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Palisaded neutrophilic and granulomatous dermatitis as a novel cause of hypercalcemia : A case report

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Title: Palisaded neutrophilic and granulomatous dermatitis as a novel cause of hypercalcemia : A case report
Authors: Kono, M. Browse this author
Hasegawa, T. Browse this author →KAKEN DB
Nagai, S. Browse this author
Odani, T. Browse this author
Akikawa, K. Browse this author
Nomura, Y. Browse this author
Sato, H. Browse this author
Kikuchi, K. Browse this author
Amizuka, N. Browse this author →KAKEN DB
Kikuchi, H. Browse this author
Keywords: 1α-hydroxylase
palisaded neutrophilic and granulomatous dermatitis
Issue Date: May-2017
Publisher: Wolters Kluwer
Journal Title: Medicine
Volume: 96
Issue: 21
Start Page: e6968
Publisher DOI: 10.1097/MD.0000000000006968
Abstract: Rationale: Palisaded neutrophilic and granulomatous dermatitis (PNGD) is a benign, inflammatory dermatosis with distinct histopathological features often observed in patients with systemic diseases. There were no reports of PNGD without underlying systemic diseases as an underlying cause of hypercalcemia. Herein, we report a case of a 62-year-old man with hypercalcemia due to PNGD, but with no underlying systemic diseases, including tuberculosis, sarcoidosis, or vasculitis. Patient concerns: Laboratory tests showed an elevated C-reactive protein level, an elevated corrected calcium level, a normal 25-hydroxyvitamin D level, and an elevated 1,25-dihydroxyvitamin D level. There were no other abnormalities to explain the hypercalcemia. Positron emission tomography-computed tomography showed abnormal uptake in his skin. Histopathological examination of the skin showed palisaded granulomatous infiltrate in the dermis. Neutrophils, degenerated collagen, and fibrin were present in the centers of the palisades without prominent mucin. There were no eosinophils, central necrosis, or necrotizing vasculitides. These features were consistent with PNGD. Diagnoses: A diagnosis of PNGD with hypercalcemia was established. Interventions: Oral prednisolone was administered to the patient. Outcomes: After treatment, his symptoms resolved, and his calcium, 1,25-dihydroxyvitamin D and CRP levels returned to normal. Skin specimens before and after treatment were assessed using immunohistochemistry for 1a-hydroxylase. Granuloma and epidermal cells were 1a-hydroxylase-positive before treatment. After treatment, the granuloma diminished in size and the 1ahydroxylase-positive areas of the epidermal cells decreased. Lessons: This case was particularly unique because increased 1a-hydroxylase expression in the granuloma and epidermal cells seemed to result in hypercalcemia due to excessive transformation of 25-hydroxyvitamin D to 1,25-dihydroxyvitamin D. Physicians should consider PNGD as an underlying cause of hypercalcemia.
Type: article
Appears in Collections:歯学院・歯学研究院 (Graduate School of Dental Medicine / Faculty of Dental Medicine) > 雑誌発表論文等 (Peer-reviewed Journal Articles, etc)

Submitter: 長谷川 智香

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