Elsevier

Endocrine Practice

Volume 19, Issue 2, March–April 2013, Pages 50-56
Endocrine Practice

Case Report
Hypercalcemia Associated with Mineral Oil-Induced Sclerosing Paraffinomas

https://doi.org/10.4158/EP12092.CRGet rights and content

ABSTRACT

Objective

Granuloma-forming diseases such as sarcoidosis are associated with extrarenal synthesis of 1,25-dihydroxyvitamin D [1,25(OH)2D]. We describe a case of extensive skin lesions associated with mineral oil injections in which we provide evidence for cutaneous granuloma synthesis of 1,25(OH)2D in the pathogenesis of the patient's hypercalcemia.

Methods

Analysis of expression of the 25(OH)D-1a-hydroxylase (1-a OHase [CYP27b1]) was carried out by immunohistochemical analysis of involved skin.

Results

In involved skin, expression of CYP27b1 was found in the dermis, where it is not normally expressed. Successful management of hypercalcemia was achieved with glucocorticoids.

Conclusions

Hypercalcemia associated with mineral oil induced skin lesions is likely driven by unregulated expression of CYP27b1 by inflammatory monocytes and macrophages infiltrating the dermis. (Endocr Pract. 2013;19:e50-e56)

Section snippets

INTRODUCTION

Hypercalcemia occurs in a variety of chronic granu-lomatous diseases such as sarcoidosis, berylliosis, slack skin granulomatous disease, Crohns disease, and certain lymphomas (1,2). The mechanism for the hypercalcemia in these diseases has been demonstrated to be increased circulating levels of dihydroxyvitamin D [1,25(OH)2D] (3) catalyzed by autonomous activity of 25-hydroxyvitamin D3 1-alpha hydroxylase (CYP27b1) (4). This enzyme is also known to be expressed by a variety of extra-renal

DISCUSSION

Extensive diagnostic evaluation of this patient's symptomatic hypercalcemia was unrevealing for the most common etiologies of hypercalcemia. Symptomatic hyper-calcemia developed 8-10 years after the appearance of mineral oil-associated skin lesions. Sarcoidosis, especially with elevated angiotensin-converting enzyme (ACE) levels is a difficult diagnosis to exclude, but the lack of suggestive history, physical, or radiographic signs make the diagnosis of sarcoidosis unlikely. Furthermore,

METHODS

Informed consent was obtained from the patient for immunostaining of skin tissue from the skin biopsy site. After approval from the Institutional Review Board and Biospecimens Subcommittee of the New York Downtown Hospital, immunohistochemical analysis of Cyp27b1 protein expression in paraffin-embedded tissue sections was carried out. Hormonal tests were performed by routine, commercial assays. For 25-OH-D, liquid chromatogra-phy-tandem mass spectrometry was used as described below: Deuterated

DISCLOSURE

The authors have no multiplicity of interest to disclose. This work was funded by the New York Downtown Hospital and the UCLA Orthopaedic Hospital Research Center.

ACKNOWLEDGMENT

The authors thank Dr. Cynthia Magro (Professor of Pathology and Laboratory Medicine at the Weill Cornell Medical College) for reviewing the skin biopsy.

REFERENCES (25)

  • Fernández-LópezE. et al.

    Subcutaneous fat necrosis of the newborn and idiopathic hypercalcemia

    Dermatologica

    (1990)
  • BalfourE. et al.

    Subcutaneous fat necrosis of the newborn presenting as a large plaque with lobulated cystic areas

    Cutis

    (2002)
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