A 36-year-old non-pregnant woman presented with a four-month history of pro
gressive visual deterioration and amenorrhea. The latest gestation was 6 ye
ars earlier. Hormonal study revealed central diabetes insipidus, hypopituit
arism, and slightly increased prolactin level. Ophthalmologic examination s
howed bilateral hemianopsia. In the magnetic resonance imaging an intrasell
ar mass with supra and retrosellar extension was found. The mass showed a p
olylobular aspect with heterogeneous signal within the tissue. The normal n
eurohypophysis could not be identified. Pterional craniotomy was performed.
The pathological examinations revealed fibrous tissue with heavy inflammat
ory infiltrate composed of lymphocytes and plasma cells, islands of eosinop
hilic epithelial cells stained positively for chromogranin, GH, ACTH, and P
RL and negatively for antibodies directed against HLA-II antigens. This cas
e of lymphocytic hypophysitis was not related to pregnancy and involved the
neurohypophysis. We discuss the features that can help to make a preoperat
ive differential diagnosis.