Letters to the Editor
molecular biology findings indicated that the first maggots removed were Lucilia sericata and the second maggots removed were Sarcophaga crassipalpis. It is remarkable that two different species of flies infested the patient’s vulvar SCC within a span of a few days. Cases of vulvar malignant wound myiases are rarely seen, especially in Japan. It is likely that the second species of maggot was deposited after removal of the first species of maggot. The former (L. sericata) is an oviparous species, while the latter (S. crassipalpis) is ovoviviparous. Female flies of the latter species deposit not eggs but larvae, which are visible as they are at least 1 mm just after deposition. Indeed, we did not notice the latter maggots when we observed the vulva for the first time. Generally, myiases of wounds or tumors are managed by treating the wounds or tumors.1 However, this patient’s recurrent vulvar SCC was terminal and incurable, and the patient was receiving palliative care. The tumor was worsening, and was sore to touch and difficult to wash. She was forced to leave it unclean and uncovered, even while sleeping. Thus, the odor of her exposed vulvar tumor could attract flies, especially during the summer. After removing the maggots from her vulvar SCC, we advised her to wear a disposable diaper because it might cause her less pain than common underwear. Since then, her myiasis has not recurred.
CONFLICT OF INTEREST:
None.
Hiroyuki HIRAOKA,1 Toshiyuki OZAWA,1 Junko SOWA-OSAKO,1 Tomoyuki ICHIMURA,2 Isao KIMATA-TERAMOTO,3 Rie ISOZUMI,3 Akira KANEKO,3 Daisuke TSURUTA1 Departments of 1Dermatology, 2Gynecology, and 3Parasitology, Osaka City University Graduate School of Medicine, Osaka, Japan doi: 10.1111/1346-8138.12911
REFERENCES 1 Francesconia F, Lupib O. Myiasis. Clin Microbiol Rev 2012; 25: 79– 105. 2 Uni S, Nakagawa K, Shibata T, Fukunaga A. Two Sarcophaga wound myiases in Japan: S. peregrina at a trichophytosis sore of an inpatient and S. crassipalpis in a post-surgical wound of a home convalescent. Med Entomol Zool 2006; 57: 235–240. 3 Kano R. Identification technique. In: Asahina S, ed. Guidance of the Inspection about the Hygiene Animal. Tokyo: Japan Environmental Sanitation Center, The Ministry of Welfare and Health of Japan, 1971; 4–27. (In Japanese). 4 Smith MA, Woodley NE, Janzen DH, Hallwachs W, Hebert PDN. DNA barcodes reveal cryptic host-specificity within the presumed polyphagous members of a genus of parasitoid flies (Diptera: Tachinidae). Proc Natl Acad Sci USA 2006; 103: 3657–3662.
Case of ossifying epithelioid hemangioendothelioma on the forearm Dear Editor, Epithelioid hemangioendothelioma (EHE) is a rare tumor of the vascular endothelium that arises in soft tissue, bone, skin and various parenchymal organs.1 It was first described by Weiss and Enzinger2 and is considered as borderline malignancy. Here, we report a case of ossifying EHE on the forearm. Characteristically, eggshell-like lamellar bone enclosed this tumor showing a well-circumscribed appearance. A 68-year-old Japanese woman presented with a 2-year history of a slowly growing nodule on her left forearm. Examination revealed a 10 mm 9 10 mm, well-circumscribed, firm, subcutaneous nodule. It was hardly movable from the underlying tissue upon palpation. Our initial clinical differential diagnoses were angioleiomyoma and neurinoma. The tumor was completely excised under local anesthesia. The histological findings revealed a well-defined tumor mass, and a continuous rim of eggshell-like bony tissue was present on the outermost part. The tumor was attached to the fascia and was in connection with a large vessel (Fig. 1a). In a higher power view, the inside of the shell exhibited sheets of tumor cells with occasional osteoid formation (Fig. 1b, c). The proliferated cells were relatively large round to short spindle-shaped cells arranged in a cord- or sheet-like pattern embed-
ded in fibromyxoid stroma (Fig. 1d). The main component cells possessed round nuclei and abundant cytoplasm with occasional characteristic intracytoplasmic vacuoles of variable sizes (Fig. 1e). Mitotic figures were occasionally seen. Immunohistochemically, the tumor cells were positive for CD34 (Fig. 1f), ERG (Fig. 1g) and factor VIII-related antigen (Fig. 1h), but negative for CD31, D2-40, epithelial membrane antigen and desmin. From these findings, we diagnosed the tumor as ossifying EHE. Weiss and Enzinger proposed the term EHE for this biologically borderline malignancy that can be classified between a hemangioma and a conventional angiosarcoma.2 Ossification in this neoplasm has been well documented, but all such cases involved small foci of metaplastic bone formation according to the limited number of cases in the published work.3,4 We previously proposed the term ossifying EHE for a well-circumscribed EHE with a shell of mature bony tissue.4 Since then, however, this type of unique EHE has not been reported. Among many other soft-tissue tumors, ossifying fibromyxoid tumor of soft tissue shows prominent ossification and some such cases were reported to have encapsulation by rim-like bony tissue that was very similar to that in our case.5 However, in ossifying fibromyxoid tumor of soft tissue, the component
Correspondence: Takeshi Nakahara, M.D., Ph.D., Division of Skin Surface Sensing, Department of Dermatology, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan. Email: [email protected]
© 2015 Japanese Dermatological Association
841
Letters to the Editor
(a)
(b)
(c)
(f)
(d)
(g)
(e)
(h)
cells are small, have small amounts of eosinophilic cytoplasm, and proliferate in cords, nests and sheets, in fibromyxoid matrix that is almost always associated with various amounts of ossification. In addition, immunohistochemical positivity of vascular endothelial markers such as ERG is useful in the differentiation of ossifying EHE from ossifying fibromyxoid tumor because the component cells of the latter are negative for such markers. The overall prognosis of EHE is unclear, and the variety of clinical behavior suggests intermediate malignancy of this neoplasm.1 We speculate that ossifying EHE behaves as a completely benign neoplasm because the bony shell may limit its infiltrating growth; however, careful follow up may be needed.
CONFLICT OF INTEREST:
None.
Takeshi NAKAHARA,1,2 Yuichi YAMADA,3 Eriko ITOH,1,2 Makiko KIDO-NAKAHARA,2 Yoshinao ODA,3 Hiromaro KIRYU,4 Masutaka FURUE2
Figure 1. (a) The nodule was a well-circumscribed lesion with a shell of mature bony tissue. (b,c) Inside the shell, there were sheets of tumor cells with occasional osteoid formation. (d,e) The proliferated cells were relatively large round to short spindle-shaped cells arranged in a cordor sheet-like pattern (hematoxylin–eosin, original magnifications: [a] 940; [b] 9200; [c] 9100; [d] 9200; [e] 9400). The main component cells possessed round nuclei and abundant cytoplasm with occasional characteristic intracytoplasmic vacuoles. Immunohistochemical staining for (f) CD34, (g) ERG and (h) factor VIII showed positive reactions in the tumor cells (original magnifications: [f] 9200; [g] 9200; [h] 9200). 1 Division of Skin Surface Sensing, 2Department of Dermatology, Department of Anatomic Pathology, Graduate School of Medical Sciences, Kyushu University, and 4Fukuoka Institute for Dermatopathology, Fukuoka, Japan 3
doi: 10.1111/1346-8138.12915
REFERENCES 1 Clarke LE, Lee R, Militello G, Elenitsas R, Junkins-Hopkins J. Cutaneous epithelioid hemangioendothelioma. J Cutan Pathol 2008; 35: 236–240. 2 Weiss SW, Enzinger FM. Epithelioid hemangioendothelioma: a vascular tumor often mistaken for a carcinoma. Cancer 1982; 5: 970–981. 3 Weidner N. Atypical tumor of the mediastinum: epithelioid hemangioendothelioma containing metaplastic bone and osteoclastlike giant cells. Ultrastruct Pathol 1991; 15: 481–488. 4 Kiryu H, Hashimoto H, Hori Y. Ossifying epithelioid hemangioendothelioma. J Cutan Pathol 1996; 23: 558–561. 5 Miettinen M, Finnell V, Fetsch JF. Ossifying fibromyxoid tumor of soft parts–a clinicopathologic and immunohistochemical study of 104 cases with long-term follow-up and a critical review of the literature. Am J Surg Pathol 2008; 32: 996–1005.
Malignant melanoma on female clitoris with bidirectional upper and lower lymphatic flow Dear Editor, Malignant melanoma in the female genital area, especially the clitoris, is relatively rare. Usually, the route from the genital area is considered to lead into the ipsilateral inguinal lymph nodes (LN). However, the precise route has not
been identified. Here, we report a case of clitoral melanoma, in which bidirectional lymphatic flows were identified. Interestingly, downward arch form bypass flows were clearly demonstrated, which drained into the inguinal LN of each side.
Correspondence: Chiaki Takahashi, M.D., Department of Dermatology, Asahikawa Medical University, 2-1-1-1 Midorigaoka-Higashi, Asahikawa, Hokkaido 078-8510, Japan. Email: [email protected]
842
© 2015 Japanese Dermatological Association
molecular biology findings indicated that the first maggots removed were Lucilia sericata and the second maggots removed were Sarcophaga crassipalpis. It is remarkable that two different species of flies infested the patient’s vulvar SCC within a span of a few days. Cases of vulvar malignant wound myiases are rarely seen, especially in Japan. It is likely that the second species of maggot was deposited after removal of the first species of maggot. The former (L. sericata) is an oviparous species, while the latter (S. crassipalpis) is ovoviviparous. Female flies of the latter species deposit not eggs but larvae, which are visible as they are at least 1 mm just after deposition. Indeed, we did not notice the latter maggots when we observed the vulva for the first time. Generally, myiases of wounds or tumors are managed by treating the wounds or tumors.1 However, this patient’s recurrent vulvar SCC was terminal and incurable, and the patient was receiving palliative care. The tumor was worsening, and was sore to touch and difficult to wash. She was forced to leave it unclean and uncovered, even while sleeping. Thus, the odor of her exposed vulvar tumor could attract flies, especially during the summer. After removing the maggots from her vulvar SCC, we advised her to wear a disposable diaper because it might cause her less pain than common underwear. Since then, her myiasis has not recurred.
CONFLICT OF INTEREST:
None.
Hiroyuki HIRAOKA,1 Toshiyuki OZAWA,1 Junko SOWA-OSAKO,1 Tomoyuki ICHIMURA,2 Isao KIMATA-TERAMOTO,3 Rie ISOZUMI,3 Akira KANEKO,3 Daisuke TSURUTA1 Departments of 1Dermatology, 2Gynecology, and 3Parasitology, Osaka City University Graduate School of Medicine, Osaka, Japan doi: 10.1111/1346-8138.12911
REFERENCES 1 Francesconia F, Lupib O. Myiasis. Clin Microbiol Rev 2012; 25: 79– 105. 2 Uni S, Nakagawa K, Shibata T, Fukunaga A. Two Sarcophaga wound myiases in Japan: S. peregrina at a trichophytosis sore of an inpatient and S. crassipalpis in a post-surgical wound of a home convalescent. Med Entomol Zool 2006; 57: 235–240. 3 Kano R. Identification technique. In: Asahina S, ed. Guidance of the Inspection about the Hygiene Animal. Tokyo: Japan Environmental Sanitation Center, The Ministry of Welfare and Health of Japan, 1971; 4–27. (In Japanese). 4 Smith MA, Woodley NE, Janzen DH, Hallwachs W, Hebert PDN. DNA barcodes reveal cryptic host-specificity within the presumed polyphagous members of a genus of parasitoid flies (Diptera: Tachinidae). Proc Natl Acad Sci USA 2006; 103: 3657–3662.
Case of ossifying epithelioid hemangioendothelioma on the forearm Dear Editor, Epithelioid hemangioendothelioma (EHE) is a rare tumor of the vascular endothelium that arises in soft tissue, bone, skin and various parenchymal organs.1 It was first described by Weiss and Enzinger2 and is considered as borderline malignancy. Here, we report a case of ossifying EHE on the forearm. Characteristically, eggshell-like lamellar bone enclosed this tumor showing a well-circumscribed appearance. A 68-year-old Japanese woman presented with a 2-year history of a slowly growing nodule on her left forearm. Examination revealed a 10 mm 9 10 mm, well-circumscribed, firm, subcutaneous nodule. It was hardly movable from the underlying tissue upon palpation. Our initial clinical differential diagnoses were angioleiomyoma and neurinoma. The tumor was completely excised under local anesthesia. The histological findings revealed a well-defined tumor mass, and a continuous rim of eggshell-like bony tissue was present on the outermost part. The tumor was attached to the fascia and was in connection with a large vessel (Fig. 1a). In a higher power view, the inside of the shell exhibited sheets of tumor cells with occasional osteoid formation (Fig. 1b, c). The proliferated cells were relatively large round to short spindle-shaped cells arranged in a cord- or sheet-like pattern embed-
ded in fibromyxoid stroma (Fig. 1d). The main component cells possessed round nuclei and abundant cytoplasm with occasional characteristic intracytoplasmic vacuoles of variable sizes (Fig. 1e). Mitotic figures were occasionally seen. Immunohistochemically, the tumor cells were positive for CD34 (Fig. 1f), ERG (Fig. 1g) and factor VIII-related antigen (Fig. 1h), but negative for CD31, D2-40, epithelial membrane antigen and desmin. From these findings, we diagnosed the tumor as ossifying EHE. Weiss and Enzinger proposed the term EHE for this biologically borderline malignancy that can be classified between a hemangioma and a conventional angiosarcoma.2 Ossification in this neoplasm has been well documented, but all such cases involved small foci of metaplastic bone formation according to the limited number of cases in the published work.3,4 We previously proposed the term ossifying EHE for a well-circumscribed EHE with a shell of mature bony tissue.4 Since then, however, this type of unique EHE has not been reported. Among many other soft-tissue tumors, ossifying fibromyxoid tumor of soft tissue shows prominent ossification and some such cases were reported to have encapsulation by rim-like bony tissue that was very similar to that in our case.5 However, in ossifying fibromyxoid tumor of soft tissue, the component
Correspondence: Takeshi Nakahara, M.D., Ph.D., Division of Skin Surface Sensing, Department of Dermatology, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan. Email: [email protected]
© 2015 Japanese Dermatological Association
841
Letters to the Editor
(a)
(b)
(c)
(f)
(d)
(g)
(e)
(h)
cells are small, have small amounts of eosinophilic cytoplasm, and proliferate in cords, nests and sheets, in fibromyxoid matrix that is almost always associated with various amounts of ossification. In addition, immunohistochemical positivity of vascular endothelial markers such as ERG is useful in the differentiation of ossifying EHE from ossifying fibromyxoid tumor because the component cells of the latter are negative for such markers. The overall prognosis of EHE is unclear, and the variety of clinical behavior suggests intermediate malignancy of this neoplasm.1 We speculate that ossifying EHE behaves as a completely benign neoplasm because the bony shell may limit its infiltrating growth; however, careful follow up may be needed.
CONFLICT OF INTEREST:
None.
Takeshi NAKAHARA,1,2 Yuichi YAMADA,3 Eriko ITOH,1,2 Makiko KIDO-NAKAHARA,2 Yoshinao ODA,3 Hiromaro KIRYU,4 Masutaka FURUE2
Figure 1. (a) The nodule was a well-circumscribed lesion with a shell of mature bony tissue. (b,c) Inside the shell, there were sheets of tumor cells with occasional osteoid formation. (d,e) The proliferated cells were relatively large round to short spindle-shaped cells arranged in a cordor sheet-like pattern (hematoxylin–eosin, original magnifications: [a] 940; [b] 9200; [c] 9100; [d] 9200; [e] 9400). The main component cells possessed round nuclei and abundant cytoplasm with occasional characteristic intracytoplasmic vacuoles. Immunohistochemical staining for (f) CD34, (g) ERG and (h) factor VIII showed positive reactions in the tumor cells (original magnifications: [f] 9200; [g] 9200; [h] 9200). 1 Division of Skin Surface Sensing, 2Department of Dermatology, Department of Anatomic Pathology, Graduate School of Medical Sciences, Kyushu University, and 4Fukuoka Institute for Dermatopathology, Fukuoka, Japan 3
doi: 10.1111/1346-8138.12915
REFERENCES 1 Clarke LE, Lee R, Militello G, Elenitsas R, Junkins-Hopkins J. Cutaneous epithelioid hemangioendothelioma. J Cutan Pathol 2008; 35: 236–240. 2 Weiss SW, Enzinger FM. Epithelioid hemangioendothelioma: a vascular tumor often mistaken for a carcinoma. Cancer 1982; 5: 970–981. 3 Weidner N. Atypical tumor of the mediastinum: epithelioid hemangioendothelioma containing metaplastic bone and osteoclastlike giant cells. Ultrastruct Pathol 1991; 15: 481–488. 4 Kiryu H, Hashimoto H, Hori Y. Ossifying epithelioid hemangioendothelioma. J Cutan Pathol 1996; 23: 558–561. 5 Miettinen M, Finnell V, Fetsch JF. Ossifying fibromyxoid tumor of soft parts–a clinicopathologic and immunohistochemical study of 104 cases with long-term follow-up and a critical review of the literature. Am J Surg Pathol 2008; 32: 996–1005.
Malignant melanoma on female clitoris with bidirectional upper and lower lymphatic flow Dear Editor, Malignant melanoma in the female genital area, especially the clitoris, is relatively rare. Usually, the route from the genital area is considered to lead into the ipsilateral inguinal lymph nodes (LN). However, the precise route has not
been identified. Here, we report a case of clitoral melanoma, in which bidirectional lymphatic flows were identified. Interestingly, downward arch form bypass flows were clearly demonstrated, which drained into the inguinal LN of each side.
Correspondence: Chiaki Takahashi, M.D., Department of Dermatology, Asahikawa Medical University, 2-1-1-1 Midorigaoka-Higashi, Asahikawa, Hokkaido 078-8510, Japan. Email: [email protected]
842
© 2015 Japanese Dermatological Association
