Transient Polyuria Related to Central Diabetes Insipidus Caused by Lymphocytic Infundibulo-neurohypophysitis in a Patient Treated for Graves' Disease

Abstract

A 45-year-old man was hospitalized because of weight loss, finger tremor, thirst, polydipsia and increased urinary frequency. He was diagnosed with Graves' disease (GD) and central diabetes insipidus (CDI). Magnetic resonance imaging revealed the enlarged posterior pituitary with thickened stalk. Histological examination obtained from biopsy of the pituitary revealed lymphocytic infundibulo-neurohypophysitis. He received treatment with thiamazole (MMI) for GD and desmopressin acetate (DDAVP) for CDI. However, DDAVP administration could be discontinued as GD was gradually improved. This course indicates that not only the recovered renal response to arginine-vasopressin but also the immunomodulative effects of MMI might attribute to the improvement of polyuria.

Manifestation of Central Diabetes Insipidus in a Patient with Thyroid Storm

“…A similar complicated case, but with a transient course of central DI, was reported by Yamazaki et al ( 9 ) showed interesting findings that may prompt reconsideration of our case. A middle-aged man diagnosed with Graves' disease and central DI had received treatment with thiamazole.…”

“…Recent research has also provided evidence that rabphilin-3A is a major autoantigen for LINH and thus might be useful as a biomarker for the diagnosis of central DI ( 8 ). Clinical relationships have been detected between LINH and other diseases, including autoimmune thyroiditis ( 9 ), systemic lupus erythematosus ( 10 ) and Behçet's disease ( 11 ) as well as a drug-induced case ( 12 ). However, the complication of DI secondary to thyroid storm has not been well documented.…”

“…Of interest, DDAVP administration was able to be discontinued due to improvement of his Graves' disease. Yamazaki et al reported that the recovery of the renal response to AVP and the immune-modulative effects of thiamazole, which inhibit prostaglandin-E2 resulting in sustaining the AVP action ( 9 ), might have contributed to the improvement of polyuria ( 9 ). In contrast, our case showed a distinct course demonstrating severe symptoms due to thyroid crisis and persistent central DI that required continuous DDAVP administration, even after the recovery from thyroid crisis.…”

Lymphocitic Infundibuloneurohypophysitis Mimicking a Pituitary Adenoma

“…3 However, according to different autopsy series occurrence of aHYP could also been shown in males. 4 aHYP most commonly presents with symptoms of sellar mass with or without varying degrees of hypopituitarism, whereas DI is uncommon.…”

An unusual cause of polyuria

“…Lymphocytic hypophysitis rarely associated with Graves’ disease may cause central diabetes insipidus which may present with polyuria in such a setting. [4] This was ruled out by a negative water deprivation test in our patient.…”