bluuebunny/crossref_metadata_2025_split huggingface dataset:
0117 Emotional Support Is the Strongest Predictor of Sleep Disturbances Among a Sample of Perinatal Women of Color
Authors: Carrasco Mary , Blanc Judite , Garchitorena Laurent , Dimanche Stacyca , Hollimon Laronda , Gabbay Vilma , Scaramutti Carolina , Jean-Louis Girardin , Seixas Azizi 
Published: April 2024

Abstract

Introduction
Racial and ethnic minorities face a higher prevalence of maternal mental health conditions (e.g. postpartum depression and anxiety), compared to their White counterparts. Yet, studies have shown White women are more likely to get a diagnosis and treatment for postpartum depression compared to Black or Latina women. Lack of emotional and social support are known to be linked with poor health outcomes that are also associated with sleep. Yet, little is known about the direct relationship between emotional support and sleep health, especially among perinatal women of color.

Methods
This work utilized data from Nurturing Moms, a pilot study investigating the effect of a virtual reality program on maternal stress among expectant (0-36 weeks gestation) and postpartum (up to 12 months after birth) women of color. Participants (n=46) completed baseline surveys that captured psychological, emotional, behavioral, and sleep data. These surveys included Patient-Reported Outcomes Measurement Information System (PROMIS) measures of Emotional Support and Sleep Disturbance, among others. For the current study, we hypothesized that emotional support will be associated with sleep disturbance outcomes. Multilinear regression was performed using SPSS to determine if emotional support predicted sleep disturbance.

Results
Twenty-three expectant (29.3±4.8 years) and twenty-three post-partum (31.6±5.1 years) women completed online baseline surveys. Emotional support was found to be significantly associated with sleep disturbance in the expectant moms cohort [F(4,18) = 4.68, p=0.009] and in the post-partum mothers cohort [F(4,18) = 5.73, p=0.004]. The regression analysis indicates that lower emotional support predicted greater sleep disturbance (β1= -0.525) among expectant mothers and (β1= -0.388) among post-partum mothers, albeit a greater magnitude was observed among expectant mothers.

Conclusion
Emotional support was found to be strongly associated with sleep disturbance in a cohort of pregnant and post-partum women of color. With recent efforts geared toward addressing the maternal health crisis, this study highlights the importance of focusing on racial-ethnic minorities when investigating the impact of support on sleep health. Further studies exploring the relationship between emotional support and sleep among perinatal women of color are needed to implement interventions that will target maternal mental health in the United States.

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K01HL135452, R01HL152453, R01HL142066, R01HL095799, R01AG072644, RO1MD004113, R01AG067523, R01AG075007
1170 Iron and Modafinil Efficacy
Authors: Rabah Hussein , Trotti Lynn Marie , Saini Prabhjyot , Rye David 
Published: April 2024

Abstract

Introduction
Central diseases of hypersomnolence (CDH) are lifelong disorders characterized by excessive sleep and daytime sleepiness. Treatment options include modafinil, armodafinil, psychostimulants, pitolisant, clarithromycin, and gamma-hydroxy butyrate. Although the mechanism of action behind efficacy is poorly understood, modafinil is often the first-line therapy. Modafinil’s efficacy relies upon the integrity of brain dopamine (DA) signaling: it increases extracellular dopamine by inhibiting the dopamine transporter (DAT), and genetic deletion of the DAT renders modafinil Synaptic dopamine signaling is further highly dependent upon brain iron availability, leading us to consider whether systemic iron deficiency might impact the efficacy of common wake-promoting agents in CDH.

Report of case(s)
A 30-year-old woman diagnosed with Idiopathic Hypersomnia (IH) at the age of 22 self referred to our sleep center expressing concern over the loss of her modafinil’s wake-promoting benefits. Her PSG/MSLT had shown a sleep efficiency of 96%, a mean sleep latency of 4.6 minutes, and a single SOREMp on her 2nd nap of 5. She was prescribed modafinil (200mg QAM increased to BID over the past year), which provided symptomatic relief for nearly a decade. Her laboratory work revealed 5% transferrin saturation, low ferritin level ( 7 ng/ml), and normal CSF iron and orexin levels of 33 ng/ml and 261.8 pg/ml, respectively; therefore, she was started on oral iron supplements. At 2 weeks follow-up, serum transferrin saturation and ferritin levels increased to 23% and 17 ng/ml, respectively. Follow-up CSF orexin level was 266.6 pg/ml , and repeat assessments of CSF iron parameters are pending. After iron repletion, modafinil was restarted and titrated over one week to 200 mg in the morning and 100 mg in the afternoon. Subjective sleepiness improved, as evidenced by the patient regaining confidence in driving safely in excess of 70 minutes continuously. Iron deficiency negatively impacts sleep, vigilance, and motor programs in human infants and impairs synaptic dopamine signaling in animal studies. This could explain why the patient responded to Modafinil after iron was repleted.

Conclusion
Pre-anemic and anemic iron deficiency should be considered a potential readily treatable cause of modafinil’s lack of efficacy by way of effects upon wake-promoting brain dopamine circuits.

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1171 Hypersomnia Secondary to Long Covid
Authors: Peth Dustin , Ibrahim Sally 
Published: April 2024

Abstract

Introduction
Recognition of Long Covid is increasing. In a recent meta-analysis in youth with Long Covid, the most prevalent clinical symptoms were mood (16.50%), fatigue (9.66%), and sleep disorders (8.42%). However, there is a paucity of data in the management of these symptoms.

Report of case(s)
We present a case of severe hypersomnolence after a mild Covid-19 infection in a 13 year-old female with no significant history, an acceptable age-appropriate sleep-wake schedule without nighttime awakenings or sleepiness prior to infection. Post-infection, she developed a one year history of sleep onset and maintenance insomnia, significant circadian delays, sleep inertia, brain fog and headaches. She was referred to sleep medicine and responded to treatment for a circadian rhythm disorder with sleep-wake restructuring, melatonin, and strict sleep hygiene. Sleep fragmentation, unrefreshing sleep, and hypersomnolence persisted; ESS=15/24. Sleep duration was often ≥12 hours in a 24 hour period. The patient could not wake easily, and often missed school or left early for sleepiness and headaches. A trial of gabapentin 200mg at bedtime failed. Sleep diagnostics were performed. Polysomnography showed no sleep disruptors, 8 hours of sleep, and some delayed slow wave sleep periods. Mean sleep latency testing (MSLT) showed MSL of 15.4min; 1 SOREM (nap #1). With a working diagnosis of hypersomnia due to medical condition vs. idiopathic hypersomnia, methylphenidate up to 36mg ER was trialed with some improvement. A recent adult case with Long Covid and ADHD-like syndrome also responded to stimulant use, though no guidelines are established for Long Covid. Oxybate was later added up to 2g once nightly which helped resolve sleep inertia, improve sleep quality and reduce sleepiness(ESS=4/24). She was able to return to school full time and reached normal functioning.

Conclusion
While the pathophysiology of Long Covid is still under investigation, symptom management of sleepiness can impact functioning. This case demonstrates a successful treatment of a patient with Long Covid and hypersomnolence with resolution of excessive sleepiness, morning inertia, and sleep fragmentation with a treatment approach similar to that of idiopathic hypersomnia, using an oxybate along with daytime stimulant use.

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1172 Hoops by Day, Ventilator by Night
Authors: Almeda Marco , Bauer Sarah , Bandyopadhyay Anuja 
Published: April 2024

Abstract

Introduction
Selenoprotein N-related myopathy (SEPN1-RM) is a rare congenital disorder often presenting in infancy or early childhood. Clinical sequelae frequently include progressive muscle weakness, spinal rigidity, respiratory insufficiency, and the need for nocturnal ventilatory support. Here, we describe a teenage athlete who presented with hypercarbia in the sleep lab, required tracheostomy for chronic mechanical ventilation, and was ultimately diagnosed with SEPN1-RM.

Report of case(s)
We present a 15-year-old male with a remote history of scoliosis repair and obstructive sleep apnea (apnea-hypopnea index [AHI] of 11.1/hour) with subsequent tonsillectomy and adenoidectomy at age 2 years. He had done well until age 15 years when he was referred to Sleep Medicine for morning headaches and daytime sleepiness. At evaluation, he reported working a physically demanding after-school job and playing on the basketball team without any issues. Repeat polysomnography showed an AHI of 88.8/hour, hypoxemia, and hypoventilation with an average transcutaneous carbon dioxide level of 82mmHg, prompting admission to the intensive care unit for further management. After failing non-invasive ventilation due to persistent hypercarbia, he underwent tracheostomy for nocturnal mechanical ventilation. He otherwise tolerated room air during the day. Other notable work-up included evidence of pulmonary hypertension on echocardiogram, oropharyngeal collapse on bronchoscopy, and restriction on pulmonary function tests. Whole exome sequencing eventually revealed SEPN1-RM. The patient was discharged after ventilator training and continues to follow outpatient.

Conclusion
To date, there is limited understanding on the pathophysiology and clinical course of patients with SEPN1-RM. Our patient’s development of severe obstructive sleep apnea necessitating mechanical ventilation, despite grossly preserved neuromuscular strength and a relatively late onset, does not follow the typical course of other pediatric neuromuscular disorders. His course adds to a small cohort of patients with this disorder and demonstrates its potentially wide clinical spectrum, suggesting further research is needed to better understand this disease process.

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None.
1173 Unmasking Severe OSA After Transvenous Phrenic Nerve Stimulation for Central Sleep Apnea
Authors: Jain Nikhita , Ibrahem Marwah , Rapoport David , Hiensch Robert 
Published: April 2024

Abstract

Introduction
In the absence of reversible etiology, central sleep apnea (CSA) may be treated with positive airway pressure (CPAP or BPAP), adaptive servo-ventilation, or supplemental oxygen. In 2017, a transvenous phrenic nerve stimulator (PNS), the remedē System™, was FDA-approved for moderate-to-severe CSA. Five-year outcomes data confirmed the device’s sustained efficacy, with improvements in symptoms, apnea-hypopnea index(AHI), and central apnea index(CAI). While the device effectively treats CSA, unrecognized upper airway obstruction may hinder clinical success. This case report describes a patient whose polysomnography (PSG) suggested candidacy for PNS; however, his airway collapsibility led to severe obstructive sleep apnea(OSA), unmasked after PNS implantation.

Report of case(s)
Our patient is a 74-year-old with longstanding severe CSA and chronic insomnia. He has a history of spasmodic dysphonia status post recurrent laryngeal nerve ablation. His unilateral vocal fold paralysis was followed by injection medialization for voice augmentation. He is also an asymptomatic carrier of myotonic dystrophy(MD), identified after his grandchild was diagnosed with MD. He presented to our institution for daytime sleepiness, disrupted sleep and snoring with complaints of PAP intolerance. PSG revealed an AHI4% of 49.1 events/hour with 0 obstructive apneas, 89 central apneas, and 53 unclassified hypopneas. Transthoracic echocardiogram was unremarkable. Pulmonary function tests were normal without evidence of neuromuscular weakness or extrathoracic obstruction. He underwent PNS implantation. Postoperatively, the patient continued endorsing unrefreshed sleep. His PNS titration study demonstrated prolonged flow limitation and an AHI4% of 35.2 events/hour with frequent obstructive events. He is now using a mandibular advancement device (MAD) with the PNS.

Conclusion
This case highlights the impact of upper airway collapsibility on PNS treatment response. Although our patient’s history may have indicated greater susceptibility to airway collapse, preoperative PSG did not reveal unequivocal OSA. However, frequent obstructive hypopneas were unmasked after PNS implantation. This patient’s upper airway collapse was underrepresented on PSG, and increased negative intrathoracic pressure during the PNS titration may have worsened the severity. Since differentiating central from obstructive hypopneas can be challenging, PNS may not be ideal for CSA patients with increased hypopnea burdens. If OSA emerges postoperatively, adjuncts like MAD, CPAP, or hypoglossal nerve stimulation may stabilize the upper airway.

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1174 Oromandibular/Faciomandibular Myoclonus; A Rare Sleep-Related Movement Disorder Captured on Polysomnography
Authors: Kramer Joshua , Garwood Mark 
Published: April 2024

Abstract

Introduction
Oromandibular myoclonus (OMM) also known as faciomandibular myoclonus (FMM) is a rare neurological sleep-related movement disorder characterized by involuntary and repetitive contractions of the facial muscles. Consequences of this disorder include nocturnal tongue and cheek biting, which can mimic signs of seizure disorders. This case report presents a patient with OMM/FMM which was captured on polysomnography.

Report of case(s)
The patient is a 25-year-old male with a medical history that includes hypertension, obesity (BMI 67), bipolar disorder, and headaches. He presented with the chief complaint of excessive daytime sleepiness. He also endorsed waking up with a sore tongue on nearly a monthly basis without obvious reason. He underwent a split-night polysomnogram with transcutaneous CO2 (TcCO2) monitoring which showed severe obstructive sleep apnea (apnea-hypopnea index of 114.6) and sleep related hypoventilation with TcCO2 levels greater than 55 mm of Hg for more than 10 consecutive minutes. During the titration portion of the polysomnogram, several very brief (less than 250 milliseconds) stereotypical jerk-like contractions of his jaw muscles with characteristics of myoclonus were observed during REM sleep. These occurred as isolated bursts and clusters of irregular bursts of three to five repetitive contractions. Increased amplitude in the submentalis EMG leads, as well as muscle artifact in the EOG and EEG channels were identified during events. EEG showed no epileptiform findings. REM sleep atonia was otherwise preserved.

Conclusion
Oromandibular/Faciomandibular myoclonus is a sleep-related movement disorder characterized by very brief (less than 250 milliseconds) forceful myoclonus of masticatory muscles in sleep. These can occur either as isolated bursts or as a cluster of regularly or irregularly occurring brief bursts. Pathophysiology is considered to be a form of brainstem reticular myoclonus, involving a circuitry of cranial nerve V and VII nuclei along polysynaptic pathways. Tongue biting is widely recognized as a useful clinical diagnostic tool for differentiating between epileptic seizures and nonepileptic events. OMM/FMM typically presents with recurrent nocturnal tongue biting, which can be misdiagnosed as nocturnal seizures. Therefore, appropriate awareness and recognition is important to avoid an epilepsy misdiagnosis which may result in lifelong anti-seizure medication use.

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1175 Alternative Treatment for REM-Sleep Behavior Disorder
Authors: Ballenberger Matthew , Koo Brian 
Published: April 2024

Abstract

Introduction
Rapid Eye Movement (REM) Sleep Behavior Disorder (RBD) is a parasomnia occurring during REM sleep, characterized by loss of normal REM sleep atonia and dream enactment behaviors. The majority of patients diagnosed with idiopathic RBD will develop an alpha-synucleinopathy neurodegenerative disease, which can complicate the treatment of RBD. First-line treatment with high-dose melatonin or clonazepam can have side effects of daytime sleepiness, dizziness, or nightmares, which can increase risk of harm to patients. This has led some patients to seek alternative treatments. We report a patient who successfully treated symptoms of RBD with edible tetrahydrocannabinol (THC) gummies.

Report of case(s)
A 67-year-old man presented to sleep clinic with symptoms of daytime sleepiness and dream enactment behavior including yelling, punching and kicking. He underwent a diagnostic polysomnogram that revealed moderate obstructive sleep apnea (OSA) but did not confirm dream enactment behavior or RSWA. He was diagnosed with RBD based on clinical criteria and started CPAP and melatonin 10 mg with initial reduction of dream enactment. At a subsequent visit he reported increased frequency of dream enactment behaviors with morning grogginess which caused him to stop melatonin and start marijuana gummies with 4.2 mg of THC purchased over-the-counter. Since starting this, his wife had reported complete resolution of dream enactment behavior.

Conclusion
Current first-line medications used to treat symptoms of RBD can cause unfavorable side effects of dizziness and daytime sleepiness which can negatively impact patients’ quality of life and compound progressive symptoms of neurodegeneration. Recent studies involving cannabinoids for RBD trialed the use of Cannabidiol (CBD), but showed no reduction in frequency of dream enactment symptoms. Our case illustrates an example of successfully using THC as an alternative treatment for dream enactment behavior, which may be more well-tolerated than first-line agents and is readily available over-the-counter in many states. A possible mechanism for the improvement in symptoms may be related to THC-predominant cannabis mediated reduction in total REM sleep and REM density. More studies are needed to determine if THC can be used as a viable alternative for treatment of RBD.

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1176 Extended Polysomnography Confirms Idiopathic Hypersomnia Diagnoses: A Case Series
Authors: Mouchati Christian , Alsheikhtaha Zahreddin , Foldvary-Schaefer Nancy 
Published: April 2024

Abstract

Introduction
Idiopathic hypersomnia (IH) is often underdiagnosed since at least 30-40% have normal mean sleep latency tests (MSLT) and test-retest reproducibility is poor. We report two cases of IH with multiple normal sleep evaluations diagnosed at Cleveland Clinic through extended polysomnography (PSG) using a 32-hr bed rest protocol as published by Evangelista et al., 2018.

Report of case(s)
Case 1: A 28-year-old female reported daytime sleepiness (Epworth Sleepiness Scale (ESS) 15), fatigue, sleep inertia, habitual sleep duration of 10-14 hr, brain fog and rare sleep-related hallucinations with symptom onset of 10 yr of age. PSG/MSLT evaluations at ages 13, 22 and 26 yr were normal, with MSLT mean sleep latencies (MSL) of 12.9, 15 and 16.8 minutes and 0 SOREMPs after normal PSG with adequate sleep. On extended PSG, total sleep time (TST) was 19.3 hr (1155 min) on the 32-hr recording and 13.1 hr (785 min) on the 24-hr recording. Case 2: A 42-year-old female reported excessive sleepiness (ESS 22), sleep inertia, memory and attention complaints, and rare sleep paralysis associated with sleep-related hallucinations beginning in the 2nd decade of life. Habitual sleep duration was 9.5 hr. PSG/MSLT evaluations at 40 and 41 yr were normal, with MLST MSL of 14.2 and 12.8 min and 0 SOREMPs and negative urine toxicology following normal PSG with adequate sleep and nocturnal sleep of 8-8.5 hr on actigraphy. On extended PSG, TST was 19.1 hr (1147 min) on the 32-hr recording and 11.7 hr (704 min) on the 24-hr recording.

Conclusion
We report two cases with IH phenotypes, multiple normal PSG/MSLT evaluations, and debilitating symptoms for over two decades. Both met the 19-hr cutoff for the diagnosis of IH using the 32-hr bed rest protocol based on published criteria and one met the 12-hr cut-off using the 24-hr protocol. This work illustrates the limitations of current IH diagnostic criteria and underscores the need to establish more accurate diagnostic test modalities for IH given the lack of biomarkers.

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1178 CASE REPORT – USE of WEARABLE OXIMETRY DEVICE to TITRATE HYPOGLOSSAL NERVE STIMULATION SETTINGS
Authors: Chaudhary Hira , Tashman Yasemin 
Published: April 2024

Abstract

Introduction
Obstructive sleep apnea (OSA) is characterized by repetitive airway narrowing or collapse during sleep. Hypoglossal nerve stimulation (HNS) is one alternative therapy. After device implantation and activation, it is patient-titrated based on symptoms. A sleep study then confirms effective settings based on an apnea-hypopnea index (AHI) below 5. Symptomatic control alone may not entail low AHI. Objective data with overnight oximetry during titration may help bridge this. We describe a patient in whom Wellue pulse oximeter was used during self-titration of the Inspire HNS.

Report of case(s)
A 63 year-old male presented in June 2022 for HNS evaluation due to gasping and witnessed apneas. AutoPAP trial had failed. In November 2022 diagnostic polysomnography revealed an AHI of 18 and lowest oxygen saturation (OS) of 80%. Sleep endoscopy in February 2023 confirmed no concentric collapse at the level of the velum. He underwent HNS implantation in May 2023 and activation in June. Initial sensation and functional thresholds were 0.6 volts. Amplitude range was 0.6 to 1.6 volts, pulse width 90 microseconds, rate 33 hertz, pause time 15 minutes, electrode configuration [+,-,+], and duration 8 hours. In July, he felt 0.7 volts controlled symptoms. A Watchpat home sleep study revealed an AHI of 46.2 and lowest OS of 82%. He purchased a pulse oximeter and was shown the oxygen desaturation index (ODI) at &gt;3%. On increasing to 1.0 volts, oximetry revealed ODI 8.65 averaged over 4 nights. At 1.1 volts, ODI improved to 0.9-4.14. Since ODI was below 5, he had subsequent in-laboratory titration, revealing at 1.1 volts the residual AHI was 2.5. The lowest OS was 89% with a mean of 95%.

Conclusion
The post-activation procedure for HNS utilizes symptom-based self-titration. Sleep studies may reveal continued apnea despite self-reported symptom control. We suspect overnight oximetry during self-titration may improve efficacy. In this case, after self-titration with oximetry to decide an amplitude setting, home and in-laboratory sleep studies confirmed effective control. This preliminarily suggests that overnight oximetry used with HNS during the self-titration phase may improve titration outcomes as confirmed by formal polysomnography.

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