bluuebunny/crossref_metadata_2025_split huggingface dataset:
1201 A Drowsy Deployment Dilemma: Case Report of a Military Deployment-Induced Kleine-Levin Syndrome Episode
Authors: Sickbert Lydia , Cocozza Victoria , Tchopev Zahari , Brock Matthew , Frey William 
Published: April 2024

Abstract

Introduction
Kleine-Levin syndrome (KLS) is a rare central disorder of hypersomnolence that typically develops in adolescent males and is accompanied by behavioral and cognitive disturbances such as altered perception or disinhibited behavior. The underlying etiology of this disease is unknown. It has a benign clinical course that often resolves after a median of fourteen years.

Report of case(s)
A 24-year-old male with an unremarkable past medical history was deployed by the United States (U.S.) Air Force to Africa. Patient had an episode of extreme hypersomnolence during his deployment associated with hearing abnormalities, anterograde amnesia, and irritability. He endorsed sleeping 20 hours straight – only waking to eat and use the restroom. He ultimately required medical evacuation to Germany. In Germany, he had an unremarkable neurology evaluation, laboratory studies, electroencephalogram (EEG), and brain magnetic resonance imaging (MRI). Upon further questioning, the patient’s first episode occurred three years ago without an identifiable trigger. Subsequent episodes lasted 1-4 weeks and recurred every 3-4 months. He was asymptomatic between episodes. Upon returning to the U.S. the patient was evaluated by sleep medicine, and a clinical diagnosis of KLS was made. The following day, the patient returned to the sleep center escorted by his work supervisor with an acute onset of hypersomnia. A multiple sleep latency test (MSLT) was performed with modifications (no polysomnography (PSG) or actigraphy prior and four naps). The patient demonstrated a mean sleep latency of 12 minutes and 37 seconds, which does not meet criteria for hypersomnolence. The patient was sent home after the MSLT with actigraphy for 2 weeks. He did not wear the device enough to allow for interpretation.

Conclusion
This is the first case report describing a military deployment-induced KLS episode. KLS is a clinical diagnosis of exclusion that does not require objective confirmation. The majority of available KLS literature states that objective measurements, such as MSLT and PSG, are not useful for diagnosis. However, KLS studies have limitations due to rarity of disease and lack of testing during episodes. We propose that the diagnostic criteria for KLS be re-evaluated to include objective data such as MSLT.

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1202 Worsening of Obstructive Sleep Apnea Following Resection of a Vagal Nerve Schwannoma
Authors: Chohan Moeed , Avidan Alon 
Published: April 2024

Abstract

Introduction
Schwannomas (SN) arising from the vagus cranial nerve (CN X) are uncommon (2–5%) benign nerve tumors. SN may promote obstructive sleep apnea (OSA) when parapharyngeal schwannoma narrows the airspace or as a direct result of injury to the recurrent laryngeal nerve which leads to vocal cord paralysis. We present an unusual presentation of worsening OSA in a patient who presented with CNX SN, underwent resection of the tumor, and developed unilateral vocal cord paralysis.

Report of case(s)
A 47-y/o male with a history of mild OSA s/p UPPP, presented with a new neck mass, worsening snoring, apneic episodes, fragmented nocturnal sleep, and worsening hypersomnolence. He was diagnosed with CN X SN and underwent left modified radical neck dissection for removal of a left-sided carotid mass which extended to the distal branches of the recurrent laryngeal nerve (RLN). Postoperatively, the patient exhibited dysphonia, dysphagia, and severe gasping at night attributed to left-sided vocal cord paralysis (VCP). Baseline Polysomnography (PSG) at age 32 before UPPP: AHI: 9 (REM:15, Supine: 18), SaO2: 89%. PSG following SN resection: AHI: 42 (REM: 21, Supine: 42), SaO2: 77%. The patient was prescribed autoPAP which resolved his symptoms.

Conclusion
The literature provides evidence of iatrogenic VCP, as a significant contributor to OSA. To our knowledge, this is the first report of VCP linked to SN of CN X. Multiple mechanisms may contribute to the OSA in our patient. (1) Resection of the CN X SN complex, including the distal branches CN X which lies near the RLN and may have been severed resulting in unilateral VCP and ultimately airflow limitations. (2) Resection of the CN X SN complex may have also altered neuronal pathways originating in the respiratory control centers in the brainstem. This mechanism, while somewhat elusive, highlights the potential role of vagal sensory innervation of the respiratory tract and provides fundamental insights into how derangement of afferent neuronal control modulated through CNX contributes to disorders of respiratory control.

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1204 Importance of Arm Electromyography for REM Without Atonia Detection
Authors: Das John , Johal Arminder , Stahl Stephanie 
Published: April 2024

Abstract

Introduction
Dream enactment raises concern for rapid-eye movement (REM) sleep behavior disorder (RBD). RBD is a parasomnia during which the patient has loss of normal atonia during REM sleep. RBD is highly associated with α-synucleinopathies (i.e., Parkinson disease, dementia with Lewy bodies, and multiple system atrophy) and can precede additional neurodegenerative signs and symptoms. REM sleep without atonia (RWA) is demonstrated through electromyography (EMG) during polysomnography. RWA requires the presence of tonic/sustained activity with ≥50% of the epoch containing excessive chin activity or phasic/transient muscle activity in ≥5 mini-epochs of 3-second duration in a 30-second epoch. We present a case that highlights the necessity of ordering polysomnography with arm EMG when evaluating for RBD.

Report of case(s)
A 73-year-old man presented to the clinic for concern of obstructive sleep apnea. He was also evaluated by neurology and given a diagnosis of essential tremor. During the initial visit, he also reported frequent “night terrors,” which he later described as dream enactment. His medications included metoprolol but no antidepressants; however, the dream enactment started prior to beta-blocker use. His physical exam was notable for right cogwheel rigidity, mild decrease in right serial finger tapping and rapid alternating movements, and mild dysphonia. A polysomnogram with arm EMG was ordered. On polysomnography, RWA was present in multiple epochs that was seen predominantly with arm (flexor digitorum superficialis) EMG. The RWA index (% of REM sleep meeting criteria for RWA) using the leg (anterior tibialis) and chin EMG was only 5%; however, the RWA index with the addition of arm EMG increased the RWA index to 64%.

Conclusion
The presence of RWA on PSG with dream enactment behavior gives this patient the diagnosis of RBD. While leg EMG is a part of routine PSG, arm EMG is not and needs to be specifically requested. Although research supports the use of arm EMG leads, we commonly find the addition of arm EMG leads is not ordered by providers when assessing for RBD. Arm EMG increases the chance of observing RWA and can be of strong importance to solidify an RBD diagnosis.

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1205 Haunted Slumbers: Decoding a Trauma-Induced Sleep Disorder in a Teen Patient
Authors: Iriana Sidney , Ibrahim Sally , Peth Dustin , Mistry Shikha , Lisio Catherine Di , Strohl Kingman 
Published: April 2024

Abstract

Introduction
This case examines a complex inpatient sleep presentation of a teenager with a history of foster care, depression, anxiety, PTSD, and ADHD, presenting with features of parasomnia. Key symptoms included excessive sleepiness, waking hallucinations, partial awareness, and significant uncontrolled movements reaching a level of severity requiring hospitalization.

Report of case(s)
A 14-year-old female presented with severe nocturnal episodes arising from sleep characterized by hallucinations of a dark figure, observed limb thrashing, screaming, and impaired arousal that gradually intensified over a 5 month period. Despite treatment for PTSD and an SSRI, her condition worsened - escalating to a severe and prolonged episode requiring EMS and hospitalization for her intense, potentially harmful movements. Concerns of parasomnia prompted inpatient sleep medicine consultation. Detailed history indicated these spells occurred primarily in early morning hours during sleep-wake transitions, with the patient having only partial recall of her movements. Each episode involved fear and visual hallucinations of threatening figures that she could recall later. Significant sleep disruption and daytime sleepiness were noted. Her psychiatric history revealed trauma from foster care. She also reported a fascination with horror media which seemed to influence these disturbances, reflecting in recurrent nightmares and similar traumatic experiences. Video EEG was negative for seizure disorder; PSG showed very mild OSA, REM with appropriate atonia, and sudden brief awakenings in both NREM and REM sleep. No spells were observed. Due her parents safety concerns, she stayed in a residential facility for intensive outpatient psychiatric therapy. Her episodes gradually improved with sleep scheduling, optimization of medications, and counseling. In outpatient therapy, she reported a swift cessation of the episodes, marked by her intentional disregard of the hallucinatory figure from her experiences.

Conclusion
This case highlights trauma-induced sleep disturbances in a young patient requiring hospitalization and inpatient sleep medicine consultation. The resolution of symptoms with changes in medication, improved sleep duration and hygiene in a residential setting underscores the importance of a comprehensive approach to managing complex sleep spells in adolescents with mood disturbance.

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1208 A Case of Complex Sleep Apnea Improved with Cardiac Resynchronization Therapy
Authors: Bhardwaj Puja , Naik Sreelatha , Suryanarayanana Manoj 
Published: April 2024

Abstract

Introduction
Congestive heart failure (CHF) is associated with higher risk of both obstructive (OSA) and central sleep apnea (CSA). While PAP therapy remains the gold standard in the treatment of obstructive sleep apnea, management of CSA needs to be tailored to the underlying pathophysiology. Optimal medical therapy for CHF is essential in the management of sleep-disordered breathing (SDB) among patients with CHF and reduced left ventricular ejection fraction (HFrEF). Cardiac resynchronization therapy (CRT) may have an important role.

Report of case(s)
We present the case of an 80 year old male, BMI 29 g/m2 who was evaluated at our sleep disorders center for the management of sleep apnea treated with CPAP 6 cm H2O and comorbid with HFrEF (ejection fraction 35% at the time of presentation) Therapy data from his CPAP device showed elevated residual AHI due to a combination of obstructive and central apneas as well as hypopneas. Significant leak and Cheyne-Stokes respiration were not detected. An empiric pressure increase was made as he was reluctant to return for PAP re-titration. He was also being treated with medical therapy for CHF and underwent CRT several months after presentation to the sleep clinic. Upon follow up he was found to have a dramatic improvement in residual events (AHI 31.2/hr- 6.7/hr) without further changes to CPAP. HFrEF increases risk of CSA through a vicious cycle stemming from significantly lower arousal threshold, high loop gain resulting in an exaggerated ventilatory response to arousals, and prolonged circulatory time leading to mismatched communication of arterial gas concentrations with the chemoreceptors. CHF may independently increase risk of OSA due to vascular engorgement of neck vessels that might affect upper airway patency especially among patients with hypervolemia. The terminal manifestation of a central apnea might be an occlusion of the pharyngeal airway requiring positive pressure to overcome the obstruction. This would explain why CPAP therapy might be effective among patients with HFrEF.

Conclusion
We conclude that the improvement in SDB in our patient is attributable to optimal medical therapy leading to euvolemia, CRT and medical therapy potentially improving cardiac systolic function and PAP therapy maintaining upper airway patency.

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1210 Initiation of Methadone for Refractory Restless Leg Syndrome with Significant Improvement of Symptoms
Authors: Damia Carla , Monceaux Brittany , Hall Catherine , Bhamrah Paul , Koothirezhi Rupa , Liendo Cesar , Ho Minh Tam , Ghildiyal Naomi , Chernyshev Oleg 
Published: April 2024

Abstract

Introduction
Restless legs syndrome (RLS) is a common sleep-related movement disorder. We are reporting a case of progressive and refractory RLS in which a patient had a long history of poorly controlled and worsening RLS with intolerance to first line medications and incomplete management of symptoms.

Report of case(s)
Patient is a 62-year-old Caucasian female with a past medical history of RLS, mild obstructive sleep apnea (diagnosed 2021), insomnia (managed with zolpidem), atrial fibrillation, coronary artery disease, obesity, diabetes, hypertension and osteoarthritis. Initial presentation to our clinic was 2 years ago. She reported RLS symptoms 20 years prior with treatment initiated 15 years ago. Symptoms started to worsen 5-6 years ago. She reported sensations in the upper extremities and symptoms starting earlier in the day. She noted intolerance, side effects to and ineffectiveness of first-line medications. Pregabalin produced oral blisters and gabapentin sedated her with minimal benefit to leg sensations. She was on ropinirole 0.25mg in the AM and 2mg in the evening. Dosage was increased but patient found that increased dosage during the day caused sedation and did not alleviate symptoms. Ferritin was low and patient had been on oral iron without change in symptoms. Iron infusion was recommended but deemed not feasible. After the first visit, patient presented 3 months later. She had required an emergency cholecystectomy and was given hydromorphone and morphine while hospitalized. These medications completely alleviated symptoms for several days. Patient had taken other opioid medications including tramadol, hydrocodone and oxycodone in the past without any effect on symptoms. Patient was continued on ropinirole 2mg at night and started on methadone 5mg daily. She has noted a significant improvement in symptoms with almost complete alleviation. She denies any side effects from this regimen and has been stable on this regimen for almost 2 years without worsening symptoms.

Conclusion
This case focuses on the benefit of specific opioids for management of refractory RLS. Initial selection of an opioid may not produce benefit but evaluating response to different medications and tailoring a treatment plan for each individual patient could produce notable alleviation of debilitating symptoms that occur with progressive and refractory RLS.

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1211 Pressure Injury of the Face After an Overnight Titration Study
Authors: Yaqoob Zaid , Jaffery Syed , Bazan Luisa 
Published: April 2024

Abstract

Introduction
We present a case of facial pressure injury related to a CPAP mask occurring within 24 hours of an overnight titration study, an uncommon finding.

Report of case(s)
Patient is a 69-year-old man with prior history of OSA who presented to the Henry Ford Sleep Medicine Clinic for reevaluation. His past medical history included class III obesity, diabetes mellitus, hypertension, coronary artery calcification, dyslipidemia and 88 pack-year history of smoking, he quit in 2010. When he was first diagnosed with OSA, he was treated with PAP therapy for 4 years without any reported skin adverse effects. He stopped using PAP therapy for 5 years prior to presentation to our clinic. A home sleep apnea test showed severe OSA with sleep related hypoxia. He underwent a titration study with the use of F&P Evora full face. CPAP was transitioned to BiPAP during the titration due to persistent hypoxia. He felt as though the plastic edge of the mask had cut into his skin during the study. Within 24 hours he noticed facial puffiness, tenderness, redness and facial wounds with increasing purulent discharge. He was seen in the sleep clinic the following day and was prescribed sulfamethoxazole-trimethoprim for a 7-day course for unstageable pressure injury to the face, which soon resolved. Patient was seen by allergy and an allergic reaction to the mask was ruled out. Further work up of hypoxia showed restrictive lung disease on pulmonary function testing and sniff test showed right diaphragmatic weakness.

Conclusion
Pressure injury to the face within 24 hours of using a mask is an uncommon complication of overnight titration studies. To our knowledge, this is the first reported case after using the F&P Evora full face mask which could have been the triggering factor for this type of injury. We suggest proper mask fitting during titration to minimize facial pressure injuries in the sleep lab.

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1212 Higher Is Not Always Better
Authors: Chang Stephanie , Seo Jiyeon , Rojanapairat Oragun 
Published: April 2024

Abstract

Introduction
Positive airway pressure (PAP) is the recommended first line treatment for obstructive sleep apnea (OSA). Overnight titration studies can be performed to find the optimal PAP settings for patients. However, manual titration studies can lead to higher PAP settings.

Report of case(s)
Cases: A 69-year-old man with a body mass index (BMI) of 33 and severe OSA on continuous PAP (CPAP) 10 cm of H2O was referred to sleep clinic for worsening daytime fatigue. He had an in-lab titration study with a full-face mask and was successfully titrated to bilevel PAP (BPAP) with a final inspiratory PAP (IPAP) of 20 cm of H2O and expiratory PAP (EPAP) of 16 cm H2O. Notably during his sleep study, CPAP improved the AHI up to 14 cm H2O, but further increase in CPAP and switching to BPAP worsened the AHI until a final setting of BPAP 20/16 cm H2O was reached. A 75-year-old man with a BMI of 20 and moderate OSA was referred to sleep clinic for persistent daytime sleepiness despite compliant auto-CPAP use with nasal pillows. He underwent an in-lab titration study with a full-face mask and was titrated to BPAP settings of 22/17 cm of H2O. Review of his sleep study showed effective therapy at CPAP of 6 cm of H2O, but further titration on CPAP and BPAP worsened the AHI until a final setting of BPAP 22/17 cm of H2O was reached. Discussion: PAP settings are an important determinant for PAP compliance as patients often do not tolerate higher settings. Manual titration studies have shown to overestimate CPAP requirements compared to the use of auto-titrating PAP in uncomplicated OSA. Furthermore, full-face masks can paradoxically worsen upper airway obstruction and increase treatment pressures compared to nasal masks. It is thought that full-face masks can cause worsening of upper airway obstruction and lead to higher pressure requirements to pneumatically stent the airways.

Conclusion
In conclusion, manual PAP titration studies may exaggerate PAP requirements, especially with the use of full-face masks. Careful review of the titration and choice of mask should be an important consideration in treating OSA.

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1213 Vagal-nerve Stimulation Induced Refractory Central Sleep Apnea
Authors: Shetty Amit , Thomas Robert , Heckman Eric 
Published: April 2024

Abstract

Introduction
Vagus nerve stimulation (VNS) is an effective therapy for epilepsy that has previously been suggested to worsen obstructive and central sleep apnea. Management of medically complex patients in the traditional sleep laboratory can present logistical barriers given the minimal medical oversight. Innovative forms of sleep monitoring offer new options to evaluate sleep serially.

Report of case(s)
A 69-year-old female with a history of medically refractory generalized epilepsy, on a vagal nerve stimulator, presented to sleep clinic in September of 2022 after undergoing a polysomnogram significant for severe sleep apnea with AHI 3% of 56, AHI 4% of 48, and oxygen nadir of 83%. A November 2022 titration was unsuccessful (residual AHI of 48 and oxygen desaturation nadir of 88%). Periodic breathing proved refractory to CPAP, BiPAP, BiPAP S/T, adaptive servo-ventilation, and attempts to mitigate high loop gain physiology with CO2 modulation techniques, acetazolamide, and supplemental oxygen. VNS setting could not be manipulated in the sleep laboratory safely. She was placed on CPAP setting of 5-10 cm H2O with compliance data showing average residual AHI of 24.3 and 29% of nights being in Hunter-Cheyne-Stokes breathing (HCSB). A planned hospital admission in July 2023 was arranged to monitor sleep with the SleepImage Ring system, which offered measurement of AHI and periodicity without the need for manual scoring. The VNS battery proved depleted during the stay but 5 nights on the same CPAP settings with the VNS off showed a residual AHI 4% of 1-3 with minimal periodic breathing. Patient underwent replacement of VNS generator with reduction of output current to 1/3 of prior value with gradual up-titration. Subsequent CPAP compliance data showed gradual increase in AHI and periodic breathing with rising VNS setting. Currently, the patient is undergoing VNS adjustments to optimize seizure control with plans to restudy sleep breathing at lower nocturnal settings when stabilized.

Conclusion
Sleep apnea induced by VNS can be refractory to therapy. Tracking sleep with the SleepImage system and PAP compliance data allowed understanding of the role of neurostimulation in inducing sleep dysfunction.

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Institute for Personalized Sleep Health, Beth Israel Deaconess Medical Center