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Ma X, Gan J, Cao D, Peng P. Extracranial and nonvaginal extragonadal malignant germ cell tumors: 12 cases at a Chinese institution over the last 38 years. Arch Gynecol Obstet 2025; 311:1637-1648. [PMID: 39774707 PMCID: PMC12055612 DOI: 10.1007/s00404-024-07889-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2024] [Accepted: 12/10/2024] [Indexed: 01/11/2025]
Abstract
PURPOSE To provide a comprehensive understanding and propose a strategy for the management of extragonadal malignant germ cell tumors (EMGCTs) arising from extracranial and nonvaginal sites. METHODS We retrospectively reviewed the cases of 12 patients with EMGCTs arising from extracranial and nonvaginal sites treated in our center over the past 38 years. Data on clinicopathological characteristics, treatment modalities, and follow-up information were analyzed. RESULTS Among 209 patients diagnosed with EMGCTs, 12 women (5.7%) with EMGCTs of extracranial and nonvaginal sites were identified. These patients had tumors in the sacrococcygeal region (n = 4), abdominal cavity (n = 3), groin region (n = 2), uterus (n = 2), and mediastinum (n = 1). The median age at diagnosis was 23 years. Symptoms included abnormal uterine bleeding (n = 3), abdominal discomfort (n = 3), compression symptoms (n = 3), palpable mass (n = 2), and asymptomatic (n = 1). Yolk sac tumors (YSTs) were the most common histologic type. The median level of serum alpha-fetoprotein (AFP), a sensitive tumor marker, was 8216 ng/ml (2.7-74,157 ng/ml). One patient started bleomycin/etoposide/cisplatin without a pathologic diagnosis based on clinical diagnosis (high AFP levels and imaging findings), and 11 patients started chemotherapy following tumor biopsy or surgical resection. During the follow-up, one patient suffered a recurrence, two patients were alive with disease, and nine patients were disease-free. CONCLUSIONS Extracranial and nonvaginal EMGCTs are a heterogeneous group of tumors due to their varied onset ages, location, and clinical presentation. An all-around clinical evaluation is crucial for selecting appropriate treatment. Most patients achieve a good prognosis after surgical resection and chemotherapy. Patients with these rare diseases may benefit from individualized treatment and timely referral to experienced medical centers.
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Affiliation(s)
- Xiao Ma
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Jingwen Gan
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Dongyan Cao
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China.
- Peking Union Medical College Hospital (Dongdan Campus), No.1 Shuaifuyuan Wangfujing Dongcheng District, Beijing, 100730, China.
| | - Peng Peng
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China.
- Peking Union Medical College Hospital (Dongdan Campus), No.1 Shuaifuyuan Wangfujing Dongcheng District, Beijing, 100730, China.
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Pathak A, Vijay N, Bhalerao A. Clinical, Diagnostic, and Intervention Approach on Yolk Sac Tumor: A Case Report. Cureus 2024; 16:e71184. [PMID: 39525133 PMCID: PMC11549645 DOI: 10.7759/cureus.71184] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 10/10/2024] [Indexed: 11/16/2024] Open
Abstract
Endodermal primitive tumors, also known as yolk sac tumors (YSTs), are rare tumors that usually develop in the gonads and are more common in females at premenarchal age. Thus, this report details a rare case of YST in a 13-year-old girl who presented to the Obstetrics and Gynecology outpatient department with complaints of pain and a mass in the abdominal region that had been gradually growing in size for two to three months. The patient also reported a history of weight loss, loss of appetite, intermittent fever, and frequent urination. Per abdomen examination resulted in a mass corresponding to 24 weeks which was firm or hard in consistency with restricted mobility, arising from the pelvis and having an irregular surface. Per rectal examination also reported the same mass on the right side. For investigation magnetic resonance imaging (MRI) was performed that stated the lesion with neoplastic etiology likely arising from the uterus. The treatment plan included a staging laparotomy with a right oophorectomy, bilateral salpingectomy, dissection of the right preaortic and pelvic lymph nodes, and infracolic omentectomy, followed by a course of chemotherapy. The diagnosis of YST was confirmed based on the intraoperative findings and the frozen section report. As this is a rare tumor gynecologists and pediatric physicians should still be aware of this tumor since it can be life-threatening and should be promptly prevented and treated.
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Affiliation(s)
- Anjanee Pathak
- Department of Obstetrics and Gynaecology, Narendra Kumar Prasadrao Salve Institute of Medical Sciences and Research Centre, Nagpur, IND
| | - Nikita Vijay
- Department of Obstetrics and Gynaecology, Narendra Kumar Prasadrao Salve Institute of Medical Sciences and Research Centre, Nagpur, IND
| | - Anuja Bhalerao
- Department of Obstetrics and Gynaecology, Narendra Kumar Prasadrao Salve Institute of Medical Sciences and Research Centre, Nagpur, IND
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Chen M, Zhang S, Jia X, Xu Y, Wei Y, Liao S. Ultrasonographic and clinicopathological features of pelvic yolk sac tumors in women: a single-center retrospective analysis. Front Oncol 2024; 14:1417761. [PMID: 38966065 PMCID: PMC11222404 DOI: 10.3389/fonc.2024.1417761] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/15/2024] [Accepted: 06/05/2024] [Indexed: 07/06/2024] Open
Abstract
Objectives Yolk sac tumors (YSTs) are rare and highly malignant ovarian malignancies that have a very poor prognosis. The aim of this study is to delineate the ultrasound and clinicopathological features of female pelvic YSTs to better understand the disease. Methods This study was a retrospective analysis of the clinicopathological and ultrasound imaging data from 16 YST patients who received treatment at our hospital between January 2012 and August 2023. Then, the ultrasound imaging characteristics were compared with pathological findings. Results Among the 16 patients, various degrees of serum AFP increase were observed, and CA125 levels increased in 58.33% (7 out of 12) of patients. Thirteen patients (81.25%) had tumors located in ovary, two patients (12.5%) had tumors located in the sacrococcygeal region, and one patient (6.25%) had tumors located in the mesentery. Pathologically, nine patients presented with simple yolk sac tumors and seven with mixed germ cell tumors. According to the ultrasound manifestations, YST lesions can be classified into three types. (1) the cystic type, was diagnosed in two patients who presented with a large cystic mass with regular morphology and clear boundary and dense liquid within the cyst; and (2) the cystic-solid mixed type, was diagnosed in 4 patients. On 2D ultrasound, the lesions showed a cystic-solid mixed echo, and color Doppler showed a rich blood flow signal in the solid region and cystic separation. made up of four cases. (3) In ten patients with the solid type, 2D ultrasound showed solid uniform echoes with clear boundaries. The "fissure sign" was observed in the lesion. Color Doppler displayed rich blood flow in the solid part, and PW showed low to moderate resistance index of artery (RI:0.21-0.63). On contrast-enhanced ultrasound (CEUS), rapid and high enhancement in the solid part and cystic separation was observed in 2 patients. Conclusions Combining ultrasound features with clinical information and tumor markers provides reliable clues for the diagnosis of YST. The application of two-dimensional ultrasound and CEUS combined with patient tumor marker levels can provide a robust reference for determining the necessity of fertility-preserving surgery and postoperative chemotherapy, which can improve clinical decision-making and patient consultation.
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Affiliation(s)
- Mei Chen
- Department of Ultrasonography, The First Affiliated Hospital of Ningbo University, Ningbo, Zhejiang, China
| | - Shengmin Zhang
- Department of Ultrasonography, The First Affiliated Hospital of Ningbo University, Ningbo, Zhejiang, China
| | - Xiupeng Jia
- Department of Histopathology, Ningbo Clinical Pathology Diagnosis Center, Ningbo, Zhejiang, China
| | - Youfeng Xu
- Department of Ultrasonography, The First Affiliated Hospital of Ningbo University, Ningbo, Zhejiang, China
| | - Yaping Wei
- Department of Ultrasonography, Ningbo Women and Children’s Hospital, Ningbo, Zhejiang, China
| | - Shusheng Liao
- Department of Ultrasound, The First Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China
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Zhang X, Yang J, Xiang Y, Pan L, Wu M, Cao D, Yang J. Advanced ovarian yolk sac tumor: upfront surgery or neoadjuvant chemotherapy followed by interval debulking? Int J Gynecol Cancer 2024; 34:99-105. [PMID: 37696647 DOI: 10.1136/ijgc-2023-004624] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 09/13/2023] Open
Abstract
OBJECTIVE To compare surgery and survival outcomes between neoadjuvant chemotherapy and primary debulking surgery in patients with advanced ovarian yolk sac tumor. METHODS In this retrospective cohort analysis, patients with stage III to IV ovarian yolk sac tumor or mixed germ cell tumors containing yolk sac tumor elements, and who underwent surgery at Peking Union Medical College Hospital between January 2011 and December 2021, were identified. Patient characteristics, treatment, and survival data were analyzed between the two groups. RESULTS A total of 40 patients were enrolled: 19 patients received neoadjuvant chemotherapy followed by interval surgery, and 21 patients were treated with primary debulking surgery. After neoadjuvant chemotherapy, the surgical conditions of patients were improved. All patients achieved cytoreduction to R0 or R1 at interval surgery. No statistical difference was found in 3-year disease-free survival and overall survival between the neoadjuvant chemotherapy group and the primary debulking surgery group (log rank p=0.4 and 0.94). Patients had less blood loss (328.4 vs 1285.7 mL, p=0.029), lower transfusion volume (1044.4 vs 3066.7 mL, p=0.011), and fewer peri-operative complications (15.8% vs 47.6%, p=0.032) at the interval debulking surgery after neoadjuvant chemotherapy compared with patients who underwent primary debulking surgery. CONCLUSION For patients with advanced-stage ovarian yolk sac tumor, neoadjuvant chemotherapy followed by interval surgery is an alternative option, especially for those who cannot tolerate the primary debulking surgery because of high tumor burden and vulnerable status.
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Affiliation(s)
- Xinyue Zhang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
| | - Jie Yang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
| | - Yang Xiang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
| | - Lingya Pan
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
| | - Ming Wu
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
| | - Dongyan Cao
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
| | - Jiaxin Yang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing, China
- National Clinical Research Center for Obstetric & Gynecologic Diseases, Beijing, China
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Hammami MB, Rezk M, Dubey D. Paraneoplastic neurologic syndrome and autoantibody accompaniments of germ cell tumors. HANDBOOK OF CLINICAL NEUROLOGY 2024; 200:431-445. [PMID: 38494295 DOI: 10.1016/b978-0-12-823912-4.00001-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 03/19/2024]
Abstract
Paraneoplastic neurologic syndromes (PNSs) are a group of diseases affecting the central and/or peripheral nervous system caused by immune-mediated processes directed toward antigens with shared expression in tumor and neural tissue. Germ cell tumors (GCTs) are associated with PNSs with varied clinical phenotypes. Early diagnosis of PNS is vital to potentially uncover and treat underlying tumors, improving the chances of recovery, and preventing permanent neurologic complications. In this chapter, we outline the pathophysiology and epidemiology of PNS. We briefly provide a summary of GCTs in males and females. We review the neural-specific autoantibodies and PNSs associated with GCTs and their clinical and radiologic accompaniments. We also provide an overview of the treatment and prognosis of these disorders.
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Affiliation(s)
- M Bakri Hammami
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States; Jacobi Medical Center/Albert Einstein College of Medicine, Bronx, NY, United States
| | - Mohamed Rezk
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States; Department of Neurology, Mayo Clinic, Rochester, MN, United States
| | - Divyanshu Dubey
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, United States; Department of Neurology, Mayo Clinic, Rochester, MN, United States.
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Zhang D, Xu H, Zhao C, Qin L, Wei R, Xi L, Li F. Detailed characterization of PD-1/PD-L1 and CTLA4 expression and tumor-infiltrating lymphocytes in yolk sac tumors. Hum Immunol 2023; 84:534-542. [PMID: 37453913 DOI: 10.1016/j.humimm.2023.07.003] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/11/2023] [Revised: 07/10/2023] [Accepted: 07/10/2023] [Indexed: 07/18/2023]
Abstract
BACKGROUND Immune checkpoint blockade (ICB) is considered as a promising approach for cancer treatment. However, the potency of ICB therapy in yolk sac tumors (YSTs) has not been confirmed, and the comprehensive analysis of tumor immune microenvironment and the expression of PD-1/PD-L1 and CTLA4 were also not thoroughly evaluated. METHODS Immunohistochemistry was performed in formalin-fixed, paraffin-embedded tumor specimens from 23 YSTs patients to detect the density and distribution of tumor-infiltrating T cells, tertiary lymphoid structures (TLSs), as well as the expression of PD-1/PD-L1 and CTLA4. RESULTS Overall, more than half (61 %) of all patients exhibited an immune-desert phenotype based on CD3+ T cells. PD-1 expression was identified in five tumor samples (21.7 %), and PD-L1 expression exhibited a different positive rate in tumor cells (TCs) and tumor-infiltrating lymphocytes (TILs) (39.1 % and 17.4 %). Noteworthily, the rate of positive CTLA4 expression in both TCs and TILs was markedly higher (69.6 % and 56.5 %) than those of PD-1 and PD-L1 expression. Furthermore, TLSs were observed in 21.74 % of all tissues, and samples with TLSs exhibited significantly higher densities of TILs and higher expression of immune checkpoint molecules, particularly PD-1/PD-L1. In addition, tumors located in testes also exhibited a higher density of TILs and higher expression of immune checkpoint molecules. CONCLUSION Generally a high frequency of CTLA4 expression was found, PD-1/PD-L1 expression, the immune-inflamed phenotype, and TLSs were low frequency in YSTs, however, YSTs in testes showed a higher density of TILs and higher expression of immune checkpoint molecules.
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Affiliation(s)
- Danya Zhang
- Cancer Biology Research Center (Key Laboratory of the Ministry of Education), Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China; Department of Gynecologic Oncology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China
| | - Hanjie Xu
- Cancer Biology Research Center (Key Laboratory of the Ministry of Education), Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China; Department of Gynecologic Oncology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China
| | - Can Zhao
- Department of Pathology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China
| | - Lingzhi Qin
- Department of Pathology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China
| | - Rui Wei
- Cancer Biology Research Center (Key Laboratory of the Ministry of Education), Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China; Department of Gynecologic Oncology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China
| | - Ling Xi
- Cancer Biology Research Center (Key Laboratory of the Ministry of Education), Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China; Department of Gynecologic Oncology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China
| | - Fei Li
- Cancer Biology Research Center (Key Laboratory of the Ministry of Education), Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China; Department of Gynecologic Oncology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430030, China.
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Khaled SS, Soliman HA, Abdel-Gabbar M, Ahmed NA, El-Nahass ES, Ahmed OM. Naringin and naringenin counteract taxol-induced liver injury in Wistar rats via suppression of oxidative stress, apoptosis and inflammation. ENVIRONMENTAL SCIENCE AND POLLUTION RESEARCH INTERNATIONAL 2023; 30:90892-90905. [PMID: 37466839 PMCID: PMC10439847 DOI: 10.1007/s11356-023-28454-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Subscribe] [Scholar Register] [Received: 03/02/2023] [Accepted: 06/22/2023] [Indexed: 07/20/2023]
Abstract
This research aimed to evaluate the preventing effects of naringin, naringenin, and their combination on liver injury induced by Taxol (paclitaxel) in Wistar rats. Male Wistar rats received 2 mg/kg Taxol intraperitoneal injections twice weekly on the second and fifth days of each week for 6 weeks. During the same period as Taxol administration, rats were given naringin, naringenin, or a combination of the two (10 mg/kg b.wt) every other day. Treatment with naringin and/or naringenin reduced the abnormally high serum levels of total bilirubin, aspartate transaminase, alanine transaminase, alkaline phosphatase, lactate dehydrogenase, and gamma-glutamyl transferase in Taxol-treated rats. It also significantly increased the level of serum albumin, indicating an improvement in the liver. The perturbed histological liver changes were markedly improved due to the naringin and/or naringenin treatment in Taxol-administered rats. Additionally, the treatments reduced high hepatic lipid peroxidation and increased liver glutathione content as well as the activities of superoxide dismutase and glutathione peroxidase. Furthermore, the treatments reduced the levels of alpha-fetoprotein and caspase-3, a pro-apoptotic mediator. The naringin and naringenin mixture appeared more effective in improving organ function and structural integrity. In conclusion, naringin and naringenin are suggested to employ their hepatoprotective benefits via boosting the body's antioxidant defense system, reducing inflammation, and suppressing apoptosis.
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Affiliation(s)
- Shimaa S. Khaled
- Biochemistry Department, Faculty of Science, Beni-Suef University, P.O. Box 62521, Beni-Suef, Egypt
| | - Hanan A. Soliman
- Biochemistry Department, Faculty of Science, Beni-Suef University, P.O. Box 62521, Beni-Suef, Egypt
| | - Mohammed Abdel-Gabbar
- Biochemistry Department, Faculty of Science, Beni-Suef University, P.O. Box 62521, Beni-Suef, Egypt
| | - Noha A. Ahmed
- Physiology Division, Zoology Department, Faculty of Science, Beni-Suef University, P.O. Box 62521, Beni-Suef, Egypt
| | - El-Shaymaa El-Nahass
- Department of Pathology, Faculty of Veterinary Medicine, Beni-Suef University, P.O. Box 62521, Beni-Suef, Egypt
| | - Osama M. Ahmed
- Physiology Division, Zoology Department, Faculty of Science, Beni-Suef University, P.O. Box 62521, Beni-Suef, Egypt
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Lu H, Jia D, Tang Q, Shu S. Primary endodermal sinus tumor originating from the sacral ligament: a case report and review of the literature. BMC Pregnancy Childbirth 2023; 23:528. [PMID: 37474890 PMCID: PMC10357729 DOI: 10.1186/s12884-023-05849-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/16/2023] [Accepted: 07/12/2023] [Indexed: 07/22/2023] Open
Abstract
BACKGROUND Endodermal sinus tumor (EST) is a malignant tumor originating from the ovary or testis. In most case, ultrasound examination shows ovarian mass. But there is a special kind of extra-gonadal endodermal sinus tumor, which occur in organs other than gonads with insidious onset. Here we reported a case of endodermal sinus tumor, which originated from the sacral ligament presenting as an acute lower abdominal pain. CASE PRESENTATION A 14-year-old girl was admitted to the hospital because of acute lower abdominal pain. The ultrasound showed a mass with 72 mm × 64 mm × 50 mm in Douglas, and there was no abnormality in bilateral ovaries and fallopian tubes. Laparoscopic exploration showed a large amount of blood clots in the pelvic cavity. After removal of the blood, we found rotten fish-like tissue in the left sacral ligament, rapid pathology suggested endodermal sinus tumor. After the operation, we retrospectively examined the value of alpha-fetoprotein (AFP), which was found to be elevated, and post-operative paraffin pathology confirmed the diagnosis. After four cycles of BEP chemotherapy, exploratory laparotomy was performed to remove the visible lesion, but postoperative pathology showed no abnormality. At the one-year follow-up, the patient remained recurrence-free. CONCLUSION Extra-gonadal germ cell tumors are rarely reported. When young teenagers complain of acute lower abdominal pain with elevated AFP, but there was no lesion in bilateral ovaries and fallopian tubes, we must think about the possibility of endodermal sinus tumors. Accurate diagnosis facilitates complete resection of lesions and improves patient's outcomes.
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Affiliation(s)
- Han Lu
- Department of Gynecology and Obstetrics, The First Affiliated Hospital of Jinan University Guangzhou, Guangzhou, 510630, Guangdong, People's Republic of China
| | - Dongsong Jia
- Department of Gynecology and Obstetrics, The First Affiliated Hospital of Jinan University Guangzhou, Guangzhou, 510630, Guangdong, People's Republic of China
| | - Qionglan Tang
- Department of Pathology, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, 510120, People's Republic of China
| | - Shanrong Shu
- Department of Gynecology and Obstetrics, The First Affiliated Hospital of Jinan University Guangzhou, Guangzhou, 510630, Guangdong, People's Republic of China.
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Ovarian yolk sac tumor in a premenarchal girl. Wien Med Wochenschr 2023; 173:70-73. [PMID: 36637654 DOI: 10.1007/s10354-022-00996-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/26/2022] [Accepted: 11/25/2022] [Indexed: 01/14/2023]
Abstract
Yolk sac tumors are highly malignant and commonly affect the ovaries, with a median age of occurrence of 23 years. We describe the case of an ovarian yolk sac tumor in a 12-year-old premenarchal girl suffering from Hashimoto's thyroiditis and chronic spontaneous urticaria, which presented as a rapidly growing solid cystic formation in the hypogastrium with an extreme increase in alpha fetoprotein (52,778 mg/ml). After ultrasound and MRI imaging, fertility-sparing staging surgery was performed, and the diagnosis of an ovarian yolk sac tumor with positive malignant cells in ascites was confirmed. The specificity of this case is the tumor classification into stage IC3 according to the FIGO and stage III according to the Children's Oncology Group criteria. The postoperative course was complicated by a pelvic abscess and a subcutaneous suture rejection reaction. Our case may incite further research on the relationship between autoimmunity and yolk sac tumors.
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Feasibility of Fertility-Sparing Surgery After Neoadjuvant Chemotherapy for Advanced Malignant Germ Cell Tumor of the Ovary. INDIAN JOURNAL OF GYNECOLOGIC ONCOLOGY 2022. [DOI: 10.1007/s40944-022-00673-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
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Gică N, Peltecu G, Chirculescu R, Gică C, Stoicea MC, Serbanica AN, Panaitescu AM. Ovarian Germ Cell Tumors: Pictorial Essay. Diagnostics (Basel) 2022; 12:diagnostics12092050. [PMID: 36140449 PMCID: PMC9498179 DOI: 10.3390/diagnostics12092050] [Citation(s) in RCA: 7] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/19/2022] [Revised: 08/11/2022] [Accepted: 08/19/2022] [Indexed: 11/16/2022] Open
Abstract
Ovarian germ cell tumors of the ovary represent a histologically heterogenous group of tumors with a high incidence at reproductive age. Patients with this pathology are very often young women with amenorrhea. The aim of this article is to present a pictorial essay of this rare pathology and to promote a national tumor registry and protocol. The treatment is individualized according to age, and fertility-sparing surgery is the actual standard of surgical treatment for young patients in early stage of the disease.
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Affiliation(s)
- Nicolae Gică
- Department of Obstetrics and Gynecology, Carol Davila University of Medicine and Pharmacy, 020021 Bucharest, Romania
- Filantropia Clinical Hospital, 011132 Bucharest, Romania
| | - Gheorghe Peltecu
- Department of Obstetrics and Gynecology, Carol Davila University of Medicine and Pharmacy, 020021 Bucharest, Romania
- Filantropia Clinical Hospital, 011132 Bucharest, Romania
- Correspondence:
| | - Raluca Chirculescu
- Department of Obstetrics and Gynecology, Carol Davila University of Medicine and Pharmacy, 020021 Bucharest, Romania
- Filantropia Clinical Hospital, 011132 Bucharest, Romania
| | - Corina Gică
- Department of Obstetrics and Gynecology, Carol Davila University of Medicine and Pharmacy, 020021 Bucharest, Romania
- Filantropia Clinical Hospital, 011132 Bucharest, Romania
| | | | - Andreea Nicoleta Serbanica
- Department of Pediatrics, Fundeni Clinical Institute, Department of Pediatrics Hematology, Carol Davila University of Medicine and Pharmacy, 020021 Bucharest, Romania
| | - Anca Maria Panaitescu
- Department of Obstetrics and Gynecology, Carol Davila University of Medicine and Pharmacy, 020021 Bucharest, Romania
- Filantropia Clinical Hospital, 011132 Bucharest, Romania
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12
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Qin L, Wang B, Wang Z, He D. Clinicopathological features, prognosis, and fertility outcomes in Chinese Han women treated for ovarian yolk sac tumor: A retrospective case series study from two tertiary-care academic medical centers. Medicine (Baltimore) 2022; 101:e29868. [PMID: 35866762 PMCID: PMC9302259 DOI: 10.1097/md.0000000000029868] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/04/2023] Open
Abstract
OBJECTIVE Ovarian yolk sac tumor (YST) is a very rare malignant tumor in young women. This study aimed to explore the clinicopathological prognostic characteristics and reproductive outcomes of Chinese Han patients. METHODS To describe a case series study, we reviewed the clinicopathological data of 50 YST patients treated from 2 tertiary medical academic medical centers from January 2009 to December 2019. The Akaike information criterion was used to select variables. The influence of relevant characteristics on prognosis factors was analyzed by the Cox proportional hazard model. RESULTS The median follow-up time was 64.5 months (range from 3 to 124 months). The median age was 22.7 years (3 to 34 years). Abdominal pain (54.0%) or mass (42.0%) were the most common clinical symptoms in the early stage of diagnosis. The tumors were located bilaterally in 4 cases. 27 patients, 7 patients, 13 patients, and 3 patients were in stage I, II, III, and IV, respectively. Twenty-one stage I patients and 12 stage II to IV patients underwent fertility-preserving surgery. Of the 50 patients who received postoperative chemotherapy, 49 received the BEP regimen. At the last follow-up, 92% of the patients were still alive. The overall survival rate and disease-free survival rate were 91.6% and 90.6%, respectively. Recurrence occurred in 7 (14%) patients with a median survival time of 16.7 months (range from 3 to 50 months). Six patients had recurrence in the abdominal space. The percentage of Ki67 (P = .01) and tumor size (P = .03) were 2 important prognostic factors in multivariate analysis. In terms of survival outcomes, fertility-preserving surgery can be equivalent to radical surgery. Sixteen patients tried to conceive, and 6 patients with advanced-stage succeeded in 10 pregnancies. Of these, 6 patients successfully gave birth to 6 healthy babies. CONCLUSIONS The diagnosis of YST of childbearing age is very rare. Because the failure of primary treatment is related to the residual disease after salvage surgery, the fertility and survival results of patients undergoing fertility-preserving surgery are promising.
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Affiliation(s)
- Li Qin
- Department of Obstetrics and Gynecology, The Central Hospital of Enshi Tujia and Miao Autonomous prefecture, Enshi, Hubei, China
| | - Bo Wang
- Department of Obstetrics and Gynecology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
| | - Zaiping Wang
- Department of Obstetrics and Gynecology, The Central Hospital of Enshi Tujia and Miao Autonomous prefecture, Enshi, Hubei, China
| | - Du He
- Department of Obstetrics and Gynecology, The Central Hospital of Enshi Tujia and Miao Autonomous prefecture, Enshi, Hubei, China
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Young RH, Wong A, Stall JN. Yolk Sac Tumor of the Ovary: A Report of 150 Cases and Review of the Literature. Am J Surg Pathol 2022; 46:309-325. [PMID: 34469331 DOI: 10.1097/pas.0000000000001793] [Citation(s) in RCA: 10] [Impact Index Per Article: 3.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
Abstract
One hundred fifty yolk sac tumors (YSTs) of the ovary in patients from 1 to 61 (mean: 21.5) years of age are described; 75% of the patients were in the second and third decades and only 1 was above 50 years of age. The clinical manifestations were typically related to a fast-growing adnexal mass; endocrine manifestations (hirsutism) were present in only 2 cases. The tumors were all unilateral and 70% were ≥15 cm; an associated dermoid cyst was present in 20 cases. The tumors were solid and cystic in 57% of the cases, 25% were multicystic, and 18% uniformly solid. The solid tissue was typically tan to pink or yellow and often friable with hemorrhage and necrosis; smaller solid neoplasms were sometime uniformly yellow. The most common histologic pattern was reticular composed of an irregular meshwork of spaces that was conspicuous in 68% of the neoplasms but present to at least a minor degree in all of them. That appearance almost always merged with small to large cysts that were prominent in about 40% of tumors. In 25% of the tumors, cysts sometimes associated with a cellular stroma (the polyvesicular pattern), were present but conspicuous in only half these cases. One third of the tumors had a labyrinthine pattern, 22% glands, and 6% a festoon pattern. Papillae with a central blood vessel (Schiller-Duval bodies) were seen in one-third of the tumors but were numerous in only 5% of them. Nonspecific appearing papillae were seen in 10% of the tumors. A solid growth of cells with pale cytoplasm was seen in one-third of the tumors but was conspicuous in only half of that subset. The solid appearance was typically reminiscent of that of dysgerminoma, but lacked the septa and lymphocytic infiltrate of that neoplasm. Nine tumors had a component of cells with scant cytoplasm resulting in a blastema-like appearance and 3 had cells with abundant clear cytoplasm. Cords and clusters of cells were common but did not dominate the microscopic appearance. The stroma typically had a nonspecific collagenous to edematous appearance. Stromal luteinization was seen in 12 tumors; in 5 this was likely due to the patient being pregnant. Two tumors had minor foci of cells that resembled hepatocytes. Hyaline bodies were seen in most of the tumors and were often conspicuous. The neoplastic cells typically had modest amounts of lightly staining cytoplasm and only mild nuclear pleomorphism. Cells lining cysts were often flattened sometimes resulting in a deceptively innocuous appearance. Many of the tumors (mostly consultation cases), caused diagnostic difficulty; tumors in the differential diagnosis included clear cell carcinoma, embryonal carcinoma, Sertoli-Leydig cell tumor, and juvenile granulosa cell tumor. The patient age and marked elevation of the serum alpha-fetoprotein level (if measured) is helpful in many of these considerations. The overtly malignant gross appearance of most YSTs contrasts with certain other tumors in the differential and the association of some YSTs with dermoid cyst and many clear cell carcinomas with endometriosis may be helpful. The vast majority of ovarian YSTs are dominated microscopically by merging of reticular and cystic patterns which, although focally mimicked by other neoplasms, are in general characteristic, and distinctive features of other neoplasms are absent. Immunohistochemistry, particularly for alpha-fetoprotein and glypican 3, and lack of staining for various markers of other neoplasms is helpful but overlap exists and these results must be considered in the context of the overall clinical, gross, and microscopic features. YSTs dominated by hepatoid and glandular features are rare and their categorization as YSTs should be done cautiously if thoroughly sampled tumors show no evidence of classic features of YST emphasized herein and first elaborated by the Danish investigator Gunnar Teilum whose seminal observations have stood the test of time.
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Affiliation(s)
- Robert H Young
- James Homer Wright Pathology Laboratories, Massachusetts General Hospital, Harvard Medical School, Boston, MA
| | - Adele Wong
- James Homer Wright Pathology Laboratories, Massachusetts General Hospital, Harvard Medical School, Boston, MA
| | - Jennifer N Stall
- Hospital Pathology Associates, Virginia Piper, Cancer Institute, Minneapolis, MN
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14
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Ma X, Cao D, Peng P, Xiao Y, Yang J, Huang H, Zhang Y, Yu M, Wang J, Zhou H, Cheng N, Wang T, Shen K. Preservation of sexual and reproductive function in the treatment of extragonadal yolk sac tumors in the female genital tract. Front Pediatr 2022; 10:1004501. [PMID: 36452350 PMCID: PMC9701820 DOI: 10.3389/fped.2022.1004501] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/27/2022] [Accepted: 10/10/2022] [Indexed: 11/15/2022] Open
Abstract
OBJECTIVE This study aimed to summarize the clinical features, treatment modalities, therapeutic effects, menstruation and fertility outcomes, and prognosis of extragonadal yolk sac tumors (YSTs) of the female genital tract. METHODS We reviewed 32 cases of extragonadal YSTs in the genital tract treated between 1983 and 2021. The medical records, including clinical characteristics, histopathology, treatments, chemo-reduced adverse events, and outcomes on long-term follow-up, were collected. RESULTS Among the 32 cases, 30 were vaginal YSTs and two were uterine YSTs (endometrial and cervical). Thirty patients (30/32, 93.8%) were <4 years. Abnormal vaginal bleeding (n = 31) and elevated serum alpha-fetoprotein level (n = 32) were the most common presentations. Vaginohysteroscopy and/or pediatric rhinoscopy were used for diagnosis in 17 pediatric patients and evaluation of chemotherapeutic efficacy in 21 pediatric patients. All the patients received combination chemotherapy. Bleomycin/etoposide/cisplatin (BEP) was chosen with prior consideration in 28 cases; 21 patients were treated with BEP alone. Yellow or grayish-yellow tissue with irregular shape was found in 66.7% of the cases during repeat examinations. Five patients underwent surgeries during repeat examinations and follow-ups, and no evidence of malignancy was noted in them. Thirty-one patients achieved complete remission. During a median follow-up of 63 months (2.4-240.3 months), two patients experienced recurrence, three died, and 29 remained disease-free. One patient recovered menstruation and five had undergone menarche. CONCLUSION BEP chemotherapy can serve as a preferred treatment modality for vaginal and uterine YSTs. Vaginohysteroscopy and pediatric rhinoscopy can be used for diagnosis and evaluation of chemotherapeutic efficacy in pediatric patients. YSTs possibly appear as yellow or grayish-yellow after chemotherapy.
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Affiliation(s)
- Xiao Ma
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Dongyan Cao
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Peng Peng
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Yu Xiao
- Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China
| | - Jiaxin Yang
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Huifang Huang
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Ying Zhang
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Mei Yu
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Jinhui Wang
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Huimei Zhou
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Ninghai Cheng
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Tao Wang
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Keng Shen
- Department of Obstetrics and Gynecology, National Clinical Research Center for Obstetric & Gynecologic Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
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15
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Afectación pulmonar extensa como presentación de un tumor del saco vitelino ovárico. OPEN RESPIRATORY ARCHIVES 2022. [PMID: 37497311 PMCID: PMC10369609 DOI: 10.1016/j.opresp.2022.100156] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/18/2022] Open
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Grindstaff S, Hanley LC, Banet N. Yolk Sac Tumor Arising in Uterine Carcinosarcoma With Endometrioid and Serous Components: A Case Report With Immunohistochemical Analysis. Int J Gynecol Pathol 2022; 41:40-44. [PMID: 33770060 DOI: 10.1097/pgp.0000000000000774] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Abstract
Yolk sac tumor of the endometrium is an uncommon neoplasm. Here we report a case of yolk sac tumor arising in a uterine carcinosarcoma, with the carcinomatous component showing both endometrioid and serous components, and the sarcomatous component showing homologous (spindled) differentiation. The yolk sac tumor showed predominant glandular configuration and was present admixed with the epithelial components. Extensive immunostaining was performed to narrow the differential diagnosis, including potentially therapeutic testing for HER-2. To our knowledge, this is the first case of carcinosarcoma with this mix of epithelial components and corresponding reporting of these immune and therapeutic markers.
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17
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Xie CL, Peng CR, Yan JX, Wang LX, Zhou P. Ovarian yolk sac tumor in a patient with sexual differentiation disorder: a case description. Quant Imaging Med Surg 2021; 11:3360-3366. [PMID: 34249660 DOI: 10.21037/qims-20-890] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/21/2020] [Accepted: 12/02/2020] [Indexed: 11/06/2022]
Affiliation(s)
- Chao Lian Xie
- Department of Radiology, Sichuan Cancer Hospital and Institute, Sichuan Cancer Center, School of Medicine, University of Electronic Science and Technology of China, Chengdu, China
| | - Chun Rong Peng
- Department of Gynecologic Oncology, Sichuan Cancer Hospital and Institute, Sichuan Cancer Center, School of Medicine, University of Electronic Science and Technology of China, Chengdu, China
| | - Jia Xin Yan
- Department of Pathology, Sichuan Cancer Hospital and Institute, Sichuan Cancer Center, School of Medicine, University of Electronic Science and Technology of China, Chengdu, China
| | - Lin Xia Wang
- Department of Radiology, Chengdu First People's Hospital, Chengdu Integrated Traditional and Western Medicine Hospital, Chengdu, China
| | - Peng Zhou
- Department of Radiology, Sichuan Cancer Hospital and Institute, Sichuan Cancer Center, School of Medicine, University of Electronic Science and Technology of China, Chengdu, China
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18
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Oh HK, Park SN, Kim BR. Laparoscopic uncontained power morcellation-induced dissemination of ovarian endodermal sinus tumors: A case report. World J Clin Cases 2021; 9:3212-3218. [PMID: 33969110 PMCID: PMC8080755 DOI: 10.12998/wjcc.v9.i13.3212] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/31/2020] [Revised: 02/03/2021] [Accepted: 03/12/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Endodermal sinus tumors (ESTs), which arise primarily in children and adolescents, account for 20% of malignant ovarian germ cell tumors, but constitute only 1% of all ovarian malignancies. Treatment of ESTs consists of surgical staging with fertility-sparing surgery and chemotherapy.
CASE SUMMARY A 15-year-old nulliparous patient was diagnosed with disseminated ovarian ESTs after laparoscopic unilateral salpingo-oophorectomy using uncontained power morcellation for treatment of a ruptured solid adnexal mass in another hospital. Exploratory laparotomy; total abdominal hysterectomy, right salpingo-oophorectomy, and lymphadenectomy were performed with optimal debulking, and surgical stage 3C was assigned to the patient.
CONCLUSION In 2014, the Food and Drug Administration noted that power morcellation was probably associated with a risk of disseminating suspected cancerous tissue. Furthermore, the use of power morcellation to remove solid adnexal mass is considered a contraindication because of the potential for a malignant tumor. This case report aims to warn of the dangers of using uncontained power morcellation to treat solid adnexal masses.
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Affiliation(s)
- Ha Kyung Oh
- Department of Obstetrics and Gynecology, Wonkwang University Hospital, Iksan 54645, Jeollabuk-do, South Korea
| | - Seong Nam Park
- Department of Obstetrics and Gynecology, Wonkwang University Hospital, Iksan 54645, Jeollabuk-do, South Korea
| | - Byoung Ryun Kim
- Department of Obstetrics and Gynecology, Wonkwang University Hospital, Iksan 54645, Jeollabuk-do, South Korea
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19
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Anfelter P, Testa A, Chiappa V, Froyman W, Fruscio R, Guerriero S, Alcazar JL, Mascillini F, Pascual MA, Sibal M, Savelli L, Zannoni GF, Timmerman D, Epstein E. Imaging in gynecological disease (17): ultrasound features of malignant ovarian yolk sac tumors (endodermal sinus tumors). ULTRASOUND IN OBSTETRICS & GYNECOLOGY : THE OFFICIAL JOURNAL OF THE INTERNATIONAL SOCIETY OF ULTRASOUND IN OBSTETRICS AND GYNECOLOGY 2020; 56:276-284. [PMID: 32119168 DOI: 10.1002/uog.22002] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/12/2019] [Revised: 02/02/2020] [Accepted: 02/24/2020] [Indexed: 06/10/2023]
Abstract
OBJECTIVE To describe the clinical and sonographic characteristics of malignant ovarian yolk sac tumors (YSTs). METHODS In this retrospective multicenter study, we included 21 patients with a histological diagnosis of ovarian YST and available transvaginal ultrasound images and/or videoclips and/or a detailed ultrasound report. Ten patients identified from the International Ovarian Tumor Analysis (IOTA) studies had undergone a standardized preoperative ultrasound examination, by an experienced ultrasound examiner, between 1999 and 2016. A further 11 patients were identified through medical files, for whom ultrasound images were retrieved from local image workstations and picture archiving and communication systems. All tumors were described using IOTA terminology. The collected ultrasound images and videoclips were used by two observers for additional characterization of the tumors. RESULTS All cases were pure YSTs, except for one that was a mixed tumor (80% YST and 20% embryonal carcinoma). Median age at diagnosis was 25 (interquartile range (IQR), 19.5-30.5) years. Seventy-six percent (16/21) of women had an International Federation of Gynecology and Obstetrics (FIGO) Stage I-II tumor at diagnosis. Fifty-eight percent (11/19) of women felt pain during the ultrasound examination and one presented with ovarian torsion. Median serum α-fetoprotein (S-AFP) level was 4755 (IQR, 1071-25 303) µg/L and median serum CA 125 level was 126 (IQR, 35-227) kU/L. On ultrasound assessment, 95% (20/21) of tumors were unilateral. The median maximum tumor diameter was 157 (IQR, 107-181) mm and the largest solid component was 110 (IQR, 66-159) mm. Tumors were classified as either multilocular-solid (10/21; 48%) or solid (11/21; 52%). Papillary projections were found in 10% (2/21) of cases. Most (20/21; 95%) tumors were well vascularized (color score, 3-4) and none had acoustic shadowing. Malignancy was suspected in all cases, except in the patient with ovarian torsion, who presented a tumor with a color score of 1, which was classified as probably benign. Image and videoclip quality was considered as adequate in 18/21 cases. On review of the images and videoclips, we found that all tumors contained both solid components and cystic spaces, and that 89% (16/18) had irregular, still fine-textured and slightly hyperechoic solid tissue, giving them a characteristic appearance. CONCLUSION Malignant ovarian YSTs are often detected at an early stage, in young women usually in the second or third decade of life, presenting with pain and markedly elevated S-AFP. On ultrasound, malignant ovarian YSTs are mostly unilateral, large and multilocular-solid or solid, with fine-textured slightly hyperechoic solid tissue and rich vascularization. © 2020 The Authors. Ultrasound in Obstetrics & Gynecology published by John Wiley & Sons Ltd on behalf of the International Society of Ultrasound in Obstetrics and Gynecology..
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Affiliation(s)
- P Anfelter
- Department of Obstetrics and Gynecology, Karolinska Institutet, Sodersjukhuset, Stockholm, Sweden
- Department of Clinical Science and Education, Karolinska Institutet, Sodersjukhuset, Stockholm, Sweden
| | - A Testa
- Department of Gynecological Oncology, Catholic University of Sacred Heart, Rome, Italy
| | - V Chiappa
- Department of Obstetrics and Gynecology, National Cancer Institute, Milan, Italy
| | - W Froyman
- Department of Obstetrics and Gynecology, University Hospital KU Leuven, Leuven, Belgium
| | - R Fruscio
- Clinic of Obstetrics and Gynecology, University of Milan Bicocca, San Gerardo Hospital, Monza, Italy
| | - S Guerriero
- Department of Obstetrics and Gynecology, University of Cagliari, Policlinico Universitario Duilio Casula, Monserrato, Cagliari, Italy
| | - J L Alcazar
- Department of Obstetrics and Gynecology, Clinica Universidad de Navarra, Pamplona, Spain
| | - F Mascillini
- Department of Gynecological Oncology, Catholic University of Sacred Heart, Rome, Italy
| | - M A Pascual
- Department of Obstetrics, Gynecology, and Reproduction, Hospital Universitario Dexeus, Barcelona, Spain
| | - M Sibal
- Department of Obstetrics and Gynecology, Manipal Hospital, Bangalore, India
| | - L Savelli
- Department of Obstetrics and Gynecology, Policlinico S. Orsola-Malpighi, Bologna, Italy
| | - G F Zannoni
- Department of Pathology, Catholic University of the Sacred Heart, Rome, Italy
| | - D Timmerman
- Department of Obstetrics and Gynecology, University Hospital KU Leuven, Leuven, Belgium
| | - E Epstein
- Department of Obstetrics and Gynecology, Karolinska Institutet, Sodersjukhuset, Stockholm, Sweden
- Department of Clinical Science and Education, Karolinska Institutet, Sodersjukhuset, Stockholm, Sweden
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Ovarian solid tumors: MR imaging features with radiologic-pathologic correlation. Jpn J Radiol 2020; 38:719-730. [PMID: 32342277 DOI: 10.1007/s11604-020-00976-8] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/27/2020] [Accepted: 04/08/2020] [Indexed: 10/24/2022]
Abstract
Ovarian solid tumors have variable histological types including benign and malignant tumors. In addition, non-neoplastic lesions sometimes show a tumor-like appearance. It is important to differentiate benign from malignant tumors. In general, low signal intensity (SI) on T2-weighted imaging (T2WI), low SI on diffusion-weighted imaging (DWI), and gradual increased pattern on dynamic contrast-enhanced magnetic resonance (MR) imaging are known to be suggestive of a benign tumor. Conversely, there are some cases in which these rules do not apply. We should, therefore, strive for a greater understanding of these exceptional cases. Several tumors show characteristic findings on MR imaging reflecting pathologic features, which leads to the correct diagnosis. Additionally, MR imaging provides important information other than the nature of tumors, such as secondary uterine changes. Furthermore, clinical findings and laboratory examination data also help in determining the correct diagnosis.
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21
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Perry MF, Jackson AL, Herzog TJ, Billingsley CC. Early diagnosis and treatment challenges of endodermal sinus tumors: A case report. Case Rep Womens Health 2020; 27:e00198. [PMID: 32300538 PMCID: PMC7152713 DOI: 10.1016/j.crwh.2020.e00198] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/02/2020] [Revised: 03/30/2020] [Accepted: 04/02/2020] [Indexed: 11/24/2022] Open
Abstract
Background Ovarian endodermal sinus tumors (ESTs) are rapidly growing and highly malignant tumors that respond well to chemotherapy. They can be difficult to diagnose and delayed diagnosis can worsen prognosis. Case We present the case of a 20-year-old woman with an EST initially misdiagnosed as a tubo-ovarian abscess who then experienced rapid progression within weeks of initial presentation and was subsequently found to have unresectable advanced stage disease. Conclusion ESTs are extremely aggressive and require prompt referral and early treatment with chemotherapy. Presenting symptoms of pain and a mass can lead to a broad range of differential diagnoses. In such patients, early consideration of tumor markers is warranted. This case report reviews the key aspects for prompt diagnosis and rapid treatment of these tumors, which significantly impacts the prognosis.
Delayed diagnosis of endodermal sinus tumors can worsen prognosis. Certain radiological signs are specific for endodermal sinus tumors. Broad differentials and early tumor markers may decrease time to diagnosis.
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Affiliation(s)
- Madeline F Perry
- University of Cincinnati College of Medicine, Cincinnati, OH, United States
| | - Amanda L Jackson
- Division of Gynecologic Oncology, University of Cincinnati, Cincinnati, OH, United States
| | - Thomas J Herzog
- Division of Gynecologic Oncology, University of Cincinnati, Cincinnati, OH, United States
| | - Caroline C Billingsley
- Division of Gynecologic Oncology, University of Cincinnati, Cincinnati, OH, United States
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22
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Stefanelli E, Talarico V, Scavone M, Carboni E, Stranieri G, Galati MC, Salerno D, Raiola G. Acute abdominal pain in an adolescent girl with an ovarian yolk sac tumor. ACTA BIO-MEDICA : ATENEI PARMENSIS 2019; 90:599-602. [PMID: 31910193 PMCID: PMC7233780 DOI: 10.23750/abm.v90i4.9017] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 11/11/2019] [Accepted: 11/12/2019] [Indexed: 11/23/2022]
Abstract
Yolk sac tumor (YST) is a rare tumor that usually occurs in the first two decades of life. It is considered the second most common malignant germ cell tumor of the ovary, characterized by a rapid growth and a bad prognosis due to the frequent metastasis. We report the case of a 12-year-old girl who came to our observation for an acute abdominal pain. Clinical examination evidenced a vague mass in the suprapubic region and a lower abdomen tenderness, the US imaging revealed a complex lesion of the left ovary (19 x 13 cm) and the alpha-fetoprotein (AFP) resulted high (5858 ng/mL). Computed tomography (CT) revealed a large pelvic mass. The treatment consisted of debulking surgery of yolk sac tumor followed by 4 cycles of BEP protocol (Bleomycin, Etoposide, Cisplatin). After 3 years of follow-up there was no evidence of disease recurrence. (www.actabiomedica.it).
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Affiliation(s)
- Ettore Stefanelli
- Department of Pediatrics, Magna Graecia University of Catanzaro, Catanzaro, Italy.
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23
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Zong X, Yang JX, Zhang Y. Persistently elevated alpha-fetoprotein associated with chronic hepatitis B during chemotherapy for malignant ovarian germ cell tumors: a case series and a review of the literature. J Ovarian Res 2019; 12:124. [PMID: 31836006 PMCID: PMC6911275 DOI: 10.1186/s13048-019-0598-x] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/05/2019] [Accepted: 12/04/2019] [Indexed: 12/17/2022] Open
Abstract
Background Alpha-fetoprotein (AFP) plays a crucial role in the management of malignant ovarian germ cell tumors (MOGCTs) and is an important reference index for chemotherapy termination. However, a high level of AFP can also be caused by several benign diseases, causing confusion and impacting treatment decisions. Case presentation We described four patients who were diagnosed with MOGCTs; the histologic subtype in two of them was mixed MOGCTs (yolk sac tumor with mature teratoma), while the rest was immature teratoma. The serum AFP level of each patient was abnormal before surgery, but it was still persistently elevated around 300 ng/ml even after additional cycles of chemotherapy. All patients were thoroughly evaluated, but we did not find any evidence of disease progression or residual tumors. Liver function tests were normal, whereas serum assays revealed positive of hepatitis B surface antigen, and two patients had a high level of HBV-DNA. They were chronic carriers of hepatitis B virus and never received relevant treatments. Then they were managed with tumor surveillance and the antiviral treatment. Thereafter, the AFP levels presented a slowly decreasing trend. Conclusions False elevation of AFP in MOGCTs is a rare condition and should be assessed with a comprehensive evaluation to avoid unnecessary treatments.
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Affiliation(s)
- Xuan Zong
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, No. 1 Shuaifuyuan, Dongcheng District, Beijing, 100730, China
| | - Jia-Xin Yang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, No. 1 Shuaifuyuan, Dongcheng District, Beijing, 100730, China.
| | - Ying Zhang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, No. 1 Shuaifuyuan, Dongcheng District, Beijing, 100730, China
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Endometrial tumors with yolk sac tumor-like morphologic patterns or immunophenotypes: an expanded appraisal. Mod Pathol 2019; 32:1847-1860. [PMID: 31375771 DOI: 10.1038/s41379-019-0341-6] [Citation(s) in RCA: 22] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/15/2019] [Revised: 07/10/2019] [Accepted: 07/10/2019] [Indexed: 11/08/2022]
Abstract
Uterine yolk sac tumors have gained increased recognition in recent years. The current study is a multi-faceted examination of yolk sac tumor-like phenotypes in endometrial tumors, based on an analysis of 3 groups of uterine tumors: Group 1: 9 endometrial tumors that had been classified as yolk sac tumor, or as having a yolk sac tumor component, were assessed with a 35-marker immunohistochemical panel, with the goal of defining their immunophenotypic spectrum; Group 2, comprised of 70 endometrial carcinomas of various histotypes, were analyzed for their expression of SALL4, Glypican-3, and AFP, to assess the specificity of these markers for yolk sac tumors relative to endometrial carcinomas; Group 3, comprised of 626 archived cases of endometrial carcinoma/carcinosarcoma, reviewed to define the frequency of yolk sac tumor-like morphology therein. Yolk sac tumor areas in the Group 1 cases were consistently immunoreactive for SALL4 and Glypican-3; variably positive for AFP (89%), Villin (89%), PLAP (78%), 34βE12 (67%), CAM 5.2 (62.5%), EMA (56%), CD117 (50%), p16 (50%), CDX2 (44%), p53 (44% aberrant), MOC31 (37.5%), CK7 (33%), GATA3 (33%), CK5 (25%), and PAX8 (11%); and were negative for CD30, Napsin A, OCT4, estrogen, androgen, and progesterone receptors. 29 (41%) of the 70 group-2 cases expressed at least one of the 3 markers, and 96% of the positive cases was a high-grade histotype. Glypican-3, SALL4, and AFP were positive in 30, 20, and 2.8% of group-2 cases respectively; however, co-expression of any 2, or all 3 markers was uncommon (<9 and 1.4% of cases respectively). Potential yolk sac tumor-like morphology was identified in 5 (0.8%) of 626 group-3 cases, and three were ultimately deemed to be true yolk sac tumor phenotypes based on their morphologic and immunophenotypic similarity to the group 1 cases. These findings highlight the broad immunophenotypic spectrum of uterine yolk sac tumors, the potential pitfalls associated with using immunophenotypes alone to define yolk sac tumor differentiation in endometrial carcinoma, and the utility and limitations of morphologic assessment to identify yolk sac tumors at this site.
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Prognostic factors and oncological outcomes of ovarian yolk sac tumors: a retrospective multicentric analysis of 99 cases. Arch Gynecol Obstet 2019; 300:175-182. [PMID: 30982145 DOI: 10.1007/s00404-019-05160-6] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/29/2018] [Accepted: 04/09/2019] [Indexed: 10/27/2022]
Abstract
PURPOSE To investigate the clinico-pathological prognostic factors and treatment outcomes in patients with ovarian yolk sac tumors (YST). METHODS A multicenter, retrospective department database review was performed to identify patients with ovarian YST who underwent surgery between 2000 and 2017 at seven Gynecologic Oncology Centers in Turkey. RESULTS The study group consisted of 99 consecutive patients with a mean age of 23.9 years. While 52 patients had early stage (stage I-II) disease, the remaining 47 patients had advanced stage (stage III-IV) disease. The uterus was preserved in 74 (74.8%) of the cases. The absence of gross residual disease following surgery was achieved in 76.8% of the cases. Of the 54 patients with lymph node dissection (LND), lymph node metastasis was detected in 10 (18.5%) patients. Of the 99 patients, only 3 patients did not receive adjuvant therapy, and most of the patients (91.9%) received BEP (bleomycin, etoposide, cisplatin) chemotherapy. Disease recurred in 21 (21.2%) patients. The 5-year disease-free survival (DFS) and overall survival (OS) in the entire cohort were 79.2% and 81.3%, respectively. In multivariate analysis, only residual disease following initial surgery was found to be significantly associated with DFS and OS in patients with ovarian YST (p = 0.026 and p = 0.001, respectively). CONCLUSIONS Our results demonstrate the significance of achieving no visible residual disease in patients with ovarian YST. Fertility-sparing approach for patients with no visible residual disease affected neither DFS nor OS. Although high lymphatic involvement rate was detected, the benefit of LND could not be demonstrated.
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Sookram J, Levin B, Barroeta J, Kenley K, Mehta P, Krill LS. A case of ovarian endometrioid adenocarcinoma with yolk sac differentiation and Lynch syndrome. Gynecol Oncol Rep 2019; 27:60-64. [PMID: 30723761 PMCID: PMC6348978 DOI: 10.1016/j.gore.2019.01.001] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/05/2018] [Revised: 12/20/2018] [Accepted: 01/03/2019] [Indexed: 01/24/2023] Open
Abstract
Ovarian endometrioid adenocarcinoma with yolk sac component has been reported in fewer than twenty cases in the literature. A majority of the diagnoses are described in postmenopausal women without specific reference to germline genetic testing. We describe, to our knowledge, the first case in the English literature of a premenopausal woman that presented with an ovarian endometrioid adenocarcinoma with focal yolk sac component and was subsequently found to have a germline MSH2 mutation confirming a diagnosis of Lynch syndrome. Concurrent diagnosis of ovarian endometrioid adenocarcinoma with yolk sac tumor and Lynch syndrome is an extremely rare finding in a young patient and requires careful follow-up. Genetics evaluation and testing may be reasonable for individuals with this rare or mixed tumor pathology at young age of onset and can have clinical utility in guiding future cancer treatment or surveillance.
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Affiliation(s)
- Janhvi Sookram
- Department of Obstetrics and Gynecology, Division of Gynecologic Oncology, MD Anderson Cancer Center at Cooper, Cooper University Health System, Camden, NJ, USA
| | - Brooke Levin
- Division of Hematology/Medical Oncology, MD Anderson Cancer Center at Cooper, Cooper University Health System, Camden, NJ, USA
| | - Julieta Barroeta
- Department of Pathology, Cooper University Health System, Camden, NJ, USA
| | - Kathy Kenley
- Cooper University Health System, Camden, NJ, USA
| | - Pallav Mehta
- Division of Hematology/Medical Oncology, MD Anderson Cancer Center at Cooper, Cooper University Health System, Camden, NJ, USA
| | - Lauren S Krill
- Department of Obstetrics and Gynecology, Division of Gynecologic Oncology, MD Anderson Cancer Center at Cooper, Cooper University Health System, Camden, NJ, USA
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Chen LH, Yip KC, Wu HJ, Yong SB. Yolk Sac Tumor in an Eight-Year-Old Girl: A Case Report and Literature Review. Front Pediatr 2019; 7:169. [PMID: 31114774 PMCID: PMC6503144 DOI: 10.3389/fped.2019.00169] [Citation(s) in RCA: 19] [Impact Index Per Article: 3.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/23/2019] [Accepted: 04/12/2019] [Indexed: 12/22/2022] Open
Abstract
Yolk sac tumor (YST), which most frequently arises in the gonads as a type of germ cell tumor, is rare in children but is highly malignant. It has been suggested that alpha-fetoprotein (AFP) can be applied as a feasible tumor marker because its level was elevated in >90% of YST. The treatment generally involves debulking surgery of tumors followed by systemic chemotherapy. Metastasis process of YST in children is different from that in adults and thus the treatment option is required. In this study, we described a rare case of YST in terms of the clinical manifestation, imaging, and histopathology findings, diagnosis and treatment in an 8-year-old girl. Furthermore, it is important to investigate more thoroughly a patient with history of intermittent abdominal pain and fever with previously multiple accesses, because these might be the critical signs for YST that should be alarmed for early treatment. Although YST is rare in children, pediatric physicians should be aware of this and prompt treatment should be addressed.
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Affiliation(s)
- Li Hsun Chen
- Asian Institute of Tele-surgery (IRCAD-Taiwan), Chang Bing Show Chwan Memorial Hospital, Lukang, Taiwan
| | - Kui-Chuen Yip
- Division of Family Medicine, Chang Bing Show Chwan Memorial Hospital, Lukang, Taiwan
| | - Hsing-Ju Wu
- Research Assistant Center, Show Chwan Memorial Hospital, Changhua, Taiwan.,Department of Medical Research, Chang Bing Show Chwan Memorial Hospital, Lukang, Taiwan
| | - Su-Boon Yong
- Institute of Medicine, Chung Shan Medical University, Taichung, Taiwan.,Division of Pediatric Allergy, Immunology and Rheumatology, Department of Pediatrics, Show Chwan Memorial Hospital, Changhua, Taiwan.,Department of Nursing, Meiho University, Pingtung, Taiwan
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Tomao F, Di Pinto A, Sassu CM, Bardhi E, Di Donato V, Muzii L, Petrella MC, Peccatori FA, Panici PB. Fertility preservation in ovarian tumours. Ecancermedicalscience 2018; 12:885. [PMID: 30679952 PMCID: PMC6345054 DOI: 10.3332/ecancer.2018.885] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/04/2018] [Indexed: 12/14/2022] Open
Abstract
A considerable number of patients with a cancer diagnosis are of childbearing age and have not satisfied their desire for a family. Despite ovarian cancer (OC) usually occurring in older patients, 3%-14% are diagnosed at a fertile age with the overall 5-year survival rate being 91.2% in women ≤44 years of age when it is found at 1A-B stage. In this scenario, testing the safety and the efficacy of fertility sparing strategies in OC patients is very important overall in terms of quality of life. Unfortunately, the lack of randomised trials to validate conservative approaches does not guarantee the safety of fertility preservation strategies. However, evidence-based data from descriptive series suggest that in selected cases, the preservation of the uterus and at least one part of the ovary does not lead to a high risk of relapse. This conservative surgery helps to maintain organ function, giving patients of childbearing age the possibility to preserve their fertility. We hereby analysed the main evidence from the international literature on this topic in order to highlight the selected criteria for conservative management of OC patients, including healthy BRCA mutations carriers.
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Affiliation(s)
- Federica Tomao
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
| | - Anna Di Pinto
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
| | - Carolina Maria Sassu
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
| | - Erlisa Bardhi
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
| | - Violante Di Donato
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
| | - Ludovico Muzii
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
| | | | | | - Pierluigi Benedetti Panici
- Department of Gynaecological and Obstetrical Sciences and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy
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Ferraro S, Panzeri A, Braga F, Panteghini M. Serum α-fetoprotein in pediatric oncology: not a children’s tale. ACTA ACUST UNITED AC 2018; 57:783-797. [DOI: 10.1515/cclm-2018-0803] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/27/2018] [Accepted: 09/11/2018] [Indexed: 12/16/2022]
Abstract
Abstract
Background
Measurement of α-fetoprotein (AFP) concentrations in the serum of infants is useful for the management of testicular germ cell tumors, hepatoblastoma and hepatocellular carcinoma. Here, we provide a critical review of the available information about pediatric reference intervals (RI), focusing on their utility in interpreting AFP as an aid for cancer diagnosis.
Content
Evidence sources in the available literature were critically appraised. Out of 3873 retrieved papers, 24 were finally selected and carefully inspected, and six of them overcame exclusion criteria (i.e. methodological limitations in the study design, statistical gaps, drawbacks in traceability of the AFP assay to higher order materials and/or biased reporting of AFP results). Preterm and term infants up to the 3rd month of life exhibited the highest average AFP concentrations, but the attempt of defining RI by data pooling and partitioning for age intervals was impeded by the wide variability of data. The inability of defining robust RI in the first months of life made difficult, if not impossible, using upper reference limits for ruling out malignancies with a single AFP result. Evaluating the behavior of AFP concentrations 5 days from the baseline result, if this exceeds risk thresholds partitioned for age, according to the formula Xt=X0*2−t/HL (where: t=days elapsed for AFP retest; HL=AFP half-life according to age; X0=AFP baseline concentration, and Xt=predicted AFP concentration at day 5), could give a better information.
Summary
Novel studies defining AFP RI in infants based on robust methodology are warranted to improve the interpretation of AFP results in pediatric oncology. In the meantime, algorithms based on both serum AFP absolute concentrations and HL may aid in cancer diagnosis.
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Affiliation(s)
- Simona Ferraro
- UOC Patologia Clinica, Ospedale “Luigi Sacco” , Via GB Grassi 74 , Milan 20157 , Italy
- Department of Biomedical and Clinical Sciences ‘Luigi Sacco’ , University of Milan, and Clinical Pathology Unit, ASST Fatebenefratelli-Sacco , Milan , Italy
| | - Andrea Panzeri
- Department of Biomedical and Clinical Sciences ‘Luigi Sacco’ , University of Milan, and Clinical Pathology Unit, ASST Fatebenefratelli-Sacco , Milan , Italy
| | - Federica Braga
- Department of Biomedical and Clinical Sciences ‘Luigi Sacco’ , University of Milan, and Clinical Pathology Unit, ASST Fatebenefratelli-Sacco , Milan , Italy
| | - Mauro Panteghini
- Department of Biomedical and Clinical Sciences ‘Luigi Sacco’ , University of Milan, and Clinical Pathology Unit, ASST Fatebenefratelli-Sacco , Milan , Italy
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Wang Y, Yang J, Yu M, Cao D, Zhang Y, Zong X, Shen K. Ovarian yolk sac tumor in postmenopausal females: A case series and a literature review. Medicine (Baltimore) 2018; 97:e11838. [PMID: 30113473 PMCID: PMC6112915 DOI: 10.1097/md.0000000000011838] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/25/2022] Open
Abstract
RATIONALE Ovarian yolk sac tumors (YSTs) are the second most common histologic type of ovarian germ cell tumors. Most patients are adolescent and young women, while cases in postmenopausal women were rarely reported. Due to its rarity, we know little about the treatment and prognosis of postmenopausal patients with ovarian YSTs. We reported 3 cases of mixed ovarian YST in postmenopausal females reviewed the related current English literature. PATIENT CONCERNS The ages of the three patients were 61, 58 and 77 respectively. The three patients came to the hospital because of the abdominal discomfort or tenderness, and the third patient also has vaginal bleeding. DIAGNOSES Imaging examination revealed pelvic mass with cystic and solid components. The elevated serum AFP level and pathologcial examination confirmed mixed ovarian YST. INTERVENTIONS All patients received surgery and chemotherapy. Two patients received PEB (cisplatin, etoposide, and bleomycin) chemotherapy initially and one patient received TC (paclitaxel carboplatin) chemotherapy. OUTCOMES One patient relapsed 8 months after diagnosis and underwent re-cytoreductive surgery. The three patients all survived at last follow-up. LESSONS The diagnosis of postmenopausal ovarian YST is relatively difficult and it can coexist with other germ cell or epithelial tumors. Postmenopausal ovarian YSTs are aggressive, and may have a worse prognosis compared with those in young patients. More aggressive treatment is needed. When YST mixed with epithelial cancer components, adjuvant chemotherapy regimen should include platinum-based chemotherapy aiming at both epithelial ovarian cancer and germ cell tumors.
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Makris GM, Fotiou A, Chrelias G, Battista MJ, Chrelias C. Giving Birth After Fertility Sparing Treatment for a Yolk Sac Tumour: Case Report. J Clin Diagn Res 2017; 11:QD12-QD13. [PMID: 28969219 DOI: 10.7860/jcdr/2017/25820.10524] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/04/2016] [Accepted: 05/23/2017] [Indexed: 11/24/2022]
Abstract
Yolk Sac Tumours (YSTs) of the ovary, also called Endodermal Sinus Tumours (ESTs), are the second most common Malignant Ovarian Germ Cell Tumours (MOGCTs), after dysgerminomas. YSTs occur primarily in children and young women. We present a case report of a 20-year-old woman who had been diagnosed with YST (tumour diameter of 29 cm). The patient underwent fertility sparing surgery and was subsequently treated with adjuvant chemotherapy (four cycles of bleomycin, etoposide and cisplatin). Two years after the diagnosis of YST, she successfully gave birth to a healthy girl (birth weight 3,500 g, Apgar score of 9-10 and a spontaneous vaginal delivery). During pregnancy, she had microscopic hematuria and bilateral nephrolithiasis, was ultrasonographically diagnosed. The conception after fertility sparing treatment of YST is possible and the birth of a healthy child is feasible. There are no current guidelines for surveillance of such patients during pregnancy; however, structured pregnancy surveillance in such survivors seems necessary.
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Affiliation(s)
- Georgios-Marios Makris
- Head, Department of Obstetrics and Gynecology, Athens Euroclinic Hospital, Athens, Greece
| | - Alexandros Fotiou
- Resident, Department of Obstetrics and Gynecology, Athens Euroclinic Hospital, Athens, Greece
| | - Georgios Chrelias
- Resident, Department of Obstetrics and Gynecology, General University Hospital "Attikon", University of Athens, Athens, Greece
| | - Marco-Johannes Battista
- Professor, Department of Gynecology and Obstetrics, University Hospital Mainz, Mainz, Germany
| | - Charalampos Chrelias
- Professor, Department of Obstetrics and Gynecology, General University Hospital "Attikon", University of Athens, Athens, Greece
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Nasioudis D, Chapman-Davis E, Frey MK, Caputo TA, Holcomb K. Management and prognosis of ovarian yolk sac tumors; an analysis of the National Cancer Data Base. Gynecol Oncol 2017; 147:296-301. [PMID: 28803748 DOI: 10.1016/j.ygyno.2017.08.013] [Citation(s) in RCA: 27] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/14/2017] [Revised: 08/07/2017] [Accepted: 08/08/2017] [Indexed: 10/19/2022]
Abstract
OBJECTIVE To evaluate the clinico-pathological characteristics, management and prognosis of women diagnosed with ovarian yolk sac tumors (OYSTs). METHODS The U.S National Cancer Data Base was queried for women diagnosed with OYST between 2004 and 2014. Overall survival (OS) was calculated following generation of Kaplan-Meir curves. Univariate analysis was performed with the log-rank test. A Cox model was constructed to determine independent predictors of mortality. RESULTS A total of 561 women were identified with a median age of 23years. The majority (58.5%) had early stage (I-II), while 29.6% and 11.9% had stage III and IV disease respectively. Five-year OS for women with stage I, II, III and IV disease were 94.8%, 97.1%, 70.9% and 51.6% respectively, p<0.001. Better 5-yr OS was observed for adolescents (94.4%) and young adults (89.3%) compared to older premenopausal (67.6%) and postmenopausal women (30.6%), p<0.001. Omentectomy, hysterectomy and lymph node sampling/dissection (LND) were not associated with better OS. Women who received adjuvant chemotherapy had superior OS compared to those who did not, p=0.016. Early disease stage, younger age and receipt of adjuvant chemotherapy, but not LND were independently associated with better mortality. CONCLUSIONS Women with OYST commonly present with early stage disease. Administration of adjuvant chemotherapy, early stage and younger age are associated with superior outcomes.
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Affiliation(s)
- Dimitrios Nasioudis
- Department of Obstetrics and Gynecology, Weill Cornell Medicine, New York, NY, USA; Department of Obstetrics and Gynecology, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.
| | - Eloise Chapman-Davis
- Department of Obstetrics and Gynecology, Weill Cornell Medicine, New York, NY, USA
| | - Melissa K Frey
- Department of Obstetrics and Gynecology, Weill Cornell Medicine, New York, NY, USA
| | - Thomas A Caputo
- Department of Obstetrics and Gynecology, Weill Cornell Medicine, New York, NY, USA
| | - Kevin Holcomb
- Department of Obstetrics and Gynecology, Weill Cornell Medicine, New York, NY, USA
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Wakiya T, Toyoki Y, Ishido K, Kudo D, Kimura N, Tsutsumi S, Odagiri T, Suto A, Uchida C, Hakamada K. Living donor liver transplantation in a pediatric patient with preexisting yolk sac tumor. Pediatr Transplant 2017; 21. [PMID: 28039901 DOI: 10.1111/petr.12856] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 11/03/2016] [Indexed: 11/26/2022]
Abstract
There is ongoing discussion regarding the indications and timing of LT for patients with a preexisting extrahepatic malignancy. We herein report a pediatric case that underwent LDLT after therapy for YST. The patient, a 13-year-old female with biliary atresia, had undergone portoenterostomy at 2 months of age. She developed a left ovarian tumor with a high serum alpha-fetoprotein concentration at 10 years of age. She underwent left oophorectomy and was diagnosed with ovarian YST (Stage I). After surgery, hepatopulmonary syndrome progressed gradually. She was examined carefully and exhibited no findings to suggest the recurrence of YST. We decided to perform LDLT at 3 years and 6 months of age after the surgery for YST. The patient is currently alive and doing well without recurrence of YST at approximately 2 years after transplantation. There is no significant difference between the recurrence rate of preexisting extrahepatic malignancy and the incidence of de novo malignancy if specific cases are selected. The indications and period from surgery for preexisting extrahepatic malignancy to LT should thus be determined according to the type and stage of cancer.
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Affiliation(s)
- Taiichi Wakiya
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Yoshikazu Toyoki
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Keinosuke Ishido
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Daisuke Kudo
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Norihisa Kimura
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Shinji Tsutsumi
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Tadashi Odagiri
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Akiko Suto
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Chiaki Uchida
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
| | - Kenichi Hakamada
- Department of Gastroenterological Surgery, Hirosaki University Graduate School of Medicine, Aomori, Japan
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Yolk sac tumor in the abdominal wall of an 18-month-old girl: a case report. J Med Case Rep 2017; 11:47. [PMID: 28215183 PMCID: PMC5317054 DOI: 10.1186/s13256-017-1216-4] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/20/2016] [Accepted: 01/21/2017] [Indexed: 11/22/2022] Open
Abstract
Background Pediatric germ cell tumors account for approximately 3.5 % of all childhood cancers for children under the age of 15 years. Up to one-third are extragonadal neoplasms. Germ cell tumors are a heterogeneous group of malignant tumors with a wide variety of histopathological features. Yolk sac tumor is the predominant variant in newborns and younger children. We report for the first time, the presentation of a primary yolk sac tumor in the abdominal wall of a small child. Case presentation An 18-month-old white girl underwent resection of a small, round subcutaneous lump (1.5×1.3×0.8 cm) of the abdominal wall in her right hypochondriac region. The histopathology was compatible with yolk sac tumor. Her alpha-fetoprotein was initially elevated but normalized after the resection. Magnetic resonance imaging of her abdomen was normal. The surgeon decided to observe and follow her alpha-fetoprotein level closely. One year after resection a local recurrence appeared and her alpha-fetoprotein rose to 58 ng/mL. The surgeon performed a wide resection of the lesion with normalization of her alpha-fetoprotein. Follow-up consisted of measuring alpha-fetoprotein, clinical evaluation, and abdominal ultrasound. Conclusions Clinicians should be aware that a yolk sac tumor can present in an unusual extragonadal place, for example in this case it was subcutaneous. In some cases, conservative treatment can be carried out with careful monitoring of the patient and their alpha-fetoprotein.
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Gevers EF, Meredith S, Shah P, Torpiano J, Peters C, Sebire NJ, Slater O, White A, Dattani MT. Cushing syndrome in a child due to pro-opiomelanocortin (POMC) secretion from a yolk sac tumor. Eur J Endocrinol 2017; 176:K1-K7. [PMID: 27879325 DOI: 10.1530/eje-16-0776] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/11/2016] [Revised: 11/14/2016] [Accepted: 11/22/2016] [Indexed: 11/08/2022]
Abstract
CONTEXT Pituitary microadenomas and adrenal tumours are the most common causes for endogenous Cushing syndrome (CS) in children. CASE DESCRIPTION We describe a two-year old girl with Cushing syndrome due to ectopic pro-opiomelanocortin (POMC) production from an abdominal yolk sac tumor. Cortisol concentrations were elevated but adrenocorticotropic hormone (ACTH) concentrations were equivocal. The use of antibodies specifically detecting ACTH precursors revealed that plasma ACTH precursors were elevated. Additionally, an ACTH assay with a low cross-reactivity for precursors showed low concentrations of ACTH. Immunohistochemistry suggested POMC but not ACTH production by the tumour. CONCLUSION We describe a yolk sac tumour as a novel source of ectopic POMC production leading to CS in a young girl.
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Affiliation(s)
- Evelien F Gevers
- Department of EndocrinologyGreat Ormond Street Hospital for Children, London, UK
| | - Suzanne Meredith
- Division of DiabetesEndocrinology and Gastroenterology, Faculty of Biology, Medicine and Health, University of Manchester, Manchester Academic Health Sciences Centre, Manchester, UK
| | - Pratik Shah
- Department of EndocrinologyGreat Ormond Street Hospital for Children, London, UK
- Section of Genetics and Epigenetics in Health and DiseaseGenetics and Genomic Medicine Programme, UCL Institute of Child Health, London, UK
| | - John Torpiano
- Department of PaediatricsPaediatric Endocrine Service, Mater Dei Hospital, Msida, Malta
| | - Catherine Peters
- Department of EndocrinologyGreat Ormond Street Hospital for Children, London, UK
| | - Neil J Sebire
- Department of HistopathologyGreat Ormond Street Hospital and Institute for Child Health (UCL), London, UK
| | - Olga Slater
- Department of OncologyGreat Ormond Street Hospital for Children, London, UK
| | - Anne White
- Division of DiabetesEndocrinology and Gastroenterology, Faculty of Biology, Medicine and Health, University of Manchester, Manchester Academic Health Sciences Centre, Manchester, UK
| | - Mehul T Dattani
- Department of EndocrinologyGreat Ormond Street Hospital for Children, London, UK
- Section of Genetics and Epigenetics in Health and DiseaseGenetics and Genomic Medicine Programme, UCL Institute of Child Health, London, UK
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de la Motte Rouge T, Pautier P, Genestie C, Rey A, Gouy S, Leary A, Haie-Meder C, Kerbrat P, Culine S, Fizazi K, Lhommé C. Prognostic significance of an early decline in serum alpha-fetoprotein during chemotherapy for ovarian yolk sac tumors. Gynecol Oncol 2016; 142:452-7. [PMID: 27401840 DOI: 10.1016/j.ygyno.2016.07.005] [Citation(s) in RCA: 20] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/18/2016] [Revised: 07/02/2016] [Accepted: 07/04/2016] [Indexed: 10/21/2022]
Abstract
BACKGROUND The ovarian yolk sac tumor (OYST) is a very rare malignancy arising in young women. Our objective was to determine whether an early decline in serum alpha-fetoprotein (AFP) during chemotherapy has a prognostic impact. METHODS This retrospective study is based on prospectively recorded OYST cases at Gustave Roussy (Cancer Treatment Center). Survival curves were estimated using the Kaplan-Meier method. The serum AFP decline was calculated with the formula previously developed and validated in male patients with poor prognosis non-seminomatous germ cell tumors. Univariate and multivariate analyses were performed using the log-rank test and logistic regression, respectively. RESULTS Data on AFP were available to calculate an early AFP decline in 57 patients. All patients had undergone surgery followed by chemotherapy. The 5-year overall survival (OS) and event-free survival (EFS) rates were 86% (95% CI: 74%-93%) and 84% (95% CI: 73%-91%), respectively. The disease stage, presence of ascites at presentation, use of the BEP regimen, serum AFP half-life and an early AFP decline were significantly predictive factors for OS and EFS in the univariate analysis. The OS rate was 100% and 49% (95% CI: 26%-72%) in patients with a favorable AFP decline and in those with an unfavorable decline, respectively (p<0.001). In the multivariate analysis, only the presence of ascites at diagnosis (RR=7.3, p=0.03) and an unfavorable early AFP decline (RR=16.9, p<0.01) were significant negative predictive factors for OS. CONCLUSIONS An early AFP decline during chemotherapy is an independent prognostic factor in patients with OYSTs. CONFLICT OF INTEREST STATEMENT No conflict of interest.
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Affiliation(s)
- Thibault de la Motte Rouge
- Department of Medical Oncology, Centre Eugène Marquis, Av de la bataille Flandres-Dunkerque, CS 44229, F-35042 Rennes, France
| | - Patricia Pautier
- Department of Cancer Medicine, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Catherine Genestie
- Department of Biopathology, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Annie Rey
- Department of Biostatistics, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Sébastien Gouy
- Department of Surgery, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Alexandra Leary
- Department of Cancer Medicine, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Christine Haie-Meder
- Department of Radiotherapy, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Pierre Kerbrat
- Department of Medical Oncology, Centre Eugène Marquis, Av de la bataille Flandres-Dunkerque, CS 44229, F-35042 Rennes, France
| | - Stéphane Culine
- Department of Medical Oncology, CHU Hôpital Saint Louis, 1 avenue Claude-Vellefaux, F-75010 Paris, France
| | - Karim Fizazi
- Department of Cancer Medicine, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France
| | - Catherine Lhommé
- Department of Cancer Medicine, Institut Gustave-Roussy, 114 rue Edouard Vaillant, F-94805 Villejuif, France.
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Abstract
ObjectiveThe aim of this study was to evaluate the clinicopathologic characteristics of patients with ovarian yolk sac tumor and the benefit of omentectomy in patients with clinical early-stage disease.MethodsThe medical records of 66 patients with ovarian yolk sac tumor were reviewed retrospectively.ResultsThere were 37, 8, 14, and 7 patients with stages I, II, III, and IV disease, respectively. Sixty-five patients received surgery and adjuvant chemotherapy, and 1 had chemotherapy only. The median follow-up was 78 months. The overall 5-year survival rate was 86.0%. Univariate analysis revealed that stage (P = 0 .022), age (P = 0.001), residual tumor (P = 0.036), and satisfactory α-fetoprotein (AFP) decline (defined as normalization of AFP after the first or second cycles of postsurgery chemotherapy, P = 0.006) were significant prognostic factors. Multivariate analysis revealed that satisfactory AFP decline was an independent significant prognostic factor for overall survival (P = 0.028). The postoperative pathology showed that only 1 (2.7%) of 37 patients who received omentectomy without gross spread had omentum metastasis microscopically. The 5-year survival rates were 89.2% and 100.0% for stage I-II patients with (36 cases) or without (9 cases) omentectomy, respectively (P > 0.05). Three of the 7 patients with recurrence were successfully salvaged and lived 38.0, 102.6, and 45.2 months after initial diagnosis.ConclusionsPostsurgery satisfactory AFP decline was an independent significant prognostic factor for patient survival. Omentectomy might not be of therapeutic significance for clinical stage I-II patients.
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Tomao F, Peccatori F, Del Pup L, Franchi D, Zanagnolo V, Panici PB, Colombo N. Special issues in fertility preservation for gynecologic malignancies. Crit Rev Oncol Hematol 2015; 97:206-19. [PMID: 26358422 DOI: 10.1016/j.critrevonc.2015.08.024] [Citation(s) in RCA: 40] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/03/2015] [Revised: 07/25/2015] [Accepted: 08/26/2015] [Indexed: 11/27/2022] Open
Abstract
Gynecologic malignancies account for 1,09 million new cancer cases worldwide consisting of about 12% of tumors affecting female population. About 10% of all female cancer survivors are younger than 40 years of age. Since cancers affecting female genital organs are usually treated by radical surgery, chemotherapy or chemoradiation approaches that induce permanent damage of reproductive functions, the development of strategies for fertility preservation represent one of the most important goals for gynecologic oncology. In this scenario, the newly defined oncofertility discipline acquires increasing interest, offering patients maximal chances to make an adequate decision about future fertility, based on their oncologic diagnosis and prognosis. However, the majority of physicians do not pay particular attention to these issues, even if impressive progresses have been made in this field in the last decades. Possibly, it is due to the lack of strong evidences from clinical trials without an adequate number of cases to establish safety and efficacy of these procedures. In this review we will discuss the most recently debated options for fertility preservation in gynecologic oncology, highlighting issues and controversies related to oncofertility.
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Affiliation(s)
- Federica Tomao
- European Institute of Oncology "IEO", Via Giuseppe Ripamonti 435, 20141 Milan, Italy; University of Rome "Sapienza", Viale del Policlinico 155, 00161 Rome, Italy.
| | - Fedro Peccatori
- European Institute of Oncology "IEO", Via Giuseppe Ripamonti 435, 20141 Milan, Italy
| | - Lino Del Pup
- National Cancer Institute "CRO", Via Franco Gallini 2, 33081 Aviano PD, Italy
| | - Dorella Franchi
- European Institute of Oncology "IEO", Via Giuseppe Ripamonti 435, 20141 Milan, Italy
| | - Vanna Zanagnolo
- European Institute of Oncology "IEO", Via Giuseppe Ripamonti 435, 20141 Milan, Italy
| | | | - Nicoletta Colombo
- European Institute of Oncology "IEO", Via Giuseppe Ripamonti 435, 20141 Milan, Italy
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Jones RG, Akande MY, Younes HK, Jagarpu J, Mba N, Savell VH, Almond SP. Yolk sac tumor of the ovary in a young girl with tuberous sclerosis: A case report and review of the literature. Gynecol Oncol Rep 2015; 10:9-12. [PMID: 26075992 PMCID: PMC4434146 DOI: 10.1016/j.gore.2014.07.005] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/26/2014] [Accepted: 07/28/2014] [Indexed: 11/30/2022] Open
Abstract
We reported the first tuberous sclerosis patient with an ovarian yolk sac tumor. Although angiomyolipoma is a common benign tumor in TS patients, abdominal malignancies must be considered.
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Affiliation(s)
- Riley G Jones
- Department of Surgery, Driscoll Children's Hospital, Corpus Christi, TX, USA
| | - Manzilat Y Akande
- Department of Pediatrics, Driscoll Children's Hospital, Corpus Christi, TX, USA
| | - Houssam K Younes
- Department of Surgery, University of Texas Medical Branch, Galveston, TX, USA
| | - Jawahar Jagarpu
- Deptartment of Pediatrics, Jackson Memorial Hospital, Miami, FL, USA
| | - Nkechi Mba
- Department of Pediatrics, Driscoll Children's Hospital, Corpus Christi, TX, USA
| | - Van H Savell
- Department of Pathology, Driscoll Children's Hospital, Corpus Christi, TX, USA
| | - Stephen P Almond
- Department of Surgery, Driscoll Children's Hospital, Corpus Christi, TX, USA
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Li X, Guo S, Wu Z, Dong P, Li Y, Zhang Z, Yao K, Han H, Qin Z, Zhou F, Liu Z. Surveillance for patients with clinical stage I nonseminomatous testicular germ cell tumors. World J Urol 2014; 33:1351-7. [PMID: 25471669 DOI: 10.1007/s00345-014-1454-7] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/30/2014] [Accepted: 11/23/2014] [Indexed: 01/22/2023] Open
Abstract
PURPOSE To assess the prognostic value of histological parameters in patients with clinical stage I nonseminomatous germ cell tumors (NSGCTs) undergoing active surveillance post-orchiectomy. METHODS Prognoses and recurrence patterns were investigated in 78 patients with CSI NSGCT who underwent orchiectomy. Immediately following orchiectomy, patients participated in active surveillance between 1999 and 2013 at Sun Yat-sen University Cancer Center, Guangzhou, China. RESULTS 23.1 % of the 78 investigated patients with CSI NSGCT relapsed, within a median time of 5.6 months It was determined using multivariate analysis that lymph vascular invasion (LVI) (OR 6.521; 95 % CI 1.872-22.721; p = 0.003) and the predominant presence of yolk sac tumor (greater than 50 %) (OR 3.537; 95 % CI 1.076-11.628; p = 0.038) independently correlated with relapse-free survival (RFS). Patients were categorized accordingly into three risk groups: low risk [<50 % presence of yolk sac tumor and LVI (-); n = 41], intermediate risk [50 % or greater presence of yolk sac tumor and LVI (+); n = 29], and high risk [50 % or greater presence of yolk sac tumor and LVI (+); n = 8]. Relapse rates of the low-risk, intermediate-risk, and high-risk groups were 7.3, 31.0, and 75.0 %, respectively. CONCLUSIONS LVI and a predominant presence of yolk sac tumor are crucial risk factors for relapse of CSI NSGCT. For patients without either of these risk factors, active surveillance post-orchiectomy is a safe and effective approach for the initial management of CSI NSGCT.
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Affiliation(s)
- Xiangdong Li
- Department of Urology, Sun Yat-sen University Cancer Center, No. 651, East Dongfeng Road, Guangzhou, 510060, Guangdong Province, China
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Zovko G, Madžarac V, Kukura V, Gašparov S, Kovačić-Vicić V, Bursać D. Successful Pregnancy After Conservative Treatment of Advanced Yolk-Sac Tumor of the Ovary. J Gynecol Surg 2014. [DOI: 10.1089/gyn.2014.0065] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Affiliation(s)
- Gojko Zovko
- Department of Obstetrics and Gynecology, Merkur University Hospital, Zagreb, Croatia
| | - Vedran Madžarac
- Department of Obstetrics and Gynecology, Merkur University Hospital, Zagreb, Croatia
| | - Vlastimir Kukura
- Department of Obstetrics and Gynecology, Merkur University Hospital, Zagreb, Croatia
| | - Slavko Gašparov
- Department of Pathology, Merkur University Hospital, Zagreb, Croatia
| | - Vesna Kovačić-Vicić
- Department of Anaesthesia and Intensive Care, Merkur University Hospital, Zagreb, Croatia
| | - Danijel Bursać
- Department of Obstetrics and Gynecology, Merkur University Hospital, Zagreb, Croatia
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Guo YL, Zhang YL, Zhu JQ. Prognostic value of serum α-fetoprotein in ovarian yolk sac tumors: A systematic review and meta-analysis. Mol Clin Oncol 2014; 3:125-132. [PMID: 25469282 DOI: 10.3892/mco.2014.417] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/20/2014] [Accepted: 09/03/2014] [Indexed: 12/22/2022] Open
Abstract
This study was conducted to determine the prognostic value of serum α-fetoprotein (AFP) levels in patients with ovarian yolk sac tumor (OYST). We performed a systematic review and meta-analysis to assess the associations between serum AFP level and prognosis in OYST. A total of 12 quantitative studies met the inclusion criteria. Preoperative AFP was not found to be associated with overall survival (OS) [odds ratio (OR)=0.84, 95% confidence interval (CI): 0.43-1.62] in OYST. However, a high postoperative AFP level was associated with worse OS (OR=0.16, 95% CI: 0.05-0.48) and relapse-free survival (RFS) (OR=0.18, 95% CI: 0.08-0.43) compared to a low postoperative AFP level in patients with OYST. In addition, a postoperative AFP level of >1,000 ng/ml was associated with a decrease in OS (OR=0.16, 95% CI: 0.05-0.50) and RFS (OR=0.21, 95% CI: 0.08-0.57). In conclusion, the postoperative, but not the preoperative, AFP level was found to be a prognostic factor in patients with OYST. In particular, a postoperative AFP level of >1,000 ng/ml was an indicator of poor prognosis in patients with OYST.
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Affiliation(s)
- Yang-Long Guo
- Department of Gynecologic Oncology, Zhejiang Cancer Hospital, Hangzhou, Zhejiang 310022, P.R. China
| | - Ying-Li Zhang
- Department of Gynecologic Oncology, Zhejiang Cancer Hospital, Hangzhou, Zhejiang 310022, P.R. China
| | - Jian-Qing Zhu
- Department of Gynecologic Oncology, Zhejiang Cancer Hospital, Hangzhou, Zhejiang 310022, P.R. China
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Rittiluechai K, Wilcox R, Lisle J, Everett E, Wallace HJ, Verschraegen CF. Prognosis of hepatoid yolk sac tumor in women: what's up, Doc? Eur J Obstet Gynecol Reprod Biol 2014; 175:25-9. [DOI: 10.1016/j.ejogrb.2013.12.029] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/15/2013] [Revised: 12/15/2013] [Accepted: 12/20/2013] [Indexed: 11/29/2022]
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Lu Y, Yang J, Cao D, Huang H, Wu M, You Y, Chen J, Lang J, Shen K. Role of neoadjuvant chemotherapy in the management of advanced ovarian yolk sac tumor. Gynecol Oncol 2014; 134:78-83. [PMID: 24582864 DOI: 10.1016/j.ygyno.2014.02.029] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/23/2013] [Revised: 02/17/2014] [Accepted: 02/20/2014] [Indexed: 10/25/2022]
Abstract
OBJECTIVE The aim of the study was to identify the role of neoadjuvant chemotherapy (NACT) in the treatment of the patients with advanced stage ovarian yolk sac tumor (OYST). METHODS The comparative study was based on 53 cases with advanced stage OYST registered at Peking Union Medical College Hospital from 1995 to 2010. Twenty one cases were treated with NACT followed by interval debulking surgery (IDS). Thirty two cases were treated with primary debulking surgery (PDS). Data on patient characteristics, treatment and survival were analyzed and compared between two groups to assess the outcome of NACT. RESULTS After NACT, the overall status of the patients was improved significantly. Patients in NACT had better optimal cytoreduction rate and less peri-operative morbidities. Seven patients (13.2%) suffered from relapse. There was a significantly better PFS for patients with ovarian tumor size >20cm in the NACT than those underwent PDS. Residual disease >2cm was the independent risk factor of relapse. CONCLUSIONS NACT is the better treatment option for some patients with advanced stage OYST, especially for those with unresectable tumors and poor general condition.
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Affiliation(s)
- Yan Lu
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Jiaxin Yang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Dongyan Cao
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Huifang Huang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Ming Wu
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Yan You
- Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Pathology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Jie Chen
- Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Pathology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Jinhe Lang
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China
| | - Keng Shen
- Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, People's Republic of China and Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Peking Union Medical College, People's Republic of China.
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Jabot G, Koskas M, Boyez A, Zurlinden O, Ou P, Shouman Claeys E. Tumeur du sac vitellin rompue dans la cavité péritonéale. IMAGERIE DE LA FEMME 2013. [DOI: 10.1016/j.femme.2013.06.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
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Loh AHP, Gee KWC, Chua JHY. Diagnostic accuracy of preoperative alpha-fetoprotein as an ovarian tumor marker in children and adolescents: not as good as we thought? Pediatr Surg Int 2013; 29:709-13. [PMID: 23653236 DOI: 10.1007/s00383-013-3316-6] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 04/12/2013] [Indexed: 12/21/2022]
Abstract
PURPOSE To evaluate the diagnostic accuracy of preoperative serum alpha-fetoprotein (AFP) levels in predicting malignancy risk in children and adolescents presenting with ovarian neoplasms. METHODS In 110 girls aged 18 and below diagnosed with ovarian neoplasms, we retrospectively correlated preoperative serum AFP levels with histological diagnosis of germ cell tumor or immature teratoma (GCT/IT) versus non-GCT/IT, and benign versus non-benign. We determined area under receiver-operating characteristic curves (AUC), sensitivity, specificity, and likelihood ratios. RESULTS Twenty patients (18.2 %) had non-benign ovarian neoplasms, of which 12 had GCT/IT (10.9 %). In diagnosing GCT/IT versus non-GCT/IT, specificity of preoperative serum AFP was 87.8 %, sensitivity 66.7 %, and AUC 0.853. Excluding infants to remove the effects of increased variance in AFP in this group, specificity improved (92.0 %), but not sensitivity (66.7 %); AUC was 0.926. Increasing AFP cutoff to two times upper normal limit improved specificity (94.9 %), but not sensitivity (66.7 %). For benign versus non-benign tumors, AFP specificity was only 88.9 % and sensitivity 50.0 %. CONCLUSION The diagnostic accuracy of preoperative serum AFP for detecting GCT/IT in girls was limited by poor sensitivity and positive predictive value. Excluding infants and raising cutoff levels improved specificity marginally. Clinicians should be aware of these limitations when using AFP in the preoperative evaluation of childhood ovarian neoplasms.
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Affiliation(s)
- Amos Hong Pheng Loh
- Department of Pediatric Surgery, KK Women's and Children's Hospital, 100 Bukit Timah Road, Singapore 229899, Singapore.
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Shigeta N, Hori K, Ito K, Watanabe H, Kimura H, Nakatsuka SI. Secondary Cytoreductive Surgery for Primary Yolk Sac Tumor Arising from the Omentum. J Gynecol Surg 2013. [DOI: 10.1089/gyn.2012.0048] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
Affiliation(s)
- Naoya Shigeta
- Department of Obstetrics and Gynecology, Kansai Rosai Hospital, Amagasaki, Hyogo, Japan
| | - Kensuke Hori
- Department of Obstetrics and Gynecology, Kansai Rosai Hospital, Amagasaki, Hyogo, Japan
| | - Kimihiko Ito
- Department of Obstetrics and Gynecology, Kansai Rosai Hospital, Amagasaki, Hyogo, Japan
| | - Hitoshi Watanabe
- Department of Radiology, Kansai Rosai Hospital, Amagasaki, Hyogo, Japan
| | - Hayato Kimura
- Department of Pathology, Kansai Rosai Hospital, Amagasaki, Hyogo, Japan
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Vui-Kee K, Mohd Dali AZH, Mohamed Rose I, Ghazali R, Jamal R, Mokhtar NM. Molecular markers associated with nonepithelial ovarian cancer in formalin-fixed, paraffin-embedded specimens by genome wide expression profiling. Kaohsiung J Med Sci 2012; 28:243-50. [PMID: 22531302 DOI: 10.1016/j.kjms.2011.11.007] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/02/2011] [Accepted: 07/11/2011] [Indexed: 01/05/2023] Open
Abstract
Nonepithelial ovarian cancer (NEOC) is a rare cancer that is often misdiagnosed as other malignant tumors. Research on this cancer using fresh tissues is nearly impossible because of its limited number of samples within a limited time provided. The study is to identify potential genes and their molecular pathways related to NEOC using formalin-fixed paraffin embedded samples. Total RNA was extracted from eight archived NEOCs and seven normal ovaries. The RNA samples with RNA integrity number >2.0, purity >1.7 and cycle count value <28 cycles were hybridized to the Illumina Whole-Genome DASL assay (cDNA-mediated annealing, selection, extension, and ligation). We analyzed the results using the GeneSpring GX11.0 and FlexArray software to determine the differentially expressed genes. Microarray results were validated using an immunohistochemistry method. Statistical analysis identified 804 differentially expressed genes with 443 and 361 genes as overexpressed and underexpressed in cancer, respectively. Consistent findings were documented for the overexpression of eukaryotic translation elongation factor 1 alpha 1, E2F transcription factor 2, and fibroblast growth factor receptor 3, except for the down-regulated gene, early growth response 1 (EGR1). The immunopositivity staining for EGR1 was found in the majority of cancer tissues. This finding suggested that the mRNA level of a transcript did not always match with the protein expression in tissues. The current gene profile can be the platform for further exploration of the molecular mechanism of NEOC.
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Affiliation(s)
- Koon Vui-Kee
- Department of Physiology, Faculty of Medicine, Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia
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Ashihara T, Nakanishi K, Hashii K, Fujimoto M, Yasuhara Y, Yoshioka S, Mikami Y, Konishi I. Ovarian yolk sac tumor in a postmenopausal woman: case report and review of the literature. Int Cancer Conf J 2012. [DOI: 10.1007/s13691-012-0019-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/28/2022] Open
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Chen CF, Wong WY, Chuang CH, Yeh YS, Tsai KB, Wang JY. Ruptured ovarian yolk sac tumor combined with hemoperitoneum in a young girl with abdominal blunt injury. GENOMIC MEDICINE, BIOMARKERS, AND HEALTH SCIENCES 2012; 4:76-78. [DOI: 10.1016/j.gmbhs.2012.04.011] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 09/20/2023]
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