Hatori Kozo, Tagawa Yasuhiro, Hatano Taku, Akiyama Osamu, Izawa Nana, Kondo Akihide, Sato Kazunori, Watanabe Ayami, Hattori Nobutaka, Fujiwara Toshiyuki
Department of Rehabilitation Medicine, Juntendo University Graduate School, Tokyo, Japan.
Department of Neurology, Faculty of Medicine, Juntendo University, Tokyo, Japan.
Prog Rehabil Med. 2023 May 12;8:20230014. doi: 10.2490/prm.20230014. eCollection 2023.
Task-specific dystonia (TSD) confined to the lower extremities (LE) is relatively rare. This report describes dystonia confined to the LE only during forward walking. This case required careful neurological and diagnostic assessment because the patient was taking several neuropsychiatric drugs that cause symptomatic dystonia, such as aripiprazole (ARP).
A 53-year-old man visited our university hospital with a complaint of abnormalities in the LE that appeared only during walking. Neurological examinations other than walking were normal. Brain magnetic resonance imaging revealed meningioma in the right sphenoid ridge. The patient had been treated for depression with neuropsychiatric medications for a long time, and his abnormal gait appeared about 2 years after additional administration of ARP. After the meningioma was removed, his symptoms remained. Surface electromyography showed dystonia in both LE during forward walking, although his abnormal gait appeared to be accompanied by spasticity. The patient was tentatively diagnosed with tardive dystonia (TD). Although dystonia did not disappear clinically, it was alleviated after discontinuing ARP. Administration of trihexyphenidyl hydrochloride and concomitant rehabilitation improved his dystonia until return to work, but some residual gait abnormalities remained.
We report an unusual case of TD with task specificity confined to the LE. The TD was induced by the administration of ARP in combination with multiple psychotropic medications. Careful consideration was required for clinical diagnosis, rehabilitation, and assessment of its relevance to TSD.
局限于下肢（LE）的任务特异性肌张力障碍（TSD）相对罕见。本报告描述了仅在向前行走时局限于下肢的肌张力障碍。该病例需要仔细的神经学和诊断评估，因为患者正在服用几种可导致症状性肌张力障碍的神经精神药物，如阿立哌唑（ARP）。
一名53岁男性因仅在行走时出现下肢异常而就诊于我院。除行走外的神经学检查均正常。脑磁共振成像显示右侧蝶骨嵴有脑膜瘤。该患者长期使用神经精神药物治疗抑郁症，在额外服用ARP约2年后出现异常步态。脑膜瘤切除后，其症状仍存在。表面肌电图显示向前行走时双下肢均有肌张力障碍，尽管其异常步态似乎伴有痉挛。该患者初步诊断为迟发性肌张力障碍（TD）。尽管肌张力障碍在临床上未消失，但停用ARP后有所缓解。给予盐酸苯海索并同时进行康复治疗，其肌张力障碍得到改善直至恢复工作，但仍残留一些步态异常。
我们报告了一例不寻常的局限于下肢的具有任务特异性的TD病例。该TD是由服用ARP联合多种精神药物诱发的。临床诊断、康复治疗以及评估其与TSD的相关性时需要仔细考虑。
