Repeat Polysomnogram Research Articles

0980 A Child with Achondroplasia, Cervicomedullary Junction Compression, Severe Obstructive Sleep Apnea and Pulmonary Hypertension

Abstract Introduction Achondroplasia is a short stature autosomal dominant skeletal dysplasia. Children with Achondroplasia are at risk for multiple medical comorbidities including sleep disordered breathing due to midface hypoplasia and craniocervical junction compression. Recent guidelines from American Academy of Pediatrics on health supervision of children with achondroplasia recommend routine clinical screening for symptoms of obstructive sleep apnea (OSA) and craniocervical junction compression in which case a polysomnogram should be considered. Report of case(s) The patient is a now 15 month old male who was initially seen in sleep medicine clinic at age 4 months for snoring and respiratory distress while awake. Polysomnogram 2 days later demonstrated severe OSA. The study demonstrated an oAHI of 25.1/hour, O2 nadir of 77%, and TcCO2 with evidence of significant hypoventilation with TcCO2 max of 67mmHg. This was adequately treated with supplemental oxygen. The patient subsequently presented to the emergency department at age 7 months with hypoxemic respiratory failure and signs of right heart failure and was started on CPAP. Echocardiogram demonstrated significant pulmonary hypertension. Subsequent CPAP titration study during admission demonstrated adequate treatment of OSA and his pulmonary hypertension significantly improved with the patient requiring no pulmonary hypertension treatment at discharge. At age 9 months, the patient was again admitted with hypoxemic respiratory failure and subsequent brain MRI demonstrated foramen magnum stenosis requiring subsequent surgical decompression. He was discharged on CPAP, however, was not tolerating this at home. Therefore, repeat polysomnogram was obtained that demonstrated severe OSA that was not responsive to supplemental oxygen. The patient was then started on BPAP therapy which he has since been tolerating well. Conclusion Children with achondroplasia are at increased risk for sleep disordered breathing and craniocervical junction compression. Our patient experienced significant complications from OSA at a young age. His sleep apnea worsened significantly in a short period making it unresponsive to previous treatment recommendations. There is a need for further studies to determine better screening guidelines for craniocervical junction narrowing and OSA. This case highlights the dynamic nature of sleep disordered breathing in achondroplasia and need for close follow up and monitoring of symptoms at all clinical encounters. Support (if any)

1025 Schizophrenic Patient with Large REM Rebound Following Treatment of Severe Obstructive Sleep Apnea

Abstract Introduction We present a case of uncontrolled schizophrenia with severe obstructive sleep apnea (OSA) and significant REM rebound after bilevel positive airway pressure (BPAP) therapy. Report of case(s) A 74-year-old female, with Schizophrenia on anti-psychotic therapy, presented to the sleep clinic due to excessive daytime sleepiness (EDS) and an Epworth score of 21/24. Diagnostic polysomnogram (PSG) reported an AHI of 162.1 events/hour with SaO2 nadir of 71% with sleep efficiency of 31.6% and no REM-sleep, consistent with severe OSA. The next morning, multiple sleep latency testing (MSLT) demonstrated mean sleep latency (MSL) of 3.6 minutes with 0 REM-onset sleep periods (SOREMPs). Patient underwent CPAP/BPAP titration which improved sleep efficiency to 88.6% and severe REM rebound at 61.7%. MSLT after the titration study demonstrated MSL of 4.0 minutes and 3 SOREMPs and was initiated on BPAP therapy at 20/13 cmH2O. A repeat PSG, 4 weeks later showed normalization of sleep architecture. After starting PAP therapy, the patient had a significant improvement in EDS but also energy and interest levels and denied feelings of sadness, racing or paranoid thoughts. Her social life improved and started exercising regularly and volunteering in the community. There was also improved blood pressure and blood sugar levels. She was followed up in the sleep clinic regularly and remained compliant with and benefiting from BPAP therapy and was continued on anti-psychotic therapy. Conclusion Untreated OSA often requires repetitive arousals to reopen the obstructed upper-airway that results in increased non-REM sleep at the expense of slow wave sleep and REM sleep. PAP therapy can cause a rebound increase in REM and SWS. OSA is under recognized in schizophrenic patients and other psychiatric disorders which results in development of significant comorbidities with greater severity of psychiatric symptoms. Elevated rates of OSA have been seen in the schizophrenic population. Treatment of OSA helps with not only physical but also psychotic symptoms. Many anti-psychotics, due to causing weight gain, may contribute to OSA. Studies also indicated that CPAP response to OSA, was as favorable in people with severe mental illness, compared to other patient groups. Support (if any) REM rebound and CPAP compliance; B. Koo, R. Wiggins, C. Molina

0864 Severe Central Sleep Apnea

Abstract Introduction Central sleep apnea (CSA) is a rare form of sleep disordered breathing with repeated apneic episodes with absence of associated respiratory effort. CSA due to medication is well described in literature with opiate therapy. Baclofen which is a gamma-aminobutyric acid-B agonist with muscle relaxant properties has been implicated in CSA. We present a rare case of CSA likely due to chronic baclofen use. Patient underwent a polysomnogram (PSG) while on baclofen therapy which revealed severe central sleep apnea. Patient was weaned off baclofen therapy completely and a subsequent PSG was performed which revealed resolution of the CSA. Methods 28 year old male with cerebral palsy and neurogenic bladder who presented for evaluation of snoring, chronic insomnia and non-refreshing sleep. Overnight PSG was performed for further evaluation of his sleep related complaints while on chronic baclofen therapy, which revealed severe central sleep apnea. Trial of positive airway pressure therapy for treatment of CSA and supplemental oxygen therapy were attempted during the PSG but had to be terminated due to intolerance. Echocardiography and magnetic resonance imaging of the brain were performed to rule out congestive heart failure and Chiari malformation respectively, which revealed no underlying pathology to explaining the CSA. CSA in this patient was thought to be associated with chronic baclofen therapy. Patient was weaned off baclofen with repeat PSG revealing resolution of the CSA. Results 1st diagnostic PSG - on Baclofen: 139 central apneas, 6 obstructive apneas, 3 mixed apnea, and 38 hypopneas. This study showed severe central sleep apnea. The apnea-hypopnea index (AHI) was 51.5, REM index 21.8 and supine index 51.5. Central apnea index 39.5. Respiratory events were associated with moderate oxygen desaturations. The mean oxygen saturation during the study was 92.8%, with a minimum oxygen saturation of 81%. The patient spent 2.9 minutes with an oxygen saturation equal to or less than 88% for the entire study. CPAP titration was attempted and aborted, due to patient having difficulty tolerating CPAP. Supplemental oxygen was briefly added at 0.5 LPM, which was also discontinued due to patient complaining of burning sensation in the nose. Head of the bed was elevated at several levels (20 degrees, 30 degrees and 45 degrees) which did not attenuate the respiratory events. 2nd Diagnostic PSG – patient weaned off baclofen for study – Sleep efficiency reduced at 59.4%. Sleep onset latency 7.5 minutes. REM latency 163 minutes. 100% of total sleep time in supine position Moderate snoring. The apnea-hypopnea index (AHI) was 4.2, REM index 36.5 and supine index 4.2. Respiratory events were mainly noted using REM sleep. Respiratory events were associated with moderate oxygen desaturations. The mean oxygen saturation during the study was 93.9%, with a minimum oxygen saturation of 83%. The patient spent 1.2 minutes with an oxygen saturation equal to or less than 88% for the entire study. Labs and imaging – Workup for severe central sleep apnea after first diagnostic sleep study – Echo and MRI brain, which revealed no underlying pathology explaining his central sleep apnea. Therefore, thought to be due to baclofen. Conclusion 28 year old male, concurrent and past medical history of cerebral palsy, neurogenic bladder. Presented, initially to sleep medicine clinic for complaints of insomnia, snoring, and non-refreshing sleep. Wheelchair bound. Has muscle spasticity, was on baclofen. Underwent initial sleep study, for snoring and insomnia. The initial PSG was on baclofen doses. His initial PSG – showed apnea-hypopnea index (AHI) of 51.5, REM index 21.8 and supine index 51.5, Central apnea index 39.5. Oxygen nadir – 81%. Patient had 139 central sleep apnea events. Patient had MRI and echo, which revealed no pathology to explain his severe central sleep apnea. Baclofen on his medicine list – was the concern for the driving force behind his central sleep apnea. As a result, baclofen was weaned and stopped. Repeat PSG – showed moderate snoring, overall AHI greatly improved from first study to 4.2. Respiratory events were associated with moderate oxygen desaturations. The mean oxygen saturation during the study was 93.9%, with a minimum oxygen saturation of 83%. The patient spent 1.2 minutes with an oxygen saturation equal to or less than 88% for the entire study. In closing – due to the side effect profile of baclofen leading to central sleep apnea in this case, recommend alternative therapy in place of baclofen due to severe central sleep apnea with baclofen use. Support (If Any) 1. Ghanavatian S, Derian A. Baclofen. In: StatPearls. Treasure Island (FL): StatPearls Publishing; May 15, 2021.2. Locatelli F, Formica F, Galbiati S, et al. Polysomnographic Analysis of a Pediatric Case of Baclofen-Induced Central Sleep Apnea. J Clin Sleep Med. 2019;15(2):351-354. Published 2019 Feb 15. doi:10.5664/jcsm.76443. Olivier PY, Joyeux-Faure M, Gentina T, et al. Severe Central Sleep Apnea Associated With Chronic Baclofen Therapy: A Case Series. Chest. 2016;149(5):e127-e131. doi:10.1016/j.chest.2015.10.001

571 Evaluating Need for Follow up Polysomnograms after Adenotonsillectomy in children

Abstract Introduction Most children with Obstructive sleep apnea (OSA) undergo repeat polysomnogram (PSG) following adenotonsillectomy (T & A). Repeat PSG is often performed regardless of presence of symptoms of sleep disordered breathing after T & A. PSG data performed before and after T & A performed at our institution over past 2 years were reviewed. Data was collected on patient characteristics such as age, sex, BMI, comorbidities and AHI on pre-T & A study. Data was then analyzed looking at patient characteristics that can help predict significant OSA (AHI >5) in post T & A study. Methods Retrospective review of 50 consecutive PSGs before and after T & A performed at our institution over past 2 years was performed. Data was collected on age, sex, Obstructive AHI, BMI, O2 nadir and comorbidities to identify patient characteristics to predict significant OSA (AHI>5) in the post T & A study. Results Age range of our cohort was 2–16 years. Average age and BMI of our cohort was 5.8 years (median 5 years) and 16.6 respectively. 8 patients had significant OSA (AHI>5) in the after T & A study. AHI (>20) and increased BMI z-score at baseline was highly predictive of significant OSA (AHI>5) in the after T & A study. Conclusion Based on our data, follow up PSG is warranted if AHI is >20/hour on baseline diagnostic PSG. Similarly, high BMI z-score at baseline study is also highly predictive of significant OSA (AHI>5) in the post T & A study. Most children with associated comorbidities with mild (AHI 2–5 hour), moderate (AHI 5–10/hour) and lower end of severe OSA (AHI 10–20/hour) do not need repeat PSG after T & A unless symptoms of sleep disordered breathing are present. We are analyzing larger PSG database to identify other patient characteristics that may signal need for follow up PSG. As PSGs are expensive and time consuming, follow up PSGs should be performed only if clinical suspicion for residual OSA is high to optimize resource utilization. Support (if any):

467 Positive Airway Pressure Tracking Systems: Outcomes of Polysomnographic Testing Prompted by the Residual Apnea Hyponea Index

Abstract Introduction To assess positive airway pressure (PAP) adherence and efficacy, tracking systems have been developed to monitor hours of CPAP use, mask leak, and residual apnea-hypopnea index (AHI) while patients are on treatment. No formal guidelines however have been developed on how to interpret and utilize this information. We looked at treatment outcomes after an in-laboratory sleep study reevaluation was made based on clinical symptoms and the residual AHI. Methods We performed a retrospective chart review of adult patients evaluated with an in-laboratory polysomnogram (PSG) based on a clinical concern for inadequately treated obstructive sleep apnea (OSA) and the residual AHI obtained from the PAP tracking system. We documented the outcomes of the repeat study and follow-up AHI after the new intervention (if recommended). We excluded patients non-adherent to PAP. Results Nine patients were identified between January 2015 and 2020 at the McGovern Medical School Outpatient Sleep Clinic. All nine patients were male with an average age of 69.2 years (range 44–84). The average AHI on the diagnostic study (CMS criteria) was 37.1 events/hour (range 17.4–67.1). The average residual AHI prompting reevaluation was 9 events/hour (median 15.9). All patients had a change in treatment based on recommendations made after their sleep study. The clinical suspicion for central events on the tracking system was confirmed on PSG on three patients who were subsequently switched to adaptive servo-ventilation. Two patients were found to have central events without a previous suspicion for central events. Four were prescribed a higher pressure or BPAP for suspected untreated OSA confirmed on the repeat PSG. All of the patients had a decreased residual AHI (average 6.3 events/hour) after treatment changes were made. Conclusion Reevaluation with a PSG after concerns of the residual AHI led to a change in diagnosis (complex sleep apnea) or the need for higher treatment pressures in our cohort. This lead to the optimization of therapy and a decrease in AHI on the tracking system post-intervention, hence justifying the repeat PSG. Exact guidelines however need to be set to standardize the recommendations with a potential cut-off residual AHI after which a repeat PSG is the standard. Support (if any):

558 A Surface Electrode Adjacent to Vagal Nerve Stimulator Lead Can Aid in Characterizing VNS Mediated Sleep Disordered Breathing

Abstract Introduction Vagal nerve stimulators (VNS) are a nonpharmacological treatment for patients with refractory epilepsy. The VNS can decrease seizure frequency by over 75% in 40% of pediatric patients with refractory epilepsy. An underrecognized side effect is sleep disordered breathing (SDB). The purpose of this study was to demonstrate how a sensor placed adjacent to the VNS lead can distinguish whether SDB is due to VNS discharge. Methods Five pediatric patients (ages: 5–8) with refractory epilepsy with VNS were referred to our sleep center for concern for SDB. Each patient underwent a polysomnogram (PSG) that included a standard PSG montage with a surface electrode placed adjacent to their left lateral neck to detect VNS discharge. VNS associated apnea hypopnea index (vAHI) was calculated by determining the number of hypopneas and obstructive apneas occurring during VNS discharge. Results Of the 5 patients, three met pediatric criteria for obstructive sleep apnea (OSA). Patient 1 had an obstructive AHI (oAHI) of 21.3 events/hr with a vAHI accounting for 79% of the total (16.8 events/hr), patient 2 had an oAHI of 16.6 events/hr with a vAHI accounting for 57% of the total (9.5 events/hr), and patient 3 had an oAHI of 1.9 events/hr with vAHI accounting for 68% of the total (1.3 events/hr). Because of these findings, the VNS settings of all 3 patients were changed with the goal of reducing SDB due to VNS discharge. Upon repeat PSG, patient 2 had reduced OSA with an oAHI of 3 events/hr, with no events associated with VNS discharge. The remaining 2 patients did not exhibit VNS associated SDB, however, both experienced increased respiratory rate during VNS discharge. Conclusion We demonstrated that a surface electrode adjacent to the VNS is able to temporally co-register VNS discharges and enabled us to directly correlate SDB to VNS stimulation in 3 patients with refractory epilepsy. Because of our findings, we titrated the VNS parameters in all 3 patients, with one showing resolution of VNS associated SDB on repeat PSG. We propose that an added surface electrode to detect VNS discharge be considered as standard practice in PSG studies of patients with VNS. Support (if any):

833 Evolution of sleep disordered breathing types in heart failure

Abstract Introduction Central sleep apnea is commonly seen in patients with heart failure. Here we present a case demonstrating shifting of predominant apneic events from central to obstructive type after placement of left ventricular assist device (LVAD) in end stage heart failure patient. Report of case(s) Case Presentation: 66 year-old African American male has past medical history of chronic congestive heart failure diabetes, hypertension, paroxysmal atrial fibrillation, anemia, hypothyroidism, chronic kidney disease and sleep apnea. Prior to his LVAD placement, his left ventricular ejection fraction (EF) was <10%. Patient was diagnosed with central sleep apnea with AHI of 58 (with 92% of apneic events being central events), oxygen nadir of 74%. Subsequently, patient had LVAD placed for symptomatic heart failure and repeat polysomnogram repeated at six month demonstrated an improved AHI of 45.8 with predominantly obstructive and mixed apneic events, with only 12.5% being central events. Conclusion This case report highlights not only the improvement of the sleep apnea in CHF treated with LVAD but also shows the shift of apneic events from predominantly central to obstructive type post LVAD. Support (if any) 1. Henein MY, Westaby S, Poole-Wilson PA, Cowie MR, Simonds AK. Resolution of central sleep apnoea following implantation of a left ventricular assist device. Int J Cardiol. 2010 Feb 4;138(3):317–9. PMID: 18752859. 2. Köhnlein T, Welte T, Tan LB, Elliott MW. Central sleep apnoea syndrome in patients with chronic heart disease: a critical review of the current literature. Thorax. 2002 Jun;57(6):547–54. PMID: 12037232 3. Monda C, Scala O, Paolillo S, Savarese G, Cecere M, D’Amore C, Parente A, Musella F, Mosca S, Filardi PP. Apnee notturne e scompenso cardiaco: fisiopatologia, diagnosi e terapia [Sleep apnea and heart failure: pathophysiology, diagnosis and therapy]. G Ital Cardiol (Rome). 2010 Nov;11(11):815–22. Italian. PMID: 21348318.

Adenoidectomy Without Tonsillectomy for Pediatric Obstructive Sleep Apnea.

The primary objective was to determine if obstructive sleep apnea (OSA) can improve after adenoidectomy. Case series with chart review. Tertiary children's hospital between 2016 and 2018. The study included children under 3.5 years with small (1+ or 2+) palatine tonsils, large (3+ or 4+) adenoids, and documented OSA on polysomnogram (PSG). Seventy-one children were included. Age at adenoidectomy was 2.0 years (95% CI, 1.8-2.2) and 71.8% were male. Mean follow-up was 2.5 years (95% CI, 2.3-2.7). Twenty-six children (36.6%) obtained a repeat PSG at a mean of 9.7 months (95% CI, 6.3-13.2) after adenoidectomy. Among those with a postoperative PSG, apnea-hypopnea index decreased in 77.0% (mean, -3.2 events/h; 95% CI, -14.1 to 7.6), and the proportion with moderate to severe OSA decreased from 65.4% to 30.8% (P = .03). Six children (23.1%) had a normal PSG after adenoidectomy. Tonsillectomy was performed in 14.1% of children at 12.1 months (95% CI, 7.5-16.7) after adenoidectomy. Despite similar preoperative PSG variables, younger children (1.5 vs 2.1 years, P = .02) were more likely to require tonsillectomy. Substantial adenoid regrowth was identified in 1 child at the time of tonsillectomy. Adenoidectomy may improve OSA in young children with large adenoids and small tonsils. However, younger age predicted the need for subsequent tonsillectomy. Prospective studies with additional PSG data are necessary to corroborate these findings.

1228 A CASE OF PULMONARY HYPERTENSION AND SEVERE OBSTRUCTIVE SLEEP APNEA IN A PEDIATRIC PATIENT WITH DOWN SYNDROME

Abstract Introduction Children with Down syndrome (DS) are at risk for obstructive sleep apnea (OSA) and related health consequences. Adherence to treatment plans, especially positive airway pressure (PAP), can be difficult for people with DS. We present a case of a pediatric patient with DS who developed pulmonary arterial hypertension (PAH) due to poor adherence of OSA therapy. Report of Case A 4-year-old male with a past medical history of DS was diagnosed with severe OSA and hypoventilation at 18 months of age – AHI 27, (oAHI 27), CO2 above 50 mmHg for 53.2% of total sleep time (TST)). A titration study showed that on a bi-level pressure (BPAP) of 11/6 cm H2O his OSA was improved – AHI 5.9 (oAHI 3.7), O2 nadir of 93%. The family attempted PAP at home but discontinued after 2 weeks. He underwent an adenoidectomy at 22 months of age and tonsillectomy and repeat adenoidectomy at 40 months of age. Patient presents to the emergency center with respiratory distress. Chest films showed cardiomegaly. Echocardiogram revealed evidence of worsening PAH (tricuspid regurgitation of 3.5-3.8 m/s and flattening of the interventricular septum). He was admitted to the pediatric intensive care unit for further management of his PAH attributed to his underlying uncorrected OSA and hypoventilation. Repeat polysomnogram showed continue severe OSA and hypoventilation – AHI 30.3 (oAHI 30.3) CO2 above 50 mmHg for 89% of TST. Repeat titration study showed that a pressure of 11/7 cm H2O improved his OSA– AHI 6.4 (oAHI 6.4) CO2 max of 52 mmHg. Treatment with BPAP was initiated prior to discharge. Repeat echocardiogram 2 months after admission showed improvement of the PAH while on BPAP without additional vasoactive drug therapy. Conclusion Children with DS are at high risk for OSA. Although treatment can be difficult, proper management of the OSA essential to preventing potential serious health consequences.

1234 Constrictive Pericarditis, an unlikely cause of Central Sleep Apnea

Abstract Introduction Central sleep apnea (CSA) and Cheyne stokes respiration (CSR) is a well-recognized complication of heart failure across multiple New York Heart Association functional classes. We present a case of CSA associated with constrictive pericarditis and normal systolic function. Report of Case 43-year-old male with past medical history of severe obstructive sleep apnea noted to have high residual apnea-hypopnea index (AHi) while using automatic continuous positive airway pressure, suspected to be central events. The patient also had progressive dyspnea on exertion, chest pain, and bilateral transudative pleural effusions. Pulmonary function testing was normal. He underwent a split night PSG which demonstrated severe OSA with AHi 81/hour during baseline. With CPAP titration, AHi was 30/hr, with predominately central events. Transthoracic echocardiogram demonstrated reduced stroke volume (LVOT VTI .085), dilated IVC, and EF 55%, septa! bounce, and annulus reversus (possible constriction). Right and left heart catheterization showed equalization of the diastolic pressures (RA 35 mm Hg, RV 48/30, PA 49/30, PCWP 29, Cl 1.6, SVR 1118 dynes, PVR 216 dynes), no coronary disease, and codominant system. Technetium 99 pyrophosphate scan demonstrated symmetric uptake in left and right ventricles, but abnormal thickening of pericardium suspicious for constrictive pericarditis. His symptoms progressed despite aggressive medical therapy and he was ultimately taken to surgery for pericardiectomy where it was noted his pericardium was extremely thickened and densely adhered to the myocardium, consistent with constrictive pericarditis. There was immediate improvement in hemodynamic status post-operatively. At post discharge follow up, he was NYHA class I, with resolution of all signs of heart failure. Repeat polysomnogram demonstrated persistent OSA, but resolution of CSA at baseline and with CPAP. Conclusion Constrictive pericarditis has not previously been reported as a cause of CSA or CSR. This patient had complete resolution of his heart failure symptoms with definitive resection of his pericardium.

1223 Central sleep apnea and Chiari 1 malformation in a pediatric patient with Klippel-Feil sequence

Abstract Introduction Klippel- Feil Sequence (KFS) is a rare congenital condition that classically presents with a triad of congenital cervical spine fusion, reduced cervical spine flexion and low posterior hairline. KFS has been associated with several comorbidities including congenital heart defects, hearing loss, renal dysfunction, Chiari malformation and sleep disordered breathing (SDB). The co-occurrence of SDB and chiari malformation type 1 (CM1) has been reported in multiple individuals. However, the pathological basis of the connection between CM1 and SDB in the setting of KFS is not clearly understood. Report of Case Here we present a pediatric case report of a patient with KFS, SDB, drooling and dysphagia. The drooling and dysphagia prompted an MRI that revealed CM1. Baseline polysomnogram (PSG) showed mild central sleep apnea with apnea-hypopnea index (AHI) 6.06 comprised of central apnea index 4.81 and obstructive apnea index 1.28. Posterior fossa decompression of this patient, following neurology recommendations, resolved majority of the symptoms, namely the drooling, nocturnal cough, dysphagia and sleep disturbances. However unexpectedly repeat polysomnogram eight weeks after posterior fossa decompression revealed worsening central sleep apnea despite the patient being clinically asymptomatic. Conclusion Taken together this case highlights the point that while it is critical to recognize the association of SDB in the setting of KFS, decompression alone may not be sufficient to completely alleviate SDB and/or certain neurological symptoms such as nocturnal coughing. Our observations are consistent with the hypothesis that SDB in a setting of KFS is multifactorial and pathophysiology is not clearly understood.

825: Can we use home sleep testing with autoscore to triage for sleep apnea in obese pregnant women?

Obstructive sleep apnea (OSA) in pregnancy has been associated with adverse pregnancy outcomes. However, there are still limited data on how best to test for and treat OSA in pregnancy. Home sleep testing (HST) devices with autoscoring capabilities may lessen the burden of testing for OSA in pregnancy, however data regarding their reliability in pregnancy are limited. Our goal was to evaluate the reliability of HST with autoscoring at low apnea hypopnea index (AHI) values. This was an ancillary study of an ongoing prospective study of OSA in obese pregnant women. For the primary study women are recruited in early pregnancy and undergo an in-lab polysomnogram (PSG) before 21 weeks’ gestation to evaluate for OSA. OSA is defined as an AHI of ≥ 5, with values of 4.6 or greater rounded up to 5. Those who test negative for OSA at the first PSG, return for a repeat evaluation between 28-32 weeks’ gestation. It is anticipated that some of these women will developed mild OSA during pregnancy, but most are expected to have a second negative study. A subgroup of these women were asked to wear an Apnea-Link HST device for 1 night, within 2 weeks of the repeat PSG. We used the Apnea-Link autoscoring algorithm to obtain a AHI for the HST. HSTs were considered valid if they had a ≥ 4 hours of nasal flow signal. We compared the HST-autoscore to PSG using the intraclass correlation coefficient (ICC), and assessed categorical agreement (AHI≥ 5 (positive) or <5 (negative)). 34 women were recruited and we obtained 21 valid HST (5 women never wore the device, 8 had an inadequate signal). 4/21 women were diagnosed with OSA by in-lab PSG. The agreement between HST-autoscore AHI and PSG AHI was good (ICC 0.73, Figure). Categorical agreement was excellent (90.5%). All negative PSG studies were negative on HST-autoscore. The 2 positive HST-autoscore studies were also positive on PSG. Of the 4 positive PSG, 2 were negative by HST-autoscore (5.01 PSG vs. 2.4 auto and 5.47 PSG vs. 4.2 auto). A low HST-autoscore of < 2 may be sufficient to rule out OSA in obese pregnant women. Our data demonstrates that HST with autoscoring may be a useful tool to help quickly triage obese women for further OSA evaluation.

1254 Resolution of persistent nocturnal hypoxia in a patient with treated obstructive sleep apnea after closure of a patent foramen ovale

A higher prevalence of patent foramen ovale (PFO) is documented in individuals with obstructive sleep apnea (OSA). Concurrent OSA and PFO can lead to more severe nocturnal desaturation although the effect of closure of PFO on nocturnal hypoxia is unclear. A 38 year old male with hypertension, tobacco use and obesity presented to the WLA-VA sleep center with a prior diagnosis of OSA. A polysomnogram (PSG) performed 7 years prior demonstrated an AHI of 99.0/hr with 4.7% of TST spent at an O2 saturation of <89% and he was successfully titrated to a CPAP of 10 cm H20. The patient was non-compliant with CPAP and was restarted on his prior settings of 10 cm H20 through the VA. Polycythemia (hgb 18) was noted and a workup was initiated. PFTs and CXR were normal and a blood gas showed a daytime room air PaO2 at 67mmHg. A 3-night oximetry showed long periods of time < 88% SaO2 while on CPAP (residual AHI < 5/hr). Supplemental oxygen with CPAP was initiated although nocturnal hypoxia persisted at 3L/min with CPAP. Echocardiogram with bubble study did not demonstrate a shunt but transcranial doppler revealed a high grade right-to-left shunt at rest, increasing with Valsalva maneuver. Cardiac catheterization showed xygenation step up from RA (74%) to RV (82%) indicating significant left to right shunt and TEE showed a PFO. Patient underwent successful percutaneous PFO closure. Repeat PSG on CPAP and room air showed resolution of nocturnal hypoxia and hgb remained normal after cessation of supplemental nocturnal oxygen. Comorbid OSA and PFO may lead to difficult to treat nocturnal hypoxia. A randomized trial is needed to assess if closure of PFO improves nocturnal hypoxia in these cases.

Comparison of treatment modalities in syndromic children with Obstructive Sleep Apnea—A randomized cohort study

IntroductionObstructive Sleep Apnea (OSA) is a common medical problem in adults that is becoming increasingly recognized in children. It occurs in the pediatric age group, from newborns to teens. More recently, many specialists have estimated OSA prevalence to be between 5 and 6%. However, in syndromic children, the prevalence of OSA can be from 50 to 100%, having a significant effect on their Quality-of-Life. As they are a challenging population for management, it is essential to evaluate them thoroughly before planning appropriate intervention. ObjectiveTo compare the efficacy of Adenotonsillectomy (T&A) and Continuous Positive Airway Pressure (CPAP) in syndromic children [Down syndrome (DS) and Mucopolysaccharidoses (MPS)] with Obstructive Sleep Apnea (OSA). Materials and methodsIn a prospective, randomized, cohort comparative study, 124 syndromic children (DS and MPS) aged between 6 and 12 years were recruited from a private MPS support group and the Down Syndrome Society, Chennai. A standard assessment was performed on all children who entered the study including a full overnight Polysomnogram (PSG), Epworth Sleepiness Scale-Children (ESS-C) and Quality-of-Life (QOL) tool OSA-18. The children with positive PSG who consented for the study (n=80) were randomly distributed to two groups, T&A group & CPAP group. The children were followed up with repeat PSG, clinical evaluation, ESS-C and Quality-of-Life (QOL) tool OSA-18 for a period of 1 year. Observation and resultsFollow-up was available for 73 syndromic children. Both the groups, T&A group and CPAP group, showed statistically significant (p<0.05) improvement in Apnea-Hypoapnea Index (AHI), ESS-C, QOL from the intervention. In our study, T&A showed equal outcome compared to CPAP. The contrasting feature between the two groups was that CPAP use gave immediate sustained improvement while T&A gave gradual progressive improvement of symptoms over a period of 1 year. ConclusionOn average, T&A gives equal outcomes as CPAP and it can be suggested as a first-line treatment in this group of syndromic children.

Medicare Long-Term CPAP Coverage Policy: A Cost-Utility Analysis

CPAP is an effective treatment for OSA that may reduce health care utilization and costs. Medicare currently reimburses the costs of long-term CPAP therapy only if the patient is adherent during a 90-day trial. If not, Medicare requires a repeat polysomnogram (PSG) and another trial which seems empirically not cost-effective. We modeled the cost-effectiveness of current Medicare policy compared to an alternative policy (clinic-only) without the adherence criterion and repeat PSG. Cost-utility and cost-effectiveness analysis. U.S. Medicare Population. N/A. N/A. We created a decision tree modeling (1) clinic only follow-up vs. (2) current Medicare policy. Costs were assigned based on Medicare reimbursement rates in 2012. Sensitivity analyses were conducted to test our assumptions. We estimated cumulative costs, overall adherence, and QALY gained for a 5-year time horizon from the perspective of Medicare as the payer. Current Medicare policy is more costly than the clinic-only policy but has higher net adherence and improved utility. Current Medicare policy compared to clinic-only policy costs $30,544 more per QALY. Current CMS policy promotes early identification of those more likely to adhere to CPAP therapy by requiring strict adherence standards. The policy effect is to deny coverage to those unlikely to use CPAP long-term and prevent wasted resources. Future studies are needed to measure long-term adherence in an elderly population with and without current adherence requirements to verify the cost-effectiveness of a policy change.

Improved sleep efficiency after anti-tumor necrosis factor α therapy in rheumatoid arthritis patients

Poor sleep health is increasingly recognized as contributing to decreased quality of life, increased morbidity/mortality and heightened pain perception. Our purpose in this study was to observe the effect on sleep parameters, specifically sleep efficiency, in rheumatoid arthritis (RA) patients treated with anti-tumor necrosis factor alpha (anti-TNF-α) therapy. This was a prospective observational study of RA patients with hypersomnolence/poor sleep quality as defined by the Epworth Sleepiness Scale (ESS) and Pittsburgh Sleep Quality Index (PSQI). Study patients underwent overnight polysomnograms and completed questionnaire instruments assessing sleep prior to starting anti-TNF-α therapy and again after being established on therapy. The questionnaire included the ESS, PSQI, the Berlin instrument for assessment of obstructive sleep apnea (OSA) risk, restless legs syndrome (RLS) diagnostic criteria, and measures of disease activity/impact. A total of 12 RA patients met inclusion criteria, of which 10 initiated anti-TNF-α therapy and underwent repeat polysomnograms and questionnaire studies approximately 2 months later. Polysomnographic criteria for OSA were met by 60% of patients. Following anti-TNF-α therapy initiation, significant improvements were observed by polysomnography (PSG) for sleep efficiency, increasing from 73.9% (SD 13.5) to 85.4% (SD 9.6) (p = 0.031), and 'awakening after sleep onset' time, decreasing from 84.1 minutes (SD 43.2) to 50.7 minutes (SD 36.5) (p = 0.048). Questionnaire instrument improvements were apparent in pain, fatigue, modified Health Assessment Questionnaire (mHAQ), and Rheumatoid Arthritis Disease Activity Index (RADAI) scores. Improved sleep efficiency and 'awakening after sleep onset' time were observed in RA patients treated with anti-TNF-α therapy.

An Angry Bed Partner

A 59-year-old man presented with the concern that his wife had witnessed him stopping breathing while asleep. Review of his sleep history also revealed agitated behaviors occurring during sleep. He reported almost nightly episodes of flailing his arms and legs about, often accompanied by angry yelling. When awoken from these episodes by his wife, he had clear dream recall, usually involving being in an argument or a fight with someone. His wife had resorted to sleeping in another bed so as not to be harmed. He had to date not suffered serious injury from one of these episodes. These behaviors were first noticed at 30 years of age. His past medical history included depression, hypertension, coronary artery disease and hypercholesterolemia, for which he took fluoxetine, clopidogrel, atenolol, ezetimibe/simvastatin, lisinopril, aspirin, and a multivitamin. Neurologic and systemic examinations were normal. There was no family history of neurodegenerative disease. Polysomnography with time-synchronized audiovisual monitoring and an expanded EEG montage showed mild OSA, with an AHI of 5. REM sleep was notable for persistent muscle tone recorded using 3 chin EMG leads. Overt yelling (“you want trouble, then keep it shut”) and swinging of the arms in a punching motion were recorded on video during REM sleep as well. An example of his REM sleep is shown in Figure 1. Figure 1 30-second epoch tracing of polysomnography recorded during REM sleep reveals persistent chin EMG tone during REM sleep, with overt whole-body movements at the end of the epoch. LEOG = Left electrooculogram, REOG = Right electrooculogram, FP1A2 – ... How would you treat this patient? Withdrawal of fluoxetine with careful psychiatric monitoring. REM sleep behavior disorder (RBD) is a REM-sleep parasomnia characterized by a lack of generalized atonia during REM sleep. Clinically, this usually presents as pronounced activity during sleep accompanied by vivid dream recall when awoken. Most often, the dream content is violent or anxious in nature.1 RBD may be idiopathic or secondary, being associated with neurodegenerative diseases, narcolepsy, cerebrovascular disease, intracranial tumors, central nervous system infection, and medication use. In particular, selective serotonin reuptake inhibitors, including fluoxetine, have been shown to cause secondary RBD.2 After a careful review of the patient's medication history, it was found that the RBD symptoms began around the time he had started taking fluoxetine 40 mg daily for severe depression symptoms. Therefore, secondary RBD (to fluoxetine) was suspected due to the start of symptoms with the start of fluoxetine therapy. Fluoxetine had fully controlled his depression symptoms during 19 years of continuous therapy. Two options were considered to treat the patient's RBD: addition of clonazepam, or withdrawal of fluoxetine with careful monitoring. The patient chose the latter option, and weaning was performed with close outpatient monitoring of his depressive symptoms. His RBD symptoms resolved around 6 weeks after discontinuation of fluoxetine, and a repeat polysomnogram was performed 3 months later. A representative REM sleep epoch is shown in Figure 2, showing a restoration of appropriate atonia in REM. The patient's clinical course demonstrates a more rapid response to withdrawal of fluoxetine compared to the original case report of this association, a major difference between the cases being patient age.2 There was a recurrence of depressive symptoms four months after discontinuing fluoxetine, and the patient was treated with bupropion, a dopaminergic/noradrenergic antidepressant without any reported tendency to induce or aggravate RBD.3 Bupropion therapy did not result in recurrence of RBD symptoms. Figure 2 0-second epoch tracing of polysomnography recorded during REM sleep shows normal reduction of EMG tone during REM sleep 3 months after fluoxetine discontinuation. LEOG = Left electrooculogram, REOG = Right electrooculogram, FP1A2 – O2A2 = Electroencephalogram ... The possibility of obstructive sleep apnea (OSA) triggering the RBD symptoms was entertained.4 Given the mild nature of the OSA, the polysomnographic findings, and the clinical response to fluoxetine withdrawal, this was not considered a major factor in this case. The patient's OSA was treated with positional therapy, and he and his wife were able to return to sleeping in the same bed with improvement in both his OSA and RBD symptoms.

The Prevalence and Natural History of Complex Sleep Apnea

Central sleep apnea (CSA) may occasionally occur in patients with obstructive sleep apnea during titration with a continuous positive airway pressure (CPAP) device. To determine the prevalence and the natural history of CPAP-emergent CSA. This is a retrospective study of 1286 patients with a diagnosis of OSAwho underwent titration with a positive airway device during a 1-year period. Patients were seen in consultation and underwent full-night attended polysomnography followed by full-night attended CPAP titration. Four weeks after CPAP therapy, patients returned to the clinic for follow-up, and objective adherence to CPAP was recorded. In patients who had CSA on CPAP, a second full-night attended CPAP titration was recommended. Eighty-four of the 1286 patients developed a central apnea index (CAI) of 5 or greater per hour while on CPAP. The incidence of CSA varied from 3% to 10% monthly, with an overall incidence of 6.5%. Forty-two of the 84 patients returned for a second CPAP titration. In 33 patients, CSA was eliminated. In each of the remaining 9 patients, the CAI remained at 5 or greater per hour, with an average of 13 per hour. These patients characteristically had the most severe OSA, and 5 had a CAI of 5 or more per hour at baseline. Two of the 9 patients were on opioids In this large retrospective study of 1286 patients with a diagnosis of OSA, 6.5% had CPAP-emergent or persistent CSA. However, CPAP-emergent CSA was generally transitory and was eliminated within 8 weeks after CPAP therapy. The prevalence of CPAP-persistent CSA was about 1.5%. Severity of OSA, a CAI of 5 or greater per hour, and use of opioids were potential risk factors.

More Treatment Options For Obstructive Sleep Apnea

Source: Guilleminault C, Quo S, Huynh NT, Li K. Orthodontic expansion treatment and adenotonsillectomy in the treatment of obstructive sleep apnea in prepubertal children. Sleep. 2008;31(7):953–958.Researchers from Stanford University and the University of California San Francisco School of Dentistry evaluated the efficacy of sequential rapid maxillary expansion (RME) using orthodontic treatment and adenotonsillectomy (T&A) in prepubertal children with obstructive sleep apnea (OSA).Patients with polysomnogram (PSG)-proven moderate OSA and a recommendation by a multidisciplinary team that both T&A and RME were needed were enrolled.Study patients were randomized to either T&A or RME as initial treatment; the efficacy of the treatments was assessed by parental reports of symptoms and repeat PSG three and six months post-intervention. If the results of the polysomnogram were abnormal or the child was still symptomatic after initial treatment, the second stage of treatment was recommended.Over a 2.5-year period, 32 children (16 boys) with a mean age of 6.5 years (range 4.7–9) were enrolled. All patients had some degree of tonsillar enlargement and high and narrow hard palates with crowded dentition. At the completion of the initial treatment each of the 32 patients improved, but the results of follow-up PSG were not statistically significant between those in the T&A and orthodontic expansion groups. Two patients assigned to the RME group had marked improvement in symptoms and did not undergo T&A. The remaining 30 patients underwent the second procedure. This led to a resolution of symptoms with confirmatory normal PSGs in 28 of the 30 children. Final PSG results were significantly improved over both entry PSG and PSG obtained after the first stage of treatment.Although improvement was seen with one or the other type of treatment in all 32 patients, 87.5% (28/32) required both T&A and RME for complete resolution of OSA. Tonsillar size was not helpful in predicting outcome. Most patients in this select population required both surgical and orthodontic intervention, regardless of the order in which they were performed, for complete resolution of symptoms.Dr. Dubik has disclosed no financial relationship relevant to this commentary. This commentary does not contain a discussion of an unapproved/investigative use of a commercial product/device.RME is a dentofacial procedure used to widen the maxilla and the maxillary dental arch and improve the nasal airway.1 The beneficial medical impact of orthodontic expansion is increasingly suggested in the sleep medicine literature2 but it is not “on the radar” in mainstream pediatrics or dentistry. Although the number of patients in this study is small and the authors do not define or grade “crowded teeth” and “high arched palate,” this article reminds us that there is more to the teeth and the jaw than cosmetics and malocclusion.Doctors and dentists often do not fully appreciate the effects of the maxilla and the mandible on the airway. Treatment with orthodontics is too often not considered in pediatric sleep medicine. And by the time it is, the growth plates may be gone and it’s too late. It would appear that for many children T&A is not enough, and that perhaps most children with OSA need both surgery and orthodontic intervention if complete resolution of symptoms is to be achieved. Indeed, some experts believe that orthodontics may have a more lasting impact on OSA in the long term.And that brings us to the insurance issue. There is no question that RME is an orthodontic procedure, but it is being performed for a medical indication and should be covered by medical insurance. This is not an elective aesthetic procedure; it is a medically indicated intervention. It seems very reasonable that if a child’s sleep-disordered breathing symptoms do not resolve with a T&A, orthodontic expansion should be considered and should be covered as a medical procedure equivalent to the preceding T&A. Considering the difficulty that children with OSA have accepting continuous positive airway pressure (CPAP) therapy, including RME as an early treatment option seems all the more reasonable. Guilleminaut, et al, are to be commended for ensuring that orthodontic expansion of the palate is considered among the therapeutic options by those who treat children with OSA or refer them for treatment.

Effect of Surgical Weight Loss on Sleep Architecture in Adolescents with Severe Obesity

Sleep duration and sleep fragmentation have been proposed to play a role in the development and progression of obesity-associated morbidity. Weight loss results in resolution of obesity-associated morbidity. Our aim was to determine the effect of weight loss on sleep architecture in adolescents with severe obesity. Retrospective analysis of polysomnograhic data from all adolescents who underwent overnight sleep study before and after weight loss surgery was performed. Polysomnographic variables of sleep architecture after weight loss were compared to baseline by paired Student's t test (normally distributed data) or Wilcoxon test (variables not meeting normality criteria). The mean (+/-SEM) age of 19 subjects meeting inclusion criteria was 16.5 +/- 0.35 years, mean body mass index was 60.3 +/- 2.11 kg/m2, and 66% were female. Obstructive sleep apnea was present in 14 subjects (74%). The average interval between the baseline and repeat polysomnograms was 0.91 +/- 0.16 years, and average weight loss was 66.4 +/- 8.8 kg. Surgical weight loss resulted in increased sleep efficiency (80.2% vs 73.1%, p = 0.01), reduced time in stage 1 sleep (3.0% vs 6.0%, p = 0.02), and reduced arousal index (7.6 +/- 0.6/h vs 11.3 +/- 1.2, p = 0.01). Our data demonstrate a marked improvement in sleep efficiency and sleep fragmentation with surgical weight loss. Given the emerging evidence that surgical weight loss results in resolution of obesity-associated psychosocial, metabolic, and cardiovascular morbidity, these results suggest that correction of sleep fragmentation could be an important but as yet underappreciated factor influencing changes in these other major comorbidities of obesity.