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Primary synovial sarcoma of the prostate metastatic to the liver and lung: a case report.
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PMID:  24969223     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
Primary synovial sarcoma of the prostate is an uncommon malignant tumor. There are few cases reported in the English medical literature to date. Here, we present a case of 22-year-old man with primary synovial sarcoma of the prostate metastatic to the liver and lung. To our knowledge, only six reports of synovial sarcoma involving the prostate have been previously published. We also reviewed the previous treatments and prognoses in previous case reports and evaluate the proper treatment for this disease.
Authors:
Qi Zhang; Huiju Wang; Ligang Ren; Xiaolong Qi; Feng Liu; Dahong Zhang
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Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2014-6-27
Journal Detail:
Title:  World journal of surgical oncology     Volume:  12     ISSN:  1477-7819     ISO Abbreviation:  World J Surg Oncol     Publication Date:  2014 Jun 
Date Detail:
Created Date:  2014-6-27     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101170544     Medline TA:  World J Surg Oncol     Country:  -    
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Languages:  ENG     Pagination:  194     Citation Subset:  -    
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Journal ID (nlm-ta): World J Surg Oncol
Journal ID (iso-abbrev): World J Surg Oncol
ISSN: 1477-7819
Publisher: BioMed Central
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Copyright © 2014 Zhang et al.; licensee BioMed Central Ltd.
open-access:
Received Day: 21 Month: 6 Year: 2013
Accepted Day: 12 Month: 6 Year: 2014
collection publication date: Year: 2014
Electronic publication date: Day: 27 Month: 6 Year: 2014
Volume: 12First Page: 194 Last Page: 194
PubMed Id: 24969223
ID: 4086276
Publisher Id: 1477-7819-12-194
DOI: 10.1186/1477-7819-12-194

Primary synovial sarcoma of the prostate metastatic to the liver and lung: a case report
Qi Zhang1 Email: clinic@zju.edu.cn
Huiju Wang2 Email: maozujiehospital@126.com
Ligang Ren1 Email: PAPERMAND@gmail.com
Xiaolong Qi1 Email: yezaiyuan2010@126.com
Feng Liu1 Email: liufeng20102011@126.com
Dahong Zhang1 Email: urology@zju.edu.cn
1Department of Urology, Zhejiang Provincial People’s Hospital, Hangzhou, China
2Key Laboratory of Gastroenterology of Zhejiang Province, Hangzhou, China

Background

Primary synovial sarcomas have been described at unusual sites, including the heart, oesophagus, larynx, pleura, lung, kidney, prostate, liver, abdominal wall, retroperitoneum and gastrointestinal tract; involvement of the genitourinary tract is exceedingly rare [1-3]. Synovial sarcoma arising primarily from the prostate is a rare occurrence, with only six previously reported cases. Synovial sarcoma of the prostate is usually identified at a late stage because of its nonspecific presentation. The purpose of our article is to add an additional case to the literature, to review the literature, and to formulate treatment recommendations. We describe a patient presenting with primary synovial sarcoma of the prostate metastatic to the liver and lung.


Case presentation

A 22-year-old man presented elsewhere with lower urinary tract symptoms and was referred to us for further evaluation because of urinary retention. No particular family history or occupational hazard was elicited. Our patient complained of dysuria, urinary frequency and nocturia five to ten times per night before urinary retention. On digital rectal examination, a large prostatic mass was palpable, and the surface was smooth. His serum prostate-specific antigen was 1.2 ng/ml (normal 4 or less). Pelvic computerized tomography (CT) imaging (Figure 1) and magnetic resonance imaging (MRI) (Figure 2) demonstrated a 14-cm mass that appeared to originate in the prostatic fascia. Another 8.5-cm mass was found in the right groin area. CT and chest radiography showed evidence of liver and lung metastasis (Figure 3). The patient’s age was younger than the common age range of patients for the commonest types of prostatic adenocarcinoma. Ultrasound-guided transrectal needle biopsy and pathologic examination revealed synovial sarcoma. The pathologic findings were consistent with the diagnosis of synovial sarcoma arising from the prostate (Figure 4). On immunohistochemical staining, the tumor cells were positive for vimentin and CD99, but negative for alpha smooth muscle actin, desmin, and S-100 protein. The presence of SYT-SSX fusion transcript was confirmed by RT-PCR using genomic DNA isolated from paraffin blocks. The patient refused to receive any therapeutic schedule including irradiation、chemotherapy and radical prostatectomy. His disease progression was rapid. At three months of follow-up, the patient had developed multiple lung metastases and subsequently died of respiratory failure.


Discussion

Synovial sarcoma is a clinically and histomorphologically well-defined soft tissue tumor that is extremely uncommon, with about 90% synovial sarcomas occurring on the extremities. In most patients with this disease, distant pulmonary metastases develop after primary tumor surgery. In the urinary system, most synovial sarcomas have been reported in the kidney [4]. Primary synovial sarcomas involving the prostate are exceedingly rare, with only a handful of cases reported in the English medical literature to date (Table 1) [5,6].

Primary prostatic sarcomas are rare tumors and most patients present with urinary obstruction, as did our patient. The pathologic findings were consistent with the diagnosis of synovial sarcoma arising from the prostate. In common with other synovial sarcomas, the immunohistochemical staining demonstrated some typical findings of synovial sarcoma: the tumor cells were positive for vimentin and CD99, but negative for CD34, Bcl-2, alpha smooth muscle actin, desmin, and S-100 protein. Finally, the presence of SYT-SSX fusion transcript confirmed the diagnosis [7,8].

Several clinicopathologic features were particular for primary synovial sarcoma of the prostate. As compared with the most common malignant tumor of the prostate adenocarcinoma, prostatic sarcoma occurs in relatively younger patients [9]. According to the previous reports, the age of diagnosis for these patients was 37 to 63 years. The age of our patient (22 years old) was younger than this range. Most patients with prostatic sarcoma present with symptoms of bladder outlet obstruction. Our patient complained of dysuria, urinary frequency and nocturia before urinary retention. The massive tumor may have been responsible for this symptom. The PSA level of prostatic adenocarcinoma is often elevated. It has been reported that the PSA level may not rise in patients with prostatic sarcoma, due to the non-epithelial origin of prostate sarcoma [9]. PSA levels in previous patients with prostatic synovial sarcoma ranged from 0.5 ng/ml to 0.9 ng/ml. Our patient had a PSA level of 1.20 ng/ml, which was still within normal range. Prostatic synovial sarcoma image findings are not well reported. In one case report by Shirakawa, T2 weighted MRI revealed a high signal mass originating in the prostatic fascia [5]. The imaging findings of primary synovial sarcoma of the prostate are not unique and pathologic examination is the only way to make a definite diagnosis.

The survival of persons with primary prostatic sarcomas is poor, with most patients dying within a few months after diagnosis and surgery. Distant pulmonary metastases are found in most patients with this disease, whether the patient has undergone surgery or not. In our case, the tumor had metastasis to the liver and lung. Treatment for this sarcoma is uncertain due to extremely limited experience but it appears that aggressive resection has to be considered for the treatment of locally confined synovial sarcoma [10]. The role of adjuvant chemotherapy and/or radiation remains unknown; however, it is strongly recommended because of the poor prognosis. Documentation of further cases is needed to establish appropriate therapy.


Conclusion

In conclusion, we demonstrate a case of primary prostatic synovial sarcoma. Due to the paucity of cases of synovial sarcoma involving the prostate, protocols for definitive treatment are not well defined. Treatment for this tumor is uncertain due to extremely limited experience, but it appears that aggressive resection should be considered as the standard therapy. The role of adjuvant chemotherapy and radiation also remains unknown. Reporting of future cases is necessary to establish appropriate therapeutic guidelines for this disease.


Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Abbreviations

CT: computerized tomography; MRI: magnetic resonance imaging; RT-PCR: reverse transcription-polymerase chain reaction; DNA: Desoxvribose Nucleic Acid.


Competing interests

The authors declare that they have no competing interests.


Authors’ contributions

QZ, LR, HW, XQ, FL and DZ participated in the admission and the care of this patient. All the authors participated in the conception, the design, data collection and interpretation, manuscript preparation and literature search. All authors have read and approved the final manuscript.


Acknowledgements

We thank all of the pathologists at the Zhejiang Provincial People’s Hospital for their assistance with the pathologic analysis. Without their efforts, this article would not be possible. This work was supported by grant from the Medicine and Health Research Foundation of Zhejiang Province (Grant Number 2013KYB018) and Natural Science Foundation of Zhejiang Province (Grant Number LQ13H050001).


References
Porter HJ 2nd,Damjanov I,Arnold P,Thrasher JB,Synovial sarcoma metastatic to the penis and prostateJ UrolYear: 200112260510.1016/S0022-5347(05)65996-411458080
Chang JM,Lee HJ,Lee SE,Byun SS,Choe GY,Kim SH,Seong CK,Kim SH,Pictorial review: unusual tumours involving the prostate: radiological-pathological findingsBr J RadiolYear: 20081297190791510.1259/bjr/6829477518662962
Varma T,Adegboyega P,Primary cardiac synovial sarcomaArch Pathol Lab MedYear: 201212445445810.5858/arpa.2011-0008-RS22458908
Kim DH,Sohn JH,Lee MC,Lee G,Yoon GS,Hashimoto H,Sonobe H,Ro JY,Primary synovial sarcoma of the kidneyAm J Surg PatholYear: 20001281097110410.1097/00000478-200008000-0000710935650
Shirakawa T,Fujisawa M,Gotoh A,Okada H,Arakawa S,Kamidono S,Complete resection of synovial sarcoma of prostatic fasciaUrologyYear: 200312364412639671
Pan CC,Chang YH,Primary synovial sarcoma of the prostateHistopathologyYear: 200612332132310.1111/j.1365-2559.2005.02236.x16430486
Iwasaki H,Ishiguro M,Ohjimi Y,Ikegami H,Takeuchi T,Kikuchi M,Kaneko Y,Ariyoshi A,Synovial sarcoma of the prostate with t (X;18)(p11.2;q11.2)Am J Surg PatholYear: 199912222022610.1097/00000478-199902000-000129989850
Jun L,Ke S,Zhaoming W,Linjie X,Xinru Y,Primary synovial sarcoma of the prostate: report of 2 cases and literature reviewInt J Surg PatholYear: 200812332933410.1177/106689690730957518387992
Sexton WJ,Lance RE,Reyes AO,Pisters PW,Tu SM,Pisters LL,Adult prostate sarcoma: the M D. Anderson cancer center experienceJ UrolYear: 200112252152510.1016/S0022-5347(05)65974-511458058
Williams DH,Hua VN,Chowdhry AA,Laskin WB,Kalapurakal JA,Dumanian GA,Dalton DP,Synovial sarcoma of the prostateJ UrolYear: 2004126 Pt 1237615126828

Figures

[Figure ID: F1]
Figure 1 

Enhanced pelvic computed tomography. (A) Evidence of liver metastasis. (B-F), Enhanced pelvic computed tomography revealed a 14-cm mass that appeared to originate in the prostatic fascia. An 8.5-cm mass was found on pelvic magnetic resonance imaging.



[Figure ID: F2]
Figure 2 

Enhanced pelvic magnetic resonance imaging. (A-C) Enhanced pelvic T2 weighted MR revealed a high signal mass originating in the prostatic fascia and an 8.5-cm mass was found in the right groin area.



[Figure ID: F3]
Figure 3 

Chest radiography. (A) Evidence of lung metastasis.



[Figure ID: F4]
Figure 4 

Pathologic analysis. (A) The pathologic findings of the tumor (H&E). (B) Immunohistochemical staining for vimentin in tumor tissue. (C) Immunohistochemical staining for CD99 in tumor tissue.



Tables
[TableWrap ID: T1] Table 1 

Clinicopathologic features, treatment, and prognosis of seven cases of primary synovial sarcoma of prostate


 
Cases
  Iwasaki Shirakawa Williams Pan Li first case Li second case This study
Age (years)
37
52
63
44
46
44
22
Clinical findings
Gross hematuria, dysuria, painful micturition
Urinary retention
Lower urinary tract symptoms
Lower urinary tract symptoms
Dysuria, painful micturition
Dysuria gradually
Dysuria, urinary frequency, nocturia, urinary retention
Serum PSA (ng/ml)
None
0.9
0.5
2.91
0.35
1.19
1.2
Extent of tumor
10-cm solid mass with necrosis, invading the prostatic urethra, seminal vesicles, retrovesical soft tissues
7-cm mass, originating in the prostatic fascia of the right lobe
8.5-cm mass extending from the mid-potion of the prostate inferiorly to the base of the penis
6-cm, well circumscribed, soft, heterogeneous mass in the right side of prostate
5.5-cm mass with partial necrosis extending to the pelvic soft tissue
12-cm mass extending to the bladder, the rectum and pelvic soft tissue, partial necrosis
14-cm mass that appeared to originate in the prostatic fascia. Another 8.5-cm mass was found in the right groin area
IHC results
VM+, EMA +
VM+
VM+, CK(CAM5.2)+, S100+, CK7+, CKAE1/3+,CAL+
VM+, CD99+, CK +, bcl-2+
VM+, CD99+, bcl-2+, E-CA+, CK/EMA +
VM+, CK+,CD99+, E-CA+, bcl-2+
VM+, CD99+
Treatment
Chemotherapy, radical prostatocystectomy
Radical prostatectomy, adjuvant chemotherapy
Irradiation with en bloc penectomy and pubectomy and ileal conduit urinary diversion
Radical prostatectomy
Radical prostatectomy, chemotherapy
The tumor could not be completely resected
None
Prognosis Died (32 months, tumor recurrence and metastasis to the lungs and liver) Alive (no recurrence or metastasis) Alive (no recurrence or metastasis) Alive (no recurrence and metastasis) Alive (no recurrence and metastasis) Died (eight months, tumor recurrence and metastasis to the costosternal junction and lung) Died (three months, multiple lung metastasis and respiratory failure


Article Categories:
  • Case Report

Keywords: Prostate, Synovial sarcoma, Metastasis.

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