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.

Aripiprazole-induced oculogyric crisis (acute dystonia) in 11 years old girl: a case report

“…The periods of OGC can take minutes or hours, it may recur or become chronic. Cases of acute-and tardive-onset OGC have been reported with olanzapine, quetiapine, clozapine, risperidone, aripiprazole, and ziprasidone [9][10][11][12][13][14] . Patients with OGC experience severe distress and may refuse treatment when it recurs frequently.…”

“…The nigrostriatal dopamine pathway projects from the substantia nigra to the putamen and caudate nucleus. Antipsychotic agents cause acute dystonia by blocking dopamine D2 receptors in caudate, putamen, and globus pallidum 14 . The possible mechanism for the development of less extrapyramidal side effects result from aripiprazole is that it has partial agonistic activity to dopamine D2 and serotonin 5HT1A receptors and full antagonistic activity to the 5HT2A receptor [15][16][17] .…”

Psychogenic blepharospasm associated with Meige’s syndrome: a case report

“…Symptoms usually begin in the fifth or sixth decade of life with a two-fold predominance of women. Blepharospasm is the most frequent initial complaint of patients, while the other dystonic movements may gradually develop [1]. In majority of cases, Meige's syndrome develops secondary to structural lesions of the basal ganglia or the rostral brainstem [2].…”

“…In majority of cases, Meige's syndrome develops secondary to structural lesions of the basal ganglia or the rostral brainstem [2]. Meige's syndrome has been reported to be induced by several antipsychotics, including risperidone, olanzapine, quetiapine, and aripiprazole [1,3]. It was previously reported that Botulinum toxin (BTX) injections had only a moderate improvement rates for Meige's syndrome [1].…”

“…Meige's syndrome has been reported to be induced by several antipsychotics, including risperidone, olanzapine, quetiapine, and aripiprazole [1,3]. It was previously reported that Botulinum toxin (BTX) injections had only a moderate improvement rates for Meige's syndrome [1]. The other treatment options in Meige's syndrome include anticholinergics, benzodiazepines, baclofen, and tetrabenazine [4].…”