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We are presenting the case of a 50 year-old patient whose symptoms started over an year ago with hiposmia and epistaxis. Multiple ENT surgeries were performed for a frontal maxillary sinusitis. The patient also developed polydipsia, reaching up to 15 liters of liquid intake per day and, soon after, inflammatory and migratory arthralgia, Raynaud phenomenon, conjunctivitis and eyelid edema. The biological and imagistic investigations showed intense inflammatory syndrome, c-ANCA antibodies, nodular cavitary lesions in both lungs, pericarditis, pituitary micro adenoma. Infectious and neoplastic causes were excluded. The final diagnosis was granulomatosis with polyangiitis with upper and lower airway, cardiac, skeletal, and endocrinological involvement. Remission of induction therapy with cyclophosphamide and pulse glucocorticoids was started with a good clinical and biological outcome and a gradual decrease of fluid intake. The presence of pituitary granuloma leading to central diabetes insipidus is a rare clinical manifestation of granulomatosis with polyangiitis, and it is reversible if the treatment is prompt, before pituitary necrosis. [ABSTRACT FROM AUTHOR]