Familial paroxysmal dystonic choreoathetosis and response to alternate‐day oxazepam therapy

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Abstract
A patient with familial paroxysmal dystonic choreoathetosis (FPDC) who was only transiently improved on daily benzodiazepine therapy showed sustained benefit from a regimen of alternate‐day oxazepam therapy. Our findings confirm that FPDC is responsive to benzodiazepine therapy and suggest that an alternate‐day dosing schedule, using a short‐acting benzodiazepine, may minimize receptor down regulation and thereby sustain the therapeutic effect.

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