TY - JOUR
T1 - Post-Encephalitic Parkinsonism and Sleep
Disorder Responsive to Immunological
Treatment: A Case Report
AU - Brunetti, Valerio
AU - Testani, Elisa
AU - Iorio, Raffaele
AU - Frisullo, Giovanni
AU - Luigetti, Marco
AU - Di Giuda, Daniela
AU - Della Marca, Giacomo
PY - 2016
Y1 - 2016
N2 - We describe a 70-year-old man who, after a viral encephalitis associated with pneumonia, progressively developed a parkinsonism
associated with lethargy. Encephalitis manifested with persistent hiccups, seizures and impairment of consciousness. After 2
weeks, the initial neurologic symptoms subsided and the patient progressively developed movement disorders (rigidity and
bradykinesia, resistant to L-DOPA), lethargy and behavioral hypersomnia. Magnetic resonance imaging showed thalamic and
hippocampal signal abnormalities, immunohistochemistry on a mouse brain substrate revealed serum autoantibodies binding to
the brainstem neuropil. Polysomnographic monitoring was consistent with a very severe disruption of sleep: the sleep-wake
cycle was fragmented, and the NREM-REM ultradian cycle was irregular. Intravenous immune globulin therapy resulted in the
complete reversal of the movement and the sleep disorders. Our observation confirms that parkinsonism and sleep disorders
may be consequences of encephalitis, that an immune-mediated pathogenesis is likely, and, consequently, that immunotherapy
can be beneficial in these patients. The polysomnographic monitoring suggests that lethargia, rather than a mere hypersomnia,
is the result of a combination between sleep disruption and altered motor control.
AB - We describe a 70-year-old man who, after a viral encephalitis associated with pneumonia, progressively developed a parkinsonism
associated with lethargy. Encephalitis manifested with persistent hiccups, seizures and impairment of consciousness. After 2
weeks, the initial neurologic symptoms subsided and the patient progressively developed movement disorders (rigidity and
bradykinesia, resistant to L-DOPA), lethargy and behavioral hypersomnia. Magnetic resonance imaging showed thalamic and
hippocampal signal abnormalities, immunohistochemistry on a mouse brain substrate revealed serum autoantibodies binding to
the brainstem neuropil. Polysomnographic monitoring was consistent with a very severe disruption of sleep: the sleep-wake
cycle was fragmented, and the NREM-REM ultradian cycle was irregular. Intravenous immune globulin therapy resulted in the
complete reversal of the movement and the sleep disorders. Our observation confirms that parkinsonism and sleep disorders
may be consequences of encephalitis, that an immune-mediated pathogenesis is likely, and, consequently, that immunotherapy
can be beneficial in these patients. The polysomnographic monitoring suggests that lethargia, rather than a mere hypersomnia,
is the result of a combination between sleep disruption and altered motor control.
KW - encephalitis, hypersonnia, parkinsonism
KW - encephalitis, hypersonnia, parkinsonism
UR - http://hdl.handle.net/10807/95489
U2 - DOI: 10.1177/1550059416645706
DO - DOI: 10.1177/1550059416645706
M3 - Article
SN - 1550-0594
VL - 2016
SP - 324
EP - 329
JO - Clinical EEG and Neuroscience
JF - Clinical EEG and Neuroscience
ER -