Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness
Yuki Omori,1 Takashi Kanbayashi,1,2 Aya Imanishi,1 Ko Tsutsui,1 Yohei Sagawa,1 Yuka S Kikuchi,1 Masahiro Takeshima,1 Kazuhisa Yoshizawa,1 Sachiko Uemura,3 Tetsuo Shimizu1,2 1Department of Neuropsychiatry, Akita University Graduate School of Medicine, Akita, Japan; 2International Institute for Integ...
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Dove Medical Press
2018
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oai:doaj.org-article:e85ca90cb28f4bba99064e23c91ee4e82021-12-02T04:00:37ZOrexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness1178-2021https://doaj.org/article/e85ca90cb28f4bba99064e23c91ee4e82018-02-01T00:00:00Zhttps://www.dovepress.com/orexinhypocretin-levels-in-the-cerebrospinal-fluid-and-characteristics-peer-reviewed-article-NDThttps://doaj.org/toc/1178-2021Yuki Omori,1 Takashi Kanbayashi,1,2 Aya Imanishi,1 Ko Tsutsui,1 Yohei Sagawa,1 Yuka S Kikuchi,1 Masahiro Takeshima,1 Kazuhisa Yoshizawa,1 Sachiko Uemura,3 Tetsuo Shimizu1,2 1Department of Neuropsychiatry, Akita University Graduate School of Medicine, Akita, Japan; 2International Institute for Integrative Sleep Medicine (WPI-IIIS), University of Tsukuba, Tsukuba, Japan; 3Department of Physical Therapy, Akita University Graduate School of Health Sciences, Akita, Japan Purpose: Myotonic dystrophy type 1 (DM1) is often characterized by excessive daytime sleepiness (EDS) and sleep-onset rapid eye movement periods caused by muscleblind-like protein 2. The EDS tends to persist even after treatment of sleep apnea. We measured the cerebrospinal fluid (CSF) orexin levels in DM1 patients with EDS and compared the clinical characteristics with narcolepsy type 1 and idiopathic hypersomnia (IHS) patients.Patients and methods: We measured the CSF orexin levels in 17 DM1 patients with EDS and evaluated subjective sleepiness using the Epworth Sleepiness Scale (ESS), objective sleepiness using mean sleep latency (MSL), and sleep apnea using apnea-hypopnea index (AHI). We compared the ESS scores and MSL between decreased (≤200 pg/mL) and normal (>200 pg/mL) CSF orexin group in DM1 patients. Furthermore, we compared the CSF orexin levels, ESS scores, MSL, and AHI among patients with DM1, narcolepsy type 1 (n=46), and IHS (n=30).Results: Seven DM1 patients showed decreased CSF orexin levels. There were significant differences in the ESS scores and MSL between decreased and normal CSF orexin groups in DM1 patients. The ESS scores showed no significant difference among patients with DM1, narcolepsy type 1, and IHS. The MSL in DM1 and IHS patients were significantly higher than narcolepsy type 1 patients (p=0.01, p<0.001). The AHI in DM1 patients was significantly higher than narcolepsy type 1 patients (p=0.042) and was insignificantly different from IHS patients. The CSF orexin levels in DM1 patients were significantly lower than IHS patients and higher than narcolepsy type 1 patients (p<0.001, p<0.001).Conclusion: The CSF orexin levels of DM1 patients moderately decreased compared to those of IHS patients as the control group. However, the EDS of DM1 patients may not be explained by only orexin deficiency. Keywords: myotonic dystrophy type 1, orexin, narcolepsy, sleep-onset rapid eye movement periods, muscleblind-like proteinOmori YKanbayashi TImanishi ATsutsui KSagawa YKikuchi YSTakeshima MYoshizawa KUemura SShimizu TDove Medical Pressarticlemyotonic dystrophy type 1orexinnarcolepsysleep-onset rapid eye movement periodsmuscleblind-like proteinNeurosciences. Biological psychiatry. NeuropsychiatryRC321-571Neurology. Diseases of the nervous systemRC346-429ENNeuropsychiatric Disease and Treatment, Vol Volume 14, Pp 451-457 (2018) |
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DOAJ |
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EN |
| topic |
myotonic dystrophy type 1 orexin narcolepsy sleep-onset rapid eye movement periods muscleblind-like protein Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429 |
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myotonic dystrophy type 1 orexin narcolepsy sleep-onset rapid eye movement periods muscleblind-like protein Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429 Omori Y Kanbayashi T Imanishi A Tsutsui K Sagawa Y Kikuchi YS Takeshima M Yoshizawa K Uemura S Shimizu T Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| description |
Yuki Omori,1 Takashi Kanbayashi,1,2 Aya Imanishi,1 Ko Tsutsui,1 Yohei Sagawa,1 Yuka S Kikuchi,1 Masahiro Takeshima,1 Kazuhisa Yoshizawa,1 Sachiko Uemura,3 Tetsuo Shimizu1,2 1Department of Neuropsychiatry, Akita University Graduate School of Medicine, Akita, Japan; 2International Institute for Integrative Sleep Medicine (WPI-IIIS), University of Tsukuba, Tsukuba, Japan; 3Department of Physical Therapy, Akita University Graduate School of Health Sciences, Akita, Japan Purpose: Myotonic dystrophy type 1 (DM1) is often characterized by excessive daytime sleepiness (EDS) and sleep-onset rapid eye movement periods caused by muscleblind-like protein 2. The EDS tends to persist even after treatment of sleep apnea. We measured the cerebrospinal fluid (CSF) orexin levels in DM1 patients with EDS and compared the clinical characteristics with narcolepsy type 1 and idiopathic hypersomnia (IHS) patients.Patients and methods: We measured the CSF orexin levels in 17 DM1 patients with EDS and evaluated subjective sleepiness using the Epworth Sleepiness Scale (ESS), objective sleepiness using mean sleep latency (MSL), and sleep apnea using apnea-hypopnea index (AHI). We compared the ESS scores and MSL between decreased (≤200 pg/mL) and normal (>200 pg/mL) CSF orexin group in DM1 patients. Furthermore, we compared the CSF orexin levels, ESS scores, MSL, and AHI among patients with DM1, narcolepsy type 1 (n=46), and IHS (n=30).Results: Seven DM1 patients showed decreased CSF orexin levels. There were significant differences in the ESS scores and MSL between decreased and normal CSF orexin groups in DM1 patients. The ESS scores showed no significant difference among patients with DM1, narcolepsy type 1, and IHS. The MSL in DM1 and IHS patients were significantly higher than narcolepsy type 1 patients (p=0.01, p<0.001). The AHI in DM1 patients was significantly higher than narcolepsy type 1 patients (p=0.042) and was insignificantly different from IHS patients. The CSF orexin levels in DM1 patients were significantly lower than IHS patients and higher than narcolepsy type 1 patients (p<0.001, p<0.001).Conclusion: The CSF orexin levels of DM1 patients moderately decreased compared to those of IHS patients as the control group. However, the EDS of DM1 patients may not be explained by only orexin deficiency. Keywords: myotonic dystrophy type 1, orexin, narcolepsy, sleep-onset rapid eye movement periods, muscleblind-like protein |
| format |
article |
| author |
Omori Y Kanbayashi T Imanishi A Tsutsui K Sagawa Y Kikuchi YS Takeshima M Yoshizawa K Uemura S Shimizu T |
| author_facet |
Omori Y Kanbayashi T Imanishi A Tsutsui K Sagawa Y Kikuchi YS Takeshima M Yoshizawa K Uemura S Shimizu T |
| author_sort |
Omori Y |
| title |
Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| title_short |
Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| title_full |
Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| title_fullStr |
Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| title_full_unstemmed |
Orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| title_sort |
orexin/hypocretin levels in the cerebrospinal fluid and characteristics of patients with myotonic dystrophy type 1 with excessive daytime sleepiness |
| publisher |
Dove Medical Press |
| publishDate |
2018 |
| url |
https://doaj.org/article/e85ca90cb28f4bba99064e23c91ee4e8 |
| work_keys_str_mv |
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