Tuo Hou-Zhen, Tian Ze-Long, Cui Yi-Nong, Ma Xiao-Yang, Xu Chun-Ling, Bi Hong-Yan, Zhang Li-Yan, Zhang Yong-Bo, Le Wei-Dong, Ondo William
Department of Neurology, Capital Medical University Affiliated Beijing Friendship Hospital, Beijing 100050, China.
Department of Neurology, The Fourth Central Hospital of Tianjin, Tianjin 300140, China.
Chronic Dis Transl Med. 2017 Sep 1;3(3):186-190. doi: 10.1016/j.cdtm.2017.08.001. eCollection 2017 Sep.
Pontine infarction is a common type of stroke in the cerebral deep structures, resulting from occlusion of small penetrating arteries, may manifest as hemi-paralysis, hemi-sensory deficit, ataxia, vertigo, and bulbar dysfunction, but patients presenting with restless legs syndrome (RLS) are extremely rare. Herein, we reported five cases with RLS as a major manifestation of pontine infarction.
Five cases of pontine infarction related RLS were collected from July 2013 to February 2016. The diagnosis of RLS was made according to criteria established by the International RLS Study Group (IRLSSG) in 2003. Neurological functions were assessed according to the National Institutes of Health Stroke Scale (NIHSS) and modified Rankin Scale (mRS). Severity of RLS was based on the International RLS Rating Scale (IRLS-RS). Sleep quality was assessed by Epworth Rating Scale (ERS), and individual emotional and psychological states were assessed by Hamilton Depression Scale (HDS) and Hamilton Anxiety Scale (HAS).
The laboratory data at the onset including hemoglobin, serum concentration of homocysteine, blood urea nitrogen (BUN), creatinine, electrolytes, and thyroid hormones were normal. The electroencephalogram (EEG), lower-extremity somatosensory evoked potential (SEP), and nerve conduction velocity (NCV) in four limbs were normal. The average period of follow-up was 34.60 ± 12.76 months. The MRI examination showed acute or subacute pontine infarction lesions, 3 cases in the rostral inner side, 1 case in the rostral lateral and inner side, and 1 case in rostral lateral side. The neurological deficits included weakness in 4 cases, contralateral sensory deficit in 1 case, and ataxia in 2 cases. All 5 patients presented with symptom of RLS at or soon after the onset of infarction and 4 patients experienced uncomfortable sensations in the paralyzed limbs contralateral to the ischemic lesion. Their neurological deficits improved significantly 2 weeks later, but the symptoms of RLS did not resolve. Among them, 3/5 patients were treated with dopaminergic drugs. At the end of the follow-up, RLS symptom eventually resolved in 3 patients but persisted in two. The IRLS-RS, NIHSS and mRS scores were significantly lower at the onset than those at the last follow-up ( = 0.035, 0.024 and 0.049, respectively). However, there was no significant difference in the ERS, HDS and HAS scores ( = 0.477, 0.226 and 0.778, respectively).
RLS can be an onset manifestation of pontine infarction, clinicians should be aware of this potential symptom. RLS usually occurs in the paralyzed limbs contralateral to the infarction lesion. The pathogenesis still needs further investigation.
脑桥梗死是脑深部结构中常见的一种卒中类型，由小的穿通动脉闭塞所致，可表现为偏瘫、偏身感觉障碍、共济失调、眩晕及延髓功能障碍，但以不安腿综合征（RLS）为主要表现的患者极为罕见。在此，我们报告5例以RLS为主要表现的脑桥梗死病例。
收集2013年7月至2016年2月期间5例与脑桥梗死相关的RLS病例。RLS的诊断依据国际RLS研究组（IRLSSG）2003年制定的标准。根据美国国立卫生研究院卒中量表（NIHSS）和改良Rankin量表（mRS）评估神经功能。RLS的严重程度基于国际RLS评定量表（IRLS-RS）。通过Epworth评定量表（ERS）评估睡眠质量，通过汉密尔顿抑郁量表（HDS）和汉密尔顿焦虑量表（HAS）评估个体的情绪和心理状态。
发病时的实验室检查数据，包括血红蛋白、血清同型半胱氨酸浓度、血尿素氮（BUN）、肌酐、电解质及甲状腺激素均正常。脑电图（EEG）、下肢体感诱发电位（SEP）及四肢神经传导速度（NCV）均正常。平均随访时间为34.60±12.76个月。MRI检查显示急性或亚急性脑桥梗死灶，3例位于脑桥内侧上部，1例位于脑桥外侧及内侧上部，1例位于脑桥外侧上部。神经功能缺损包括4例肌无力，1例对侧感觉障碍，2例共济失调。所有5例患者在梗死发作时或发作后不久均出现RLS症状，4例患者在缺血灶对侧的瘫痪肢体有不适感。2周后其神经功能缺损明显改善，但RLS症状未缓解。其中，3/5的患者接受了多巴胺能药物治疗。随访结束时，3例患者的RLS症状最终缓解，但2例仍持续存在。发病时的IRLSSG、NIHSS及mRS评分显著低于末次随访时（分别为P=0.035、0.024和0.049）。然而，ERS、HDS及HAS评分无显著差异（分别为P=0.477、0.226和0.778）。
RLS可为脑桥梗死的首发表现，临床医生应意识到这种潜在症状。RLS通常发生在梗死灶对侧的瘫痪肢体。其发病机制仍需进一步研究。
