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.
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Video 1: Three months after continuing the administration of THP. THP improved the blepharospasm only minimally; however it did not influence the involuntary spasmodic contractions of the bilateral orbicularis oculi, levator labii superioris, and platysma. The leftward lingual deviation and cervical rotation also remained the same (MPG 2928 kb)
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Video 2: Nine months after continuing the administration of THP and 6 months after the first subcutaneous injection of BTX. Note that his blepharospasm almost disappeared. Also the movements such as the compression and sideward drawing of his lips resolved, however, it resulted in the obtrusiveness of the continuous leftward lingual deviation and mastication-like movements (MPG 1244 kb)
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Video 3: Ten months after continuing the administration of THP, 7 weeks after starting the ARP, and 3 weeks after the second injection of BTX. His residual spasmodic lingual deviation to the left and mastication-like movements were remarkably improved (MPG 2140 kb)
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Saito, T., Katayama, T., Sawada, J. et al. Combination therapy for segmental craniocervical dystonia (Meige syndrome) with aripiprazole, trihexyphenidyl, and botulinum toxin: three cases reports. Neurol Sci 36, 243–245 (2015). https://doi.org/10.1007/s10072-014-1927-x
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DOI: https://doi.org/10.1007/s10072-014-1927-x