Differential characteristics of repeated polysomnography and multiple sleep latency test parameters in narcolepsy type 1 and type 2 patients: a longitudinal retrospective study

Author(s):  
Yoo Hyun Um ◽  
Jihye Oh ◽  
Sung-Min Kim ◽  
Tae-Won Kim ◽  
Ho-Jun Seo ◽  
...  
2018 ◽  
Vol 27 (5) ◽  
pp. e12700 ◽  
Author(s):  
Yu-Shu Huang ◽  
Christian Guilleminault ◽  
Cheng-Hui Lin ◽  
Chia-Hsiang Chen ◽  
Wei-Chih Chin ◽  
...  

Neurology ◽  
2019 ◽  
Vol 93 (11) ◽  
pp. e1034-e1044 ◽  
Author(s):  
Fabio Pizza ◽  
Lucie Barateau ◽  
Isabelle Jaussent ◽  
Stefano Vandi ◽  
Elena Antelmi ◽  
...  

ObjectiveTo validate polysomnographic markers (sleep latency and sleep-onset REM periods [SOREMPs] at the Multiple Sleep Latency Test [MSLT] and nocturnal polysomnography [PSG]) for pediatric narcolepsy type 1 (NT1) against CSF hypocretin-1 (hcrt-1) deficiency and presence of cataplexy, as no criteria are currently validated in children.MethodsClinical, neurophysiologic, and, when available, biological data (HLA-DQB1*06:02 positivity, CSF hcrt-1 levels) of 357 consecutive children below 18 years of age evaluated for suspected narcolepsy were collected. Best MSLT cutoffs were obtained by receiver operating characteristic (ROC) curve analysis by contrasting among patients with available CSF hcrt-1 assay (n = 228) with vs without CSF hcrt-1 deficiency, and further validated in patients without available CSF hcrt-1 against cataplexy (n = 129).ResultsPatients with CSF hcrt-1 deficiency were best recognized using a mean MSLT sleep latency ≤8.2 minutes (area under the ROC curve of 0.985), or by at least 2 SOREMPs at the MSLT (area under the ROC curve of 0.975), or the combined PSG + MSLT (area under the ROC curve of 0.977). Although specificity and sensitivity of reference MSLT sleep latency ≤8 minutes and ≥2 SOREMPs (nocturnal SOREMP included) was 100% and 94.87%, the combination of MSLT sleep latency and SOREMP counts did not improve diagnostic accuracy. Age or sex also did not significantly influence these results in our pediatric population.ConclusionsAt least 2 SOREMPs or a mean sleep latency ≤8.2 minutes at the MSLT are valid and reliable markers for pediatric NT1 diagnosis, a result contrasting with adult NT1 criteria.Classification of evidenceThis study provides Class III evidence that for children with suspected narcolepsy, polysomnographic and MSLT markers accurately identify those with narcolepsy type 1.


2021 ◽  
Vol 12 ◽  
Author(s):  
Fang Deng ◽  
Yanan Zhang ◽  
Ran Zhang ◽  
Qi Tang ◽  
Zhenni Guo ◽  
...  

Objective: We aimed to investigate the dynamic cerebral autoregulation (dCA) in patients with central disorders of hypersomnolence during wakefulness.Methods: Thirty-six patients with central disorders of hypersomnolence were divided into three groups according to polysomnography and multiple sleep latency test results: the idiopathic hypersomnia group (IH), narcolepsy type 1 without rapid-eye-movement sleep behavior disorder group (NT1-RBD), and narcolepsy type 1 with rapid-eye-movement sleep behavior disorder group (NT1 + RBD), with 12 patients in each group. Twelve sex- and age-matched healthy controls were recruited. We assessed the Epworth sleepiness scale (ESS) and dCA of all subjects. dCA was assessed by analyzing the phase difference (PD) using transfer function analysis. The ESS and dCA were analyzed before and after standardized treatment in 24 patients with narcolepsy type 1.Results: The overall PD of the IH, NT1-RBD, and NT1 + RBD groups were lower than that of the control group (P < 0.001). There were no significant differences between the overall PD of the NT1-RBD and NT1 + RBD group (P > 0.05). The ESS scores decreased and the overall PD increased after treatment in 24 patients with narcolepsy type 1 (P < 0.001). Multivariable analysis showed that mean sleep latency in multiple sleep latency test was independently associated with impaired overall PD (P < 0.05).Conclusions: The dCA is impaired in patients with central disorders of hypersomnolence. The impairment of dCA occurs irrespective of NT1-RBD/+RBD. The ESS score and dCA improved in patients with narcolepsy type 1 after medication treatment. The mean sleep latency in multiple sleep latency test was independently associated with impaired dCA.Clinical Trial Registration:www.ClinicalTrials.gov, identifier: NCT02752139.


2017 ◽  
Vol 13 (12) ◽  
pp. 1441-1444 ◽  
Author(s):  
Yoo Hyun Um ◽  
Tae-Won Kim ◽  
Jong-Hyun Jeong ◽  
Ho-Jun Seo ◽  
Jin-Hee Han ◽  
...  

SLEEP ◽  
2020 ◽  
Vol 43 (Supplement_1) ◽  
pp. A477-A477
Author(s):  
Kamal Patel ◽  
Bianca J Lang

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


Author(s):  
Sulaiman Alhifzi ◽  
Nevin Zaki ◽  
Aljohara S. Almeneesier ◽  
Ahmed S. BaHammam

Despite varied classification systems, hypersomnolence disorders (or central disorders of hypersomnolence) are a group of disorders with a common symptom of excessive daytime sleepiness. In addition to a thorough clinical interview and examination, the assessment of hypersomnolence may require clinical investigations such as polysomnography and the multiple sleep latency test. This chapter examines the disorders of hypersomnolence with an emphasis on their clinical features and treatment strategies. The authors discuss the four main types of hypersomnolence disorders, narcolepsy type 1, narcolepsy type 2, idiopathic hypersomnia and Kleine–Levin syndrome, as well as hypersomnia due to a medical disorder, medication, or substance use; a psychiatric disorder; and insufficient sleep syndrome. It also discusses the relationships between hypersomnolence and psychiatric disorders.


1989 ◽  
Vol 47 (1) ◽  
pp. 76-79 ◽  
Author(s):  
Rubens Reimão ◽  
Aron Diament

A case of periodic hypersomnia in an 11-year-old female with the unique features of mental deficiency, incontinentia pigmenti, acanthosis nigricans and hereditary multiple exostosis (diaphysial aclasis) is reported. The clinical, Polysomnographic and Multiple Sleep Latency test features of this case with a follow up of seven years are consistent with a diagnosis of periodic (intermittent) excessive somnolence. The unique presentation, however, does differ from Kleine-Levin syndrome and suggests a relationship between the predominantly ectodermal, congenital disorders and the sleep-wake, pattern dysfunction.


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