Orbit, 2014; 33(4): 295–297 ! Informa Healthcare USA, Inc. ISSN: 0167-6830 print / 1744-5108 online DOI: 10.3109/01676830.2014.902477

C ASE REPORT

Bilateral Symmetrical Subepidermal Calcified Nodules of the Eyelids Mehmet Talay Koylu1, Yusuf Uysal2, Murat Kucukevcilioglu3, Osman Melih Ceylan4, and Mehmet Salih Deveci5 1

Tatvan Military Hospital Ophthalmology Clinic, Bitlis, Turkey, 2Gulhane Military Medical School, Associate Professor of Ophthalmology, Ankara, Turkey, 3University of Iowa, Department of Ophthalmology and Visual Sciences, Iowa City, Iowa, USA, 4Ardahan Military Hospital Ophthalmology Clinic, Ardahan, Turkey, and 5 Gulhane Military Medical School, Professor of Pathology, Ankara, Turkey

ABSTRACT A subepidermal calcified nodule is an uncommon variant of calcinosis cutis and only a limited number of cases have been reported about the eyelid nodules in the literature. A 20-year-old male was referred to our department with symmetrical nodules on both upper eyelids enlarging over 3 years. Both nodules were removed by excisional biopsy. After the histopathologic evaluation, the diagnosis was subepidermal calcified nodule. He had a complete recovery with no recurrence and acceptable aesthetic appearance. As a rare entity, subepidermal calcified nodule should be thought in differential diagnosis of eyelid nodular lesions and symmetrical appearance may be seen. Keywords: Calcinosis cutis, eyelid, idiopathic, subepidermal calcified nodule, symmetrical

CASE REPORT

Calcinosis cutis is a group of disorders in which insoluble compounds of calcium are deposited within the skin.1 The disease is classified according to aetiology: dystrophic, iatrogenic, metastatic, and idiopathic.2 Dystrophic calcification results from an underlying disease, a preexisting dermal nodule or trauma.3 Iatrogenic calcinosis cutis is due to recent hospitalization with calcium-containing medical devices or medications.2 Idiopathic calcinosis cutis is not associated with previous trauma or disease in otherwise healthy individuals.3 Subepidermal calcified nodule (SCN) is a rare variant of idiopathic calcinosis cutis that usually develops in the first two decades of life as a solitary white or yellow nodule on the face of children.3,5 We present a rare case of subepidermal calcified nodules of the eyelids which have symmetrical appearance.

A 20-year-old male was referred to our department with bilateral symmetrical upper eyelid lesions. He noted painless increase in size of lesions over three years and there was no history of trauma or evidence of any systemic disease (Figure 1A). Both nodules were white and yellowish, hard, freely mobile which were measured 10  7 mm in diameter on the right and 9x6 mm on the left side with hyperkeratinization on the surface (Figure 1B and C). Patient’s systemic evaluation including calcium, phosphate and parathyroid hormone were unremarkable. Our clinical preliminary diagnoses were keratoacanthoma, squamous papilloma and epidermal cyst, respectively. Both nodules were removed by excisional biopsy under local anesthesia. On histopathologic evaluation with hematoxylin-eosin-stained

Received 24 November 2013; Revised 13 February 2014; Accepted 3 March 2014; Published online 15 May 2014 Correspondence: Dr. Mehmet Talay Koylu, Tatvan Military Hospital, Tatvan, Bitlis, Turkey, 13200. Tel: +905303273844. Fax: +904348276916. E-mail: [email protected]

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296 M. T. Koylu et al. sections, the lesions were lined by hyperplastic and hyperkeratotic epidermis (Figure 2A), and were composed of many large and small rounded bluish calcified concretions localized within the dermis (Figure 2B). The diagnosis was SCN for both of the lesions. He had a complete recovery with no recurrence and acceptable aesthetic appearance.

COMMENT SCN of the eyelid is an unusual diagnosis. The clinical appearance is usually single, solitary, painless, whiteyellowish, raised nodule with papillomatous features.1,3,5 SCN were not suspected in any of the cases before the pathologic specimen confirmed the diagnosis in the literature.1,3,5 In differential diagnosis of SCN; pilomatrixoma, warty papilloma, cysts, keratoakanthoma, juvenile xanthogranuloma,

FIGURE 1. (A) A clinical photograph showing bilateral symmetrical subepidermal calcific nodules on upper eyelids. (B–C) Both upper eyelid nodules were white-yellowish, hard, freely mobile with hyperkeratinization on the surface.

cutaneous horn, molluscum contagiosum, xanthelasma, milia and tumour should be thought.1,3 Pilomatrixoma would present as a solitary, painless, irregularly shaped subcutaneous mass beneath red-blue discoloration of the overlying skin frequently in children6. Papillomas would present with multiple lesions1. Cysts would have keratinizing wall1. The pathogenesis of SCN is not clear.1,3 It is not associated with metastatic process, previous skin disease or other medical problems.3 Serum levels of calcium and phosphate are normal.1 The presence of bilateral and symmetric cases suggests that systemic factors may play role rather than local factors. Our case had the typical features described in the literature with the addition of symmetrical appearance, and the size. Only a few bilateral cases were reported in the literature, but symmetry of lesions were not as prominent as our case.3,7–9 The nodules of our case were measured 10  7 mm in diameter on the right and 9  6 mm on the left side, which were larger than those in the current literature (mean diameter, 3 mm; range, 1–5 mm).1,3,5 The histopathologic features of SCN include homogenous basophilic subepithelial calcific deposits beneath the acanthotic, papillomatous epidermis in the upper dermis on haematoxylin–eosin-stained preparations.1,5 We observed the same histopathologic findings and uncommon surface hyperkeratinization as well. History of increase in size of lesions over 3 years, hyperkeratinization on the surface may show the process of self-resolution. Complete surgical excision is curative with no recurrence.3,5 Both nodules were removed by excisional biopsy and complete recovery with no recurrence and an acceptable aesthetic appearance was achieved in our case. In conclusion, SCN of the eyelid should be thought in differential diagnosis of eyelid nodular lesions and symmetrical appearance may be seen.

FIGURE 2. (A) Full-mount histopathologic appearance of the lesion lined by hyperplastic (short arrow) and hyperkeratotic (long arrow) epidermis. (B) Large (long arrow) and many small (short arrow) rounded bluish calcified concretions within the dermis [Haematoxylin and eosin stain, 40 (A), 200 (B)]. Orbit

Subepidermal Calcified Nodules of the Eyelids

ACKNOWLEDGMENTS This case was presented as a poster presentation at the 16th Afro Asian Congress of Ophthalmology held in Istanbul between 13–16 June 2012.

DECLARATION OF INTEREST The authors have no financial support or conflicts of interest about this case. The authors alone are responsible for the content and writing of the paper.

REFERENCES 1. Nguyen J, Jakobiec FA, Hanna E, Fay A. Subepidermal calcified nodule of the eyelid. Ophthal Plast Reconstr Surg 2008;24:494–495.

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2. Jun I, Kim SE, Lee SY, et al. Calcinosis cutis at the tarsus of the upper eyelid. Korean J Ophthalmol 2011;25: 440–442. 3. Nico MMS, Bergonse FN. Subepidermal calcified nodule: report of two cases and review of the literature. Pediatr Dermatol 2001;18:227–229. 4. Moutopoulos CH, Cooper J, Paek A, Chien J. Multiple calcified deposits along the eyelid margins secondary to chronic renal failure and hyperparathyroidism. Optometry 2005;76:181–184. 5. Kim HJ, Johnson PB, Kropinak M, et al. Subepidermal calcified nodules of the eyelid. Ophthal Plast Reconstr Surg 2009;25:489–490. 6. Levy J, Ilsar M, Deckel Y, et al. Eyelid pilomatrixoma: a description of 16 cases and a review of the literature. Surv Ophthalmol 2008;53:526–535. 7. Carvounis PE, Santi M, Stern JB, Miller M. Subepidermal calcified nodules. Ophthalmologica 2005;219:112–114. 8. Salem AM, Yasser HA, Ammar CA. Bilateral subepidermal calcified nodules of the eyelid. Saudi J Ophthalmol 2011; 25(1):81–83. 9. Zeglaoui F, Khaled A, Fazaa B, et al. Multiple subepidermal calcified nodules on the eyelids with eruptive syringomas: A possible ethiopathogenic relationship. J Eur Acad Dermatol Venereol 2009;23(3):337–339.

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Bilateral symmetrical subepidermal calcified nodules of the eyelids.

A subepidermal calcified nodule is an uncommon variant of calcinosis cutis and only a limited number of cases have been reported about the eyelid nodu...
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