Carbamazepine
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Reactions 900 - 4 May 2002 S Carbamazepine Vanishing bile duct syndrome and Stevens-Johnson syndrome in a child: case report A 4-year-old boy developed vanishing bile duct and Stevens- Johnson syndromes after receiving carbamazepine therapy for seizures. The boy had been receiving carbamazepine [dosage not stated] for 4 months when he was admitted to hospital with a fever and a rapidly progressive necrotising rash. One month prior to admission, his serum carbamazepine concentration was 8 µg/ml. On admission, the boy had discrete and confluent erythematous macules over his face, trunk, extremities and genitalia; he also had erosions and crusting over his lips. His abdomen was distended and splenomegaly was noted. Laboratory tests revealed the following levels: total bilirubin 4 mg/dl, ALT 673 IU/L, AST 444 IU/L, alkaline phosphatase 481 IU/L, γ-glutamyl transpeptidase (GGT) 613 IU/L and cholesterol 200 mg/dl. An abdominal ultrasound revealed a heterogenic liver and a skin biopsy confirmed a diagnosis of Stevens-Johnson syndrome. Carbamazepine was discontinued and therapy with methylprednisolone and ursodeoxycholic acid was started. Over the next 2 days, the boy developed leucopenia. A liver biopsy on day 4 revealed that intrahepatic bile ducts were inconspicuous with epithelial destruction and intraepithelial lymphocytic infiltrates. The findings were consistent with early vanishing bile duct syndrome. The boy’s pancreatitis, hepatitis and Stevens-Johnson syndrome resolved rapidly, but his GGT and cholesterol levels remained elevated. On day 16, tacrolimus was started and, after one month of triple therapy, a repeat liver biopsy showed resolution of abnormalities. GGT and cholesterol levels normalised after 6 weeks. Author comment: ‘Our patient is the first child reported to have carbamazepine-associated acute VBDS [vanishing bile duct syndrome] and, to our knowledge, only the third case of pediatric acute drug-related VBDS in the literature.’ Garcia M, et al. Efficacy of early immunosuppressive therapy in a child with carbamazepine-associated vanishing bile duct and Stevens-Johnson syndromes. Digestive Diseases and Sciences 47: 1771-82, Jan 2002 - USA 800899392 1 Reactions 4 May 2002 No. 900 0114-9954/10/0900-0001/$14.95 Adis © 2010 Springer International Publishing AG. All rights reserved
Transcript of Carbamazepine
Reactions 900 - 4 May 2002
SCarbamazepine
Vanishing bile duct syndrome and Stevens-Johnsonsyndrome in a child: case report
A 4-year-old boy developed vanishing bile duct and Stevens-Johnson syndromes after receiving carbamazepine therapy forseizures.
The boy had been receiving carbamazepine [dosage notstated] for 4 months when he was admitted to hospital with afever and a rapidly progressive necrotising rash. One monthprior to admission, his serum carbamazepine concentrationwas 8 µg/ml.
On admission, the boy had discrete and confluenterythematous macules over his face, trunk, extremities andgenitalia; he also had erosions and crusting over his lips. Hisabdomen was distended and splenomegaly was noted.Laboratory tests revealed the following levels: total bilirubin 4mg/dl, ALT 673 IU/L, AST 444 IU/L, alkaline phosphatase 481IU/L, γ-glutamyl transpeptidase (GGT) 613 IU/L andcholesterol 200 mg/dl. An abdominal ultrasound revealed aheterogenic liver and a skin biopsy confirmed a diagnosis ofStevens-Johnson syndrome.
Carbamazepine was discontinued and therapy withmethylprednisolone and ursodeoxycholic acid was started.Over the next 2 days, the boy developed leucopenia. A liverbiopsy on day 4 revealed that intrahepatic bile ducts wereinconspicuous with epithelial destruction and intraepitheliallymphocytic infiltrates. The findings were consistent with earlyvanishing bile duct syndrome.
The boy’s pancreatitis, hepatitis and Stevens-Johnsonsyndrome resolved rapidly, but his GGT and cholesterol levelsremained elevated. On day 16, tacrolimus was started and,after one month of triple therapy, a repeat liver biopsy showedresolution of abnormalities. GGT and cholesterol levelsnormalised after 6 weeks.
Author comment: ‘Our patient is the first child reported tohave carbamazepine-associated acute VBDS [vanishing bileduct syndrome] and, to our knowledge, only the third case ofpediatric acute drug-related VBDS in the literature.’Garcia M, et al. Efficacy of early immunosuppressive therapy in a child withcarbamazepine-associated vanishing bile duct and Stevens-Johnson syndromes.Digestive Diseases and Sciences 47: 1771-82, Jan 2002 - USA 800899392
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Reactions 4 May 2002 No. 9000114-9954/10/0900-0001/$14.95 Adis © 2010 Springer International Publishing AG. All rights reserved
