PHOX2B mutations are associated with neurocristopathies-Hirschsprung disease (HSCR) and congenital central hypoventilation syndrome (CCHS)-and peripheral neuroblastic tumours. PHOXB2 plays an important role in the diagnostics of these conditions.Genotyping of a PHOX2B pathogenic variant is required to establish a diagnosis of CCHS. In HSCR patients, PHOX2B immunohistochemical staining has proven to be a valuable tool in identifying this disease. Furthermore, PHOXB2 is a predisposition gene for neuroblastoma, in which PHOX2B immunohistochemical staining can be used as a highly sensitive and specific diagnostic marker. The utility of PHOX2B immunohistochemistry in pheochromocytoma and paraganglioma has also been studied but yields conflicting results.In this review, an overview is given of PHOX2B, its associated diseases and the usefulness of PHOX2B immunohistochemistry as a diagnostic tool.


Assuntos
Proteínas de Homeodomínio , Hipoventilação , Imuno-Histoquímica , Neuroblastoma , Fatores de Transcrição , Humanos , Proteínas de Homeodomínio/genética , Fatores de Transcrição/genética , Hipoventilação/congênito , Hipoventilação/diagnóstico , Hipoventilação/genética , Neuroblastoma/diagnóstico , Neuroblastoma/genética , Neuroblastoma/patologia , Apneia do Sono Tipo Central/diagnóstico , Apneia do Sono Tipo Central/genética , Biomarcadores Tumorais/genética , Biomarcadores Tumorais/análise , Doença de Hirschsprung/diagnóstico , Doença de Hirschsprung/genética , Doença de Hirschsprung/patologia , Mutação , Neoplasias das Glândulas Suprarrenais/genética , Neoplasias das Glândulas Suprarrenais/diagnóstico , Neoplasias das Glândulas Suprarrenais/patologia , Predisposição Genética para Doença
3.
Sleep Med ; 116: 32-40, 2024 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-38417306

RESUMO

OBJECTIVES: Chiari type 1 malformation (CM1) may occasionally lead to central sleep apnea (CSA). We studied, in a large clinical cohort of pediatric CM1 patients, the effect of CM1 on breathing during sleep. METHODS: This is a retrospective single pediatric pulmonology center study with a systematic evaluation of pediatric CM1 patients under age 18 with polysomnography (PSG) during 2008-2020. Children with syndromes were excluded. All patients had undergone head and spine magnetic resonance imaging. RESULTS: We included 104 children with CM1 with a median age of 7 (interquartile range (IQR) 5-13) years. The median extent of tonsillar descent (TD) was 13 (IQR 10-18) mm. Syringomyelia was present in 19 children (18%). Of all children, 53 (51%) had normal PSG, 35 (34%) showed periodic breathing or central apnea and hypopnea index ≥5 h-1, and 16 (15%) displayed features of compensated central hypoventilation and end-tidal or transcutaneous carbon dioxide 99th percentile level above 50 mmHg. TD had the best predictive value for central breathing disorders. In a linear model, both age (61%) and TD (39%) predicted median breathing frequency (R = 0.33, p < 0.001). CONCLUSIONS: Although severe CSA is a rare complication of brainstem compression in pediatric patients with CM1, short arousal-triggered episodes of periodic breathing and mild compensated central hypoventilation are common. TD shows the best but still poor prediction of the presence of a central breathing disorder. This highlights the use of PSG in patient evaluation. Posterior fossa decompression surgery effectively treats central breathing disorders.


Assuntos
Malformação de Arnold-Chiari , Transtornos Respiratórios , Apneia do Sono Tipo Central , Criança , Humanos , Pré-Escolar , Adolescente , Apneia do Sono Tipo Central/complicações , Hipoventilação/complicações , Estudos Retrospectivos , Malformação de Arnold-Chiari/complicações , Sono , Transtornos Respiratórios/complicações
6.
Eur Arch Otorhinolaryngol ; 281(6): 3125-3130, 2024 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-38227284

RESUMO

PURPOSE: Investigate the effect of adenotonsillectomy on mixed apnea index (MAI) and central apnea index (CAI) in children with moderate-to-severe obstructive sleep apnea syndrome (OSAS). METHODS: Observational retrospective analysis of polysomnographic data in children diagnosed with moderate-to-severe OSAS and without comorbidity, submitted to adenotonsillectomy. RESULTS: Data were available for 80 children, 55 boys and 25 girls, with a median age of 3.6 years (2.1-5.9). Before surgery AHI was 14.1 (11.0-18.4) per hour, with a median preoperative OAI of 7.1 (4.1-10.6), MAI of 1.2 (0.6-1.6) and CAI of 1.0 (0.4-2.0). Adenotonsillectomy caused significant improvements in MAI, from 1.2 (0.6-1.6) to 0.5 (0.1-0.8) (p < 0.001) and CAI from 1.0 (0.4-2.0) to 0.5 (0.1-0.9) (p < 0.001). This represents a normalization of MAI in 91.7% and CAI in 75.6% of children that had an abnormal value prior surgery. CONCLUSION: Non obstructive apneas are common in children with OSAS. Adenotonsillectomy caused significant decrease not only in OAI, but also in MAI and CAI in children with moderate-to-severe OSAS.


Assuntos
Adenoidectomia , Polissonografia , Apneia Obstrutiva do Sono , Tonsilectomia , Humanos , Tonsilectomia/métodos , Masculino , Feminino , Adenoidectomia/métodos , Apneia Obstrutiva do Sono/cirurgia , Estudos Retrospectivos , Pré-Escolar , Criança , Apneia do Sono Tipo Central/cirurgia , Apneia do Sono Tipo Central/etiologia , Resultado do Tratamento , Índice de Gravidade de Doença
7.
Eur Respir Rev ; 33(171)2024 Jan 31.
Artigo em Inglês | MEDLINE | ID: mdl-38296343

RESUMO

The aim of this review is to summarise evidence that became available after publication of the 2017 European Respiratory Society statement on the diagnosis and management of obstructive sleep apnoea syndrome (OSAS) in 1- to 23-month-old children. The definition of OSAS in the first 2 years of life should probably differ from that applied in children older than 2 years. An obstructive apnoea-hypopnoea index >5 events·h-1 may be normal in neonates, as obstructive and central sleep apnoeas decline in frequency during infancy in otherwise healthy children and those with symptoms of upper airway obstruction. A combination of dynamic and fixed upper airway obstruction is commonly observed in this age group, and drug-induced sleep endoscopy may be useful in selecting the most appropriate surgical intervention. Adenotonsillectomy can improve nocturnal breathing in infants and young toddlers with OSAS, and isolated adenoidectomy can be efficacious particularly in children under 12 months of age. Laryngomalacia is a common cause of OSAS in young children and supraglottoplasty can provide improvement in children with moderate-to-severe upper airway obstruction. Children who are not candidates for surgery or have persistent OSAS post-operatively can be treated with positive airway pressure (PAP). High-flow nasal cannula may be offered to young children with persistent OSAS following surgery, as a bridge until definitive therapy or if they are PAP intolerant. In conclusion, management of OSAS in the first 2 years of life is unique and requires consideration of comorbidities and clinical presentation along with PSG results for treatment decisions, and a multidisciplinary approach to treatment with medical and otolaryngology teams.


Assuntos
Obstrução das Vias Respiratórias , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Tonsilectomia , Lactente , Recém-Nascido , Humanos , Pré-Escolar , Criança , Apneia Obstrutiva do Sono/diagnóstico , Apneia Obstrutiva do Sono/epidemiologia , Apneia Obstrutiva do Sono/terapia , Adenoidectomia/efeitos adversos , Adenoidectomia/métodos , Tonsilectomia/efeitos adversos , Tonsilectomia/métodos , Obstrução das Vias Respiratórias/diagnóstico , Obstrução das Vias Respiratórias/etiologia , Obstrução das Vias Respiratórias/terapia
8.
Int J Pediatr Otorhinolaryngol ; 177: 111863, 2024 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-38262224

RESUMO

OBJECTIVE: The purpose of this study was to assess the effects of adenotonsillectomy (A&T) on reducing central sleep apnea (CSA) in children and adolescents with obstructive sleep apnea (OSA). METHODS: A review of the PubMed database was conducted. Two researchers independently reviewed the articles from the literature search and selected papers for further review if they met inclusion criteria. Included studies were prospective studies and case series whose patients were children 18 years or younger undergoing adenotonsillectomy for obstructive sleep apnea with reported pre and postoperative central apnea indexes (CAI). RESULTS: Of the 107 articles initially identified, 18 underwent full length review, and ultimately 15 for final review. All studies reported marked improvement of central sleep apnea indexes after adenotonsillectomy. Two studies found resolution of CSA in 66.7 % and 73.7 % of patients respectively. The remaining four studies found significant reductions in CAI in 43.9 %-93 % of patients. The degree of reduction varied from 40.9 % to 80 %. DISCUSSION: Adenotonsillectomy improves and at times resolves CSA in pediatric patients with concomitant OSA.


Assuntos
Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Tonsilectomia , Adolescente , Criança , Humanos , Apneia do Sono Tipo Central/cirurgia , Apneia do Sono Tipo Central/complicações , Estudos Prospectivos , Adenoidectomia , Apneia Obstrutiva do Sono/cirurgia , Apneia Obstrutiva do Sono/complicações
9.
Pediatr Pulmonol ; 59(3): 642-651, 2024 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-38088209

RESUMO

RATIONALE: The use of long-term noninvasive respiratory support is increasing in children along with an extension of indications, in particular in children with central nervous system (CNS) disorders. OBJECTIVE: The aim of this study was to describe the characteristics of children with CNS disorders treated with long-term noninvasive respiratory support in France. METHODS: Data were collected from 27 French pediatric university centers through an anonymous questionnaire filled for every child treated with noninvasive ventilatory support ≥3 months on 1st June 2019. MAIN RESULTS: The data of 182 patients (55% boys, median age: 10.2 [5.4;14.8] years old [range: 0.3-25]) were collected: 35 (19%) patients had nontumoral spinal cord injury, 22 (12%) CNS tumors, 63 (35%) multiple disabilities, 26 (14%) central alveolar hypoventilation and 36 (20%) other CNS disorders. Seventy five percent of the patients were treated with noninvasive ventilation (NIV) and 25% with continuous positive airway pressure (CPAP). The main investigations performed before CPAP/NIV initiation were nocturnal gas exchange recordings, alone or coupled with poly(somno)graphy (in 29% and 34% of the patients, respectively). CPAP/NIV was started in an acute setting in 10% of the patients. Median adherence was 8 [6;10] hours/night, with 12% of patients using treatment <4 h/day. Nasal mask was the most common interface (70%). Airway clearance techniques were used by 31% of patients. CONCLUSION: CPAP/NIV may be a therapeutic option in children with CNS disorders. Future studies should assess treatment efficacy and patient reported outcome measures.


Assuntos
Doenças do Sistema Nervoso Central , Ventilação não Invasiva , Apneia do Sono Tipo Central , Masculino , Criança , Humanos , Adolescente , Feminino , Ventilação não Invasiva/métodos , Pressão Positiva Contínua nas Vias Aéreas/métodos , Resultado do Tratamento , Doenças do Sistema Nervoso Central/complicações , Doenças do Sistema Nervoso Central/terapia
10.
J Clin Sleep Med ; 20(4): 505-514, 2024 Apr 01.
Artigo em Inglês | MEDLINE | ID: mdl-37950451

RESUMO

STUDY OBJECTIVES: The aims of this study were to characterize obstructive sleep apnea (OSA) care pathways among commercially insured individuals in the United States and to investigate between-groups differences in population, care delivery, and economic aspects. METHODS: We identified adults with OSA using a large, national administrative claims database (January 1, 2016-February 28, 2020). Inclusion criteria included a diagnostic sleep test on or within ≤ 12 months of OSA diagnosis (index date) and 12 months of continuous enrollment before and after the index date. Exclusion criteria included prior OSA treatment or central sleep apnea. OSA care pathways were identified using sleep testing health care procedural health care common procedure coding system/current procedural terminology codes then selected for analysis if they were experienced by ≥ 3% of the population and assessed for baseline demographic/clinical characteristics that were also used for model adjustment. Primary outcome was positive airway pressure initiation rate; secondary outcomes were time from first sleep test to initiation of positive airway pressure, sleep test costs, and health care resource utilization. Associations between pathway type and time to treatment initiation were assessed using generalized linear models. RESULTS: Of 86,827 adults with OSA, 92.1% received care in 1 of 5 care pathways that met criteria: home sleep apnea testing (HSAT; 30.8%), polysomnography (PSG; 23.6%), PSG-Titration (19.8%), Split-night (14.8%), and HSAT-Titration (3.2%). Pathways had significantly different demographic and clinical characteristics. HSAT-Titration had the highest positive airway pressure initiation rate (84.6%) and PSG the lowest (34.4%). After adjustments, time to treatment initiation was significantly associated with pathway (P < .0001); Split-night had shortest duration (median, 28 days), followed by HSAT (36), PSG (37), PSG-Titration (58), and HSAT-Titration (75). HSAT had the lowest sleep test costs and health care resource utilization. CONCLUSIONS: Distinct OSA care pathways exist and are associated with differences in population, care delivery, and economic aspects. CITATION: Wickwire EM, Zhang X, Munson SH, et al. The OSA patient journey: pathways for diagnosis and treatment among commercially insured individuals in the United States. J Clin Sleep Med. 2024;20(4):505-514.


Assuntos
Síndromes da Apneia do Sono , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Adulto , Humanos , Estados Unidos , Apneia Obstrutiva do Sono/diagnóstico , Apneia Obstrutiva do Sono/terapia , Síndromes da Apneia do Sono/complicações , Sono , Polissonografia/métodos , Apneia do Sono Tipo Central/complicações
11.
J Clin Sleep Med ; 20(3): 478-481, 2024 Mar 01.
Artigo em Inglês | MEDLINE | ID: mdl-37861394

RESUMO

Congenital central hypoventilation syndrome (CCHS), a rare disease caused by paired-like homeobox 2B variants, affects control of breathing. We report on a 21-month-old boy with CCHS caused by a novel nonpolyalanine repeat mutation, neuroblastoma, severe obstructive and central sleep apnea, and sleep-related hypoxemia without hypoventilation. At 10 months, due to persistent central sleep apnea during serial polysomnography, bilevel positive airway pressure therapy was initiated despite the absence of hypoventilation. Nonpolyalanine repeat mutations are associated with severe phenotypes requiring continuous assisted ventilation, Hirschsprung's disease, and neural crest tumors; however, our patient had a relatively milder respiratory phenotype requiring sleep-only assisted ventilation without tracheostomy. Although alveolar hypoventilation is the hallmark of CCHS, our patient lacked hypoventilation. Bilevel positive airway pressure could be considered in some infants with CCHS requiring sleep-only assisted ventilation for tracheostomy avoidance. Our case demonstrates the expanding phenotypic spectrum in CCHS and the importance of formulating an individualized care plan. CITATION: Fain ME, Raghunandan S, Pencheva B, Leu RM, Kasi AS. Images: atypical presentation of congenital central hypoventilation syndrome in an infant with central and obstructive sleep apnea. J Clin Sleep Med. 2024;20(3):478-481.


Assuntos
Hipoventilação/congênito , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Masculino , Lactente , Humanos , Hipoventilação/complicações , Hipoventilação/genética , Hipoventilação/terapia , Apneia do Sono Tipo Central/complicações , Apneia do Sono Tipo Central/genética , Apneia do Sono Tipo Central/terapia , Apneia Obstrutiva do Sono/complicações , Apneia Obstrutiva do Sono/terapia , Sono
12.
Lung ; 201(6): 625-634, 2023 12.
Artigo em Inglês | MEDLINE | ID: mdl-37987861

RESUMO

PURPOSE: Guidelines recommend considering an initial trial of continuous positive airway pressure (CPAP) to treat central sleep apnea (CSA). However, practice patterns vary widely. This study investigated predictors for an initial trial of CPAP in patients with central apneas and whether those factors predict adequate treatment response in patients receiving an initial CPAP trial. METHODS: Charts of patients receiving a diagnostic code for CSA following a sleep study during 2016-2018 at a single center were reviewed. Patient factors, initial treatment prescriptions, and subsequent changes to therapy were extracted from electronic health records. Regression models were used to estimate factors associated with an initial CPAP prescription and the likelihood of an adequate CPAP response (no subsequent therapy change and no discontinuation of therapy) among patients prescribed CPAP. RESULTS: 429/588 (73%) patients with central apneas received an initial trial of CPAP. Younger age, diagnosis by home sleep testing, non-opiate etiology of central apneas, and a lower proportion of central apneas at diagnosis were independently associated with a higher likelihood of an initial CPAP trial. A lower proportion of central apneas was associated with a higher probability of adequate response, while current smoking and opiate-related central apneas predicted an unsuccessful CPAP trial. A new finding was that older age predicted a lower likelihood of an initial CPAP prescription but did not predict an unsatisfactory response to CPAP. CONCLUSION: Clinicians may incorrectly weigh certain clinical and sleep study characteristics when deciding whether to trial CPAP for patients with central apneas.


Assuntos
Apneia do Sono Tipo Central , Humanos , Pressão Positiva Contínua nas Vias Aéreas , Polissonografia , Apneia do Sono Tipo Central/diagnóstico , Apneia do Sono Tipo Central/terapia , Ensaios Clínicos como Assunto
13.
Int J Pediatr Otorhinolaryngol ; 175: 111750, 2023 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-37844425

RESUMO

STUDY OBJECTIVE: Assess the prevalence of and risk factors for pediatric pulmonary hypertension (PH) in the 2016 Kids' Inpatient Database (KID), including obstructive sleep apnea (OSA) and obesity. METHODS: Retrospective cross-sectional cohort study utilizing 6,081,132 weighted pediatric discharges from the 2016 KID. Study variables included age, length of stay, mortality, gender, hospital region, primary payer, race, median household income for patient's ZIP code, OSA, central sleep apnea (CSA), obesity, Down syndrome, sickle cell disease (SCD), thalassemia, congenital heart disease (CHD), hypertension, asthma and chronic lung disease of prematurity (CLDP). PH was the primary outcome of interest. Bivariate and multivariable logistic regression models were utilized with odds ratios and 95 % confidence intervals. RESULTS: The mean age was 3.76 years, the mean hospital length of stay was 3.85 days, 48.9 % were male, 52.6 % had government health insurance, 51.0 % were White, 16.1 % were Black, 21.1 % were Hispanic, 5.0 % were Asian or Pacific Islander, 0.80 % were Native American and 6.1 % identified as "other". The prevalence of PH was 0.21 % (12,777 patients). There were 37,631 patients with OSA and the prevalence of PH among this cohort was 3.3 %, over 10x greater than the overall prevalence of PH in the 2016 KID (0.21 %). Risk factors associated with PH included CLDP, CHD, Down syndrome, asthma, OSA, CSA, hypertension, SCD, obesity, race/ethnicity, government insurance, age, male gender (p < 0.0001), and hospital region (p = 0.0002). CONCLUSIONS: Several risk factors were independently associated with PH, such as OSA, CSA, obesity, asthma, and insurance status. Prospective multi-institutional studies are needed to assess the relationships between these risk factors, severity metrics, and causative links in the development of PH; in addition to identifying children with OSA who are most likely to benefit from cardiopulmonary screening prior to adenotonsillectomy. LEVEL OF EVIDENCE: Level III.


Assuntos
Asma , Síndrome de Down , Cardiopatias Congênitas , Hipertensão Pulmonar , Hipertensão , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Humanos , Criança , Masculino , Pré-Escolar , Feminino , Hipertensão Pulmonar/epidemiologia , Estudos Retrospectivos , Síndrome de Down/complicações , Estudos Prospectivos , Pacientes Internados , Estudos Transversais , Apneia Obstrutiva do Sono/diagnóstico , Hipertensão/complicações , Asma/complicações , Cardiopatias Congênitas/epidemiologia , Cardiopatias Congênitas/complicações , Fatores de Risco , Obesidade/complicações , Apneia do Sono Tipo Central/complicações
14.
Eur Arch Otorhinolaryngol ; 280(12): 5205-5217, 2023 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-37642712

RESUMO

PURPOSE: To evaluate the effects of adenotonsillectomy on improving central sleep apnea events in children with obstructive sleep apnea (OSA). METHODS: We searched four online databases for relevant articles published from inception until October 2022. We included studies that measured the number of central apnea events per sleep and central apnea-hypopnea index (CAHI) or central apnea index (CAI) scores in children with OSA before and after adenotonsillectomy. Our primary outcomes were changes in CAI scores, the number of central apnea events per sleep, and CAHI scores after surgery. Our secondary outcomes were changes in total and mixed apnea events, improvement of sleep outcomes, and differences in oxygen or carbon dioxide saturation during sleep. We performed meta-analyses by pooling the mean changes of all included studies with a 95% confidence interval using Stata 17. Subsequently, we performed subgroup analyses based on the presence of comorbidities. RESULTS: We included 22 studies comprising 1287 patients. Central and total sleep apnea parameters, except for CAHI and mixed apnea index scores, showed significant improvements after surgery. In addition, all respiratory parameters and second and third stages of non-rapid eye movement sleep showed significant postsurgical improvements. Patients with comorbidities showed significant improvements only in the total apnea-hypopnea index, oxygen desaturation index, and minimal oxygen saturation. CONCLUSION: Adenotonsillectomy improves central apnea events in patients with OSA but not in those with comorbidities.


Assuntos
Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Tonsilectomia , Criança , Humanos , Apneia do Sono Tipo Central/etiologia , Adenoidectomia , Apneia Obstrutiva do Sono/complicações , Apneia Obstrutiva do Sono/cirurgia , Oxigênio
15.
Artigo em Chinês | MEDLINE | ID: mdl-37551575

RESUMO

Two children with late-onset congenital central hypoventilation syndrome were reported, one of whom was male and had no abnormal manifestations after birth, respiratory failure occurs at the age of 1 year and 6 months. After being hospitalized, he was treated with oxygen inhalation and non-invasive ventilation, but carbon dioxide retention could not be corrected. After one month of tracheal intubation, he was failure to wean from ventilator, so tracheostomy was performed. He needs a ventilator to help breath while sleeping, and can breath autonomously during the day without ventilator. The other case was a female, with no abnormalities after birth. At the age of 11 months, she developed respiratory failure. During sleep, the child needs non-invasive assisted ventilation through a nasal mask, and during the day, she breathed autonomously.Two patients were followed up forever 2 years and their growth and development were normal.


Assuntos
Apneia do Sono Tipo Central , Humanos , Criança , Masculino , Feminino , Lactente , Apneia do Sono Tipo Central/terapia , Respiração Artificial , Hipoventilação/terapia , Hipoventilação/congênito , Oxigênio
16.
J Sleep Res ; 32(5): e13914, 2023 10.
Artigo em Inglês | MEDLINE | ID: mdl-37128177

RESUMO

Children with genetic skeletal disorders have variable conditions that can lead to sleep-disordered breathing, and polysomnography is the gold standard for diagnosing this condition. We aimed to review polysomnography findings, to assess the severity of sleep apnea, and to investigate the clinical variables predictive of sleep-disordered breathing in these patients. We retrospectively collected the medical records of patients with genetic skeletal disorders who underwent polysomnography for 5 years. Twenty-seven children with various genetic skeletal disorders, including achondroplasia (14), Crouzon syndrome (3), acromesomelic dysplasia Maroteaux type (3), Apert syndrome (2), osteopetrosis (1), Jeune dysplasia (1), Desbuquois dysplasia (1), acrodysostosis (1), and spondyloepiphyseal dysplasia (1) were enrolled. The median age at the first polysomnography was 58 (1st-3rd quartile: 31-113) months. The overall sleep-disordered breathing results were: 19 (70.3%) had obstructive sleep apneas (OSA) (4 mild, 6 moderate, 9 severe), 2 (7.4%) had central apneas, 4 (14.8%) had nocturnal hypoventilation. There was a significant correlation between non-ambulatory status with both total AHI and OSA (p < 0.001, rho: -0.66/p = 0.04, rho: 0.38, respectively). Nine patients received positive airway pressure titration, and the oAHI values of all returned to the normal range. These patients were started with positive airway pressure treatment. Our cohort showed that the majority of the patients with skeletal dysplasia had sleep apnea syndrome characterised mainly by OSA, highlighting the importance of polysomnography screening for sleep disorders. Positive airway pressure therapy represents an effective treatment for sleep-disordered breathing in those patients.


Assuntos
Síndromes da Apneia do Sono , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Humanos , Criança , Pré-Escolar , Estudos Retrospectivos , Polissonografia , Síndromes da Apneia do Sono/diagnóstico
17.
J Sleep Res ; 32(4): e13851, 2023 08.
Artigo em Inglês | MEDLINE | ID: mdl-36807952

RESUMO

Sleep-disordered breathing is an important health issue for children. The objective of this study was to develop a machine learning classifier model for the identification of sleep apnea events taken exclusively from nasal air pressure measurements acquired during overnight polysomnography for paediatric patients. A secondary objective of this study was to differentiate site of obstruction exclusively from hypopnea event data using the model. Computer vision classifiers were developed via transfer learning to either normal breathing while asleep, obstructive hypopnea, obstructive apnea or central apnea. A separate model was trained to identify site of obstruction as either adeno-tonsillar or tongue base. In addition, a survey of board-certified and board-eligible sleep physicians was completed to compare clinician versus model classification performance of sleep events, and indicated very good performance of our model relative to human raters. The nasal air pressure sample database available for modelling comprised 417 normal, 266 obstructive hypopnea, 122 obstructive apnea and 131 central apnea events derived from 28 paediatric patients. The four-way classifier achieved a mean prediction accuracy of 70.0% (95% confidence interval [67.1-72.9]). Clinician raters correctly identified sleep events from nasal air pressure tracings 53.8% of the time, whereas the local model was 77.5% accurate. The site of obstruction classifier achieved a mean prediction accuracy of 75.0% (95% confidence interval [68.7-81.3]). Machine learning applied to nasal air pressure tracings is feasible and may exceed the diagnostic performance of expert clinicians. Nasal air pressure tracings of obstructive hypopneas may "encode" information regarding the site of obstruction, which may only be discernable by machine learning.


Assuntos
Síndromes da Apneia do Sono , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Humanos , Criança , Pressão do Ar , Síndromes da Apneia do Sono/diagnóstico , Apneia Obstrutiva do Sono/diagnóstico , Aprendizado de Máquina
18.
Ann Am Thorac Soc ; 20(5): 721-727, 2023 05.
Artigo em Inglês | MEDLINE | ID: mdl-36638772

RESUMO

Rationale: Hunter-Cheyne-Stokes breathing with central sleep apnea (CSA) is prevalent in some patients with heart failure with reduced ejection fraction (HFrEF). Theoretical models of Hunter-Cheyne-Stokes breathing predict that a low metabolic rate (MR) predisposes one to CSA. Objectives: In this study, we examined the role of MR in the pathogenesis of CSA. Methods: A physiological study was conducted in a sleep laboratory at a U.S. Department of Veterans Affairs medical center. Patients were 28 consecutive male Veterans with stable HFrEF. After an adaptation night, polysomnography, left ventricular ejection fraction, pulmonary function tests, carbon dioxide production ([Formula: see text]), and arterial blood samples were obtained under strict standardized conditions. Physiological variables were then entered into regression models to examine the association with CSA. Results: Body mass index varied from 20 to 40 kg/m2, and [Formula: see text] ranged from 167 to 434 ml/min. In the final regression model, low [Formula: see text] and low body mass index were associated with CSA index. [Formula: see text] had the strongest association (95% confidence interval, -0.36 to -0.06; P = 0.007). Conclusions: In patients with HFrEF, a low MR and related low [Formula: see text], but not low oxygen consumption, were associated with CSA. Mechanistically, in the face of low MR and [Formula: see text], a given change in ventilation results in large swings in partial pressure of CO2, thus promoting CSA. To our knowledge, this is the first study in humans that shows this association.


Assuntos
Insuficiência Cardíaca , Apneia do Sono Tipo Central , Humanos , Masculino , Dióxido de Carbono , Volume Sistólico/fisiologia , Função Ventricular Esquerda/fisiologia , Respiração de Cheyne-Stokes/complicações
19.
J Clin Sleep Med ; 19(4): 843-849, 2023 04 01.
Artigo em Inglês | MEDLINE | ID: mdl-36708265

RESUMO

This is the first case report of sleep-disordered breathing in patients with pontine tegmental cap dysplasia, a very rare neurological disorder characterized by an anatomic malformation in the pons. Patients present with hypotonia, cognitive dysfunction, and cranial nerve palsies (eg, hearing loss, trigeminal anesthesia, and swallow dysfunction). Extensive studies have demonstrated the relevance of different pontine neuronal nuclei in breathing regulation, which are structurally abnormal in pontine tegmental cap dysplasia. We present detailed polysomnography data for 3 patients aged 41 years, 20 years, and 1.5 years revealing significant central sleep apnea. We discuss our experience with managing their sleep-disordered breathing in the setting of multiple cranial nerve palsies and corneal anesthesia, and its relative contraindication of noninvasive positive pressure ventilation treatment. CITATION: Ju Wang J-D, Doherty D, Ramirez J-M, Chen M. Central sleep apnea in patients with pontine tegmental cap dysplasia treated with supplemental oxygen: a case report. J Clin Sleep Med. 2023;19(4):843-849.


Assuntos
Anormalidades Múltiplas , Doenças dos Nervos Cranianos , Apneia do Sono Tipo Central , Humanos , Apneia do Sono Tipo Central/complicações , Apneia do Sono Tipo Central/terapia , Respiração com Pressão Positiva , Oxigênio
20.
Sleep Breath ; 27(1): 245-253, 2023 03.
Artigo em Inglês | MEDLINE | ID: mdl-35394577

RESUMO

PURPOSES: The prevalence of sleep-disordered breathing (SDB) is high in patients with heart failure (HF), while the prevalence of SDB in HF with different left ventricular ejection fractions (LVEF) has rarely been reported. We aimed to explore the prevalence and clinical characteristics of SDB in patients with HF having different LVEF. METHODS: Patients with stable HF were consecutively enrolled. All patients underwent portable overnight cardiorespiratory polygraphy and echocardiography. According to their LVEF, the patients were divided into the HFrEF (HF with reduced EF, EF < 40%), HFmrEF (HF with mid-range EF, 40 ≤ EF < 50), and HFpEF groups (HF with preserved EF, EF ≥ 50%). The prevalence and clinical data of SDB among the 3 groups were then compared. RESULTS: A total of 252 patients, including 134 men, were enrolled in the study. The prevalence of SDB in patients with HF was 70%. Obstructive sleep apnea (OSA) was diagnosed in 48% and central sleep apnea (CSA) in 22%. The prevalence of SDB in the HFrEE, HFmrEF, and HFpEF groups was 86%, 86%, and 62%, respectively (P = 0.001). The prevalence of OSA among the 3 groups was 42%, 47%, and 49%, respectively (P = 0.708), while the prevalence of CSA among the 3 groups was 44%, 40%, and 13% (P < 0.001). Logistic regression analysis revealed that age and BMI were independent risk factors for OSA in patients with HF, while LVEF and smoking were independent risk factors for CSA in patients with HF. Correlational analyses revealed that LVEF was negatively correlated with apnea-hypopnea index (AHI) (r = -0.309, P < 0.001) and central apnea index (CAI) ( r = -0.558, P < 0.001), while there was no significant correlation with obstructive apnea index (OAI). The ROC curve revealed that LVEF could predict the occurrence of CSA and SDB, with AUC = 0.683 (95%CI 0.600-0.767, P < 0.001) and AUC = 0.630 (95%CI 0.559-0.702, P = 0.001), but not of OSA. CONCLUSIONS: SDB was highly common in HF, and the prevalence of SDB was different in HF with different LVEF, mainly due to the difference in cardiac functions. The prevalence and severity of SDB in HFrEF and HFmrEF were significantly higher than those in HFpEF, which was mainly related to the increase in CSA. When HFmrEF was similar to HFrEF in cardiac functions, the prevalence, type, and severity of SDB were similar between the two groups. Changes in LVEF had a significant impact on CAI, but not on OAI. LVEF can predict the occurrence of CSA and SDB to a certain extent.


Assuntos
Insuficiência Cardíaca , Síndromes da Apneia do Sono , Apneia do Sono Tipo Central , Apneia Obstrutiva do Sono , Masculino , Humanos , Volume Sistólico , Função Ventricular Esquerda , Insuficiência Cardíaca/diagnóstico , Insuficiência Cardíaca/epidemiologia , Prevalência , Polissonografia , Síndromes da Apneia do Sono/diagnóstico , Síndromes da Apneia do Sono/epidemiologia , Apneia do Sono Tipo Central/diagnóstico , Apneia do Sono Tipo Central/epidemiologia , Apneia Obstrutiva do Sono/diagnóstico , Apneia Obstrutiva do Sono/epidemiologia
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