Nazeer, Kabir KhanAlatrash, SalhinElsheikh, SawsanJacob, Saiju2025-07-182025-07-182025-07-14Nazeer KK, Alatrash S, Elsheikh S, Jacob S. Xanthomatous hypophysitis relapsing and remitting over two decades. Pract Neurol. 2025 Jul 14;25(4):359-3621474-776610.1136/pn-2024-00443039884847pn-2024-00443039884847https://westmid.openrepository.com/handle/20.500.14200/8029Inflammation of the pituitary gland can be primary (without another underlying cause) or secondary (associated with a systemic inflammatory condition). Primary hypophysitis is very rare, among which xanthomatous hypophysitis as a histological type is extremely unusual. A woman in her late 50s presented with recurrent pituitary lesions over 20 years. Her general practitioner had diagnosed panhypopituitarism in her 30s; a decade later, she had presented to ophthalmology with visual loss and restricted visual fields, and a pituitary lesion was found. This recurred several times requiring multiple resections. Histopathology showed atypical inflammation in keeping with xanthomatous hypophysitis; this responded well to corticosteroid therapy. Xanthomatous hypophysitis is a rare form of steroid-responsive primary pituitary inflammation, to consider in the differential diagnosis of recurring pituitary lesions.en© Author(s) (or their employer(s)) 2025. No commercial re-use. See rights and permissions. Published by BMJ Group.EndocrinologyNeurologyOphthalmologyJournal Article