Bilateral paramedian thalamic infarctions (BPTIs) due to artery of Percheron occlusion are known to cause hypersomnia. However, the role of hypocretin-1, a wake-promoting peptide that is located at the lateral hypothalamus, in hypersomnia in these patients remains unclear.
To clarify the role of hypocretin-1 in hypersomnia in patients with BPTIs, hypocretin-1 levels in the cerebrospinal fluid (CSF) were measured in 6 patients with BPTIs: 2 with rostral midbrain involvement (BPT+RMI) and 4 without midbrain involvement (BPT-MI).
CSF hypocretin-1 levels were decreased in 2 patients with BPT+RMI and were within normal ranges in 4 patients with BPT-MI. Hypersomnia was noted in all the patients. In one BPT+RMI patient, hypersomnia was improved within 2 weeks and decreased CSF hypocretin-1 levels were reversed (acute phase (on day 9), 109.2 pg/mL; chronic phase (at 3 months), 323 pg/mL), whereas another BPT+RMI patient who displayed coma in the acute phase had decreased CSF orexin levels (107 pg/mL) at day 49 and exhibited severe disability.
Hypocretin deficiency was not involved in hypersomnia observed in BPT-MI patients; however, CSF hypocretin-1 levels were reduced in BPT+RMI patients. Reduced CSF hypocretin-1 levels in the chronic phase may possibly predict a poor clinical outcome in patients with Percheron artery infarction.
aDepartment of Neurology, Dokkyo Medical University, Tochigi, Japan
bDepartment of Neurology, Dokkyo Medical University Koshigaya Hospital, Saitama, Japan
cDepartment of Clinical Medicine for Nursing, Dokkyo Medical University School of Nursing, Tochigi, Japan
dDepartment of Nephrology, Shimonoseki City Hospital, Yamaguchi, Japan
eDepartment of Neurology, Fujita Health University School of Medicine, Aichi, Japan
fDepartment of Pediatrics, Epilepsy Center, Nishi-Niigata Chuo National Hospital, Niigata, Japan
gDepartment of Neuropsychiatry, Akita University School of Medicine, Akita, Japan
hInternational Institute for Integrative Sleep Medicine (WPI-IIIS), University of Tsukuba, Tsukuba, Japan.
Correspondence: Tomoyuki Miyamoto, Department of Neurology, Dokkyo Medical University Koshigaya Hospital, 2-1-50, Minami-Koshigaya, Koshigaya, Saitama, 343-8555, Japan (e-mail: firstname.lastname@example.org).
Abbreviations: AOP = artery of Percheron, BPT+RMI = bilateral paramedian thalamic infarction with rostral midbrain involvement, BPTI = bilateral paramedian thalamic infarction, BPT-MI = bilateral paramedian thalamic infarction without midbrain involvement, CSF = cerebrospinal fluid, MRI = magnetic resonance imaging.
There are no financial disclosures in regard to this article.
Authors’ contributions: study concept and design: KS, TM, MM, TK; acquisition of data: TM, MM, HM, KN, JT; analysis of data: KS, TM, MM, TK; drafting of the manuscript: KS; critical revision of the manuscript: TM, MM, KH, TS.
The authors declare that no potential conflicts of interest exist.
This is an open access article distributed under the Creative Commons Attribution License 4.0, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0
Received February 24, 2016
Received in revised form June 22, 2016
Accepted June 24, 2016