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Introduction: Trihexyphenidyl is 1 of the most effective agents for treatment of young-onset dystonia. As such, women of childbearing potential use trihexyphenidyl despite inadequate information about potential effects on pregnancy, labor, and fetal development.

Case Report: We report 2 uncomplicated pregnancies in 1 woman with early-onset, sporadic, primary generalized dystonia (DYT1 negative) treated with high dosage trihexyphenidyl and review the literature on antidystonic agents and pregnancy.

Conclusion: Although there is limited data, our case demonstrates that high-dosage trihexyphenidyl treatment is not necessarily a contraindication to pregnancy.

(C) 2011 Lippincott Williams & Wilkins, Inc.