Introduction and importancePituicytomas are extremely rare cancers of the sellar and suprasellar region that appear from the infundibulum or posterior pituitary. World Health Organization in 2007, described pituicytoma as a low-grade tumour (Grade I) in the taxonomy of CNS cancers. The tumour can frequently simulate a pituitary adenoma and is also linked with hormonal disorders. Distinguishing a pituitary adenoma from a pituicytoma can be challenging. We present a rare case report where an elderly female showed high levels of prolactin mainly due to mass effects along with diagnostic, imaging, and immunohistochemical characteristics of pituicytoma.Case presentationA 50-year-old female known case of hypothyroidism, complained of headache associated with dizziness and blurry vision. Her prolactin levels were high which led to the suspicion of pituitary involvement and underwent MRI. The imaging study revealed a well-defined, completely suprasellar, homogenously enhancing mass lesion arising from the left lateral aspect of the pituitary infundibulum. The initial differential diagnosis from the imaging included an ectopic pituitary gland, adenoma, pituicytoma, or hypothalamic glioma. She underwent a right supra-orbital craniotomy for debulking of the pituitary stalk lesion. The histopathological diagnosis was pituicytoma, WHO grade I.Clinical discussionThe clinical manifestations are mostly depended upon the tumour mass and position. They typically present due to mass effects leading to hormonal disorders. The imaging studies are the backbone of the clinical diagnosis along with the histopathological findings. Surgical resection is the preferred treatment for pituicytoma, with an exceptionally low recurrence rate (4.3 %) following complete removal.ConclusionPituicytomas are slow-growing, benign glial growths. It is challenging to diagnose before surgery as its clinical manifestations and imaging findings look like those of non-functional pituitary adenomas. The effective treatment for pituicytoma is gross total resection by the endoscopic method or transcranial technique.