Correspondence
Primary cutaneous sweat gland carcinoma ABSTRACT Primary cutaneous sweat gland carcinoma is a rare neoplasm of malignant sweat gland lesions. It is characterized clinically with non‑symptomatic, slow‑growing nodules. We report the case of a patient with cutaneous sweat gland carcinoma with local recurrence and metastasis to the lung that was treated with surgical resection therapy and chemotherapy. The initial neoplasm was excised but biopsy was not performed. The tumor then recurred 7 years later, was re‑excised, biopsy was performed, and diagnosed as a low‑grade hidradenocarcinoma. We presented a very good result of chemotherapy in the treatment of this rare malignant disease. It demonstrates that adjunct chemotherapy is effective to control the condition of malignant sweat‑gland carcinomas patient. KEY WORDS: Chemotherapy, metastasis, recurrence, surgical resection, sweat gland carcinoma
INTRODUCTION Malignant sweat‑gland tumors are rare neoplasms originating from the epidermal sweat ducts. The tumor is often located on the head, neck, and perineum as an asymptomatic tumor in the early stage. It started as a slow‑growing benign tumor with a rare incidence of distant metastases. Due to its low incidence and high rate of local recurrence, classical standard of diagnosis and therapy remains to be elucidative. CASE REPORT A 35‑year‑old woman presented with a long history of right‑shoulder cutaneous lesion. In the early 2003s, the patient found an exospheric mass with pain on the right shoulder and gradually developed. The mass was excised without biopsy in a local hospital. In 2010s, the patient suffered a second surgery because of the local recurrence. Biopsy suggested it was a low‑grade hidradenocarcinoma. Owning to the economic reason, the sufferer did not receive any treatment. The patient again noticed a small tumor and the tumor had progressively enlarged on the right shoulder in the area of the previous operation site after the primary operation [Figure 1]. Aggressive excision was performed on the patient. After the pathological review, the final diagnosis was low‑grade hidradenocarcinoma [Figure 2]. The patient complained of chest distress and her trouble became aggravated in July, 2011. The chest computed tomography (CT) in July 2011 revealed multiple lung metastases [Figure 3a]. After that, the patient chose to accept six cycles of chemotherapy with cisplatin plus vinorelbine (NP) 390
protocol (vinorelbine 40 mg IV D1, 8 and Cisplatin 30 mg IV D2‑5, repeat every 21 days). There were remissions of clinical symptoms and lesion of lung during the treatment [Figure 3b]. The patient appears to have stable disease at the end of six cycles of chemotherapy [Figure 3c]. DISCUSSION Sweat‑gland carcinoma affects both sexes equally in the fifth to sixth decade of life and usually shows as a solitary, firm nodule positioned on the head, neck, or more rarely, other sites. We present a case of primary sweat‑gland carcinoma of the skin in the right shoulder. It is difficult to make a proper diagnosis since this type of tumor is characterized by varying clinical appearance and histological features. The advantage of diagnosis is that this disease is easy to detect early since it arise from the surface of the body. However, considerable number of disadvantages increased the difficulty of diagnosis. First, a benign tumor must be eliminated in the differential diagnosis since it is a slow‑growing carcinoma. Second, when a node is recognized in the predilection site such as head, neck, and perineum, the possible inspection methods include ultrasound, CT, and biopsy must be carried out. Finally, due to the limited amount of literature and cases documented, it is important to have a basic knowledge of this disease process as a differential diagnosis when dealing with cutaneous lesions.
Xiao-Xia Wang, Hai-Yan Wang1, Jun-NianZheng1, Jian-Chao Sui Department of Clinical Oncology, Wendeng Central Hospital of Weihai, The Affiliated Hospital of Weifang Medical College,Weihai, 1 The Affiliated Hospital of Xuzhou Medical College, Xuzhou, China For correspondence: Prof. Jian-Chao Sui, Wendeng Central Hospital of Weihai, The Affiliated Hospital of Weifang Medical College,Weihai 264400, China.3 East Mi-shan Road, Weihai, Shandong, P.R.China. E-mail: jianchaosui @163.com
Access this article online Website: www.cancerjournal.net DOI: 10.4103/0973-1482.136667 PMID: *** Quick Response Code:
Therapy consists mainly of surgical excision, chemotherapy, and radiation.[1‑3] Surgery is the first Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Wang, et al.: Sweet gland carcinoma
compared to conventional surgical excision. The use of Mohs’ micrographic surgery was proposed by Wildemore recently in order to decrease the recurrence rate of this type of tumor.[7] Without therapy, the course of the disease is very slow and long duration, but multiple recurrences have been described. The role of radiation therapy in sweat‑gland carcinoma has changed from radio‑resistance in earlier reports[8] to good local control in recent studies[9] with technological advances in radiation therapy over the years. However, it still needs to be elucidated and hopefully, future studies will provide a better understanding of the radiation therapy.
Figure 1: The patient again noticed several small tumor and the tumor had progressively enlarged on the right shoulder in the area of the previous operation site in 2010s
a
b
Figure 2: (a) The definitive histopathology showed small cellular nests of adenocarcinoma lying in pools of extracellular mucin that were separated by fibrocollagenous septae with involvement of underlying muscle. There was no evidence of lymphatic invasion (H and E, ×10), (b) H and E, ×20
The role of chemotherapy in sweat‑gland carcinoma remains unclear. In one report, no response was observed in four cases of pediatric Eccrine porocarcinoma (EPC) treated with a combination of 5‑fluorouracil, doxorubicin, and cyclophosphamide after 1 year of therapy.[10] In the case of our patient, she initially suffered a local excision. We recommend that the disease should initially be treated with surgical excision with broad tumor margins and regional lymph node dissection, which would decrease the recurrence rate and distant metastasis of this type of tumor. The tumor in our patient had already spread to the lungs at the time of initial diagnosis in our department. The patient was, therefore, treated with chemotherapy which consisted of six cycles of Navelbine and cisplatin. After the combination chemotherapy, there were no significant changes in the size and number of primary and metastasis lesions. Our case demonstrates that aggressive surgery with adjunct chemotherapy is effective to control the condition of patient. Given the history of our case, cutaneous sweat‑gland carcinoma with local recurrence and metastasis to the lung have a good response to chemotherapy. It is necessary to carry out a detailed systematic examination, such as whole‑body CT and RI in the case of local recurrence and lymph‑node metastasis. ACKNOWLEDGMENTS
a
b
This project is supported by grants from the Program for New Century Excellent Talents in University (NCET‑08‑0700).
REFERENCES
c Figure 3: (a)The computed tomography of July 2011 revealed multiple lung metastases, (b)The computed tomography of December 2011 revealed remissions of lung lesion, (c)The computed tomography of April 2012 displayed the disease was stable
choice for management of such a sweat‑gland tumor since this type of carcinoma typically features a poor response to chemotherapy and radiotherapy.[1] Recent studies suggest that Mohs’ micrographic surgery is the method of choice[4‑6]
1. Chintamani, Sharma R, Badran R, Singhal V, Saxena S, Bansal A. Metastatic sweat gland adenocarcinoma: A clinico‑pathological dilemma. World J Surg Oncol 2003;1:13. 2. Dahill SW, Seywright M. Synchronous occurrence of cutaneous lymphadenoma and syringoid eccrine carcinoma in a single patient. Histopathology 1998;33:89‑90. 3. Ramos D, Monteagudo C, Carda C, Montesinos E, Ferrer J, Peydro‑Olaya A. Clear cell syringoid carcinoma: An ultrastructural and immunohistochemical study. Am J Dermatopathol 2000;22:60‑4. 4. Evans AT, Parham DM, Van Niekerk LJ. Metastasising eccrine syringomatous carcinoma. Histopathology 1995;26:185‑7. 5. McKee PH, Fletcher CD, Rasbridge SA. The enigmatic eccrine epithelioma (eccrine syringomatous carcinoma) Am J Dermatopathol 1990;12:552‑61.
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
391
Wang, et al.: Sweet gland carcinoma
6. Cottel WI. Eccrine epithelioma: Case report. J Dermatol Surg Oncol 1982;8:610‑1. 7. Wildemore JK, Lee JB, Humphreys TR. Mohs surgery for malignant eccrine neoplasms. Dermatol Surg 2004;30:1574‑9. 8. el‑Domeiri AA, Brasfield RD, Huvos AG, Strong EW. Sweat gland carcinoma: A clinico‑pathologic study of 83 patients. Ann Surg 1971;173:270‑4. 9. Shiohara J, Koga H, Uhara H, Takata M, Saida T. Eccrine porocarcinoma: Clinical and pathological studies of 12 cases. J Dermatol 2007;34:516‑22.
392
10. Chow CW, Campbell PE, Burry AF. Sweat gland carcinomas in children. Cancer 1984;53:1222‑7.
Cite this article as: Wang X, Wang H, Zheng J, Sui J. Primary cutaneous sweat gland carcinoma. J Can Res Ther 2014;10:390-2. Source of Support: Program for New Century Excellent Talents in University (NCET‑08‑0700), Conflict of Interest: None declared.
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Copyright of Journal of Cancer Research & Therapeutics is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Primary cutaneous sweat gland carcinoma ABSTRACT Primary cutaneous sweat gland carcinoma is a rare neoplasm of malignant sweat gland lesions. It is characterized clinically with non‑symptomatic, slow‑growing nodules. We report the case of a patient with cutaneous sweat gland carcinoma with local recurrence and metastasis to the lung that was treated with surgical resection therapy and chemotherapy. The initial neoplasm was excised but biopsy was not performed. The tumor then recurred 7 years later, was re‑excised, biopsy was performed, and diagnosed as a low‑grade hidradenocarcinoma. We presented a very good result of chemotherapy in the treatment of this rare malignant disease. It demonstrates that adjunct chemotherapy is effective to control the condition of malignant sweat‑gland carcinomas patient. KEY WORDS: Chemotherapy, metastasis, recurrence, surgical resection, sweat gland carcinoma
INTRODUCTION Malignant sweat‑gland tumors are rare neoplasms originating from the epidermal sweat ducts. The tumor is often located on the head, neck, and perineum as an asymptomatic tumor in the early stage. It started as a slow‑growing benign tumor with a rare incidence of distant metastases. Due to its low incidence and high rate of local recurrence, classical standard of diagnosis and therapy remains to be elucidative. CASE REPORT A 35‑year‑old woman presented with a long history of right‑shoulder cutaneous lesion. In the early 2003s, the patient found an exospheric mass with pain on the right shoulder and gradually developed. The mass was excised without biopsy in a local hospital. In 2010s, the patient suffered a second surgery because of the local recurrence. Biopsy suggested it was a low‑grade hidradenocarcinoma. Owning to the economic reason, the sufferer did not receive any treatment. The patient again noticed a small tumor and the tumor had progressively enlarged on the right shoulder in the area of the previous operation site after the primary operation [Figure 1]. Aggressive excision was performed on the patient. After the pathological review, the final diagnosis was low‑grade hidradenocarcinoma [Figure 2]. The patient complained of chest distress and her trouble became aggravated in July, 2011. The chest computed tomography (CT) in July 2011 revealed multiple lung metastases [Figure 3a]. After that, the patient chose to accept six cycles of chemotherapy with cisplatin plus vinorelbine (NP) 390
protocol (vinorelbine 40 mg IV D1, 8 and Cisplatin 30 mg IV D2‑5, repeat every 21 days). There were remissions of clinical symptoms and lesion of lung during the treatment [Figure 3b]. The patient appears to have stable disease at the end of six cycles of chemotherapy [Figure 3c]. DISCUSSION Sweat‑gland carcinoma affects both sexes equally in the fifth to sixth decade of life and usually shows as a solitary, firm nodule positioned on the head, neck, or more rarely, other sites. We present a case of primary sweat‑gland carcinoma of the skin in the right shoulder. It is difficult to make a proper diagnosis since this type of tumor is characterized by varying clinical appearance and histological features. The advantage of diagnosis is that this disease is easy to detect early since it arise from the surface of the body. However, considerable number of disadvantages increased the difficulty of diagnosis. First, a benign tumor must be eliminated in the differential diagnosis since it is a slow‑growing carcinoma. Second, when a node is recognized in the predilection site such as head, neck, and perineum, the possible inspection methods include ultrasound, CT, and biopsy must be carried out. Finally, due to the limited amount of literature and cases documented, it is important to have a basic knowledge of this disease process as a differential diagnosis when dealing with cutaneous lesions.
Xiao-Xia Wang, Hai-Yan Wang1, Jun-NianZheng1, Jian-Chao Sui Department of Clinical Oncology, Wendeng Central Hospital of Weihai, The Affiliated Hospital of Weifang Medical College,Weihai, 1 The Affiliated Hospital of Xuzhou Medical College, Xuzhou, China For correspondence: Prof. Jian-Chao Sui, Wendeng Central Hospital of Weihai, The Affiliated Hospital of Weifang Medical College,Weihai 264400, China.3 East Mi-shan Road, Weihai, Shandong, P.R.China. E-mail: jianchaosui @163.com
Access this article online Website: www.cancerjournal.net DOI: 10.4103/0973-1482.136667 PMID: *** Quick Response Code:
Therapy consists mainly of surgical excision, chemotherapy, and radiation.[1‑3] Surgery is the first Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Wang, et al.: Sweet gland carcinoma
compared to conventional surgical excision. The use of Mohs’ micrographic surgery was proposed by Wildemore recently in order to decrease the recurrence rate of this type of tumor.[7] Without therapy, the course of the disease is very slow and long duration, but multiple recurrences have been described. The role of radiation therapy in sweat‑gland carcinoma has changed from radio‑resistance in earlier reports[8] to good local control in recent studies[9] with technological advances in radiation therapy over the years. However, it still needs to be elucidated and hopefully, future studies will provide a better understanding of the radiation therapy.
Figure 1: The patient again noticed several small tumor and the tumor had progressively enlarged on the right shoulder in the area of the previous operation site in 2010s
a
b
Figure 2: (a) The definitive histopathology showed small cellular nests of adenocarcinoma lying in pools of extracellular mucin that were separated by fibrocollagenous septae with involvement of underlying muscle. There was no evidence of lymphatic invasion (H and E, ×10), (b) H and E, ×20
The role of chemotherapy in sweat‑gland carcinoma remains unclear. In one report, no response was observed in four cases of pediatric Eccrine porocarcinoma (EPC) treated with a combination of 5‑fluorouracil, doxorubicin, and cyclophosphamide after 1 year of therapy.[10] In the case of our patient, she initially suffered a local excision. We recommend that the disease should initially be treated with surgical excision with broad tumor margins and regional lymph node dissection, which would decrease the recurrence rate and distant metastasis of this type of tumor. The tumor in our patient had already spread to the lungs at the time of initial diagnosis in our department. The patient was, therefore, treated with chemotherapy which consisted of six cycles of Navelbine and cisplatin. After the combination chemotherapy, there were no significant changes in the size and number of primary and metastasis lesions. Our case demonstrates that aggressive surgery with adjunct chemotherapy is effective to control the condition of patient. Given the history of our case, cutaneous sweat‑gland carcinoma with local recurrence and metastasis to the lung have a good response to chemotherapy. It is necessary to carry out a detailed systematic examination, such as whole‑body CT and RI in the case of local recurrence and lymph‑node metastasis. ACKNOWLEDGMENTS
a
b
This project is supported by grants from the Program for New Century Excellent Talents in University (NCET‑08‑0700).
REFERENCES
c Figure 3: (a)The computed tomography of July 2011 revealed multiple lung metastases, (b)The computed tomography of December 2011 revealed remissions of lung lesion, (c)The computed tomography of April 2012 displayed the disease was stable
choice for management of such a sweat‑gland tumor since this type of carcinoma typically features a poor response to chemotherapy and radiotherapy.[1] Recent studies suggest that Mohs’ micrographic surgery is the method of choice[4‑6]
1. Chintamani, Sharma R, Badran R, Singhal V, Saxena S, Bansal A. Metastatic sweat gland adenocarcinoma: A clinico‑pathological dilemma. World J Surg Oncol 2003;1:13. 2. Dahill SW, Seywright M. Synchronous occurrence of cutaneous lymphadenoma and syringoid eccrine carcinoma in a single patient. Histopathology 1998;33:89‑90. 3. Ramos D, Monteagudo C, Carda C, Montesinos E, Ferrer J, Peydro‑Olaya A. Clear cell syringoid carcinoma: An ultrastructural and immunohistochemical study. Am J Dermatopathol 2000;22:60‑4. 4. Evans AT, Parham DM, Van Niekerk LJ. Metastasising eccrine syringomatous carcinoma. Histopathology 1995;26:185‑7. 5. McKee PH, Fletcher CD, Rasbridge SA. The enigmatic eccrine epithelioma (eccrine syringomatous carcinoma) Am J Dermatopathol 1990;12:552‑61.
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
391
Wang, et al.: Sweet gland carcinoma
6. Cottel WI. Eccrine epithelioma: Case report. J Dermatol Surg Oncol 1982;8:610‑1. 7. Wildemore JK, Lee JB, Humphreys TR. Mohs surgery for malignant eccrine neoplasms. Dermatol Surg 2004;30:1574‑9. 8. el‑Domeiri AA, Brasfield RD, Huvos AG, Strong EW. Sweat gland carcinoma: A clinico‑pathologic study of 83 patients. Ann Surg 1971;173:270‑4. 9. Shiohara J, Koga H, Uhara H, Takata M, Saida T. Eccrine porocarcinoma: Clinical and pathological studies of 12 cases. J Dermatol 2007;34:516‑22.
392
10. Chow CW, Campbell PE, Burry AF. Sweat gland carcinomas in children. Cancer 1984;53:1222‑7.
Cite this article as: Wang X, Wang H, Zheng J, Sui J. Primary cutaneous sweat gland carcinoma. J Can Res Ther 2014;10:390-2. Source of Support: Program for New Century Excellent Talents in University (NCET‑08‑0700), Conflict of Interest: None declared.
Journal of Cancer Research and Therapeutics - April-June 2014 - Volume 10 - Issue 2
Copyright of Journal of Cancer Research & Therapeutics is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
