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CASE REPORT
Year : 2019  |  Volume : 64  |  Issue : 5  |  Page : 411-413
Zosteriform lymphangitic metastases of eccrine porocarcinoma


1 Department of Oncology, Hainan Provincial Hospital of TCM, Haikou, China
2 Department of Dermatology, Hainan Provincial Hospital of Skin Disease, Haikou, China

Date of Web Publication5-Sep-2019

Correspondence Address:
Jiejie Lu
No. 33, Southern Road of Longkun, Haikou
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_440_18

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   Abstract 


Eccrine porocarcinoma (EPC) is a rare malignant neoplasm which originates from the intraepidermal portion of eccrine sweat glands or acrosyringium. Here, we report a unique case of cutaneous metastases of EPC presenting with an erosive plaque on the left thumb and multiple nodules on left forearm. The histopathological examination of the wrist lesion revealed islands of basaloid tumor cells with eosinophilic cytoplasm, downward infiltrating growths, ductal differentiation, and intracytoplasmic lumen formations and focally connected to the epidermis. Acrosyringeal differentiation was confirmed by positive immunohistochemical staining using antibodies to carcinoembryonic antigen (CEA) and epithelial membrane antigen (EMA) in some of porocarcinoma cells. Lymphatic embolization was shown by immunostaining of podoplanin. To the best of our knowledge, this is the first reported EPC case with a primary lesion on the left thumb and zosteriform growth pattern of lymphangitic metastases on left forearm. Our case highlights the diagnostic challenges and reveals lymphangitic metastasis mode of EPC.


Keywords: Eccrine porocarcinoma, lymphangitic metastasis, zosteriform


How to cite this article:
Zhang Y, Zhang M, Wu W, Lu J. Zosteriform lymphangitic metastases of eccrine porocarcinoma. Indian J Dermatol 2019;64:411-3

How to cite this URL:
Zhang Y, Zhang M, Wu W, Lu J. Zosteriform lymphangitic metastases of eccrine porocarcinoma. Indian J Dermatol [serial online] 2019 [cited 2019 Sep 23];64:411-3. Available from: http://www.e-ijd.org/text.asp?2019/64/5/411/265943





   Introduction Top


Eccrine porocarcinoma (EPC) is a rare malignant neoplasm which originates from the intraepidermal portion of eccrine sweat glands or acrosyringium. Here, we report a unique case of EPC with a primary lesion on the left thumb and zosteriform growth pattern of lymphangitic metastases on left forearm.


   Case Report Top


In May 2018, a 53-year-old man was referred to our department with a 1-year history of erosive plaque on the left thumb and multiple nodules on left forearm. One year ago, he noticed the asymptomatic plaque on left thumb without obvious predisposing factors. The lesion was gradually enlarging, and ulcerated. The patient was treated with antibiotics and daily dressing at a local clinic. However, his symptoms did not improve. Two months ago, multiple papules and nodules developed progressively on the dorsum of left hand and forearm. His past medical history was unremarkable.

On physical examination, there was an erosive plaque on the pulp of his left thumb with nail bed involved [Figure 1]a. The patient was also found to have multiple well-demarcated papules and nodules on the dorsum of the left thumb, hand, and forearm, measuring approximately from 0.2 to 1.5 cm in diameter [Figure 1]b. There was no palpable regional lymphadenopathy. No obvious abnormality was detected in routine tests of blood, urine and stool, hepatic and renal function tests, blood sugar, lipid profile, and electrocardiogram. Histological findings presented as islands of basaloid tumor cells with eosinophilic cytoplasm, downward infiltrating growth pattern, ductal differentiation, and intracytoplasmic lumen formations, and focally connected to the epidermis [Figure 1]c. Immunohistochemistry (IHC) staining pattern was consistent with malignant appendage tumor of eccrine differentiation. IHC showed positive expression of epithelial membrane antigen (EMA) [Figure 2]a, carcinoembryonic antigen (CEA) [Figure 2]b, and podoplanin [Figure 2]c.
Figure 1: Clinicopathological features of EPC in our case. A 53-year-old man presented erosive plaque on left thumb (a) and multiple nodules with lymphangitic metastasis of left forearm (b). The histopathological examination of the wrist lesion revealed islands of basaloid tumor cells with eosinophilic cytoplasm, downward infiltrating growths, ductal differentiation, and intracytoplasmic lumen formations and focally connected to the epidermis (c) (H and E, ×100)

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Figure 2: Immunohistochemical findings of the left wrist tumor. Acrosyringeal differentiation was confirmed by positive immunohistochemical staining using antibodies to CEA (a) (×100) and EMA (b) (×100) in some of porocarcinoma cells. Lymphatic embolization was shown by immunostaining of podoplanin (c) (arrow) (×100)

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Based on clinicopathological features, we diagnosed lymphangitic metastasis of EPC. The patient refused further examination and treatment in our hospital, and then underwent thumb amputation and wide surgical excision of the tumor lesions in another hospital in June 2018. The patient died from brain and lung metastasis of EPC in October 2018.


   Discussion Top


Until now, approximately 300 cases of EPC have been reported worldwide since this disease was first described by Pinkus and Mehregan in 1963.[1] The pathogenesis of EPC was unclear and usually occurs in elderly patients with major involvement in the lower extremities.[2] Only 8% of EPC occur on the upper extremity, and 3% are on the hand.[3] In this case, the primary lesion occurred on the thumb, which was the second case reported.

The lesions of EPC had various but nonspecific morphologies, so EPC is a challenge to diagnose because it can mimic many other dermatological diseases. We report a unique case of cutaneous metastases of EPC clinically presenting with a zosteriform pattern of multiple reddish and well-demarcated papules and nodules. Zosteriform presentation of papules and plaques have been reported in several kinds of skin diseases, such as sporotrichosis, leishmaniasis, seborrheic keratosis, palisaded-encapsulated neuroma, eccrine spiradenoma, cutaneous leiomyoma, zosteriform cutaneous metastases from malignant tumor, and so on.[4],[5],[6],[7],[8],[9],[10],[11]

The accurate diagnosis and differential diagnosis of EPC relies on specific histological features. Current immunohistological markers were reported to be helpful. CEA and EMA are often used as the principal indicators of ductal differentiation owing to their high sensitivity. Positive immunohistochemical staining to human cytokeratin (CK), CEA, and EMA can confirm acrosyringeal differentiation.[12] In this report, immunohistochemical studies showed that CEA and EMA were expressed in some of the porocarcinoma cells, which confirmed the diagnosis of EPC. Podoplanin, recognized by monoclonal antibody D2-40, is a useful marker for lymphatic endothelial cells and lymphangiogenesis. Immunostaining of podoplanin clearly revealed extensive lymphatic invasion by the tumor cell.[13] In this case, podoplanin staining demonstrated intralymphatic neoplastic cells, which proved pathologically that metastasis of EPC occurs primarily through the lymphatic system.

EPC has a high tendency of metastatic spread. Local recurrence, nodal recurrence, and distant metastases were observed in 17%–20%, 10.4%–20%, and 8.3%–11.4% of cases, respectively.[3],[14],[15] Cutaneous metastasis can be explained by the direct lymphatic infiltration of cancer cells and posterior reinvasion of the epidermis.[16] Zosteriform dissemination was usually regarded as the initial lymphatic spread of the tumor cells,[17],[18] which also was observed in our case.

There is no established standard modality of treatment for EPC, especially in cases of metastasis. Conventional wide-local excision with broad tumor margins was currently the first-line treatment.[19] Mohs micrographic surgery seems to be effective in eradicating early EPC and minimizing the risk of recurrence.[20],[21] In the case of lymphadenopathy, sentinel lymph node biopsy might be useful in determining subclinical metastasis in the regional lymph nodes.[22] In cases of metastasis, various combinations of excision, radiotherapy, and chemotherapy, including paclitaxel, docetaxel, interferon-α, and cetuximab, have been used.[23],[24],[25],[26] However, the long-term prognosis is still unfavorable. In case of recurrent or metastatic EPC, the prognosis is actually poor, and mortality rate of metastatic EPC patients can be up to 67%.[14] Tumor thickness, tumor size, mitosis, lymphovascular invasion, lymphnode metastasis, and infiltrative growth pattern are correlated with poor prognosis.[22],[27]

To the best of our knowledge, this is the first reported EPC case with a primary lesion on the left thumb and zosteriform growth pattern of lymphangitic metastases on left forearm. Our case highlights the diagnostic challenges and reveals lymphangitic metastasis mode of EPC.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Pinkus H, Mehregan, AH. Epidermotropic eccrine carcinoma. A case combining features of eccrine poroma and Paget's dermatosis. Arch Dermatol 1963;88:597-606.  Back to cited text no. 1
    
2.
Salih AM, Kakamad FH, Essa RA, Rauf GM, M SA, S HM, et al. Porocarcinoma: A systematic review of literature with a single case report. Int J Surg Case Rep 2017;30:13-6.  Back to cited text no. 2
    
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Robson A, Greene J, Ansari N, Kim B, Seed PT, Mckee PH, et al. Eccrine porocarcinoma (malignant eccrine poroma): A clinicopathologic study of 69 cases. Am J Surg Pathol 2001;25:710-20.  Back to cited text no. 3
    
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Syuto T, Yamanaka M, Amano H, Ishikawa O. Two cases of cutaneous sporotrichosis with atypical clinical features: A clinical study of 38 cases with sporotrichosis from Gunma university hospital. Japanese J Dermatol 2009;119:49-53.  Back to cited text no. 4
    
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Omidian M, Mapar M. Chronic zosteriform cutaneous leishmaniasis. Indian J Dermatol Venereol Leprol 2006;72:41.  Back to cited text no. 5
[PUBMED]  [Full text]  
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Halder C, Sen S, Gangopadhyay A, Bala S. Zosteriform palisaded encapsulated neuroma: An unusual presentation. Indian J Dermatol 2013;58:492.  Back to cited text no. 7
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Alfonso-Trujillo I, Arteaga-Hernández E, Pérez-Suárez JC. Eccrine spiradenoma in a zosteriform distribution: Presentation of a case. Actas Dermosifiliogr (English Edition) 2009;100:619-20.  Back to cited text no. 8
    
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Riera-Leal L, Guevara-Gutiérrez E, Barrientos-García JG, Madrigal-Kasem R, Briseño-Rodríguez G, Tlacuilo-Parra A. Eccrine porocarcinoma: Epidemiologic and histopathologic characteristics. Int J Dermatol 2015;54:580-6.  Back to cited text no. 12
    
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Plaza JA, Ortega PF, Stockman DL, Suster S. Value of p63 and podoplanin (d2-40) immunoreactivity in the distinction between primary cutaneous tumors and adenocarcinomas metastatic to the skin: A clinicopathologic and immunohistochemical study of 79 cases. J Cutan Pathol 2010;37:403-10.  Back to cited text no. 13
    
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Snow SN, Reizner GT. Eccrine porocarcinoma of the face. J Am Acad Dermatol 1992;27:306-11.  Back to cited text no. 14
    
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Skowron F, Poulhalon N, Balme B, Touzet S, Thomas L. Thomas. Primary eccrine porocarcinoma: A clinicopathological study of 50 cases. Ann Dermatol Venereol 2014;141:258-64.  Back to cited text no. 15
    
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GonzálezLópez MA, VázquezLópez F, Soler T, GómezDiéz S, Garcia, YH, Manjón JA, et al. Metastatic eccrine porocarcinoma: A 5.6-year followup study of a patient treated with a combined therapeutic protocol. Dermatol Surg 2003;29:1227-32.  Back to cited text no. 16
    
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Ando KI, Goto Y, Kato K, Murase T, Matsumoto Y, Ohashi M. Zosteriform inflammatory metastatic carcinoma from transitional cell carcinoma of the renal pelvis. J Am Acad Dermatol 1994;31:284-6.  Back to cited text no. 17
    
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Nina S, Müller, Carolin Z, Wolfgang H, Lonsdorf AS. Zosteriform metastases of eccrine porocarcinoma mimicking eruptive seborrhoeic keratoses. Acta Derm Venereol 2015;95:245-6.  Back to cited text no. 18
    
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Nazemi A, Higgins S, Swift R, In G, Miller K, Wysong A. Eccrine porocarcinoma: New insights and a systematic review of the literature. Dermatol Surg 2018;44:1247-61.  Back to cited text no. 19
    
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Borgognoni L, Pescitelli L, Urso C, Brandani P, Sestini S, Chiarugi C, et al. A rare case of anal porocarcinoma treated by electrochemotherapy. Future Oncol 2014;10:2429-34.  Back to cited text no. 23
    
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Gutermuth J, Audring H, Voit C, Trefzer U, Haas N. Antitumour activity of paclitaxel and interferon-alpha in a case of metastatic eccrine porocarcinoma. J Eur Acad Dermatol Venereol 2004;18:477-9.  Back to cited text no. 24
    
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Plunkett TA, Hanby AM, Miles DW, Rubens RD. Metastatic eccrine porocarcinoma: Response to docetaxel (Taxotere) chemotherapy. Ann Oncol 2001;12:411-4.  Back to cited text no. 25
    
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Ponzetti A, Ribero S, Caliendo V, Spadi R, Macripò G, Lista P. Long-term survival after multidisciplinary management of a metastatic sarcomatoid porocarcinoma with repeated exercises, radiotherapy, chemotherapy and cetuximab: Case report and review of literature. G Ital Dermatol Venereol 2017;152:66-70.  Back to cited text no. 26
    
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Montes-Torres A, Pérez-Plaza A, Llamas-Velasco M, Gordillo C, De AD, García-García C, et al. Eccrine porocarcinoma with extensive cutaneous metastases. Int J Dermatol 2016;55:e156-60.  Back to cited text no. 27
    


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