bluuebunny/crossref_metadata_2025_split huggingface dataset:
0115 How Pre-Sleep Negative Affect and Rumination Impact Sleep Onset
Authors: Strauel Caroline , Krizan Zlatan 
Published: April 2024

Abstract

Introduction
Getting a good night’s sleep is critical to several aspects of daily functioning and health, but many choices may hinder an individual’s onset of sleep. The impact of negative emotion on subsequent sleep has received limited attention and causal claims require strong experimental methodology. One contributor to delayed sleep onset may be rumination, as ruminating individuals’ may negative emotions may persevere longer. The current study aimed to examine the impact of emotions on sleep immediately after experiencing an emotionally disturbing video and the role of individual differences, specifically rumination tendencies.

Methods
Using a within-subjects, at-home design, participants (N=149) encountered two nights of control videos and two nights of emotionally distressing videos immediately before their normal sleep time. Rumination tendencies were measured at baseline using the Rumination-Reflection Questionnaire by Trapnell and Campbell. Mood and negative affect were recorded before and after watching the videos and their subsequent sleep was recorded using actigraphy. Participants self-reported sleep-onset latency (SOL) in minutes.

Results
The manipulation was effective at inducing negative emotions including anger (d = 0.596, p&lt; 0.001), disgust (d = 1.513, p&lt; 0.001), and fear (d = 1.058, p&lt; 0.001). A paired samples t-test showed no meaningful differences on self-reported SOL between control and experimental nights (p=.596). While control nights did have a slightly higher average SOL than experimental, the averages differed by 1.1 minutes. Preliminary analysis revealed no significant correlations of rumination on experimental or control SOL, with rumination correlated to overall SOL 0.05.

Conclusion
Despite effective induction of pre-sleep negative affect, preliminary analyses showed there were no substantive differences between control and experimental conditions in participants’ self-reported SOL (despite adequate statistical power), although the reports may be inaccurate due to self-report biases. Future analyses of actigraphic data will evaluate more objective aspects of sleep and roles of other individual differences.

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N/A
1151 Pharmacological Treatment of Pediatric Insomnia--A Successful Trial of Doxepin
Authors: Tu Xinhang , Matarese Christine , Lloyd Robin , Sorensen Channing , Schmidt Scott , Baughn Julie 
Published: April 2024

Abstract

Introduction
Insomnia is a common sleep disorder in pediatric patients with neurodevelopmental disabilities. There are no FDA approved medications for insomnia in the pediatric population, and treatment can be challenging. We present a case of successful treatment of sleep maintenance insomnia in a 15-year-old male with doxepin with supportive actigraphy data.

Report of case(s)
A 15-year-old male with a past history pertinent for a genetic neurodevelopmental disorder, autism spectrum disorder (ASD) and attention deficit hyperactivity disorder (ADHD) was evaluated in Pediatric Sleep Medicine Clinic for long standing difficulties with sleep maintenance. He experienced frequent, prolonged nocturnal awakenings and early morning awakenings. Previous polysomnogram was negative for sleep disordered breathing. He previously trialed clonidine, guanfacine, trazodone, melatonin, and mirtazapine without significant benefit. Baseline actigraphy demonstrated frequent and prolonged nocturnal awakenings and early morning awakenings. He was initiated on doxepin 3 mg at bedtime, and it was subsequently increased to 6 mg at bedtime. Parental report and repeat actigraphy 4 months later (on doxepin 6 mg) demonstrated improvement in nocturnal awakenings and early morning awakenings.

Conclusion
Up to now, there are no FDA approved medications for the treatment of insomnia in the pediatric population. A prior study of patients with ASD and ADHD demonstrated the use of doxepin provided clinical improvement in sleep onset and maintenance insomnia based on subjective evidence with parental report. In this case, doxepin improved subjective report of insomnia and correlated with objective findings on actigraphy.

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NA
1152 A Journey Through Kleine-Levin Syndrome: A Longitudinal Case Report
Authors: Tu Xinhang , Lipford Melissa 
Published: April 2024

Abstract

Introduction
Kleine-Levin Syndrome (KLS) is a rare disease with intermittent episodes of hypersomnia with behavioral abnormalities. We present a case of KLS with successful symptomatic treatment with modafinil and eventual remission.

Report of case(s)
A 19-year-old healthy male with no prior sleep disorders was referred for evaluation of hypersomnia. He developed leg cellulitis requiring antibiotic therapy. Shortly afterwards, he developed abnormal behaviors --quitting school, estrangement from friends, sleeping 20 hours a day, irritability, and suppressed appetite. He also demonstrated hypersexuality and disinhibited behaviors. After 10 days, he abruptly returned to baseline and had limited recall of the prior events. Three months later, he had a second episode with similar behavioral and sleep changes, again followed by spontaneous return to baseline after 10 days. Two months later, a similar spell occurred, prompting arrangement for psychiatric admission and initiation of quetiapine for a proposed diagnosis of rapid-cycling bipolar disorder. His symptoms again resolved spontaneously. Diagnostic tests including liver function tests, thyroid function tests, ammonia level, urine drug screen, Lyme disease, MRI brain, EEG, and CSF analysis were unremarkable. He was eventually referred to the Sleep Medicine clinic for evaluation. There were no symptoms suggesting other comorbid sleep disorders, and he had a normal neurologic and physical exam at time of presentation. A clinical diagnosis of KLS was made. The patient and family deferred prophylactic or abortive treatment (such as lithium, carbamazepine, or steroids) due to potential for side effects. Modafinil provided symptomatic benefits during the three milder episodes he had over the next few years. Episodes subsequently resolved and the patient is currently in remission (over 5 years) without sequalae.

Conclusion
KLS is a rare disease with typical onset during young adulthood, manifesting with abrupt onset of recurrent episodes of hypersomnia and prolonged sleep duration, lasting from days to several weeks. The diagnosis is clinical and requires at least one additional feature including: cognitive dysfunction, derealization, major apathy, or disinhibited behaviors. Patients revert to baseline between the episodes. Stimulants such as modafinil can promote wakefulness during the episodes.

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1154 Life Threatening Obstructive Sleep Apnea in a Patient with Joubert Syndrome and Pierre Robin Sequence
Authors: Isaacs Thomas , Malhotra Sonal 
Published: April 2024

Abstract

Introduction
Joubert syndrome is a recessive, multisystem genetic disorder classically resulting in cerebellar and brainstem malformations recognizable as the “Molar Tooth Sign” on magnetic resonance imaging (MRI). Respiratory complications in these patients include disordered control of breathing, hypoventilation, central sleep apnea and hyperpnea. Obstructive sleep apnea and Pierra Robin syndrome have also been reported, however the use of mandibular distraction in the population has not been well described.

Report of case(s)
Here we present a 17-month-old male who was diagnosed with Joubert syndrome at 2 months of age after MRI imaging demonstrated the “Molar Tooth Sign” and whole exome sequencing identified a CEP41 mutation. He was also found to have micrognathia, retrognathia and glossoptosis consistent with Pierre Robin sequence. Polysomnography (PSG) in the prone position (as patient did not tolerate the supine position) demonstrated an obstructive apnea hypopnea index (oAHI) of 19.3. Based on this study, mandibular distraction was performed. Following this procedure, the patient was weaned to room air. His respiratory status later worsened, with frequent apneas requiring bilevel positive pressure support (BPAP). A titration PSG was performed, but no acceptable BPAP setting was found. Tracheostomy was discussed, and ultimately the family declined. The patient improved on BPAP with intermittent breaks with low flow nasal oxygen when awake. On follow up, the patient’s respiratory status improved and he was tolerating BPAP therapy with sleep without issue. However, during a respiratory illness the patient unfortunately passed away.

Conclusion
Respiratory and sleep-related complications are frequent in patients with Joubert syndrome. This case emphasizes the importance of the recognition and treatment of these complications and points to the need for further research of this population. This case is also unusual as the use of mandibular distraction in patients with Joubert syndrome is not frequently described.

Support (if any)
Bachmann-Gagescu, Ruxandra, et al. "Healthcare recommendations for Joubert syndrome." American journal of medical genetics Part A 182.1 (2020): 229-249.
1155 Exploring Vagus Nerve Stimulation (VNS) and Its Relationship with Sleep Apnea: Implications and Insights
Authors: Cardona-Quiñones Roberto , Hernandez Saidy Salem , Vargas-Perez Noel 
Published: April 2024

Abstract

Introduction
Obstructive Sleep Apnea (OSA) is characterized by recurrent partial or complete blockage of the upper airway during sleep, leading to disruptions in breathing and intermittent oxygen desaturation, putting patients with epilepsy at risk for seizure exacerbations. However, literature about the development or progression of OSA in patients with a vagus nerve stimulator (VNS) refractory epilepsy is limited.

Report of case(s)
Case of a 42-year-old female with history of right lobe temporal epilepsy since childhood, temporal lobe surgery for epilepsy, currently on VNS who complained of snoring and daytime sleepiness. An Epworth Sleepiness Scale was obtained with a score of 18/24 (high). Physical examination revealed mallampati score of II, BMI: 24.62 kg/m², 139 lb weight, 14-inch neck circumference, and a STOP-Bang sleep score of 2/8 (low-risk). During the initial polysomnography (PSG), the patient exhibited mild to moderate OSA, with associated hypoxemia and disruption in sleep architecture, as reflected by the baseline apnea/hypopnea index (AHI) of 13.5/hr (normal: &lt; 5/hr). The REM AHI (AASM) was 12.3/hr, and the respiratory events and desaturation pattern exhibited an atypically periodic nature, occurring approximately every 3.6 minutes with fixed respiratory events lasting around 29-30 seconds. This periodicity was likely associated with the activation of the VNS implant.

Conclusion
To our knowledge, this case is novel and important in raising awareness of screening for sleep-related disorders, given the limited research about VNS leading to such manifestations. What is significant about this case is that routine tools typically used did not provide sufficient clarity for diagnosing this patient. Instead, a thorough evaluation was required where there are no clear guidelines to evaluate possible VNS-induced sleep-related disorders. The mechanisms involved are complex and diverse. Some proposed causes include respiratory disturbances with changes in airflow, primarily attributable to vocal cord paresis, supraglottic spasm, laryngeal muscle stimulation, or alterations to the brainstem's respiratory center and relay of information, among other possible mechanisms. Further studies are necessary to understand these manifestations, especially in underserved regions like the Puerto Rican and Hispanic populations, where access to sleep studies and research is limited.

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1156 “NIV Nap” to Improve PAP Acceptability and Hypoventilation in Neuromuscular Disease
Authors: Thetford Jay , Chang Melisa , Eng Kevin 
Published: April 2024

Abstract

Introduction
Respiratory failure is the most common cause of death in patients with several types of neuromuscular disease (NMD), including myotonic dystrophy type 1 (MD1). Non-invasive ventilation (NIV) is the cornerstone of treatment, but consistent use and acceptability remain a challenge. While nap-titrations have improved CPAP adherence in obstructive sleep apnea (OSA), we present a case where increased use and optimization of therapy was achieved through a daytime nap NIV titration.

Report of case(s)
47-year-old man with MD1, obesity , and mild OSA with long-standing PAP intolerance presented with dyspnea and hypoxia. Blood gas was suggestive of hypoventilation and work-up for hypoxia including CT chest with pulmonary artery protocol and echocardiogram with bubble were unremarkable. BIPAP and supplemental oxygen were applied with subsequent improvement. Pulmonary function testing was also consistent with hypoventilation, featuring MIP of 14 cmH2O (15% predicted), FVC of 3.30L (65% predicted), and no diffusion impairment. Shortly after discharge, patient was transitioned to NIV with iVAPS with EPAP 5 cmH2O, PS 4-12 cmH2O, TV 6cc/kg, and RR 12 with 4L oxygen. Despite consistent use, ongoing hypoxia was noted on home oximetry testing. The patient was then brought into the sleep lab for a daytime NIV titration study, or “NIV Nap.” During this 2-hour study, he was found to have persistent obstructive events, and was titrated to BIPAP S/T with EPAP 10 cm H2O, IPAP 16 cm H2O, RR 16, and I-time of 0.8-1.5 seconds which achieved optimal therapy for his OSA and hypoventilation. Following this titration, he was able to weaned off of nocturnal oxygen and reported improved symptoms. At 30-day follow-up, remote data demonstrated NIV usage 100% of days, &gt;4-hour/day usage of 87%, residual AHI of 1.3/hr, average TV of 519 mL, and average MV of 10.8 L/min.

Conclusion
While daytime nap titrations have been used with good effect in patients with OSA, this tool has been underutilized in patients with neuromuscular disease and complex hypoventilation. Daytime “NIV Naps” can allow for increased acceptability of treatment while assuring safe and effective NIV therapy, and in this case demonstrated an increase in use.

Support (if any)
N/A
1157 Obstructive Sleep Apnea Symptoms Caused by an Occult Seizure Disorder with a Brain Tumor Focus
Authors: Ibrahem Marwah , Jain Nikhita , Bronstein Jason 
Published: April 2024

Abstract

Introduction
Complex nocturnal behaviors are one of the most difficult diagnostic challenges in sleep medicine. The sensitivity of electroencephalography (EEG) varies with the study duration, epileptogenic lesion location, electrode placement, and the presence of events. A normal isolated EEG is insufficient to exclude an underlying seizure disorder particularly when diagnostic uncertainty remains. Polysomnography (PSG) is the gold standard for the evaluation of obstructive sleep apnea (OSA), and that has demonstrated a bidirectional relationship between OSA and comorbid epilepsy. This case report highlights an adolescent male with seizure-like symptoms initially diagnosed with OSA with refractory nocturnal episodes on continuous positive airway pressure (CPAP) for years until his eventual clinical diagnosis that was prompted by a repeat PSG.

Report of case(s)
A 14-year-old biological male with a history of snoring and excessive daytime sleepiness reported multiple episodes of unresponsiveness during sleep associated with shaking activity and self-reported desaturation at home. Initial episodes terminated upon repositioning of his body, however episodes and symptoms persisted despite treatment with positional therapy and CPAP. He had no clinical episodes during an unrevealing 72-hour EEG study. A repeat diagnostic PSG demonstrated an apnea-hypopnea index (AHI4%) of 1.1 events/hour, an AHI3A of 5.3 events/hour, and an SpO2 nadir of 92%. When the lights were turned on the patient exhibited repetitive facial movement upon awakening with poly-spikes on the corresponding EEG leads. This was followed by an inpatient admission for continuous EEG that demonstrated focal to bilateral tonic-clonic seizure during sleep of left-sided origin. MRI showed a 1.7 cm left basal temporal lobe tumor that was likely the seizure focus. He was discharged on levetiracetam, and ultimately underwent a posterior temporal lesion resection with resolution of his symptoms, including paroxysmal events and excessive daytime sleepiness.

Conclusion
Suspected nocturnal seizures often requires comprehensive and repeated testing modalities as an interictal EEG may fail to detect a seizure disorder and the presence of OSA does not eliminate the possibility of comorbid epilepsy. It is paramount to maintain a heightened index of suspicion as untreated OSA and epilepsy may have additive detrimental health effects. Appropriate identification as in our case may have a curative effect.

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1163 Central Sleep Apnea: A Possible Rare Adverse Effect of Vagus Nerve Stimulation
Authors: Todd Michael , Nelson Michele , Taylor Samuel , Alattar Maha , Rath Subhendu 
Published: April 2024

Abstract

Introduction
Vagus nerve stimulation (VNS) is a neuromodulation therapy for treatment of drug-resistant epilepsy (DRE). VNS therapy is known to affect sleep by increasing risk for obstructive sleep apnea (OSA). We present the case of a young woman with DRE and mild OSA who was found to have central sleep apnea (CSA) five years after VNS placement. Based on our literature review, there are limited case reports on this topic.

Report of case(s)
A 22-year-old female with a history of Tuberous Sclerosis and DRE, couple months’ status post-VNS placement presented for snoring and gasping arousals. She underwent Polysomnography (PSG), which documented mild OSA, with an apnea-hypopnea index (AHI) of 8.6 and associated oxygen desaturation nadir of 82%. Average transcutaneous CO2 (TcCO2) monitoring during supine wakefulness was 43.1 mmHg and during sleep was 44.2 mmHg respectively. She had a titration PSG, which noted that a Continuous Positive Airway Pressure (CPAP) setting of 6 cm of water was effective in treating her OSA. CSA was not observed in either of those studies. Five years later, the patient returned for a routine follow-up. Her home CPAP data review showed that despite a four-hour compliance of 90%, the average CPAP-generated AHI was 14.9. Titration PSG was performed, which showed frequent obstructive and central apneas. Bilevel PAP in ST mode was tested at 15/10 cm of water with a respiratory rate of 10 per minute, which helped apneas. However, obstructive and central hypopneas persisted in this setting. Additionally, global hypercarbia was observed with an average TcCO2 value of 51.9 mmHg during supine wakefulness and 60mmHg (412 minutes &gt; 55mmHg) during sleep, respectively. An MRI two years prior to this titration PSG showed no structural cause for the central apnea. She has never been on opioids and her BMI (30.5) has remained largely unchanged in the past five years. She is planned to have a follow up for further evaluation including changes in VNS settings and a retitration PSG study.

Conclusion
CSA remains an unexplored possible adverse effect of VNS implantation. Further investigations are needed to establish a direct correlation between VNS implantation and CSA.

Support (if any)
none
1165 A Challenging Case of Chronic Fentanyl Use Inducing Central Sleep Apnea After Tracheostomy
Authors: Alsaggaf Mohammed , Karroum Elias 
Published: April 2024

Abstract

Introduction
It’s always challenging to diagnose central sleep apnea (CSA) in related to chronic opioid use.

Report of case(s)
A 66 years old male with a significant history of myasthenia gravis, obstructive sleep apnea, depression, and asthma. Initial home sleep apnea test (HSAT) AHI: 65. However he could not tolerate PAP therapy and eventually underwent an oral device. The repeated HSAT AHI while on oral appliance was 30.3. Due to poor compliance, he has been off both treatments for a few years until he was seen in our clinic with excessive daytime sleepiness (EDS), fatigue, loud snoring and witnessed gasping events. His Epworth scale was 24. His medications were remarkable for fentanyl 150 mcg patch daily for chronic pain. His BMI is 28. A new in-lab polysomnography (PSG) showed AHI of 57.9 with mild central apnea index 5.1. BIPAP 20/16 was recommended under titration. The patient declined PAP therapy and surgery. Subsequently, he underwent tracheostomy and his symptoms were much improved. However he was still having residual EDS. A repeated PSG was done with his tracheostomy with AHI: 107 but all the apnea and hypopnea events were central in origin. The study showed a very irregular and ataxic breathing without Cheyne-Stokes breathing. His ECHO shows an ejection fraction of 51%. All other blood work up was unremarkable. It was believed that his central apnea had been induced by chronic opioid use based on Morphine Milligram Equivalents (MME). Prevalence of CSA in patients taking chronic opioid is 24%. CSA due to opioids is associated with abnormal breathing patterns (ataxic, biot and/ or cluster). The morphine equivalent daily dose (MEDD) or Morphine Milligram Equivalents (MME) is strongly associated with the severity of sleep breathing disorders (SBD) predominantly CSA. Studies have shown that ataxic breathing during NREM was more frequent at MED ≥ 200. Our patient's fentanyl dose of 150 mcg patch equals 360 MME which puts him at an extreme risk of CSA.

Conclusion
It’s important to calculate MME among patients with opioid to avoid risk of inducing central sleep apnea.

Support (if any)
None
1166 Long-term Buprenorphine Use and Prevalence of Sleep Apnea: A Case Series
Authors: Eshghabadi Mohammad Amin , Javaheri Shahrokh , Aboelnasr Amr 
Published: April 2024

Abstract

Introduction
It has been well documented that opiate use is a risk factor for developing sleep apnea. We suspect that buprenorphine being a partial-opioid-agonist to also be a risk factor for developing sleep apnea. Study by Farney et al showed an association between Buprenorphine use and sleep apnea. But the literature also has contradictory findings in which a publication by Grunstein et al showed that by switching from methadone to buprenorphine pre-existing sleep apnea was eliminated, although this study only observed the effects of only short-term buprenorphine use. There is a gap in understanding the prevalence and specific risk factors associated with sleep apnea in chronic buprenorphine.

Report of case(s)
Patient 1: 46-year-old male. Medical History: Obesity, hypertension, coronary artery disease. Former smoker (15 pack year smoking history) Buprenorphine Use: 6 years for opioid dependence. Sleep Study: Central sleep apnea- hypopnea index of 10.3, unresponsive to CPAP titration. Patient 2: 66-year-old male Medical history: Chronic back pain, smoker Buprenorphine Use: 6 years for chronic back pain. Sleep Study: Central sleep apnea- hypopnea index of 4.5, unresponsive to CPAP titration. Patient 3: 47-year-old female. Medical History: Obesity, narcolepsy. Buprenorphine Use: 6 years. Sleep Study: No evidence of central sleep apnea on initial and subsequent studies. Patient 4: 28-year-old male. Medical History: Obesity. Buprenorphine Use: 8 years for chronic pain or opioid dependence. Sleep Study: No central sleep apnea on initial study. Subsequent study revealed central sleep apnea with central apnea- hypopnea index (AHI) of 2.9. Patient 5: 68-year-old male. Medical History: Atrial fibrillation, hyperlipidemia, chronic kidney disease, cigarette smoker, obesity. Buprenorphine Use: 8 years for chronic pain or opioid dependence. Sleep Study: Initial study showed central AHI of 0.5. Subsequent study revealed an increase in central AHI to 13.1.

Conclusion
The presented cases raise awareness of a possible link between long- term buprenorphine use and sleep apnea. This case series underscores the importance of monitoring individuals on long-term buprenorphine therapy for the potential development of sleep apnea. Further research is needed to elucidate the underlying mechanisms and explore management strategies for individuals facing this complication of buprenorphine therapy.

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