“Man-in-the-barrel” syndrome as delayed manifestation of extrapontine and central pontine myelinolysis: Beneficial effect of intravenous immunoglobulin

• Published in: Journal of the neurological sciences Vol. 237; no. 1; pp. 103 - 106
• Main Authors: Deleu, Dirk, Salim, Khalid, Mesraoua, Boulenouar, El Siddig, Abbas, Al Hail, Hassan, Hanssens, Yolande
• Format: Journal Article
• Language: English
• Published: Shannon Elsevier B.V 15.10.2005; Elsevier Science
• Subjects: Arm; Biological and medical sciences; Brain - pathology; Central pontine myelinolysis; Extrapontine myelinolysis; Female; Humans; Hyponatremia; Hyponatremia - blood; Immunization, Passive; Immunoglobulins, Intravenous - therapeutic use; Intravenous immunoglobulin; Magnetic Resonance Imaging; Malformations of the nervous system; Man-in-the-barrel syndrome; Medical sciences; Metabolic diseases; Middle Aged; Myelinolysis, Central Pontine - complications; Myelinolysis, Central Pontine - pathology; Myelinolysis, Central Pontine - therapy; Neurology; Other nutritional diseases (malnutrition, nutritional and vitamin deficiencies...); Paralysis - etiology; Paralysis - pathology; Paralysis - therapy; Sepsis - complications; Sodium - blood; Water-Electrolyte Balance - physiology; Man-in-the-barrel syndrome; Intravenous immunoglobulin; Central pontine myelinolysis; Hyponatremia; Magnetic resonance imaging; Extrapontine myelinolysis; Human; Hydroelectrolytic balance disorder; Immunoglobulins; Nervous system diseases; Intravenous administration; Inorganic element; Nuclear magnetic resonance imaging; Metabolic disorder; Sodium; Man-in-the- barrel syndrome
• ISSN: 0022-510X; 1878-5883
• DOI: 10.1016/j.jns.2005.05.012

“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.