Botulinum-toxin/clonazepam/unspecified anticholinergics

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Lack of efficacy: 2 case reports In a case series, two men [including one man in his early 30s; ages at event onset not stated] were described, of whom one man exhibited lack of efficacy during treatment with botulinum-toxin for dystonia while the other man exhibited lack of efficacy during treatment with botulinum-toxin, unspecified anticholinergics and clonazepam for dystonia [not all routes stated; dosages not stated]. Patient 1: An 19-year-old man had KMT2B mutation and generalised dystonia. His development slowly decelerated after birth, which resulted in short stature, cognitive decline, microcephaly and dysarthria. A left foot inversion developed at the age of 6 years while myoclonic movements in upper extremities and truncal bending developed at the age of 8 years. At the age of 15 years, the truncal bending worsened. He became wheelchair dependent. Over the time, he received various unspecified oral medications along with botulinum-toxin injection. However, the treatment was ineffective. Due to this, he was referred to hospital for surgical treatment (current presentation; at the age of 19 years). He underwent a left pallidotomy. Consequently, his right hand myoclonus improved. But, his trunk bending remained. Later, he underwent a right pallidothalamic tractotomy. Both his trunk bending and left hand myoclonus were improved. Eventually, he was able to walk independently. At two-year follow-up, his dystonic symptoms were stable. Patient 2: A 34-year-old man had KMT2B mutation and generalised dystonia. At 10 years of the age, he developed bilateral jerky hand movements. Later, he developed neck and trunk tilting to the left at the age of 32 years. He had received various oral medications including unspecified anticholinergics and clonazepam, as well as botulinum-toxin injections. However, the treatment was not effective. One year later, his axial symptoms deteriorated suddenly. He was referred to hospital for surgical treatment (current presentation; at the age of 34 years). He underwent a right pallidothalamic tractotomy. Consequently, his left hand myoclonic movements improved. However, his axial symptoms were still disabling. He was scheduled the contralateral side surgery. No significant postoperative complications were noted. Horisawa S, et al. Radiofrequency ablation for DYT-28 dystonia: short term follow-up of three adult cases. Annals of Clinical and Translational Neurology 7: 2047-2051, No. 803514922 10, Oct 2020. Available from: URL: http://doi.org/10.1002/acn3.51170

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Reactions 14 Nov 2020 No. 1830