A Talerman

C Bokemeyer

Chao-Fu Wang

D Cao

Ding-Wei Ye

G Rosti

GC Toner

GJ Bosl

H Robova

J Prim

J-P Tasu

JP Shah

K Mei

M Kebapci

P Albers

R Gupta

RM Bukowski

SB Saxman

Xu-Dong Yao

Y-J Hse

Ya-Ping Hong

World Journal of Surgical Oncology

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WORLD JOURNAL OF SURGICAL ONCOLOGY Yao et al. World Journal of Surgical Oncology 2012, 10:189http://www.wjso.com/content/10/1/189CASE REPORT Open AccessPrimary yolk sac tumor of seminal vesicle: a casereport and literature reviewXu-Dong Yao1*†, Ya-Ping Hong1,2†, Ding-Wei Ye1,2 and Chao-Fu Wang3AbstractBackground: Yolk sac tumor (endodermal sinus tumor) is a rare malignant germ cell tumor arising in the testis orovary. Extragonadal yolk sac tumor is even rarer and has only been described in case reports. Due to the rarity ofthe tumors, the appropriately optimal treatment remains unclear. We report a case of yolk sac tumor in theseminal vesicle.Case: A 38-year-old Asian male presented with gross hematuria and hemospermia. Transrectal ultrasound scanshowed a solid mass in the left seminal vesicle and the scrotal sonography showed no abnormalities. Bilateralseminal vesicles were resected, and histopathological examination showed a typical pattern of yolk sac tumor (YST).The patient responded poorly to comprehensive treatment of radiotherapy, chemotherapy and surgeries,developed systemic multiple metastases, and died of cachexia one and half years after diagnosis.Keywords: Yolk sac tumor, Seminal vesicle, ExtragonadalBackgroundYolk sac tumor (YST, also called endodermal sinustumor) most frequently occurs in the gonads and has apoor prognosis, if not treated aggressively [1].Extragonadal yolk sac tumor (YST) is rare, and the mostcommon sites of origin are, in order of frequency, the medi-astinum, the retroperitoneum, the sacrococcygeal regionand the pineal gland [2]. Due to the rarity of this tumor, thereported cases were treated by varied surgeries with orwithout adjuvant therapy. Thus, the optimal treatmentremains unclear. To add to the literature, we report a caseof yolk sac tumor originating in the left seminal vesicle.Case presentationA 38-year-old Asian male presented to his urologist withgross hematuria and hemospermia of a half-year dur-ation. The remainder of his physical examination, med-ical history and surgical history were unremarkable. Hehad no history of cryptorchidism, testicular swelling ortesticular atrophy, and no family history of testicularcancer. A transrectal ultrasound scan performed on the* Correspondence: yaoxudong67@sina.com†Equal contributors1Department of Urology, Fudan University Shanghai Cancer Center, 270Dong’an Road, Shanghai 200032, ChinaFull list of author information is available at the end of the article© 2012 Yao et al.; licensee BioMed Central LtdCommons Attribution License (http://creativecreproduction in any medium, provided the orday of presentation showed a solid mass of the left sem-inal vesicle. A puncture biopsy of the left seminal vesicleconfirmed diagnosis of germ cell tumor. No tumorouslesions were found in other sites. At presentation, levelsof serum tumor markers were obtained: α-fetoprotein(AFP) was found to be elevated to 1,740.9 ug/l (normalless than 10 ng/ml); serum β-human chorionic gonado-tropin (β-hCG) (normal less than 10 mIU/ml) andprostate-specific antigen (PSA) (normal less than 4.0 ng/ml) were within normal range. The patient was givenfour cycles of chemotherapy consisting of bleomycin (15mg D-2, 9, and 16), cisplatin (30 mg/m2 for four con-secutive days), and etoposide (100 mg/m2 for four con-secutive days). After the chemotherapy, serum AFP wasfound to be decreased to 58.09 ug/l, and the symptom ofhemospermia disappeared.The patient was then transferred to our hospital. Hehad normal testes on palpation. And a scrotal sonog-raphy was performed and revealed no abnormalities.Magnetic resonance imaging (MRI) reported an abnor-mal signal mass in the left seminal vesicle (Figure 1).Positron emission tomography/computed tomography(PET/CT) showed tumor activity in the left seminalvesicle (Figure 2). Extensive examination for metastases,including chest X-ray film and abdominal computedtomography (CT) showed no abnormalities. Resection of. This is an Open Access article distributed under the terms of the Creativeommons.org/licenses/by/2.0), which permits unrestricted use, distribution, andiginal work is properly cited.Figure 1 Magnetic resonance imaging (MRI): an abnormal signal mass in the left seminal vesicle.Yao et al. World Journal of Surgical Oncology 2012, 10:189 Page 2 of 5http://www.wjso.com/content/10/1/189the bilateral seminal vesicles was performed. The leftseminal vesicle was obviously swollen (4. × 3.0 × 2.5 cm)and was adhering widely to its surroundings. Theresected tumor was a solid encapsulated mass, and thecut surface showed cystic fish-like changes (Figure 3).Although the right seminal vesicle was slightly swollen,there was no sign of tumor involvement.Histological evaluation of the resected specimen exhib-ited a malignant tumor composed of neoplastic cells pro-liferating in a microcystic or reticular pattern of growth.The neoplastic cells had highly atypical middle- or large-sized nuclei, with Schiller-Duval bodies partially mimick-ing papillary structures (Figure 4). Immunohistochemicalstain for α-fetoprotein was positive (Figure 5). HistologicalFigure 2 Positron emission tomography/computedtomography (PET/CT). There was still tumor activity in the leftseminal vesicle.features of the resected specimen were interpreted as ayolk sac tumor. The cutting edge of the left seminal vesiclewas not involved. Plus, the right seminal vesicle was freeof the tumor. Therefore, we diagnosed his disease as ayolk sac tumor originating in the left seminal vesicle. Aftersurgery, serum AFP decreased to 29.7 ug/l, yet it increasedto 79.43 ug/l at four weeks. After the surgery, the patientreceived four cycles of chemotherapy consisting of bleo-mycin (30 U D-2, 9, and 16), cisplatin (30 mg/m2 for fiveconsecutive days), and etoposide (100 mg/m2 for five con-secutive days). On completion of the chemotherapy,serum AFP was found to have decreased to 53.24 ug/l andremained stable. However, at week 6 after chemotherapy,serum AFP again elevated to 184.9 ug/ml. Positron emis-sion tomography (PET) of the pelvis revealed lymph nodeFigure 3 The resected specimen with the tumor showingcapsulated appearance and cut surface cystic, fish-like changes.Figure 4 Histopathology of the resected specimen. A. The resected specimen was a malignant tumor composed of neoplastic cellsproliferating in a microcystic or reticular growth pattern. B. Neoplastic cells with highly atypical middle-or large-sized nuclei and Schiller-Duvalbodies partially mimicking papillary structures.Yao et al. World Journal of Surgical Oncology 2012, 10:189 Page 3 of 5http://www.wjso.com/content/10/1/189metastases in the region of the left iliac vessels. Thus, re-section of the pelvic lymph nodes was performed.Enlarged lymph nodes were not found in the areas of theexternal iliac, internal iliac and obturator. The left side ofthe prostate was fixed to the pelvic wall. Histologicalevaluation of the specimens exhibited proliferation in theleft obturator lymph tissues but showed no evidence oflymphatic metastasis. Fibrous connective tissue outsidethe bladder was infiltrated by lymphocytes.On the postoperative Day 3, adjunct radiotherapy wasintroduced, but the response was not good, with AFPremaining at a high level since then. The patient pre-sented rapid multiple systemic metastases and died ofcachexia one and half years after diagnosis.DiscussionYST is a malignant germ cell tumor that usually arisesin the gonads and is characteristically accompanied byan elevation of the serum α-fetoprotein level. YST israrely found extragonadally. Case reports have describedFigure 5 Immunohistochemical stain for alpha-fetoprotein waspositive.the occurrences of this entity in the pineal gland, headand neck, mediastinum, lung, stomach, liver, omentum,retroperitoneum, prostate, intrarenal, vagina, vulva, ura-chus and presacral regions [2-6].The typical morphologic structures of endodermalsinus pattern known as sinuses of Duval, Schiller-Duvalbodies, or glomerulus-like structures, superficially re-semble immature renal glomeruli [7]. The three sensitivediagnostic markers for yolk sac tumor are alpha-fetopro-tein, glypican-3 and SALL4 [3,7].The histogenesis of extragonadal YST remains contro-versial. These tumors appear to arise from germ cellsmisplaced during embryogenesis, and malignant trans-formation leads to primary germ cell tumors at extrago-nadal sites [8].Due to the rarity of the extragonadal YST, there is nodetailed description of its epidemiology and its clinicalfeatures in particular. Additionally, due to the particu-larly aggressive nature of YST, the information derivedfrom germ cell tumor (GCT) studies may have limitedapplicability. From the case reports we have, the age dis-tribution of extragonadal YST, similar to that of the tes-ticular tumors, appears to be bimodal with an early peakin early infancy and a later peak in adolescence. YSTmay grow to a large size with no or relatively few symp-toms, and the symptoms may vary due to origin sites.The majority of YSTs produce high levels of AFP, whichhelps in monitoring the response to the treatment. Inthe past, YSTs were nearly uniformly fatal, regardless ofthe primary location. However, the survival rate of YSTpatients has been significantly improved because of theapplication of Cisplatin-based multiagent chemotherapy[1-6,9].In our case, the patient had no history of swollen tes-tes or testicular atrophy and had normal testes on palpa-tion.Also, a scrotal sonography was performed andrevealed no abnormalities in either side of the testes.With spatial resolution boosted by a high-frequencyYao et al. World Journal of Surgical Oncology 2012, 10:189 Page 4 of 5http://www.wjso.com/content/10/1/189transducer, a testicular tumor may be nonpalpable butstill visible sonographically [10,11]. Sonographic findingsrange from echogenic foci or hypoechoic areas and het-erogeneous lesions to microlithiasis and macrocalcifica-tions [11,12]. It should be noted that normalsonographic findings were more frequent before 1990[11]. Therefore, we did not consider the diagnosis of theprimary testicular tumor regressed with metastasis inthe seminal vesicle. However, since the testes were notresected, we could not completely exclude this possibil-ity without histological evaluation.A correct diagnosis was made as late as half a yearafter the symptoms appeared due to the unspecificsymptoms (gross hematuria and hemospermia) and rar-ity of the site of extragonadal YST (the seminal vesicle).This may be one of the contributors to the poor progno-sis. From the clinical courses of this patient, we can seethat the patient’s general response to the treatment isnot very good as compared with recent reports. Jay P.Shah [13] reported a five-year survival rate of 65%. Thepossible reasons for the poor prognosis of our patientwere the advanced age, the race and the site of the ori-ginal tumor. The median age at the time of diagnosis ofYSTs is 18, occurrence after 30 is exceptional [14], andprognosis in the infancy cohort is better than that in theadolescent cohorts. The age of our patient (38) may be acontributor to the poor diagnosis. In Jay P. Shah’s report,African-Americans had poorer survival compared withCaucasians. Yu-juwei Hse [15] reported the treatmentresults in Chinese patents with nonseminomatous extra-gonadal germ cell tumor (NSEGCT) are not as good as inpatients in Western countries. Saxman et al. [16] reportedthe experience with chemotherapy in Indians withNSEGCT. Only 5 of 73 patients (7%) were long-term dis-ease-free survivors after chemotherapy. More investiga-tions need to be done to determine whether race is one ofthe contributors to the poor prognosis of EGCT. Severalreports showed that EGCT in mediastina has a worse out-come than EGCT in other sites [15-17]. To our know-ledge, there is no report about YST primarily arising inthe seminal vesicle. The seminal vesicle as the site of ori-gin contributes to the patient’s poor prognosis.ConclusionsWe present a rare case of yolk sac tumor originating inthe left seminal vesicle. Extragonadal YST is an aggres-sive germ cell tumor, and the treatment results are stillunsatisfactory. More investigations are warranted to de-fine the optimal treatment.ConsentWritten informed consent was obtained from thepatient’s elder brother for publication of this case reportand any accompanying images. A copy of the writtenconsent is available for review by the Editor-in-Chief ofthis journal.AbbreviationsYST: yolk sac tumor; AFP: α-fetoprotein; CT: computed tomography;MRI: magnetic resonance imaging; PSA: prostate-specific antigen; PET/CT: Positron emission tomography/computed tomography; GCT: germ celltumor; EGCT: extragonadal germ cell tumor; NSEGCT: nonseminomatousextragonadal germ cell tumor.Competing interestsThe authors declare that they have no competing interests.Authors’ contributionsXDY and HYP conceived the concept for the study, participated in draftingthe manuscript, and conducted a critical review. Both authors contributedequally to this work. DWY participated in revising the manuscript critically forimportant intellectual content. CFW prepared the histological figures. Allauthors read and approved the final manuscript.Author details1Department of Urology, Fudan University Shanghai Cancer Center, 270Dong’an Road, Shanghai 200032, China. 2Department of Oncology, ShanghaiMedical College, Fudan University, Shanghai, China. 3Department ofPathology, Fudan University Shanghai Cancer Center, Shanghai, China.Received: 12 June 2012 Accepted: 11 August 2012Published: 14 September 2012References1. Bosl GJ, Motzer RJ: Testicular germ-cell cancer. N Engl J Med 1997,337:242–253. Erratum in: N Engl J Med 1997, 337:1403.2. Toner GC, Geller NL, Lin SY, Bosl GJ: Extragonadal and poor risknonseminomatous germ cell tumors. Survival and prognostic features.Cancer 1991, 67:2049–2057.3. Mei K, Liu A, Allan RW, Wang P, Lane Z, Abel TW, Wei L, Cheng H, Guo S,Peng Y, Rakheja D, Wang M, Ma J, Rodriguez MM, Li J, Cao D: Diagnosticutility of SALL4 in primary germ cell tumors of the central nervoussystem: a study of 77 cases. Mod Pathol 2009, 22:1628–1636.4. Saxman SB, Nichols CR, Einhorn LH: Salvage chemotherapy in patientswith extragonadal nonseminomatous germ cell tumors: the IndianaUniversity experience. J Clin Oncol 1994, 12:1390–1393.5. Cao D, Liu A, Wang F, Allan RW, Mei K, Peng Y, Du J, Guo S, Abel TW, LaneZ, Ma J, Rodriguez M, Akhi S, Dehiya N, Li J: RNA-binding protein LIN28 isa marker for primary extragonadal germ cell tumors: animmunohistochemical study of 131 cases. Mod Pathol 2011, 24:288–296.6. Gupta R, Mathur SR, Arora VK, Sharma SG: Cytologic features ofextragonadal germ cell tumors: a study of 88 cases with aspirationcytology. Cancer 2008, 114:504–511.7. Talerman A: Germ cell tumors of the ovary. In Blaustein's Pathology of theFemale Genital Tract. 5th edition. Edited by Kurman RJ. New York: Springer-Verlag; 2002:967–1033.8. Albers P, Düsseldorf: 10th edition of the European Association of UrologyGuidelines. In Edited by Parsons KF. The European Association of Urology;2010.9. Bukowski RM, Wolf M, Kulander BG, Montie J, Crawford ED, Blumenstein B:Alternating combination chemotherapy in patients with extragonadalgerm cell tumors. A Southwest Oncology Group study. Cancer 1993,71:2631–2638.10. Prim J: Spontanheilung eines bösartigen wahrschein-lichchorionepitheliomatosen: gewaches im hoden. Arch Pathol Anat 1927,265:239–241.11. Tasu J-P, Faye N, Eschwege P, Rocher L, Bléry M: Imaging of burned-outtestis tumor: five new cases and review of the literature. J UltrasoundMed 2003, 22:515–521.12. Kebapci M, Can C, Isiksoy S, Aslan O, Oner U: Burned-out tumor of thetestis presenting as supraclavicular lymphadenopathy. Eur Radiol 2002,12:371–373.13. Shah JP, Kumar S, Bryant CS, Ali-Fehmi R, Malone JM Jr, Deppe G, Morris RT:A population-based analysis of 788 of yolk sac tumors: a comparison ofmales and females. Int J Cancer 2008, 123:2671–2675.Yao et al. World Journal of Surgical Oncology 2012, 10:189 Page 5 of 5http://www.wjso.com/content/10/1/18914. Robova H, Rob L, Hrehorcak M, Zoban P, Prusa R: Endodermal sinus tumordiagnosed in pregnancy: a case report. Int J Gynecol Cancer 2007,17:914–933.15. Hse Y-J, Pai L, Chen YC, Ho CL, Kao WY, Chao TY: Extragonadal germ celltumor in Taiwan: an analysis of treatment results of 59 patients. Cancer2002, 95:766–774.16. Bokemeyer C, Hartmann JT, Fossa SD, Droz JP, Schmol HJ, Horwich A, GerlA, Beyer J, Pont J, Kanz L, Nichols CR, Einhorn L: Extragonadal germ celltumors: relation to testicular neoplasia and management options. APMIS2003, 111:49–59. discussion 59–63.17. Rosti G, De Giorgi U, Wandt H, Lioure B, Leyvraz S, Kolbe K, Papiani G,Ballardini M, Kulekci A, Demirer T, Solid Tumours Working Party: First-linehigh-dose chemotherapy for patients with poor prognosis extragonadalgerm cell tumors: the experience of the European Bone MarrowTransplantation (EBMT) Solid Tumors Working Party. Bone MarrowTransplant 2004, 34:1033–1037.doi:10.1186/1477-7819-10-189Cite this article as: Yao et al.: Primary yolk sac tumor of seminal vesicle:a case report and literature review. World Journal of Surgical Oncology2012 10:189.Submit your next manuscript to BioMed Centraland take full advantage of: • Convenient online submission• Thorough peer review• No space constraints or color ﬁgure charges• Immediate publication on acceptance• Inclusion in PubMed, CAS, Scopus and Google Scholar• Research which is freely available for redistributionSubmit your manuscript at www.biomedcentral.com/submit

Primary yolk sac tumor of seminal vesicle: a case report and literature review

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Primary yolk sac tumor of seminal vesicle: a case report and literature review

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