Scientific Letter
Mature Cystic Teratoma of the Lung With a Somatic-Like Malignant Component in a Patient With Testicular Germ-Cell Tumor
Teratoma quístico maduro de pulmón con componente maligno de tipo somático en un paciente con un tumor testicular de células germinales
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Cells display eosinophilic cytoplasm, rounded nuclei and fine granular chromatin. Metastatic lesion: Mature glandular tissue staining positive for CDX2 (B) and CK7 (C). (D) Tumor cells staining positive for chromogranin-A.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">In urological oncology, the diagnosis of <span class="elsevierStyleItalic">teratoma with a somatic-like malignant component</span> refers to the presence of a malignant neoplasm, e.g. carcinoma or sarcoma, either within a teratoma of the genital tract (usually the testis) or, more commonly, in a metastasis, following initial cisplatin therapy.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The somatic-type malignancy (STM) most widely observed in germ cell tumors (GCT) is rhabdomyosarcoma; neuroendocrine carcinoma is extremely rare.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report on a 40-year-old patient who had undergone surgery seven years earlier for a malignant mixed germ-cell tumor of the right testicle with pulmonary and retroperitoneal metastasis, and received cisplatin therapy. At recent check-up, CT scans revealed some enlargement of known lung lesions; blood alpha-fetoprotein levels were also slightly elevated (AFP: 9<span class="elsevierStyleHsp" style=""></span>ng/ML). Since needle-biopsy findings were inconclusive, lung nodules were all excised by videothoracoscopy. Histological examination of them revealed a multicystic lesion containing solid areas. The cystic component was lined by squamous, respiratory and intestinal epithelium, a finding consistent with a mature teratoma. The solid component comprised tumor cell nests displaying peripheral pallisading, an organoid arrangement with rosette formation, focal necrosis and a high mitotic rate (6/10 HPF) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). No elements characteristic of immature teratoma were observed. Immunohistochemical analysis revealed patchy focal positivity for CK7, CK20, CDX2 and SOX2 in the cylindrical epithelial cells of the cystic component (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B and C). Solid-component cells stained positive for cytokeratin (AE1/AE3), chromogranin-A (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D), synaptophysin, and cytoplasmic OCT4, and negative for CD56. Staining for neural development transcription factors (TTF1, ASCL1 and POU3F2) was positive only for POU3F2 (nuclear). Since tests using a broad battery of antibodies (CDX2, TTF1, CK7 and CK20) yielded negative results, it was not possible to identify the original tissue of the neuroendocrine component.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The final pathological diagnosis was atypical carcinoid tumor in a metastatic mature cystic teratoma. Surgical margins were tumor free, and postoperative recovery was good, with no complications.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Two histogenetic hypotheses have been put forward to account for this association. The first is that cisplatin therapy selectively excludes immature components, whilst chemotherapy-resistant mature components survive and in some cases grow. The second is that chemotherapy induces the transformation of immature malignant teratoma into mature teratoma, a phenomenon first reported by DiSaia et al. in ovarian GCTs, and termed chemotherapeutic retroconversion.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Somatic-type malignancy occurs in 3–6% of GCTs, almost always in postpubertal patients (aged 15–68).<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2,4</span></a> Although most affected patients have teratomatous GCTs,<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2,5</span></a> somatic-type malignancy has occasionally been reported in other germ-cell tumors including yolk-sac and spermatocytic tumors.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> The time elapsing between GTC diagnosis and the appearance of STMs tends to vary widely, ranging from simultaneous presentation to a latency period of up to 30 years. The interval for carcinomas (108 months) is longer than for sarcomas (20 months).<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Reported STMs include various types of sarcoma, epithelial malignancies and hematological malignancies.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> Sarcomas are undoubtedly the most common type of malignancy; although rhabdomyosarcomas account for over half the STMs recorded, leiomyosarcomas, angiosarcomas and other STMs have occasionally been reported.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">8,9</span></a> Carcinomas tend to be mostly to be classed as adenocarcinomas NOS<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a>; there are very few reports of neuroendocrine tumors, particularly in testicular lesions.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Generally speaking, the prognosis for these tumors is not affected when the STM is located in the testicle; however, development of STM in metastases—as in the present case—is associated with an increased risk of mortality.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The role of chemotherapy in teratomas with malignant neuroendocrine transformation has not been fully established.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> Complete surgical resection has traditionally been the treatment of choice when malignant transformation occurs at a single site. Metastatic or unresectable tumors will generally require multimodal management including chemotherapy in addition to loco-regional approaches. Although patients tend to respond poorly to cisplatin-based treatments, some tumors may respond to the specific chemotherapy used for the somatic counterpart.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Summarizing, in this rare case a patient with mixed germ-cell tumor of the testis and metastasis, treated with chemotherapy, seven years later developed an atypical carcinoid tumor, with a somatic-like malignant component, in a metastatic mature cystic teratoma of the lung. Comprehensive pathological examination is essential for the identification of somatic neoplasms within germ cell tumor metastases.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 946 "Ancho" => 1257 "Tamanyo" => 484715 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Solid islets and cell clusters separated by a fibrous stroma. Cells display eosinophilic cytoplasm, rounded nuclei and fine granular chromatin. Metastatic lesion: Mature glandular tissue staining positive for CDX2 (B) and CK7 (C). 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 9 | 2 | 11 |
| 2024 October | 56 | 31 | 87 |
| 2024 September | 49 | 21 | 70 |
| 2024 August | 71 | 47 | 118 |
| 2024 July | 66 | 15 | 81 |
| 2024 June | 65 | 30 | 95 |
| 2024 May | 72 | 19 | 91 |
| 2024 April | 55 | 31 | 86 |
| 2024 March | 64 | 20 | 84 |
| 2024 February | 49 | 17 | 66 |
| 2023 March | 16 | 4 | 20 |
| 2023 February | 84 | 16 | 100 |
| 2023 January | 45 | 26 | 71 |
| 2022 December | 81 | 26 | 107 |
| 2022 November | 96 | 21 | 117 |
| 2022 October | 122 | 50 | 172 |
| 2022 September | 53 | 30 | 83 |
| 2022 August | 58 | 49 | 107 |
| 2022 July | 56 | 52 | 108 |
| 2022 June | 59 | 45 | 104 |
| 2022 May | 64 | 36 | 100 |
| 2022 April | 85 | 43 | 128 |
| 2022 March | 76 | 46 | 122 |
| 2022 February | 94 | 42 | 136 |
| 2022 January | 96 | 27 | 123 |
| 2021 December | 71 | 33 | 104 |
| 2021 November | 61 | 43 | 104 |
| 2021 October | 51 | 43 | 94 |
| 2021 September | 50 | 50 | 100 |
| 2021 August | 34 | 44 | 78 |
| 2021 July | 19 | 33 | 52 |
| 2021 June | 51 | 61 | 112 |
| 2021 May | 63 | 31 | 94 |
| 2021 April | 126 | 100 | 226 |
| 2021 March | 74 | 37 | 111 |
| 2021 February | 48 | 24 | 72 |
| 2021 January | 44 | 19 | 63 |
| 2020 December | 44 | 25 | 69 |
| 2020 March | 12 | 11 | 23 |
| 2020 February | 42 | 17 | 59 |
| 2020 January | 41 | 21 | 62 |
| 2019 December | 41 | 21 | 62 |
| 2019 November | 38 | 19 | 57 |
| 2019 October | 30 | 12 | 42 |
| 2019 September | 31 | 13 | 44 |
| 2019 August | 34 | 19 | 53 |
| 2019 July | 39 | 17 | 56 |
| 2019 June | 30 | 12 | 42 |
| 2019 May | 0 | 2 | 2 |
| 2019 March | 1 | 2 | 3 |
| 2019 January | 4 | 4 | 8 |
| 2018 December | 24 | 6 | 30 |
| 2018 October | 2 | 2 | 4 |
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