title: Combination therapy for segmental craniocervical dystonia (Meige syndrome) with aripiprazole, trihexyphenidyl, and botulinum toxin: three cases reports.
abstract: Segmental craniocervical dystonia is characterized by blephalospasm and oromandibular dystonia and is also called Meige syndrome. The current treatment strategy including botulinum toxin (BTX) injections has not yet attained an acceptable level. We describe a long-term favorable response of a novel combination therapy with aripiprazole (ARP), trihexyphenidyl (THP), and BTX in three patients with segmental craniocervical dystonia. The symptoms of three patients responded promptly to the combination therapy with ARP 3-6 mg daily, THP 2-8 mg daily, and BTX. Although the patients were required to receive a BTX 50-100 IU injection every 3-6 months, their symptoms were kept in a satisfactory condition for up to 2 years without any adverse effects. ARP possesses the potential for dramatically improving dystonia. THP has the possibility to enhance the efficacy of ARP and prolong the effective period of BTX. It may be an important requisite to give all three agents together for a successful treatment. The combination therapy with ARP, THP, and BTX was well-tolerated and useful in controlling the symptoms of segmental craniocervical dystonia, however, the reason why this combination therapy succeeded is unknown. A further long-term follow-up is required to monitor the delayed neurological adverse effects.
authors.affiliation: Division of Cardiovascular, Respiratory and Neurology, Department of Internal Medicine, Asahikawa Medical University, Midorigaoka Higashi 2-1-1-1, Asahikawa, Hokkaido, 078-8510, Japan, tsukasa@asahikawa-med.ac.jp.
authors.forename: Tsukasa
authors.initials: T
authors.lastname: Saito
authors.forename: Takayuki
authors.initials: T
authors.lastname: Katayama
authors.forename: Jun
authors.initials: J
authors.lastname: Sawada
authors.forename: Kohei
authors.initials: K
authors.lastname: Kano
authors.forename: Asuka
authors.initials: A
authors.lastname: Asanome
authors.forename: Kae
authors.initials: K
authors.lastname: Takahashi
authors.forename: Nobuyuki
authors.initials: N
authors.lastname: Sato
authors.forename: Naoyuki
authors.initials: N
authors.lastname: Hasebe
mesh_terms: D000328:Adult; D018726:Anti-Dyskinesia Agents / Q000008:administration & dosage*; D000068180:Aripiprazole / Q000008:administration & dosage*; D001905:Botulinum Toxins / Q000008:administration & dosage*; D004359:Drug Therapy, Combination; D005260:Female; D006801:Humans; D008297:Male; D008538:Meige Syndrome / Q000188:drug therapy*; D008875:Middle Aged; D016896:Treatment Outcome; D014282:Trihexyphenidyl / Q000008:administration & dosage*
chemical_list: D018726:Anti-Dyskinesia Agents; D014282:Trihexyphenidyl; D000068180:Aripiprazole; D001905:Botulinum Toxins