Abstract
Purpose
Yolk sac tumors (YST) are a rare and aggressive germ cell tumor. We aimed to conduct a population-based cohort study and develop a nomogram to predict overall survival (OS) in pediatric patients with YST.
Methods
The Surveillance, Epidemiology, and End Results (SEER) database was used to identify all pediatric patients with YST diagnosed between 2000 and 2018. The log-rank test was used to compare survival curves. To examine the impact of each factor on overall survival, a multivariate Cox proportional hazards model was created. Based on the results of the Cox regression model, a nomogram was constructed.
Results
A total of 520 YST patients were identified. Overall survival rates for all patients were 92.2% at 3-year and 90.3% at 5-year, respectively. The outcome of Cox proportional hazard regression revealed that age, gender, primary sites, and treatment regimens were important independent predictors in this model. Based on the Cox regression model, we created a nomogram for predicting OS in pediatric YST patients. The chance of death increased with age in patients. Furthermore, patients with extra-gonadal YST have a lower survival rate than those with gonadal YST.
Conclusions
Our study revealed that age, gender, and primary site were found to be the most important predictors of the overall survival of pediatric YST, providing crucial epidemiological information for clinical management.
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Availability of data and materials
The dataset used and analyzed during the current study are available from the corresponding author on reasonable request.
References
Feng S, Huang S, Tong Y et al (2016) RNA-binding protein LIN28 is a sensitive marker of pediatric yolk sac tumors. Pediatr Surg Int 32(8):819–825
Dede M, Pabuccu R, Yagci G et al (2004) Extragonadal yolk sac tumor in pelvic localization. A case report and literature review. Gynecol Oncol 92(3):989–991
Yang H, Wu Z, Tian Y et al (2022) Yolk Sac Tumor in an infant with androgen insensitivity syndrome: a case report and review of the literature. Int J Surg Pathol 30(7):804–809
McKenney JK, Heerema-McKenney A, Rouse RV (2007) Extragonadal germ cell tumors: a review with emphasis on pathologic features, clinical prognostic variables, and differential diagnostic considerations. Adv Anat Pathol 14:69–92
Bayar GR, Gulses A, Sencimen M et al (2010) Oral metastasis of the mediastinal germ cell tumor (yolk sac). J Craniofac Surg 21(6):1828–1830
Unal O, Beyazal M, Avcu S et al (2011) Metastasis of testicular yolk sac tumor to cauda equina. Fetal Pediatr Pathol 30(3):150–155
Thomas WJ, Kelleher JF, Duval-Arnould B (1981) Successful treatment of metastatic extragonadal endodermal sinus (yolk sac) tumor in childhood. Cancer 48(11):2371–2374
Arumugam D, Thandavarayan P, Chidambaram L et al (2016) Primary Nasopharngeal Yolk Sac tumor: a case report. J Clin Diagn Res 10(5):ED06–ED07
Young RH (2014) The yolk sac tumor: reflections on a remarkable neoplasm and two of the many intrigued by it-Gunnar Teilum and Aleksander Talerman-and the bond it formed between them. Int J Surg Pathol 22(8):677–687
Geng R, Zheng Z, Lin Y et al (2021) Clinical characteristics and prognostic factors of male yolk sac tumor: a Surveillance, Epidemiology, and End Results program study. World J Urol 39(4):1211–1217
Wang T, Wang B, Wang SX et al (2021) Clinicopathologic characteristics and survival of patients with rare malignant ovarian yolk sac tumors: a population-based analysis. Curr Med Sci 41(2):342–347
Tamura D, Maeda D, Sato T et al (2020) An extragonadal yolk sac tumor presumed to be of postmeiotic germ cell origin by genetic zygosity analysis via single nucleotide polymorphism array. Genes Chromosomes Cancer 59(3):209–213
Ravishankar S, Malpica A, Ramalingam P et al (2017) Yolk Sac tumor in extragonadal pelvic sites: still a diagnostic challenge. Am J Surg Pathol 41(1):1–11
Cornejo KM, Frazier L, Lee RS et al (2015) Yolk Sac tumor of the testis in infants and children: a clinicopathologic analysis of 33 cases. Am J Surg Pathol 39(8):1121–1131
Wruck W, Bremmer F, Kotthoff M et al (2021) The pioneer and differentiation factor FOXA2 is a key driver of yolk-sac tumour formation and a new biomarker for paediatric and adult yolk-sac tumours. J Cell Mol Med 25(3):1394–1405
Terenziani M, Piva L, Spreafico F et al (2002) Clinical stage I nonseminomatous germ cell tumors of the testis in childhood and adolescence: an analysis of 31 cases. J Pediatr Hematol Oncol 24(6):454–458
Shah JP, Kumar S, Bryant CS et al (2008) A population-based analysis of 788 cases of yolk sac tumors: a comparison of males and females. Int J Cancer 123(11):2671–2675
Zhou X, Zhao L, Feng X et al (2022) Relapsed and refractory yolk sac tumor of the peritoneum (mesentery): a case report and literature review. Front Oncol 12:928234
Zeremski V, Mawrin C, Fischer T et al (2017) Diagnostic and therapeutic challenges in extragonadal yolk sac tumor with hepatoid differentiation: a case report. Mol Clin Oncol 6(1):79–82
Sekar MD, Pradeep I, Srinivas BH et al (2023) Primary Pediatric Yolk Sac tumor of liver with lung metastasis: an unusual presentation with diagnosis aided by LIN28 immunohistochemistry. Int J Surg Pathol. https://doi.org/10.1177/10668969231157777
Rudaitis V, Mickys U, Katinaitė J et al (2016) Successful treatment of advanced stage yolk sac tumour of extragonadal origin: a case report and review of literature. Acta Med Litu 23(2):110–116
Kapoor G, Advani SH, Nair CN et al (1995) Pediatric germ cell tumor. An experience with BEP. sJ Pediatr Hematol Oncol 17(4):318–324
Funding
This study was supported by the Department of Health of Zhejiang Province (Grant Number 2022RC234 to Shaoguang Feng) and Annual Scientific Project Fund of Xuzhou Children’s hospital (Grant Number 22040406 to Xu Liu).
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XL: conception and design of study, acquisition of data, revising the manuscript. SF: conception and design of study, acquisition of data, drafting the manuscript. LZ: conception and design of study, acquisition of data, drafting the manuscript. LL: conception and design of study, acquisition of data, analysis and/or interpretation of data, drafting the manuscript. all authors read and approved the final manuscript as submitted.
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Liu, X., Feng, S., Zhao, L. et al. Clinical characteristics and prognostic models of gonadal and extra-gonadal yolk sac tumors: a population-based analysis in children and adolescents. World J Urol 41, 3009–3017 (2023). https://doi.org/10.1007/s00345-023-04616-4
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DOI: https://doi.org/10.1007/s00345-023-04616-4