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Clinical case
Bilateral and synchronous testicular teratoma: a case report and literature review
Teratoma testicular bilateral sincrónico: reporte de un caso y revisión de la literatura
Pedro Matabuena-Tameza,
Corresponding author
pmatabuena@hotmail.com

Corresponding author at: Prol. Privada Zaragoza 16, Torre 2 Consultorio 506, Santiago de Querétaro, C.P. 76030, Querétaro, Mexico. Tel.: +442 2153075; fax: +442 1923021.
, Patricio Canepa-Fernándezb, Luis César Valencia-Garcíac, César Gutiérrez-Samperiod, Marco Alonso Gallegos-Coronae
a Servicio de Cirugía Oncológica, Hospital Regional de Zona 1, Instituto Mexicano del Seguro Social, Santiago de Querétaro, Querétaro, Mexico
b Facultad de Medicina, Universidad Autónoma de Querétaro, Santiago de Querétaro, Querétaro, Mexico
c Servicio de Cirugía General, Centro Médico ABC, México, D.F., Mexico
d Investigación y Docencia, Universidad Autónoma de Querétaro, Santiago de Querétaro, Querétaro, Mexico
e Servicio de Patología, Hospital Regional de Zona 1, Instituto Mexicano del Seguro Social, Santiago de Querétaro, Querétaro, Mexico
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Background</span><p id="par0005" class="elsevierStylePara elsevierViewall">Testicular germ-cell carcinoma is the most frequent neoplasm in males aged 15&#8211;35<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">1</span></a>&#59; it is bilateral in 2&#8211;3&#37;&#44; and synchronous in 20&#8211;25&#37; of cases&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">2</span></a> Approximately 80&#37; of tumors are seminomas&#44; and the remaining 20&#37; have been identified as mixed teratomas&#44; yolk sac tumors and both pure and mixed embryonal cell carcinomas&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">3</span></a> Most synchronous tumors are of the same histological type&#46;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Medical records and physical examination are used in the diagnosis of testicular tumors&#46; Generally the tumors present as a painless increase in intrascrotal volume&#44; although 10&#37; of patients present with acute pain&#46; Diagnosis is commonly delayed due to the patient&#39;s reluctance to seek medical advice&#46; Scrotal ultrasound testing is the most useful diagnostic tool&#44; with tumor sensitivity in almost 100&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0125"><span class="elsevierStyleSup">5&#44;6</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">&#945;-Fetoprotein&#44; lactate dehydrogenase and human chorionic gonadotropin tumor markers both assist diagnosis and enable tumor staging whilst simultaneously serving to aid follow-up and prognosis&#46; They should be determined prior to orchiectomy&#44; one week after surgery&#44; and subsequent to the administration of chemotherapy&#44; due to their kinetics and mean life span&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">7</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Tumor staging is the cornerstone to testicular cancer treatment&#46; Computed tomography should therefore be used to assess probable metastases sites&#44; sensitivity ranges from 70&#37; to 80&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">8</span></a> 10&#37; of patients present with metastasis at the time of diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">9</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The <span class="elsevierStyleItalic">aim</span> of this article is to report a case of bilateral synchronous testicular teratoma&#44; the first manifestation of which was a retroperitoneal tumor&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Clinical case</span><p id="par0030" class="elsevierStylePara elsevierViewall">A 19 year-old male&#44; with a family history of testicular cancer presented with sudden abdominal pain in the upper left quadrant&#46; This was a moderate to severe&#44; transfixing pain coming from the left iliac fosse&#44; and was accompanied by repeated bouts of nausea and vomiting&#46; Physical examination revealed a voluminous mesogastrium tumor which extended into deeper locations&#44; and was accompanied by slight pain and no peritoneal irritation&#59; the testicles were clinically with no palpable tumor activity&#46; Abdominal X-rays showed the presence of the tumor and from the contrast-enhanced CT scan of the abdomen and pelvis we were able to determine its retroperitoneal location &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Tumor markers presented with a significantly elevated &#945;-fetoprotein of 32&#44;108&#46;6<span class="elsevierStyleHsp" style=""></span>ng&#47;ml&#44; lactate dehydrogenase of 1184<span class="elsevierStyleHsp" style=""></span>U&#47;l and fraction&#946; of human chorionic gonadotropin of 10&#44;728<span class="elsevierStyleHsp" style=""></span>mIU&#47;ml&#46; Testicular ultrasound showed a complex bilateral lesion in the right testicle of 20<span class="elsevierStyleHsp" style=""></span>mm&#44; and a solid&#44; hypervascularised lesion of 19<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>15<span class="elsevierStyleHsp" style=""></span>mm &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; in the left testicle&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Exploratory laparomoy was performed which revealed highly vascular&#44; retroperitoneal malignancy&#44; which had infiltrated the mesenterium and displaced all intestinal loops&#46; Incisional biopsies were taken of the tumor&#44; reporting mixed germ-cell tumor&#44; with areas of choriocarcinoma and embryonal carcinoma &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">Systemic treatment was initiated and 6 cycles of chemotherapy were administered with bleomycin&#44; etoposide and cisplatin&#44; with partial tumor response&#46; After terminating chemotherapy the patient subsequently underwent bilateral radical orchiectomy with pathology reporting the presence of bilateral synchronous testicular teratoma&#46; Unresectible retroperitoneal disease persisted &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">The patient was administered a second line of chemotherapy&#44; based on vincristine&#44; etoposide&#44; ifosfamide and cisplatinum&#46; Nevertheless&#44; the disease progressed&#44; spreading to the liver and lung&#44; and the patient died 2 years after diagnosis &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>&#41;&#46;</p><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Discussion</span><p id="par0055" class="elsevierStylePara elsevierViewall">Germ cell tumors represent 95&#37; of testicular cancers&#46;<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">10</span></a> They are rare diseases which mainly affect young adults and teenagers&#46;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">11</span></a> An estimated 8&#44;590 new cases are detected annually in the United States&#44; with a survival rate of up to 5 years in 96&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">12</span></a> Malignant testicular tumors rank twelfth in the number of hospital discharges for malignant tumors&#44; according to the data base of the Registro Histopatol&#243;gico de Neoplasias Malignas in Mexico&#46;<a class="elsevierStyleCrossRef" href="#bib0165"><span class="elsevierStyleSup">13</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Identifiable risk factors which predispose the development of germ cell tumors include&#58; a prior history of testicular tumor&#59; a family history of the disease&#59; undescended testicles&#59; testicular dysgenics&#44; and Klinefelter syndrome&#46;<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">14</span></a> A change in the short arm of chromosome 12&#59; i&#40;12p&#41; has been identified as a possible cause&#44; and a mutation in gene DAD-R &#40;apoptosis-inducing agent&#41; has also been reported&#46;&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0175"><span class="elsevierStyleSup">15&#44;16</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Primary presentation of these tumors may be extra-gonadal in location&#44; due to embryonic origin and testicular descent&#46; It is difficult to distinguish a retroperitoneum primary germ cell tumor from metastatic disease of a clinically undetected gonadal tumor or one that has regressed&#46; This clinical condition is known as tumor burn-out&#46;<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">14</span></a> It is important to recall that initial signs of these tumors may be voluminous abdominal metastases&#44; with no primary testicular tumor having been detected&#46; This therefore delays diagnosis and treatment&#59; examination and ultrasound imaging of the testicles are therefore important&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">In the clinical case presented&#44; there was no palpable tumor on examination but the doppler ultrasound scan revealed a complex&#44; highly vascular cystic lesion in both testicles&#46; This situation is considered infrequent&#44; given that bilateral synchronous testicular cancer is extremely rare&#46;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">11&#44;17</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Based on the <span class="elsevierStyleItalic">American Joint Committee on Cancer</span> &#40;AJCC&#41; classification scale we could state that our case was a stage IIIC &#40;pTXN3MOS3&#41; when diagnosed&#46;<a class="elsevierStyleCrossRef" href="#bib0190"><span class="elsevierStyleSup">18</span></a> There is a risk classification scale for the patient with advanced disease&#44; from the Grupo Colaborativo Internacional de C&#225;ncer de C&#233;lulas Germinales&#46; This classification takes several prognostic factors into account&#44; including histological prognosis&#44; the spread of the disease and the serum levels of the tumor markers&#44; from which 3 risk groups are established&#58; low&#44; intermediate and high&#46;<a class="elsevierStyleCrossRef" href="#bib0195"><span class="elsevierStyleSup">19</span></a> In accordance with this classification our patient was in the high risk category&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">The presence of an active tumor in the testicles after chemotherapy is a clearly proven fact&#44; with the result that surgical exploration of the testicles and even orchiectomy are options to consider whilst taking into account individual patient characteristics&#46;<a class="elsevierStyleCrossRef" href="#bib0200"><span class="elsevierStyleSup">20</span></a> In our case inguinal extraction from the testicle was performed and transoperative tested biopsies were taken&#46; Pathology reported testicular tumor&#44; and we therefore decided to perform bilateral radical orchiectomy&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Conclusions</span><p id="par0085" class="elsevierStylePara elsevierViewall">Statistics show that approximately 90&#37; of patients diagnosed with germ cell tumors can be cured&#46; However&#44; delay in diagnosis correlates with an advanced clinical stage and poor diagnosis&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">The clinical evolution of the patient was slow&#44; given the aggressive biological nature of the tumor&#44; and late stage diagnosis&#46; Despite treatment with chemotherapy and bilateral orchiectomy&#44; the disease progressed and the patient died as a result&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0085">Conflict of interests</span><p id="par0095" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare&#46;</p></span></span>"
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            2 => array:2 [
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    "fechaRecibido" => "2014-05-19"
    "fechaAceptado" => "2014-10-03"
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          "clase" => "keyword"
          "titulo" => "Keywords"
          "identificador" => "xpalclavsec607288"
          "palabras" => array:3 [
            0 => "Synchronous bilateral testicular teratoma"
            1 => "Metastases"
            2 => "Testicular germ-cell carcinoma"
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          "clase" => "keyword"
          "titulo" => "Palabras clave"
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          "palabras" => array:3 [
            0 => "Teratoma testicular sincr&#243;nico bilateral"
            1 => "Met&#225;stasis"
            2 => "C&#225;ncer testicular de c&#233;lulas germinales"
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    "resumen" => array:2 [
      "en" => array:3 [
        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Background</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Testicular germ-cell carcinoma is the most frequent neoplasm in males aged 15&#8211;35 years old&#46; It is bilateral in 2&#8211;3&#37;&#44; and synchronous in 20&#8211;25&#37; of the cases&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Clinical case</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The case is presented of a 19 year-old male&#44; with abdominal pain&#46; Physical examination revealed abdominal mass in the umbilical region&#44; and the computed tomography scan showed a retroperitoneal tumor&#44; with &#945;-fetoprotein&#44; lactate dehydrogenase&#44; and human chorionic gonadotropin above limits&#46; Testicular ultrasound showed bilateral lesions&#46; Exploratory laparotomy was performed&#44; identifying an unresectable retroperitoneal tumor&#46; Biopsies were taken&#44; reporting mixed germ cell tumor composed of choriocarcinoma and embryonal carcinoma&#46; Six cycles of chemotherapy were given&#44; based on bleomycin&#44; etoposide and cisplatin&#44; with partial tumor response&#46; Later on&#44; the patient underwent bilateral radical orchiectomy&#44; with pathology reporting a synchronous bilateral testicular teratoma&#46; A second line of chemotherapy was given&#44; based on vincristine&#44; etoposide&#44; ifosfamide and cisplatinum&#46; Nevertheless&#44; the disease progressed&#44; with metastatic dissemination and the patient died&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Discussion</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Germ cells tumors can be presented in primary extra-gonadal locations&#46; It is difficult to distinguish a retroperitoneum primary germ cell tumor from metastatic disease of a clinically undetected gonadal tumor or one that has regressed&#44; like the situation described in the case presented&#46;</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conclusions</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Ninety percent of patients diagnosed with germ cell tumors can be cured&#46; However&#44; delay in diagnosis correlates with an advanced clinical stage and poor prognosis&#46;</p></span>"
        "secciones" => array:4 [
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        "resumen" => "<span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Antecedentes</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El c&#225;ncer testicular de c&#233;lulas germinales es la neoplasia m&#225;s frecuente en hombres de 15 a 35 a&#241;os de edad&#59; es bilateral en el 2 al 3&#37;&#44; y sincr&#243;nico en el 20 al 25&#37; de los casos&#46;</p></span> <span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Caso cl&#237;nico</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Masculino de 19 a&#241;os de edad&#44; con dolor abdominal y tumor palpable en mesogastrio&#46; En la tomograf&#237;a se encontr&#243; un tumor retroperitoneal&#44; y por laboratorio se detect&#243; elevaci&#243;n de &#945;-fetoprote&#237;na&#44; deshidrogenasa l&#225;ctica y gonadotropina cori&#243;nica humana&#46; En el ultrasonido testicular se identifican lesiones bilaterales&#46; Se realiz&#243; laparotom&#237;a exploradora&#44; identific&#225;ndose tumor retroperitoneal irresecable&#44; y se tomaron biopsias incisionales compatibles para tumor de c&#233;lulas germinales mixto&#44; con &#225;reas de coriocarcinoma y carcinoma embrionario&#46; Se administraron 6 ciclos de quimioterapia con bleomicina&#44; etop&#243;sido y cisplatino&#44; obteni&#233;ndose una respuesta tumoral parcial&#46; Posteriormente se realiz&#243; orquiectom&#237;a radical bilateral&#44; con reporte patol&#243;gico de teratoma bilateral sincr&#243;nico&#46; Se inici&#243; segunda l&#237;nea de quimioterapia con vincristina&#44; etop&#243;sido&#44; ifosfamida y platino&#59; sin embargo&#44; la enfermedad progres&#243;&#44; presentando diseminaci&#243;n metast&#225;sica y provocando el deceso del paciente&#46;</p></span> <span id="abst0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Discusi&#243;n</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Los tumores de c&#233;lulas germinales pueden presentarse en sitios primarios extragonadales&#46; Es dif&#237;cil distinguir un tumor de c&#233;lulas germinales primario del retroperitoneo&#44; de una enfermedad metast&#225;sica derivada de un tumor gonadal no detectado cl&#237;nicamente&#44; o que ha involucionado&#44; situaci&#243;n que se describe en el caso cl&#237;nico presentado&#46;</p></span> <span id="abst0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conclusi&#243;n</span><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">El 90&#37; de los pacientes diagnosticados con tumor de c&#233;lulas germinales pueden ser curados&#59; sin embargo&#44; un retraso en el diagn&#243;stico se correlaciona con una etapa cl&#237;nica m&#225;s avanzada y un pron&#243;stico desfavorable&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Matabuena-Tamez P&#44; Canepa-Fern&#225;ndez P&#44; Valencia-Garc&#237;a LC&#44; Guti&#233;rrez-Samperio C&#44; Gallegos-Corona MA&#46; Teratoma testicular bilateral sincr&#243;nico&#58; reporte de un caso y revisi&#243;n de la literatura&#46; Cirug&#237;a y Cirujanos&#46; 2015&#59;83&#58;527&#8211;531&#46;</p>"
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          "en" => "<p id="spar0050" class="elsevierStyleSimplePara elsevierViewall">Ultrasound scan of the right testicle which shows a cystic fine-tissued tumor with irregular hypoechoic areas&#46;</p>"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Tumor testicular bilateral metacr&#243;nico asociado a microlitiasis"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "M&#46;A&#46; Feria-Flores"
                            1 => "R&#46; Guti&#233;rrez-Lerma"
                            2 => "S&#46;C&#46; Lara-Miranda"
                            3 => "J&#46;F&#46; L&#243;pez-Verdugo"
                            4 => "L&#46;C&#46; Urbina-Bernal"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:5 [
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Article information
ISSN: 24440507
Original language: English
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