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Palisaded neutrophilic and granulomatous dermatitis as a novel cause of
hypercalcemia: A case report
#MMPMID28538395
Kono M
; Hasegawa T
; Nagai S
; Odani T
; Akikawa K
; Nomura Y
; Sato H
; Kikuchi K
; Amizuka N
; Kikuchi H
Medicine (Baltimore)
2017[May]; 96
(21
): e6968
PMID28538395
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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.
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