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The Journal of Pathology Jan 2022Germ cell tumors (GCTs) originate during the histogenesis of primordial germ cells to mature gametes. Previous studies identified five histogenic mechanisms in ovarian...
Germ cell tumors (GCTs) originate during the histogenesis of primordial germ cells to mature gametes. Previous studies identified five histogenic mechanisms in ovarian mature teratomas (type I: failure of meiosis I; type II: failure of meiosis II; type III: duplication of the genome of a mature gamete; type IV: no meiosis; and type V: fusion of two different ova), but those of other GCTs remain elusive. In this study, we analyzed 84 GCTs of various pathologic types to identify the histogenesis using single-nucleotide polymorphism array by analyzing copy-neutral loss of heterozygosity (CN-LOH) and copy number alterations (CNAs). We detected types I and II in ovarian teratomas, type III in ovarian teratomas and yolk sac tumors (YSTs), and type IV in all GCT types. The GCTs with multiple-type histogenesis (I-IV) (ovarian mature/immature teratomas and YST) show meiotic CN-LOH with scant CNAs. Type IV-only GCTs are either with mitotic CN-LOH and abundant CNAs (seminoma, dysgerminoma, testicular mixed GCTs) or with scant CNAs and no CN-LOH (pediatric testicular and mediastinal teratomas). The development sequences of CN-LOH and CNA are different between the multiple type (I-IV) GCTs and type IV-only GCTs. We analyzed two different histologic areas in eight GCTs (one mature teratoma with a mucin-secreting adenoma, two immature teratomas, and five mixed GCTs). We found that GCTs (mature teratoma, immature teratoma, and mixed GCT) showed different genomic alterations between histologic areas, suggesting that genomic differences within a GCT could accompany histologic differentiation. Of note, we found evidence for collision tumors in a mixed GCT. Our data indicate that GCTs may have various histogenesis and intratumoral genomic differences, which might provide important information for the identification of GCTs, especially for those with different histologic areas. © 2021 The Pathological Society of Great Britain and Ireland. Published by John Wiley & Sons, Ltd.
Topics: Humans; Loss of Heterozygosity; Male; Molecular Biology; Neoplasms, Germ Cell and Embryonal; Ovarian Neoplasms; Seminoma; Teratoma; Testicular Neoplasms
PubMed: 34561860
DOI: 10.1002/path.5799 -
Medicine Jun 2023There is currently no bibliometric study on teratomas in the literature. This study aims to analyze the published articles on teratomas to provide an overview of the... (Review)
Review
There is currently no bibliometric study on teratomas in the literature. This study aims to analyze the published articles on teratomas to provide an overview of the subject, determine global productivity, and identify current research trends. Additionally, data on different components of scientific output (countries, journals, institutions, authors) were analyzed. A total of 4209 articles published on teratomas between 1980 and 2022 were analyzed using various bibliometric and statistical methods. Bibliometric network visualization maps were used to determine trending topics, citation analyses, and international collaborations. Spearman correlation coefficient was used for correlation analysis. The top 3 countries that made the most contributions to the literature were the USA (1041, 24.7%), Japan (501, 11.9%), and India (310, 7.3%). The top 3 active institutions were the University of California System (n = 78), University of London (64), and Harvard University (62). The top 3 productive journals were the Journal of Pediatric Surgery (n = 141), Pediatric Surgery International (n = 70), and Journal of Pediatric Surgery Case Reports (69). The most productive author was Ulbright TM. (n = 18). The most studied topics from past to present were ovarian cancer/ovarian teratoma/ovarian torsion, mature cystic teratoma/dermoid cyst, sacrococcygeal teratoma, germ cell tumors, immature teratoma, malignant transformation, mediastinal teratoma/mediastinum, neonate/newborn/infant, prenatal diagnosis, testis/testicular cancer/teratoma, ultrasonography/ultrasound, magnetic resonance imaging, chemotherapy, growing teratoma syndrome, surgery, retroperitoneal teratoma/retroperitoneum, laparoscopic surgery/laparoscopy, children/child, and fetal surgery/fetus. We identified trend research topics in the field of teratomas in recent years, including mature cystic teratoma, ovarian teratoma/neoplasm, ovarian cancer, ovarian torsion, growing teratoma syndrome, recurrence, pediatric, testicular cancer, anti-n-methyl-d-aspartate receptor encephalitis, immature teratoma, retroperitoneal, struma ovarii, and carcinoid. The research leadership in the development of teratoma literature was determined by countries with major economies such as the USA, Japan, India, the UK, China, Turkey, South Korea, and other European countries (France, Germany, Italy).
Topics: Child; Female; Humans; Infant; Infant, Newborn; Male; Pregnancy; Ovarian Neoplasms; Ovarian Torsion; Teratoma; Testicular Neoplasms
PubMed: 37390229
DOI: 10.1097/MD.0000000000034208 -
European Journal of Obstetrics,... Jul 2021Extragonadal teratomas (EGTs) are rare and the commonest intra-abdominal subtype is omental. We present two cases: 1) a parasitic omental teratoma likely secondary to... (Review)
Review
INTRODUCTION
Extragonadal teratomas (EGTs) are rare and the commonest intra-abdominal subtype is omental. We present two cases: 1) a parasitic omental teratoma likely secondary to auto-amputation of an ovarian teratoma with subsequent omental reimplantation and 2) an omental immature teratoma likely due to parthenogenetic activation of displaced primordial germ cells. We subsequently conduct a systematic review to characterise EGTs.
METHODS
We sourced for English, peer-reviewed case reports of extragonadal teratomas in women and female adolescents aged 11 and above published from inception of each database through 31st June 2020 following PRISMA guidelines. Two authors reviewed each case for appropriateness and each case was graded for methodological quality utilising a modified Newcastle Ottawa Scale. PROSPERO Registration Number: CRD42020190131 RESULTS: Upon literature review between 1920-2020, from an initial screen of 818 articles, 67 articles were selected featuring 70 cases. One case featured an immature teratoma while the remaining were mature. Omental EGTs were the most common (56.5 %) followed by Pouch of Douglas and uterosacral ligament (23.2 %) and upper abdomen (14.5 %). There were statistically significant differences in EGT mean sizes between each location with the largest being in the upper abdomen (10.9 cm) and the smallest being in the adnexa or hernia (6.2 cm). Auto-amputation was deemed the commonest cause amongst omental EGTs (55.3 %) and Pouch of Douglas and uterosacral ligament EGTs (37.5 %) while 70 % of upper abdominal EGTs were likely due to displaced primordial germ cells. We characterise clinical features associated with each pathogenic mechanism and imaging characteristics of EGTs. Characterisation of EGT tumour marker profiles was limited as only 42.9 % of cases reported them but 19.2-25.0 % had raised tumour markers. The main risks are torsion, rupture, immature components and potential malignant change of the cell lines. Treatment is largely surgical. The mean size of EGTs approached laparoscopically and via laparotomy was 5.23 cm and 9.16 cm respectively.
CONCLUSIONS
While rare, EGTs should be considered when evaluating pelviabdominal masses with imaging characteristics consistent with teratomas. Confirmation is usually intraoperative and a laparoscopic approach is reasonable if there is good surgeon comfort and the size is about 5 cm.
Topics: Abdominal Wall; Adolescent; Female; Humans; Omentum; Ovarian Neoplasms; Teratoma; Uterus
PubMed: 34022590
DOI: 10.1016/j.ejogrb.2021.05.005 -
Neuropediatrics Apr 2022The rate of anti-N-methyl-D-aspartate receptor encephalitis (NMDARE) in ovarian teratomas is unknown. We aim to identify the prevalence of NMDARE as well as volumetric...
BACKGROUND
The rate of anti-N-methyl-D-aspartate receptor encephalitis (NMDARE) in ovarian teratomas is unknown. We aim to identify the prevalence of NMDARE as well as volumetric and histopathologic characteristics of ovarian teratomas in patients with versus without.
METHODS
We performed a retrospective cohort study to identify patients with confirmed ovarian teratomas and the characteristics of teratomas in NMDARE compared with non-NMDARE patients. Patients aged between 0 and 21 years with confirmed histopathological diagnosis of ovarian teratoma after resection were included. The rate of NMDARE in ovarian teratomas was identified. Moreover, volumes of ovarian teratomas and the frequency of neuronal glial elements on histopathology in NMDARE versus non-NMDARE patients were assessed.
RESULTS
Five out of one-hundred-and-sixty-three (3.07%) patients with histopathology confirmed ovarian teratomas were diagnosed with NMDARE. Age was not different between the NMDARE (mean: 13.8 years, standard deviation: 3.9) and non-NMDARE groups (median: 14, interquartile range [IQR]: 5). Teratoma volumes from NMDARE patients were smaller than those of non-NMDARE patients (median 28.3 cm with IQR of 431.2 and median 182.8 with IQR of 635.0, respectively). Both age and NMDARE diagnosis were statistically significant variables in the analysis of variance on a multiple linear regression model. Age ( = 0.013) had a positive correlation with teratoma size, whereas presence of NMDARE had a negative correlation ( = 0.008).
CONCLUSION
The rate of NMDARE in ovarian teratomas is low and NMDARE patients have smaller teratomas than non-NMDARE. Further studies are needed to understand the timing of anti-NMDA receptor antibodies in teratomas and the development of NMDARE.
Topics: Adolescent; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Child; Child, Preschool; Female; Humans; Infant; Infant, Newborn; Ovarian Neoplasms; Receptors, N-Methyl-D-Aspartate; Retrospective Studies; Teratoma; Young Adult
PubMed: 34872133
DOI: 10.1055/s-0041-1740352 -
Scientific Reports Jul 2023Epilepsy is a common neurological disease. Systemic tumors are associated with an increased risk of epileptic events. Paraneoplastic encephalitis related to gonadal...
Epilepsy is a common neurological disease. Systemic tumors are associated with an increased risk of epileptic events. Paraneoplastic encephalitis related to gonadal teratoma is frequently accompanied by seizures and life-threatening status epilepticus (SE). However, the risk of epilepsy in gonadal teratoma has not been studied. This study aims to investigate the relationship between epileptic events and gonadal teratoma. This retrospective cohort study used the Korean National Health Insurance (KNHI) database. The study population was divided into two study arms (ovarian teratoma vs. control and testicular teratoma vs. control) with 1:2 age and gender-matched control groups without a history of gonadal teratoma or other malignancy. Participants with other malignancies, neurologic disorders, and metastatic brain lesions were excluded. We observed the occurrence of epileptic events during the observation period (2013-2018) and investigated the risk of epileptic events in each gonadal teratoma group compared to controls. In addition, the influence of malignancy and tumor removal was investigated. The final analysis included 94,203 women with ovarian teratoma, 2314 men with testicular teratoma, and controls. Ovarian teratoma is associated with a higher risk of epilepsy without SE (HR, 1.244; 95% CI 1.112-1.391) and epilepsy with SE (HR, 2.012; 95% CI 1.220-3.318) compared to the control group. The risk of epilepsy without SE was higher in malignant ovarian teratoma (HR, 1.661; 95% CI 1.358-2.033) than in benign (HR, 1.172; 95% CI 1.037-1.324). Testicular teratoma did not show significant relations with epileptic events. The risk of epileptic events showed a tendency to decrease after removing the ovarian teratoma. This study found that ovarian teratoma is associated with a higher risk of epileptic events, especially in malignant tumors, whereas testicular teratoma did not show significant differences in epileptic events compared to the control group. This study adds to the current understanding of the association between gonadal teratoma and epileptic events.
Topics: Male; Humans; Female; Retrospective Studies; Teratoma; Epilepsy; Status Epilepticus
PubMed: 37433861
DOI: 10.1038/s41598-023-38255-x -
Journal of Pediatric Surgery Jun 2022With increased surgeon comfort using laparoscopy, we hypothesized resection of pediatric ovarian dermoids using laparoscopy would yield a shorter length of stay and no...
BACKGROUND
With increased surgeon comfort using laparoscopy, we hypothesized resection of pediatric ovarian dermoids using laparoscopy would yield a shorter length of stay and no increase in morbidity or recurrence compared to laparotomy.
METHODS
A retrospective review was performed amongst eleven pediatric hospitals. Patients aged 2 to 21 who underwent resection of an ovarian dermoid from 2010 to 2020 were included. Patient characteristics, operative details, and outcomes by approach were evaluated using Chi-squared and Wilcoxon-Mann tests.
RESULTS
466 patients were included, with a median age of 14.4 and median follow-up of 4.0 months. 279 patients underwent laparoscopy (60%), 139 laparotomy (30%), and 48 laparoscopy converted to laparotomy (10%). There were no differences in rates of tumor spillage by approach (p = 0.15). 65% underwent ovarian-sparing surgery and 35% underwent oophorectomy. Length of stay was significantly shorter amongst patients who underwent laparoscopy (1 day versus 2 days for laparotomy and converted, p<0.0001). There were no differences in rates of suspected recurrence or reoperation (p = 0.19 and p = 0.57, respectively).
CONCLUSION
Patients who underwent laparoscopy experienced no differences in the rates of tumor spillage, recurrence, or reoperation and had a shorter length of stay compared to laparotomy. Laparoscopy is an acceptable approach for resection of pediatric ovarian dermoids.
Topics: Child; Dermoid Cyst; Female; Humans; Infant; Laparoscopy; Laparotomy; Ovarian Neoplasms; Postoperative Complications; Retrospective Studies; Teratoma
PubMed: 35292164
DOI: 10.1016/j.jpedsurg.2022.01.053 -
Journal of Minimally Invasive Gynecology May 2021To review short- and long-term complications associated with intraoperative rupture of benign ovarian cysts. (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
To review short- and long-term complications associated with intraoperative rupture of benign ovarian cysts.
DATA SOURCES
The Cochrane Central Register of Controlled Trials, BIOSIS, Medline (Ovid), Web of Science, ClinicalTrials.gov, and Google Scholar were searched using the following terms and their combinations: "spillage," "rupture," "leakage," "ovarian cyst," "teratoma," "dermoid," "operative," "surgery," "outcome."
METHODS OF STUDY SELECTION
Randomized controlled and observational studies evaluating the operative outcomes of surgical treatment of ovarian cysts with intraoperative spillage compared with those of surgical treatment of ovarian cysts without spillage were included. A systematic review and meta-analysis following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines was performed.
TABULATION, INTEGRATION, AND RESULTS
A total of 28 studies were included in the qualitative analysis and 12 in the quantitative analysis. Ovarian cyst diameter was not found to be associated with the risk for spillage (relative risk [RR] 0.75; 95% confidence interval [CI], -0.33 to 1.82). Intraoperative benign ovarian cyst rupture was not associated with adverse short- and long-term outcomes such as reoperation (RR 1.16; 95% CI, 0.39-3.48), infertility (RR 0.73; 95% CI, 0.15-3.63), transient fever (RR 3.22; 95% CI, 0.83-12.51), and readmission (RR 1.00; 95% CI, 0.33-2.98). However, intraoperative spillage was found to be associated with increased risk for benign recurrence (RR 3.1; 95% CI, 1.05-9.14). A subgroup analysis of the studies that included only dermoid cysts showed an association between intraoperative cyst rupture and postoperative chemical peritonitis (RR 9.36; 95% CI, 1.20-73.28).
CONCLUSION
Intraoperative ovarian cyst spillage of a benign cyst is associated with limited adverse clinical outcomes. Although the surgical approach (minimally invasive vs open) should not be affected by the concern regarding an intraoperative cyst rupture, maximal efforts should be made to prevent intra-abdominal spillage.
Topics: Female; Humans; Laparoscopy; Neoplasm Recurrence, Local; Ovarian Cysts; Peritonitis; Postoperative Complications; Teratoma
PubMed: 33279627
DOI: 10.1016/j.jmig.2020.11.025 -
Journal of Ultrasound in Medicine :... Sep 2022To describe the ultrasonographic signs of ovarian collision tumors and evaluate the malignancy risk using the O-RADS system.
OBJECTIVES
To describe the ultrasonographic signs of ovarian collision tumors and evaluate the malignancy risk using the O-RADS system.
METHODS
This was a retrospective analysis of 25 ovarian collision tumors from 8739 patients between May 2010 and January 2020. All clinical characteristics, ultrasound images, and histological findings were collected and analyzed. Using the O-RADS lexicon descriptors, the O-RADS score was determined by two senior doctors. Lesions with O-RADS scores of 1 to 3 were classified as benign tumors, and lesions with O-RADS scores of 4 to 5 were classified as malignant tumors.
RESULTS
The mean age of the 25 patients was 30.4 years. Histological findings showed that all tumors were a combination of mature cystic teratomas and cystadenomas/cystadenocarcinomas. There were 11 benign tumors of O-RADS 2 or 3, including 3 uniocular cysts, 3 dermoid cysts, and 5 dermoid cysts with an anechoic fluid cavity. There were 12 benign tumors of O-RADS 4, including 3 uniocular-multilocular cysts with solid components, 2 multilocular cysts with irregular walls, and 8 multilocular cysts with dermoid sacs. One borderline tumor of O-RADS 4 was a multilocular cyst with irregular septation. One malignant tumor of O-RADS 5 was a multilocular cystic tumor with solid components and ascites.
CONCLUSIONS
The most common ovarian collision tumor was the coexistence of a mature cystic teratoma and a cystadenoma. The O-RADS system was able to sensitively detect malignant tumors in this series. A typical dermoid cyst with an anechoic cavity or a multilocular cyst with a dermoid sac may signify a benign collision tumor.
Topics: Adult; Dermoid Cyst; Female; Humans; Ovarian Neoplasms; Retrospective Studies; Risk Assessment
PubMed: 34931350
DOI: 10.1002/jum.15919 -
Reproductive Biology and Endocrinology... Nov 2020Ovarian teratoma-associated anti-N-methyl-D-aspartate receptor encephalitis (NMDAR-E) is a severe autoimmune neurological disorder, and the influence of teratoma-induced...
BACKGROUND
Ovarian teratoma-associated anti-N-methyl-D-aspartate receptor encephalitis (NMDAR-E) is a severe autoimmune neurological disorder, and the influence of teratoma-induced autoantibodies on the pathogenesis remains unclear.
METHODS
Ovarian teratoma tissues were collected from teratoma patients with and without NMDAR-E. Proteins were extracted and then analyzed using iTRAQ-coupled LC-MS/MS, which was followed by bioinformatics analysis. Candidate proteins were verified by Western blotting and immunohistochemistry.
RESULTS
In total, 36 differentially expressed proteins (DEPs) were identified between the control group and NMDAR-E group, and the bioinformatics analysis revealed that the DEPs were mainly involved in immune-related pathways, especially HLA-A and HLA-DRB1. The western blotting results for HLA-A and HLA-DRB1 were consistent with the results of the iTRAQ analysis. Additionally, the immunohistochemical data revealed that the aggregation of HLA-A (+) and HLA-DRB1 (+) cells was more apparent in the teratoma tissues of NMDAR-E patients compared with that in the tissues of controls.
CONCLUSION
Our investigation indicated that HLA-A and HLA-DRB1 might be involved in mediating ovarian teratoma-associated NMDAR-E. These findings provide new insights into the pathophysiological mechanisms and provide information for the functional exploration of proteins in the future.
Topics: Adolescent; Adult; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Case-Control Studies; China; Female; Genetic Association Studies; Genetic Predisposition to Disease; HLA-A Antigens; HLA-DRB1 Chains; Humans; Ovarian Neoplasms; Teratoma; Young Adult
PubMed: 33160385
DOI: 10.1186/s12958-020-00661-5 -
Archives of Gynecology and Obstetrics Aug 2020Anti-NMDA receptor antibody (anti-NMDAr) encephalitis, although still a rare condition, is well known to neurologists as it is the leading cause of non-infectious acute... (Review)
Review
BACKGROUND
Anti-NMDA receptor antibody (anti-NMDAr) encephalitis, although still a rare condition, is well known to neurologists as it is the leading cause of non-infectious acute encephalitis in young women. However, this is less well known to gynecologists, who may have a decisive role in etiological management. Indeed, in 30-60% of cases in women of childbearing age, it is associated with the presence of an ovarian teratoma, whose removal is crucial in the resolution of symptomatology.
OBJECTIVES
Primary objective of our work was to present a review in a very schematic and practical way for gynecologists, about the data on anti-NMDAr encephalitis in terms of epidemiology, clinical symptomatology, treatment and prognosis. The second objective was to propose a decision tree for gynecologists to guide them, in collaboration with neurologists and anesthesiologists, after the diagnosis of NMDAr encephalitis associated with an ovarian mass.
METHOD
We conducted an exhaustive review of existing data using PubMed and The Cochrane Library. Then, we illustrated this topic by presenting two typical cases from our experience.
RESULTS
Anti-NMDA antibody encephalitis association with an ovarian teratoma is common, especially in women of reproductive age. Complementary examinations in search of an ovarian teratoma must therefore be systematic to envisage a possible surgical excision that may improve patient prognosis.
CONCLUSION
Anti-NMDA antibody encephalitis should not be ignored by gynecologists whose role in management is central.
Topics: Adult; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Antibodies; Female; Humans; Ovarian Neoplasms; Physical Examination; Prognosis; Receptors, N-Methyl-D-Aspartate; Reproduction; Teratoma; Young Adult
PubMed: 32556515
DOI: 10.1007/s00404-020-05645-9