bluuebunny/crossref_metadata_2025 huggingface dataset:
1164 Sleep Conversion of Atrial Bigeminy to Sinus Rhythm and Using Oximetry for Heart Rate
Authors: Lisio Catherine Di , Strohl Kingman
Published: April 2024

Introduction

Patient presented for PSG with initial wake, N1 period showing bigeminy on ECG that converted to sinus rhythm during N2, N3, and REM. SR persisted despite periods of hypoxemia until the end of the study. Furthermore, the heart rate, as generated using oximetry and Polysmith, would not recognize the bigeminal beat underestimating heart rate by half.

Report of case(s)

A middle-aged female was referred to Sleep Medicine for difficulty falling asleep, staying asleep, snoring, witnessed apneas. Epworth Sleepiness Scale score (ESS) was 9/24. PSG was performed using Polysmith software. Epochs showed: Wake: bigeminy on EKG and corresponding plethysmography with one large peak (the visible P wave) and one small peak (where P is hidden in the preceding T wave). Only the first P wave is used to generate heart rate. A clear transition can be seen within a single epoch in N1: EKG changes from bigeminy to SR with corresponding plethysmography showing two peaks (as in wake) that convert to a single peak in sinus rhythm. Heart rate is then correctly accounted for midway through the epoch. N2 and REM: SR, single peak on plethysmography, accurate heart rate continue. ECG approximately one week prior to PSG showed sinus bradycardia.

Conclusion

While there has been no mention of atrial bigeminy and sleep in cardiovascular or sleep medicine literature, one hypothetical reason for its resolution in sleep is the reduction of sympathetic and cholinergic tone. As for the spurious heart rate on oximetry, software did not recognize the smaller plethysmography peak, underestimating heart rate by half during periods of bigeminy. Atrial bigeminy is associated with heart failure, alcohol, electrolyte derangement, caffeine and usually left untreated in asymptomatic patients. Patients with symptoms, such as palpitations, dizziness, near syncope, or syncope can be treated with β-blockers, calcium-channel blockers, or catheter ablation. While this patient did not complain of symptoms, recent labs did show hypomagnesemia and hypokalemia.

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1168 Snoring Cessation in a Case of Vagal Nerve Stimulator Induced Obstructive Hypopnea
Authors: McFarlane Robin , Nguyen Andrea , Chaung Matthew , Lee-Iannotti Joyce
Published: April 2024

Introduction

Vagal nerve stimulators (VNS) have been found to induce sleep disordered breathing and many patients will seek sleep evaluation after the development of symptoms. Snoring is a common reason patients may present. Once therapy is initiated, the lack of snoring is often used as a clinical marker for reduced apnea. Here we present the counterintuitive case of a VNS patient who had treatment-refractory obstructive sleep apnea with snoring that halted during apneic episodes.

Report of case(s)

A 24-year-old male, past medical history of medically refractory epilepsy w/ VNS, developmental delay with minimal verbal output, sleep apnea treated with CPAP (Continuous Positive Airway Pressure), presented for a split night polysomnogram based on reports of witnessed nocturnal apneas despite treatment with CPAP. To find new CPAP settings for worsening apnea we performed a split night polysomnogram. During the baseline diagnostic portion of the study the apnea hypopnea index was 40.9 events/hour with severe obstructive sleep apnea consisting of 12 obstructive apneas and 32 hypopneas. Respiratory events were noted to have remarkable chronicity, lasting 30 seconds every 1 min and 10 seconds. The obstructive events were also seen during wake cycles. We discovered his VNS settings were programmed for a 1 minute and 10 second OFF cycle with 30 second ON cycle, coinciding with the periodicity of his apneic events. Additionally, snoring occurred throughout the study but ceased during the hypopneas. VNS artifact was noted in the EKG lead during the 30 second periods of hypopnea in a dramatic time locked obstructive pattern, suggesting VNS induced collapse of the upper airway during VNS ON phase.

Conclusion

This is a report of VNS induced cessation of snoring with paradoxical worsening of hypopneas, as well as awake hypopnea events. VNS can induce sleep apnea and our study shows lack of snoring is not a reliable indicator of lack of hypopnea due to the adduction of vocal cords and resultant laryngeal collapse. We suggest that all patients with a VNS implanted device undergo a sleep study to evaluate for VNS induced OSA after implantation of VNS, and a follow up with a sleep study titration of VNS in select cases.

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1177 The Patient Who Never Stopped Dreaming
Authors: Gupta Shalini , Avidan Alon
Published: April 2024

Introduction

Sleep-related hallucination (SRH) is a parasomnia that may be encountered in the form of altered dream mentation in α-synucleinopathies, particularly in the setting of dementia with Lewy bodies (DLB). Visual hallucinations (VH) are complex, well-formed, episodes that represent a core diagnostic criterion for DLB. We report a patient who presented with REM sleep behavior disorder (RBD), VH, and perceiving the world as if he were in a persistent dream-like state coinciding with new cognitive deficits.

Report of case(s)

A 74 y/o male presented with a subjective perception of “being in a constant dream-like state” for the past 2 years. He describes “atypical nightmares and hypnopompic visual hallucinations” The Patient describes that objects do not feel real, but appear drawn out as in a cartoon. He feels “disconnected” from the environment and perceives images as “distant, distorted, unreal, but also very colorful, vivid, saturated as a cartoon animation sequence. He no longer feels safe to drive and his wife reveals a failure to appreciate depth leading to an increased frequency of falls. Ophthalmologic exam was unremarkable including normal visual acuity, field testing, and color vision. ESS: 8, Sleep log: subjective total sleep- 7-8 hrs/night. Polysomnography conforms the presence of dream enactment behaviors and REM sleep without atonia meeting the diagnostic criteria for RBD. AHI-3, SaO2: 92%. REM latency: 92 minutes.

Conclusion

People with DLB experience a spectrum of sleep disorders, especially RBD, but also experience a range of sleep and wake-related complex visual hallucinations. The latter may reflect impairment in sensory integration, particularly during the transformation from mild cognitive impairment (MCI) to DLB due to alteration in visual sensory processing. Sleep clinicians serve a critical role in the evaluation of SRH, and differentiating these from other mimics, such as VH related to neuropsychiatric conditions. Due to their bizarre and perplexing presentation, visual-spatial processing disorders (VSP) in DLB may be stigmatized, dismissed, or attributed to RBD. Sleep clinicians must therefore include recognizing VSP in any patient presenting with SRH.

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1184 The Leptin Puzzle: Decoding Genes, Morbid Obesity, Sleep-Related Disorders and Its Influence
Authors: Colon-Soto Jose , Vega-Sanabria Bryan , Cardona-Quiñones Roberto , Balsalobre-Vélez Alexandra , De Jesus-Rojas Wilfredo
Published: April 2024

Introduction

The role of early-onset obesity-related genetic predisposition and leptin receptor variants have been studied in previous studies. However, sleep-related disorders related to a genetic predisposition, leading to obesity, and how leptin could play a role in sleep-related disorders have been limited. In this study, we explore a case of how leptin receptor variants could play a role in the relationship between obesity and sleep-related disorders.

Report of case(s)

Case of a 17-year-old Hispanic female presented with a history of frequent snoring, gasping for air while sleeping, daytime sleepiness, sleep onset, maintenance difficulties, and limb movements at sleep. Polysomnography (PSG) was performed and later diagnosed with Obstructive Sleep Apnea (OSA). Respiratory disturbances were associated with oxygen desaturation down to a nadir of 68 % during sleep and a mean oxygen saturation of 91%, with an AHI of 22.7 events/hr. Additional genetic studies were performed to assess the causes of obesity, presenting with BBS9 heterozygous gene for a sequence variant defined as c.396GC, which is predicted to result in the amino acid substitution p.Gln132His. Furthermore, this patient is heterozygous in the LEPR gene for a sequence variant defined as c.658GA, which is predicted to result in the amino acid substitution p.Val220Ile, which has been reported in the heterozygous state in severe obesity, which increases leptin levels due to mutations in its receptor.

Conclusion

Leptin dysregulation has been associated with an increased predisposition to obesity, which could lead to sleep-related disorders, such as OSA. What is novel in our case is that our patient has an LEPR gene heterozygous variant, leading to an increase in leptin levels, a greater increase in metabolic dysregulation, and increased body weight, thus worsening her OSA. At the same time, research has shown that conditions such as OSA also increase leptin levels, which could worsen her OSA and prognosis. Further research is needed to assess LEPR and its role in OSA, especially in Hispanic populations, where research is limited.

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1186 Phrenic Nerve Stimulation to Improve Central Sleep Apnea and Quality of Life in Palliative Patients: A Case Report
Authors: Chaung Matthew , Nguyen Andrea , Thacker Stacy , Lee-Iannotti Joyce
Published: April 2024

Introduction

Central sleep apnea (CSA) is a devastating disease and occurs in approximately 40% of patients with a history of stroke, neurological conditions, and heart failure(Joseph et.al). Typical symptoms include disrupted sleep or paroxysmal nocturnal hypoxemia. The gold standard for diagnosis is polysomnography and treatment is typically positive airway pressure (PAP)(Ishikawa et. al). REMEDE stimulates the phrenic nerve transvenously, providing innovative therapeutic alternatives for patients who cannot tolerate PAP therapies(Joseph et. al).

Report of case(s)

A 77-year-old male with history of advanced Parkinson’s disease (PD) established with palliative care, was referred to the sleep clinic for additional management of severe central sleep apnea refractory to bilevel positive airway pressure (Bilevel PAP ST). Despite initial Bilevel PAP settings of 22/18 cm of water with a backup rate of 12 bpm, the patient faced difficulties using his machine. His apnea hypopnea index (AHI) remained in the moderate range at 18 events/hour despite PAP therapy. Attempts to adjust pressure settings, implement acclimation measures, and mask changes proved unsuccessful. Due to persistent symptoms and poor quality of life (QoL), the patient was subsequently considered for phrenic nerve stimulation, meeting inclusion criteria. Six months post-implantation, he experienced significant improvement in his reported QoL (improved mood, less sleepy during the day with improved Epworth Sleepiness scores), confirmed from his wife. Unfortunately, he passed away 11 months after implantation due to PD-related complications.

Conclusion

Phrenic nerve stimulation emerges as a novel and effective treatment for CSA, particularly for patients intolerant to Bilevel PAP therapy. This case study highlights the positive impact of phrenic neuromodulation on QoL and sleep improvement, even in patients with palliative conditions and advanced neurodegenerative diseases. Even in patients with end-stage Parkinson disease neurosurgery to replace deep brain stimulation generators is low risk(Carlson et. al). Moreover, nutrition and swallowing can be difficult in end stage Parkinson disease and there is a role for percutaneous endoscopic gastrostomy (PEG) tube to also improve QoL(Schindler et. al). We assert that methods aimed at enhancing sleep, albeit invasive and surgical, should be considered viable options for patients categorized as palliative or terminal, with the overarching goal of improving their remaining days(Lilley et. al).

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0119 Symptoms of Post-Traumatic Stress Disorder and Self-Rated Multidimensional Sleep Health
Authors: Cassell Jack , Marquez Francisco , Piechota Amanda , Decker Andrea , Halvorson Emily , Cribbet Matthew
Published: April 2024

Introduction

Sleep disturbances are common among individuals with posttraumatic stress disorder (PTSD). Individuals with PTSD experience heightened sensitivity in brain regions involved in fear and emotion regulation, which may impair sleep onset, maintenance, depth, duration and quality. This suggests that PTSD may be influencing multiple aspects of sleep at once. Thus, the goal of this study was to examine associations between symptoms of PTSD, and self-rated multidimensional sleep health.

Methods

A community sample of midlife adults living in the deep south (N=70; Age=52.93±9.05; Female=75.7%; 11.42% African American) completed a demographic measure, the PTSD Checklist for DSM-5 (PCL-5), and a self-rated measure of multidimensional sleep health (created by summing scores on items assessing satisfaction, duration, regularity, alertness, efficiency and timing). Bivariate correlations were conducted to test the association between symptoms of PTSD and sleep health. Follow-up multiple regression analyses were used to examine the association between sleep health and symptoms of PTSD, while adjusting for sex, age, and BMI. Exploratory analyses were conducted to determine which aspects of sleep health were driving any significant associations with PTSD symptoms.

Results

Bivariate correlations revealed significant negative relationships between symptoms of PTSD and self-rated multidimensional sleep health (r=-0.349, p=0.003). After accounting for age, sex, and BMI, regression analyses demonstrated a significant inverse relationship between symptoms of PTSD and sleep health, such that a one unit increase in PTSD symptoms corresponded to a 0.09 decrease in a multidimensional measure of sleep health (b=-0.099, t=-3.24, p=.002, R2=0.144). In separate multiple regression models, accounting for BMI, age and sex, greater PTSD symptoms were associated with poor sleep satisfaction (b=-0.021, t=-2.49, p=.015, R2=0.137) and poor sleep efficiency (b=-0.029, t=-2.90, p=.005, R2=0.131), but unrelated to other aspects of sleep health (ps > .05).

Conclusion

Symptoms of PTSD were significantly negatively associated with self-reported multidimensional sleep health. These results were driven primarily by poor sleep efficiency and satisfaction with sleep. These findings add to growing literature on PTSD and sleep and suggest specific targets for interventions.

Support (if any)

Support was provided by the Deep South Resource Center for Minority Aging Research (RCMAR) P30 AG0301054 from the NIA.
1190 Periodic Limb Movement Disorder Masked by Concurrent Focal Aware Seizures
Authors: Rasul Ammar , Khan Anees , Peters Jon , Motamedi Gholam
Published: April 2024

Introduction

Sleep disorders such as sleep apnea and sleep-related movement disorders are more common in patients with epilepsy compared to the general population. Complex rhythmic motor events related to periodic limb movement disorder (PLMD) can overlap with the hypermotor phenomenon of nocturnal seizures, making it a challenging diagnosis solely based on clinical description. The recognition of these repetitive stereotypical movements from seizures would be essential to guide targeted therapy.

Report of case(s)

We report a patient with focal aware seizures who concurrently experienced sensory symptoms typical of restless leg syndrome (RLS) as her ictal symptoms, as well as paroxysmal nocturnal motor symptoms meeting the diagnostic criteria of PLMD. A 51-year-old right-handed female presented with two years of seizures and nocturnal episodes primarily characterized by creepy crawly feeling in both her upper and lower extremities as well as her trunk with intact awareness. The patient also endorsed non-restorative sleep and daytime sleepiness. During an admission to the epilepsy monitoring unit, several habitual events were recorded that were consistent with fronto-temporal seizures. Interestingly, the majority of her ictal events started with the same creepy crawly sensation. Blood count, serum chemistry, and liver and renal function tests were unremarkable, except for very low serum ferritin levels (4.5 ng/ml) which, in combination with her sensory symptoms raised suspicion for PLMD. Subsequent polysomnography (PSG) showed mild OSA (AHI 9.8/h) and moderate to severe PLMD (43 events/h). Symptoms improved after iron supplementation along with better seizure control (adding lamotrigine to her levetiracetam). A repeat ferritin improved to normal range (46 ng/ml).

Conclusion

Our case demonstrates that clinical presentation of RLS/PMLD can be indistinguishable from the primary seizure semiology. RLS/PLMD can be a consideration in patients with epilepsy complaining of sensory ictal semiology as seen in this case and may warrant further work-up.

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1191 A Pediatric Case of Treatment Emergent Central Sleep Apnea During High Flow Nasal Cannula Polysomnography
Authors: Giles Cristina , Wisniewski Benjamin , Hawkins Stephen
Published: April 2024

Introduction

High flow nasal cannula (HFNC) is an alternative therapy to continuous positive airway pressure (CPAP) in pediatric populations with behavioral intolerance or contraindications to CPAP. Studies evaluating HFNC as a treatment modality in the pediatric literature have been limited.

Report of case(s)

A previously healthy 12-year-old male with a history of obesity presented with partial and full thickness burns to his face, neck, chest, and bilateral upper-extremities following a bonfire accident. His burns covered 15% of his total body surface area. He required hospitalization for acute burn management and skin grafting. During his hospitalization, he was observed to have snoring, witnessed apneas, and nocturnal hypoxemia concerning for underlying obstructive sleep apnea. He was unable to utilize a non-invasive positive pressure mask interface due to his extensive facial dressings and pressure sensitive skin grafts. He was discharged home on high flow nasal cannula at 10-15 Liters/minute. The patient experienced weight gain following hospitalization, possibly related to Cyproheptadine use for burn-related itching. Approximately 10 months after discharge, the patient underwent split-night HFNC polysomnography. The diagnostic portion of his study revealed severe obstructive sleep apnea with an OAHI of 28.8 events/hour. There were no observed central or mixed respiratory events and no REM stage sleep during the diagnostic portion of the study. After initiating HFNC at 40 L/minute and 34 degrees Celsius per sleep laboratory protocol, the OAHI decreased to 0.0 events/hour despite REM rebound, and the CAHI increased to 16.8 events/hour. This study was conducted at approximately the same altitude as the patient’s home.

Conclusion

This patient experienced treatment emergent central sleep apnea during HFNC polysomnography. Treatment emergent central sleep apnea with HFNC has previously only been described in a limited number of pediatric patients with underlying chronic lung disease, developmental delay, or genetic syndromes. This case highlights HFNC as an alternative treatment modality to CPAP as well as the need for further understanding of HFNC titration in pediatric sleep laboratories.

Support (if any)

The Breathing Institute at Children’s Hospital Colorado
1194 A Case of Unsuccessful Positive Airway Pressure Titration
Authors: Walia Neal , Chang Melisa , Zeidler Michelle
Published: April 2024

Introduction

Oronasal masks are commonly used interfaces when treating obstructive sleep apnea (OSA). Masks modalities are often chosen based on patient preference and comfort however, oronasal masks may cause worsening of OSA in some individuals.

Report of case(s)

An 83 year old male presented for snoring, witnessed apneas, and daytime somnolence. A split-night PSG was ordered for evaluation. During the diagnostic portion of the study, the patient exhibited moderate OSA with an apnea hypopnea index (AHI) of 27/hr. The titration portion of the study was initiated with an oronasal (full face) interface. Continuous positive airway pressure (CPAP) pressure was titrated from 5cm H2O to 15 cm H20 with worsening of obstructive events and a residual AHI of 21 to 40/hr. Bilevel positive airway pressure (BiPAP) was then initiated and titrated from 19/15 cm H2O to 21/17 cm H20 with a continued residual AHI of 40/hr. No central apneas were noted. Upon review, suspicion arose whether the oronasal mask exacerbated the patient’s OSA. A repeat titration PSG was conducted with a nasal mask. With this mask a pressure of 8cm H20 resulted in a residual AHI of 0/hr. The patient was prescribed APAP 6-10 cm H20 with a nasal mask which resulted in excellent compliance and a residual AHI of 3.6/hr.

Conclusion

This case report presents a patient with worsening OSA due to use of an oronasal interface. Prior studies have suggested that oronasal masks produce less retropalatal airway opening than nasal masks. There may also be an effect on anterior-posterior distance of the mandible in the setting of exhalation. When possible, oronasal masks should be avoided in lab or home titration studies as they may lead to worsening OSA leading to escalation of PAP pressures and/or adjustment in titration modality.

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1195 Sleep-related Hallucinations
Authors: Deshpande Pushkar , Vyata Vishruth , Diaz-Abad Montserrat , Konikkara John
Published: April 2024

Introduction

Sleep-related hallucinations, encompassing visual and auditory experiences pose a complex clinical challenge. Here, we illustrate 2 cases with diverse underlying etiologies.

Report of case(s)

Case 1: 76-year-old male with 3 months of nocturnal visual hallucinations, occurring 3-5 nights per week. These ranged from familiar and unfamiliar faces and characters reminiscent of movies. Symptoms occurred when waking up in the middle of the night, with difficulty returning to sleep. Polysomnography (PSG) unveiled moderate obstructive sleep apnea (OSA) without findings indicative of parasomnias and no symptoms on the night of PSG. Brain MRI revealed chronic infarcts involving right basal ganglia and bilateral optic radiations. Lesions of structures that commonly cause visual hallucinations (optic lens, retina, thalamus, midbrain) were absent. The frequency of episodes decreased to 1-2 per month after initiating CPAP therapy. Potential mechanism of improvement could be PAP mediated optimization of perfusion to the ischemic penumbra. Case 2: 93-year-old male with an eight-year history of insomnia due to musical hallucinations (MH). Auditory experiences included church and semi-classical music, with 80% being familiar tunes. The patient, a former church choir member, has a history of progressive hearing loss and tinnitus. Brain MRI/MRA revealed chronic periventricular microvascular ischemic changes. Extended EEG showed no evidence of seizures. Audiometry confirmed moderate-severe bilateral sensorineural hearing loss. No response was seen to Levetiracetam, Gabapentin, Donepezil and behavioral therapy. Home sleep study revealed severe OSA. CPAP therapy did not result in improvement of MH. MH improved significantly with Lorazepam. Hypoacusis, psychiatric disorders, focal brain lesions, epilepsy and intoxication are common etiologies for MH. The etiology in this case is very likely due to hypoacusis. The hypothesis is deafferentation resulting in release phenomenon or spontaneous activity in the auditory pathway, manifesting as MH.

Conclusion

Sleep-related and nocturnal hallucinations warrant a comprehensive evaluation. Differential diagnoses include neurological causes, including stroke, seizure, Multiple Sclerosis, tumor, vascular lesions, and neurodegenerative disorders. Other etiologies include psychiatric disorders, medications, alcohol, otologic and ophthalmic pathologies. It is also reasonable to evaluate for comorbid OSA as appropriate treatment may decrease the frequency of episodes and improve overall sleep in some patients.

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1196 Use of Home Sleep Apnea Testing to Investigate Persistent Hypoxia in the Remote Setting
Authors: Epstein Sam , Eng Kevin
Published: April 2024

Introduction

Nocturnal oxygen titrations can be resource intensive, and often require the patient to undergo an attended polysomnogram (PSG). Veterans who live in remote areas are often reluctant to undergo PSG, particularly in those with multiple medical comorbidities that prohibit prolonged travel. We present a case of a veteran with severe OSA and persistent nocturnal hypoxia who underwent a remote oxygen titration using home sleep apnea testing (HSAT) equipment.

Report of case(s)

79-year-old male with history of hypertension and diabetes diagnosed with severe OSA (AHI 38 events/hour), with significant oxygen nadir of 65%. He was prescribed automatic positive airway pressure (APAP) of 6-16 cmH2O and a follow up 3-night oximetry was ordered which showed persistent nocturnal hypoxia. Remote monitoring showed regular PAP use with AHI < 5/hr during the nights of oximetry. Unfortunately, the patient was unable to travel for attended PAP and oxygen titration study for further diagnostic assessment. It was instead decided to repeat 3-night oximetry with use of supplemental oxygen (1, 2, 3 liters/minute for nights one, two, and three respectively). Results again showed persistent nocturnal hypoxia with AHI < 5/hr on remote data monitoring. 3-night HSAT connected to the patient’s APAP device was ordered, with bleed in oxygen at 2, 3, and then 4 liters/minute over the consecutive nights of the study. Results included the C-flow channel, which surprisingly showed persistent snoring and obstructive events (average AHI of 8/h) despite APAP, a discordant finding to that on remote data monitoring. The decision was made to transition patient to an automatic bilevel positive airway pressure mode with two-liter oxygen bleed in. This resulted in adequate treatment of OSA and nocturnal hypoxia as confirmed by follow up HSAT. It is notable that ongoing diagnostic evaluation of hypoxia is underway.

Conclusion

Aside from its promise in diagnosing moderate to severe OSA, HSAT over oximetry alone may provide additional diagnostic information (C-flow channel) in patients with residual nocturnal hypoxia despite PAP. In patients who are not able to undergo formal attended titration studies such as those who live in remote settings, this is a potential alternative.

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1197 Cataplexy - Management Conundrums
Authors: Buntak Vesna , Masroor Kiran , Geer Jacqueline
Published: April 2024

Introduction

Creating a guideline for the management of patients with narcolepsy is complex due to the number of potential mechanisms that could account for the increased risk of perioperative complications: aggravation of narcolepsy itself, dysautonomia, narcolepsy-related medications, anesthesia interactions, etc.

Report of case(s)

An 80-year-old woman with a history of narcolepsy type 1, depression, restless leg syndrome, obstructive sleep apnea, and atrial fibrillation presented to our sleep clinic to establish care. Her narcolepsy was managed with strategic naps, but no medications. She had previously experienced rare episodes of cataplexy, described as the inability to talk, and syncope. While on anticoagulation for her atrial fibrillation, she sustained a fall during an episode of cataplexy resulting in brain bleed and eye loss, which prompted her to start pitolisant. Subsequent polysomnography showed an apnea-hypopnea index of 72/hr with nadir oxygen of 74%, requiring Adaptive Servo-Ventilation (ASV), which she did not tolerate. She noted cognitive decline and extreme weakness resulting in hospitalization, during which neurology reported frequent myoclonic jerks and excessive sleepiness. Given her untreated sleep apnea, we recommended re-initiation of ASV during the inpatient hospital stay, which improved her symptoms significantly. She subsequently developed an unrelated need for urgent bowel surgery, necessitating cessation of oral intake, which was temporally related to debilitating and refractory cataplexy. Following recovery from this acute hospitalization, she recovered and was again seen in the clinic, with no further episodes of cataplexy and improved sleepiness in the setting of ASV compliance and resumption of her pitolisant.

Conclusion

This case emphasizes the importance of controlling even rare cataplexy in the setting of potentially catastrophic outcomes. It also raises concerns about the management of narcolepsy with cataplexy in the perioperative period, which currently lacks quality data and guidelines.

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1200 Overstimulated: A Case of Hypoglossal Nerve Stimulator Over-titration
Authors: Seibert Katherine , Ryden Armand , Chang Melisa
Published: April 2024

Introduction

Hypoglossal nerve stimulation (HNS) is an effective treatment for obstructive sleep apnea (OSA) that stimulates nerves that protrude the tongue and move the hyoid bone anteriorly to restore airway patency during sleep. After implantation, the stimulator voltage is titrated to an optimal therapeutic level. We describe the case of a patient whose OSA was effectively treated at a lower voltage and then demonstrated worsening sleep apnea after over-titration to a higher voltage.

Report of case(s)

A 37-year-old healthy man with a body mass index of 32 kg/m2 presented with snoring. A polysomnogram revealed an apnea hypopnea index (AHI) of 53.9 events per an hour consistent with severe OSA. He was intolerant of continuous positive airway pressure (CPAP) and trial of a mandibular advancement device (MAD) was ineffective. The patient subsequently underwent hypoglossal nerve stimulator implantation without complications. After an appropriate healing period, his device was activated with a noted functional amplitude of 0.8V and an allowed range of 0.8V to 1.8V. During self-titration, the patient noted improvement of snoring and apneas at an amplitude of 1.6V. However when he increased the amplitude further he noted recurrence of his OSA symptoms. He underwent an attend polysomnography titration study that revealed an estimated AHI of less than 10 with his HNS set to 1.6V representing an appropriate treatment response. However, when his HNS was titrated up to 1.7V, his estimated AHI increased to 26.4 consistent with moderate OSA. The patient’s device was set to a voltage of 1.6V.

Conclusion

HNS is an important treatment option for patients with OSA who are intolerant of CPAP. After implantation, attention must be given to careful titration of the stimulation voltage during an attended polysomnogram to avoid over titration which can lead to an increased number of obstructive events in some patients. While the mechanism of this phenomenon is currently unknown, theoretical mechanisms include stimulation of the lateral division of the hypoglossal nerve triggering tongue retraction at higher voltages and genioglossus muscle fatigue.

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1203 When the Options Run Out… Less Is More… a Case of Pediatric Narcolepsy with Limited Treatment Options
Authors: Khan Yousaf , Revana Amee
Published: April 2024

Introduction

We report a pediatric patient with Narcolepsy Type 1 with significant daytime impairment who faced challenges with the standard treatment options of pediatric narcolepsy.

Report of case(s)

The patient is a 16 year old female with confirmed diagnosis of narcolepsy type 1 (mean sleep latency < 5 minutes, 4 sleep onset REM periods). Her symptoms (excessive daytime sleepiness (PDSS 26), cataplexy, hypnagogic/hypnopompic hallucinations and depression) gradually started at the age of 12 years of age and worsened overtime. Cataplexy initially involved eye and facial twitching that were triggered by laughter or anger which gradually progressed to generalized loss of tone. As the initial management of narcolepsy, she was started on atomoxetine with no change in symptoms. She transitioned to methylphenidate which improved her daytime sleepiness minimally but with no improvement of the cataplexy. Due to further adverse effects with stimulants, she started sodium oxybate with low dose dextroamphetamine. Initially, the combination improved wakefulness but then with dose titration, the patient developed severe depression and suicidal ideation after 8 months of use. She also demonstrated manic episodes and anorexia. With each treatment regimen, her depression and anxiety vacillated in severity. Lastly, a trial of pitolisant was titrated to 13.35 mg. She developed heart palpitations with the higher doses, so low dose Pitolisant 8.9 mg daily was continued. At this dose sleepiness improved, with some improvement in functionality. Venlafaxine 225 mg daily was added to the regimen to control cataplexy and depression with good benefit. With the low dose pitolisant and venlafaxine combination, she has been able to participate in extracurricular activities at school.

Conclusion

Pediatric narcolepsy can be challenging to treat as there are limited treatment options. We presented a case of a teen who had multiple symptoms of narcolepsy with coinciding depression and anxiety. Despite multiple drug regimens, her symptoms improved with off label use of Pitlosant , interestingly at low dose. Although clinical manifestations of pediatric narcolepsy may be life altering for the patient, close clinical follow up is imperative to screen for adverse effects after implementing a treatment plan.

Support (if any)

None
1207 Intercostal Monitoring as a Novel Approach to Polysomnography During Hypoglossal Nerve Stimulator Titration
Authors: Sedar Cole , Andre Aguillon
Published: April 2024

Introduction

Hypoglossal nerve stimulation is an alternative treatment modality for patients with obstructive sleep apnea who are not able to achieve ideal compliance rates with PAP therapy, and is becoming more widespread, with growing numbers of patients considering and undergoing implantation. Protocol for the only commercially available hypoglossal nerve stimulator (HNS) in the US includes a follow up titration polysomnogram (PSG) after implantation for device adjustment. This is typically performed using a standard PSG setup, which does not include leads for monitoring of the intercostals. Following implantation of HNS in our patient, we added intercostal electromyography to the standard PSG during HNS titration study, allowing us to directly monitor activation of the intercostal muscle group responsible for the sensing lead on the HNS. A 2016 study comparing intercostal EMG activity to respiratory inductance plethysmography (RIP) monitoring for differentiating obstructive from central events showed an increase in sensitivity for chest wall EMG in detecting respiratory effort, with 10.4% of events initially detected as central by RIP monitoring being reclassified as obstructive by chest well EMG, and 2.8% noted to be mixed rather than central4

Report of case(s)

Our patient is a 46-year-old female with a past medical history May-Thurner syndrome and nutcracker syndrome s/p stenting, Ehlers-Danlos syndrome, mitral valve regurgitation, Barrett’s esophagus, chronic migraines, thyroid cancer s/p radical resection and resultant hypothyroidism, Insomnia, gastroparesis s/p total colectomy, melanoma in situ s/p resection, cholecystectomy, and spinal nerve stimulator implantation. She was initially diagnosed with OSA in 2022 with an AHI 21.8 events per hour, nadir SpO2 87%. She trialed CPAP, but was intolerant to therapy. She underwent HNS implantation on 3/9/23. The device was activated in the clinic and she underwent polysomnography on 7/31/23 for voltage and setting titration. During the study, we monitored electromyography of the intercostals along with a standard PSG montage.

Conclusion

Intercostal electromyography, in addition to standard polysomnographic montage allowed for direct visualization of the intercostal contraction and subsequent activation of the hypoglossal nerve stimulator. This expanded montage provides more information for the titration study, and may allow for more precise HNS device settings given the increased sensitivity in detecting respiratory effort.

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1209 A Case of Morquio Syndrome Presenting as Respiratory Failure Due to Severe OSA and Successful Long-term Treatment
Authors: Vyata Vishruth , Deshpande Pushkar , Guntupalli Vineeta , Diaz-Abad Montserrat , Glick Danielle , Lasso-Pirot Anayansi
Published: April 2024

Introduction

Morquio Syndrome is a lysosomal storage disease (mucopolysaccharidosis type 4) characterized by the accumulation of glycosaminoglycans (GAG) and causes craniofacial, skeletal, and soft-tissue abnormalities. The pathophysiology of obstructive sleep apnea (OSA) is multifactorial due to GAG accumulation in upper airways, adenoids, trachea, and bronchi causing narrowing and collapsibility of the airways.

Report of case(s)

A nine-year-old male with Morquio Syndrome presented to the emergency department with difficulty staying awake for 5 days and an upper respiratory tract infection. He would doze off and wake up every few minutes several times a day. He had no prior history of somnolence. He was admitted to the ICU given the severity of the presentation and was clinically started on BiPAP. There was also a history of loud snoring but no witnessed apneas. An in-laboratory polysomnogram was performed to confirm his diagnosis of severe OSA and sleep-related hypoxemia, with an apnea-hypopnea index of 34.1, minimum oxygen saturation 78%, and time < 90% of 74.8 minutes. After a titration study, he was started on noninvasive ventilation (NIV) with bilevel PAP. He remained compliant with outpatient NIV therapy and did not have any hospital admission for respiratory illness. Seven years later, he had a repeat titration study and was switched to NIV with AVAPS. He had one emergency visit for dyspnea and tested positive for influenza. A CT at that time showed severe tracheal stenosis of 8 mm at the thoracic inlet. On direct visualization with laryngoscopy, the stenosis was noted to be around 3 mm, probably due to dynamic airway obstruction. He improved with symptomatic treatment and was discharged the next day. Despite having significant airway abnormalities, he has never required intubation for respiratory failure, likely attributable to consistent use of NIV therapy.

Conclusion

Symptoms of OSA can be subtle, and if unrecognized, can potentially progress to respiratory failure. It is crucial, particularly in children with airway abnormalities, to actively screen for symptoms of OSA and initiate appropriate treatment. Our case highlights successful long-term treatment spanning more than a decade in an individual with Morquio Syndrome and avoidance of tracheostomy using NIV with bilevel PAP and AVAPS therapy.

Support (if any)
0121 Discrimination Attributed to Personal Characteristics and Sleep Health Among Black/African American Young Adults
Authors: Collie Angel , Dadzie Adwoa , Master Lindsay , Reichenberger David , Mathew Gina , Chang Anne-Marie , Hale Lauren , Buxton Orfeu
Published: April 2024

Introduction

Perceived discrimination is associated with sleep health problems. This association is even stronger among Black/African Americans, who are typically exposed to more frequent discrimination in the US. Limited research examines how perceptions of discrimination impact young adults’ sleep health, particularly using objective measures. The current study investigated the association between perceived discrimination experiences and sleep health among Black/African American young adults.

Methods

Data from age 22 wave of the Future of Families and Child Wellbeing, Young Adult Sleep Study (YASS) included Black/African American young adults (n = 221) who self-reported experiences of discrimination due to their race/skin color, gender, and age. Wrist-worn actigraphy measures across ~2 weeks included sleep duration, sleep maintenance efficiency, and wake after sleep onset (WASO). Multiple linear regression models examined the association between perceived discrimination experiences based on personal attributes and dimensions of sleep adjusting for age, gender, and educational attainment.

Results

Among this sample of Black/African American young adults, perceived experiences of discrimination in their life were not associated with actigraphic sleep duration. Perceived discrimination attributed to race/skin color, age, or gender were not associated with sleep duration. Perceived experiences of discrimination in their life were also not associated with actigraphic sleep maintenance efficiency. Perceived discrimination attributed to age was associated with lower sleep maintenance efficiency (-3.01%, p<.001) and more WASO (+10.64 minutes/night, p=.004) compared to young adults who did not perceive age discrimination. Perceived discrimination attributed to gender was associated with lower sleep maintenance efficiency (-2.12%, p=.023) and more WASO (+9.47 minutes/night, p=.019), and were not different between women and men. Gender did not modify associations of sleep measures with perceived discrimination attributed to race/skin color or age.

Conclusion

Black/African American young adults’ perceived discrimination due to specific personal attributes (age and gender) are linked to lower sleep maintenance efficiency and more WASO. Future longitudinal research should examine whether perceived discrimination in young adulthood predicts poorer sleep health across time.

Support (if any)

R25-HL147668, R01HD073352, R01HD36916, R01HD39135, and R01HD40421
1216 Resolution of Headaches Related to Idiopathic Intracranial Hypertension After Treatment with Lemborexant
Authors: Sabbagh Riad Al , McCann Peter , Arjyal Sushrusha
Published: April 2024

Introduction

The orexin (hypocretin) system has attracted interest for its role in the regulation of multiple behavioral processes including feeding, sleep-wake cycle and reward-related behaviors. Dual orexin receptor antagonists (DORAs) target both orexin receptors 1 and 2 and these drugs currently have FDA-approval for insomnia treatment. The orexin system has also been implicated in pain processing through modulation of nociceptive pathways at the periaqueductal gray. Idiopathic intracranial hypertension (IIH) is characterized by evidence of raised intracranial pressure with or without papilledema without an underlying cause. Symptoms include chronic headaches, visual disturbance, and pulsatile tinnitus as well as back and neck pain. Management of IIH can be challenging and includes weight loss, carbonic anhydrase inhibitors and CSF shunting procedures all of which can have variable success in reducing headache frequency. Here we present a case of Lemborexant treatment for chronic insomnia with subsequent resolution of headaches related to IIH.

Report of case(s)

A 68-year-old male with a 25-year history of headaches secondary to IIH presented to the sleep medicine clinic for chronic insomnia. He had persistent headaches despite treatment with acetazolamide, zonisamide, nortriptyline and therapeutic lumbar punctures. He reported a history of insomnia for over 20 years. Polysomnography showed no evidence of OSA and actigraphy was consistent with his self-reported insomnia. He was started on mirtazapine for insomnia which was discontinued due to intolerance, and he was started on Lemborexant. When he was seen at follow up one month later, he reported improved sleep, achieving 7-8 hours of restful sleep per night. He also noted complete resolution of his daily headaches and had since stopped all medications required for his IIP treatment. This improvement in his IIH symptoms persisted during his two-month follow-up visit.

Conclusion

This is the first case report to our knowledge showing an improvement in headaches related to IIH in a patient prescribed lemborexant. The relationship between chronic pain and sleep disturbance is complex and there may be a subset of patients with chronic pain including headaches and insomnia who experience improvements in their pain through treatment of their sleep disturbance with DORAs.

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0122 Development and Initial Validation of the Danieli-Arizona Border Intergenerational Transmission of Stress Scale
Authors: Valencia Dora , Begay Tommy , Rasmussen Elizabeth , Ochoa Sylvia , Hernandez Edlin , Molina Patty , Carrillo Ivan , Grandner Michael A
Published: April 2024

Introduction

Intergenerational transmission of stress is an underrecognized potential contributor to sleep and mental health disparities among at-risk populations. No existing tool captures this construct as experienced at the US-Mexico Border.

Methods

The DABITSS was originally developed based on the Danieli Inventory of Multigenerational Legacies of Trauma developed for families of Holocaust survivors and was adapted to the border region through a series of focus groups. The end result is a scale in both English and Spanish that consists of 3 subscales: Parents (46 items, parental behaviors indicating transmission of stress), Adaptations (25 items, thoughts, feelings, and behaviors that represent internalization of stress), and Experiences (35 items, events that may induce intergenerational stress). Total scores for the Parents and Adaptations subscales range from 0-100, for a total score of 0-200. The Experiences scale ranges from 0-35. Scale and total scores were evaluated relative to insomnia (Insomnia Severity Index), depression (PHQ9 scale), and adverse childhood experiences (ACEs questionnaire). Models were adjusted for age, sex, ethnicity (Mexican or White), survey language (English or Spanish), immigrant status, and acculturation.

Results

DABITS scores on the Parents subscale ranged from 0.54-63.98 (M=28.99, SD=15.31); scores on the Adaptations subscale ranged from 5-65 (M=33.90, SD=14.51); scores on the Experiences subscale ranged from 0-20 (M=5.11, SD=3.57), and total scores [(Parents+Adaptations)/2] ranged from 10.54-123.06 (M=62.89, SD=26.27). Insomnia severity was associated with Parents (B=0.85, 95%CI [0.10, 1.60]), Adaptations (B=1.16, 95%CI [0.50,1.81]), and total score (B=2.01, 95%CI [0.76,3.25]). Depression was associated with Parents (B=1.09, 95%CI [0.25,1.93]), Adaptations (B=1.29, 95%CI [0.55,2.02]), Experiences (B=0.25, 95%CI [0.06,0.45]), and total score (B=2.38, 95%CI [0.99,3.77]). ACEs score was associated with Parents (B=2.91, 95%CI [1.15,4.67]), Adaptations (B=1.87, 95%CI [0.24,3.51]), Experiences (B=0.99, 95%CI [0.61,1.36]), and total score (B=4.79, 95%CI [1.78, 7.79]). Additional analyses are planned for other sleep (duration, quality, disorders, sleepiness) and mental health (anxiety, stress) measures.

Conclusion

The DABITSS scale represents multiple dimensions of intergenerational stress (how it was passed on, how it is experienced, and what may have triggered it). These dimensions are related to sleep and mental health.

Support (if any)

R01MD011600