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Disease Markers 2021To study the clinical characteristics and surgical outcomes of anti-NMDAR encephalitis and the immunopathology of associated 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.
Topics: Adolescent; Adult; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Autoantibodies; Biomarkers; China; Female; Follow-Up Studies; Humans; Immunohistochemistry; Ovarian Neoplasms; Ovariectomy; Ovary; Receptors, N-Methyl-D-Aspartate; Retrospective Studies; Teratoma; Treatment Outcome; Young Adult
PubMed: 34093900
DOI: 10.1155/2021/9990382 -
Acta Medica (Hradec Kralove) 2022Mature cystic teratoma is the most common type of ovarian tumor in children. Adnexal torsion is the main complication of mature ovarian teratoma. The synchronous...
Mature cystic teratoma is the most common type of ovarian tumor in children. Adnexal torsion is the main complication of mature ovarian teratoma. The synchronous bilateral incidence of mature cystic teratoma in premenarchal girls is known to be rare. However, the incidence of adnexal torsion is higher in young girls. A 10-year-old girl presenting with acute abdomen was managed by emergency laparotomy. Bilateral mature ovarian teratoma with adnexal torsion of the right ovary was found. The right ovarian tissue was not viable due to torsion and an oophorectomy was necessary. Cystectomy with preservation of the ovarian tissue of the left ovary was performed. Histopathological diagnosis was bilateral synchronous mature teratoma with necrosis of the right adnexa. Although the risk of malignancy of torsed ovaries and mature teratomas in premenarchal girls is low, their removal is recommended to prevent adnexal torsion. Decision between ovarian tissue sparing surgery or oophorectomy depends on the risk of malignancy, fertility preservation and the avoidance of early menopause.
Topics: Child; Female; Humans; Ovarian Neoplasms; Ovarian Torsion; Teratoma
PubMed: 35793507
DOI: 10.14712/18059694.2022.14 -
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 -
Diagnostic and Interventional Imaging 2021
Topics: Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Brain; Female; Humans; Ovarian Neoplasms; Teratoma
PubMed: 34147389
DOI: 10.1016/j.diii.2021.04.006 -
Oncology 2023The aim of the study was to evaluate the perioperative risks and outcomes of ultra-radical surgery in patients with extensive metastatic ovarian growing teratoma...
INTRODUCTION
The aim of the study was to evaluate the perioperative risks and outcomes of ultra-radical surgery in patients with extensive metastatic ovarian growing teratoma syndrome (GTS).
METHODS
We conducted a retrospective study of patients with extensive metastatic ovarian GTS treated in our hospital between 2000 and 2022. Patients' clinical characteristics, surgical treatment, and outcomes were evaluated.
RESULTS
Overall, 13 patients were identified, and the median age at diagnosis of ovarian immature teratoma (IT) was 24 years (range: 5-37). The median interval between IT diagnosis and presenting GTS was 8 months (range: 2-60), with a median surgery delay of 5 months (range: 3-300). Peritoneum and liver were the most commonly affected sites (100%), followed by bowel (12 patients, 92.3%), diaphragm (12 patients, 92.3%), adnexa (9 patients, 69.2%), omentum (8 patients, 61.5%), uterus (7 patients, 53.8%), in the descending order. The mean operation time was 316 min (range: 180-625), and the mean blood loss volume was 992 mL (range: 200-5,000). Peritoneal metastasectomy (13 patients, 100%), diaphragmatic metastasectomy (12 patients, 92.3%), metastasis removal from the bowel (8 patients, 61.5%), partial hepatectomy (4 patients, 30.8%), bowel excision and anastomosis (1 patient, 7.7%) were also applied to achieve optimal debulking. R0 was achieved in 9 (69.2%) patients. A high rate of intraoperative blood transfusion (8 patients, 61.5%) and admission to the intensive care unit (9 patients, 69.2%) were observed, and the median postoperative hospitalization time was 8 days (range: 4-22). After a median follow-up of 3.3 years, 9 patients were free of disease, and 4 were alive with stable residual diseases.
CONCLUSION
The survival outcomes in extensive metastatic ovarian GTS were satisfactory after ultra-radical surgery, while a proper therapeutic plan should be established due to the high perioperative risks.
Topics: Female; Humans; Child, Preschool; Child; Adolescent; Young Adult; Adult; Retrospective Studies; Teratoma; Ovarian Neoplasms; Prognosis; Cytoreduction Surgical Procedures
PubMed: 38096801
DOI: 10.1159/000533411 -
Frontiers in Immunology 2023Immature ovarian teratomas are a type of malignant germ cell tumor composed of complicated cell types and are characterized by pathological features of immature...
INTRODUCTION
Immature ovarian teratomas are a type of malignant germ cell tumor composed of complicated cell types and are characterized by pathological features of immature neuroectodermal tubules/rosettes. However, there is a lack of understanding of patient-derived immature ovarian teratomas (PDT) at the single cell level. Moreover, whether stem cell lines derived from immature teratomas (CDT) can be used as models for research on PDT remains to be elucidated.
METHODS
Single-cell RNA sequencing (scRNA-seq) and subsequent bioinformatic analysis was performed on three patient-derived immature ovarian teratomas (PDT) samples to reveal the heterogeneity, evolution trajectory, and cell communication within the tumor microenvironment of PDT. Validations were conducted in additional seven samples through multiplex immunofluorescence.
RESULT
A total of qualified 22,153 cells were obtained and divided into 28 clusters, which can match to the scRNA-seq annotation of CDT as well as human fetal Cell Atlas, but with higher heterogeneity and more prolific cell-cell crosstalk. Radial glia cells (tagged by SOX2) and immature neuron (tagged by DCX) exhibited mutually exclusive expression and differentiated along distinct evolutionary trajectory from cycling neural progenitors. Proportions of these neuroectodermal cell subtypes may play important roles in PDT through contributing to the internal heterogeneity of PDTs. Moreover, the immune cells in PDTs were infiltrated rather than teratoma-derived, with more abundant macrophage in immature neuron than those in radial glia cells, and the infiltrated macrophage subtypes (i.e., M1 and M2) were significantly correlated to clinical grade. Overall, suppressed evolution process and transcriptome regulation in neuroectodermal cells, reduced cell-cell crosstalk, higher M1/M2 proportion ratio, and enhanced T cell effects in tumor microenvironment are enriched in patients with favorable prognosis.
DISCUSSION
This study provides a comprehensive profile of PDT at the single cell level, shedding light on the heterogeneity and evolution of neuroectodermal cells within PDTs and the role of immune cells within the tumor microenvironment. Also, our findings highlight the potential usage of CDTs as a model for research on PDT.
Topics: Female; Humans; Transcriptome; Teratoma; Ovarian Neoplasms; Neoplasms, Germ Cell and Embryonal; Tumor Microenvironment
PubMed: 36936909
DOI: 10.3389/fimmu.2023.1131814 -
International Journal of Paleopathology Sep 2017This paper discusses the differential diagnosis of an unusual calcified mass found in the pelvic cavity of 45+-year-old female excavated from 15th-18th century Lisbon...
This paper discusses the differential diagnosis of an unusual calcified mass found in the pelvic cavity of 45+-year-old female excavated from 15th-18th century Lisbon (Portugal). The mass is relatively large, irregularly shaped, and exhibits a concave base with malformed teeth embedded within its inner surface. Considering its macroscopic and radiological characteristics, several conditions were considered in the differential diagnosis, namely eccyesis, fetus in fetu, lithopaedion, and ovarian teratoma. However, the morphological features of the specimen, such as its structure, morphology, and dimensions, are diagnostic of a teratoma. Its location and the sex of the individual are more specifically compatible with a calcified ovarian teratoma. With regional and temporal variations in the frequency of tumours, the report of new cases becomes imperative, especially from geographic regions where few cases have been identified. In fact, this appears to be the first case of ovarian teratoma detected in the Portuguese archaeological record and adds to the few palaeopathological cases described in the osteoarchaeological literature worldwide.
Topics: Female; Humans; Middle Aged; Diagnosis, Differential; History, 15th Century; History, 16th Century; History, 17th Century; History, 18th Century; Ovarian Neoplasms; Paleontology; Portugal; Teratoma
PubMed: 28888390
DOI: 10.1016/j.ijpp.2017.05.001 -
Pediatric Blood & Cancer Apr 2020To describe characteristics and outcome of pediatric ovarian immature teratomas (IT) to better define the place of chemotherapy. (Clinical Trial)
Clinical Trial
OBJECTIVE
To describe characteristics and outcome of pediatric ovarian immature teratomas (IT) to better define the place of chemotherapy.
METHODS
Children with ovarian IT enrolled in TGM95 and TGM2013 studies were analyzed. Norris grading and International Federation of Gynecology and Obstetrics staging system were used.
RESULTS
Thirty-six cases were identified with a median age of 11 years (range = 1-18): 35 of 36 stage I (17 stage IA, 13 stage IC, and 5 stage IX), including seven patients with gliomatosis peritonei (GP), and 1 stage IIIB (IT peritoneal implants). Centrally reviewed Norris grading was performed in 31 cases: 14 grade I and 17 grade II/III tumors. All patients underwent upfront surgery: 19 unilateral oophorectomy, 14 unilateral adnexectomy, 2 unilateral cystectomy, and 1 bilateral cystectomy. No extensive GP surgery was performed. Six patients received adjuvant vinblastin, bleomycin, and cisplatinum because of tumor rupture (n = 5, including two patients with GP) or stage III (n = 1). After a median follow-up of 39.5 months (range = 6-238), two events occurred 10 and 11 months after diagnosis: one bilateralization (initial stage IX, grade I) and one IT peritoneal relapse (initial stage IA, grade II), respectively. Both were successfully rescued by platinum-based chemotherapy and delayed surgery. No stage IC patients treated without adjuvant chemotherapy relapsed (four grade I and three grade III). None of the seven patients with GP progressed. Five-year event-free survival and overall survival were 94% (95% CI = 81-98%) and 100%.
CONCLUSIONS
The current series confirms the excellent prognosis of pediatric ovarian IT, arguing for conservative surgical approach in GP and against systematic adjuvant chemotherapy, even in ruptured tumors.
Topics: Adolescent; Antineoplastic Combined Chemotherapy Protocols; Bleomycin; Child; Child, Preschool; Cisplatin; Disease-Free Survival; Female; Follow-Up Studies; France; Humans; Infant; Neoplasm Grading; Ovarian Neoplasms; Ovariectomy; Survival Rate; Teratoma; Vinblastine
PubMed: 31981415
DOI: 10.1002/pbc.28186 -
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 -
Open Veterinary Journal Sep 2023Teratomas are rare types of germ cell neoplasms composed of various differentiated or undifferentiated tissues. (Review)
Review
BACKGROUND
Teratomas are rare types of germ cell neoplasms composed of various differentiated or undifferentiated tissues.
CASE DESCRIPTION
A 25-week-old female control FVB /n mouse in a 4-week toxicity study presented abdominal distension and poor body condition. It was euthanized, and the necropsy examination revealed a large mass connected to the tip of the right uterine horn, occupying the entire abdominal cavity. Microscopically, this mass showed areas of epidermal differentiation, with laminated keratin and sebaceous glands, differentiation into respiratory and digestive epithelium, cartilage, bone, and extensive areas of differentiation into the nervous tissue, being classified as an ovarian teratoma.
CONCLUSION
As far as authors know, the occurrence of ovarian teratomas in the FVB/n mouse strain has never been previously described.
Topics: Mice; Female; Animals; Ovarian Neoplasms; Teratoma; Rodent Diseases
PubMed: 37842105
DOI: 10.5455/OVJ.2023.v13.i9.19