Shioya Ayako, Takuma Hiroshi, Shiigai Masanari, Ishii Akiko, Tamaoka Akira
Department of Neurology, Faculty of Medicine, University of Tsukuba, Japan.
Department of Neurology, Faculty of Medicine, University of Tsukuba, Japan.
J Neurol Sci. 2014 Aug 15;343(1-2):203-5. doi: 10.1016/j.jns.2014.05.055. Epub 2014 Jun 2.
A 52-year-old Japanese woman complaining of horizontal double vision for 10 days was admitted to our hospital. Neurological examination revealed left abducent nerve palsy and muscle swelling in her thighs. Brain MRI showed obstruction in the spinal fluid space of the left Dorello's canal, which transmits a portion of the abducent nerve. In Ga-67-enhanced citrate scintigraphy, wide accumulation was seen in her bilateral thighs, lower legs, and gluteus muscles. Muscular MRI showed a star-shaped central structure on short tau inversion recovery (STIR) images, and the three stripes sign on T2-weighted images. These MRI findings indicated nodular-type muscular sarcoidosis. A muscle biopsy from the quadriceps femoris showed granulomatous epithelioid giant cells and non-necrotizing chronic lymphadenitis, which also indicate sarcoidosis. Her condition was considered to be caused by sarcoid granulomas obstructing Dorello's canal. She was treated with oral prednisolone (1 mg·kg(-1)·day(-1)) and her symptoms and MRI findings improved. This is the first known report of abducent nerve impairment in Dorello's canal, other than fetal hypoplasia. Brain MRI, muscular MRI, and muscle biopsy are useful for the diagnosis of abducent nerve palsy, and it is important to consider Dorello's canal obstruction by sarcoidosis. Complete remission can be achieved with proper treatment.
一名52岁的日本女性因出现水平复视10天而入院。神经系统检查发现其左侧展神经麻痹及大腿肌肉肿胀。脑部磁共振成像（MRI）显示，在传导展神经一部分的左侧多雷洛管脑脊液间隙存在梗阻。在镓-67增强柠檬酸盐闪烁扫描中，其双侧大腿、小腿及臀肌出现广泛放射性聚集。肌肉MRI在短tau反转恢复（STIR）图像上显示出星状中央结构，在T2加权图像上显示出三条带征。这些MRI表现提示为结节型肌肉结节病。股四头肌肌肉活检显示有肉芽肿性上皮样巨细胞及非坏死性慢性淋巴结炎，这也提示结节病。她的病情被认为是由结节病肉芽肿阻塞多雷洛管所致。给予她口服泼尼松龙（1毫克·千克⁻¹·天⁻¹）治疗，其症状及MRI表现均有改善。这是除胎儿发育不全外，关于多雷洛管展神经损伤的首例已知报道。脑部MRI、肌肉MRI及肌肉活检对展神经麻痹的诊断很有用，且考虑结节病导致多雷洛管梗阻很重要。经过适当治疗可实现完全缓解。
