"Man-in-the-barrel" syndrome as delayed manifestation of extrapontine and central pontine myelinolysis: beneficial effect of intravenous immunoglobulin.
"Man-in-the-barrel" syndrome has been rarely described following osmotic myelinolysis. We report a case of a 45-year-old woman admitted with septicemia and severe hyponatremia. She presented with a "man-in-the-barrel" syndrome which developed more than 10 days after rapid correction of the hyponatremia. There was radiological evidence of central pontine and extrapontine myelinolysis. Three days after completing a course of intravenous immunoglobulin therapy (0.4 g/kg body weight/day for 5 days) there was considerable improvement (Expanded Disability Status Scale score improved 30%). This case, reported for its peculiar mode of development, unusual presentation and challenging therapeutic response to intravenous immunoglobulin, highlights the enigmatic and unpredictable aspects of osmotic myelinolysis.
Deleu
Dirk
D
Department of Medicine (Neurology), Hamad Medical Corporation, P.O. Box 3050, Doha, State of Qatar. doc_deleu@hotmail.com
Salim
Khalid
K
Mesraoua
Boulenouar
B
El Siddig
Abbas
A
Al Hail
Hassan
H
Hanssens
Yolande
Y
D001132:Arm; D001921:Brain / Q000473:pathology; D005260:Female; D006801:Humans; D007010:Hyponatremia / Q000097:blood; D007116:Immunization, Passive; D016756:Immunoglobulins, Intravenous / Q000627:therapeutic use*; D008279:Magnetic Resonance Imaging; D008875:Middle Aged; D017590:Myelinolysis, Central Pontine / Q000150:complications* / Q000473:pathology / Q000628:therapy*; D010243:Paralysis / Q000209:etiology* / Q000473:pathology / Q000628:therapy*; D018805:Sepsis / Q000150:complications; D012964:Sodium / Q000097:blood; D014882:Water-Electrolyte Balance / Q000502:physiology
D016756:Immunoglobulins, Intravenous; D012964:Sodium