Ovarian Mature Teratoma Research Articles

Prevalence of metachronous contralateral mature ovarian teratoma: A systematic review.

There is increasing recognition that contralateral metachronous tumor may occur following treatment of unilateral mature ovarian teratoma. We aimed to define this risk to guide appropriate surveillance strategies. We undertook a systematic review of three large medical databases (Ovid Medline, Embase, and Cochrane Controlled Trials Register) to April 2020 using a defined search strategy. From 1831 articles retrieved, 23 were included, reporting 1101 girls with unilateral mature ovarian teratomas. The intensity and duration of follow-up varied between studies, with only five reporting close surveillance. Overall prevalence of metachronous contralateral mature teratoma was 2.1%, with a prevalence per study of 0%-23% (median 0%). Prevalence was higher (7%) among studies with more robust surveillance. These data suggest a small but real risk of metachronous contralateral tumors. Surveillance ultrasonography is proportionate and indicated alongside further prospective data collection to record the natural history and impact of surveillance in greater detail.

Methylation profile of imprinted genes provides evidence for teratomatous origin of a subset of mucinous ovarian tumours.

Mucinous ovarian tumours are sometimes associated with mature teratomas. It is suggested that the mucinous tumours in this setting are derived from teratomas, but there remains the possibility of collision or metastasis from extra-ovarian sites. Because mature ovarian teratomas are considered to be parthenogenetic tumours that arise from a single oocyte/ovum, they have only a maternal genome and therefore show maternal genome imprinting. If mucinous ovarian tumours originate from teratomas, their genome imprinting is theoretically maternal. One of the most important mechanisms of genome imprinting is DNA methylation. In the present study, we analysed a total of 28 mucinous ovarian tumours (7 with teratomas, 21 without teratomas; 14 malignant, 14 borderline) to clarify the methylation profiles of their imprinted genes using methylation-specific multiplex ligation-dependent probe amplification (MS-MLPA) of 21 imprinting control regions (ICRs) of nine imprinted genes/gene clusters using formalin-fixed, paraffin-embedded samples. All cases lacked evidence of an extra-ovarian primary mucinous tumour. In all seven mucinous tumours with teratomas, the overall methylation profile of mucinous tumours was comparable to that of teratomas, although some ICRs showed aberrant methylation. In contrast, all but one of the mucinous tumours without teratomas showed somatic or irregular methylation patterns. Morphologically, there was little teratomatous tissue in some mucinous tumours carrying teratoma-type methylation profiles, suggesting that mucinous tumours overwhelmed ancestral teratomas. In conclusion, the methylation profile of imprinted genes provides evidence that a subset of mucinous ovarian tumours originated from mature teratomas. Genome imprinting-based analysis is a promising strategy to verify the teratomatous origin of human tumours. © 2021 The Pathological Society of Great Britain and Ireland. Published by John Wiley & Sons, Ltd.

Mature cystic ovarian teratomas: Relationship between histopathological contents and clinical features

Objectives: Mature cystic teratomas (MCT) originate from three germ layers: ectoderm, mesoderm and endoderm. The predictive significance of tumor markers in mature cystic teratomas is still unclear. In this study, we aimed to evaluate the predictive value of tumor markers in ovarian MCTs and histopathological contents of tumor. Materials and Methods: The data of 106 patients who were operated for ovarian MCT between 2015 and 2020 were analyzed retrospectively. In addition, slides in the pathology archive were re-examined under a microscope to evaluate their histopathological contents. Results: The average age of the patients was 35.7; 26.7 in the cystectomy group and 42.1 in the oophorectomy group (p = 0.000). The ratio of tumor markers exceeding the cut off value for carbohydrate antigen (CA 19-9), lactate dehydrogenase (LDH), cancer antigen (CA 125), carcinoma antigen (CA 15-3), carcinoembryonic antigen (CEA) were 45.2%; 47.7%; 8.15%; 12.3%; 6.9%, respectively. Histopathological examination of MCTs revealed 99.1% ectoderm, 45.3% mesodermal, 39.6% endodermal origin. Conclusion: Mature cystic teratoma is a benign tumor of the reproductive age that contains three germ layers in various proportions. LDH and CA 19-9 can be a helpful tool in predicting mature cystic teratomas.

Anti-N-Methyl-D-Aspartate Receptor Encephalitis Associated with Ovarian Teratoma in South China-Clinical Features, Treatment, Immunopathology, and Surgical Outcomes of 21 Cases.

Objective To study the clinical characteristics and surgical outcomes of anti-NMDAR encephalitis and the immunopathology of associated teratomas. Methods Twenty-one patients were enrolled in this retrospective study, who were diagnosed with anti-NMDAR encephalitis with ovarian teratoma and admitted to two tertiary hospitals in South China from July 2014 to December 2019. The clinical data of patients were reviewed. Comparisons were made between the patients with different outcomes after surgery. Immunohistochemical analyses of associated ovarian teratomas were performed. Results The mean age of the patients was 24.33 ± 5.12 years. The peak seasons of disease onset were autumn and winter (30.61% and 32.65%). The symptoms could be divided into 8 categories, including psychiatric abnormalities, seizures, movement dysfunction, consciousness disorders, autonomic dysregulation, speech disturbance, central hypoventilation, and memory deficits. All patients developed four or more categories of symptoms within the first four weeks. Twelve patients (57.1%) had a maximum mRS of 5, and 11 patients (52.4%) were admitted to ICU. Twenty patients received surgery, and only 3 patients were diagnosed pathologically with immature ovarian teratomas, while the other 17 patients had mature ovarian teratomas. After surgery, 17 patients (85.0%) got clinical improvement. The central hypoventilation symptom and mature ovarian teratomas were associated with surgical outcome. Immunohistochemical analysis revealed that there were NMDAR-positive neural tissues in all 8 teratomas and in which 3 cases also contained large numbers of NMDAR-positive sebaceous glands and squamous epithelial tissues. Conclusion The disease is of high prevalence in autumn and winter. The central hypoventilation symptom and mature ovarian teratomas were associated with surgical outcome. NMDAR-positive neural tissue is not the only etiological factor of encephalitis. We speculate that encephalitis development in some patients may result from NMDAR expression in sebaceous glands and squamous epithelial tissues.

“Black Ovaries”: An Uncommon Case of First Systemic Recurrence of Melanoma

Here we describe a rare case of a 48-year-old woman with a previous history of malignant melanoma (pT2a pathological stage, IB clinical stage) occurring about five years previously. She complained of abdominal pain and pelvic discomfort, diagnosed as a consequence of a bilateral ovarian solid masses completely occupying the recto-uterine space. She underwent laparotomy surgery with total hysterectomy and bilateral salpingo-oophorectomy. Gross examination of the excised material revealed an unusual grey-black bilateral ovarian tumor; a histopathological diagnosis of ovarian bilateral metastatic melanoma was made. Imaging study (CT/MRI) did not reveal metastasis in other zones of the body. Melanoma metastasis usually affects the skin, liver, brain and lungs, and rarely gynecological localizations. On the other hand, most bilateral ovarian tumors are comprised of serous carcinoma, mature teratoma and gastrointestinal carcinoma metastasis. Exceptionally, primary ovarian melanoma may arise in mature ovarian cystic teratomas.

CT and MRI characteristics of ovarian mature teratoma in patients with anti-N-methyl-D-aspartate receptor encephalitis

PurposeThe 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 MethodsA 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. ResultsIn 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. ConclusionBy 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.

Ovarian Teratoma and N-Methyl-D-Aspartate Receptor Autoimmune Encephalitis: Insights Into Imaging Diagnosis of Teratoma and Timing of Surgery

Mature ovarian teratoma is the most common type of germ cell tumor and mostly detected incidentally in women in second or third decade. Recent researches have provided strong evidence between mature ovarian teratoma and encephalitis, which is caused due to the presence of anti-N-methyl-D-aspartate (NMDA) antibodies. We would like to report four patients who had typical neuropsychiatric manifestations, such as abnormal behavior, speech disorder, seizures, movement disorders, loss of consciousness, and autonomic dysfunction. All the patients had prolonged course of disease before the diagnosis of NMDA encephalitis had been made. The importance of timely evaluation of symptomatic young women with serum and cerebrospinal fluid (CSF) NMDA receptor (NMDAR) antibodies is emphasized. Imaging ranging from transvaginal ultrasound to positron emission tomography-computed tomography (PET-CT) will help in arriving at a probable diagnosis. Surgical treatment is the cornerstone of management, and patients responded well post surgery with almost full recovery, with addition of immunotherapy, physiotherapy, rehabilitation providing a vital role. There is significant variation in clinical presentation of encephalitis and thus in time to diagnosis for those with non-specific symptoms, particularly psychiatric ones. The index of suspicion of anti-NMDA encephalitis should be high in young females and involvement of a gynecology team needs to be done. Surgical removal of the teratoma is the key and the gynecology team should not be deterred by the acute symptoms and debilitating nature of the symptoms J Clin Gynecol Obstet. 2021;10(1):22-27 doi: https://doi.org/10.14740/jcgo715

Clinical analysis of 12 cases of ovarian cystic mature teratoma with malignant transformation into squamous cell carcinoma.

ObjectiveThis study aimed to examine the clinicopathological characteristics, treatment, and prognostic factors in 12 cases of malignant transformation of mature cystic teratoma of the ovary (MCTO).MethodsWe performed a retrospective study of 12 patients with malignant transformation of MCTO who were admitted to the Affiliated Hospital of Qingdao University from 2003 to 2019. We examined case records, clinical parameters, and biological assessments.ResultsThe median age of the patients was 56.5 years and seven of them were postmenopausal. The average tumor size was 18.5 cm. All patients had pelvic masses at their first hospital visit. Nine of the patients had discomfort in the lower abdomen, two presented with a lower abdominal palpable mass, and three were complicated by fever. The median follow-up time was 73 months (12‒193 months). Ten patients survived with a disease-free status and two died.ConclusionsThere is a low incidence of malignant transformation of MCTO, and its most common histological type is squamous cell carcinoma. Age and tumor size are important factors in malignant transformation of teratomas. While there is a lack of treatment guidelines for malignant transformation of MCTO, early diagnosis and treatment may be beneficial for these patients.

Rare case of BRAF V600E mutant anaplastic pleomorphic xanthroastrocytoma in a 5-year survivor of acute lymphoblastic leukaemia

A 20-year-old woman with a history of acute lymphoblastic leukaemia treated at 13 years old with radiation and chemotherapy and a benign cystic mature ovarian teratoma diagnosed at 18 years...

Rapid malignant transformation of mature ovarian cystic teratoma into undifferentiated carcinoma

Abstract Introduction: Malignant transformation of mature cystic teratoma (MCT) is extremely rare and has poor prognosis. We report a case of malignant transformation of ovarian MCT into undifferentiated carcinoma, that showed extremely rapid progression after surgery. Patient concerns: A 51-year-old woman presented with complaints of abdominal swelling, pain and fever. Computed tomography (CT) and transvaginal ultrasonography revealed a cystic mass in the pelvis. Diagnosis: Based on the CT findings, ovarian tumor with possible rupture was provisionally diagnosed. However, post-operative histopathology showed malignant transformation of mature ovarian cystic teratoma into undifferentiated carcinoma. Interventions: Total abdominal hysterectomy with bilateral salpingo-oophorectomy and omentectomy was performed. Outcomes: Positron emission tomography with 18-fluorodeoxyglucose (FDG-PET) after surgery showed multiple FDG uptake in liver, paraaortic lymph nodes and mesentery. Adjuvant chemotherapy was started, but the patient died 3 months after the surgery due to disease progression. Autopsy findings confirmed the metastases of undifferentiated carcinoma in liver, mesentery and left lung. Conclusions: Malignant transformation of MCT into undifferentiated carcinoma can be rapid and has poor outcomes. Pre-operative diagnosis is very difficult. Moreover, limited literature makes it difficult to establish the standardized treatment strategy for these patients. This case report shows that progression of this malignancy can be rapid despite surgery and chemotherapy.

High Frequency of Malignant Transformations on Ovarian Mature Teratomas at a Single University Hospital, Jakarta, Indonesia (2015-2018).

High Frequency of Malignant Transformations on Ovarian Mature Teratomas at a Single University Hospital, Jakarta, Indonesia (2015-2018).

Lack of genetic homozygosity in prepubertal teratomas: divergent pathogenesis distinct from that of teratomas in adolescents

AbstractIn adults, both immature and mature ovarian teratomas show frequent genetic homozygosity consistent with tumorigenesis involving germ cells after meiosis I. Investigation into genetic zygosity of various teratomas in children has been limited. Thirteen sacrococcygeal, 12 ovarian, and 3 testicular teratomas in children 18 years or younger were retrieved from our departmental archives and histologically reviewed. Tumor and paired normal tissues were microdissected and subjected to short tandem repeat (STR) genotyping. DNA genotyping was informative in 12 sacrococcygeal teratomas, 8 ovarian teratomas, and 3 testicular teratomas. Sacrococcygeal teratomas included seven mature teratomas, four immature teratomas, and one mixed germ cell tumor with patient age ranging from 0 days to 3 years. All but two patients were female. Ovarian teratomas included five mature and three immature teratomas with patient age ranging from 2 to 18 years. Testicular teratomas included two mature teratomas and one immature teratoma with patient age ranging from 3 months to 3 years. All sacrococcygeal, testicular, and ovarian teratomas in patients younger than 4 years showed no evidence of genetic homozygosity by STR genotyping. In contrast, all four ovarian teratomas in patients older than 9 years showed either partial or complete homozygosity. In conclusion, unlike adolescent and adult ovarian teratomas, prepubertal sacrococcygeal and gonadal teratomas lack genetic homozygosity, supporting the hypothesis that teratomas before puberty develop at an early stage of germ cell development different from that of teratomas in adolescents and adults.To investigate genetic homozygosity in pediatric teratomas, this study analyses 13 sacrococcygeal, 12 ovarian, and 3 testicular teratomas in children by short tandem repeat (STR) genotyping. While genetic homozygosity is frequent in adult ovarian teratomas, no evidence of genetic homozygosity is seen in patients younger than 4 years, arguing pediatric teratomas arise at an earlier stage of germ cell development.

Unusual Case of Pseudomyxoma Peritonei of a Mature Teratoma in Adult Men

Pseudomyxoma peritonei (PMP) is a rare disease usually originating from appendiceal neoplasm and its incidence is 1-2 cases per million. Extra-appendicular origin is less common and is related to ovary, urachus, stomach, colon and pancreas cancer. A few cases of mature ovarian teratoma associated with PMP have been described. We report an extremely rare case of a man treated for a mature teratoma recurrence as a PMP. A 57-year-old man with multiple surgeries for mature teratoma excision as a newborn was addressed to our institution. At the age of 55, a surgical exploration found a low-grade PMP. The patient was asymptomatic, and surveillance was performed. Nine months later, a sub-complete cytoreductive surgery (CRS) with Mitomycin C HIPEC (hyperthermic intraperitoneal chemotherapy) was performed. One year later, the residual mass volume was treated by a new CRS with Mitomycin C HIPEC. After a two-year follow-up patient has no recurrence. PMP extra-appendiceal origin in less than 10%. This is the first reported case of PMP originating as a mature teratoma recurrence in a man. A few cases describe PMP from ovarian teratoma in women. Despite PMP’s different origins, gold standard treatment still remains CRS and HIPEC.

Ovarian Mature Cystic Teratoma Combined with Mucinous Boderline Tumor-A Case Report with Literature Review

&lt;p&gt;Mature ovarian cystic teratoma with co-existing mucinous borderline tumor is an extremely rare finding in literature. Here, we report a case of bilateral mature cystic teratoma of the ovary with mucinous borderline tumors and intraepithelial carcinoma in a very young, 26-year-old female. The case was diagnosed histologically through immunohistochemical support. The biological behavior of mature ovarian cystic teratoma with mucinous borderline tumor is between those of benign and malignant tumors, having low malignant potential. Since the patient had given birth to a child, a radical operation was performed and no&amp;nbsp;&lt;br /&gt;tumor recurrence was observed after one year follow-up.&lt;/p&gt;

Ovarian Sparing Surgery in Mature Ovarian Teratomas in Children: A 20-Year Single-Center Experience.

Despite its benign nature, possible bilateral presentation, and a very good prognosis, ovarian sparing tumorectomy (OST) in mature ovarian teratoma (MOT) is not commonly performed. Unilateral oophorectomy has physiological consequences, while bilateral is devastating. The aim of this study is promotion of OST in MOT among children. We reviewed 120 patients operated for MOT between August 1999 and 2019. Ovariectomy was performed in 15 patients (14 between 1999 and 2009 and 1 after 2010). In 105 girls, OST was possible including 32 with ovarian torsion. The approach was laparoscopy in 30 girls (11 conversions) and laparotomy in 94. Intra-abdominal spillage occurred in 30% of laparoscopic dissections. Postoperative morbidity was not associated with surgical approach (p = 0.613) or presence of adnexal torsion (p = 0,608). Follow-up was from 4 months to 9 years (median = 4 years) with access to 90% of patients. Bilateral lesions were observed in six (synchronous in five and metachronous in one) patients. Recurrence appeared in three patients operated via laparotomy and OST (after 12, 46, and 74 months). In one girl, asynchronous contralateral MOT was found 5 years after unilateral oophorectomy. Ovarian regeneration after torsion was observed in sonography in 84.4% of the patients. None of the patients experienced chemical peritonitis or malignant tumor transformation. OST is safe and effective and should be the first-line procedure in children. Laparoscopy and laparotomy constitute a complementary approach to MOT. Ultrasound follow-up is necessary to monitor recurrence, contralateral disease, and ovarian regeneration.

Mature Ovarian Cystic Teratoma Mimicking a Bladder Diverticulum in a 16-year-old Female

A 16-year-old female was incidentally found to have an abnormal pelvic ultrasound while undergoing evaluation for mild scoliosis. A large, thick-walled, lobular, fluid-filled structure was found at midline in the pelvis that initially resembled bladder diverticula due to its anatomical position and sonographic appearance. Voiding cystourethrogram and computed tomography later revealed a large cystic mass originating from the right ovary. This report outlines a case of a mature cystic teratoma convincingly mimicking bladder anomaly on sonography.

Methylation profiles of imprinted genes are distinct between mature ovarian teratoma, complete hydatidiform mole, and extragonadal mature teratoma

Mature ovarian teratoma is considered to be a parthenogenetic tumor that arises from a single oocyte/ovum. Conversely, complete hydatidiform mole (CHM) is androgenetic in origin: classic CHM arises from a single or two sperm. Since mature ovarian teratoma and CHM have only maternal and paternal genomes, respectively, their genome imprinting is theoretically reverse, but this has yet to be investigated. Genome imprinting in struma ovarii, a special form of mature teratoma, remains unclear. Although a mature teratoma can rarely arise in extragonadal sites, its genome imprinting, as well as cell origin, is poorly understood. One of the most important mechanisms of genome imprinting is DNA methylation. To investigate the methylation profile of imprinted genes, we performed methylation-specific multiplex ligation-dependent probe amplification (MS-MLPA) of 21 imprinting control region (ICRs) of 9 imprinted genes/gene clusters in formalin-fixed, paraffin-embedded samples obtained from 12 mature ovarian teratomas, 6 struma ovarii, 10 CHMs, and 7 extragonadal (1 sacrococcygeal, 6 mediastinal) mature teratomas of females. In mature ovarian teratomas, ICRs of maternally and paternally imprinted genes showed high and low levels of methylation, respectively, and this pattern was almost reverse in CHMs. In CHMs, however, some ICRs showed aberrant methylation. The methylation profile of struma ovarii was comparable to that of mature ovarian teratomas, except for an adenomatous tumor. In extragonadal mature teratomas, the methylation pattern was somatic or irregular. In conclusion, mature ovarian teratomas/struma ovarii, CHMs, and extragonadal mature teratomas showed distinct methylation profiles of imprinted genes. Ovarian teratomas and CHMs are most likely to inherit their methylation profiles from their ancestral germ cells, although some aberrant methylation suggests a relaxation of imprinting in CHMs and a subset of struma ovarii. Extragonadal mature teratomas may carry a methylation profile of misplaced primordial germ cells or possibly somatic cells that have been reprogrammed in vivo.

Gliomatosis peritoneal asociada a teratoma maduro de ovario

Los teratomas de ovario son tumores compuestos por tejidos derivados de las tres líneas germinales embrionarias. La gliomatosis peritoneal es una afección poco frecuente caracterizada por implantes de tejido glial en el peritoneo comúnmente asociada con teratoma inmaduro de ovario, no así con teratomas maduros. El diagnóstico definitivo se realiza por histología. No existen pautas sobre cómo realizar el seguimiento de los pacientes con esta afección. El pronóstico es favorable. Sin embargo, los implantes pueden experimentar regresión fibrótica o excepcionalmente degenerar a glioblastoma. Si están presentes tejido glial inmaduro u otros componentes teratomatosos en el peritoneo o epiplón, el tratamiento será similar al del carcinoma metastásico de ovario. En pacientes con enfermedad peritoneal residual, es necesario un seguimiento a largo plazo, debido al riesgo de recurrencia y transformación maligna. Se presenta un caso de gliomatosis peritoneal asociado a teratoma maduro de ovario.

The Poké Ball sign in mature ovarian cystic teratoma.

The Poké Ball sign in mature ovarian cystic teratoma.

Generation of human androgenetic induced pluripotent stem cells

In humans, parthenogenesis and androgenesis occur naturally in mature cystic ovarian teratomas and androgenetic complete hydatidiform moles (CHM), respectively. Our previous study has reported human parthenogenetic induced pluripotent stem cells from ovarian teratoma–derived fibroblasts and screening of imprinted genes using genome-wide DNA methylation analysis. However, due to the lack of the counterparts of uniparental cells, identification of new imprinted differentially methylated regions has been limited. CHM are inherited from only the paternal genome. In this study, we generated human androgenetic induced pluripotent stem cells (AgHiPSCs) from primary androgenetic fibroblasts derived from CHM. To investigate the pluripotency state of AgHiPSCs, we analyzed their cellular and molecular characteristics. We tested the DNA methylation status of imprinted genes using bisulfite sequencing and demonstrated the androgenetic identity of AgHiPSCs. AgHiPSCs might be an attractive alternative source of human androgenetic embryonic stem cells. Furthermore, AgHiPSCs can be used in regenerative medicine, for analysis of genomic imprinting, to study imprinting-related development, and for disease modeling in humans.