阴茎上皮样肉瘤1例报告并文献复习
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摘要
目的:探讨阴茎上皮样肉瘤的临床、病理特征、诊断、治疗方法及预后。方法:回顾性分析1例阴茎上皮样肉瘤患者的临床资料及诊治流程并结合文献复习。结果:69岁男性患者,因发现阴茎根部进行性增大包块3月入院就诊。下腹部CT示阴茎根部大小5.9 cm×5.6 cm×6.5 cm软组织肿块,倾向恶性肿瘤,肉瘤可能性大。行阴茎根部肿瘤切除术,术后病理诊断:镜下见肿瘤细胞呈圆形或卵圆形,染色深浅不一,部分呈空泡状,核仁明显。免疫组化染色结果:CK广谱(-),vimentin(部分+),EMA(大部分+), CD34(血管+),CD31(血管+),CD99(+),S-100(-),Bcl-2(-),HMB45(-),SMA(-),AFP(-)。术后患者恢复可出院,未行进一步治疗。术后7月患者发现左侧腹股沟出现进行性增大包块,术后10月患者复查盆腔CT示阴茎根部上份盆壁、左侧睾丸及左侧腹股沟区见多发软组织肿块,最大者位于左侧腹股沟区,大小约7.6 cm×6.8 cm,考虑局部复发并左侧睾丸、腹股沟转移,患者自行放弃行进一步治疗,最后随访日期为2018年1月。结论:阴茎上皮样肉瘤是一种罕见的阴茎间质恶性肿瘤,确诊依赖于病理学及特异性免疫组化染色结果,手术切除是其主要治疗方法,术后是否行放化疗治疗尚存争议,其易局部复发及区域淋巴结转移,总体预后较差。
Objective:To investigate the clinical,pathological features,diagnosis,treatment and prognosis of penile epithelioid sarcoma.Methods:This is A retrospective analysis of a case of penile epithelioid sarcoma in clinical data and diagnosis and treatment process combined with literature review.Results:69-year-old male patient,due to the founding of the penis root progressive enlargement mass was admitted to the hospital for 3 months.The lower abdomen CT showed a soft tissue mass of 5.9 cm×5.6 cm×6.5 cm in the base of the penis,which tended to be a malignant tumor,and the possibility of sarcoma was large.Resection of penile root tumor and postoperative pathological diagnosis microscopically showed the tumor cells were round or oval,with different staining depths,some vacuoles,and obvious nucleoli.Immunohistochemical staining results showed CK broad spectrum(-),vimentin(part+),EMA(most +),CD34(vessel+),CD31(vessel+),CD99(+),S-100(-),Bcl-2(-),HMB45(-),SMA(-),AFP(-).The patient recovered after surgery and was discharged without further treatment.After 7 months,the patient found a progressive enlargement of the mass in the left groin.After 10 months,the patient underwent pelvic CT to show the upper pelvic wall of the penis,the left testis and the left groin area with multiple soft tissue masses.The largest one was left.The lateral groin area was about 7.6 cm×6.8 cm.Considering local recurrence and left testicular and groin metastasis,the patient gave up treatment for further treatment.The final follow-up date was January 2018.Conclusion:Penile epithelioid sarcoma is a rare malignant tumor of the penis.The diagnosis depends on the pathological and specific immunohistochemical staining results.Surgical resection is the main treatment.There is controversy whether radiotherapy and chemotherapywe received after operation.It is easy to local recurrence and regional lymph node metastasis,and the overall prognosis is poor.
Objective:To investigate the clinical,pathological features,diagnosis,treatment and prognosis of penile epithelioid sarcoma.Methods:This is A retrospective analysis of a case of penile epithelioid sarcoma in clinical data and diagnosis and treatment process combined with literature review.Results:69-year-old male patient,due to the founding of the penis root progressive enlargement mass was admitted to the hospital for 3 months.The lower abdomen CT showed a soft tissue mass of 5.9 cm×5.6 cm×6.5 cm in the base of the penis,which tended to be a malignant tumor,and the possibility of sarcoma was large.Resection of penile root tumor and postoperative pathological diagnosis microscopically showed the tumor cells were round or oval,with different staining depths,some vacuoles,and obvious nucleoli.Immunohistochemical staining results showed CK broad spectrum(-),vimentin(part+),EMA(most +),CD34(vessel+),CD31(vessel+),CD99(+),S-100(-),Bcl-2(-),HMB45(-),SMA(-),AFP(-).The patient recovered after surgery and was discharged without further treatment.After 7 months,the patient found a progressive enlargement of the mass in the left groin.After 10 months,the patient underwent pelvic CT to show the upper pelvic wall of the penis,the left testis and the left groin area with multiple soft tissue masses.The largest one was left.The lateral groin area was about 7.6 cm×6.8 cm.Considering local recurrence and left testicular and groin metastasis,the patient gave up treatment for further treatment.The final follow-up date was January 2018.Conclusion:Penile epithelioid sarcoma is a rare malignant tumor of the penis.The diagnosis depends on the pathological and specific immunohistochemical staining results.Surgical resection is the main treatment.There is controversy whether radiotherapy and chemotherapywe received after operation.It is easy to local recurrence and regional lymph node metastasis,and the overall prognosis is poor.
引文
[1] Velazquez EF,Melamed J,Barreto JE,et al.Sarcomatoid carcinoma of the penis:A clinicopathologic study of 15 cases[J].American Journal of Surgical Pathology,2005,29(9):1152.
[2] Hornick JL,Dal CP,Fletcher CD.Loss of INI1 expression is characteristic of both conventional and proximal-type epithelioid sarcoma[J].American Journal of Surgical Pathology,2009,33(4):542-550.
[3] Tao X,Chen LY,Chen H,et al.Clinicopathological and immunohistochemical analysis of epithelioid sarcoma[J].Journal of Fujian Medical University,2016,50(2):98-102.[陶璇,陈林莺,陈虹,等.上皮样肉瘤的临床病理及免疫组织化学分析[J].福建医科大学学报,2016,50(2):98-102.]
[4] Rossi G,Ferrari G,Longo L,et al.Epithelioid srcoma of the penis:A case report and review of the literature[J].Pathology International,2010,50(7):579-585.
[5] Nishibaba R,Higashi Y,Goto Y,et al.Epithelioid sarcoma with multiple lesions on the left arm:A case report[J].Journal of Medical Case Reports,2016,10(1):295.
[6] Hauck EW,Schmelz HU,Diemer T,et al.Epithelioid sarcoma of the penis—a rare differential diagnosis of Peyronie's disease[J].International Journal of Impotence Research,2003,15(5):378-382.
[7] Usta MF,Adams DM,Zhang J,et al.Penile epithelioid sarcoma and the case for a histopathological diagnosis in Peyronie's disease[J].Bju International,2015,91(6):519-521.
[8] Yu H,Wu B,Liu H,et al.Epithelioid sarcoma:CT and MRI findings with corresponding pathological basis[J].Journal of Clinical Radiology,2016,35(6):964-968.[余洪,吴博,刘衡,等.上皮样肉瘤CT和MRI表现及其病理基础[J].临床放射学杂志,2016,35(6):964-968.]
[9] Yang Z,Xia Y,Xiao M,et al.Epithelioid sarcoma:A case report[J].Journal of Clinical Dermatology,2014,43(2):92-94.[杨珍,夏颖,肖敏,等.上皮样肉瘤[J].临床皮肤科杂志,2014,43(2):92-94.]
[10] Alikhan MB,Pease G,Watkin W,et al.Primary epithelioid sarcoma of the kidney and adrenal gland:Report of 2 cases with immunohistochemical and molecular cytogenetic studies[J].Human Pathology,2017,61:158-163.
[11] Colombo F,Franceschelli A,Schiavina R,et al.Epithelioid sarcoma of the penis:A case report[J].Journal of Sexual Medicine,2013,10(11):2871-2874.
[12] Thway K,Jones RL,Noujaim J,et al.Epithelioid sarcoma:Diagnostic features and genetics[J].Advances in Anatomic Pathology,2016,23(1):41-49.
[13] Pan ZH,Kong DJ.A case of MRI findings and pathology review of epithelioid sarcoma[J].Journal of Clinical Radiology,2013,32(3):360-361.[潘志华,孔德军.上皮样肉瘤MRI表现及病理回顾一例[J].临床放射学杂志,2013,32(3):360-361.]
[14] Zhu XW,Guo JP,Chen H,et al.Deep sarcoma of the penis:A report of 2 cases and review of the literature[J].National Journal of Andrology,2007,13(10):915-917.[朱选文,郭君平,陈晖,等.阴茎海绵体肉瘤(附2例报告并文献复习)[J].中华男科学杂志,2007,13(10):915-917.]
[15] Sundaram A,Elangovan A,Rajwanshi A,et al.Proximal-type Epithelioid Sarcoma of Vulva:Cytopathological diagnosis of a rare neoplasm[J].Cytopathology,2018,29(5):471-473.
[16] Outani H,Imura Y,Tanaka T,et al.Clinical outcomes of patients with epithelioid sarcomas:Impact and management of nodal metastasis[J].International Journal of Clinical Oncology,2018,23(1):181-188.
[17] Kim C,Yoo KH,Kim MH,et al.Different subtypes of epithelioid sarcoma and their clinical implication:Long-term multi-institutional experience with a rare sarcoma[J].Apmis,2017,125(3):223-229.
[18] Jones RL,Constantinidou A,Olmos D,et al.Role of palliative chemotherapy in advanced epithelioid sarcoma[J].Am J Clin Oncol,2012,35(4):351-357.
[19] Echchaoui A,Sadrati Y,Elbir Y,et al.Proximal-type epithelioid sarcoma:A new case report and literature review[J].Pan Afr Med J,2016,24:238.
[20] Sambri A,Bianchi G,Cevolani L,et al.Can radical margins improve prognosis in primary and localized epithelioid sarcoma of the extremities[J]?Journal of Surgical Oncology,2017,117(6):1204-1210.
[21] Pradhan A,Grimer RJ,Abudu A,et al.Epithelioid sarcomas:How important is loco-regional control[J]?Eur J Surg Oncol,2017,43(9):1746-1752.
[22] Lowentrit B,Parsons JK,Argani P,et al.Pediatric epithelioid sarcoma of the penis[J].The Journal of Urology,2004,172(1):296-297.
[23] De Visscher SA,Van Ginkel RJ,Wobbes T,et al.Epithelioid sarcoma:Still an only surgically curable disease[J].Cancer,2010,107(3):606-612.
[24] Moses KA,Sfakianos JP,Winer A,et al.Non-squamous cell carcinoma of the penis:Single-center,15-year experience[J].World Journal of Urology,2013,32(5):1347-1353.
[25] Siontis BL,Chugh R,Schuetze SM.The potential of emerging therapeutics for epithelioid sarcoma[J].Expert Opinion on Orphan Drugs,2017,5(5):983-989.
[2] Hornick JL,Dal CP,Fletcher CD.Loss of INI1 expression is characteristic of both conventional and proximal-type epithelioid sarcoma[J].American Journal of Surgical Pathology,2009,33(4):542-550.
[3] Tao X,Chen LY,Chen H,et al.Clinicopathological and immunohistochemical analysis of epithelioid sarcoma[J].Journal of Fujian Medical University,2016,50(2):98-102.[陶璇,陈林莺,陈虹,等.上皮样肉瘤的临床病理及免疫组织化学分析[J].福建医科大学学报,2016,50(2):98-102.]
[4] Rossi G,Ferrari G,Longo L,et al.Epithelioid srcoma of the penis:A case report and review of the literature[J].Pathology International,2010,50(7):579-585.
[5] Nishibaba R,Higashi Y,Goto Y,et al.Epithelioid sarcoma with multiple lesions on the left arm:A case report[J].Journal of Medical Case Reports,2016,10(1):295.
[6] Hauck EW,Schmelz HU,Diemer T,et al.Epithelioid sarcoma of the penis—a rare differential diagnosis of Peyronie's disease[J].International Journal of Impotence Research,2003,15(5):378-382.
[7] Usta MF,Adams DM,Zhang J,et al.Penile epithelioid sarcoma and the case for a histopathological diagnosis in Peyronie's disease[J].Bju International,2015,91(6):519-521.
[8] Yu H,Wu B,Liu H,et al.Epithelioid sarcoma:CT and MRI findings with corresponding pathological basis[J].Journal of Clinical Radiology,2016,35(6):964-968.[余洪,吴博,刘衡,等.上皮样肉瘤CT和MRI表现及其病理基础[J].临床放射学杂志,2016,35(6):964-968.]
[9] Yang Z,Xia Y,Xiao M,et al.Epithelioid sarcoma:A case report[J].Journal of Clinical Dermatology,2014,43(2):92-94.[杨珍,夏颖,肖敏,等.上皮样肉瘤[J].临床皮肤科杂志,2014,43(2):92-94.]
[10] Alikhan MB,Pease G,Watkin W,et al.Primary epithelioid sarcoma of the kidney and adrenal gland:Report of 2 cases with immunohistochemical and molecular cytogenetic studies[J].Human Pathology,2017,61:158-163.
[11] Colombo F,Franceschelli A,Schiavina R,et al.Epithelioid sarcoma of the penis:A case report[J].Journal of Sexual Medicine,2013,10(11):2871-2874.
[12] Thway K,Jones RL,Noujaim J,et al.Epithelioid sarcoma:Diagnostic features and genetics[J].Advances in Anatomic Pathology,2016,23(1):41-49.
[13] Pan ZH,Kong DJ.A case of MRI findings and pathology review of epithelioid sarcoma[J].Journal of Clinical Radiology,2013,32(3):360-361.[潘志华,孔德军.上皮样肉瘤MRI表现及病理回顾一例[J].临床放射学杂志,2013,32(3):360-361.]
[14] Zhu XW,Guo JP,Chen H,et al.Deep sarcoma of the penis:A report of 2 cases and review of the literature[J].National Journal of Andrology,2007,13(10):915-917.[朱选文,郭君平,陈晖,等.阴茎海绵体肉瘤(附2例报告并文献复习)[J].中华男科学杂志,2007,13(10):915-917.]
[15] Sundaram A,Elangovan A,Rajwanshi A,et al.Proximal-type Epithelioid Sarcoma of Vulva:Cytopathological diagnosis of a rare neoplasm[J].Cytopathology,2018,29(5):471-473.
[16] Outani H,Imura Y,Tanaka T,et al.Clinical outcomes of patients with epithelioid sarcomas:Impact and management of nodal metastasis[J].International Journal of Clinical Oncology,2018,23(1):181-188.
[17] Kim C,Yoo KH,Kim MH,et al.Different subtypes of epithelioid sarcoma and their clinical implication:Long-term multi-institutional experience with a rare sarcoma[J].Apmis,2017,125(3):223-229.
[18] Jones RL,Constantinidou A,Olmos D,et al.Role of palliative chemotherapy in advanced epithelioid sarcoma[J].Am J Clin Oncol,2012,35(4):351-357.
[19] Echchaoui A,Sadrati Y,Elbir Y,et al.Proximal-type epithelioid sarcoma:A new case report and literature review[J].Pan Afr Med J,2016,24:238.
[20] Sambri A,Bianchi G,Cevolani L,et al.Can radical margins improve prognosis in primary and localized epithelioid sarcoma of the extremities[J]?Journal of Surgical Oncology,2017,117(6):1204-1210.
[21] Pradhan A,Grimer RJ,Abudu A,et al.Epithelioid sarcomas:How important is loco-regional control[J]?Eur J Surg Oncol,2017,43(9):1746-1752.
[22] Lowentrit B,Parsons JK,Argani P,et al.Pediatric epithelioid sarcoma of the penis[J].The Journal of Urology,2004,172(1):296-297.
[23] De Visscher SA,Van Ginkel RJ,Wobbes T,et al.Epithelioid sarcoma:Still an only surgically curable disease[J].Cancer,2010,107(3):606-612.
[24] Moses KA,Sfakianos JP,Winer A,et al.Non-squamous cell carcinoma of the penis:Single-center,15-year experience[J].World Journal of Urology,2013,32(5):1347-1353.
[25] Siontis BL,Chugh R,Schuetze SM.The potential of emerging therapeutics for epithelioid sarcoma[J].Expert Opinion on Orphan Drugs,2017,5(5):983-989.