bluuebunny/crossref_metadata_2025 huggingface dataset:
0914 Perception of Sleep Medicine (SM) Training among Internal Medicine (IM) Trainees in Puerto Rico
Authors: Irizarry Juan , Rivera-Diaz Alba , Padilla-Diaz Miriam , Fernandez-Hawa Patricia , Jimenez-Davila Lissette , Rodriguez-Cintron William
Published: May 2023

Introduction

Awareness of the importance of SM among trainees at our IM Training Program (IMTP) has not been documented, despite the high prevalence of Sleep Disordered Breathing (SDB) within our clinical population. Recently, the Indiana University Sleep Questionnaire (IUSQ) was administered to quantify the perception of SM in IM residents.

Methods

We administered the IUSQ questionnaire through an anonymous computer survey sample of IM residents at our ACGME accredited IMTP.

Results

A total of 52/61 (85%) residents from the Veterans Administration Caribbean Health Care System (VACHS) IMTP participated. There were 18 PGY-1, 16 PGY-2, 15 PGY-3 and 3 PGY-4 residents whom the majority strongly agreed or agreed that the burden of SDB was large in the general population (63% & 35%), that it is important for their training (48% & 46%) and for their general practice (54% & 38%). They were mostly neutral or disagreed about getting adequate training in SDB during residency (38% & 40%). The majority strongly agreed or agreed that sleep specialist is needed for managing SDB (27% & 48%) and 9.6% disagreed with this. Many do not have interest in pursuing a sleep medicine fellowship answering neutral, disagree and strongly disagree (23%, 38% &23%). The majority strongly agree and agree that a sleep fellowship is important in treating sleep disorders (48% & 33%). The residents are mostly neutral or disagree when it comes to their confidence in treating sleep apnea (40% & 48%) and treating other sleep disorders (46% & 38%).

Conclusion

Despite the high prevalence of SDB in the population they manage, residents do not feel they are getting enough training in the management of sleep disorders. They acknowledge the importance of SM education for the latter purpose, but the majority does not want to pursue formal subspecialty training in SM. In addition, they do not feel comfortable managing SDB and other sleep disorders. Interestingly this mismatch between burden of disease and the need for disease management proficiency is similar to one found previously in a different resident population to which the same IUSQ was administered. An educational approach toward residents may furtherly change these perceptions.

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0918 Elevated stress during the first-year of medical school negatively impacts sleep quality
Authors: Fisher Fielding , Marquardt Matthew , Grandner Michael , Khan Meena
Published: May 2023

Introduction

Poor sleep health is widespread within healthcare and is notoriously neglected during residency; however, little is known about sleep quality during the pre-clinical years of medical school. Stress contributes to poor sleep quality and is commonplace in medical education. This study sought to understand how stress impacts sleep among first-year medical students.

Methods

A one-time, 130-item survey was distributed to the first-year medical school class three months into the first semester. Quantitative and open-text questions sought to understand student demographics, mental health, sleep quality, and sleep-related behaviors, beliefs, and knowledge. Highly prevalent anxiety was defined as feeling nervous/anxious/on edge for more than half of the days during the previous two weeks. Odds ratios (OR) include 95% confidence intervals and were determined using binary logistic regression. Statistical significance was considered a p-value ≤0.05.

Results

138 of the 209 students (66%) in the first-year medical school class completed the survey. Demographics mirrored those of the whole class: 46% male, 54% female, and average age 23.5. Short sleep was prevalent with a mean (standard deviation) sleep time of 6.85 (0.86) hours. Additionally, more than three-quarters reported suboptimal sleep: 56% rated quality as average, and 21% rated quality as poor/very poor. 33% of respondents reported difficulty falling asleep at least once per week. A lack of restorative sleep was also common: 38% felt that sleep was rarely or never refreshing. Over half of respondents (51%) attributed poor sleep to either stress or school, and 29% of students reported elevated or highly prevalent anxiety. Elevated anxiety predicted suboptimal sleep quality (OR 14.67, 1.70-126.76, p=0.015) and a lack of restorative sleep (OR 6.00, 1.52-23.71, p=0.011). Similarly, suboptimal sleep quality was associated with highly prevalent anxiety (OR 44.0, 4.62-418.93, p< 0.001) and difficulty falling asleep (OR 4.14, 1.76-9.75, p=0.001).

Conclusion

Poor sleep and stress are both prevalent during the first year of medical school, with students directly linking stress from their academic workload to impaired sleep quality and insufficient sleep duration. Due to the pervasiveness of stress within medical education, future interventions should help students improve their sleep during both low and high stress time periods.

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0921 Insomnia Symptoms and Environmental Disruptors: A preliminary evaluation of Veterans within a VA subacute rehabilitation unit
Authors: Boyle Julia , Boeve Angelica , Moye Jennifer , Masse Soraya , Driver Jane , Ruopp Marcus , O'Malley Kelly
Published: May 2023

Introduction

Insomnia symptoms during rehabilitation admission are associated with slower rehabilitation progression, increased healthcare utilization, worse functional recovery one-month post discharge, and decreased survival rates one-year post discharge. Insomnia symptoms are complex, multifactorial (e.g., insomnia severity, sleep need, daytime sleepiness), and are often unintentionally worsened by rehabilitation admission due to environmental factors (e.g., light, noise) and behavioral factors (e.g., increased time in bed). The aim of this clinical innovation project was to conduct a preliminary evaluation of Veterans’ sleep at admission and discharge in a VA subacute rehabilitation unit for the purposes of informing clinical interventions.

Methods

Veterans admitted to the VA subacute rehabilitation unit between March and August 2022 (N=67) were approached within 7 days of admission to complete a brief evaluation (Insomnia Severity Index [ISI], Sleep Need Questionnaire [SNQ], Epworth Sleepiness Scale [ESS]). Veterans also answered questions regarding environmental sleep disruptors. Following the evaluation, Veterans were offered sleep resources and behavioral treatment options. Veterans repeated the evaluation within 7 days of discharge and were offered outpatient referrals for their sleep difficulties. Pearson correlations were used to determine associations between length of stay (LOS), ISI, SNQ, and ESS scores at admission and discharge.

Results

33 of 67 (49.2%) Veterans (Mage=73.3±8.8; 96.9% male; 78.8% white) completed admission and discharge evaluations. Over 80% of Veterans reported at least 1 environmental disruptor at admission and discharge. The number of environmental disruptors at discharge was correlated with ISI (r =.549, p=.001) and SNQ (r =.475, p=.005) scores at discharge. LOS was correlated with ESS scores at discharge (r =.524, p=.002).

Conclusion

This preliminary evaluation demonstrated relationships between higher numbers of environmental disruptors and greater insomnia severity and sleep need at discharge from a VA subacute rehabilitation unit. Additionally, longer durations of admission were associated with higher levels of daytime sleepiness at discharge. Future evaluations should include larger sample sizes to determine causality between these variables in Veteran populations. Given the impact of insomnia symptoms on rehabilitation progression, future evaluations should also consider physical function and sleep variables post-discharge.

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0939 Obstetrician-Gynecologists’ knowledge and practice patterns regarding sleep disorders in pregnant patients
Authors: Raglan Greta , Swanson Leslie , Schulkin Jay
Published: May 2023

Introduction

Prenatal sleep quality is an important factor in pregnancy and delivery outcomes and prenatal sleep disorders have been linked to a variety of negative maternal/fetal outcomes. At the same time, pregnancy itself is a risk factor for the development of several sleep disorders including insomnia, sleep apnea, and restless legs syndrome. Obstetrician-gynecologists (ob-gyns) are well-positioned to provide access to resources or treatment for sleep disorders early in the presentation of symptoms, and thus could contribute to overall sleep health and positive birth outcomes.

Methods

A 104-question survey was developed to assess ob-gyns’ knowledge and treatment patterns as they relate to screening and treating sleep disorders among pregnant patients. It was distributed to randomly selected physicians registered with the Pregnancy-Related Care Research Network (PRCRN; N= 220).

Results

Response rate was 46%. Of those surveyed, 89.2% reported that they never screen for sleep disorders, or do so only when their patients report symptoms. When asked about various risks associated with maternal sleep disorders, providers recognized, on average, 60.9% these associations. Roughly half of providers did not recognize associations between maternal sleep disorders and gestational diabetes (49%), maternal mortality (54.9%), premature delivery (51%), or low infant birth weight (50%). Only 18.6% recognized the association between sleep disorders and cesarean delivery. While 99.0% of respondents endorsed that sleep quality is important to wellness, and 90.2% reported that it is an important issue for their patients, only 14.7% and 11.8% reported confidence in their own ability to diagnose and treat sleep disorders, respectively.

Conclusion

There are significant opportunities for educating ob-gyns about sleep disorders and their impact on perinatal outcomes. Additional resources aimed at increasing screening of pregnant patients during perinatal visits could increase early interventions and improve maternal/fetal outcomes.

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0942 Sleep duration benefits for Spanish-speaking Hispanic/Latine respondents of the 2020 BRFSS
Authors: Valencia Dora , Mendoza Javier , Esquivel Denise Rodriguez , Killgore William D S , Armenta M Denisse , Grandner Michael
Published: May 2023

Introduction

Habitual sleep duration is associated with many aspects of health. Previous studies show that some groups of Hispanic/Latine Americans are somewhat protected from sleep disparities seen in other groups, but also that this may depend on acculturation. This study expanded this finding to nationally-representative data and explored some potential pathways that may link acculturation and sleep in Hispanics/Latinos.

Methods

Data from the 2020 Behavioral Risk Factor Surveillance System (BRFSS) were used (N=309,548 provided complete data). Habitual sleep duration was self-reported in hours and converted to minutes for analysis. Race/ethnicity was self-reported as Non-Hispanic White, Black/African American, Hispanic/Latine, Asian/Pacific-Islander, American Indian/Alaskan Native, and Multiracial/Other. The Hispanic/Latine group was further divided into those who chose to complete the survey in English versus Spanish. Covariates included age, sex, education level, household income, employment status, overall health, and mental health. Linear regression examined sleep duration (minutes) as the outcome, with race/ethnicity as the predictor. Race/ethnicity-by-covariate interactions were explored.

Results

Compared to non-Hispanic Whites, Blacks/African Americans reported 7.4 minutes less sleep (p< 0.0005), English-speaking Hispanics/Latine reported 4 fewer minutes (p=0.038), Spanish-speaking Hispanic/Latine reported 16.9 more minutes (p< 0.0005), Asians/Pacific Islanders reported 4.7 fewer minutes (p=0.042), no difference was seen for American Indians/Alaskan Natives, and Multiracial/Others reported 9.5 fewer minutes (p< 0.0005). Significant interactions were seen for age, education, income, employment, health, and mental health (all p< 0.05). Sleep duration benefits of speaking Spanish were evident especially in younger groups (under age 50, peaking age 30), those with more education, those with higher income, those in better health, those with fewer mental health problems, and those who were employed or were students.

Conclusion

Spanish-speaking Hispanics/Latine Americans were the only race/ethnicity group to report more sleep than non-Hispanic White adults and showed markedly different relationships versus when including English-speaking Hispanic/Latine adults. This advantage was differentially experienced, though, and depended on age, socioeconomics, and health status. Also, the BRFSS did not ask about primary household language nor English fluency. Future research is needed to explicitly explore acculturation-related explanations and develop linguistically- and culturally-targeted interventions for sleep health, especially for addressing apparent generational and socioeconomic differences.

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0960 Use of an Innerspring Mattress Improves Deep Sleep and WASO
Authors: Rus Holly , Danoff-Burg Sharon , Burke Colin , Saba Shaddy , Weaver Morgan , Carmon Kiara , Ledesma Duvia Lara , Upadhyaya Devanshi
Published: May 2023

Introduction

Previous research has shown that comfort, including comfort of the sleep surface, is essential to sleep quality. In addition to comfort, the structure and firmness of a mattress may also play a role when it comes to sleep quality. More empirical research is needed to establish the impact of particular mattresses on both objective and self-reported measures of sleep quality. This study compared sleep on an innerspring mattress to participants’ prior sleep on their original mattress.

Methods

Healthy adults (68% female, ages 23-69) participated in a 10-week field study, using a pre-post intervention design. During the 4-week baseline period, participants used their regular mattress at home. During the 4-week intervention period, which occurred following a 2-week adjustment period, they used a Sleepy’s by Sealy mattress at home. Sleep was measured objectively using SleepScore Max every night and by daily and pre-post self-report. Multilevel regression and paired t-tests were used to test for statistical significance.

Results

There were over 757 nights of tracked sleep across 18 participants. Objective sleep measurements showed decreased WASO, both in duration (-13 minutes, p=.024) and proportion of the night (p=.025). Also, there was an increase in deep sleep (+12 minutes, p=040). Self-report measures revealed that the innerspring mattress felt more comfortable (p<.001). Improvements also were found for a variety of perceived sleep outcomes, including time to fall asleep, waking less often, spending less time awake, longer sleep duration, better sleep quality, and feeling more rested in the morning (ps<.05).

Conclusion

Objectively-measured sleep and self-reported sleep improved when using the innerspring mattress compared to healthy adults’ original mattresses. Furthermore, qualitative and quantitative self-report results suggested that the intervention was perceived as comfortable.

Support (if any)

Mattress Firm INC
0961 Applying Fractal Analysis Towards the Understanding of Long-COVID-Induced Insomnia
Authors: Emerling Max , Rouen Alexandre , Taieb Jonathan , Salmon Dominique , Léger Damien
Published: May 2023

Introduction

As part of the recent international COVID Sleep Study-II, long COVID syndrome was found to be associated with chronic insomnia. However, an article submitted for publication out of the Hôpital Hôtel-Dieu found no significant difference between long-COVID-related insomnia and standard insomnia with respect to routine polysomnographic (PSG) parameters. In light of these findings, we seek to better understand the difference between long COVID insomnia and standard chronic insomnia by conducting a fractal analysis of the raw PSG signals of our subjects.

Methods

15 long COVID patients with complaints of chronic insomnia were included as subjects, as well as 34 matched controls without long COVID who had similar complaints of chronic insomnia. All participants underwent one night of polysomnographic recording. We perform our analysis on movements of the abdomen (as an approximation of deep breathing quality) and on EEG waves, considering that shortness of breath and cognitive dysfunction are commonly associated with long COVID. Building upon an existing repertoire of medical research using the fractal dimension (FD) of patients’ signal data to differentiate pathologic and physiologic processes, we make use of Detrended Fluctuation Analysis (DFA) to calculate the FD of the abdomen (FDA) and of an aggregated EEG wave (FDE) for each patient.

Results

We find a decrease in the FDA (p=0.036) and increase in the FDE (p=0.012) of long COVID insomniacs with respect to those of our control group. These results are shown to be significant after applying a Holm-Bonferroni correction for multiple hypotheses.

Conclusion

Despite our small data sample, an in-depth analysis of PSG signals shows significant differences in the fractal nature of long-COVID-related insomnia with respect to standard chronic insomnia. Our results furthermore match our expectations, given that related research outside of the COVID domain indicates a decrease in the FD of muscle movements and an increase in the FD of EEG waves in unhealthy individuals. Further research could employ more participants in order to extend our results to other data channels outside of EEG signals and the abdomen.

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0969 Severe persistent specific sleep disorders in post-acute sequelae of SARS-CoV-2
Authors: Czuma Richard , Thomas Robert , Maley Jason , Gott Danielle
Published: May 2023

Introduction

Viral infections have long been associated with sleep disorders, especially hypersomnia following the acute phase The Covid-19 epidemic has resulted in a post-viral syndrome called post-acute sequelae of SARS-CoV-2 (PASC), or Long-Covid. Sleep has been reported to be severely disturbed during acute Covid-19 infection, and is a major feature of PASC. Much of the subjective data is based on questionnaires, with a paucity of objective sleep evaluations including polysomnography. We share our early experience with diagnosis and management of sleep disorders in PASC.

Methods

PASC patients are seen in a dedicated Long-Covid clinic at the Beth Israel Deaconess Medical Center for a comprehensive evaluation. The patients presented with new onset persistent sleep impairments shortly following acute COVID-19 infection, without pre-existing sleep impairments, in association with a clinical diagnosis of long covid. Sleep symptoms (excessive sleepiness, hypersomnia/long sleep, and insomnia) are identified and those who consider these a key symptom are referred to the Covid Sleep Clinic, for formal sleep evaluation by RJT. Diagnostic evaluations include polysomnography (extended for hypersomnia diagnosis), Multiple Sleep Latency Test for excessive daytime sleepiness, and home sleep apnea test for suspected sleep apnea. Sleep logs and sleep quality using a Ring oximeter-based SleepImage system complements evaluation of sleep-wake instability. Standard management is offered based on the ensuing diagnosis.

Results

Twenty-six patients (age range 22 to 58 years, 16 females) have been evaluated in this sleep pathology enriched pathway. Specific new sleep disorders diagnosis so far have included narcolepsy without cataplexy (3), hypersomnia with long sleep (3), hypersomnia without long sleep (3), rapid eye movement sleep disorder (2), persistent insomnia (3), a sleep-wake instability syndrome we call cyclical insomnia (2), and sleep apnea (7). Treatment has included CPAP, stimulants, modafinil, and sodium oxybate. Clinical responses to treatment have so far been as expected. One patient with cyclical insomnia has had a marked subjective and objective improvement with low dose (250 mg) sodium valproate.

Conclusion

PASC/Long-Covid can induce specific sleep syndromes. These include narcolepsy, hypersomnia, persistent insomnia, sleep-wake instability, and REM-behavior disorder.

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0971 Case Report: Sleep-Related Painful Erections Treated with Sodium Oxybate
Authors: Chaudhary Hira , Zeidman Eric , Punjani Nahid , Tashman Yasemin
Published: May 2023

Introduction

Sleep-related painful erection (SRPE) is a rare parasomnia characterized by painful penile erections during rapid eye movement sleep. Normal, painless erections continue to occur while awake. Approximately thirty cases have been reported in the literature. Multiple theories about potential pathophysiology have been proposed including neurologic dysfunction of the ischiocavernosus and bulbocavernosus, correlation with obstructive sleep apnea, and/or autonomic dysfunction. There is no known single effective treatment to date resulting in durable response despite multiple attempts at therapy.

Report of case(s)

A 39-year-old male presented with SRPE. This started in 2018 and occurred in the second half of the night every twenty minutes, contributing to discomfort and poor sleep quality. While awake, his erections were painless. He was prescribed baclofen at doses up to 30mg daily decreasing episode frequency to 1-6 times per night, but disabling somnolence precluded continuation. Pelvic physiotherapy was unhelpful while imipramine and clonazepam provided partial relief. Tadalafil was ineffective and cinitapride was not available in the United States. Oxycodone used for pain after a motor vehicle accident did effectively treat him for five hours per night, so morphine was trialed. This was effective for two weeks, but symptoms returned. In April 2022, he was begun on sodium oxybate. With an initial dose of 2.25 g twice nightly, symptoms were treated for four hours a night. After increasing to 2.75 g twice nightly, the SRPE was effectively treated. As of September 2022, he is taking sodium oxybate 3 g at bedtime, 3 g 3.5 hours later and the final 3 g at the 7th hour, promoting 9.5 hours of sleep without SRPE.

Conclusion

This 39-year-old male who presented with SRPE was trialed on multiple therapies without success, and ultimately successfully treated with an appropriate and titrated dose of sodium oxybate. This may represent a new treatment option, which we hypothesize may act by modulating GABA to result in relaxation of the bulbospongiosus and ischiocavernosus muscles. Further studies should include the study of both the efficacy of this drug for SRPE as well as potential mechanisms of action.

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0983 Hand Dominance in Sexsomnia: A Clue to Pathophysiology?
Authors: Badami Varun , Avidan Alon , Schenck Carlos
Published: May 2023

Introduction

Sexsomnia is a variant of confusional arousal, a NREM parasomnia (NREMP), typically consisting of sexual behaviors manifesting upon partial arousal from deep NREM sleep; however, its pathophysiology is not well understood, and literature is limited. We present a series of patients with non-dominant hand sexsomnia behaviors that may reveal important new clues about the neurophysiologic mechanisms of sexsomnias.

Report of case(s)

Case 1: 22 y/o right-handed (RH) female, no history of NREMP, or snoring. Presenting complaint (PC): 8-year history of sleep masturbatory behavior (MB) including partial awakening from MB with bloody fingers (left hand) during her menstrual period in the setting of delayed sleep phase disorder. Case 2: 30 y/o ambidextrous male with a history of NREMP. PC: MB involving the left hand, observed by bed partner (BP) in the setting of weight gain and apneas. PSG: AHI 17/h and PLMI 23/h. Case 3: 33 y/o RH female with a history of NREMP. PC: left-handed sleep MB observed by BP in the setting of multiple sclerosis with C2-3 and C4 right posterolateral demyelinating lesions on C-spine MRI. Case 4: A 44 y/o RH male with OCD, depression, and no history of NREMP. PC: bilateral MB and dream enactment behaviors observed by BP, in the setting of sertraline. Events resolved upon switching to quetiapine. Case 5: A 59- y/o RH female with multiple system atrophy (MSA) and RBD, no previous NREMP. PC: sleep MB, noted by BP involving the left hand in the setting of worsening dysautonomia. Dream enactment occurred bilaterally. PSG: AHI 34/h, PLMI: 70/h and REM sleep without atonia. Spells improved with melatonin and PAP.

Conclusion

This case series highlights a pattern of adult sleep MB involving the non-dominant (left) hand in 4/5 cases and the left hand in one ambidextrous patient. There was no evidence of gender predilection. We speculate that sexsomnia originates from central pattern generators in the brainstem and spinal cord, as opposed to the cerebral cortex (which would likely involve the dominant hand). The lack of involvement of cerebral motor control is further substantiated by amnesia for sexsomnia events.

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0985 Oxybate induced mania
Authors: Barber Cameron , Czuma Richard , Campbell Kimberly , Thomas Robert
Published: May 2023

Introduction

Hypersomnia sleep disorders are characterized by overwhelming daytime sleepiness. Treatment options include sodium oxybate the sodium salt of γ-hydroxybutyric acid (GHB). Oxybate, an endogenous metabolite of GABA, acts through GABAB noradrenergic and dopaminergic neurons, as well as at thalamocortical neurons. < 1% may experience suicidal ideations or psychosis.

Report of case(s)

A 17-year-old female with obesity, spina bifida occulta follows with sleep for idiopathic hypersomnia with long sleep (TST 12.6 hours on polysomnography) and a delayed circadian rhythm. Her typical sleep zone is 11:00PM to 10:00AM, but she can easily sleep until 2:00PM. She started sodium oxybate to minimize her sleep burden and improve daytime wakefulness. She initially developed insomnia with concern for mania and migraines with photophobia and phonophobia. After 26 days, oxybate was discontinued. Her migraines persisted daily. Her sleep schedule and mood returned to baseline within 17 days thereafter. A 48-year-old female with anxiety/depression, PTSD has follows with sleep since age 42 for unspecified hypersomnia, either narcolepsy type 1 with long sleep versus idiopathic hypersomnia. Initial polysomnography/MSLT on fluoxetine revealed AHI 1.6, with a mean sleep latency of 6.4 minutes without SOREMPs. Her typical sleep zone was 8:00PM to 9:00AM. She was started on sodium oxybate for her cataplexy and daytime sleepiness. She had remarkable improvement in her symptoms after 4 days. After 35 days on 9 grams nightly she developed anxiety, extracorporeal sensations, and paranoia. The dose was decreased to 7.5 grams nightly. By 7 months of therapy her paranoia progressed to the point where she threatened her landlord and was facing legal consequences. Sodium oxybate was discontinued. At 53-years-old she was trial on mixed-salts oxybate. After three months of therapy, she became hypomanic with severe pressured speech despite a euthymic mood. She reported going without sleep entirely for 3-4 days at a time using this oxybate. Mixed-salts oxybate was discontinued and 36 days later with in addition to quetiapine, her hypomania was nearly resolved and she was able to get 8-9 hours of restful sleep.

Conclusion

Hypomania appears to be a rare but serious side effect of oxybate in long-sleepers. Discontinuing oxybate may not be sufficient therapy.

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0991 Dual Therapy to Treat Circadian Rhythm Disturbance in Unique Genetic Mutation of Smith-Magenis Syndrome
Authors: Cocozza Victoria , Kim David , Long Benjamin , Hansen Shana
Published: May 2023

Introduction

Smith-Magenis syndrome (SMS) is a rare disorder associated with several genetic mutations. Patients with SMS experience circadian rhythm sleep-wake disorders, which may lead to significant behavioral concerns. A proposed etiology of sleep disturbances is diurnal melatonin secretion. Treatment approaches aim at blocking the abnormal morning secretion of melatonin and providing exogenous melatonin in the evening.

Report of case(s)

We present a patient with SMS having persistent sleep-wake disturbance despite dual therapy with beta-1 adrenergic antagonist in the morning and over-the-counter melatonin at night. Our patient switched to therapy with beta-1 adrenergic antagonist in the mornings and melatonin receptor agonist, tasimelteon, at night with significant improvement. In addition, our patient has mutations in JAKMIP1 and ZBTB18 genes. ZBTB18 mutation is a novel mutation in the setting of clinical features of SMS.

Conclusion

To the authors’ knowledge, this is the first case report of successful combination therapy to optimize treatment of sleep-wake disturbances in patients with SMS.

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0995 Sleep-related Hypoventilation in a 62-Year-Old Female with a Rare Genetic Neuromuscular Disorder
Authors: Gadodia Ritika , Dorsch Jennifer , Mora Jorge
Published: May 2023

Introduction

Central Core Myopathy (CCM) is a rare genetic neuromuscular disorder involving a mutation in the ‘Ryanodine Receptor’ (RyR1) gene, also known as the gatekeeper of calcium within the muscle cells. CCM typically manifests as slowly progressive proximal muscle weakness Currently, there is limited data regarding the management of nocturnal hypoventilation in adults with neuromuscular disorders.

Report of case(s)

We present a 62-year-old woman with CCM, as identified via RyR1 mutation (c14818G>A) on genetic testing, as well as muscle biopsy. This genotype is associated with < 10% respiratory muscle involvement. She presented to the sleep clinic with complaints of morning headaches, non-restorative sleep, and daytime sleepiness. Her polysomnogram (PSG) showed an overall Apnea-Hypopnea-Index in the normal range. However, there was a progressive increase in transcutaneous carbon dioxide levels (PtcCO2), reaching a peak of 53 mmHg during rapid eye movement (REM) sleep from 38 mmHg during wakefulness, consistent with sleep-related neuromuscular hypoventilation. On further evaluation, this patient had no evidence of neuromuscular restrictive lung physiology or overt diaphragmatic dysfunction, as suggested by < 20% variation in the supine forced vital capacity. Moreover, her negative inspiratory force (NIF) value was not less than negative 40. However, in the setting of her sleep-related symptoms and evidence of nocturnal hypoventilation, our team decided to trial non-invasive positive pressure ventilation (NIPPV). Since we had concerns about respiratory alkalosis in NREM sleep in the setting of normal body mass index, normal pulmonary function tests, and hypoventilation limited to REM sleep, the patient was sent for in-lab titration of pressures with measurement of PtcCO2 to appropriately recommend NIPPV settings. The study demonstrated bi-level positive airway pressure settings of IPAP 7 cmH20, EPAP 5 cmH20, and a backup rate of 12/min resulting in sustained eucarbia and steady PtcCO2 levels during both REM and NREM sleep.

Conclusion

This case highlights the importance of evaluation for nocturnal hypoventilation in symptomatic patients with rare neuromuscular disorders and the potential for symptomatic improvement with treatment with NIPPV. Moreover, the case demonstrates the importance of in-lab titration of pressure settings for NIPPV when hypoventilation is limited to REM sleep, due to concern for iatrogenic hyperventilation and respiratory alkalosis.

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0998 Duloxetine as a Possible Effective Treatment of Klein-Levin Syndrome Case Report
Authors: Hall Samuel , Ibrahim Sally
Published: May 2023

Introduction

Kleine Levin Syndrome (KLS), or recurrent hypersomnia, is a rare sleep disorder characterized by recurrent episodes of severe hyper-somnolence lasting several days to several weeks. Because the etiology is unknown, treatment is mostly supportive. Some pharmacologic agents have been attempted to treat symptoms during episodes with limited success.

Report of case(s)

Patient is a 17 year-old with a history attention-deficit hyperactivity disorder and generalized anxiety disorder who presented to the clinic due to recurrent episodes of severe hypersomnolence associated with derealization and cognitive dysfunction during all episodes, anxiety of being alone, and other symptoms in some episodes such as altered taste affecting appetite, and binge eating. The patient was diagnosed with KLS and tracked episodes which lasted 11-12 days (average) with recurrence every 10-17 days. Labs, 72 hour EEG, MRI and MRA were largely unremarkable. The family declined trial of Lithium. Pharmacogenetic testing showed significant gene-drug interaction with duloxetine requiring higher drug dosage for effects. After psychiatry started duloxetine, the patient experienced a 7 month period without episodes. Then episodes recurred for unclear reason while on duloxetine but with slightly less duration (6-12 days) and shorter time between episodes. Duloxetine was discontinued due to lack of mood effects. Clarithromycin was trialed next for use during episodes without changes. Episodes were tracked for several months with some predictability. Duloxetine was restarted a second time after several months, and the patient showed mild improvement in event duration down to 8-11 days for a few months. Time between episodes also shortened to 8-10 days. Anxiety was only slightly reduced between and during the episodes, though they still continued to struggle with derealization and ability to focus during episodes that effected school and work functioning. Interestingly, they also started to have more insomnia between episodes while on duloxetine.

Conclusion

KLS is a rare disorder with limited data on effective treatments. To date, this is the first report of a prolonged brief KLS remission with duloxetine. Norepinephrine and serotonin modulation may limit episode duration. More research is needed to determine whether duloxetine can modulate the effects of KLS.

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None
1004 A novel use of adaptive servo ventilation for VNS-induced central sleep apnea in an adolescent with refractory epilepsy
Authors: Jones Brandon , Matarese Christine , Wong-Kisiel Lily , Baughn Julie
Published: May 2023

Introduction

Epilepsy is frequently encountered in the pediatric population and some patients are treated by stimulation of the vagus nerve to reduce their seizure frequency. Vagus nerve stimulation (VNS) may induce central sleep apnea. Adaptive servo ventilation is a known treatment option for central sleep apnea in certain adult patients with heart failure and Cheyne-Stokes respiration but is infrequently utilized in the pediatric population.

Report of case(s)

A 17-year-old male with a history of intractable generalized epilepsy, VNS placement, obstructive sleep apnea (OSA) and central sleep apnea (CSA) presented to our tertiary care center for management of his sleep disordered breathing. Initial management of sleep disordered breathing included CPAP for OSA. After VNS placement, the patient began to have a high residual apnea-hypopnea index on data card download of his CPAP. Polysomnography (PSG) was performed on CPAP and CSA events were noted that correlated with VNS firing. BiPAP was titrated and was unsuccessful in treating the CSA. VNS adjustment did not impact the sleep disordered breathing. Repeat titration with adaptive servo ventilation successfully treated the OSA and CSA. In follow up, the patient is tolerating ASV with improvement in his nocturnal sleep disordered breathing events and his daytime functioning.

Conclusion

Central sleep apnea can be induced by vagus nerve stimulation. This unique case documents successful treatment of vagus nerve stimulation induced central sleep apnea by adaptive servo ventilation in an adolescent patient.

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1008 Excessive daytime sleepiness in myotonic dystrophy treated with modafinil
Authors: Lee Ian , Hekmat Anahid
Published: May 2023

Introduction

Physiology of excessive daytime sleepiness (EDS) in myotonic dystrophy type 1 (DM1) remains incompletely understood and best practice treatment approaches are unclear.

Report of case(s)

We present a 73-year-old man with atrial fibrillation, implantable cardioverter defibrillator for inducible ventricular tachycardia, hypertension, and severe obstructive sleep apnea (OSA) was referred after a recent diagnosis of myotonic dystrophy type 1 (DM1) for EDS. He presented with foot pain, balance issues, and falls. A subdural hemorrhage from a fall prompted thorough evaluation including electromyography. This showed length-dependent polyneuropathy type pattern with features of subacute and chronic denervation change and myotonic discharges in right vastus medialis and bilateral tibialis anterior and gastrocnemius muscles. Myotonic Syndrome Advanced Evaluation revealed with a repeat expansion mutation in DMPK gene (85 and 13 CTG repeats). He was previously diagnosed severe OSA with an AHI of 64 and titrated onto bilevel. He recalled no improvement in symptoms with bilevel. He noted sleeping 10-12 hours per night and feeling rested. However, he takes multiple times per day for hours at a time. When actively working on tasks he feels alert and focused. Epworth sleepiness scale (ESS) was 12. Polysomnogram on bilevel followed by MSLT demonstrated control of OSA and mean sleep onset latency of 8 minutes and 5 sleep onset REM periods. With relatively little impairment and a desire for intermittent improved wakefulness, he was started on modafinil 200mg daily as needed. He reported a robust response to modafinil without the need for additional naps or cardiac complications thus far. Repeat ESS only improved to 10.

Conclusion

This case highlights the need for a precision medicine approach as subpopulations exist even within the specific diagnosis excessive daytime sleepiness in myotonic dystrophy. People with DM1 have been similarly reported with little improvement in ESS but dramatic subjective improvement in symptoms with modafinil. A Cochrane Review and a European Medicines Agency recommendations on modafinil are not supportive of its use in DM1 despite a subpopulation with a clear response. Interestingly, clinical measures do not seem to predict response to modafinil, suggesting unmeasured and, potentially, varied pathologies of excessive daytime sleepiness in DM1.

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1020 Modafinil Induced Psychosis
Authors: Patel Harvey , Patel Harvey
Published: May 2023

Introduction

Modafinil has been used for the treatment of numerous different conditions, including daytime somnolence (EDS). Modafinil is a non-amphetamine central nervous system (CNS) stimulant with wakefulness-promoting properties. There have been few reported cases of psychosis associated with modafinil use, however a large majority of those were seen in patients with a history of primary psychiatric disturbances.

Report of case(s)

We present a case of a 48 year old female coming for evaluation of excessive daytime somnolence. She completed polysomnography and a multi sleep latency test (MSLT) which revealed mildly elevated AHI, but no sleep onset rapid eye movements (SOREMPs). A diagnosis of idiopathic hypersomnia was subsequently made and 200mg of modafinil up to twice daily was prescribed for her. Patient initially tolerated the modafinil well, however she began having delusions and she subsequently required psychiatric evaluation. These delusions primarily consisted of her believing she was under surveillance and had conspiracy theories against her. She also described having auditory hallucinations as well, especially voices telling her about these conspiracy theories. She also demonstrated poor insight into her medical condition as she refused to discontinue modafinil. Upon evaluation, routine labs including complete blood count, chemistry profile and urine drug screens were completed to rule out any secondary causes of psychosis. It was therefore recommended by psychiatry to discontinue modafinil as there was concern that the delusions were occurring secondary to medication use. The patient's psychotic delusions seemed to dissipate after this. A multidisciplinary meeting was held regarding management of patients' excessive daytime sleepiness (EDS) and a decision was made to begin the patient on pitolisant, a histamine receptor antagonist. The patient was able to achieve great benefit with this medication.

Conclusion

The presented case illustrates a patient who began having psychosis after beginning modafinil 200mg twice daily. Psychosis is a much more rare side of modafinil and providers should become aware of it. Therefore, patients who are prescribed modafinil should be carefully followed for psychosis development, even when they are previously healthy and have no history of any primary psychiatric conditions.

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1024 A Challenging Inspire Case: All Roads Do Not Necessarily Lead to Rome
Authors: Call Justin , Punj Mantavya , Werninck Natalia , Colaco Clinton , Arunthari Vichaya , Colaco Brendon
Published: May 2023

Introduction

Hypoglossal nerve stimulation (HNS) for the treatment of obstructive sleep apnea (OSA) in patients who have failed positive airway pressure (PAP) therapy for one reason or the other is now standard of care. Once the device is implanted and activated, voltage to stimulate the nerve has to be titrated over time, ultimately leading to an HNS titration study. There are 4 possible electrode configurations to stimulate different fibers in the nerve that can be employed for treatment. We present a case of a patient who was a challenge with worsening of OSA with Inspire.

Report of case(s)

A 65-year-old woman with hypertension, and severe OSA, intolerant of positive airway pressure or oral appliances was implanted with Inspire. Post activation, there was symptomatic improvement with reduction of overall apnea-hypopnea index (AHI) from 34 per hour of sleep to 6.3 per hour. However, patient slept exclusively on the sides with evidence of significant residual disease in REM sleep. During subsequent weeks, patent reported inability to restrict position and discomfort and anxiety associated with up titration. Both awake endoscopy and advanced overnight HNS titration were pursued. All 4 electrode configurations were tested during both procedures. With the exception of (minus-plus-minus) configuration, there was worsening of sleep disordered breathing and higher residual AHI of 60 per hour on polysomnography and evidence of poor tongue motion and collapsed velopharynx on awake endoscopy. Inspire settings were readjusted to (minus-plus-minus) configuration with instructions for very slow outpatient uptitration and co-management of anxiety and discomfort with anxiolytics and sleep aids.

Conclusion

We make two important conclusions from this case. Firstly, although there are established criteria for patient selection for hypoglossal nerve stimulation therapy, certain other factors that impact success which are not as easily defined include the patients’ expectations, tolerance of discomfort, propensity to anxiety and personality type which impact the ability to successfully titrate voltage with the Inspire device over time. Secondly, as the tongue is a complex muscle, both under titration and over titration may be counterproductive and additionally, electrode configuration may also have varying effects on tongue protrusion and impact success.

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1027 Title: Hypersomnolence following resection of a craniopharyngioma
Authors: Rehman Sofiya , Khan Safia , Carter Gregory , Luong Shan
Published: May 2023

Introduction

Craniopharyngiomas are rare tumors that arise from Rathke’s pouch remnants along the pituitary-hypothalamic axis. The tumor and its treatment can lead to significant neurological and endocrinological complications including alterations in appetite, endocrine insufficiency, circadian rhythms and sleep. We report the case of a woman who developed hypersomnia following resection of a craniopharyngioma

Report of case(s)

This 38-year-old female with neither significant past medical history of sleep, medical, neurological, or psychiatric disorders nor a family history of narcolepsy presented with galactorrhea and amenorrhea. Imaging showed a pituitary tumor. She underwent resection of the tumor with a tissue diagnosis of an adamantinomatous craniopharyngioma. MRI post procedure showed post-surgical changes without new lesions. Diabetes insipidus and panhypopituitarism developed for which appropriate replacement therapy was initiated. She gained approximately one hundred pounds in the weeks post-surgery. Her family described new onset snoring in supine sleeping position and witnessed apneas. She denied hypnagogic hallucinations, cataplexy, or sleep paralysis. She presented to the sleep medicine clinic a few months later for evaluation of persistent hypersomnolence.

Conclusion

The most common sleep disorders post craniopharyngioma resection are sleep-related breathing disorder, hypersomnia, and circadian rhythm disorder. Sleep and appetite are felt to be altered because of direct surgical compression, injury or vascular compromise of structures involved in the control of sleep, wakefulness, and appetite. This can produce hypothalamic dysfunction induced obesity with secondary sleep-related breathing disorder and worsening hypersomnolence. In this woman the temporal relationship between the surgery and the onset of symptoms is most suggestive of this hypothalamic injury, resulting in abnormal regulation of both appetite and sleep. Improvements in weight control and hypersomnia with use of stimulant therapy in the setting of secondary obstructive sleep apnea have been reported and in addition to standard positive airway pressure therapies give hope for the management of these complications of craniopharyngioma resection.

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1029 Inspire Confidence In CPAP
Authors: Rodriguez Nathaniel
Published: May 2023

Introduction

Obstructive sleep apnea (OSA) is defined as the repetitive episodes of complete or partial occlusion of the airway occurring during sleep. OSA is thought to play a role in many different diseases including anxiety, depression, excessive day time sleepiness, and GERD. Treatment of OSA can often be difficult as many patients cannot tolerate CPAP.

Report of case(s)

The patient is a 76-year-old male who was diagnosed with OSA in 1991. He had undergone Uvulopalatopharyngoplasty in 1997 and maxillomandibular advancement in 1999 but continued to have snoring and witnessed apneas and excessive daytime sleepiness (EDS). A repeat PSG in 2019 showed severe sleep apnea with an AHI of 37.3. The patient had been unable to tolerate his CPAP at home due to discomfort from the pressure. He was then referred for evaluation and subsequent placement of a hypoglossal nerve stimulator known as inspire by ENT. On follow-up at the sleep clinic, the patient complained that he was still having daytime sleepiness and snoring despite compliance with INSPIRE. A home study was done to evaluate the effectiveness of INSPIRE and his AHI was found to be 46. INSPIRE was reconfigured and a repeat sleep study was done which showed similar results. A decision was made to try combination therapy with INSPIRE and CPAP at a lower pressure than previously. His original CPAP settings were 12cmH20 and with combo therapy we were able to change this to autoPAP 7-10, which the patient tolerated well and showed good control of his sleep apnea with an AHI of 4.6.

Conclusion

For those with moderate to severe OSA, the gold standard therapy is CPAP, but not every patient can tolerate CPAP therapy. One of the reasons is the CPAP pressure being too high. In 2014 the FDA approved a new treatment option for those with moderate to severe sleep apnea with the hypoglossal nerve stimulator. In our patient, multiple different INSPIRE configurations were attempted but he still had uncontrolled OSA and EDS. We were able to use combination therapy with INSPIRE and CPAP to adequately control his OSA without needing the high pressure that he could not tolerate previously.

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