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Journal of Ovarian Research Aug 2018Although teratomas are the most common histologic subtype of childhood ovarian germ cell tumors, their appropriate treatment in this age group still remains unclear....
BACKGROUND
Although teratomas are the most common histologic subtype of childhood ovarian germ cell tumors, their appropriate treatment in this age group still remains unclear. Paucity of research dedicated exclusively to both mature and immature teratomas of the ovary, contribute to decision making difficulties. Therefore, we decided to review retrospectively our experience in treatment of pediatric ovarian teratomas in order to assess the epidemiology, presenting features, and diagnostic as well as surgical management of these lesions.
RESULTS
The study comprised 58 patients. Fifty percent of patients were between 9 and 15 years old. Mature teratoma was diagnosed in 55(94.83%) patients, while 3(5.17%) patients presented with immature teratoma. Twenty eight (50.91%) girls with mature teratoma had laparotomy and 23 (41.82%) had laparoscopy performed as an initial operative approach. Ovarian tissue sparing technique (preservation of the ovarian tissue of the affected gonad) was applied in only 11.11% of patients operated in the first study period (years 1999-2003) and increased to 40.54% in the second half of our study (years 2004-2016). The extent of gonadal resection was not related with the size of the lesion. Bilateral lesions were noted in 8 patients with mature teratoma. All girls with immature teratoma were subjected to formal laparotomy. Two patients had stage III of the disease and one had stage IV. They underwent at least resection of the affected gonad. Adjuvant chemotherapy was given to all girls with immature teratoma after the surgery.
CONCLUSIONS
Under particular conditions ovarian-sparing surgery might be successfully applied in children with mature teratoma. Laparotomy is the treatment of choice in large masses, suspicious for malignancy and if surgical staging is required. High quality prospective multi-institutional studies are required in order to get an objective insight into biology and prognostic factors of teratomas in children.
Topics: Adolescent; Chemotherapy, Adjuvant; Child; Child, Preschool; Female; Humans; Infant; Infant, Newborn; Laparoscopy; Neoplasm Staging; Ovarian Neoplasms; Ovary; Retrospective Studies; Teratoma; Ultrasonography
PubMed: 30165903
DOI: 10.1186/s13048-018-0448-2 -
The Journal of International Medical... Apr 2017Objective To investigate the role of ovarian cystectomy in patients with early-stage immature teratoma. Methods A retrospective review was undertaken on patients...
Objective To investigate the role of ovarian cystectomy in patients with early-stage immature teratoma. Methods A retrospective review was undertaken on patients diagnosed pathologically with immature teratoma and with malignant lesions confined to the ovary. Patients were included if they had been treated between January 1997 and December 2015 at the Obstetrics and Gynaecology Hospital of Fudan University, Shanghai, China. Relevant demographic and clinical data were retrieved from the medical records. Results Forty-three patients were included in the study; 14 underwent ovarian cystectomy (group 1) and 29 underwent unilateral salpingo-oophorectomy (USO; group 2). Three of the patients who underwent USO relapsed and required a second surgical intervention. The 5-year disease-free survival rates were 100% and 88%for groups 1 and 2, respectively. There were no significant differences between the two groups in terms of survival or postoperative fertility outcomes. Univariate and multivariate analysis further revealed that ovarian cystectomy was not a poor prognostic indicator for disease-free survival. Conclusion These current data suggest that ovarian cystectomy can be considered for patients with apparent early-stage immature teratoma as it preserves fertility as much as possible without adversely impacting upon survival.
Topics: Adolescent; Adult; Child; Disease-Free Survival; Female; Fertility; Humans; Neoplasm Recurrence, Local; Neoplasm Staging; Ovarian Neoplasms; Ovariectomy; Ovary; Pregnancy; Reoperation; Retrospective Studies; Teratoma
PubMed: 28415950
DOI: 10.1177/0300060517692149 -
Journal of Neuro-ophthalmology : the... Mar 2022Opsoclonus-myoclonus syndrome (OMS) is a rare syndrome characterized by opsoclonus, which is irregular, spontaneous, multivectorial saccadic eye movements, along with...
Opsoclonus-myoclonus syndrome (OMS) is a rare syndrome characterized by opsoclonus, which is irregular, spontaneous, multivectorial saccadic eye movements, along with diffuse or focal myoclonus and sometimes ataxia. OMS is associated with paraneoplastic etiologies in 20%-40% of cases, with small-cell lung and breast cancers the most common associated primary neoplasms in adults, whereas neuroblastoma is more common in children and ovarian teratoma may occur in women younger than 30 years. Onconeural antibodies are often not identified. In existing literature, paraneoplastic OMS precedes identification of the neoplasm, and neurological recovery depends on treatment of the underlying cancer. We describe a 27-year-old woman with the delayed onset of OMS one month after resection of ovarian teratoma, likely due to immune trigger from antigen exposure at the time of resection. She was treated with intravenous methylprednisolone, immunoglobulins, and eventually rituximab with resolution of her symptoms. Identification of OMS after tumor resection and prompt immunotherapy are critical for neurologic recovery. At 30-month follow-up, this patient had not experienced recurrence of OMS.
Topics: Adult; Child; Female; Humans; Methylprednisolone; Opsoclonus-Myoclonus Syndrome; Ovarian Neoplasms; Teratoma
PubMed: 34417773
DOI: 10.1097/WNO.0000000000001384 -
The Turkish Journal of Pediatrics 2017Atıcı A, Yılmaz E, Karaman A, Apaydın S, Afşarlar ÇE. Tuba-ovarian auto-amputation caused by ovarian teratoma in an adolescent girl. Turk J Pediatr 2017; 59:...
Atıcı A, Yılmaz E, Karaman A, Apaydın S, Afşarlar ÇE. Tuba-ovarian auto-amputation caused by ovarian teratoma in an adolescent girl. Turk J Pediatr 2017; 59: 90-92. Ovarian auto-amputation is an extremely rare condition commonly encountered in the perinatal period. Spontaneous or secondary torsion of the ovary caused by an ovarian lesion may result in infarction and subsequent auto-amputation of the ovary. This paper demonstrates a case that underwent laparoscopic appendectomy with an incidental calcified auto-amputated right ovary. A 16-year-old adolescent girl was admitted to our department with a history of one-day abdominal pain. Physical examination of the patient revealed abdominal tenderness and rigidity on right lower quadrant. Her white blood cell count was 11x103/mL, and C-reactive protein was 69 mg/L. The patient underwent a laparoscopic appendectomy with a provisional diagnosis of acute appendicitis, and further exploration revealed a 2x2 cm white ovoid mass floating freely in the pelvis. The left ovary was clearly identified in its usual localization, but the right tuba was blindly ending without any fimbria or ovary. Postoperative course of the patient was uneventful, and she was discharged on postoperative day 2. The histopathological examination revealed a necrotic calcified ovarian teratoma. Auto-amputated ovary is a rare occasion mostly encountered during perinatal period, and it may be unilateral or bilateral. An auto-amputated ovarian mass may rarely be a teratoma although the most common cause of auto-amputation during perinatal and adolescent period is ovarian torsion due to an ovarian cyst.
Topics: Adolescent; Amputation, Surgical; Fallopian Tubes; Female; Humans; Laparoscopy; Ovarian Neoplasms; Ovary; Teratoma
PubMed: 29168372
DOI: 10.24953/turkjped.2017.01.017 -
Scientific Reports Feb 2021Teratomas in mice, composed of different tissue types, are derived from primordial germ cells (PGCs) in the foetal gonads. The strongest candidate gene in the testicular...
Teratomas in mice, composed of different tissue types, are derived from primordial germ cells (PGCs) in the foetal gonads. The strongest candidate gene in the testicular teratoma locus (Ter) responsible for testicular teratoma formation was identified as mutation in Dnd1, Dnd1R178*. However, the phenotype of mice with a mutated Dnd1 gene was germ cell loss. This suggests that other genes are involved in teratoma formation. Testicular teratomas can also be induced experimentally (experimentally testicular teratomas: ETTs) in 129/Sv mice by transplanting E12.5 foetal testes into adult testes. Previously, we mapped the ett1 locus, which is the locus responsible for ETT formation on chromosome 18. By exome sequence analysis of the 129 and LTXBJ (LT) strains, we identified a missense mutation in the melanocortin 4 receptor (MC4R) gene among 8 genes in the ett1 region. The missense mutation causes a substitution of glycine 25 by serine. Thus, this gene is a candidate for ETT formation. We established the LT-ett1 congenic strain, which introduced the locus responsible for ETT formation genetically into the genomes of a testicular teratoma non-susceptible strain. In this study, we crossed LT-ett1 and a previously established LT-Ter strain to establish the double congenic strain LT-Ter-ett1. Also, we established a strain with a point mutation in the MC4R gene of the LT strain by genome editing, LT-MC4R. Furthermore, double genetically modified strain LT-Ter-MC4R was established to address the relation between Ter and MC4R. Surprisingly, highly developed ovarian teratomas (OTs), instead of testicular teratomas, appeared not only in the LT-Ter-MC4R and LT-MC4R strains but also in the LT-ett1 and LT-Ter-ett1 strains. The incidence of OT formation was high in double genetically modified strains. The results demonstrated that MC4R is one of the genes responsible for OT formation. It was suggested that the effect of the missense mutation in MC4R on teratoma formation was promoted by abnormal germ cell formation by the mutation in DND1.
Topics: Amino Acid Substitution; Animals; CRISPR-Cas Systems; Female; Gene Editing; Male; Mice; Mutation; Mutation, Missense; Neoplasm Proteins; Oocytes; Ovarian Neoplasms; Point Mutation; Receptor, Melanocortin, Type 4; Teratoma; Testicular Neoplasms
PubMed: 33568756
DOI: 10.1038/s41598-021-83001-w -
Brain : a Journal of Neurology Nov 2022Penfield’s motor homunculus anthropomorphizes the cerebral level of motor control, the upper motor neuron. However, it leaves the cranial and spinal motor neurons...
Penfield’s motor homunculus anthropomorphizes the cerebral level of motor control, the upper motor neuron. However, it leaves the cranial and spinal motor neurons unrepresented. Here Ravits and Stack redress the imbalance by presenting a lower motor neuron homunculus.
Topics: Female; Humans; Motor Neurons; Teratoma; Ovarian Neoplasms
PubMed: 36029046
DOI: 10.1093/brain/awac310 -
Taiwanese Journal of Obstetrics &... Oct 2017Benign mature teratoma during pregnancy is common, mostly discovered incidentally by antenatal sonography. However, repeated pregnancy coincident with ovarian mature... (Review)
Review
OBJECTIVE
Benign mature teratoma during pregnancy is common, mostly discovered incidentally by antenatal sonography. However, repeated pregnancy coincident with ovarian mature teratoma is rarely reported. The cases of teratoma with rapid growing characteristics are even more unique.
CASE REPORT
A 17-year-old woman was pregnant at 6 weeks of gestation with a left ovarian teratoma. She underwent artificial abortion followed by surgical removal of the teratoma. However, eleven years after the surgery, a right ovarian teratoma was found incidentally by antepartum sonography at 21 weeks of gestation. The right ovarian teratoma developed uneventfully, with rapid growth during pregnancy. Abdominal delivery at term was accomplished without any complication.
CONCLUSION
Younger patients and patients with bilateral or large size dermoid cysts should be followed up closely. Further studies are needed for better understanding of its natural clinical course and the mechanism of progression. The treatment options should be made individually, weighing the risks of torsion, rupture, or obstruction of labor versus the potential for unnecessary surgical risk to mother and fetus.
Topics: Adolescent; Birth Intervals; Female; Gravidity; Humans; Live Birth; Ovarian Neoplasms; Pregnancy; Pregnancy Complications, Neoplastic; Teratoma; Ultrasonography, Prenatal
PubMed: 29037561
DOI: 10.1016/j.tjog.2017.08.021 -
Medicine May 2024Trophoblastic neoplasms are often associated with pregnancy, and nongestational trophoblastic neoplasms are extremely rare. Nongestational ovarian choriocarcinoma (NGCO)... (Review)
Review
INTRODUCTION
Trophoblastic neoplasms are often associated with pregnancy, and nongestational trophoblastic neoplasms are extremely rare. Nongestational ovarian choriocarcinoma (NGCO) is a highly aggressive germ cell-derived tumor frequently presenting with early hematogenous metastasis.
PATIENT CONCERNS
Herein, we report a case of a 28-year-old unmarried woman with regular menstruation who experienced vaginal bleeding 1 week after her last menstrual cycle. Doppler ultrasound revealed bilateral adnexal masses and elevated serum human chorionic gonadotropin (hCG) levels. The patient was initially misdiagnosed as presenting an ectopic pregnancy.
DIAGNOSIS
The final pathology confirmed an International Federation of Gynecology and Obstetrics stage IA NGCO with bilateral mature teratoma of the ovary. This is an extraordinary instance of ovarian choriocarcinoma which emerged without any prior gestation, and the patient's lack of a history of pregnancy made the diagnosis ignored.
INTERVENTIONS
After initial surgery and 1 cycle of bleomycin, etoposide, and cisplatin (BEP) chemotherapy, a laparoscopic fertility-preserving comprehensive staging surgery was performed. Two cycles of chemotherapy with BEP were administered as supplemental therapy postsurgery, and leuprorelin was administered to protect ovarian function.
OUTCOMES
Menstruation resumed 4 months after chemotherapy completion, and tumor indicators were within the normal range. No signs of recurrence were observed at the 36-month follow-up.
CONCLUSION
NGCO should be considered if a female patient exhibits irregular vaginal bleeding and masses in the adnexal area. The present case and our literature review also highlighted that fertility-sparing surgery and multidrug chemotherapy are effective methods for treating NGCO.
Topics: Humans; Female; Adult; Ovarian Neoplasms; Teratoma; Choriocarcinoma, Non-gestational; Antineoplastic Combined Chemotherapy Protocols; Etoposide; Pregnancy; Bleomycin
PubMed: 38701311
DOI: 10.1097/MD.0000000000036996 -
Anales de Pediatria Dec 2023
Topics: Female; Humans; X-Rays; Ovarian Neoplasms; Radiography; Teratoma
PubMed: 37932160
DOI: 10.1016/j.anpede.2023.08.017 -
Annals of Neurology Mar 2018N-methyl-D-aspartate receptor (NMDAR) antibody encephalitis is mediated by immunoglobulin G (IgG) autoantibodies directed against the NR1 subunit of the NMDAR. Around...
INTRODUCTION
N-methyl-D-aspartate receptor (NMDAR) antibody encephalitis is mediated by immunoglobulin G (IgG) autoantibodies directed against the NR1 subunit of the NMDAR. Around 20% of patients have an underlying ovarian teratoma, and the condition responds to early immunotherapies and ovarian teratoma removal. However, despite clear therapeutic relevance, mechanisms of NR1-IgG production and the contribution of germinal center B cells to NR1-IgG levels are unknown.
METHODS
Clinical data and longitudinal paired serum NR1-reactive IgM and IgG levels from 10 patients with NMDAR-antibody encephalitis were determined. Peripheral blood mononuclear cells from these 10 patients, and two available ovarian teratomas, were stimulated with combinations of immune factors and tested for secretion of total IgG and NR1-specific antibodies.
RESULTS
In addition to disease-defining NR1-IgG, serum NR1-IgM was found in 6 of 10 patients. NR1-IgM levels were typically highest around disease onset and detected for several months into the disease course. Moreover, circulating patient B cells were differentiated into CD19 CD27 CD38 antibody-secreting cells in vitro and, from 90% of patients, secreted NR1-IgM and NR1-IgG. Secreted levels of NR1-IgG correlated with serum NR1-IgG (p < 0.0001), and this was observed across the varying disease durations, suggestive of an ongoing process. Furthermore, ovarian teratoma tissue contained infiltrating lymphocytes which produced NR1-IgG in culture.
INTERPRETATION
Serum NR1-IgM and NR1-IgG, alongside the consistent production of NR1-IgG from circulating B cells and from ovarian teratomas suggest that ongoing germinal center reactions may account for the peripheral cell populations which secrete NR1-IgG. Cells participating in germinal center reactions might be a therapeutic target for the treatment of NMDAR-antibody encephalitis. Ann Neurol 2018;83:553-561.
Topics: Adolescent; Adult; Aged; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Autoantibodies; Female; Germinal Center; HEK293 Cells; Humans; Immunoglobulin G; Immunoglobulin M; Longitudinal Studies; Middle Aged; Ovarian Neoplasms; Prospective Studies; Receptors, N-Methyl-D-Aspartate; Teratoma; Young Adult
PubMed: 29406578
DOI: 10.1002/ana.25173