851 Self-Medication with Caffeine for 31 Years: A Case of Undiagnosed Childhood Narcolepsy Type II

SLEEP ◽  
2021 ◽  
Vol 44 (Supplement_2) ◽  
pp. A331-A331
Author(s):  
Moustapha Doulaye Seydou ◽  
Christian Karime ◽  
Brenda Wyrick ◽  
Amir Khan
Keyword(s):  
Sleep Latency ◽  
Treatment Plan ◽  
Sleep Onset ◽  
Panic Attacks ◽  
Apnea Hypopnea Index ◽  
Type Ii ◽  
Obstructive Sleep ◽  
Sleep Benefit ◽  
Patient Reported ◽  
Multiple Episodes

Abstract Introduction Narcolepsy represents a relatively rare chronic neurological sleep disorder. While peak incidence occurs in adolescence and early-adulthood, reports indicate a substantial number of children under 10 remain undiagnosed or are misdiagnosed. The present case describes a female with undiagnosed narcolepsy type II self-medicating with caffeinated beverages from the age of 7. Report of case(s) A 40-year-old female presented at our clinic with excessive daytime fatigue and hypersomnolence despite adequate sleep (Epworth sleepiness scale= 16/24). The patient denied snoring, sleep paralysis, catalepsy, and hypnagogic/hypnopompic hallucinations. Symptoms began at the age of 7 and steadily worsened, with teachers reporting significant concentration difficulties and multiple episodes of unintentional sleep onset in the classroom. The patient reported heavily relying on caffeinated beverages from the age of 7 to remain awake and focused on classroom activities. Starting at the age of 7, the patient consumed on average a caffeine-equivalent of 1 espresso shot/day (64mg caffeine/day). This increased to 4–6 espresso shots/day (256-384mg caffeine/day) by the age of 12 and 5–9 espresso shots/day (320-576mg caffeine/day) by the age of 18. At the age of 25, the patient developed severe anxiety with panic attacks and episodes of suicidal ideation. With multiple episodes of sleep onset while operating a motor vehicle, a near-accident prompted medical evaluation. Diagnosed with general anxiety disorder and idiopathic hypersomnolence, escitalopram and armodafinil were started with limited effect. The patient continued self-medicating with caffeinated beverages until age 38 when she was diagnosed with narcolepsy type II. Sodium oxybate was subsequently added to her treatment plan with initial sleep benefit and caffeine reduction. A repeat mean sleep latency test confirmed narcolepsy type II (mean sleep latency= 3 minutes, mean rapid eye movement [REM] sleep latency= 3 minutes). Polysomnography was later performed due to non-resolving symptoms, revealing mild obstructive sleep apnea (REM apnea-hypopnea index= 13.5/hour). Continuous positive airway pressure was added to the treatment regime with significant sleep benefit. Conclusion We describe a case of undiagnosed childhood narcolepsy type II necessitating significant caffeine consumptions in order to maintain classroom performance. With known anxiety-provoking effects of caffeine, the case highlights the importance of addressing childhood narcolepsy. Support (if any):

857 Too sleepy: A pediatric case of Prader Willi Syndrome and Narcolepsy

SLEEP ◽  
2021 ◽  
Vol 44 (Supplement_2) ◽  
pp. A333-A333
Author(s):  
Elizabeth Lam ◽  
Sonal Malhotra ◽  
Daniel Glaze
Keyword(s):  
Daytime Sleepiness ◽  
Sleep Latency ◽  
Chromosome Region ◽  
Genetic Disorder ◽  
Sleep Onset ◽  
Apnea Hypopnea Index ◽  
Prader Willi Syndrome ◽  
Obstructive Sleep ◽  
Patient Reported

Abstract Introduction Prader Willi syndrome (PWS) is a genetic disorder due to deletion of the paternal copies of genes within the chromosome region 15q11-q13. Individuals with PWS are commonly seen with obesity, sleep disordered breathing, and excessive daytime sleepiness (EDS). While the exact cause of EDS in individuals with PWS is not fully understood, there have been reports of PWS with narcolepsy-like syndrome. We report a case of a patient with PWS with findings suggesting the diagnosis of Narcolepsy Type 2. Report of case(s) Our patient is a 12-year-old male with PWS and 2nd degree heart block who was evaluated in our pediatric sleep center. He has a previous diagnosis of mild obstructive sleep apnea (OSA) with an apnea hypopnea index (oAHI) of 3.2. At 12 years of age, mother and patient reported that he had increased snoring, weight gain, EDS with a Pediatric Daytime Sleepiness Score (PDSS) of 10 and frequent refreshing naps during the daytime. Patient denied cataplexy during that visit. Subsequently, 2-week actigrapghy was performed which demonstrated an average total night sleep of 8 hours and 8 minutes. Overnight PSG with Multiple Sleep Latency Test (MSLT) demonstrated an oAHI of 4.8, total sleep time of 6.88 hours. During the MSLT, the mean sleep latency was 6.2 minutes and 5 sleep onset REM periods were observed over 5 nap opportunities. At his follow-up visit, methylphenidate was initiated after clearance by his cardiologist. Patient and mother opted for medical management of his mild OSA with Fluticisone and Montelukast. At his follow-up appointment, the patient had improvement in daytime sleepiness with a PDSS of 2 despite taking his Methylphenidate at night. Patient was instructed to switch to morning Methylphenidate dosing to optimize treatment of his EDS. Conclusion EDS is a common complaint seen in patients with PWS, however the etiology of it is not entirely understood. It is thought to be centrally mediated with components of hypersomnia and narcolepsy like-symptoms. More research is needed to better understand, diagnose and adequately treat patients with PWS and EDS. Support (if any):

818 A Ghost from the Past: Unmasked Insomnia Affecting Hypoglossal Nerve Stimulation Therapy Adherence

SLEEP ◽  
2021 ◽  
Vol 44 (Supplement_2) ◽  
pp. A319-A320
Author(s):  
Elena Stuewe ◽  
Aarti Grover ◽  
Peter Ostrow ◽  
Greg Schumaker ◽  
Joel Oster ◽  
...  
Keyword(s):  
Nerve Stimulation ◽  
Hypoglossal Nerve ◽  
Sleep Onset ◽  
Chronic Insomnia ◽  
Apnea Hypopnea Index ◽  
Therapy Adherence ◽  
Hypoglossal Nerve Stimulation ◽  
Obstructive Sleep ◽  
Patient Reported ◽  
History Of

Abstract Introduction Hypoglossal nerve stimulation (HNS) is an efficacious option for treating moderate to severe obstructive sleep apnea (OSA). However, there is sparse evidence regarding tolerance and adherence to HNS therapy in patients with a diagnosis of insomnia. Report of case(s) A 57-year-old man with well-controlled depression presented for evaluation for HNS therapy. He had been diagnosed with moderate OSA with an apnea-hypopnea index of 22/hour, intolerant of continuous positive airway pressure and mandibular advancement device. He underwent uvulopalatopharyngoplasty without significant improvement. At the time of initial evaluation, he denied history of insomnia and prior sleep aid use. He subsequently underwent successful HNS device implantation and activation. One week after HNS initiation, the patient reported new symptoms of significant difficulty with sleep onset and inability to fall back asleep, which was worse than his untreated OSA symptoms. Device interrogation did not reveal any hardware problems. Adjustments to start delay, pause time and device configuration with awake endoscopy did not improve tolerance. Subsequently, the patient disclosed a remote history of insomnia, which was treated with multiple hypnotics in addition to cognitive-behavioral therapy for insomnia (CBTi) and had resolved. He was diagnosed with recurrent chronic insomnia, for which eszopiclone was initiated without significant improvement. He eventually agreed to CBTi, with partial improvement in device tolerance and improvement in insomnia symptoms. Conclusion This case highlights that HNS therapy adherence can be affected by prior history of, or a current diagnosis of insomnia. Our patient had a predisposition for insomnia that was well controlled prior to HNS therapy initiation. The onset of recurrent insomnia with HNS activation suggests that HNS was a precipitating factor for his now chronic insomnia. Although there is insufficient evidence to suggest whether history of insomnia should affect the decision to initiate HNS therapy, this case illustrates the importance of screening for insomnia at pre-implant evaluation. Our center is now routinely screening for a history of insomnia to identify patients who may benefit from treatment prior to HNS implantation. Larger studies are needed to explore a possible relationship between insomnia and HNS adherence. Support (if any):

scholarly journals 1227 A CASE OF A FEARFUL SLUMBER

SLEEP ◽  
2020 ◽  
Vol 43 (Supplement_1) ◽  
pp. A468-A469
Author(s):  
Aristotle Asis ◽  
Annise Georgette Wilson ◽  
Philip Mani Alapat
Keyword(s):  
Sleep Latency ◽  
Sleep Onset ◽  
Therapeutic Interventions ◽  
Apnea Hypopnea Index ◽  
Sleep Paralysis ◽  
Obstructive Sleep ◽  
Respiratory Disturbance ◽  
Fear And Anxiety ◽  
Daytime Function ◽  
Isolated Sleep Paralysis

Abstract Introduction Isolated sleep paralysis (ISP) occurs when rapid eye movement (REM)-based atonia intrudes into wakefulness, outside the context of narcolepsy, substance abuse, mental disorder or other medical conditions. No “gold standard” assessment and diagnostic instrument currently exists. Report of Case A 63-year old female with hypersomnia and positive airway pressure (PAP)-controlled obstructive sleep apnea was referred for recurrent episodes of paralysis during sleep-wake transitions, lasting 15-20 seconds, occurring every 2-3 years since the age of 15, and associated with fear and anxiety. Episodes were more frequent in the last 2 years after significant sleep deprivation and starting a weight loss supplement, BIO-X4, which contains green tea and probiotics. No cataplexy, or history of traumatic brain injury and stroke were identified. Epworth Sleepiness Scale score was 14 on armodafinil. Reported sleep amounts were regularly scheduled 6-7-hour periods, with no suggestion of circadian dysfunction. In 2016, polysomnogram showed Apnea-Hypopnea index of 2.6/hour, Respiratory Disturbance Index of 13.8/hour with oxygen nadir of 92% in the setting of hypersomnia. Continuous PAP of 11 cmH20 was initiated after a successful titration with controlled residual AHI during follow-ups. Multiple Sleep Latency Test during the same time revealed mean sleep latency of 5.5 minutes and no sleep-onset REM with 5 naps. Brain imaging and electroencephalogram were both normal as well as drug panel, blood counts, metabolic profile and thyroid function. Decreased episodes and severity of recurrent ISP were reported after discontinuation of the supplement. Apart from anxiety related to the episodes, the patient denied any interference with daytime function. Conclusion Isolated sleep paralysis is an important sleep disorder that requires proper evaluation to rule out competing diagnoses and consideration of therapeutic interventions. Likely associated with a lack of understanding and available literature, the prevalence in the general population is likely higher than what is currently perceived.

scholarly journals 1253 Multiple sleep onset REM episodes in middle age woman with excessive daytime sleepiness – Is this automatically assumed narcolepsy?

SLEEP ◽  
2020 ◽  
Vol 43 (Supplement_1) ◽  
pp. A477-A477
Author(s):  
Kamal Patel ◽  
Bianca J Lang
Keyword(s):  
Daytime Sleepiness ◽  
Sleep Latency ◽  
Sleep Onset ◽  
Sleep Time ◽  
Multiple Sleep Latency Test ◽  
Sleep Onset Latency ◽  
Onset Latency ◽  
Obstructive Sleep ◽  
Falling Asleep ◽  
Multiple Sleep Latency

Abstract Introduction Presence of sleep onset REM episodes often raises concerns of narcolepsy. However other conditions have shown to have presence of sleep on REM episodes which include but not limited to obstructive sleep apnea, sleep wake schedule disturbance, alcoholism, neurodegenerative disorders, depression and anxiety Report of Case Here we present a case of 30 year old female with history of asthma, patent foraman ovale, migraine headache, and anxiety who presented with daytime sleepiness, falling asleep while at work, occasional scheduled naps, non-restorative sleep, sleep paralysis, and hypnopompic hallucination. Pertinent physical exam included; mallampati score of 4/4, retrognathia, high arched hard palate, crowded posterior oropharynx. She had a score of 16 on Epworth sleepiness scale. Patient previously had multiple sleep latency test at outside facility which revealed 4/5 SOREM, with mean sleep onset latency of 11.5 minutes. She however was diagnosed with narcolepsy and tried on modafinil which she failed to tolerate. She was tried on sertraline as well which was discontinued due to lack of benefit. She had repeat multiple sleep latency test work up which revealed 2/5 SOREM, with mean sleep onset latency was 13.1 minutes. Her overnight polysomnogram prior to repeat MSLT showed SOREM with sleep onset latency of 10 minutes. Actigraphy showed consistent sleep pattern overall with sufficient sleep time but was taking hydroxyzine and herbal medication. Patient did not meet criteria for hypersomnolence disorder and sleep disordered breathing. Conclusion There is possibility her medication may have played pivotal role with her daytime symptoms. We also emphasize SOREMs can be present in other disorders such as anxiety in this case and not solely in narcolepsy

scholarly journals Randomized controlled trial of an integrated approach to treating insomnia and improving the use of positive airway pressure therapy in veterans with comorbid insomnia disorder and obstructive sleep apnea

SLEEP ◽  
2020 ◽  
Author(s):  
Cathy A Alessi ◽  
Constance H Fung ◽  
Joseph M Dzierzewski ◽  
Lavinia Fiorentino ◽  
Carl Stepnowsky ◽  
...  
Keyword(s):  
Obstructive Sleep Apnea ◽  
Sleep Apnea ◽  
Airway Pressure ◽  
Positive Airway Pressure ◽  
Sleep Onset ◽  
Apnea Hypopnea Index ◽  
Sleep Onset Latency ◽  
Pressure Therapy ◽  
Comorbid Insomnia ◽  
Obstructive Sleep

Abstract Study Objectives Cognitive behavioral therapy for insomnia (CBTI) for comorbid insomnia and obstructive sleep apnea (OSA) has had mixed results. We integrated CBTI with a positive airway pressure (PAP) adherence program and tested effects on sleep and PAP use. Methods 125 veterans (mean age 63.2, 96% men, 39% non-Hispanic white, 26% black/African American, 18% Hispanic/Latino) with comorbid insomnia and newly-diagnosed OSA (apnea-hypopnea index ≥ 15) were randomized to 5-weekly sessions integrating CBTI with a PAP adherence program provided by a “sleep coach” (with behavioral sleep medicine supervision), or 5-weekly sleep education control sessions. Participants and assessment staff were blinded to group assignment. Outcomes (baseline, 3 and 6 months) included Pittsburgh Sleep Quality Index (PSQI), 7-day sleep diary (sleep onset latency [SOL-D], wake after sleep onset [WASO-D], sleep efficiency [SE-D]), 7-day actigraphy (SE-A), and objective PAP use (hours/night and nights ≥ 4 h). Insomnia Severity Index (ISI), Epworth Sleepiness Scale (ESS), and Functional Outcomes of Sleep Questionnaire-10 (FOSQ-10) were also collected. Results Compared to controls, intervention participants showed greater improvement (baseline to 3 and 6 months, respectively) in PSQI (−3.2 and −1.7), SOL-D (−16.2 and −15.5 minutes), SE-D (10.5% and 8.5%), SE-A (4.4% and 2.6%) and more 90-day PAP use (1.3 and 0.9 more hours/night, 17.4 and 11.3 more nights PAP ≥ 4 h). 90-day PAP use at 3 months was 3.2 and 1.9 h/night in intervention versus controls. Intervention participants also had greater improvements in ISI, ESS, and FOSQ-10 (all p < 0.05). Conclusions An intervention integrating CBTI with a PAP adherence program delivered by a supervised sleep coach improved sleep and PAP use in adults with comorbid insomnia and OSA. Trial Registration ClinicalTrials.gov Study name: Novel Treatment of Comorbid Insomnia and Sleep Apnea in Older Veterans URL: https://clinicaltrials.gov/ct2/results?cond=&term=NCT02027558&cntry=&state=&city=&dist= Registration: NCT02027558

scholarly journals 0879 Outcomes of Adenotonsillectomy in Children with Down Syndrome and Obstructive Sleep Apnea

SLEEP ◽  
2020 ◽  
Vol 43 (Supplement_1) ◽  
pp. A335-A335
Author(s):  
A Morello Gearhart ◽  
B Gunaratnam ◽  
E Senthilvel
Keyword(s):  
Obstructive Sleep Apnea ◽  
Down Syndrome ◽  
Sleep Apnea ◽  
Sleep Onset ◽  
Sleep Efficiency ◽  
Apnea Hypopnea Index ◽  
P Value ◽  
Children With Down Syndrome ◽  
Arousal Index ◽  
Obstructive Sleep

Abstract Introduction Obstructive sleep apnea (OSA) is highly prevalent in children with Down Syndrome (DS). The aim of this study was to assess the effectiveness of adenotonsillectomy (T&A) on polysomnographic parameters of children with DS. Methods Retrospective chart review of children with DS who underwent T&A between 2012-2019 was performed. Preoperative OSA severity was categorized by obstructive apnea-hypopnea index (OAHI): mild = 1-4.9 events/h; moderate = 5-9.9 events/h; severe ≥ 10 events/h. Results We identified 43 DS children with pre and post T&A polysomnographic data in a population of 162 DS patients. A total of 25 were male, mean age 5.1 years (± 3.8 years) and 56% Caucasians. Preoperative data showed 19% mild OSA, 30% moderate and 51% severe. Postoperatively, apnea-hypopnea index (AHI) normalized in 9.3%, 37.2% had mild OSA, 18.6% moderate and 34.9% severe. Overall, T&A resulted in significant improvement (p-value <0.05) in mean AHI, (18.51 ± 28.05 vs 11.72 ± 16.43), SaO2 nadir (80.00 ± 14.82 vs 85.51 ± 5.94), sleep efficiency (81.97 ± 11.15 vs 85.9 ± 8.28), arousal index (16.14 ± 10.23 vs 14.45 ± 12.34), and wake after sleep onset (67.19 ± 46.89 vs 50.55 ± 40.83) and no statistical difference (p-value >0.05) in end-tidal carbon dioxide (43.86 ± 9.56 vs 44.17 ± 3.78), Rapid Eye Movement (REM)% (15.86 ± 7.75 vs 15.92 ± 7.41), sleep latency (24.03 ± 34.39 vs 22.55 ± 21.11), and central apnea index (0.86 ± 1.38 vs 0.66 ± 0.82) in pre and post T&A data. There was no statistically significant difference in pre and post T&A polysomnographic parameters between 17 DS and 17 age and gender-matched non-DS control subjects. Conclusion Adenotonsillectomy resulted in improvement in AHI, oxygen desaturation nadir, sleep efficiency, arousal index and wake after sleep onset. However, a significant portion of children with DS continued to have moderate to severe OSA after T&A. Support None.

358 Daridorexant Improves Sleep in Patients with Mild/Moderate Obstructive Sleep Apnea

SLEEP ◽  
2021 ◽  
Vol 44 (Supplement_2) ◽  
pp. A143-A143
Author(s):  
Marie-Laure Boof ◽  
Ingo Fietze ◽  
Katharina Lederer ◽  
Anne-Sophie Guern ◽  
Vincent Lemoine ◽  
...  
Keyword(s):  
Obstructive Sleep Apnea ◽  
Sleep Apnea ◽  
Sleep Duration ◽  
Sleep Onset ◽  
Sleep Architecture ◽  
Sleep Stages ◽  
Apnea Hypopnea Index ◽  
Treatment Difference ◽  
Obstructive Sleep ◽  
Sleep Parameters

Abstract Introduction Daridorexant is a dual orexin receptor antagonist developed for the treatment of insomnia. The effect of the highest phase-3 dose of 50 mg daridorexant on nighttime respiratory function was evaluated in patients with mild/moderate obstructive sleep apnea (OSA). This study showed that repeated doses of daridorexant had no clinically meaningful effect on the apnea-hypopnea index (AHI) or on peripheral oxygen saturation. In the same study, the effect on objective sleep parameters was also explored by polysomnography (PSG). Methods In this randomized, double-blind, placebo-controlled, two-period, crossover study, daridorexant or placebo was administered in each period once daily for 5 consecutive nights to 28 patients. Treatment difference (daridorexant – placebo) for total sleep time (TST), latency to persistent sleep (LPS), and wake after sleep onset (WASO) was analyzed for Night 5 using linear mixed-effects modeling. In addition, sleep was further explored based on sleep duration during each hour of PSG recording, duration of the different sleep phases (rapid eye movement [REM], non-REM [including N1 to N3 sleep stages]), as well as number and mean/longest duration of awakenings. Results Of 28 patients enrolled, 25 completed the study and were included in the analysis (n=15/10 with mild/moderate OSA; mean [standard deviation] AHI: 16.3 [8.2] events/h). One patient had mild insomnia symptoms at baseline. Compared to placebo, daridorexant prolonged mean TST by 38.8 min (90% confidence interval: 19.7–57.9), shortened mean LPS by 17.2 min (-35.5–1.02), and reduced mean WASO by 31.0 min (-47.3 to 14.7). Sleep architecture was maintained as no treatment differences in the duration of the evaluated sleep stages were observed when normalized to TST. Sleep duration was prolonged in the second part of the night. mean and longest duration of awakenings were decreased by a mean (90% CI) of 2.0 min (-3.1 to 0.9) and 16.3 min (-24.1 to -8.6), respectively, without treatment difference for the total number of awakenings. Conclusion Daridorexant improved objective sleep parameters in patients with mild to moderate OSA without modifying sleep architecture. Support (if any) Funded by Idorsia Pharmaceuticals Ltd.

Predictors of Patient-Reported Dysphagia and Reflux Symptoms in Obstructive Sleep Apnea

2021 ◽  
pp. 1-7
Author(s):  
Ankita M. Bhutada ◽  
William A. Broughton ◽  
Brenda L. Beverly ◽  
Dahye Choi ◽  
Sandip Barui ◽  
...  
Keyword(s):  
Obstructive Sleep Apnea ◽  
Sleep Apnea ◽  
Predictive Factors ◽  
Assessment Tool ◽  
Apnea Hypopnea Index ◽  
Reflux Symptom Index ◽  
Clinical Variables ◽  
Reflux Symptoms ◽  
Obstructive Sleep ◽  
Patient Reported

Purpose: Despite the reported high prevalence of dysphagia and reflux, patients with obstructive sleep apnea (OSA) are not routinely screened for dysphagia or reflux during conventional OSA management. The purpose of this exploratory study was to (a) identify prevalences of dysphagia and reflux self-reported symptoms in patients with OSA and (b) determine associations between dysphagia and reflux symptoms and demographic and clinical variables. Method: A chart review was completed on 75 patients with treated OSA during routine medical management at a university-affiliated sleep center. All participants completed the 10-Item Eating Assessment Tool (EAT-10) and Reflux Symptom Index (RSI) questionnaires, which are patient-reported outcome measures (PROMs) of swallowing and reflux symptoms, respectively. Relevant demographic and clinical variables were extracted. A binary logistic regression was computed to examine the relationships between dysphagia and reflux PROMs and demographic and clinical variables. Result: Sixty-three participants met eligibility criteria ( M age = 64 years). In addition, 14% ( n = 9) and 11% ( n = 7) of patients with OSA syndrome reported swallowing and reflux symptoms, respectively. Among the demographic and clinical variables investigated, there were no significant predictive factors for EAT-10 scores. Age and apnea–hypopnea index were significant predictive factors for RSI scores. Conclusions: To our knowledge, this is the first preliminary study to investigate swallowing and reflux symptoms concurrently in OSA, as well as potential mitigating factors. Although our findings suggest a relatively low percentage of patients with OSA report dysphagia and reflux symptoms, the true prevalences of swallowing and reflux disorders may be higher based on previous evidence suggesting that pharyngeal afferent (sensory) dysfunction may cause patients to underestimate their symptoms.

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