bluuebunny/crossref_metadata_2025_split huggingface dataset:
815 Australian Surgery Trainee Education for Contemporary Airway Management of OSA: A Pilot Randomised Controlled Study
Authors: Hayward Nathan , Sideris Anders , Marshall Nathaniel , Burri Michael , Mackay Stuart G 
Published: May 2021

Abstract

Introduction
In Australia, ASOHNS delivers no formal curriculum for training of OHNS, or levels of competency required, to assess and treat complex OSA patients. Australian OHNS trainee confidence, knowledge and exposure to complex multi-level OSA surgery is lacking. Lack of exposure to sufficient complex OSA surgery case load has been identified as a major weakness in training within a recently published international survey. This study was a randomized clinical trial evaluating the effect of Australian OHNS trainee exposure to education materials compared with no exposure, on Sleep Surgery specific examination performance (multiple choice and short written answer).

Methods
70 accredited and 45 unaccredited OHNS trainees were invited to participate in this trial. Participants were randomly assigned to Sleep Surgery educational material exposure or no exposure to those materials. Those randomized to the exposure group were provided educational material and were given 2 weeks exposure time prior to the exam. Each participant then complete an online examin, consisting of 40 multiple choice questions and 1 short answer question (marked by a field expert). Differences between exposure and control group means were tested using independent t-tests.

Results
24 trainees were allocated to exposure and 22 to control. 33 participants attempted the examination. The were no significant differences between groups in the multiple choice (mean difference 1.3 ± 1.6 [3.3%], p=0.41) or written exam test scores (mean difference 1.8 ± 1.2 [9.0%], p=0.14). Accredited trainees performed better in the written exam (mean difference 2.6 ± 1.1 [13.0%], p=0.03). The mean test score in a separate exploratory group of 2 sleep fellowship trained OHNS was considerably higher in both exams.

Conclusion
This study suggests that exposure to formal education material may improve understanding of sleep surgery. Accredited trainees performed better than unaccredited trainees but the difference was small. Poor test performance in both groups may indicate further formal sleep surgery teaching is required in the ASOHNS training curriculum. Further research is required to identify the best ways possible to educate OHNS trainees in the complex and nuanced decision making required for OSA patients.

Support (if any)
Illawarra Health and Medical Research Institute Grant 2019.
832 Dose-Dependent Quetiapine-Induced Somnambulism and Sleep-Related Eating Disorder
Authors: Pham Tuyet , Malhotra Sonal 
Published: May 2021

Abstract

Introduction
Parasomnias are abnormal sleep-related movements that can occur during non-rapid eye movement sleep, rapid eye movement sleep, or transition of sleep. The prevalence of parasomnias in young children ranges from 9–40% which may be underestimated as this relies on parental recall. There are multiple reported cases of pharmacologically-induced parasomnias. Quetiapine is an atypical antipsychotic medication associated with somnambulism and sleep-related eating disorder.

Report of case(s)
A 9-year-old female with a history of attention deficit hyperactivity disorder, post-traumatic-stress disorder, depression, and sexual abuse during childhood presented to the Sleep Medicine Clinic for two years of worsened sleepwalking and sleep eating. Her medications included Methylphenidate, Quetiapine, Clonidine, and Duloxetine. She has had parasomnias since she was 3-years-old, initially presenting as abnormal sleeping positions (standing or sitting). She was initiated on Seroquel at 4-years-old, but parasomnias worsened over the last two years when Quetiapine was increased from 50 mg to 200 mg for behavioral and mood issues. Her somnambulism began to occur nightly. The family was required to remove all items from her bedroom except for the bed to prevent major injuries. She also had significant changes to her eating habit: she would eat two to three times her normal quantity as well as eating while asleep. The family would find her eating ice cream, chips, grapes, cold tortillas, or anything she was able to access. Fortunately, she did not consume raw meat or other frozen foods. The child did not have any recollection of eating at night. Psychiatry worked with her to cross-taper Quetiapine and Topiramate. At the lower dose of Quetiapine, she had exacerbation of her mood symptoms, paranoia, and insomnia; therefore, Topiramate was discontinued and Quetiapine was titrated to 150 mg with improvement in mood symptoms, insomnia, and resolution of sleep-related eating disorder. She continues to have somnambulism.

Conclusion
This case illustrates that quetiapine-induced somnambulism and sleep-related eating disorder can be dose-dependent; thus, important for clinicians to educate patients and/or family members of adverse effects while titrating quetiapine.
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846 Pulmonary Arterial Hypertension as a Sequela of Untreated Obstructive Sleep Apnea in a Patient with Prader-Willi Syndrome
Authors: Emanuel Hina , Kaplan Kevin 
Published: May 2021

Abstract

Introduction
Prader-Willi Syndrome (PWS) is a complex neurogenetic disorder characterized by hypotonia, behavioral problems, endocrinopathies, sleep and respiratory abnormalities. Morbidity and mortality in the PWS population is attributable to obesity, cardiovascular problems, and sleep apnea. We report a patient with PWS presenting with pulmonary arterial hypertension (PAH) due to untreated obstructive sleep apnea (OSA).

Report of case(s)
Our patient is a 17-year-old female with a past medical history of PWS, scoliosis, obesity (BMI 52.46), hypogonadotropic hypogonadism, and type II diabetes. Baseline echocardiogram (ECHO) performed at age 9 revealed an estimated right ventricular systolic pressure (eRVSP) of 32mmHg above right atrial pressure (RAP), tricuspid regurgitation (TR) at 2.8 m/sec with no interventricular septal flattening (IVSF) and right ventricle (RV) systolic dysfunction suggestive of mild PAH. Given significant scoliosis the patient did not qualify for growth hormone therapy. She underwent a polysomnogram (PSG) at age 14 showing severe obstructive sleep apnea; apnea-hypopnea index (AHI) of 22.6 (oAHI 22.6). Patient was subsequently lost to follow up until presenting in acute respiratory failure at age 17. She required endotracheal intubation and was extubated to bilevel PAP (BPAP) with inability to wean off BPAP. At that time an ECHO revealed eRVSP of 55 mmHg above RAP, IVSF, TR at 3.7 m/sec, and RV systolic dysfunction suggestive of moderate to severe PAH and developing right sided heart failure. A PAP titration PSG during this admission revealed hypoxemia with oxygen saturation less than 90% (O2 nadir 70%) 12.6% of total sleep time (TST) and hypoventilation (transcutaneous CO2 max of 57 mmHg with an elevation above 50 mmHg for 100% of TST). Using an inspiratory PAP (IPAP) of 24 cmH2O and expiratory PAP (EPAP) of 14 cmH20 with supplemental O2 of 4LPM the respiratory events and hypoxemia resolved but there was persistence of hypoventilation. Tadalafil was initiated for PAH and BPAP therapy for OSA. Follow up visits 4- and 8-weeks post discharge shows improving PAH (TR 3.6 m/sec, eRSVP 52 mmHg, and mild IVSF) due to BPAP and tadalafil therapies.

Conclusion
This case highlights the importance of treating OSA in patients with PWS to prevent cardiorespiratory complications and reduce morbidity and mortality.

Support (if any)
None
848 Refractory Hypoventilation in Congenital Central Hypoventilation Syndrome: A Case for Average Volume-Assured Pressure Support
Authors: Gubichuk Megan , McHugh Erin , Malhotra Sonal , Sockrider Marianna , Kancherla Binal 
Published: May 2021

Abstract

Introduction
Congenital Central Hypoventilation Syndrome (CCHS) is a rare cause of alveolar hypoventilation in children resulting in lifelong ventilatory support. In older children requiring nocturnal support alone, the use of diaphragmatic pacing in conjunction with or independent of non-invasive ventilatory (NIV) support has been demonstrated to improve quality of life. We present a case of refractory hypoventilation despite escalation of NIV.

Report of case(s)
20-year-old female with Hirschsprung’s disease and CCHS (20/26 polyalanine repeats) with history of invasive ventilation via tracheostomy who underwent bilateral diaphragmatic phrenic nerve stimulator placement at 13 years-of-age with subsequent tracheostomy decannulation. Diaphragmatic pacing was discontinued three years later in the setting of pneumonia and patient discomfort because of receiver positioning. At that time, she had improved subjective sleep quality and adequate ventilatory support on bi-level positive airway pressure (PAP) despite discontinuation of diaphragmatic pacing. Titration of bi-level PAP was done via polysomnogram four years later demonstrating nocturnal hypoventilation with transcutaneous CO2 values greater than 50 mmHg for 80% of the study and an oxygen nadir of 87% despite titration of inspiratory pressure and respiratory rate to maximize ventilatory assistance. The patient was subsequently admitted to the intensive care unit for transition to non-invasive average volume-assured pressure support (AVAPS) mode. Ventilation improved with nocturnal pCO2 values via capillary blood gas of 31 mmHg and 45 mmHg at 2 am and 6 am respectively. The patient was discharged on AVAPS therapy while undergoing evaluation to resume diaphragmatic pacing via cervical phrenic nerve stimulators for improved comfort.

Conclusion
Several ventilatory strategies may be employed in the care of patients with CCHS, with individualization of support based on phenotype, comorbidities, and patient and family preference. This case highlights the unique challenges of adequately ventilating patients as they age. The use of NIV via an AVAPS mode in patients with CCHS has been infrequently reported in the literature, though is promising in reported efficacy with regards to ensured ventilation. This, in conjunction with diaphragmatic pacing, may allow patients to achieve appropriate ventilation while maintaining quality of life, and could be considered in patients with refractory hypoventilation despite other modes of NIV.
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852 Severe Central Sleep Apnea in a Patient with Cornelia de Lange Syndrome
Authors: Willis Aaron , Petitto Lacie , Revana Amee 
Published: May 2021

Abstract

Introduction
Cornelia de Lange syndrome (CdLS) is a rare multisystem disorder with characteristic facial dysmorphisms, upper airway structural abnormalities, and varying degrees of intellectual and neurologic deficits. Affected patients can have many sleep problems including chronic insomnia and obstructive sleep apnea; however, there is no literature indicating central sleep apnea as a common sleep disorder in this patient population. We describe a patient with CdLS with an unusual presentation of both severe obstructive and central sleep apnea.

Report of case(s)
The patient is a 12-year-old female with CdLS, global developmental delay, repaired bilateral cleft-palate, and oropharyngeal dysphagia. She was referred for diagnostic polysomnography for evaluation of snoring. Physical examination findings were significant for distinctive features of the head and face including microcephaly, micrognathia, and synophrys. Neurologically, the patient was non-verbal and wheelchair bound. Pre-evaluation screening indicated no concerns from the caregiver regarding problems with sleep initiation, maintenance, or daytime symptoms. Polysomnography (PSG) at age 7 years revealed severe obstructive and central sleep apnea with an apnea-hypopnea index (AHI) of 78.2/hr. The majority of these were central events (68.44/hr). There were no epileptiform foci recorded. Bilevel Positive Airway Pressure (BPAP) in Spontaneous/Timed (S/T) mode was titrated during the study which resolved all central events. Magnetic resonance imaging of the brain was obtained indicating mild hypoplasia of the corpus callosum. Since the initial PSG, 5 additional routine studies were conducted and progressive worsening of central sleep apnea was noted with the highest recorded AHI of 108.8/hr (central events: 81.4/hr). Apneic events were fairly managed with BPAP in ST mode.

Conclusion
Sleep-disordered breathing in CdLS is an increasingly diagnosed entity. While previous literature indicates airway obstruction as a common cause for sleep apnea in children with CdLS, our case suggests that clinicians should consider central causes of sleep apnea in these patients. Given the sub-clinical progression of sleep apnea in this case study, routine follow up PSGs should be considered. Further research is necessary to increase knowledge of the incidence and management of central sleep apnea in children with CdLS.
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853 Severe Obstructive Sleep Apnea after Treatment with Phrenic Nerve Implant for Central Sleep Apnea: A Case for Dual Stimulation?
Authors: Sussman Sarah , Ananth Ashwin , Fares Elie , Huntley Colin , Boon Maurits , Fast Zhanna 
Published: May 2021

Abstract

Introduction
The remedē® system is a transvenous phrenic nerve stimulator used to treat central sleep apnea (CSA). It is a safe and effective implantable device that has demonstrated significant improvements in objective sleep and quality of life. While complex sleep apnea represents development of central sleep apnea during treatment with continuous positive airway pressure (CPAP) for obstructive sleep apnea, and has been well-described in the literature, we present a novel case of predominantly central sleep apnea treated with phrenic nerve stimulation with augmentation of obstructive events thereafter. This patient is, at the time of submission, being evaluated for hypoglossal nerve stimulation.

Report of case(s)
A 63-year-old male patient with a past medical history of hypertension, atrial fibrillation, asthma, chronic obstructive pulmonary disease, GERD, obesity, and prior pulmonary embolism presented to the sleep medicine clinic with complaints of difficulty initiating and maintaining sleep, snoring, multiple nocturnal awakenings, excessive daytime sleepiness, and physical fatigue. On exam, BMI was 30.4, and airway classification was Mallampati 4. Split night polysomnography revealed severe mixed sleep apnea with an overall apnea hypopnea index (AHI) of 58.6 and a central AHI of 53.8 per hour after successful PAP titration at 7 cm H2O. He underwent phrenic nerve stimulator implantation and activation without complications. Phrenic nerve stimulation titration study four months after activation showed a normal central AHI of 4.6 per hour at a therapeutic voltage of 1.6 to 1.9mA, but significantly worse OSA with obstructive AHI of 53.1. In follow up, he reported intolerance of CPAP due to significant nasal congestion as well as asthma. Presently, he is scheduled for drug induced sleep endoscopy to visualize airway obstruction and is being evaluated for Inspire hypoglossal nerve stimulation and nasal surgery.

Conclusion
The treatment of sleep apnea has recently evolved rapidly with the development of implantable devices to treat both central and obstructive sleep apnea. We present a novel case of the first known patient to be considered for dual nerve stimulation for sleep apnea. Further study needs to be done to determine the safety and efficacy of concurrent implantable devices for the simultaneous treatment of OSA and CSA.
Support (if any):
856 Sleep-writing, sleep-talking in uncontrolled REM-predominant obstructive sleep apnea
Authors: Ho Minh Tam , Ghildiyal Naomi , Liendo Cesar , Monceaux Brittany , Asghar Sheila , Anne Pratibha , Koothirezhi Rupa , Okorie Ugorji , Chernyshev Oleg 
Published: May 2021

Abstract

Introduction
Abnormal movements and behaviors during sleep are part of a larger group of nocturnal events that may occur during the sleep-wake cycle and/or the transitions into and out of sleep. We are presenting the case of OSA-related confusional arousals associated with sleep-writing and sleep-talking during REM-sleep.

Report of case(s)
46 year old female with history of CAD, HTN, RLS, Anxiety, Depression, and REM predominant OSA (AHI of 2.9 per hour of sleep, REM AHI of 40 per hour of sleep, and oxygen saturation nadir of 91%), noncompliant with PAP therapy, returned to reestablish sleep medicine care and restart PAP therapy. The patient reported worsening of her OSA symptoms while being without PAP therapy. In addition to traditional OSA symptoms (snoring, frequent nighttime awakenings, restless legs, daytime sleepiness/fatigue), she reported episodes of sleep-talking, sleep-writing with demonstrated evidence of these events in her personal diary. She maintains a collective diary that incorporates her blood pressure readings, her weight loss accounts with records of meals in a day, as well as a separate log of letters that she will write and mail. In this diary, she has noticed sleep writing instances in each field, with no recollections subsequently on the act of writing them. The patient reported that these episodes of sleep writing would occur 3–4 times in a week, during this period of PAP noncompliance. Last reported instance of her sleep writing was October 2020. After re-initiation of PAP therapy, the patient has not reported further episodes of sleep-writing or sleep-talking.

Conclusion
The sleep-writing is a very rare clinical symptom in the presentation of REM-predominant OSA as well as in REM/NREM parasomnias. We were not able to come across a case of it in the sleep literature review. Sleep-talking is a well-documented phenomenon. Confusional arousals may be responsible for symptoms of sleep-writing and sleep-talking in this case. We may repeat a sleep study with split protocol and parasomnia montage using AutoBipap if needed to investigate further. Further research should be done to explore the nature and correlation of sleep-writing in clinical practice.

Support (if any)
N/A
860 Variable Sleep Apnea Syndromes Complicated by Neurosurgeries in a Patient with Chiari Malformation Type 1
Authors: Ananth Ashwin , Fares Elie , Sagalow Emily , Nemargamulla Swetha , Fast Zhanna 
Published: May 2021

Abstract

Introduction
Chiari malformations (CM) are congenital conditions defined by craniocervical junction anatomic anomalies with downward displacement of cerebellar structures. Sleep-disordered breathing (SDB) including obstructive sleep apnea (OSA), bradypnea, central sleep apnea (CSA), and hypoventilation are described in CM patients.

Report of case(s)
31-year-old male with history of CM type 1 presented to the sleep medicine clinic for management of OSA and CSA diagnosed at age 16. PSG showed an apnea-hypopnea index (AHI) of 14.2, RDI of 29, and no central events. He was started on auto-CPAP with pressures of 5–20 with subsequent visits showing high residual AHI. A subsequent CPAP-titration study resulted in a pressure of 16cm H2O yielding AHI of 0. CPAP pressure was fixed, but a high residual AHI persisted despite excellent compliance. A split-night study resulted in a BiPAP prescription, for which a titration study noted PAP-emergent CSA. He was started on auto Bilevel with IPAP of 30, EPAP of 8 and backup rate of 12bpm. Despite treatment, elevated AHI persisted so he was switched to adaptive servo-ventilation (ASV) with nightly oxygen blended in resulting in controlled complex sleep apnea. SDB management was complicated by multiple neurosurgical decompressions. During the most recent procedure, he was found to have syringomyelia, syringobulbia, and a mass at the brainstem. Surgery, including placement of a 4th ventricle stent and mass excision, initially led to a decrease of residual AHI to a low of 0.7, before progressively increasing to 15. A repeat PSG demonstrated severe OSA (AHI of 35), without evidence of CSA or nocturnal hypoventilation. ASV was resumed and patient’s AHI progressively decreased to an average of 8, with improvement in his sleep apnea symptoms.

Conclusion
SDM in CM patients can be explained by condition-related anatomical changes and depression of respiratory centers due to possible extrinsic compression leading to complex sleep apnea presentations. While it is unclear why this patient’s sleep apnea improved then worsened after his latest decompression surgery, we believe that CSF recirculation and postoperative inflammation may be responsible. Close monitoring of SDB in patients with CM is important as they may require advanced therapies for proper control.
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861 Vascular dementia presenting as rapidly progressive dementia secondary to zolpidem
Authors: Barber Cameron , Carroll Dylan , Suryadevara Bhavani 
Published: May 2021

Abstract

Introduction
Rapidly progressive dementia is a condition with a wide differential which remains difficult to accurately diagnose. The potential pathologies responsible include thyroid, vitamin, and electrolytes abnormalities, infectious, and malignant causes. Vascular dementia, however, typically has a slow and insidious presentation. Zolpidem (Ambien) is among the top 50 prescribed medications in the US.

Report of case(s)
An 84-year-old Caucasian male with a past medical history of insomnia, and sleep apnea who is noncompliant with CPAP presented after a fall associated with altered mental status. He has taken zolpidem 10 mg nightly for over six years. The patient and wife reported notable personality changes beginning six months prior, as well as four months of progressively worsening auditory and visual hallucinations. Additionally, the patient noted developing urinary incontinence, and worsening gait steadiness with recurrent falls. The patient then developed sleep-wake inversion during the three weeks prior to his fall, and an outpatient referral to neurology was subsequently sent for dementia evaluation. On the night prior to his presentation, the patient took his usual nighttime zolpidem at 22:00 and later fell and was unable to get up. Subsequent testing was negative for reversible causes of dementia and MRI Brain revealed only chronic microvascular disease. His zolpidem dose was decreased to 5 mg and scheduled earlier which resulted in the resolution of his hallucinations, gait abnormalities, and acute encephalopathy.

Conclusion
One month later, the patient presented to the hospital after a repeat fall secondary to taking his zolpidem at his previously scheduled time. Once more, his dosage was further decreased to 2.5 mg and scheduling earlier, resulting again, in the complete resolution of his symptoms. Zolpidem, has an increased potential for delirium in elderly patients and especially those with dementia. Chronic use of zolpidem with insidiously progressive vascular dementia led to a worsening delirium which resolved after adjustment of timing and reduction of zolpidem dosing.
Support (if any):