bluuebunny/crossref_metadata_2025_split huggingface dataset:
1179 An Unshakeable Case of Refractory RLS- Androgen Receptor Inhibitor Therapy as a Cause of RLS Exacerbation
Authors: Baskaran Berty , Lipford Melissa 
Published: April 2024

Abstract

Introduction
Restless leg syndrome (RLS/WED) is a neurological sleep condition which is characterized by an uncontrollable urge to move an individual’s legs and sometimes arms when at rest. Symptoms typically arise in the evening and nighttime hours and can be associated with severe insomnia and negative impact on quality of life. Here we present a case of severe RLS/WED exacerbations temporally associated with androgen receptor inhibitor therapy.

Report of case(s)
A 58 year old male was referred to the sleep medicine clinic for refractory RLS/WED. He was previously treated with high dose ropinirole, which was associated with severe augmentation. Trials of gabapentin, pregabalin, tramadol, and hydrocodone were associated with persistence of severe symptoms. He was eventually treated with methadone at a dose of 10 mg in addition to Rotigotine patch at a dose of 2 mg, with adequate symptom relief. He was later diagnosed with prostate cancer, and initiated on enzalutamide, an androgen receptor inhibitor. Within two weeks of starting this medication, he had a severe exacerbation of his RLS symptoms and associated insomnia. The medication was discontinued, with subsequent abatement of RLS symptoms. He was then switched to apalutamide by his urologist (another androgen receptor inhibitor), but again developed severe RLS exacerbation. Dosing of the Rotigotine patch and methadone were increased to 4 mg and 12.5 mg daily respectively. In addition, he was started on pramipexole 0.25 mg, which was later increased to 0.5 mg, but he continued to have severe residual symptomatology. After discussing with his oncologist, he went on a trial of 3-4 weeks where apalutamide was discontinued. During this time, his RLS symptoms resolved, and he was sleeping well. He then restarted apalutamide and again experienced a similar a severe RLS flare.

Conclusion
Our case demonstrates an association between androgen receptor inhibitor therapy and RLS exacerbations. Further work is needed to understand the possible relationships between RLS and these common treatments for prostate cancer.

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1180 Restlessness Is a Pain: Methadone Induced Augmentation
Authors: Bliton Kyle , Jordan Melissa , Patterson Patricia , Warren Josh 
Published: April 2024

Abstract

Introduction
Characterized by a sensation of discomfort in the lower extremities during times of inactivity at night, relieved with movement, and resulting in sleep disturbances, restless leg syndrome (RLS) is a common concern addressed in Sleep Medicine. Providers are equipped with an array of effective non-medicinal and pharmaceutical treatment options including alpha-2-ligands, dopaminergic agents, and opioids. The phenomenon of augmentation is a well described complication of dopamine therapy in patients with RLS wherein symptoms intensify in severity and may spread to include the upper extremities. Here we describe an unexpected case of methadone induced augmentation in a patient with treatment refractory RLS.

Report of case(s)
Our patient is a 75 year old male who initially presented to our clinic in February 2021 with concern for a &gt;10 year history of treatment refractory RLS. Patient exhibited symptoms typical for RLS with absence of daytime symptoms, muscle cramping, neuropathy, and lower back pain. He had undergone a sleep study in 2013 which demonstrated mild sleep apnea with an AHI of 6.4/hr and periodic limb movement disorder with a PLMI of 105.9/hr. Over the last several years he had failed multiple treatment regimens including low dose lorazepam, Lyrica, Baclofen, Amantadine, and Ambien all of which were ineffective. Both Gabapentin (titrated to 600mg maximum dose) and Mirapex were discontinued due to side effects including itching and constipation/anosmia respectively. Ropinirole was discontinued due to augmentation characterized by worsening symptom severity. Ferritin levels were optimized with iron supplementation. Non-medical interventions including efforts at improved hydration, a structured exercise regimen, and reduced caffeine intake were employed without symptom improvement. He would eventually be initiated on Methadone at a dose of 10mg with marked symptom improvement over the initial 2 months; however, symptoms would once again worsen and progress to involve his arms. Patient self weaned methadone to a 2.5mg dose with good response.

Conclusion
Though previously reported with Tramadol use, augmentation is rare phenomenon outside the use of dopaminergic agents. We were unable to find other reports of augmentation in the setting of methadone use; however, our patient exhibited worsening symptom severity and progression with improvement on dose reduction.

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1181 Rock Around the Clock: Non-24 Sleep-Wake Disorder Successfully Treated with Circadian Entrainment, a Case Series
Authors: Benavides David , Benge Elizabeth , Epstein Lawrence 
Published: April 2024

Abstract

Introduction
Non-24-hour sleep-wake rhythm disorder (N24SWD) is a circadian rhythm disorder characterized by an inability to entrain to the 24-hour environment. It can occur in both blind and sighted individuals- usually with a longer sleep period, advanced temperature rhythm, and higher sensitivity to light than normal controls. Current therapies, derived from treating N24SWD in the blind and the human phase response curve, typically involve timed melatonin administration. It is more complex in sighted individuals, requiring multiple time cues such as light, melatonin, social interactions, feeding, and activity. We present two cases of non-24 sleep-wake disorder in sighted individuals who were successfully treated with circadian entrainment.

Report of case(s)
Report of Cases: Case 1: Sighted 31-year-old unemployed male with ADHD, presented with an irregular sleep pattern. He reported a lack of routine, prolonged dim basement computer use, and a circadian rhythm appearing longer than 24 hours. Actigraphy and sleep diaries confirmed N24SWD. Comprehensive measures, including timed light and melatonin therapy, blue light blocking glasses, and activity scheduling, led to a subjective "miraculous" improvement, with follow-up actigraphy confirming resolution. Case 2: Sighted 23-year-old female with autism and affective disorders presented with shifting sleep times alternating with prolonged video-game use. Actigraphy and sleep diaries were consistent with N24SWD. Following a prescribed schedule with light therapy, timed melatonin, and activity scheduling led to circadian alignment, confirmed by sleep diaries and patient report.

Conclusion
Effectively managing N24SWD in sighted individuals is challenging, as evidenced by limited treatment success and total cases documented in the literature, including challenges in long-term patient adherence. This case series highlights two successful instances of entrainment using a combination of timed melatonin, bright and low light therapy and activity scheduling, supported by actigraphy and diaries reflecting adaptation. These cases provide insights into contributors to the development of N24SWD and methods for resolving them.

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1182 A Case of Hypersomnolence in a Solid-Organ Transplant Recipient
Authors: Mukharesh Lana , Revana Amee 
Published: April 2024

Abstract

Introduction
Epstein-Barr virus (EBV) infects more than 90% of humans worldwide, with no evidence of active infection and persists for life. In pediatric renal transplant recipients, EBV primary infection or reactivation occur more frequently (63% and 44%, respectively). There is limited data to suggest duration of symptoms secondary to EBV and treatment options for pediatric hypersomnia secondary to EBV infection.

Report of case(s)
We describe a case of hypersomnia in a 7-year-old male with ADHD. He had received a renal transplant three years prior, secondary to renal dysplasia from a deceased donor who was EBV positive. He had a baseline polysomnogram (PSG) completed for symptoms of snoring, witnessed apneas and nocturnal enuresis, and was diagnosed with severe obstructive sleep apnea. He was started on CPAP and demonstrated excellent adherence to positive airway pressure (PAP). At age 9, he developed hypersomnolence, where he would sleep for 12-16 hours a day, fall asleep in school, during meals and vacation trips. The parents reported he was diagnosed with EBV a few months prior (EBV IgG and EBV nuclear antigen positive). Completed diagnostic work up included PAP device interrogation with adherence data, an actigraphy, a diagnostic PSG while on PAP and a multiple sleep latency test (MSLT). All the work up was reassuring, except for the MSLT where the patient had a sleep onset latency of 11 minutes and 3 out of 5 naps with sleep onset REM periods. Over the course of his evaluation, his pediatric daytime sleepiness scale ranged between 20-28 (&gt;15 is significant). He was started on atomoxetine, with some reported improvement in hypersomnolence. Repeat PSG revealed primary snoring; therefore, PAP was discontinued. At age 15, his hypersomnolence had resolved, but conversely, exhibited difficulty initiating and maintaining sleep with higher doses of atomoxetine, therefore it was discontinued after 5 years of treatment.

Conclusion
In pediatric solid-organ transplant recipients with EBV infection, it is imperative to screen for excessive daytime sleepiness in order to facilitate therapies to improve overall quality of life, as well as recognize that the excessive daytime sleepiness may be a short-term symptom.

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1183 Chasing Sleep: Circadian Rhythm Sleep Disorder - Free-Running Type
Authors: Truong Khanh , Patel Abha 
Published: April 2024

Abstract

Introduction
Circadian rhythm sleep disorder, free-running type (CRSD-FRT) is a disorder caused by a circadian pacemaker that is out of sync with the 24-hour light-dark cycle. CRSD-FRT is rare in sighted persons but prevalent in totally blind individuals. In sighted people, CRSD-FRT can be associated with psychiatric conditions such as depression, but the etiology remains unknown. It is postulated that sighted persons may have reduced light sensitivity for phase-resetting effects. Symptoms of CRSD-FRT include insomnia, excessive daytime sleepiness and functional impairment. Here we present a unique case of CRSD-FRT in a sighted female with a history of depression.

Report of case(s)
A 27-year-old sighted female with history of depression previously treated with an SSRI presented to the sleep clinic with a chief complaint of chronic insomnia. Historically, she went to bed at 3 A.M. and awoke at 2 P.M. Sleep onset latency was normal. Estimated total sleep time was 10 hours. She denied history of any other sleep disorders. She experienced a similar delayed sleep routine as a teenager and assumed it was due to late-night gaming. She tried multiple sleep aids including trazodone, melatonin, and diphenhydramine, unsuccessfully. She described a “normal” sleeping schedule once per month, but then her sleep schedule began to move later and later, until it was normal again. She worked from home and was self-employed, giving her flexibility over her schedule. Her Epworth Sleepiness Score was 13 at the initial visit. Interestingly, her sibling had a similar sleeping pattern. Actigraphy was done which confirmed patient’s diagnosis of CRSD-FRT. She was advised to start Tasimelteon.

Conclusion
The patient was previously misdiagnosed with insomnia. CRSD-FRT is rare in sighted persons and may be underdiagnosed. Our patient’s CRSD-FRT may be linked to her history of depression and SSRI use. According to one study, a clinical dose of SSRI resulted in 47% increase in melatonin suppression, which increases the human circadian system’s responsiveness to light. Her flexible work from home schedule may also be a contributing factor. When changing or eliminating social time cues could precipitate the development of CRSD-FRT due to variations in the length of circadian rhythm.

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1187 Non-24-hour Sleep-Wake Disorder in Transgender Male Receiving Exogenous Testosterone
Authors: Collins Joseph , Espiritu Joseph 
Published: April 2024

Abstract

Introduction
Little is known about the effects of hormonal therapy for adolescents undergoing transgender care. We present a case of a biologic female being treated with exogenous testosterone for transgender care who develops non-24-hour circadian rhythm disorder (N24SWD), a rare sleep-wake disorder, most commonly seen in individuals who are totally blind.

Report of case(s)
A 19-year-old transgender man (female at birth) presented to our clinic with complaints of inconsistent sleep schedule and daytime sleepiness. They have a past medical history of anxiety, obsessive-compulsive disorder (OCD), and social phobia. As a younger teenager, the patient reported having a delayed sleep phase pattern. A year ago, the patient started exogenous testosterone injections every 2 weeks. Over the past year, the patient noticed a change in sleep pattern, described as going to bed and waking up 1-2 hours later, with each passing day. This was verified with 4 weeks of sleep diaries. The patient was diagnosed with N24SWD and was prescribed melatonin 0.5 mg 6 hours before bedtime without benefit. Other behavioral modifications with set wake times, bright light therapy, and morning exercises were not helpful. We prescribed tasimelteon, a melatonin receptor agonist, but it was denied by the health insurance. The current management plan is to optimize sleep behaviors and continue psychiatric follow-up for mood disorders while appealing the denial of tasimelteon.

Conclusion
Transgender individuals are particularly vulnerable to sleep disorders. Testosterone replacement therapy has been linked to sleep disordered breathing, insomnia, and sleep disruption. It has not been well established what role hormone therapy, and specifically testosterone in this case, plays on an individual’s circadian rhythm. Previous studies have demonstrated that melatonin production decreases more abruptly for women going through menopause and that delayed sleep phase in teenagers may be due to hormonal changes during puberty, although pathophysiology is not fully understood5. A similar case of a transgender male receiving exogenous testosterone was recently reported. More research is needed to elucidate the potential adverse effect of testosterone replacement on the circadian rhythm of the growing population of patients undergoing transgender care.

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1189 Intriguing Correlation Between Melatonin and Restless Legs Syndrome
Authors: Buntak Vesna , Shukla Anant , Tobias Lauren 
Published: April 2024

Abstract

Introduction
A temporal relationship between the onset of melatonin secretion at night and the worsening of restless legs syndrome (RLS) symptoms has been described in the literature. As a frequently used adjunctive to help combat insomnia, melatonin continues to be widely used.

Report of case(s)
A 65-year-old man with RLS, migraines, and onset and maintenance insomnia presented to our clinic with severe insomnia. He takes melatonin 3 mg and falls asleep in his chair while watching television. Then, he moves to the bed and has difficulty falling asleep. He will occasionally take extra doses of melatonin. He wakes up multiple times for a variety of reasons. His wake time is 4.30 AM for work. He had a home sleep apnea test that was negative. He describes crawling and achy feelings in his legs throughout the day. Sensation is associated with an urge to move or walk. These achy feelings are worse at nighttime. He currently takes 1200 mg of gabapentin three times a day. In the last year, his dose tripled despite adequate ferritin levels. Our patient did not recognize any temporal correlation between starting melatonin and worsening of his RLS but was agreeable with stopping melatonin. Within a week of stopping melatonin, his RLS symptoms became controlled and were no longer interrupting his sleep. We continued to work on his sleeping habits.

Conclusion
This case adds to increasing evidence of a correlation between melatonin and RLS. No substantial evidence was found during our literature search. One theory suggests that melatonin can inhibit dopamine release by suppressing calcium influx into stimulated nerve endings. Considering the widespread use of melatonin and the detrimental effect of RLS on quality of life, more studies are needed to evaluate this hypothesis.

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1192 A Case of Worsening Exploding Head Syndrome When Using Positive Airway Pressure
Authors: Nguyen Thien , Chang Melisa 
Published: April 2024

Abstract

Introduction
Exploding head syndrome (EHS) is a rare parasomnia with unclear etiology. There are limited research studies to confirm the exact predisposing and precipitating factors for its onset and frequency. Some published case reports suggest symptomatic improvement with the treatment of comorbid obstructive sleep apnea (OSA). This case presents a patient who received automatic positive airway pressure (APAP) treatment for OSA with worsening EHS symptoms.

Report of case(s)
A 41-year-old male with obesity (BMI of 33) and chronic back pain complained of visual and audio sensations of explosion, vibration, and light when falling asleep and waking up in the middle of the night without any associated physical pain. These events would occur twice a month and were accompanied by waking up three to four times nightly due to loud snoring. A home sleep study (HSAT) was completed and consistent with mild obstructive sleep apnea, demonstrating a respiratory event index (REI) of 7.4 events per hour. The patient was diagnosed with exploding head syndrome and OSA. He was started on APAP therapy. At the six-week follow-up, he noted worsening frequency of EHS events to twice a week. During this time, he lost his job. At the three-month follow-up since starting APAP, he reported further worsening of his EHS episodes to three times a week. The patient was not compliant with APAP usage. The compliance data revealed average usage of 2-3 hours a night with residual AHI of 1.2/hour.

Conclusion
EHS is a rare disorder with unknown predisposing and precipitating factors. This patient noted worsening of his EHS after the initiation of APAP therapy for his obstructive sleep apnea. However, the patient was not fully compliant with PAP therapy. He also underwent further stressors during this period. This case raises consideration regarding the side effects of APAP therapy on the severity of EHS when treating OSA.

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1193 Oxycardiorespirogram? More Than a Fancy Word, an Alternate Way to Determine Respiratory Support in a Complex Infant
Authors: Wang Victoria , Teran Jose , Revana Amee 
Published: April 2024

Abstract

Introduction
Obtaining diagnostic cardiorespiratory data via a polysomnogram in extremely young infants can be challenging. A multi-channel oxycardiorespirogram (OCRG) can be used to evaluate cardiorespiratory status in infants during wake and sleep states. The study records heart rate and rhythm, respiratory rate, oxygen saturation, and chest wall movement over a course of 4-8 hours. During the study, supplemental oxygen may be provided and adjusted according to the patient’s needs. We present a case of an infant in which the OCRG was able to guide the amount of respiratory support as an alternate diagnostic tool.

Report of case(s)
Patient is a 2-month-old female born at 35 weeks gestation with prenatally diagnosed myelomeningocele, hydrocephalus (s/p ventriculoperitoneal shunt placement), absent cerebellum, and marked hypoplasia of the brainstem. She was admitted to NICU on room air and then developed frequent episodes of apnea associated with desaturations requiring escalation of respiratory support to intubation with mechanical ventilation. She was gradually weaned to high flow nasal cannula of 2 liters per minute (LPM). Capillary blood gas (CBG) at baseline was normal. A multi-channel OCRG to better characterize her apneic events was ordered. Apneas occurred most frequently during sleep and were associated with oxygen desaturations. Independent episodes of desaturations occurred during feeds and during sleep with an oxygen nadir or 86% and 75%, respectively. The frequency of the apneic/desaturation events improved with 0.5LPM supplemental oxygen given via conventional nasal cannula (NC). With these results, we recommended that she be discharged home on 24 hour supplemental oxygen support of 0.5LPM NC. At subsequent outpatient pulmonary clinic visits at 1-2 month intervals, the OCRG study recorded a decrease in frequency of the apneic episodes associated with desaturations over time. We have been able to wean her daytime supplemental oxygen use to 0.25LPM NC and keep the nighttime supplemental oxygen the same.

Conclusion
Our case highlights the importance of considering alternative diagnostic and monitoring techniques such as a multi-channel OCRG to determine supplemental oxygen respiratory support needs for patients who have frequent apneic episodes in both awake and sleep states in the neonatal and infant period.

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1198 Acute Onset Vision Loss in a Patient with NAION and OSA
Authors: Andersen Karl , Zacharias Rajesh , Grover Aarti , Schumaker Greg , Oster Joel , Ostrow Peter , He Wei 
Published: April 2024

Abstract

Introduction
Non-arteritic anterior ischemic optic neuropathy (NAION) is the most common optic neuropathy in middle-aged and older-aged adult. There are an estimated 6000 new cases per year and is most commonly diagnosed in patients between 61-72 years of age. It affects men and women equally. There is a slightly higher incidence in white patients. 55-59% of patients with NAION also have OSA.

Report of case(s)
A 51-year-old man with a past medical history of hypertension and erectile dysfunction developed painless left inferior altitudinal visual field loss and blurriness in April 2023. He was seen in the Ophthalmology Department and was found to have left unilateral optic disc swelling on fundoscopic examination. An MRI of his brain, orbits, and face were normal. He was referred to Neuro-Ophthalmology where a slit lamp and fundus exam was performed and was significant for 360 degrees of disc swelling with heme seen inferiorly and normal appearing vessels and macula. A follow up fundoscopic examination one month later was significant for a hyperemic nerve and nasal blurriness in the left eye. No changes were seen in the right eye. Based on these findings, he was diagnosed with NAION. A HST was ordered by his Ophthalmologist that showed moderate OSA with an AHI of 24.3/h, an O2 nadir of 81%, and an ODI of 32.3. He was then seen in the Sleep Medicine clinic and prescribed CPAP. At his two month follow up appointment, he had an improvement in his daytime sleepiness and concentration but still had residual visual deficits in his left eye, including blurriness, light sensitivity, and difficulty focusing.

Conclusion
NAION is a rare condition with untreated OSA as a known risk factor due to nocturnal hypoxia decreasing perfusion to the optic nerve. This case reinforces the notion that the hypoxic burden of untreated OSA is a systemic problem and can lead to damage throughout the body. In this case, the benefit of treating this patient’s OSA is that his contralateral eye, the right eye, is also vulnerable to developing the same ischemic optic neuropathy that the left eye suffered from.

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