Carpal tunnel syndrome and benign recurrent intrahepatic cholestasis are rare conditions in childhood. Benign intrahepatic cholestasis is characterized by repeated self-limited attacks of cholestasis that can start at any age and last from weeks to months. The patients are asymptomatic between these attacks. We report a 16 year-old male patient with benign recurrent intrahepatic cholestasis who developed carpal tunnel syndrome during a cholestatic attack. He was admitted with complaints of jaundice, pruritus and pain, tingling and muscle weakness in both hands for 15 days. Nerve conduction studies revealed findings compatible with carpal tunnel syndrome. He was started on ursodeoxycholic acid, fat soluble vitamins and cholestyramine and cholestasis regressed after four weeks of therapy. With the improvement of cholestasis, the symptoms of carpal tunnel syndrome also disappeared. In conclusion, benign recurrent intrahepatic cholestasis can be a rare cause of carpal tunnel syndrome in childhood. We also advocate treating the underlying disease as an appropriate conservative treatment before surgery.
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Metoclopramide is widely used as an antiemetic and a prokinetic agent. Both the antiemetic properties and side effects of the drug are the result of dopamine receptor antagonism within the central nervous system. Therapeutic doses of metoclopramide can produce adverse effects. A 5-month-old girl was referred to our emergency department with the pre-diagnosis of afebrile convulsion. In her medical history, she was mistakenly given 2 mg/kg metoclopramide within a 24 h period, after which she became hypertonic and exhibited intermittent opisthotonos. Complete blood count, electrolytes, liver and renal function tests, blood gas analysis, and urinalysis were all within normal limits. Electroencephalogram, brain CT and cerebrospinal fluid examination were normal. Metoclopramide treatment was discontinued and she was treated with biperiden, which led to an improvement in symptoms after 15 minutes and complete remission in 60 minutes. Intermittent opisthotonos may be confused with convulsion in infant and thus lead to an unnecessary hospital admission. Physicians should be aware that metoclopramide is widely used in the pediatric population and children are susceptible to the side effects of metoclopramide and the side effects may present as “intermittent opisthotonos” as observed in our patient.