Obstructive Sleep Apnea and Otolaryngologic Manifestations in Children with Pseudohypoparathyroidism

Background/Aims: Pseudohypoparathyroidism (PHP) is a rare, genetic disorder. Patients with PHP may have increased prevalence of obstructive sleep apnea (OSA) but this has not been prospectively studied. Methods: We enrolled children aged 6–18 years with PHP and matched controls. Evaluation included physical examination, medical history, and polysomnography. Results: Fifteen children with PHP type 1A (PHP1A) and 15 controls completed the study. Both groups were obese (BMI 32.2 ± 8.7 vs. 31.7± 6.5). The majority of PHP1A patients required tympanostomy tubes (86.7%) and adenotonsillectomy (73.3%). The primary outcome, i.e., the obstructive disturbance index, was significantly higher in PHP1A children versus controls (1.8 ± 2.3 vs. 0.6 ± 0.5, p = 0.045). Children with PHP1A were more likely to have OSA compared with controls (60.0 vs. 13.3%, p = 0.008). Three siblings with PHP type 1B (PHP1B) were also studied (BMI 25.9 ± 9.0). None had a history of adenotonsillectomy, one had tympanostomy tubes. The obstructive disturbance index (2.0 ± 2.3) was similar to that of children with PHP1A. Two (66.7%) PHP1B participants had OSA. Conclusion: Children with PHP1A are at an increased risk for OSA compared with similarly obese peers. They also have higher rates of otitis media and adenotonsillar hypertrophy. Screening for OSA should be considered in all patients with PHP1A and possibly PHP1B though more research is needed.

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845 Progressive external ophthalmoplegia in sleep apnea presenting as floppy eyelid syndrome

SLEEP ◽

10.1093/sleep/zsab072.842 ◽

2021 ◽

Vol 44 (Supplement_2) ◽

pp. A329-A329

Author(s):

Pratibha Anne ◽

Rupa Koothirezhi ◽

Ugorji Okorie ◽

Minh Tam Ho ◽

Brittany Monceaux ◽

...

Keyword(s):

Obstructive Sleep Apnea ◽

Surgical Treatment ◽

Sleep Apnea ◽

Genetic Disorder ◽

Deltoid Muscle ◽

Progressive External Ophthalmoplegia ◽

Ataxic Gait ◽

Pressure Setting ◽

Obstructive Sleep ◽

History Of

Abstract Introduction Floppy eye lid syndrome (FES) is known to be associated with Obstructive sleep apnea (OSA) and chronic progressive external ophthalmoplegia (CPEO) is a rare genetic disorder with mitochondrial myopathy that may present with isolated eye lid ptosis in the initial stages. In a patient with loud snoring and obesity, treating obstructive sleep apnea may improve Floppy eyelid syndrome. Report of case(s) 52-year-old African – American male with past medical history of Hypertension, obesity, glaucoma, CPEO status bilateral blepharoplasty with failed surgical treatment. Patient was referred to Sleep medicine team to rule out Obstructive Sleep Apnea aa a cause of possible underlying FES and residual ptosis. On exam, patient was noted to have bilateral brow and eyelid ptosis and mild ataxic gait. MRI brain with and without contrast was unremarkable. Deltoid muscle biopsy was suggestive of possible congenital myopathy and mild denervation atrophy. Polysomnogram showed severe OSA with AHI of 74.1 per hour and patient was initiated on Auto CPAP at a pressure setting of 7–20 cm H2O. CPAP treatment improved snoring, OSA and subjective symptoms of excessive day time sleepiness but did not improve the residual ptosis. Conclusion Treatment of severe OSA in a patient previously diagnosed with CPEO and failed surgical treatment with bilateral blepharoplasty, did not alter the course of residual ptosis/ floppy eyelids even though his other sleep apnea symptoms have improved. Support (if any) 1. McNab AA. Floppy eyelid syndrome and obstructive sleep apnea. Ophthalmic Plast Reconstr Surg. 1997 Jun;13(2):98–114. doi: 10.1097/00002341-199706000-00005. PMID: 9185193.

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Compared to Individuals with Mild to Moderate Obstructive Sleep Apnea (OSA), Individuals with Severe OSA Had Higher BMI and Respiratory-Disturbance Scores

Life ◽

10.3390/life11050368 ◽

2021 ◽

Vol 11 (5) ◽

pp. 368

Author(s):

Leeba Rezaie ◽

Soroush Maazinezhad ◽

Donald J. Fogelberg ◽

Habibolah Khazaie ◽

Dena Sadeghi-Bahmani ◽

...

Keyword(s):

Obstructive Sleep Apnea ◽

Sleep Apnea ◽

Multiple Regression ◽

Daytime Sleepiness ◽

Respiratory Disturbance Index ◽

Disturbance Index ◽

Related Data ◽

Obstructive Sleep ◽

Increased Risk ◽

Respiratory Disturbance

Objective: Individuals with obstructive sleep apnea (OSA) are at increased risk to suffer from further somatic and sleep-related complaints. To assess OSA, demographic, anthropometric, and subjective/objective sleep parameters are taken into consideration, but often separately. Here, we entered demographic, anthropometric, subjective, and objective sleep- and breathing-related dimensions in one model. Methods: We reviewed the demographic, anthropometric, subjective and objective sleep- and breathing-related data, and polysomnographic records of 251 individuals with diagnosed OSA. OSA was considered as a continuous and as categorical variable (mild, moderate, and severe OSA). A series of correlational computations, X2-tests, F-tests, and a multiple regression model were performed to investigate which demographic, anthropometric, and subjective and objective sleep dimensions were associated with and predicted dimensions of OSA. Results: Higher apnea/hypopnea index (AHI) scores were associated with higher BMI, higher daytime sleepiness, a higher respiratory disturbance index, and higher snoring. Compared to individuals with mild to moderate OSA, individuals with severe OSA had a higher BMI, a higher respiratory disturbance index (RDI) and a higher snoring index, while subjective sleep quality and daytime sleepiness did not differ. Results from the multiple regression analysis showed that an objectively shorter sleep duration, more N2 sleep, and a higher RDI predicted AHI scores. Conclusion: The pattern of results suggests that blending demographic, anthropometric, and subjective/objective sleep- and breathing-related data enabled more effective discrimination of individuals at higher risk for OSA. The results are of practical and clinical importance: demographic, anthropometric, and breathing-related issues derived from self-rating scales provide a quick and reliable identification of individuals at risk of OSA; objective assessments provide further certainty and reliability.

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Abstract P068: Obstructive Sleep Apnea is Associated with an Increase Risk of Osteoporosis

Circulation ◽

10.1161/circ.131.suppl_1.p068 ◽

2015 ◽

Vol 131 (suppl_1) ◽

Author(s):

Anawin Sanguankeo ◽

Sikarin Upala

Keyword(s):

Obstructive Sleep Apnea ◽

Sleep Apnea ◽

Primary Outcome ◽

Meta Analysis ◽

Systemic Disease ◽

Obstructive Sleep ◽

Increased Risk ◽

Increase Risk ◽

Osteoporosis Risk ◽

Stimulation Of

Background: Obstructive sleep apnea (OSA) is thought to be a systemic disease and has been associated with many disorders such as metabolic, endocrine, and especially cardiovascular diseases. One of the consequences of OSA is hypoxia, which can lead to a reduction in growth of osteoblast and a stimulation of osteoclast. Our meta-analysis was conducted to determine the risk of osteoporosis in patients with OSA compared to controls. Objectives: Eligible studies assessing the effects of obstructive sleep apnea on osteoporosis risk were comprehensively searched in PubMed/MEDLINE, EMBASE, and CENTRAL from their inception to September 2014. Two authors independently assessed article quality and extracted the data. Primary outcome were number of participants, prevalence, or risk ratio of osteoporosis in OSA and controls. Results: From 40 full-text articles, 3 studies involving 113,090 participants were included in the meta-analysis that were based on the random effects model. Compared with controls, participants who were diagnosed with obstructive sleep apnea had increased risk of osteoporosis (pooled risks ratio, 1.85; 95% CI, 1.34, 2.56). Conclusion: Patients with OSA had a higher risk of developing osteoporosis. Further study is needed to evaluate the possible mechanisms between these two conditions and to find potential treatment for OSA that could prevent osteoporosis.

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Tonsils and Adenoids: An Update

Pediatrics in Review ◽

10.1542/pir.16.1.17 ◽

1995 ◽

Vol 16 (1) ◽

pp. 17-21

Author(s):

Ellen S. Deutsch ◽

Glenn C. Isaacson

Keyword(s):

Obstructive Sleep Apnea ◽

Sleep Apnea ◽

Sleep Disturbances ◽

Recurrent Infection ◽

The United States ◽

Cor Pulmonale ◽

Clinical Aspects ◽

Adenotonsillar Hypertrophy ◽

Obstructive Sleep ◽

Increased Risk

Pediatricians frequently face the decision of whether to recommend that a child undergo a tonsillectomy, an adenoidectomy, or both. The reputation and the indications for these procedures have evolved over the years and continue to evoke controversy. The total number of tonsillectomies and adenoidectomies performed in the United States has decreased substantially over the past 30 to 40 years, although the proportion of procedures performed for obstructive sleep apnea (OSA) has increased. We will address the indications (Table), techniques, and complications of tonsillectomies and adenoidectomies, as well as current ideas concerning OSA. The palatine tonsils, adenoids, and lingual tonsils comprise a circle of lymphoid tissue in the nasopharynx and oropharynx, sometimes referred to as Waldeyer ring (Fig. 1). Although this tissue is immunologically active, no postoperative immune deficits generally have been acknowledged other than an increased risk of complications from polio1 prior to the availability of polio vaccines. Clinical Aspects The most common problems attributed to the tonsils and adenoids are recurrent infection and OSA, or its milder manifestation, adenotonsillar hypertrophy with obstruction. Chronic middle ear disease can be related to adenoid pathology. OBSTRUCTIVE SLEEP APNEA OSA either is becoming more common in children or is being recognized more frequently. Sleep disturbances, cor pulmonale, pulmonary hypertension, and congestive heart failure are potential complications of OSA.

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An Overnight Rhythmic Sleep-Wake Pattern in Stroke and Obstructive Sleep Apnea: A Case Report and Literature Review

Journal of Sleep sciences ◽

10.18502/jss.v5i1.4571 ◽

2020 ◽

Author(s):

Hamed Amirifard ◽

Arezu Najafi ◽

Ania Rahimi-Golkhandan ◽

Mahsa Shojaie

Keyword(s):

Obstructive Sleep Apnea ◽

Case Report ◽

Sleep Apnea ◽

Comorbid Condition ◽

Cpap Titration ◽

Obstructive Sleep ◽

Increased Risk ◽

Sleep Breathing Disorder ◽

History Of ◽

Titration Study

Background and Objective: Obstructive sleep apnea (OSA) is a common sleep breathing disorder and a comorbid condition in patients with stroke. The prevalence of sleep disorders as a stroke consequence is high. Being independent of vascular risk factors, OSA is associated with increased risk of ischemic stroke. Here, we report a patient with OSA and stroke. Case Report: This case report presents a patient with a history of stroke and hemiparesis who underwent polysomnog-raphy (PSG) for evaluating OSA because of snoring, witnessed apnea, and excessive daytime sleepiness (EDS). Then the subject underwent continuous positive airway pressure (CPAP) titration for treating OSA. In first night of PSG, there was a repetitive cyclic pattern of sleep and arousals. After CPAP titration study and with CPAP pressure of 15 cmH2o, OSA and rhythmic changes of electroencephalography (EEG) were resolved. Conclusion: OSA as a risk factor for stroke can be associated with PSG cyclic wake-sleep pattern in these patients, and this pattern can be resolved by CPAP therapy.

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419 STOP-Bang Score and History of Radiation Predicts Risk of Obstructive Sleep Apnea in Cancer Patients: A Machine Learning Study

SLEEP ◽

10.1093/sleep/zsab072.418 ◽

2021 ◽

Vol 44 (Supplement_2) ◽

pp. A166-A166

Author(s):

Ankita Paul ◽

Karen Wong ◽

Anup Das ◽

Diane Lim ◽

Miranda Tan

Keyword(s):

Machine Learning ◽

Obstructive Sleep Apnea ◽

Sleep Apnea ◽

Cancer Patients ◽

Prediction Accuracy ◽

Screening Tools ◽

Cancer Type ◽

Sleep Study ◽

Obstructive Sleep ◽

History Of

Abstract Introduction Cancer patients are at an increased risk of moderate-to-severe obstructive sleep apnea (OSA). The STOP-Bang score is a commonly used screening questionnaire to assess risk of OSA in the general population. We hypothesize that cancer-relevant features, like radiation therapy (RT), may be used to determine the risk of OSA in cancer patients. Machine learning (ML) with non-parametric regression is applied to increase the prediction accuracy of OSA risk. Methods Ten features namely STOP-Bang score, history of RT to the head/neck/thorax, cancer type, cancer stage, metastasis, hypertension, diabetes, asthma, COPD, and chronic kidney disease were extracted from a database of cancer patients with a sleep study. The ML technique, K-Nearest-Neighbor (KNN), with a range of k values (5 to 20), was chosen because, unlike Logistic Regression (LR), KNN is not presumptive of data distribution and mapping function, and supports non-linear relationships among features. A correlation heatmap was computed to identify features having high correlation with OSA. Principal Component Analysis (PCA) was performed on the correlated features and then KNN was applied on the components to predict the risk of OSA. Receiver Operating Characteristic (ROC) - Area Under Curve (AUC) and Precision-Recall curves were computed to compare and validate performance for different test sets and majority class scenarios. Results In our cohort of 174 cancer patients, the accuracy in determining OSA among cancer patients using STOP-Bang score was 82.3% (LR) and 90.69% (KNN) but reduced to 89.9% in KNN using all 10 features mentioned above. PCA + KNN application using STOP-Bang score and RT as features, increased prediction accuracy to 94.1%. We validated our ML approach using a separate cohort of 20 cancer patients; the accuracies in OSA prediction were 85.57% (LR), 91.1% (KNN), and 92.8% (PCA + KNN). Conclusion STOP-Bang score and history of RT can be useful to predict risk of OSA in cancer patients with the PCA + KNN approach. This ML technique can refine screening tools to improve prediction accuracy of OSA in cancer patients. Larger studies investigating additional features using ML may improve OSA screening accuracy in various populations Support (if any):

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827 Central Sleep Apnea in a patient with Multiple Sclerosis

SLEEP ◽

10.1093/sleep/zsab072.824 ◽

2021 ◽

Vol 44 (Supplement_2) ◽

pp. A322-A323

Author(s):

Rahul Dasgupta ◽

Sonja Schütz ◽

Tiffany Braley

Keyword(s):

Multiple Sclerosis ◽

Obstructive Sleep Apnea ◽

Sleep Apnea ◽

Sleep Disordered Breathing ◽

Central Sleep Apnea ◽

Progressive Multiple Sclerosis ◽

Obstructive Sleep ◽

Increased Risk ◽

Demyelinating Lesions ◽

The Brain

Abstract Introduction Sleep-disordered breathing is common in persons with multiple sclerosis (PwMS), and may contribute to debilitating fatigue and other chronic MS symptoms. The majority of research to date on SDB in MS has focused on the prevalence and consequences of obstructive sleep apnea; however, PwMS may also be at increased risk for central sleep apnea (CSA), and the utility of methods to assess CSA in PwMS warrant further exploration. We present a patient with secondary progressive multiple sclerosis who was found to have severe central sleep apnea on WatchPAT testing. Report of case(s) A 61 year-old female with a past medical history of secondary progressive multiple sclerosis presented with complaints of fragmented sleep. MRI of the brain, cervical spine, and thoracic spine showed numerous demyelinating lesions in the brain, brainstem, cervical, and thoracic spinal cord. Upon presentation, the patient noted snoring, witnessed apneas, and daytime sleepiness. WatchPAT demonstrated severe sleep apnea, with a pAHI of 63.3, and a minimum oxygen saturation of 90%. The majority of the scored events were non-obstructive in nature (73.1% of all scored events), and occurred intermittently in a periodic fashion. Conclusion The differential diagnosis of fatigue in PwMS should include sleep-disordered breathing, including both obstructive and central forms of sleep apnea. Demyelinating lesions in the brainstem (which may contribute to impairment of motor and sensory networks that control airway patency and respiratory drive), and progressive forms of MS, have been linked to both OSA and CSA. The present data illustrate this relationship in a person with progressive MS, and offer support for the WatchPAT as a cost-effective means to evaluate for both OSA and CSA in PwMS, while reducing patient burden. PwMS may be at increased risk for CSA. Careful clinical consideration should be given to ordering appropriate sleep testing to differentiate central from obstructive sleep apnea in PwMS, particularly for patients with demyelinating lesions in the brainstem. Support (if any) 1. Braley TJ, Segal BM, Chervin RD. Obstructive sleep apnea and fatigue in patients with multiple sclerosis. J Clin Sleep Med. 2014 Feb 15;10(2):155–62. doi: 10.5664/jcsm.3442. PMID: 24532998; PMCID: PMC3899317.

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Effect of continuous positive airway pressure treatment of obstructive sleep apnea-hypopnea in multiple sclerosis: A randomized, double-blind, placebo-controlled trial (SAMS-PAP study)

Multiple Sclerosis Journal ◽

10.1177/13524585211010390 ◽

2021 ◽

pp. 135245852110103

Author(s):

Sulaiman Khadadah ◽

R John Kimoff ◽

Pierre Duquette ◽

Vincent Jobin ◽

Yves Lapierre ◽

...

Keyword(s):

Multiple Sclerosis ◽

Obstructive Sleep Apnea ◽

Sleep Apnea ◽

Continuous Positive Airway Pressure ◽

Airway Pressure ◽

Primary Outcome ◽

Positive Airway Pressure ◽

Apnea Hypopnea Index ◽

Double Blind ◽

Obstructive Sleep

Objective: The aim of this study was to evaluate the effect of continuous positive airway pressure (CPAP) treatment on the Fatigue Severity Scale (FSS, preplanned primary outcome), another fatigue measure, sleep quality, somnolence, pain, disability, and quality of life in multiple sclerosis (MS) patients with obstructive sleep apnea-hypopnea (OSAH). Methods: In a randomized, double-blind trial (NCT01746342), MS patients with fatigue, poor subjective sleep quality, and OSAH (apnea-hypopnea index of ⩾ 15 events per hour/sleep), but without severe OSAH (apnea-hypopnea index > 30, and 4% oxygen desaturation index > 15 events/hour or severe somnolence), were randomized to fixed CPAP or sham CPAP for 6 months. Outcome assessments were performed at 3 and 6 months. Results: Of 49 randomized patients, 34 completed the protocol. Among completers, FSS did not improve with CPAP compared to sham at 6 months. FSS tended to improve ( p = 0.09), and sleepiness (Epworth Sleepiness Scale) improved significantly ( p = 0.03) at 3 months with CPAP compared to sham, but there were no other improvements with CPAP at either study evaluation. Conclusion: In non-severe OSAH patients, CPAP did not significantly improve the primary outcome of FSS change at 6 months. In secondary analyses, we found a trend to improved FSS, and a significant reduction in somnolence with CPAP at 3 months.

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