835 Ictal Central Apneic Events Detected on Polysomnogram: An Educational Case Report

SLEEP ◽  
2021 ◽  
Vol 44 (Supplement_2) ◽  
pp. A325-A325
Author(s):  
Shanti Shenoy ◽  
Hai Chen ◽  
Elias Karroum

Abstract Introduction Ictal central apneas (ICA) are frequently observed in focal epilepsy, mostly with temporal lobe seizures, and have been considered as potential biomarkers of sudden unexpected death in epilepsy (SUDEP), particularly when they are prolonged and associated with significant hypoxemia. We present an interesting educational case report of occurrence of such ictal apneic events as recorded during a nocturnal diagnostic polysomnogram (PSG). Report of case(s) A 39-year-old woman with history of left focal epilepsy, hypertension, and headaches was referred to the sleep clinic for loud snoring, witnessed apneic events, and excessive daytime sleepiness. She subsequently underwent a diagnostic (PSG) that demonstrated severe obstructive sleep apnea (apnea-hypopnea index of 63.1) associated with significant hypoxemia (nadir SpO2 of 58%). In addition, the patient had one ictal discharge detected on the PSG’s limited electroencephalogram that occurred in N2 sleep and lasted for almost three minutes with a focal onset and progression in the left hemisphere. The ictal discharge was briefly preceded by central apneic events that continued to occur during and shortly after the termination of the ictal discharge. These ICA events were associated with severe oxygen desaturations down to an SpO2 of 62%. The only time during the PSG recording that the patient had central apneic events was around the ictal event. There were no behavior changes on the video during the seizure, but the ictal discharge was associated with a sustained increase in the mentalis muscles activity and a brief tachycardia. The patient’s neurologist was alerted about the above findings on PSG. The patient was taking a lower dose then prescribed of her anti-epileptic medication (topiramate) that was adjusted, and the patient was counseled on the risks associated with the above findings and positive airway pressure therapy was recommended for her severe sleep apnea. Conclusion The above case report illustrates the importance of polysomnography (specifically the recording of respiratory variables rarely performed in epilepsy monitoring units) in the evaluation of patients with epilepsy given that central apneic events (ICA and post-convulsive central apneas) potentially underlie SUDEP, the most common cause of mortality in refractory epilepsy patients and usually occurring during sleep. Support (if any) None

SLEEP ◽  
2020 ◽  
Author(s):  
Cathy A Alessi ◽  
Constance H Fung ◽  
Joseph M Dzierzewski ◽  
Lavinia Fiorentino ◽  
Carl Stepnowsky ◽  
...  

Abstract Study Objectives Cognitive behavioral therapy for insomnia (CBTI) for comorbid insomnia and obstructive sleep apnea (OSA) has had mixed results. We integrated CBTI with a positive airway pressure (PAP) adherence program and tested effects on sleep and PAP use. Methods 125 veterans (mean age 63.2, 96% men, 39% non-Hispanic white, 26% black/African American, 18% Hispanic/Latino) with comorbid insomnia and newly-diagnosed OSA (apnea-hypopnea index ≥ 15) were randomized to 5-weekly sessions integrating CBTI with a PAP adherence program provided by a “sleep coach” (with behavioral sleep medicine supervision), or 5-weekly sleep education control sessions. Participants and assessment staff were blinded to group assignment. Outcomes (baseline, 3 and 6 months) included Pittsburgh Sleep Quality Index (PSQI), 7-day sleep diary (sleep onset latency [SOL-D], wake after sleep onset [WASO-D], sleep efficiency [SE-D]), 7-day actigraphy (SE-A), and objective PAP use (hours/night and nights ≥ 4 h). Insomnia Severity Index (ISI), Epworth Sleepiness Scale (ESS), and Functional Outcomes of Sleep Questionnaire-10 (FOSQ-10) were also collected. Results Compared to controls, intervention participants showed greater improvement (baseline to 3 and 6 months, respectively) in PSQI (−3.2 and −1.7), SOL-D (−16.2 and −15.5 minutes), SE-D (10.5% and 8.5%), SE-A (4.4% and 2.6%) and more 90-day PAP use (1.3 and 0.9 more hours/night, 17.4 and 11.3 more nights PAP ≥ 4 h). 90-day PAP use at 3 months was 3.2 and 1.9 h/night in intervention versus controls. Intervention participants also had greater improvements in ISI, ESS, and FOSQ-10 (all p < 0.05). Conclusions An intervention integrating CBTI with a PAP adherence program delivered by a supervised sleep coach improved sleep and PAP use in adults with comorbid insomnia and OSA. Trial Registration ClinicalTrials.gov Study name: Novel Treatment of Comorbid Insomnia and Sleep Apnea in Older Veterans URL: https://clinicaltrials.gov/ct2/results?cond=&term=NCT02027558&cntry=&state=&city=&dist= Registration: NCT02027558


2020 ◽  
Vol 57 (7) ◽  
pp. 808-818
Author(s):  
Alfred Lee ◽  
Brian L. Chang ◽  
Cynthia Solot ◽  
Terrence B. Crowley ◽  
Vamsee Vemulapalli ◽  
...  

Objective: To determine pre- and postoperative prevalence of obstructive sleep apnea (OSA) in patients with 22q11.2 deletion syndrome (DS) undergoing wide posterior pharyngeal flap (PPF) surgery for velopharyngeal dysfunction (VPD). Design: Retrospective study using pre- and postoperative polysomnography (PSG) to determine prevalence of OSA. Medical records were reviewed for patients’ medical comorbidities. Parents were surveyed about snoring. Setting: Academic tertiary care pediatric hospital. Patients: Forty patients with laboratory confirmed 22q11.2DS followed over a 6-year period. Interventions: Pre- and postoperative PSG, speech evaluation, and parent surveys. Main Outcome Measure: Severity and prevalence of OSA, defined by obstructive apnea hypopnea index (OAHI), before and after PPF surgery to determine whether PPF is associated with increased risk of OSA. Results: Mean OAHI did not change significantly after PPF surgery (1.1/h vs 2.1/h, P = .330). Prevalence of clinically significant OSA (OAHI ≥ 5) was identical pre- and postoperatively (2 of 40), with both cases having severe-range OSA requiring positive airway pressure therapy. All other patients had mild-range OSA. Nasal resonance was graded as severe preoperatively in 85% of patients. None were graded as severe postoperatively. No single patient factor or parent-reported concern predicted risk of OSA (OAHI ≥ 1.5). Conclusions: Patients with 22q11.2DS are medically complex and are at increased risk of OSA at baseline. Wide PPF surgery for severe VPD does not significantly increase risk of OSA. Careful perioperative planning is essential to optimize both speech and sleep outcomes.


2014 ◽  
Vol 21 (4) ◽  
pp. 260-264 ◽  
Author(s):  
Essam M. Madbouly ◽  
Rashid Nadeem ◽  
Mahwish Nida ◽  
Janos Molnar ◽  
Saurabh Aggarwal ◽  
...  

2021 ◽  
Vol 49 (2) ◽  
pp. 030006052199222
Author(s):  
Xiaorong Yang ◽  
Min Xie ◽  
Lu Tan ◽  
Taomei Li ◽  
Xiangdong Tang

Obstructive sleep apnea (OSA) is characterized by repetitive intermittent oxygen desaturation during sleep. Carbon monoxide poisoning (COP) is the second most common cause of death among non-medicinal poisonings, and oxygen therapy is the current standard of treatment for COP. We herein report a case of a 50-year-old woman diagnosed with severe OSA associated with COP. Both the OSA and COP gradually resolved by automatic continuous positive airway pressure (CPAP) therapy. New OSA symptoms appeared following the development of delayed encephalopathy after acute COP (DEACMP) 3 weeks later. Severe OSA was diagnosed 76 days after COP with an apnea–hypopnea index of 66 events/hour, and CPAP therapy was immediately administered. The patient’s DEACMP symptoms and OSA both improved with CPAP therapy (her apnea–hypopnea index decreased to 32.4 and 16.5 events/hour at 161 and 204 days after COP, respectively). To our knowledge, this is the first case report of OSA caused by COP based on the occurrence and disappearance of OSA symptoms and laboratory findings associated with the emergence and improvement of DEACMP.


2020 ◽  
Vol 92 (9) ◽  
pp. 39-43
Author(s):  
E. M. Elfimova ◽  
O. O. Mikhailova ◽  
N. T. Khachatryan ◽  
A. Y. Litvin ◽  
I. E. Chazova ◽  
...  

Aim.To study the effectiveness of prolonged use of PAP therapy (positive airway pressure therapy) in eliminating sleep respiratory disorders and associated cardiac conduction disturbances. Materials and methods.We included 21 patients who were examined at the Myasnikov Institute of Clinical Cardiology, National Medical Research Center of Cardiology, regarding cardiac rhythm and conduction disturbances, as well as obstructive sleep apnea and who have been on PAP therapy for more than 12 months. The average age was 66.5 [63.5; 73.2] years, body mass index 33.0 [30.2; 38.5] kg/m2, apnea-hypopnea index 65.0 [59.0; 86.3]/h. At the time of analysis, 15 patients continued to use PAP therapy (mean time of use: 6.0 years [4.7; 9.2]) and 6 patients refused long-term use of PAP therapy, mean time to use PAP therapy until failure amounted to 2.82.1 years. Results.PAP therapy lead to a persistent decrease in apnea-hypopnea index of 63.6/h to 3.7/h was (p=0.0002). 86% of patients met the criteria for adherence to PAP therapy (use 4 hours/night, more than 70% of nights). Initially, before the use of PAP therapy, all cardiac conduction disorders were during sleep and exceeded 3 seconds, with fluctuations from 3.1 to 10.6 seconds. PAP therapy appeared to be effective in all patients: no asystoles, duration of more than 3 seconds, were detected. Conclusion.In obstructive sleep apnea patients with concomitant nighttime cardiac conduction disturbances, the long-term use of PAP therapy is effective and with good adherence.


SLEEP ◽  
2020 ◽  
Vol 43 (Supplement_1) ◽  
pp. A473-A473
Author(s):  
Brittany Monceaux ◽  
Megan Smalley ◽  
Ugorji Okorie ◽  
Edmond Roberts ◽  
Cesar Liendo ◽  
...  

Abstract Introduction Weill-Marchesani Syndrome (WMS) is a rare systemic genetic connective tissue disorder which usually presents with symptoms of short stature, limited joint movement, and eye problems such as glaucoma and microspherophakia. This genetic condition is associated with fibrous tissue hyperplasia. WMS is inherited as autosomal dominant or autosomal recessive patterns in families leading to a variability in presenting phenotype. Few papers have been written on airway management during anesthesia but as far as we know, this is the first case report on obstructive sleep apnea management in a patient with WMS. Report of Case A 9 year old boy with a past medical history of Methylene THF Reductase deficiency, von Willebrand’s Disease, seizure disorder, premature birth, developmental delays and Weill-Marchesani syndrome was referred to Sleep Medicine due to tonsillar hypertrophy (3+), snoring and witnessed apneas. Upon physical examination, patient had mid-facial hypoplasia, retropositioning of the mandible, high arched palate, Mallampti class IV, maxillary hypoplasia and mandibular hypoplasia. He had been evaluated by ENT which determined the patient to be too high risk due to his medical conditions for T&A. The patient had a polysomnogram in 2018 indicating OSA with an apnea-hypopnea index of 4.2 and a minimum oxygen saturation of 91%. After a CPAP titration study, the patient was started on Auto CPAP of 5-15 cmH2O and has shown improvement in symptoms based on subjective and objective compliance report. Patient has been able to tolerate PAP therapy well with 100% compliance greater than 4 hours per night. Conclusion This case is the first illustrating OSA in a patient with Weill-Marchesani Syndrome. In WMS, the causes of OSA are not only due to tonsillar hypertrophy, but multifactorial, including craniofacial abnormalities. Given the high risk of surgical complications in WMS patients, PAP therapy appears to be a reasonable option for OSA management.


Blood ◽  
2015 ◽  
Vol 126 (23) ◽  
pp. 3348-3348
Author(s):  
Jihyun Song ◽  
Krishna Sundar ◽  
Perumal Thiagarajan ◽  
Josef T. Prchal

Abstract The number of red blood cells (RBC) is tightly controlled to maintain tissue oxygen delivery. During hypoxia, RBC mass is increased by erythropoietin (EPO), which is regulated by hypoxia-inducible transcription factors (HIFs). Neocytolysis is a pathophysiological mechanism that overcorrects the high RBC mass generated during chronic hypoxemia by preferential destruction of young RBCs after normoxia is restored. We recently showed that neocytolysis is caused by excessive mitochondrial-derived reactive oxygen species (ROS) in reticulocytes mediated by downregulation of HIF-regulated Bnip3L (Bnip3L regulates mitophagy) and a decrease in catalase in hypoxia-generated erythrons. Catalase changes are due to hypoxia-induced miR-21 that downregulates catalase. We showed that removal of ROS in plasma by PEG-catalase attenuated neocytolysis, indicating that excessive ROS in plasma from reticulocytes diffuses into RBCs, resulting in preferential destruction of hypoxia-born RBCs with low catalase (Song J, et al. J Mol. Med., 2015 93:857-66). Neocytolysis is well described in astronauts and mountaineers, and here we report this phenomenon in obstructive sleep apnea (OSA). OSA results in chronic intermittent hypoxia (CIH), which is associated with increased incidence of cardiovascular disease, insulin resistance, and cancer. However, the mechanisms of CIH-induced morbidities are still unknown. Despite CIH, polycythemia is rarely present in OSA; our analysis of 190 consecutive OSA patients at the University of Utah showed that only 4% of OSA patients had polycythemia that corrects after therapy with continuous positive airway pressure therapy (CPAP). Several studies reported that CIH also increases ROS. We hypothesize that neocytolysis might explain why most patients with OSA do not have polycythemia, and that increased ROS in blood cells during OSA may account for oxidant related injury seen with OSA associated diseases. We analyzed rate of erythropoiesis by EPO levels, HIFs (by HIF-regulated genes transcripts), catalase and miR-21, mitochondrial mass and Bnip3L transcripts, and ROS in blood cells in 30 OSA patients without polycythemia and without chronic cardiopulmonary disease before and after 3 months of CPAP. Patients ranged in OSA severity according to an apnea-hypopnea index from mild to severe. An average of 99.0 minutes was spent with oxygen saturations of 89% or lower, with a 4% oxygen desaturation index of 20.5/hr. The average lowest oxygen saturation value was 78.0%. EPO was higher before CPAP (p=0.0054), indicating that uncorrected OSA augments erythropoiesis in spite of normal hematocrit. We then reasoned that CIH of OSA might cause overcorrection of increased erythropoiesis by neocytolysis. We first tested HIFs changes during CIH indirectly by measuring selected HIF-regulated gene transcripts (GLUT1, HK1, PDK1, NOX2, SOD2, TFRC, and EGLN1) in blood cells. We found that CIH-mediated downregulation of HIF-regulated gene transcripts is consistent with intermittent rapid abrogation of apnea cycles. Further, blunted HIFs levels were differently regulated in reticulocytes, platelets and granulocytes. ROS were found to be increased during OSA in B-cells, T-cells, monocytes, granulocytes, and mature RBCs, but not in platelets. The elevated ROS corrected after CPAP treatment. These changes of ROS in blood cells coexisted with changes in mitochondrial mass and BNIP3L transcript levels; CPAP treatment decreased mitochondrial mass in reticulocytes and granulocytes but not in lymphocytes or monocytes. Catalase levels in granulocytes, platelets and reticulocytes were lower in uncorrected OSA and corrected after CPAP treatment; reverse correlation with miR-21 was found. These results indicate that neocytolysis occurs in uncorrected OSA resulting in increased mitochondrial mass in reticulocytes (and presumably also in erythroid progenitors) and granulocytes and that excessive ROS generated by increased mitochondrial mass preferentially destroy RBCs with low catalase,resulting in an absence of polycythemia in untreated OSA patients, supporting the central role of HIFs in OSA pathophysiology. Furthermore, increased ROS production may be causally related to other pathologies in OSA such as increased sympathetic tone due to stimulation of carotid bodies by ROS. Disclosures No relevant conflicts of interest to declare.


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