bluuebunny/crossref_metadata_2025_split huggingface dataset:
1019 New onset OSA after long term Vagus Nerve Stimulator implantation in a patient with Lennox-Gastaut syndrome: A case report
Authors: Patel Harshana , Fatima Maryam , Naik Sreelatha 
Published: May 2023

Abstract

Introduction
Implantable vagus nerve stimulation (VNS) is an established treatment for drug resistant epilepsy. New onset obstructive sleep apnea (OSA) is a rare side effect of VNS. There are few studies describing VNS induced sleep apnea in adults. [1-2] A study of 16 patients with drug-refractory epilepsy found 5 patients with normal AHI develop mild OSA after VNS treatment for 3 months. [2] However, no literature exists regarding OSA after long term VNS implantation. Untreated OSA in patients with epilepsy can impact epilepsy outcomes. We report a case of new onset OSA after long term VNS implantation, refractory to PAP therapy.

Report of case(s)
A 36 year-old man with Lennox-Gastaut syndrome presented with gasping during sleep and daytime hypersomnolence. He underwent VNS implantation at the age of 12. A prior PSG in 2018 showed no sleep apnea. Repeat PSG showed VNS-induced OSA (AHI 10.1/hour). After failed CPAP titration, BPAP-S at 25/16 cm H2O improved OSA. Review of therapy data on follow up showed AHI of 13.7/hour and CAI of 10.9/hour, which is concerning for treatment induced central sleep apnea on BPAP. Diagnostic study with VNS turned off revealed mild OSA (AHI 5.8/hour). A multidisciplinary approach involving ENT for drug-induced sleep endoscopy to optimize VNS stimulation parameters along with neurology and sleep involvement is planned. The optimized VNS setting could be used during planned sleep times using a VNS device with dual-programming capability.

Conclusion
To our knowledge, there is no literature on development of sleep apnea after long term VNS implantation. Although the exact mechanism is unknown, VNS can cause sleep apnea or worsen sleep apnea in patients with a preexisting diagnosis. [1] This case demonstrates need for continuous monitoring and screening for sleep apnea in patients who have been implanted with VNS, even if baseline and follow up PGSs are normal. Current literature suggests discontinuation of VNS to resolve VNS-induced OSA, but we describe a case here where VNS is needed for ongoing management of epilepsy. Further studies are needed to identify treatment options for patients that cannot discontinue VNS therapy who also cannot be successfully treated with PAP if they develop VNS-induced OSA.

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1022 Two Teenagers with New Onset Insomnia post COVID
Authors: Popoola Taiye , Wani Anna , Srikrishnan Anand , Rovira Elisa Basora 
Published: May 2023

Abstract

Introduction
Sleep disturbances are increasingly seen in COVID-19 infection. Multiple reports have described the onset of insomnia, excessive daytime sleepiness, and fatigue in patients with both acute COVID infection and long COVID syndrome. Most of these are limited to adults. We report the cases of 2 teenagers with new onset chronic insomnia post COVID-19 infection.

Report of case(s)
The first patient is a 14-year-old male who presented with sleep onset and sleep maintenance insomnia, excessive daytime sleepiness, difficulty concentrating, fatigue, and chronic cough, with onset five months prior presentation to the sleep clinic. He developed these symptoms after testing positive for COVID-19. He had significant impairment with multiple school absences and tardies for which he required accommodation. He completed a 2-week actigraphy test which showed prolonged sleep latencies and insufficient sleep. He was scheduled for polysomnography (PSG) followed by a multiple sleep latency test (MSLT) but could not proceed to MSLT due to insufficient sleep the night before. The PSG did not reveal either obstructive sleep apnea or periodic limb movements of sleep. His laboratory testing was significant for a low ferritin level of 21. He was referred for IV iron infusion due to intolerance for oral iron. He was started on melatonin without benefit. He was offered clonidine and trazodone but declined. The second patient, a 17-year-old male, presented with insomnia of 3 months duration which started after an acute COVID infection. In addition to insomnia, he had comorbid mood symptoms. He went from being engaged in school and work to being unmotivated and listless. His actigraphy testing revealed he was having sufficient sleep on several days during which he reported not getting any sleep. He failed trials of multiple medications including melatonin, trazodone, hydroxyzine, and gabapentin. He also denied benefit with CBT-I.

Conclusion
These case reports link COVID 19 and chronic insomnia in pediatric patients. While it is yet unclear whether the development of sleep disturbances post COVID is as a result of an intrinsic characteristic of the COVID-19 virus or the biopsychosocial effects of the pandemic, it is important to develop and implement targeted interventions focused on recognizing and treating insomnia early.

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1025 Schizophrenic Patient with Large REM Rebound Following Treatment of Severe Obstructive Sleep Apnea
Authors: Qasmi Syed Talha , Aguillon Andre 
Published: May 2023

Abstract

Introduction
We present a case of uncontrolled schizophrenia with severe obstructive sleep apnea (OSA) and significant REM rebound after bilevel positive airway pressure (BPAP) therapy.

Report of case(s)
A 74-year-old female, with Schizophrenia on anti-psychotic therapy, presented to the sleep clinic due to excessive daytime sleepiness (EDS) and an Epworth score of 21/24. Diagnostic polysomnogram (PSG) reported an AHI of 162.1 events/hour with SaO2 nadir of 71% with sleep efficiency of 31.6% and no REM-sleep, consistent with severe OSA. The next morning, multiple sleep latency testing (MSLT) demonstrated mean sleep latency (MSL) of 3.6 minutes with 0 REM-onset sleep periods (SOREMPs). Patient underwent CPAP/BPAP titration which improved sleep efficiency to 88.6% and severe REM rebound at 61.7%. MSLT after the titration study demonstrated MSL of 4.0 minutes and 3 SOREMPs and was initiated on BPAP therapy at 20/13 cmH2O. A repeat PSG, 4 weeks later showed normalization of sleep architecture. After starting PAP therapy, the patient had a significant improvement in EDS but also energy and interest levels and denied feelings of sadness, racing or paranoid thoughts. Her social life improved and started exercising regularly and volunteering in the community. There was also improved blood pressure and blood sugar levels. She was followed up in the sleep clinic regularly and remained compliant with and benefiting from BPAP therapy and was continued on anti-psychotic therapy.

Conclusion
Untreated OSA often requires repetitive arousals to reopen the obstructed upper-airway that results in increased non-REM sleep at the expense of slow wave sleep and REM sleep. PAP therapy can cause a rebound increase in REM and SWS. OSA is under recognized in schizophrenic patients and other psychiatric disorders which results in development of significant comorbidities with greater severity of psychiatric symptoms. Elevated rates of OSA have been seen in the schizophrenic population. Treatment of OSA helps with not only physical but also psychotic symptoms. Many anti-psychotics, due to causing weight gain, may contribute to OSA. Studies also indicated that CPAP response to OSA, was as favorable in people with severe mental illness, compared to other patient groups.

Support (if any)
REM rebound and CPAP compliance; B. Koo, R. Wiggins, C. Molina
1026 Severe Sleep Inertia Managed with Bupropion
Authors: Qasmi Syed Talha , Cavins Gayle , Rulong Geoffrey 
Published: May 2023

Abstract

Introduction
Severe sleep inertia (SI) can often be managed with behavioral modifications and stimulants. This patient’s sleep inertia was resistant to such therapies but had remarkable improvement with Bupropion.

Report of case(s)
An 11-year-old male with a history of seizures presented with hypersomnolence, SI and difficulty arousing in the morning for 3 years. There was no reported cataplexy, with occasional hypnogogic hallucinations and sleep paralysis. Family denied witnessed apneas or snoring. He had previously undergone an adenotonsillectomy at 6 years of age for obstructive sleep apnea. When initially seen, he was not taking antiseizure medications, Epworth score was 14 and BMI was 30.5 kg/m2. Polysomnogram showed an AHI of 1.5 events/hour with REM latency of 179.5 minutes. Multiple sleep latency testing showed a mean sleep latency of 4 minutes, 7 seconds with 3 SOREM periods: consistent with narcolepsy. Melatonin and methylphenidate were initially prescribed, followed by dextroamphetamine-amphetamine, but were discontinued due to worsening hypersomnolence. Armodafinil improved his overall daytime sleepiness, however, severe SI persisted intermittently in the morning and with naps to the point of EMS being called on multiple occasions. EEG and brain-MRI were normal. For suspected cataplexy, venlafaxine was tried and not beneficial. Calcium-magnesium-potassium and sodium oxybate (XW) lead to no improvement in his SI. Therefore, Methylphenidate-20mg (MP) and then Armodafinil-50mg were added before bedtime, however, his SI persisted with frequently missed school. Bedtime Bupropion 150 mg was added with an immediate response. He has not missed school or daytime activities with Bupropion.

Conclusion
Sleep inertia is typically a characteristic of idiopathic hypersomnia rather than narcolepsy and is related to the abnormal transition of sleep to wakefulness resulting in reduced alertness, impaired performance, and desire to return to sleep. Such pronounced sleep inertia is atypical and caused significant quality of life impairment in our patient. Nighttime melatonin, XW, MP, and bupropion for SI has been described in the literature1. Our patient failed multiple treatments before bupropion which was immediately successful.

Support (if any)
1. Treatment of severe morning sleep inertia with bedtime long-acting bupropion and/or long-acting methylphenidate in a series of 4 patients. Schenck, MD1 ; Golden, MD2 ; Millman, MD3
1028 Congenital Central Hypoventilation in a patient with MCEP2 duplication syndrome
Authors: Rehman Sofiya , Srikrishnan Anand , Wani Anna 
Published: May 2023

Abstract

Introduction
Congenital central hypoventilation (CCHS) is a rare disorder due to impairment of autonomic nervous system clinically defined by a significantly reduced or abolished ventilatory response to hypercapnia and hypoxemia in the absence of pulmonary, neuromuscular, or cardiac disease. There is limited knowledge on associated respiratory manifestations and sleep-disordered breathing in children with the MECP2 duplication syndrome. Although sleep-disordered breathing and nocturnal hypoventilation are currently not well recognized in these children, we present a case of a patient with MECP2 mutation and congenital hypoventilation syndrome.

Report of case(s)
24-week-old male born full-term via c-section presented with recurrent respiratory problems, laryngeal cleft, dysphagia, snoring and apneic episodes. Previous genetic work up showed chromosome 15q11.2 microdeletion. Past medical history was significant for neonatal ICU stay for respiratory distress immediately following birth. Tonsils are 1+ on exam. PSG showed profound central sleep apnea (CAI 300/h) with average apneic episodes lasted for 7 seconds and very significant periodic breathing (88%) and mild obstructive apnea. Mild hypoxemia was also seen with SpO2 in low 90s without any significant hypercapnia. Bradypnea with resp rate 10-20/m was seen. PHOX2B gene testing was negative. Genetic testing with SNP array showed MECP2 duplication syndrome. His development is delayed, currently at the age of 20 months, including cognitive, speech and motor function for which he is on therapy. Tracheostomy was recommended, however family preferred to continue with non-invasive ventilation while awaiting diaphragmatic pacing.

Conclusion
Respiratory manifestations in MECP2 duplication syndrome patients are common, with important impact and even a possible fatal outcome. Although sleep-disordered breathing and nocturnal hypoventilation is currently not cited as an important symptom in these children, there have been several case reports of CCHS associated with this. PSG should be considered in patients with MCEP2 duplication syndrome for timely diagnosis. Different treatment modalities (ENT surgery, CPAP and NIV) can be applied successfully to treat respiratory conditions associated with MECP2 duplication syndrome.

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1030 Alice in wonderland: A rare side effect of montelukast
Authors: Rojas Edward , Stansbury Robert , Azzouz Mouhannad , Sharma Sunil , Pham Christopher , Coutras Steven , Clawges Heather 
Published: May 2023

Abstract

Introduction
Alice in wonderland syndrome (AIWS), also known as Todd’s syndrome, is a neuropsychological condition that causes a set of symptoms with alteration of body image. These distortions include appearing smaller(micropsia) or larger(macropsia) or appearing to be closer or farther than they are. The exact cause of AIWS is currently unknown. Some associations include, migraine, temporal lobe epilepsy, brain tumors, psychoactive drugs or Epstein-Barr-virus. The treatment for this disease primarily aims at treating the underlying reason as to why the patient had the event.

Report of case(s)
7-year-old boy presents to sleep clinic for nighttime hallucinations. Nightly episodes described as body parts that seem abnormally large or small and people talking very fast. Events lasted 15 minutes. Patient had full recall and developed anxiety when falling asleep. Medications at presentation: vitamins, melatonin, Montelukast. Montelukast was discontinued. Referred to neurology for video EEG, which was negative for frontal lobe seizures. Due to restless sleep and snoring, sleep study and ferritin was ordered. He was found to have mild OSA with an AHI of 2.2 and a ferritin level of 28. The patient was not considered a surgical candidate for his mild OSA; therefore, watchful waiting and re-initiation of montelukast proceeded with initiation of oral iron. At sleep clinic follow up, family noted improvement of hallucinations for 2-3 months and then restart, with worsening. Upon medication review, Montelukast was the inciting factor to the presence and resolution of hallucinations.

Conclusion
In this patient the montelukast seemed to be the triggering factor of AIWS. It has been suggested that the inhibition of leukotriene receptors in the brain could be responsible for this drug reaction. No other obvious triggers, or results of work up were able to better explain his resolution once he discontinued montelukast. This just further promotes close pharmacologic vigilance in the pediatric population.

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1033 The link between vision loss and sleep
Authors: Safarpour Yasaman , Taweesedt Pahnwat , Gill Kirat 
Published: May 2023

Abstract

Introduction
Non-arteritic ischemic optic neuropathy (NAION) is the most common optic nerve disorder among patients over the age of 50. Several risk factors have been associated with NAION including systemic hypertension, diabetes mellitus, advanced age, and certain optic disc morphologies. A significant association between obstructive sleep apnea (OSA) and NAION has also been reported. Here, we present a case of a middle-aged man with a recent diagnosis of NAION who was subsequently found to have OSA.

Report of case(s)
A 53-year-old man with a remote history of head injury with concussion and tobacco use was referred to the Sleep Medicine Clinic from Ophthalmology for evaluation of obstructive sleep apnea (OSA). The patient initially presented to Ophthalmology due to acute onset, painless, left-sided, inferior vision loss. The patient was otherwise healthy and denied a history of diabetes mellitus or hypertension. He did report alcohol and cigar use on the night prior to the acute onset vision loss. He denied fevers, chills, joint or jaw pain, scalp tenderness, or jaw claudication. On initial evaluation he was found to have significant left eye optic disc edema. Blood test revealed normal complete blood count (CBC), erythrocyte sedimentation rate (ESR), and C-Reactive protein (CRP). His orbit and brain magnetic resonance imaging with and without contrast revealed no significant findings. He was subsequently diagnosed with left NAION. Further review of symptoms was positive for snoring, dry mouth, nighttime awakenings, and unrefreshing sleep, concerning for OSA and leading to referral to our Sleep Medicine Clinic. During initial consultation in Sleep Medicine, he denied symptoms concerning for any other sleep disorders. He subsequently underwent a home sleep test (WatchPAT) which confirmed the diagnosis of OSA. The apnea-hypopnea index (AHI) was 7.9 and the lowest oxygen saturation was 88%.

Conclusion
Given the reported association between NAION and OSA, a careful history should be obtained to screen for OSA when NAION is diagnosed.

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1037 Central sleep apnea due to high altitude periodic breathing
Authors: Srivali Narat , Tobias Lauren 
Published: May 2023

Abstract

Introduction
Many popular tourist destinations are at high-altitude. Visitors commonly report poor sleep quality during the first few nights after arriving, but some do not resolve over time and daytime symptoms may develop that require medical attention.

Report of case(s)
A previously healthy 76-year-old American man presented with progressive fatigue and frequent nocturnal awakenings to a clinic in Bogota, Columbia (altitude 2640 meters), where he had been living for eighteen months during the COVID-19 pandemic lockdown due to limited ability to travel. An arterial blood gas on room air revealed hypocapnia with a partial pressure of CO2 of 25 mmHg. A polysomnography (PSG) performed in Bogota demonstrated an apnea-hypopnea index (AHI) of 98 events/hour consisting of predominately central as well as obstructive respiratory disturbances (Figure 1). After a titration study, he was placed on treatment with continuous positive airway pressure (CPAP) at 10 cmH2O along with oxygen 1 L/min, but he had difficulty acclimating to treatment. The patient presented to our institution two months after returning to the USA with significantly improved symptoms despite remaining off CPAP. Repeat PSG revealed a substantial reduction in the AHI at 15 events/hour consisting of only central respiratory disturbances in the supine position during transitional N1/N2 sleep (Figure 2). Positional therapy was recommended. A repeat home sleep study performed three months later to reassess disease severity during non-supine sleep showed resolution of respiratory disturbances (Figure 3). High-altitude periodic breathing is a common phenomenon, characterized by alternating periods of absent respiratory efforts with periods of hyperventilation without another etiology (1, 2). It occurs due to the interaction of hypocapnia and increased loop-gain (2). Central sleep apnea (CSA) is considered a disorder only when associated with symptoms, not simply due to an elevated central apnea index (3). The more rapid ascent and the higher the altitude, the greater risk of developing periodic breathing. Descending to a lower altitude will often resolve apnea after few days but the phenomenon can persist for up to months (2, 4) as our patient.

Conclusion
CSA develops commonly upon ascent to high altitude. When feasible, symptomatic patients should be encouraged to descend to a lower altitude.

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1039 The Progression of Sleep-Related Breathing Disorders in a Patient with Joubert syndrome
Authors: Taweesedt Pahnwat , Safarpour Yasaman , Okorie Caroline 
Published: May 2023

Abstract

Introduction
Joubert syndrome (JS) is a rare autosomal recessive disease characterized by malformations of the cerebellar vermis, hypotonia, developmental delay, and respiratory variability. Among the potential health challenges that children with congenital malformations of the central nervous system including JS, may face, sleep concerns are frequent and may include sleep-related breathing disorders (SRBDs).

Report of case(s)
A 17-month-old female with JS was referred to the pediatric pulmonary clinic due to disrupted and fragmented sleep. She had frequent nocturnal awakenings, snoring, mouth breathing, and witnessed apneas. There was no family history of SRBDs. Physical exam was notable for high-arched palate, diffuse hypotonia, and intermittent periods of tachypnea followed by apneas, even when awake. Her polysomnography (PSG) demonstrated an apnea-hypopnea index (AHI) of 27.1/hour, with the obstructive apnea-hypopnea index (oAHI) of 18/hr with central apnea index (CAI) of 9.1/hr and nadir oxygen saturation (SpO2) of 87%. An attempt was made to initiate supplemental oxygen; however, the patient did not tolerate the cannula. At that time, neither adenoid nor tonsillar hypertrophy were noted and surgical intervention was not indicated. The repeat PSG one year later demonstrated AHI of 9.1/hour, oAHI of 4.7/hr with CAI of 4.4/hr and nadir SpO2 of 84%. At this time, she did demonstrate enlarged adenoids and tonsils and underwent adenotonsillectomy with some clinical improvement in snoring. Her mother noted continued pauses in breathing and post operative PSG demonstrated AHI of 12.8/hour, oAHI of 1.4/hr and CAI of 11.4/hr and nadir SpO2 of 79%. The patient had demonstrated worsening episodic tachypnea and central apneas and the decision was made to attempt initiation of a respiratory assist device to regulate breathing and ventilation. Due to the patient’s age, intensive therapy desensitization is required to allow her to tolerate positive airway pressure therapy.

Conclusion
Patients with JS are at high risk for complex SRBDs as part of their clinical presentation. It is therefore important to carefully monitor the character of sleep-disordered breathing, offering appropriate treatments where indicated. It is important to note the added challenges of young pediatric patients who often require deliberate desensitization interventions to proceed with successful treatment.

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