title: Hypercalcemia induced by Rosai-Dorfman disease in a hemodialysis patient: histological evidence of extrarenal calcitriol overproduction.
abstract: Rosai-Dorfman disease (RDD) is a rare disorder characterized by the proliferation of histiocytes in the sinus of the affected lymph nodes, which leads to massive lymphadenopathy. RDD usually presents as an increased inflammatory response and lymph node swelling. We herein report the case of a hemodialysis patient with a fever, hypercalcemia and increased serum calcitriol level who was histologically diagnosed to have RDD. Immunohistochemistry revealed an increased expression of 1α-hydroxylase by histiocytes in the dilated sinus, indicating the extrarenal overproduction of calcitriol. Treatment with oral prednisolone decreased the serum levels of inflammatory markers and calcitriol, normalized the serum calcium level and mitigated the systemic lymph node enlargement.
authors.affiliation: Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan.
authors.forename: Shunsuke
authors.initials: S
authors.lastname: Yamada
authors.forename: Mai
authors.initials: M
authors.lastname: Uemura
authors.forename: Masanori
authors.initials: M
authors.lastname: Tokumoto
authors.forename: Tadashi
authors.initials: T
authors.lastname: Nagara
authors.forename: Hideko
authors.initials: H
authors.lastname: Noguchi
authors.forename: Minako
authors.initials: M
authors.lastname: Hirahashi
authors.forename: Toshiaki
authors.initials: T
authors.lastname: Nakano
authors.forename: Kazuhiko
authors.initials: K
authors.lastname: Tsuruya
authors.forename: Takanari
authors.initials: T
authors.lastname: Kitazono
mesh_terms: D000284:Administration, Oral; D000305:Adrenal Cortex Hormones / Q000008:administration & dosage; D002117:Calcitriol / Q000378:metabolism; D015618:Histiocytosis, Sinus / Q000150:complications* / Q000628:therapy; D006801:Humans; D006934:Hypercalcemia / Q000175:diagnosis* / Q000188:drug therapy* / Q000209:etiology / Q000378:metabolism; D008297:Male; D008875:Middle Aged; D011239:Prednisolone / Q000008:administration & dosage; D006435:Renal Dialysis
chemical_list: D000305:Adrenal Cortex Hormones; D011239:Prednisolone; D002117:Calcitriol