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Transient total sleep loss in cerebral Whipple's disease: a longitudinal study
Author(s) -
Voderholzer Ulrich,
Riemann Dieter,
Gann Horst,
Hornyak Magdolna,
Juengling Freimut,
Schumacher Martin,
Reincke Martin,
Von Herbay Axel,
Nishino Seiji,
Mignot Emmanuel,
Berger Mathias,
Lieb Klaus
Publication year - 2002
Publication title -
journal of sleep research
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.297
H-Index - 117
eISSN - 1365-2869
pISSN - 0962-1105
DOI - 10.1046/j.1365-2869.2002.00311.x
Subject(s) - fatal familial insomnia , medicine , circadian rhythm , whipple's disease , insomnia , brainstem , melatonin , sleep (system call) , endocrinology , cerebrospinal fluid , anesthesia , disease , psychiatry , prion protein , computer science , operating system
SUMMARY A case with transient, almost complete sleep loss caused by cerebral manifestation of Whipple's disease (WD) is presented. Cerebral WD is rare and in most cases occurs after gastrointestinal infection. In our case, a progressive and finally almost complete sleep loss was the initial and predominant symptom. Polysomnographic studies in several consecutive nights and over 24 h showed a total abolition of the sleep–wake cycle with nocturnal sleep duration of less than 15 min. Endocrine tests revealed hypothalamic dysfunction with flattening of circadian rhythmicity of cortisol, TSH, growth hormone and melatonin. Cerebrospinal fluid (CSF) hypocretin was reduced. [ 18 F]Deoxyglucose positron emission tomography (FDG‐PET) revealed hypermetabolic areas in cortical and subcortical areas including the brainstem, which might explain sleep pathology and vertical gaze palsy. In the course of treatment with antibiotics and additional carbamazepine for 1 year, insomnia slowly and gradually improved. Endocrine investigations at 1‐year follow‐up showed persistent flattening of circadian rhythmicity. The FDG‐PET indicated normalized metabolism in distinct regions of the brain stem which paralleled restoration of sleep length. The extent of sleep disruption in this case of organic insomnia was similar to cases of familial fatal insomnia, but was at least partially reversible with treatment.