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(a)

(b)

(c)

(d)

Figure 1 Clinical and histopathological features. (a) Red nodules of varying sizes aggregated on the extensor surface of the left thumb. (b,c) Dense cellular infiltrates comprising mature plasma cells throughout the entire dermis. Hematoxylin and eosin, original magnification: (b) 940, (c) 9400. (d) Infiltrative cells were positive for CD138 (diaminobenzidine, original magnification, 9400) Table 1 Clinical profiles of reported cases

Case

Sex/age

Locations

Disease duration (years)

1 2 3 4 5

F/7 F/15 M/7 F/7 F/16

Axillary and scapular regions Left anterior tibia Right anterior tibia Right buttock Left hand

4 12 2 5 1

Funding sources: None. Conflicts of interest: None. References

F, female; M, male.

verify the possibility of cutaneous plasma cells contributing to autoantibody production. Yohei Shiba, MD Takahiro Satoh, MD, PhD Department of Dermatology National Defense Medical College Tokorozawa Japan Yohei Shiba, MD Department of Dermatology National Defense Medical College, 3-2 Namiki Tokorozawa 359-8513 Japan E-mail: [email protected]

International Journal of Dermatology 2014, 53, e389–e409

1 Leonard AL, Meehan SA, Ramsey D, et al. Cutaneous and systemic plasmacytosis. J Am Acad Dermatol 2007; 56: S38–S40. 2 Arico M, Bongiorno MR. Primary cutaneous plasmacytosis in a child. Is this a new entity? J Eur Acad Dermatol Venereol 2002; 16: 164–167. 3 Gilliam AC, Mullen RH, Oviedo G, et al. Isolated benign primary cutaneous plasmacytosis in children: two illustrative cases. Arch Dermatol 2009; 145: 299–302. 4 Ahn JJ, Yang YS, Shin MK, et al. Case of isolated benign primary cutaneous plasmacytosis in a child. J Dermatol 2011; 38: 364–367.

An unusual case of nevus of Ota combined with nevus spilus

Editor, Nevus of Ota is a dermal melanocytic disorder of unknown cause. Nevus spilus presents as a light tan patch with darkly pigmented, nevomelanocytic macular and/or papular elements. We describe a patient in whom both types of lesion overlapped in the same area.

ª 2014 The International Society of Dermatology

Correspondence

A 7-year-old Korean girl presented with a compounded pigmented lesion on her face. On examination, a speckled, brownish, pigmented patch was found on the inner and lower areas of the right periocular region, and a bluish patch was found on the upper and lower eyelids, divided by the right eye and partly overlapping with the speckled brownish patch (Fig. 1). The bluish patch had been present since birth, but the speckled brownish patch had not become discernible until one year prior to the patient’s presentation at our clinic. No other pigmentary

Figure 1 Clinical examination in a 7-year-old girl shows a bluish pigmented patch on both the upper and lower eyelids, overlapping a brownish patch with dark brown speckles on the right side of the malar and nasal root area

(a)

changes or physical abnormalities were noted. The patient had no family history of pigment disorders, and her medical history was unremarkable. A specimen from the overlapping bluish–brown area revealed findings of nevus of Ota in the dermis and nevus spilus in the epidermis and upper dermis. There were slight elongations of the rete ridges and increased melanin pigments in the epidermal basal layer, along with junctional nests of nevus cells at some of the rete ridges and adjacent upper dermis. In the dermis, spindle-shaped melanocytes filled with brown melanin granules were scattered among the collagen fibers and loosely aggregated around blood vessels (Fig. 2). The pathogeneses of nevus of Ota and nevus spilus are not well understood. The exact etiologies are unknown but are postulated to include a defect in neural crest melanoblasts, as well as possible roles of genetic and environmental factors.1 The hypothesis that ectopic dermal melanocytes have failed to reach their proper location has been commonly accepted.2 Dermal melanocytoses or dermal dendritic melanocytic proliferations are characterized by the presence of ectopic melanin-producing dendritic melanocytes in the dermis. They include nevi of Ito, nevi of Ota, Mongolian spots, and dermal melanocyte hamartomas. They share similar histopathologic characteristics, which include elongated dermal melanocytes that are scattered between collagen fibers in the dermis. They differ in the concentration and location of the melanocytes. There are many epidermal melanocytic lesions, such as those of ephelides, nevus spilus, Becker’s nevus, and lentigo and cafe au lait macules.3 They all share similar histopathologic features of epidermal pigmentation. Classically, the background hyperpigmentation of nevus spilus is described histologically as having the features of a lentigo or cafe au lait macule.4

(b)

Figure 2 Histopathology of the biopsy specimen reveals (a) heavy pigmentation of the basal layer of the epidermis and scattered fusiform melanocytes in the dermis, and (b) diffuse lentiginous epidermal pigmentation with junctional nests of nevus cells at some of the rete ridges. (Hematoxylin and eosin stain; original magnification [a] 9100, [b] 9200) ª 2014 The International Society of Dermatology

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There has been conflict over whether nevus spilus is a congenital or an acquired lesion and over its nomenclature as nevus spilus or speckled lentiginous nevus.5 Part of this controversy is related to the natural history of these lesions. They are often present at birth as lightly hyperpigmented patches that look similar to cafe au lait macules and may take several years to reach their more readily recognizable spotted and speckled form.5 In this sense, the term congenital can thus encompass lesions that are programmed from birth, as well as those that are overtly apparent at birth.5 The present case is notable for several points. Firstly, it represents a rare manifestation of disease in which unrelated lesions of different pathogeneses involved the same area of skin and were partly overlapping. Secondly, the timing of the appearance of the recently developed epidermal lesions accorded with current thinking on the nature of nevus spilus. Lastly, as the nevus of Ota lesion in this patient was divided by her right eye, we can infer details of its time of appearance in the facial area; this is most likely to have occurred after eyelid fusion during gestation weeks 8–9 but before the reopening of the eyelid in gestation month 6.6 Jiehyun Jeon, MD, PhD Yoo Sang Baek, MD Chil Hwan Oh, MD, PhD Hae Jun Song, MD, PhD Department of Dermatology College of Medicine Korea University Seoul South Korea E-mail: [email protected]

References 1 Vaidya DC, Schwartz RA, Janniger CK. Nevus spilus. Cutis 2007; 80: 465–478. 2 Nordlund JJ, Ortonne JP. Mechanisms that cause abnormal skin color. In: Nordlund JJ, Boissey RE, Hearing VJ, King R, Oetting W, Ortonne J-P eds. The Pigmentary System: Physiology and Pathophysiology. New York, NY: Oxford University Press, 1998: 489–502. 3 Kim S, Park JH, Kim JA, et al. Congenital combined dermal and epidermal melanocytosis: a new entity? J Eur Acad Dermatol Venereol 2007; 21: 1282–1283. 4 Stewart DM, Altman J, Mehregan AH. Speckled lentiginous nevus. Arch Dermatol 1978; 114: 895– 896. 5 Schaffer JV, Orlow SJ, Lazova R, et al. Speckled lentiginous nevus: within the spectrum of congenital melanocytic nevi. Arch Dermatol 2001; 137: 172– 178. International Journal of Dermatology 2014, 53, e389–e409

6 Hamming N. Anatomy and embryology of the eyelids: a review with special reference to the development of divided nevi. Pediatr Dermatol 1983; 1: 51–58.

The blue nails of argyria

Argyria is an adverse side effect of silver, characterized by bluish-gray discoloration of the nail, skin, conjunctiva, and internal organs. It is considered a benign condition. Here we report a case of argyria in a 68year-old man who demonstrated bluish discoloration of the nails typical of argyria (Fig. 1). The patient reported here has been using anti-tumor necrosis factor (TNF) agents for ankylosing spondylitis for the last 10 years. As a preventative measure against increased risks of infection from the anti-TNF agents, he has been self-medicating with Silver Liquid 400 PPM for the last five years. As per website information (www. vrp.com), this product contains mild silver protein. Combining silver with protein is supposed to enhance the mineral’s potency. The use of silver as an antibiotic is not new.1 Silver is credited with an ability to provide broad-spectrum immune support. In the early 20th century, with the production of new and synthetic alternatives, the use of silver in healthcare has waned. Because of the popularity of alternative medicine in the USA, there has been a resurgence in the use of silver products among patients who are using immunosuppressives, including patients who are on biologics.2 However, silver should not be considered harmless; rarely it can cause systemic side effects that include retinal toxicity, hepatitis, nephritis, and neurotoxicity.3–5 As physicians, it is critical for us to educate our patients that over-the-counter (OTC) medicines are not without side effects, and it is also important for patients to discuss with their physicians before consuming OTC medicines.

Figure 1 Bluish-gray discoloration of all of a patient’s nails

following systemic use of colloidal silver ª 2014 The International Society of Dermatology

An unusual case of nevus of Ota combined with nevus spilus.

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