Li Jiangang, Chen Yingcong, Liu Jianxue, Mai Xingsheng, Jing Shaohua
Departments of Neurology.
Departments of Ultrasonic of Medicine.
Medicine (Baltimore). 2020 Oct 23;99(43):e22920. doi: 10.1097/MD.0000000000022920.
Reversible splenial lesion syndrome (RESLES) is a recently identified clinico-radiological syndrome, the etiology is miscellaneous. Atrial septal defect (ASD) as an underlying etiology for RESLES has not been reported. We first report a rare case of RESLES associated with ASD. The clinical, radiological, and ultrasonic profiles were presented and the pathophysiological mechanism was analyzed.
A 23-year-old man presented with headache, drowsiness, occasional paraphasia, and paroxysmal dry cough. Brain magnetic resonance imaging (MRI) on admission showed an ovoid isolated lesion in the splenium of corpus callosum, which exhibited hyperintensity on diffusion-weighted imaging and hypointensity on apparent diffusion coefficient, and completely disappeared on the follow-up MRI 14 days later. ASD was found by transthoracic echocardiography, Right-to-left shunts were detected on color Doppler of transesophageal echocardiography, and microemboli were captured by transcranial Doppler ultrasound.
According to his clinical history and imaging results, we confirmed the diagnosis of RESLES associated with ASD.
The patient was treated by oral aspirin and lopidogrel sulfate to inhibit platelet aggregation. In addition, oral nimodipine to suppress vasoconstriction.
After 14 days treatment, all the symptoms presenting on admission resolved completely. Subsequently, a repair surgery of ASD under thoracoscopy was successfully performed.
To our knowledge, this is the first reported case of ASD may be an underlying etiology for RESLES and need require an etiotropic treatment.
可逆性胼胝体压部病变综合征（RESLES）是一种最近才被确认的临床放射学综合征，其病因多种多样。房间隔缺损（ASD）作为RESLES的潜在病因尚未见报道。我们首次报告了1例与ASD相关的RESLES罕见病例。介绍了其临床、放射学及超声特征，并分析了病理生理机制。
一名23岁男性，出现头痛、嗜睡、偶尔言语错乱及阵发性干咳。入院时脑部磁共振成像（MRI）显示胼胝体压部有一个椭圆形孤立病灶，在扩散加权成像上呈高信号，在表观扩散系数成像上呈低信号，14天后的随访MRI显示病灶完全消失。经胸超声心动图发现ASD，经食管超声心动图彩色多普勒检测到右向左分流，经颅多普勒超声捕获到微栓子。
根据其临床病史及影像学结果，确诊为与ASD相关的RESLES。
给予患者口服阿司匹林及硫酸氯吡格雷以抑制血小板聚集。此外，口服尼莫地平以抑制血管收缩。
经过14天治疗，入院时出现的所有症状完全缓解。随后，成功进行了胸腔镜下ASD修补手术。
据我们所知，这是首例报道的ASD可能是RESLES潜在病因的病例，且需要进行针对性治疗。
