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Case report The authors present a case of a patient with an abdominal CSF pseudocyst that resulted from an allergic reaction to silicone. The patient underwent repair surgery of the meningomyelocele associated with the Chiari II malformation, and the V-P shunt was instituted at 6 months of age. A formation of the abdominal CSF pseudocyst and the consequent shunt malfunction were observed 40 days after the V-P shunt. An increase in the number of the peripheral eosinophils and serum immunoglobulin E (IgE), and an infiltration of eosinophils in the specimen harvested from the pseudocyst wall suggested an allergic reaction as the cause of the pseudocyst. A sixth operation to revise the V-P shunt was performed using the shunt system made of ‘extracted silicone’, which was produced extracting the allergic substances. Outcome The serum IgE was normalized after surgery and the abdominal CSF pseudocyst has not recurred for 22 months. [ABSTRACT FROM AUTHOR]