April - June 2022, Volume 18, Issue 2

Borawski B, Kuca P, Zielinski G, Maksymowicz M, Witek P

Pathologically-Confirmed Isolated Hypothalamo-Pituitary Sarcoidosis Refractory to Pulse-Dose Glucocorticoids and Successfully Treated with Methotrexate

Acta Endo (Buc) 2022, 18 (2): 244-249
doi: 10.4183/aeb.2022.244

Isolated sarcoidosis of the hypothalamic-pituitary system is a very rare form of neurosarcoidosis. A high index of suspicion is required for diagnosis and the choice of therapy embodies another challenge due to lack of standardized protocols. Glucocorticoids are the mainstay of initial treatment, whereas the second and third-line therapy include immunomodulators and cytotoxic drugs, in addition to monoclonal antibodies. This report presents an unusual case of panhypopituitarism in a 32-year-old previously healthy male patient due to isolated hypothalamo-pituitary sarcoidosis confirmed histologically, refractory to pulsedose glucocorticoids and then successfully treated by methotrexate. Based on our report, in patients requiring additional therapy usage of the methotrexate as the second line agent should be considered, however the time frame and the dosing schedule of methotrexate are still unknown and deserve further investigation.

Keywords: Sarcoidosis, neurosarcoidosis, hypothalamus, pituitary, methotrexate, isolated.

Correspondence: Bartlomiej Borawski MD, Brodnowski Hospital of the Mazovian Region in Warsaw, Kondratowicza 8, Warszawa, 03-242, Poland, E-mail: bborawski94@gmail.com