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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 Dec 2015Ovarian preservation is desirable in children with ovarian tumors. However, the diagnostic and treatment strategies are heterogeneous. The aim of this study was to...
BACKGROUND/PURPOSE
Ovarian preservation is desirable in children with ovarian tumors. However, the diagnostic and treatment strategies are heterogeneous. The aim of this study was to investigate the management and preservation of ovarian tissue in order to identify the factors associated with ovarian preservation.
METHODS
Thirty-seven patients (41 ovaries) were surgically treated for ovarian tumors. Four cases were bilateral. The data on the patient symptoms at presentation, imaging, treatment, outcome, pathology, and status of ovarian preservation were retrospectively analyzed for each patient.
RESULTS
Histological examinations revealed 25 mature teratomas, 5 immature teratomas, 5 cystadenomas, 2 dysgerminomas, and other tumors. Ovarian torsion occurred in 16 ovaries (39%). Ovary-sparing surgery was performed in 22 ovaries (53.7%). Successful ovarian preservation was significantly associated with a smaller tumor size, benign pathology, and a lower degree of torsion (P<0.01).
CONCLUSIONS
Because the prognosis was favorable in most cases, the preservation of fertility and gonadal function should be a goal in the surgical treatment of ovarian tumors. We recommend ovary-sparing surgery as the first-line treatment for all pediatric ovarian tumors other than those that are preoperatively diagnosed as being malignant and those in which emergent surgical intervention is indicated owing to the suspicion of ovarian torsion.
Topics: Adolescent; Child; Child, Preschool; Cystadenoma; Dysgerminoma; Female; Humans; Ovarian Diseases; Ovarian Neoplasms; Ovariectomy; Prognosis; Retrospective Studies; Teratoma; Torsion Abnormality; Young Adult
PubMed: 26385567
DOI: 10.1016/j.jpedsurg.2015.08.036 -
Gynecologic Oncology Apr 2020To evaluate the diagnostic, surgical, and oncological outcomes of patients with growing teratoma syndrome (GTS).
OBJECTIVES
To evaluate the diagnostic, surgical, and oncological outcomes of patients with growing teratoma syndrome (GTS).
METHODS
Patients diagnosed with ovarian immature teratoma (IMT) between 1980 and 2018 at Peking Union Medical College Hospital (PUMCH) were evaluated for the development of GTS. Their clinical characteristics, surgical and pathological data, and oncological outcomes were collected.
RESULTS
Between 1980 and 2018, 175 cases of IMT were referred to PUMCH. Thirty-five patients subsequently developed GTS with a crude rate of approximately 20%. The median interval between the initial diagnosis of IMT and the first occurrence of GTS was 18.5 months (range, 6-78 months). Residual disease (P < 0.001) and gliomatosis peritonei (GP) at initial surgery (P = 0.023) were independent risk factors for GTS development. Fertility-sparing surgery for GTS was performed in 27 patients and four patients achieved five singleton pregnancies. The median follow-up time was 73 months (range, 11-401 months). Eleven patients developed at least one recurrence. Residual disease after GTS surgery was associated with GTS recurrence (P = 0.001). By the end of follow-up, 27 patients were alive without disease and the other eight patients were alive with disease.
CONCLUSION
The presence of residual disease and GP at initial surgery are risk factors for GTS. Complete surgical resection is the cornerstone for treatment of GTS. The presence of residual disease after surgery for GTS is a risk factor for GTS recurrence. Fertility-sparing surgery should be performed because spontaneous pregnancy is possible. The overall prognosis of GTS is excellent.
Topics: Adolescent; Adult; Child; Female; Fertility Preservation; Humans; Neoplasm Staging; Neoplasm, Residual; Organ Sparing Treatments; Ovarian Neoplasms; Prognosis; Syndrome; Teratoma; Treatment Outcome; Young Adult
PubMed: 31954532
DOI: 10.1016/j.ygyno.2019.12.042 -
Diagnostic and Interventional Imaging 2021The purpose of this study was to determine the computed tomography (CT) and magnetic resonance imaging (MRI) characteristics of ovarian mature teratoma in patients with...
PURPOSE
The purpose of this study was to determine the computed tomography (CT) and magnetic resonance imaging (MRI) characteristics of ovarian mature teratoma in patients with anti-N-methyl-d-aspartate receptor encephalitis (NMDAR-E).
MATERIALS AND METHODS
A total of 125 women (mean age, 40.9±17.8 [SD] years; age range: 12-85 years) with 146 histopathologically or radiologically proven ovarian mature teratomas who underwent preoperative CT and MRI examinations were retrospectively included. Eight patients with 11 teratomas had NMDAR-E, whereas 117 patients with 135 teratomas did not have NMDAR-E. CT and MRI examinations were retrospectively reviewed and teratomas in patients with NMDAR-E were compared to those in patients without NMDAR-E. Comparisons were performed using Mann-Whitney U test or Fisher exact test.
RESULTS
In patients with NMDAR-E, maximum diameter of teratomas (26.1±9.3 [SD] mm), prevalence of teeth/calcification (36%) and rate of occupation by fat components (26%) were lower than those in patients without NMDAR-E (67.0±37.6 [SD] mm [P<0.01]; 75% [P<0.05]; and 65%[P<0.01], respectively). More than 75% of space was occupied by fat components in 76/135 teratomas (56%) in patients without NMDAR-E, whereas this was not observed in any teratoma in patients without NMDAR-E.
CONCLUSION
By comparison with teratomas in patients without NMDAR-E, teratomas in patients with NMDAR-E are smaller, have few teeth/calcification, and the amount of space occupied by fat components is smaller.
Topics: Adolescent; Adult; Aged; Aged, 80 and over; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Child; Female; Humans; Magnetic Resonance Imaging; Middle Aged; Ovarian Neoplasms; Retrospective Studies; Teratoma; Tomography, X-Ray Computed; Young Adult
PubMed: 33785312
DOI: 10.1016/j.diii.2021.02.010 -
Virchows Archiv : An International... Aug 2018Although ovarian monodermal teratomas, including struma ovarii and carcinoids, are closely associated with mature teratomas, their genetic basis is poorly understood. A...
Although ovarian monodermal teratomas, including struma ovarii and carcinoids, are closely associated with mature teratomas, their genetic basis is poorly understood. A series of mature and monodermal ovarian teratomas were analyzed by short tandem repeat genotyping to evaluate their genetic zygosity and its associations. Informative DNA genotyping data were obtained for ten mature teratomas, six struma ovarii, and three carcinoids (one insular, one trabecular, and one mucinous). A homozygous genotype was present in five of the ten (50%) mature teratomas, three of the six (50%) struma ovarii, and one of the three (33%) ovarian carcinoids. There was no significant difference in genetic zygosity between mature and monodermal teratomas. Patients' age was not correlated with the genetic zygosity: the youngest age in the homozygous tumor group of patients was 4 years. It is suggested that an oocyte after meiosis I, which has escaped from meiotic arrest, is a significant cause of these tumors. Although one mature teratoma was a rare case with lactating adenoma-like breast tissue, its genetic zygosity was concordant with that of the surrounding teratomatous tissue. In one ovarian carcinoid, the carcinoid and accompanying teratomatous components showed matching zygosity at all but one locus: the carcinoid was heterozygous but teratoma was homozygous at one pericentromeric locus. This suggests that not all carcinoids are secondary neoplasms arising from a fully developed mature teratoma: some are neoplasms deviating from a developing mature teratoma.
Topics: Adolescent; Adult; Aged, 80 and over; Carcinoid Tumor; Child, Preschool; Female; Genotype; Humans; Middle Aged; Ovarian Neoplasms; Struma Ovarii; Teratoma
PubMed: 29455317
DOI: 10.1007/s00428-018-2319-z -
Journal of Neuroimmunology Sep 2023Resection of an underlying ovarian teratoma in patients with N-Methyl-d-Aspartate receptor (NMDAR)-antibody encephalitis is supported by pathophysiological studies... (Meta-Analysis)
Meta-Analysis Review
Resection of an underlying ovarian teratoma in patients with N-Methyl-d-Aspartate receptor (NMDAR)-antibody encephalitis is supported by pathophysiological studies demonstrating the production of NMDAR antibodies within the teratoma. This systematic review assesses the clinical effect of teratoma resection and compares early versus late resection. Literature search was performed on the first of October 2022 (MEDLINE, Embase, CENTRAL, Web of Science). Original studies including more than three patients with NDMAR encephalitis and associated ovarian teratoma were included and evaluated with the Study Quality Assessment Tool for risk of bias. Fourteen studies referring to 1499 patients were included and analyzed in four syntheses using the fixed Mantel-Haenszel method. The rate of relapse in patients with ovarian teratoma resection was lower than in patients without resection (risk ratio for relapse 0.30, 95% CI 0.17-0.51), however the certainty level of evidence is very low. Despite some evidence pointing to a beneficial effect of early teratoma resection in patients with NMDAR-antibody encephalitis, systematically accessible data are insufficient to provide recommendations for or against resection, as well as for timing of surgery. The authors received no financial support for the research, authorship, or publication of this article. For the systematic review no clinical-trial database registration had been done.
Topics: Female; Humans; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Receptors, N-Methyl-D-Aspartate; Neoplasm Recurrence, Local; Ovarian Neoplasms; Teratoma; Autoantibodies
PubMed: 37499300
DOI: 10.1016/j.jneuroim.2023.578153 -
Journal of Healthcare Engineering 2022Anti-N-methyl-D-aspartate (NMDA) receptor encephalitis is a rare form of autoimmune encephalitis caused by anti-NMDA receptor antibodies. This disease mainly affects... (Review)
Review
BACKGROUND
Anti-N-methyl-D-aspartate (NMDA) receptor encephalitis is a rare form of autoimmune encephalitis caused by anti-NMDA receptor antibodies. This disease mainly affects women of childbearing age and is commonly associated with ovarian teratoma. However, the relationship between anti-NMDA receptor encephalitis and ovarian teratoma and the role of anti-NMDA receptor antibody in the relationship remain unclear.
OBJECTIVES
This study aimed to describe 15 cases of anti-NMDA receptor encephalitis (5 with ovarian teratoma), review literature, and reinforce the gynecologist's knowledge of this disorder.
METHODS
Clinical data of 15 patients from January 2015 to December 2020 admitted to The Second Hospital of Hebei Medical University were collected and analyzed. The diagnosis of anti-NMDA receptor encephalitis was based on the presence of anti-NMDA receptor antibodies in cerebrospinal fluid (CSF) and/or serum. Laparoscopic teratoma removal was performed in patients with ovarian teratoma. All patients had received immunotherapy. In addition, a review of the literature was performed to reinforce the gynecologist's knowledge of this disorder.
RESULTS
A total of 15 patients with anti-NMDA receptor encephalitis were screened, of whom 5 patients were confirmed with ovarian teratoma by pathology. The most common symptoms of anti-NMDAR encephalitis with teratoma are fever (5/5, 100%), seizure (5/5, 100%), mental and behavioral disorders (4/5, 80%), and decreased consciousness (4/5, 80%). Conversely, the most common symptoms of patients without teratoma were neuropsychiatric symptoms, including headache (6/10, 60%) and mental and behavioral disorders (7/10, 70%). All patients underwent immunotherapy, including steroids, intravenous immunoglobulin (IVIG), plasma exchange, and cyclophosphamide, and 4 out of 5 patients with ovarian teratomas underwent surgical treatment. All patients had a good outcome after systemic, surgical, and immunotherapy treatment. No patient who underwent surgical treatment developed a recurrence. Conversely, 2 of 10 patients without teratoma developed an anti-NMDA receptor encephalitis recurrence.
CONCLUSIONS
Patients with anti-NMDA encephalitis show severe mental and neurological symptoms. Resection of teratoma is beneficial to the relief or disappearance of symptoms and has a good prognosis. This disorder should be fully recognized by gynecologists, who play an important role in diagnosis and treatment.
Topics: Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Female; Humans; Ovarian Neoplasms; Retrospective Studies; Teratoma
PubMed: 35340259
DOI: 10.1155/2022/4299791 -
Revue Medicale de Liege Jan 2022Summmary : Teratomas are the most common histologic type of germ cell tumors in pediatrics. There are two types of teratomas, mature, benign and immature, malignant.... (Review)
Review
Summmary : Teratomas are the most common histologic type of germ cell tumors in pediatrics. There are two types of teratomas, mature, benign and immature, malignant. Initial diagnosis is essential for optimal management. This work, based on a clinical case, aims to review the clinical, radiological, biological and histological characteristics allowing them to be differentiated.
Topics: Child; Humans; Ovarian Neoplasms; Teratoma
PubMed: 35029339
DOI: No ID Found