Langerhans cell histiocytosis of the central nervous system (CNS) usually involves the hypothalamic-pituitary axis, and T1-weighted MR images normally demonstrate infundibular thickening and/or a mass lesion in the hypothalamus and the absence of a posterior pituitary "bright spot". We recently encountered a case of CNS Langerhans cell histiocytosis with no posterior pituitary "bright spot" and with lesions involving the cerebellum and basal ganglia but not the hypothalamic-pituitary axis.