Eur J Dermatol 2014; 24(2): 248-77

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A rare case of pigmented epithelioid melanocytoma on the penis as a divided nevus

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Figure 1. The two black lesions on the penis (A). Dermoscopic views of the lesions on the prepuce (B) and the glans (C). The former lesion was more irregular in shape than the latter. Histopathological staining of the lesion on the prepuce: Hematoxylin-eosin staining yielded no remarkable findings in the epidermis (D). Heavily melanin-containing epithelioid cells had infiltrated beneath uninvolved papillary dermis and some of these cells were localized in part of the epidermal rete ridge (E). These cells reacted with S100-specific antibodies (F) and, at the peripheral lesion, melanophages reacted with CD68specific antibodies (G). (F) and (G): Stained with biotinylated second antibodies and alkaline phosphatase-labeled streptavidine after bleaching. EJD, vol. 24, n◦ 2, March-April 2014

To cite this article: Uehara M, Sato S, Kato A, Shimizu F, Matsuda K, Ooatari M, Yokoyama S, Hatano Y, Fujiwara S. A rare case of pigmented epithelioid melanocytoma on the penis as a divided nevus. Eur J Dermatol 2014; 24(2): 248-77 doi:10.1684/ejd.2014.2272

doi:10.1684/ejd.2014.2272

Sixteen cases of “divided” or “kissing” nevus on the genital mucosa have been reported [1-7], including one case of divided melanoma on the penis [3]. Pigmented epithelioid melanocytoma is a borderline melanocytic tumor encompassing epithelioid blue nevus, with or without Carney complex, and most tumors previously considered so-called “animal-type melanomas” [8]. Although rare pigmented epithelioid melanocytoma or epithelioid blue nevus on the penis has been reported [9], we found no reports of divided nevus with epithelioid blue nevus or with pigmented epithelioid melanocytoma. Here, we report the first case of pigmented epithelioid melanocytoma on the penis as a divided nevus, with dermoscopic findings, and we review the literature on divided nevus on the penis. A 9-year-old boy presented at our hospital with two pigmented lesions on his penis. He was born after an uneventful pregnancy. His family, including a younger brother, was healthy, with no pigmented lesions. Such pigmented lesions had not been recognized on the boy before the age of eight. Our patient had a 5 × 5 mm black plaque on his prepuce and a 6 × 6 mm black plaque on his glans (figure 1A). Dermoscopically, both lesions had homogeneous blue pigmentation, which suggested blue nevus, but the lesion on the prepuce had a more irregular profile (figures 1B,C). We decided to perform an excisional biopsy, above the layer of subcutaneous tissue, of the black lesion on the prepuce only, with a 2-mm measurable margin, to rule out malignancy because of the lesion’s irregular shape and unknown growth rate. We did not remove the lesion on the glans, fearing that we might disturb the line of urination. Histopathologically, heavily melanin-containing epithelioid cells had infiltrated beneath uninvolved papillary dermis (figure 1D) and almost all of these cells reacted with S100-specific (figure 1F) and HMB45-specific (data not shown) antibodies. Some of similar cells were localized in part of the rete ridge of the epidermis (figure 1E). A few spindle-shaped melanin-containing cells were observed. At the peripheral lesion, we found melanophages which reacted with CD68specific antibodies (figure 1G). Mitotic figures were absent. We diagnosed the lesion as divided pigmented epithelioid melanocytoma of the penis. Five years after surgery, there has been no local recurrence. The residual plaque on the glans expanded to 8 × 9 mm but enlargement appeared to be proportional to the patient’s growth and the shape was essentially

unchanged. Dermoscopy revealed that the central heavy black pigmentation, covered with a whitish-blue veil, had an indistinct border surrounded by light brown pigmentation. These are common features of blue nevus. The former is an indicator of heavily melanin-containing cells in the superficial dermis. Among sixteen cases of divided nevus of the penis [1-7], only one case of malignant melanoma after a diagnosis of divided nevus has been reported [3]. In that case, both congenital melanocytic nevi, on the prepuce and on the glans, had changed rapidly in size and color within a year and surgical excision with a 0.5 to 1.0-cm margin was performed. However long-term follow-up and prognosis were

not described. In our case, no rapid changes in size and color were observed in the remaining lesion on the glans over five subsequent years and, histologically, no malignant signs were found in the excised lesion. Therefore, we did not excise the lesion on the glans. Zembowicz et al. reported that sentinel lymph node metastasis may be found in up to 46% of cases of pigmented epithelioid melanocytoma, but favorable outcomes after 5year follow-up were described [8, 10]. The lesion on our patient’s glans is a pigmented epithelioid melanocytoma, namely, a borderline melanocytic tumor. Thus follow-up of this lesion without total excision is unprecedented and challenging, and considerable vigilance is required. Changes in dermoscopic findings should allow early detection of malignant change. We believe that the lesion was present at birth, according to embryogenetic hypothesis on divided nevus [2], but only became pigmented at the age of 8. In conclusion, pigmented epithelioid melanocytoma is a borderline melanocytic tumor with a low risk of local recurrence. Some cases without Carney complex have been reported [8], but the lesion is very rare in genital regions. Our unusual case involved a divided nevus on the penis. Diligent follow-up is necessary, not only of the lesion on the glans, but also the prepuce from which the tumor was removed.  Disclosure. Financial support: none. Conflict of interest: none. 1

Department of Plastic Surgery, Department of Diagnostic Pathology, 3 Department of Dermatology, Faculty of Medicine, Oita University, Hasama, Yufu, 879-5593, Japan 2

Miyuki UEHARA1 Seiichi SATO1 Aiko KATO1 Fumiaki SHIMIZU1 Kaho MATSUDA1 Miwako OOATARI1 Shigeo YOKOYAMA2 Yutaka HATANO3 Sakuhei FUJIWARA3

1. Desruelles F, Lacour JP, Mantoux F, Ortonne JP. Divided naevus of the penis: an unusual location. Arch Dermatol 1998; 134: 879-80. 2. Kono T, Nozaki M, Kikuchi Y, et al. Divided naevus of the penis: a hypothesis on the embryological mechanism of its development. Acta Derm Venereol 2003; 83: 155-6. 3. Egberts F, Egberts J-H, Schwarz, Hauschild A. Kissing melanoma or kissing nevus of the penis? Urology 2007; 69: 384.e5-7. 4. Higashida Y, Nagano T, Oka M, Nishigori C. Divided naevus of the penis. Acta Derm Venereol 2010; 90: 319. 5. Palmer B, Hemphill M, Wootton C, et al. Kissing nevus discovered at circumcision consult. J Pediatr Urol 2010; 6: 318-9. 6. Zhou C, Xu H, Zang D, Du J, Zhang J. Divided nevus of the penis. Eur J Dermatol 2010; 20: 527-8. 7. Yun SJ, Wi HS, Lee J-B, et al. Kissing nevus of the penis. Ann Dermatol 2011; 23: 512-4. 8. Zembowicz A, Carney JA, Mihm MC. Pigmented epithelioid melanocytoma. Am J Surg Pathol 2004; 28: 31-40. 9. Izquierdo MJ, Pastor MA, Carrasco L, et al. Epithelioid blue naevus of the genital mucosa: report of four cases. Br J Dermatol 2001; 145: 496-501. 10. Mandal RV, Murali R, Lundquist KF, et al. Pigmented epithelioid melanocytoma: favorable outcome after 5-year follow-up. Am J Surg Pathol. 2009; 33: 1778-82. doi:10.1684/ejd.2014.2272

EJD, vol. 24, n◦ 2, March-April 2014

Primary cutaneous follicle center lymphoma, diffuse type, with atypical clinical features A 33 year old male presented with an ulcerated tumor of 15 cm diameter with raised borders on the antecubital space of the right arm (figure 1A). The lesion evolved over a 2month period and clinically resembled an expanding lesion of pyoderma gangrenosum. Hematoxylin and eosin stained sections showed an ulcerated epidermis and diffuse infiltration of cutis and subcutis with medium-sized to large neoplastic lymphoid cells (figure 1C). Most cells had cleaved or irregular nuclei with dispersed chromatin or indistinct nucleoli, while the rest presented large round nuclei with prominent nucleoli (figure 1D). Necroses were absent. Immunohistochemical study revealed the following immunophenotype: CD45+, CD20+, CD79a+, PAX5+, CD10+, BCL6+, CD30++/-, MUM1–/+, CD45RA–/+, CD45RO-, CD3-, CD5-, CD23-, CCND1-, BCL2-, HLA-DR–/+, ␬-, ␭- (figures 1E-H). Mild positivity to p53 was observed in 40% of cells. Ki67/MIB1 proliferative index was 90%. EBER was negative. CD23 and CD21 immunostains did not reveal follicular dendritic cells. Histologic findings set the diagnosis as primary cutaneous follicle center lymphoma diffuse type (PCFCLDT). Initial staging with CT scans revealed enlarged right axillary lymph nodes. Lymph node and bone marrow trephine biopsies were negative for infiltration. The patient received six cycles of rituximab-CHOP/21 regimen, followed by involved–field irradiation. After the 2nd cycle of therapy the tumor completely resolved, leaving an ulcerated area which gradually healed (figure 1B). He was in complete remission at the end of chemotherapy and received consolidation radiotherapy. Primary cutaneous B-cell lymphomas with a large cell morphology and diffuse pattern of growth are an ongoing issue. In the WHO-EORTC classification for cutaneous lymphomas, these lymphomas are divided into 3 groups: primary cutaneous follicle center lymphoma (PCFCL), primary cutaneous large B-cell lymphoma, leg type (PCLBCL-LT) and primary cutaneous large B-cell lymphoma, other (PCLBCL-other) for diffuse large B-cell

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Figure 1. Ulcerated tumor with raised borders, which evolved during a 2-month period (A) and its remission at the end of chemotherapy (B). Skin biopsy: Diffuse infiltration of cutis and subcutis with medium-sized to large neoplastic lymphoid cells (C, D) positive to CD20 (E), PAX5 (F), CD10 (G) and BCL6 (H) immunostains (C: H&E ×200, D: H&E ×400, E: IHC ×400, F: IHC ×100, G: IHC ×400, H: IHC ×100).

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A rare case of pigmented epithelioid melanocytoma on the penis as a divided nevus.

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