bluuebunny/crossref_metadata_2025_split huggingface dataset:
0830 A Pediatric Case of Myoclonic Seizures Precipitating Central Sleep Apnea And Sequences of Periodic Breathing During Sleep
Authors: Wisniewski Benjamin , Witt Rochelle , Dye Thomas , Arya Ravindra , Simakajornboon Narong 
Published: May 2022

Abstract

Introduction
Myoclonic seizures are known to be influenced by the sleep-wake cycle. Although myoclonic seizures have been shown to alter sleep architecture, little is known about their influence on sleep-disordered breathing.
Report of Cases: A 7-year-old male with a history of generalized epilepsy of unknown etiology with absence as well as myoclonic seizures, poor impulse control, and fine motor delay was referred for restless sleep and frequent nocturnal awakenings. A routine electroencephalography (EEG) performed 33 months prior to presentation, at an outside institution, reportedly showed 3.0 Hz generalized frontally dominant spike and wave discharges lasting less than two seconds. Video EEG (vEEG) revealed diffuse bilateral poly-spike and slow wave discharges, predominantly during sleep, and myoclonic seizures with abrupt whole-body jerks. Polysomnography, at this time, was characterized by mild central sleep apnea (AHI: 3.7; OAHI: 0.0; CAI: 2.8; REM AHI: 14.4). His lamotrigine dosing was increased to from 1.8 mg/kg/day to 3.6 mg/kg/day, at presentation, with seizure control for three months but he developed a recurrence of myoclonic seizures in the setting of sleep deprivation. A repeat vEEG was similar to previous findings and his lamotrigine was increased further and clobazam was added. Due to worsening behaviors, clobazam was discontinued and a weaning plan for lamotrigine was formulated. Repeat polysomnography demonstrated worsening central sleep apnea (AHI: 18.3, OAHI: 2.3), without hypoventilation, characterized by sequences of periodic breathing that occurred in conjunction with myoclonic seizures. The majority of seizure-induced periodic breathing episodes were observed during a one-hour period of NREM sleep near the end of the study. Stereotyped epileptiform activity occurred in conjunction with a resumption of hyperventilatory respirations following apneic events. The increased respiratory drive, however, did not appear to be clinically epileptic in nature as the epileptiform activity either preceded or trailed the respirations. A variable REM and then NREM predominance of central events on successive polysomnograms further suggests that his central events are not intrinsic but that they are precipitated by epileptiform activity. Zonisamide, an anticonvulsant with carbonic anhydrase
inhibitory properties was initiated and is being titrated to therapeutic goal.

Conclusion
This case illustrates an unusual presentation of myoclonic seizure-induced periodic breathing during sleep.

Support (If Any)
Cincinnati Children’s Research Foundation.
0836 Atypical presentation of Biot’s breathing in a patient with Arnold Chiari Malformation II with complex sleep apnea/Sleep hypoventilation/hypoxemia not on opioid and with no history of meningitis
Authors: Pant Alok , Ghildiyal Naomi , Khan Yousaf , Baldridge Scott , Ho Minh , Monceaux Brittany , Koothrezhi Rupa , Liendo Cesar , Chernyshev Oleg 
Published: May 2022

Abstract

Introduction
Sleep disordered breathing is very common in Arnold Chiari malformation but Biots’s breathing has not been reported in these cases.
Report of Cases: 3-year-old with history of Arnold Chiari II malformation with hydrocephalus (s/p VP shunt and surgical decompression), spina bifida myelomeningocele (s/p in utero repair), subglottic stenosis with prior tracheostomy and eventual decannulation presented for evaluation of sleep disordered breathing. Presenting symptoms included witnessed apneas, cyanosis, daytime sleepiness and frequent awakenings. Physical examination was largely unremarkable.
Initial PSG demonstrated complex sleep apnea, with an AHI 20.6, REM AHI 57.8 per hour of
sleep, ETCO2 peak of 69 cm H20 and an O2 saturation nadir of 34%, with sleep related
hypoventilation/hypoxemia and Biot's breathing in the absence of opioid use and CNS infection.
During titration study, Biot’s breathing, complex apnea and sleep related hypoventilation/hypoxemia responded well to BiPAP ST of IPAP 18 cm H20 and EPAP 14 cm H20 BUR of 12 and oxygen of 1 L/min. She later developed intolerance to BiPAP due to high
pressures and was decreased to BIPAP 13/11 cm H2O. Later the patient discontinued the use of
BIPAP due to intolerance and was switched to night time O2 at 3-4 L/min. Per the parents, the
patient has been maintaining her oxygen saturation in the absence of BIPAP therapy with
oxygen use. Due to COVID, patient was unable to follow up but will be scheduled for a repeat
PSG in the near future. She followed with Neurosurgery for Arnold Chiari II and they
recommended no surgical intervention at this time due to functional VP shunt.

Conclusion
This is an atypical presentation of Biot’s breathing in the absence of CNS infections and opioid use in a patient with Arnold Chiari malformation II. Patient has complex sleep apnea, initially well controlled with BiPAP ST, but developed BiPAP intolerance. She is on oxygen with good control of hypoxemia in the absence of BiPAP therapy.

Support (If Any)
0841 Case Report: Can LSD be Associated with Chronic Exploding Head Syndrome?
Authors: Manikat Feby Puravath , Kushida Clete 
Published: May 2022

Abstract

Introduction
Exploding head syndrome is a rare phenomenon characterized by a loud imagined noise at sleep onset. Less commonly, it is associated with a simultaneous stab of pain in the forehead. It may result in recurring arousals and anxiety regarding the events. The underlying pathophysiology remains unknown.
Report of Cases: A 27 year old male with no past medical history presented to the sleep medicine clinic with a 2-year history of sleep-onset pain between his eyes, like “a flash of
lightning,” associated with moderate to severe pain. He experiences “brain zaps” or “a jolt of electricity between his eyes every night.” He would yell and jolt out of bed due to this sensation. These episodes occur approximately 60 to 90 minutes after sleep onset almost every night, jolting him from sleep and lasting approximately 1 minute. He also has difficulty seeing in the dark and also eye twitching. He experiences palpitations, diaphoresis, and a sense of fear often with these episodes. He denies any neurological deficits or muscle jerks. He is not on any daily
medications. He does not regularly use any sleep aids but has tried melatonin 3 mg in
the past. Evaluation by ophthalmology has ruled out ophthalmologic and neuro ophthalmic
causes. He does report use of illicit substances including lysergic acid diethylamide (LSD),
marijuana, methylenedioxymethamphetamine (MDMA), and cocaine. He did not experience any unpleasant effects after his first use of LSD, however within minutes of his second ingestion, he felt a sharp pain on his nasal bridge and forehead 10/10 in intensity. He states that since then, this pain has recurred almost every night. He does not have a family history of any neurologic disorders or migraines. The current plan for workup includes an in lab polysomnography with seizure montage and, if negative, a trial of a tricyclic antidepressant is planned.

Conclusion
This case illustrates the potential issues after LSD use causing severe dopaminergic and serotonin surge with drug use and the value of a complete history. Awareness of a possible correlation is clinically useful and may serve as a caution in the use of recreational drugs.

Support (If Any)
0842 Narcolepsy or Sarcoidosis?
Authors: Alam Ariful , Pham Christopher , Stansbury Robert 
Published: May 2022

Abstract

Introduction
Narcolepsy is a common cause of chronic sleepiness, affecting 1 in 2000 people. Despite the frequency of narcolepsy, the average time from the onset of symptoms to diagnosis is 5 to 15 years, and may remain undiagnosed in as many as half of all affected people. The etiology of this problem is largely unknown and usually multifactorial. The prevalence of these sleep disorders is increased in sarcoidosis compared to the general population. We present a case of narcolepsy in a patient with biopsy proven extrapulmonary sarcoidosis.
Report of Cases: 42-year-old female has a history of Sjogren’s syndrome (SSA/SSB positive, ANA positive 1:160), antiphospholipid syndrome with past left lower extremity deep venous thrombosis and positive LAC and anticardiolipin antibody, and Raynaud’s syndrome for more than 15 years, asthma, pleurisy, irritable bowel syndrome, GERD, depression, migraine and fibromyalgia. Her primary sleep complaint is excessive daytime sleepiness and nonrestorative sleep requiring frequent napping. She exhibited intermittent hypnagogic hallucinations but denies cataplexy. She has no history of smoking and denies drinking caffeine/alcohol. Has 3-5 isolated awakenings at night otherwise denies insomnia. She had a score of 17 on the Epworth scale. Her in-lab polysomnography demonstrated an AHI of 0/hr, sleep latency of 4.5 minutes and REM latency of 31.5 minutes. No other abnormalities noted. She then underwent a multiple
sleep latency test. Sleep was achieved in all the nap trials with a mean sleep latency of 1.2 minutes with three Sudden Onset REM Sleep recorded meeting criteria for narcolepsy. A subsequent PET/CT scan was obtained due to elevated liver enzymes and positive MRI findings, which incidentally showed a 2 mm enhancing focus involving the medial right cerebellar peduncle. She underwent biopsy which showed granulomatous disease consistent with sarcoidosis. Patient is currently successfully treated with methylphenidate for her underlying narcolepsy.

Conclusion
We present a patient with narcolepsy and multiple underlying autoimmune diseases including biopsy indicating extrapulmonary sarcoidosis. We, furthermore, present radiographic evidence in her brain that may have contributed to her narcolepsy.

Support (If Any)
0843 Sexsomnia in a female patient with nightmares and obstructive sleep apnea: a case report
Authors: Bigman Daniel , Greenough Glen 
Published: May 2022

Abstract

Introduction
Sexsomnia has been described as various sleep-related abnormal sexual behaviors primarily associated with confusional arousals and more rarely linked to rapid eye movement (REM) sleep behavior disorder (RBD). Clinical descriptions are based on case reports and review series, which leads to diagnostic ambiguity, challenges with appropriate classification, and forensic and legal ramifications
Report of Cases: We report a 34 year old female with a past medical history of polycystic ovarian syndrome and childhood post-traumatic stress disorder. Family history significant for parent with lifelong parasomnias. The patient was referred to Sleep Medicine with concern for snoring, sleepiness, and sleep behaviors. The patient reports sleep talking since childhood which predate trauma, traumatic themed nightmares, and ex-husband and current spouse reporting sleep-related masturbation, sexual advances described as coital-like pelvic movements which often lead to intercourse and subsequent amnesia. In addition, spouse describes bruxism and ongoing night terrors. Physical examination notable for body mass index 44 kg/m2, high arched palate, Mallampati class III, and 18 inch neck circumference. The patient underwent a polysomnagram which demonstrated fragmented sleep architecture, total arousal index up to 20.3/hr, and totalsleep time in stage N3 20%, Four awakenings fromN3, and findings of N3 partial arousal. The patient was observed in v-PSG to have periods of nocturnal lagophthalmos, finger movements, and vocalizations during NREM sleep. However, many of these observations were in the setting of respiratory events. The apnea-hypopnea index was 16.9/hr and a minimum oxygen saturation of 81% and mean saturation of 95%. A total of 3.1 minutes were spent less than or equal to 90%. TCO2 was without concern for hypoventilation. The patient was started on Auto CPAP 5-15cmH2O with resolution of snoring and some improvement to sleepiness. However, spouse reports ongoing sexsomnia in the same frequency despite treatment of obstructive sleep apnea.

Conclusion
This case reports a female adult with persistent sexsomnia despite early treatment of obstructive sleep apnea which highlights the possibility of co-occurring sleep disorder. The case does support current limited literature around sexsomnias having features of partial arousals from NREM sleep given the co-occuring history of night terrors, elevated N3 awakenings, and evidence of cortico-cortical dissociation.

Support (If Any)
None
0850 Exploding Head syndrome and Hypersomnia
Authors: Murray Robert , Stager Lindsay , Maddox Mary 
Published: May 2022

Abstract

Introduction
Exploding head syndrome (EHS) is a hypnagogic parasomnia where the patient experiences a loud noise or flash of light and often reports distress secondary to the stimulus. EHS is relatively uncommon, and to our knowledge, there has been only one published report of comorbid EHS and narcolepsy, describing a 38-year-old male. This report focuses on a pediatric case of EHS, evaluating possible comorbid narcolepsy and sleep apnea.
Report of Cases: Eight-year-old male was referred for excessive daytime sleepiness. He endorsed trouble staying awake in school, both in the morning and after lunch time. His Epworth in clinic was a 16, despite nightly sleep duration ≥8 hours. He also endorsed weakness secondary to strong emotions and was observed to have difficulty grasping and holding a pencil while tickled. Patient and his caregiver denied snoring, witnessed apnea, dream enactment, and symptoms of restless leg syndrome. However, the patient did admit to auditory hallucinations, occurring about twice per week during the transition from wakefulness to sleep. He described the hallucination as a banging sound and the sound of clinging bracelets, noting he has searched for the source of the stimulus, but has never been able to find a cause. Patient’s mother denied hearing similar noises. No visual hallucinations or sleep paralysis noted. Polysomnography revealed 0 Central Sleep Apneas, 0 Mixed Apneas, 10 Obstructive Apneas, and 12 hypopneas.
The mean duration of these events was 0 seconds. The patient’s Apnea-Hypopnea Index (AHI) was 2.5 and had a REM AHI of 8.3. Patient was supine 75% of the night.

Conclusion
This case illustrates a unique hypnagogic parasomnia in a pediatric patient. At this point, the patient’s symptoms would suggest EHS characterized by auditory hallucinations that involve hearing the banging sound, upon transition from sleep to wake. The patient also exhibits some narcolepsy type symptoms - hypersomnia, Epworth of 16, and subtle symptoms of cataplexy. In lab PSG – showed overall AHI of 2.5, REM AHI of 8.3- oxygen nadir 90%. Patient referred to ENT for tonsillectomy and adenoidectomy. With follow up NPSG and MSLT if still sleepy. He was referred to behavioral sleep medicine clinic to address anxiety secondary to EHS, but did not attend.

Support (If Any)
0854 A case of complete heart block in a patient with severe obstructive sleep apnea
Authors: Jamnadas Jeeten , Acho Megan , Farrehi Peter 
Published: May 2022

Abstract

Introduction
Obstructive sleep apnea (OSA) has been associated with bradycardic arrythmias and cases of transient heart block. Heart block occurring during sleep has been described in up to 10% of patients with obstructive sleep apnea, most commonly during stage R sleep and through a mechanism known as vagal activation. Stage R sleep is associated with desynchronization of respiratory and cardiovascular functions, and the excessive autonomic response (vagal activation) occurs due to the stronger stimulation of chemoreceptors and baroreceptors induced by hypoxemia, intrathoracic pressure swings, and hemodynamic alterations. Treatment of the underlying sleep-disordered breathing will typically treat the bradycardic arrythmia as well.
Report of Cases: A 49-year-old male with a BMI of 30 kg/m , neck circumference of 16 inches, and obstructive sleep apnea presented to the sleep center to establish care for management of his preexisting OSA. He was previously diagnosed with moderate OSA in 2007 but had discontinued therapy shortly after due to Continuous Positive Airway Pressure (CPAP) intolerance. The patient complained of snoring, nocturia, and excessive daytime sleepiness (18/24 on Epworth Sleepiness Scale). During the split night study, the patient was noted to have 2:1 second degree heart block followed by a transient episode of complete heart block, manifested by 4 non-conducted p-waves. The patient remained asymptomatic throughout the night. Upon study completion, the patient denied any prior chest pain, presyncope, syncope, or medications that might cause heart block.

Conclusion
Bradyarrythmias can be seen in severe obstructive sleep apnea in the setting of excessive vagal stimulation through increased stimulation of chemoreceptors and baroreceptors. Treatment with PAP therapy can lead to prevention of heart block in 80-90% of these patients.

Support (If Any)
0855 A challenging case of nocturnal visual hallucinations in an elderly woman
Authors: Van den Bulcke Laura , Van Den Bossche Maarten 
Published: May 2022

Abstract

Introduction
Nocturnal hallucinations can be part of a wide array of different disorders, like sleep disorders (e.g. narcolepsy), visual impairment (e.g. Charles-Bonnet syndrome), neurodegenerative disorders (e.g. Lewy Body Dementia) and psychiatric disorders (e.g. schizophrenia). We explore the differential diagnosis, the challenging diagnostic workup and the limited therapeutic options in this patient with complex visual nocturnal hallucinations.
Report of Cases: A 64 year old woman was referred with detailed and vivid visual nocturnal hallucinations (seeing her dog smoking a cigar, seeing someone in the room moving her bed, etc.). The hallucinations started 1-2 months ago, were mainly present at night (both when falling asleep and during the night) but occasionally also during the day. She had also been suffering from worsening daytime sleepiness for 3-4 months, and from memory problems for 1 year.
Cataplexy and sleep paralysis were absent. Clinical neurological examination was normal. The
patient had been diagnosed in the past with OSAS. She had been treated with CPAP for 3 years
and recent polysomnography showed good control of her apnea. She had also been diagnosed
with Crohn’s disease, COPD and cardiomyopathy. Because of her age, the vivid hallucinations, and the memory problems, we first wanted to rule out Lewy Body Dementia. Neuropsychological testing ruled out dementia. MRI of the brain showed some white matter lesions, without substantial atrophy. At a subsequent multidisciplinary consultation, her cardiologist considered her cardiomyopathy as a contraindication for stimulants. Therefore, and because we believed the a priori probability for narcolepsy to be low, an additional MSLT was not considered useful. Based on further pulmonary tests, we hypothesized that hypoxia could play a role in the hallucinations and hypersomnolence. The hallucinations improved over time, the hypersomnolence remained, but we found there were no good treatment options available, considering the cardiac contraindications for stimulants.

Conclusion
There is a wide-ranging differential diagnosis for nocturnal visual hallucinations. The specific patient characteristics are important both for further diagnosis and treatment options.

Support (If Any)
Supported by the Funds Malou Malou, Perano, Georgette Paulus, JMJS Breugelmans and Gabrielle, François and Christian De Mesmaeker, Managed by the King Baudouin Foundation of Belgium, No. 2021-J1990130-222081.
0857 How To Fix Mask Leak For Those With No Teeth
Authors: Kozlowski Conrad , Shamim-Uzzaman Afifa 
Published: May 2022

Abstract

Introduction
Mask fit is a crucial factor for patient adherence and effective use of PAP therapy. However, patients using dentures may face significant challenges to achieving a good mask seal, resulting in mask leak and inefficacy of therapy.

Report of Cases
A 67-year-old male with Obstructive Sleep Apnea (OSA) previously adherent to Bi-level Positive Airway Pressure (BPAP) therapy presented to clinic for complaints of mask leak. Due to a suspected dental infection, he had had all his teeth removed three years prior to this visit and had been instructed by his dentist to remove the dentures each night when he slept. However, removal of the dentures affected the fit of his full-face mask, and he experienced significant mask leak with return of pre-PAP symptoms. He was a habitual mouth-venter and preferred full-face masks. Despite trialing different masks, he was no longer able to tolerate his BPAP secondary to the leak and discontinued PAP therapy altogether. During his visit to the sleep center, a mask refit was performed with and without his dentures in place. While no masks sealed effectively with his dentures out, a satisfactory alternative was found if he wore his dentures. He was counseled to wear his dentures while using PAP, remove his dentures for the recommended minimum amount of time during the day instead of removing them for sleep, and to follow up with his denturist/prosthodontist for further instructions.

Conclusion
Edentulous patients may pose difficulties with mask fit, and a multi-modal approach may be necessary to effectively treat their sleep apnea. Luckily, our patient was able to find a mask that he found comfortable while wearing his dentures and had minimal leak. Alternatives may include consideration of positional therapy with lower pressures for a potential nasal mask, weight loss, hypoglossal nerve stimulator, or referral to an oral surgeon, denturist, or prosthodontist for guidelines on sleeping with dentures. Close monitoring of symptoms and download data is recommended following oral surgery and switch of mask interface.

Support (If Any)