Journal of Dermatology 2015; 42: 425–443

LETTERS TO THE EDITOR

Unusual case of pyogenic granuloma-like Kaposi’s sarcoma on the sole Dear Editor, Superficially located nodular Kaposi’s sarcoma, which may be difficult to distinguish from pyogenic granulomas, are referred to as pyogenic granuloma-like Kaposi’s sarcoma.1 Herein, we report an unusual case of Kaposi’s sarcoma mimicking a pyogenic granuloma and stress the importance of not overlooking this diagnosis. An 81-year-old Korean man presented with a single painless nodule on the medial side of the left sole that had increased gradually in size over the course of 4 months. A physical examination revealed a 1.5 cm 9 1.5 cm, circumscribed, red, exophytic nodule (Fig. 1a,b). The patient did not show other abnormal physical findings, and did not have a history of cutaneous tumors including Kaposi’s sarcoma. The clinical findings suggested a possible diagnosis of pyogenic granuloma. Skin biopsy revealed closely packed spindle cells, with proliferation of vascular endothelial cells, and extravasated erythrocytes. Slit-like, dilated vascular spaces were observed, without lobulation (Fig. 1c,d). The vessels and spindle cell areas were positive for CD31, CD34 and D2-40 markers (Fig. 1e,f), and immunostaining for human herpesvirus 8 (HHV-8) was positive (Fig. 1g). The lesion was diagnosed as Kaposi’s sarcoma, and treated with wide local excision. There have been several reports of Kaposi’s sarcomas with clinicohistological features similar to those of pyogenic granulomas.2,3 This new entity, pyogenic granuloma-like Kaposi’s sarcoma, is considered a true Kaposi’s sarcoma because of its clinical course and the presence of HHV-8 DNA.2 The difference in immunohistochemical staining of markers such as CD31, CD34 or factor VIII can be helpful in distinguishing pyogenic granuloma-like Kaposi’s sarcoma from pyogenic granuloma. Kaposi’s sarcoma also needs to be differentiated from malignant vascular tumors, such as angiosarcoma or lymphangiosarcoma, both of which usually have a characteristic “dissecting collagen” pattern and markedly atypical cells lining the endothelium, which are not seen in Kaposi’s sarcoma. However, differential diagnosis from other vascular tumors is often difficult. If pathological and other immunohistochemical analyses are not sufficient to confirm the diagnosis, D2-40 and HHV-8 staining can be helpful in differentiating Kaposi’s sarcoma from other vascular tumors. Two cases of pyogenic granuloma-like Kaposi’s sarcoma were recently reported in Asian patients,4,5 but they differed from the present case in that one patient4 had multiple lesions of Kaposi’s sarcoma and the other5 had a history of Kaposi’s sarcoma. The present patient presented with only a sole

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Figure 1. Clinical and histopathological findings of pyogenic granuloma-like Kaposi’s sarcoma. (a,b) A 1.5 cm 9 1.5 cm, circumscribed, red, ulcerative, exophytic nodule observed on the medial side of the patient’s left sole. (c) Histopathological analyses revealed a relatively well-defined tumor in the upper dermis (hematoxylin–eosin [HE], original magnification 940). (d) Spindle cell proliferation with numerous intravascular and extravasated erythrocytes was observed. Increased slit-like vascular channels were closely packed, displaying a back-toback appearance (HE, 9200). (e,f) Endothelial marker CD31 and lymphatic marker D2-40 were positive. (g) Immunostaining for human herpesvirus 8 was also positive.

nodule and had no history of Kaposi’s sarcoma. Therefore, this case highlights the need to be aware of this rare variant of Kaposi’s sarcoma, even in cases with a single nodule suspected of a vascular origin, and a biopsy including immunohistochemical staining for HHV-8 and D2-40 needs to be performed. In conclusion, this is the first report of a case of pyogenic granuloma-like Kaposi’s sarcoma in the Asian population, which appeared as a single nodule on the sole without any

Correspondence: Kwang Ho Kim, M.D., Department of Dermatology, Hallym University Sacred Heart Hospital, 22, Gwanpyeong-ro 170beon-gil, Dongan-gu, Anyang, 431-796, Gyeonggi-do, Korea Email: [email protected]

© 2015 Japanese Dermatological Association

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Letters to the Editor

other lesions. Cases of Kaposi’s sarcoma clinically mimicking pyogenic granuloma have rarely been reported in dermatology journals, and the present case is valuable especially in that it is the first report of pyogenic granuloma-like sarcoma, which appeared as a sole nodule, in the Asian population.

CONFLICT OF INTEREST:

None.

Min Kyung LEE, Sang Hyeon KU, Eun Byul CHO, Eun Joo PARK, Kwang Ho KIM, Kwang Joong KIM Department of Dermatology, College of Medicine, Hallym University, Anyang, Korea

REFERENCES 1 Urquhart JL, Uzieblo A, Kohler S. Detection in HHV-8 in pyogenic granuloma-like Kaposi sarcoma. Am J Dermatopathol 2006; 28: 317– 321. 2 Ryan P, Aarons S, Murray D et al. Human herpes virus 8(HHV-8) detected in two patients with Kaposi’s sarcoma-like pyogenic granuloma. J Clin Pathol 2002; 55: 619–622. 3 Cabibi D, Cacciartore M, Viviano E, Guarnotta C, Aragona F. ‘Pyogenic granuloma-like Kaposi’s sarcoma’ on the hands: immunohistochemistry and human herpesvirus-8 detection. J Eur Acad Dermatol Venereol 2009; 23: 587–589. 4 Kim HJ, Kim JY, Choi JE, Kye YC, Ahn HH, Seo SH. A firm, bluish, hemorrhagic nodule on the left heel. Int J Dermatol 2013; 52: 131–132. 5 Lee DY, Park SW. Kaposi sarcoma mimicking pyogenic granuloma below the toenail. Int J Dermatol 2014; 53: e202–e204.

doi: 10.1111/1346-8138.12811

Case of angiosarcoma on the abdominal wall, an extremely rare variant, putatively shared the pathogenesis with Stewart–Treves syndrome Dear Editor, Cutaneous angiosarcoma is an uncommon soft tissue sarcoma typically appearing on the head and neck of the elderly.1 Herein, we report an extremely rare case of angiosarcoma on the right upper abdominal wall formed after ipsilateral radical mastectomy and postoperative radiotherapy. A 78-year-old Japanese woman was referred to us with a 3-month history of growing skin nodules and spreading induration on her right upper to lateral abdominal wall. The nodules were surgically removed at another institution but recurred. She had undergone right radical mastectomy and postoperative radiotherapy 7 years before the lesions emerged. Her stature was normal (145.5 cm, 43.60 kg, body mass index 20.3). On physical examination, a yellow to brown, large, stiff plaque with multiple, dark red papules and nodules up to 20 mm in diameter was observed on her right upper to lateral abdominal wall (Fig. 1a,b). Some petechiae were noted on the plaque. Laboratory studies revealed no abnormal findings. Positron emission tomography/computed tomography detected multiple bone and liver metastasis. Histopathological investigation of the plaque lesion detected irregularly demarcated nodules invading into the fibrous septa and the lobules of the subcutis (Fig. 1c). Cords of cells infiltrating into the dermis was also noted (Fig. 1c). The nodule mainly consisted of irregularly anastomosing vascular structures lined with enlarged endothelial cells (Fig. 1d) and sheets of pleomorphic epithelioid cells with enlarged nuclei and substantial amount of mitotic figures (Fig. 1e). The tumor cells showed positive staining for CD31 (Fig. 1f) and factor VIII-related antigen (Fig. 1g). No pathologi-

cal feature indicating proliferation of lymphatic vessels was noted. Based on these findings, the diagnosis of angiosarcoma was made. She was treated with monthly chemotherapy consisting of gemcitabine (1000 mg/m2 on days 1 and 8) and docetaxel (900 mg/m2 on day 8) and achieved partial response. However, progressive thrombocytopenia due to multiple bone metastasis appeared and the patient died of multiple organ failure 6 months after her first visit. A published work review found 19 cases of angiosarcoma on the abdominal wall (Table S1). They were mostly female with a history of cervical cancer operation with postoperative irradiation. Morbid obesity was noted in seven cases. Interestingly, four out of these seven cases were without the record of surgery. These findings indicated that any condition potentially affecting lymphatic flow of the abdominal wall could cause angiosarcoma, similar to how Stewart–Treves syndrome develops secondarily to persisting postoperative lymphedema. Lymphatic drainage in the abdominal wall above the umbilicus flows to the ipsilateral axial lymph nodes.2 In our case, past remedies might have caused lymphedema in the chest to upper abdomen. Kaplan– Meier analysis of the reported cases elucidated that the 1-year estimated survival rate was 46% while the rate accounted for approximately 70% in common forms of angiosarcomas on the head and neck,3–5 suggesting that the prognosis may be poorer. This study encourages physicians to include close inspection of the abdominal wall in postoperative follow up of uterus or ovarian malignancies as well as breast cancer, especially in obese individuals.

Correspondence: Manabu Ohyama, M.D., Ph.D., Department of Dermatology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan. Email: [email protected]

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© 2015 Japanese Dermatological Association

Unusual case of pyogenic granuloma-like Kaposi's sarcoma on the sole.

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