Sato Sumito, Shibahara Ichiyo, Inoue Yusuke, Hide Takuichiro, Kumabe Toshihiro
Department of Neurosurgery, Kitasato University School of Medicine, Sagamihara, Kanagawa, Japan.
Department of Neurosurgery, Kitasato University School of Medicine, Sagamihara, Kanagawa, Japan.
World Neurosurg. 2019 Mar;123:464-468.e1. doi: 10.1016/j.wneu.2018.11.161. Epub 2018 Nov 26.
Hypertrophic olivary degeneration (HOD) is a rare neurological condition of trans-synaptic degeneration caused by disruption of the dentatorubro-olivary pathway. We present new radiologic findings of HOD in 2 cases of brainstem lymphoma.
A 35-year-old woman (Case 1) and a 69-year-old man (Case 2) presented with remarkably similar clinical courses. The primary lesion was located at the dorsal pons extending to the midbrain. Pathologic diagnosis of diffuse large B-cell lymphoma was obtained after surgical resection. Complete remission of the primary lesion was achieved by treatment with 3 courses of high-dose methotrexate and radiotherapy. Arterial spin-labeling and T-weighted imagings showed high signal intensity in the inferior olive (IO) at some time after the operation. Slight contrast enhancement in the IO was also found in Case 1. These radiologic findings nearly misled us into a diagnosis of recurrence of lymphoma. Signal intensity in the IO on arterial spin-labeling imaging changed with time. Normalized regional cerebral blood flow (rCBF) in the IO was defined as a percentage of rCBF to the global cerebral blood flow calculated using automated software. Chronologic change in normalized rCBF in the IO revealed a large peak in Case 1, but only a mild increase in Case 2. Neurological findings demonstrated severe oculopalatal tremor in Case 1 and mild palatal tremor in Case 2.
Hyperperfusion and contrast enhancement in the IO were found in 2 patients with HOD. These findings may be confused with recurrence of malignant tumor.
肥大性橄榄核变性(HOD)是一种罕见的跨突触变性神经疾病,由齿状红核橄榄束通路中断引起。我们报告2例脑干淋巴瘤患者HOD的新影像学表现。
一名35岁女性(病例1)和一名69岁男性(病例2)临床表现极为相似。原发灶位于脑桥背侧并延伸至中脑。手术切除后病理诊断为弥漫性大B细胞淋巴瘤。经3个疗程大剂量甲氨蝶呤治疗及放疗后,原发灶完全缓解。术后一段时间,动脉自旋标记和T加权成像显示下橄榄核(IO)呈高信号强度。病例1的IO也有轻微强化。这些影像学表现几乎误导我们诊断为淋巴瘤复发。动脉自旋标记成像上IO的信号强度随时间变化。IO的标准化局部脑血流量(rCBF)定义为使用自动化软件计算的rCBF占全脑血流量的百分比。IO标准化rCBF的时间变化显示病例1有一个大峰值,而病例2只有轻度增加。神经学检查发现病例1有严重的动腭震颤,病例2有轻度腭震颤。
2例HOD患者均发现IO有血流灌注增加和强化。这些表现可能与恶性肿瘤复发相混淆。