Hypercalcemia Induced by Rosai-Dorfman Disease in a Hemodialysis Patient: Histological Evidence of Extrarenal Calcitriol Overproduction

Shunsuke Yamada; Mai Uemura; Masanori Tokumoto; Tadashi Nagara; Hideko Noguchi; Minako Hirahashi; Toshiaki Nakano; Kazuhiko Tsuruya; Takanari Kitazono

doi:10.2169/internalmedicine.53.3001

CASE REPORTS

Hypercalcemia Induced by Rosai-Dorfman Disease in a Hemodialysis Patient: Histological Evidence of Extrarenal Calcitriol Overproduction

Shunsuke Yamada, Mai Uemura, Masanori Tokumoto, Tadashi Nagara, Hideko Noguchi, Minako Hirahashi, Toshiaki Nakano, Kazuhiko Tsuruya, Takanari Kitazono

Author information

Shunsuke Yamada
Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan
Department of Internal Medicine, Fukuoka Dental College, Japan
Mai Uemura
Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan
Masanori Tokumoto
Department of Internal Medicine, Fukuoka Dental College, Japan
Tadashi Nagara
Division of Nephrology, Japanese Red Cross Fukuoka Hospital, Japan
Hideko Noguchi
Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan
Minako Hirahashi
Department of Anatomic Pathology, Pathological Sciences, Graduate School of Medical Sciences, Kyushu University, Japan
Toshiaki Nakano
Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan
Kazuhiko Tsuruya
Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan
Department of Integrated Therapy for Chronic Kidney Disease, Graduate School of Medical Sciences, Kyushu University, Japan
Takanari Kitazono
Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Japan

Keywords: histiocytes, 25-hydroxyvaitamin D-1α-hydroxylase, hypercalcemia, lymphadenopathy, Rosai-Dorfman disease

JOURNAL OPEN ACCESS

2014 Volume 53 Issue 24 Pages 2783-2787

DOI https://doi.org/10.2169/internalmedicine.53.3001

Details

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.

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