Limb-shaking syndrome derived from the contralateral hemisphere following unilateral revascularisation for moyamoya disease.
Limb-shaking syndrome derived from the contralateral hemisphere following unilateral revascularisation for moyamoya disease.
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DOI：
10.25259/sni_937_2021
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发表时间：
2021
影响因子：
--
通讯作者：
Nakada M
中科院分区：
文献类型：
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作者：
Demura M;Oishi M;Uchiyama N;Mohri M;Miyashita K;Nakada M
烟雾病是一种罕见的慢性狭窄闭塞性脑血管疾病。它可能具有与脑中风相关的各种临床症状，包括运动麻痹、感觉障碍、癫痫发作或头痛。然而，烟雾病患者很少出现不自主运动障碍，包括肢体震动综合征，以前没有报道在该疾病的血运重建术后发生肢体震动综合征。虽然分水岭移位在血运重建术后可引起短暂的神经功能恶化，但在血运重建术后，源自对侧半球的症状是罕见的。在这里，我们报告一例烟雾病，其中患者在单侧血运重建后发展为来自对侧半球的肢体颤抖综合征。一个16岁的女孩表现为短暂的左上肢和下肢麻木和头痛。基于数字减影血管造影，她被诊断为症状性烟雾病。单光子发射计算机断层扫描（SPECT）显示右侧脑血流量（CBF）减少，并在此侧进行了直接和间接旁路手术。术后立即出现右上肢不自主运动。SPECT显示双侧额叶CBF减少。随后，患者被诊断为抖肢综合征。在进行左半球血运重建术后，患者症状缓解，SPECT成像证实双侧额叶CBF改善。烟雾病的血运重建可导致分水岭转移，这可引起由血运重建侧对侧半球异常引起的肢体抖动综合征。对于烟雾重建术后新发肢体震颤综合征的患者，低灌注区域的治疗可能需要额外的血运重建术。
Moyamoya disease is a rare chronic steno-occlusive cerebrovascular disease. It may have variable clinical symptoms associated with cerebral stroke, including motor paralysis, sensory disturbances, seizures, or headaches. However, patients with moyamoya disease rarely present with involuntary movement disorders, including limb-shaking syndrome, with no previous reports of limb-shaking syndrome occurring after revascularization procedures for this disease. Although watershed shifts can elicit transient neurological deterioration after revascularisation, symptoms originating from the contralateral hemisphere following the revascularization procedure are rare. Here, we report the case of moyamoya disease wherein the patient developed limb-shaking syndrome derived from the contralateral hemisphere after unilateral revascularisation. A 16-year-old girl presented with transient left upper and lower limb numbness and headache. Based on digital subtraction angiography, she was diagnosed with symptomatic moyamoya disease. Single-photon emission computed tomography (SPECT) showed decreased cerebral blood flow (CBF) on the right side, and she underwent direct and indirect bypasses on this side. Involuntary movements appeared in her right upper limb immediately postoperatively. SPECT showed decreased CBF to the bilateral frontal lobes. Subsequently, the patient was diagnosed with limb-shaking syndrome. After performing left-hemispheric revascularisation, the patient’s symptoms resolved, and SPECT imaging confirmed improvements in CBF to the bilateral frontal lobes. Revascularization for moyamoya disease can lead to watershed shifts, which can induce limb-shaking syndrome derived from abnormalities in the contralateral hemisphere of the revascularized side. For patients with new-onset limb-shaking syndrome after moyamoya revascularisation procedures, additional revascularization may be warranted for treatment of low perfusion areas.