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鞘内注射巴氯芬泵作为一种针对一例对多次毁损治疗无效的严重迟发性肌张力障碍的临时措施。

Intra thecal baclofen pump as an ad hoc measure for a case of severe tardive dystonia refractory to multiple lesioning.

作者信息

Kayastha Jessica, Shrestha Resha, Shrestha Bishal, Gurung Pritam, Dabadi Sambardhan, Dhungel Raju Raj, Pant Basant

机构信息

Department of Neurosurgery, Annapurna Neurological Institute and Allied Sciences, Maitighar, Kathmandu, Nepal.

出版信息

Ann Med Surg (Lond). 2023 Apr 11;85(5):1839-1842. doi: 10.1097/MS9.0000000000000126. eCollection 2023 May.

DOI:10.1097/MS9.0000000000000126
PMID:37228997
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10205201/
Abstract

UNLABELLED

Tardive dystonia is an infrequent ailment in patient reliant with chronic antipsychotic medication. The front-line envoy in the treatment of this illness is set into motion with oral agents including baclofen, benzodiazepines, and other antispasmodics. Regardless of an extensive therapy, the patients are not able to control of their spasticity/ dystonia. The authors reported a case of severe tardive dystonia treated with baclofen therapy in a patient frigid to medical therapy and multiple lesioning.

CASE REPORT

A 31-year-old female, diagnosed as a case of depressive illness and being managed with neuroleptic medications, who went onto develop tardive dystonia progressively worsening over a 4-year duration. After a comprehensive and meticulous evaluation of her neurological and psychological stratum, globus pallidus interna lesioning was reputed as the best course of action. As intended, staged lesioning was executed bilaterally with a trivial resolution eventually succumbing into recurrence, compelling a repeat lesioning. It was inaptly discouraging to see her crippled with the plight. Determined, not to give upon her, a way out with a baclofen therapy was proposed. A test dose with a 100 mcg of baclofen with an increment up to 150 mcg over a 3-day period demonstrated a promising prospect. On that account, the insertion of the baclofen pump was performed with an outstanding aftermath in her neurological endeavor.

CLINICAL DISCUSSION

Tardive dystonia is believed to be caused by striatal dopamine receptor super-sensitivity persuaded by the dopamine-antagonizing action of antipsychotic drugs. The first line of treatment being oral agents including oral baclofen, benzodiazepines, and antispasmodics. If the patient suffers from an early-onset primary generalized dystonia, then treatment with deep brain stimulation of the globus pallidus interna is the approved and preferred treatment approach. Recurrence of the symptoms despite of multiple lesioning can be overcome by intrathecal baclofen pump infusion as stated by many research. It is not uncommon to face complications in such a procedure, but the benefits outreach the risk, which makes it a choice of treatment.

CONCLUSION

The use of a continuous intrathecal baclofen pump for cases with tardive dystonia refractory to conventional therapy, it has been approved as one of the safest and capable procedures.

摘要

未标注

迟发性肌张力障碍在长期依赖抗精神病药物治疗的患者中是一种罕见疾病。治疗该疾病的一线药物包括口服药物,如巴氯芬、苯二氮䓬类药物及其他抗痉挛药物。尽管进行了广泛治疗,患者仍无法控制其痉挛/肌张力障碍。作者报告了一例严重迟发性肌张力障碍患者,该患者对药物治疗和多次毁损治疗均无反应,最终采用巴氯芬治疗。

病例报告

一名31岁女性,被诊断为抑郁症并接受抗精神病药物治疗,在4年时间里逐渐发展为迟发性肌张力障碍且病情不断恶化。在对其神经和心理层面进行全面细致评估后,双侧内侧苍白球毁损术被认为是最佳治疗方案。按计划进行了双侧分期毁损术,最初病情有所缓解,但最终复发,不得不再次进行毁损术。看到她因病情而致残令人非常沮丧。决心不放弃她,于是提出采用巴氯芬治疗。给予100微克巴氯芬的试验剂量,并在3天内逐渐增加至150微克,结果显示出良好前景。因此,植入巴氯芬泵后,她的神经功能恢复情况良好。

临床讨论

迟发性肌张力障碍被认为是由抗精神病药物的多巴胺拮抗作用导致纹状体多巴胺受体超敏所致。一线治疗药物为口服药物,包括口服巴氯芬、苯二氮䓬类药物及抗痉挛药物。如果患者患有早发性原发性全身性肌张力障碍,那么内侧苍白球深部脑刺激术是公认的首选治疗方法。许多研究表明,尽管进行了多次毁损术,症状仍复发,但鞘内注射巴氯芬泵输注可克服这一问题。在这种手术中出现并发症并不罕见,但益处大于风险,这使其成为一种治疗选择。

结论

对于传统治疗难治的迟发性肌张力障碍病例,使用持续鞘内巴氯芬泵治疗已被确认为最安全有效的方法之一。

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