We report a mentally retarded 30-year-old woman with partial trisomy of chromosome 9 (46,XX-6,+der(6)t(6,9)pat) who has had epilepsy since age 11 months. She had been treated with various combinations of drugs. After 1 year of treatment with valproate (VPA) and ethosuximide (ESM), the patient developed arthralgias, muscle weakness, fatigue, and fever. Laboratory examination showed increased sedimentation rate, hypergammaglobulinemia, and high titers of antinuclear antibodies (ANA). The possibility of VPA-induced systemic lupus erythematosus (SLE) was considered. This diagnosis was supported by detection of antihistone antibodies and the HLA-DR4 antigen. VPA dosage was tapered and discontinued, with accompanying resolution of clinical, immunological and hematological signs of SLE 6 weeks after VPA discontinuation. This is the fourth reported case of VPA-induced SLE.

Valproate-induced systemic lupus erythematosus in a patient with partial trisomy of chromosome 9 and epilepsy / Gian Luigi, Gigli; Anna, Scalise; Pauri, Flavia; Giulia, Silvestri; Marina, Diomedi; Fabio, Placidi; Grazia M., Pomponi; Cesare, Masala. - In: EPILEPSIA. - ISSN 0013-9580. - ELETTRONICO. - 37:6(1996), pp. 587-588. [10.1111/j.1528-1157.1996.tb00614.x]

Valproate-induced systemic lupus erythematosus in a patient with partial trisomy of chromosome 9 and epilepsy

PAURI, FLAVIA;
1996

Abstract

We report a mentally retarded 30-year-old woman with partial trisomy of chromosome 9 (46,XX-6,+der(6)t(6,9)pat) who has had epilepsy since age 11 months. She had been treated with various combinations of drugs. After 1 year of treatment with valproate (VPA) and ethosuximide (ESM), the patient developed arthralgias, muscle weakness, fatigue, and fever. Laboratory examination showed increased sedimentation rate, hypergammaglobulinemia, and high titers of antinuclear antibodies (ANA). The possibility of VPA-induced systemic lupus erythematosus (SLE) was considered. This diagnosis was supported by detection of antihistone antibodies and the HLA-DR4 antigen. VPA dosage was tapered and discontinued, with accompanying resolution of clinical, immunological and hematological signs of SLE 6 weeks after VPA discontinuation. This is the fourth reported case of VPA-induced SLE.
1996
drug toxicity; hla-hr4; systemic lupus erythematosus; trisomy 9p; valproate
01 Pubblicazione su rivista::01a Articolo in rivista
Valproate-induced systemic lupus erythematosus in a patient with partial trisomy of chromosome 9 and epilepsy / Gian Luigi, Gigli; Anna, Scalise; Pauri, Flavia; Giulia, Silvestri; Marina, Diomedi; Fabio, Placidi; Grazia M., Pomponi; Cesare, Masala. - In: EPILEPSIA. - ISSN 0013-9580. - ELETTRONICO. - 37:6(1996), pp. 587-588. [10.1111/j.1528-1157.1996.tb00614.x]
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11573/131120
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