Two cases of spontaneous improvement of optic pathway lesions in neurofibro
matosis type 1 are reported. At time of diagnosis the children were aged 21
and 32 months respectively; they have been followed by both MRI and clinic
al evaluation for 5 and 4 years. MRI findings of the first 19 months of fol
low-up for Case 1 have been described by us before. On MRI serial evaluatio
n, Case 1 showed an almost complete normalization of the size of lesions an
d a resolution of enhancement, whereas Case 2 showed a slight decrease in t
he size of lesions and a resolution of enhancement. From the clinical point
of view Case 1 showed a normalization of his clinical signs, whereas in Ca
se 2, a visual improvement was only slight, if at all present, In Case 1, t
he clinical improvement seemed to follow the spontaneous regression of the
lesions detected by MRI.