A previously healthy 19-year-old Chinese man presented with weakness of his right limbs that rapidly worsened over a short interval. He had initially felt a weakness of his right lower limb six months prior to presentation. An X-ray of his right ankle at that time had been normal and no treatment was given. Two months ago, our patient felt a weakness of his right upper limb and the symptoms of his right lower limb worsened. At the same time, he developed fever (highest temperature, 39.5°C), blurred vision in his left eye and a mild episodic headache lasting for several minutes on each occasion. No nausea or vomiting occurred. Computed tomography at an outside hospital showed numerous low-density cysts and calcifications scattered throughout both sides of his brain. The boundary of each cyst was clear with a high-density ring, sometimes calcified. Brain magnetic resonance imaging demonstrated extensive cerebral white matter leukoencephalopathy. Numerous cysts of various sizes were scattered throughout his hemispheres, thalamus, basal ganglia and left ventricle. The boundaries of the cysts were hyperintense on both T1- and T2-weighted images and gave a low signal on the fluid-attenuated inversion recovery image. Enhancement was observed in the lining of the cyst wall. The cystic content was heterointense in the T1-weighted and fluid-attenuated inversion recovery images. A physical examination showed that his vision was 0.15 in his left eye and 0.5 in his right eye. He had grade-4 strength in his right limbs and a positive Babinski sign on his right. The suspected diagnosis included parasitic infection, glioma and leukoencephalopathy. To make a correct diagnosis, we performed a large excisional biopsy of the left frontal cyst, including the adjacent brain tissue. Analysis of the cyst fluid did not suggest malignancy or infection (parasitic in particular). The biopsy specimen of his white matter and cyst wall revealed a pronounced reactive gliosis with conspicuous formation of Rosenthal fibers. In addition, focal hemosiderin deposits, which indicate previous hemorrhage, and microcalcifications were observed. Many ectatic vessels and angiomatous changes with cellulose-like degeneration were observed. A neuro-ophthalmologic examination was performed, but no evidence of Coats retinopathy was found. A visual field examination showed irregular visual field defects of both eyes. Visual-evoked potentials disclosed a mild prolongation of P100 latency of his left eye and an increased amplitude of both eyes. The results of serological and immunological tests were within the normal range. We diagnosed our patient with LCC.