We report the case of a patient with bilateral blephaloptosis associated with a recurrence of diffuse large B-cell lymphoma (DLBCL) in the midbrain. A 70-year-old man experienced acute onset bilateral blephaloptosis; the other ocular movements, except for medial rectus muscle in the right eye, were not impaired. Pupils were isocoric and light reflexes were prompt. Other cranial nerves were intact. Gadolinium-enhanced MRI revealed abnormal enhancement in the midbrain and peri-ventricular regions. FDG-PET revealed an abnormal positive signal in the midbrain, similar to the distribution seen in the MRI scan. Cytology of the cerebrospinal fluid showed large atypical lymphocytes. These findings suggest that the recurrence of DLBCL in the midbrain caused bilateral blephaloptosis. The oculomotor fascicle is localized in the paramedian ventral midbrain. The fascicular fibers are divided topographically into four regions; the lateral, medial, rostral and caudal regions. In three-dimensional arrangement of the oculomotor fascicle, the fibers to the levator palpebrae superioris muscle and medial rectus muscles are located adjacently in caudal regions. Thus, we speculate that recurrence of DLBCL in the midbrain involving the right oculomotor fascicle caused blephaloptosis in the right eye, and then, infiltration of DLBCL to the left oculomotor fascicle subsequently caused blephaloptosis in the left eye. This is a valuable case to be documented in which neurological site of lesions consistent with those are found in radiological study.
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