Yolk sac tumor of the endometrium in a post-menopausal woman: Case report and review of the literature.
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ABSTRACT:
SUBMITTER: Sinha R
PROVIDER: S-EPMC8050376 | biostudies-literature | 2021 May
REPOSITORIES: biostudies-literature
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Yolk sac tumor of the endometrium in a post-menopausal woman: Case report and review of the literature.
Sinha Risha R Bustamante Bethany B Truskinovsky Alexander A Goldberg Gary L GL Shih Karin K KK
Gynecologic oncology reports 20210316
•Seven of eighteen postmenopausal female endometrial YST cases were pure YST.•IHC supports somatic tumor cell <i>retro</i>-differentiation yielding extra-gonadal YST.•Studying genetic alterations in endometrial YST may elucidate its histiogenesis. ...[more]
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Project description:This is a case report of a rare finding of an extragonadal yolk sac tumor in a 37-year-old patient who presented with shortness of breath and abdominal bloating. During imaging and staging surgery, the findings were strongly suggestive of an extragonadal advanced tumor presenting with peritoneal dissemination, predominantly affecting omentum, with no clear primary origin. Histology revealed an extragonadal yolk sac tumor in a pure form outside the ovaries. Lacking an obvious origin elsewhere, the tumor was highly suspected to have truly originated from the omentum. The patient underwent surgery and four cycles of chemotherapy consisting of cisplatin, etoposide, and bleomycin. One-year outpatient follow-up thereafter showed no relapse. We herein discuss a possible site of the tumor origin and its development, as well as diagnostic challenges and disease prognosis.
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Project description:IntroductionOvarian yolk sac tumors after the menopause are very rare.Case presentationWe report an atypical case of yolk sac tumor in an octogenarian woman, who presented to the hospital with anasarca, ascites, toxic syndrome, and an abdominal mass. Serum alpha-fetoprotein (AFP) levels were highly increased (246,720 ng/mL), and malignant cells with positive AFP immunohistochemical expression were detected in a diagnostic paracentesis. The definitive diagnosis was established by histopathological examination of the surgical specimen and immunohistochemical detection of positivity for AFP, glypican 3, cytokeratin AE1/AE3, and focal positivity for SALL4 and CK7. Adjuvant chemotherapy was refused by the patient. Postoperative AFP values decreased to normal limits. One year after surgery, she remains in good clinical condition without evidence of tumor recurrence. In a review of the literature, 11 cases of yolk sac tumors in postmenopausal women were collected. The mean age of the patients was 63.4 years (range 58-82 years). Frequent findings were FIGO stage II, cytoreductive surgery, immunohistochemical analyses of a large panel of markers, postoperative chemotherapy, and no tumor recurrence (except in 1 patient who died), although the length of follow-up was relatively short or unclearly stated.ConclusionThe present case report illustrates that yolk sac tumors in very old women may have a favorable clinical course without signs of recurrence even in the absence of radical surgical resection with curative intent and associated adjuvant chemotherapy.
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Project description:Yolk sac tumors (YSTs) of the endometrium and the broad ligament are very rare, with only 29 cases and one case of each other reported before in the English literature. Due to lack of standard guidelines, the treatment strategies of these diseases are controversial. Here, we share two cases of YSTs originating from the endometrium and the broad ligament respectively and review related literature. A 35-year-old woman was diagnosed with endometrial YST in our center and underwent surgery followed by chemotherapy with BEP (bleomycin, cisplatin and etoposide) regimen for six courses. After follow-up for 21 months, there is still no evidence of relapse. Another 36-year-old woman was admitted to our department with YST of the broad ligament. She was treated with surgery followed by chemotherapy with BEP regimen and was lost to follow-up after completing therapy. The case of endometrial YST we shared was similar to cases reported before, while the case with YST of the broad ligament we shared was the second case reported worldwide. Both of these two cases were treated with surgery combined with chemotherapy with BEP regimen.
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Project description:BackgroundYolk sac tumor (YST), or endodermal sinus tumor, is classically associated with pediatric populations. Metastasis to the spine rarely occurs, usually involving the lower thoracic or lumbar vertebrae. The objective of this report is to present a rare case of YST metastasis to the lower cervical and upper thoracic vertebrae in an adult male. A case-based review of the literature on metastatic YSTs was also performed as an update to the relevant literature.Case descriptionA 28-year-old male with a history of YST presented to our institution with urinary retention, increasing weakness in the upper extremities, and acute onset lower extremity weakness. Computed tomography (CT) and magnetic resonance imaging (MRI) scans confirmed evidence of metastasis from a known YST with symptomatic cord compression. The patient was treated with surgical excision via decompressive laminectomies with instrumentation as described, and histopathologic analysis of the specimen confirmed YST metastasis. His disease recurred one year after index surgery. He succumbed to his disease despite repeated debulking.ConclusionsMetastasis of YST is rare, but metastasis to lower cervical and upper thoracic vertebrae is possible. YSTs are usually treated via primary surgical resection. Systemic chemotherapy and radiation may prevent recurrence. However, individualized treatment is imperative for improved patient outcomes.
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Project description:BackgroundYolk sac tumor is the most common malignant nonseminomatous germ-cell tumor in children characterized by elevated level of α-fetoprotein (AFP), accounting for 70%-80% of all cases. However, giant yolk sac tumors that involve the entire testicle may be misdiagnosed by color Doppler ultrasonography as orchitis. Therefore, we described a case of a 2-year-old pediatric patient with a giant testicular yolk sac tumor that was misdiagnosed by ultrasonography as orchitis, in order to evaluate the role of measuring AFP levels in the initial diagnosis to aid in the accuracy of the definitive diagnosis of testicular yolk sac tumor.Case presentationA 2-year-old boy received outpatient visits for unintentional swelling of the right scrotum for 7 days. Physical examination showed a rubbery swelling of the right scrotum with rejective touch. Then, the patient underwent perineal color Doppler ultrasonography in outpatient visits. The result showed a right testicle size of 29 mm × 22 mm × 20 mm with heterogeneous echogenicity and abundant blood flow, supporting the initial diagnosis of orchitis. However, the initial surgeon was skeptical of the ultrasonography diagnosis. Thus, the patient was admitted to the Department of Andrology on day 2 for further serological and imaging examination. The serum AFP level on day 3 was 323.77 ng/ml. The results of CT and MRI showed a giant tumor of the right testis (26 mm × 21 mm × 29.6 mm) with multiple lymphoid hyperplasia in the inguinal region bilaterally. The patient received radical orchidectomy without lymph node dissection on day 9. The results of postoperative pathological examination confirmed giant testicular yolk sac tumor (T1N0M0S1, Stage Is) and was positive for AFP and SALL4 in immunohistochemistry staining. The patient received three courses of bleomycin-etoposide-cisplatin chemotherapy in the Department of Pediatrics after multidisciplinary team meeting on postoperative days 14, 37, and 58, respectively. During chemotherapy and follow-up, the patient's AFP and lactate dehydrogenase levels continued to decline, and eventually remained within normal range on postoperative day 84.ConclusionMeasuring the AFP level was necessary for initial diagnosis and follow-up in pediatric cases of testicular enlargement. Radical orchidectomy combined with postoperative bleomycin-etoposide-cisplatin adjuvant chemotherapy was an effective treatment strategy for pediatric giant testicular yolk sac tumors.
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Project description:A vaginal yolk sac tumor (YST) is a rare malignant germ cell tumor for infants and children, and it has been >50 years since the first case was reported. The treatment strategy has changed markedly in the past 50 years, from radical surgical treatment to conservative surgery combined with chemotherapy, and then to combined chemotherapy alone. The present study reports the case of a primary vaginal YST in a 13-month-old girl that was successfully treated by tumor resection combined with chemotherapy. The clinical symptoms, imaging features and treatment characteristics are described in detail, as well as the postoperative treatment. There was no local recurrence or metastasis for the 2 years of follow-up to date. A literature review was also conducted to investigate the clinicopathological features, treatment and prognosis of this tumor. Overall, surgery combined with bleomycin, etoposide and carboplatin combination chemotherapy can be an effective option for vaginal YST.
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Project description:Non-gestational ovarian choriocarcinoma (NGOC) is a rare phenomenon seldom reported in the literature. Patients often present with abdominopelvic pain, and sometimes a palpable adnexal mass. Surgical excision is paramount in treating this malignancy; however, fertility-preserving care is a debated topic among gynecologic oncologists. Most patients reported in the literature are nulliparous women of child-bearing age. It is important to consider fertility preservation whilst balancing oncologic outcomes. We present a case of an 18-year-old nulliparous female with stage IIB NGOC that had disease progression in the lungs and pelvis shortly after undergoing fertility-preserving surgery. She required emergent completion surgery and received etoposide (E), methotrexate (M), actinomycin-D (A), cyclophosphamide (C) and vincristine (O) (EMA-CO) with complete response. She remains disease-free after 21-months.
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Project description:Here, we report the first adult case of pancreatic yolk sac tumor with ectopic adrenocorticotropic hormone (ACTH) syndrome. The patient was a 27-year-old woman presenting with abdominal distension, Cushingoid features, and hyperpigmentation. Endogenous Cushing's syndrome was biochemically confirmed. The ACTH level was in the normal range, which raised the suspicion of ACTH precursor-dependent disease. Elevated ACTH precursors were detected, supporting the diagnosis of ectopic ACTH syndrome. Functional imaging followed by tissue sampling revealed a pancreatic yolk sac tumor. The final diagnosis was Cushing's syndrome due to a yolk sac tumor. The patient received a steroidogenesis inhibitor and subsequent bilateral adrenalectomy for control of hypercortisolism. Her yolk sac tumor was treated with chemotherapy and targeted therapy. Cushing's syndrome secondary to a yolk sac tumor is extremely rare. This case illustrated the utility of ACTH precursor measurement in confirming an ACTH-related pathology and distinguishing an ectopic from a pituitary source for Cushing's syndrome.
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Project description:IntroductionYolk sac tumor is the second most common germ cell ovarian carcinoma. Neoadjuvant chemotherapy (NACT) is proposed as an alternative option for patients with advanced disease who are not eligible for primary debulking. We presented a case with a systematic review of advanced yolk sac tumors of the ovary with poor performance who gain benefit from NACT.Case presentationA 24-year-old female underwent NACT followed by surgery after being diagnosed with an advanced yolk sac tumor. A literature search was done based on the clinical question using the Patient/Problem, Intervention, Comparison, and Outcome (PICO) approach. Pubmed and Google Scholar were used to search the literature.DiscussionOut of 111 manuscripts found, 2 articles were retrieved for detailed evaluation. The patient showed a complete response in tumor size, histopathology, and tumor markers after the NACT procedure followed by surgery.ConclusionsNACT is a suitable option for advanced yolk sac tumors of the ovary.
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Project description:BackgroundYolk sac tumor (YST) is a germ cell neoplasm that arises predominantly in the gonads, but can also derive from somatic neoplasms in extragonadal locations. These latter cases have been documented in several organs, although reports from the urinary tract are limited. To our knowledge, this is the first report of a bladder urothelial carcinoma with a predominant component of YST differentiation.Case presentationWe present a unique case of a 76-year-old man with a recurrent urinary bladder tumor, initially interpreted as a high grade urothelial carcinoma with glandular differentiation. In the recurrent tumor, diverse histological patterns were identified, including glandular, hepatoid and sarcomatoid. This tumor showed positivity for AFP, GLP3 and SALL4, and negativity for CK7 and EMA. Fluorescent in situ hybridization study showed a polysomic pattern of chromosome 12. All these findings led to the final diagnosis of a YST derived from urothelial carcinoma.ConclusionsYST differentiation should be considered in the differential diagnosis of a high grade urothelial carcinoma, particularly when glandular and other unusual patterns are observed.
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