-
Neurology(R) Neuroimmunology &... Nov 2020Our aim was to identify patients with probable anti-NMDA receptor encephalitis among historical medical cases.
OBJECTIVE
Our aim was to identify patients with probable anti-NMDA receptor encephalitis among historical medical cases.
METHOD
A case report published in leading Hungarian-, German- and Italian-language medical journals in the early 1840s was revisited.
RESULTS
In 1830, an 18-year-old, healthy woman suffered epileptic seizures, followed by a 6-day-long state characterized by catalepsy, unresponsiveness, motionless, and light breathing. Her symptoms regularly returned in the following 1.5 years. Meanwhile, a progressively growing huge abdominal tumor appeared. One day, she suddenly started vomiting a large amount of foul-smelling pus mixed with blood, accompanied by bone fragments. Pus mixed blood with some membranous substance was also evacuated through the anus and vagina. After this event, she completely recovered; 1.5 years later, she married and later gave birth to 3 healthy children. The patient remained healthy during the 11-year follow-up.
CONCLUSIONS
We suggest that in the description of a paraneoplastic case, an anti-NMDA receptor encephalitis can be dated back as far as to the 19th century, with an especially rare type of resolution: the disappearance of the symptoms after the spontaneous elimination of an ovarian teratoma.
Topics: Adolescent; Adult; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Female; History, 19th Century; Humans; Ovarian Neoplasms; Teratoma; Young Adult
PubMed: 32978290
DOI: 10.1212/NXI.0000000000000887 -
Journal of Gynecology Obstetrics and... Jun 2021Ovarian masses in pediatric population are the most common genital neoplasms, and these masses are often benign. The purpose of this study is to evaluate the pediatric...
PURPOSE
Ovarian masses in pediatric population are the most common genital neoplasms, and these masses are often benign. The purpose of this study is to evaluate the pediatric ovarian masses operated in our hospital.
METHOD
The records of patients, under the age of 18 who were operated in our hospital due to ovarian mass between 2012 and 2018 were reviewed retrospectively. Clinical findings, operational procedures, histopathologies, tumor markers and radiological images were evaluated.
FINDINGS
During the study, 146 patients (5 patients were bilateral) were evaluated. The average age of the study patients was 14.01 ± 4.02 years. 107 of the study patients were benign, 37 were malignant and 2 were borderline. The most common symptom in benign masses was tenderness in lower abdominal (75.7 %). 124 of the patients (86.1 %) were in post-menarche period. 34 of the patients had ovarian torsion. Open surgery was conducted on 79.5 % (116/146) of the patients, and laparoscopic surgery was conducted on 20.5 % (30/146). The rate of oophorectomy was 24.6 % (36/146) throughout the operations. The most frequently conducted surgical procedure was cyst excision in benign masses and oophorectomy in malignant masses. In neoplastic masses, the ratio of pelvic mass palpation; and in non-neoplastic masses, lower abdominal tenderness was more apparent. The rate of ovarian torsion was 23.6 % (25/107) in benign masses and 24.3 % (9/37) in malignant masses.
OUTCOMES
Pediatric and adolescent ovarian masses are mostly benign and majority of these occur at post-menarche period. The most common symptom was pelvic tenderness in benign masses, and palpable pelvic masses in malignant masses. For future fertility and low incidence of malignancy in these patients, ovarian preserving surgery should be considered for the first operation.
Topics: Abdominal Pain; Adolescent; Child; Cystadenocarcinoma; Dysgerminoma; Female; Humans; Laparoscopy; Ovarian Cysts; Ovarian Neoplasms; Ovarian Torsion; Ovariectomy; Retrospective Studies; Teratoma
PubMed: 32889112
DOI: 10.1016/j.jogoh.2020.101901 -
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 -
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 -
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 -
Journal of Minimally Invasive Gynecology Jan 2020Laparoscopic cystectomy for ovarian teratomas has the advantages of a minimally invasive approach [1]. The standardization and description of the technique are the...
STUDY OBJECTIVE
Laparoscopic cystectomy for ovarian teratomas has the advantages of a minimally invasive approach [1]. The standardization and description of the technique are the main objectives of this video (Video 1). We described the surgery in 10 steps [2], which could help make this procedure easier and safer.
DESIGN
A step-by-step video demonstration of the technique.
SETTING
A French university tertiary care hospital.
PATIENTS
Patients with ovarian teratomas with indication for laparoscopic cystectomy [3]. The local institutional review board ruled that approval was not required for this video article because the video describes a technique and does not report a clinical case.
INTERVENTIONS
Standardized laparoscopic cystectomies were recorded to realize the video.
MEASUREMENTS AND MAIN RESULTS
This video presents a systematic approach to cystectomy for teratoma clearly divided into 10 steps: (1) planning of the surgery, (2) ergonomy and materials, (3) exploration and cytology, (4) prevention of peritoneal spillage [4], (5) mobilization of the ovary, (6) incision of the ovary, (7) dissection, (8) hemostasis, (9) exteriorization of the cyst, and (10) washing and exploration.
CONCLUSION
Standardization of laparoscopic cystectomy for ovarian teratoma could make this procedure easier and safer to perform. The 10 steps presented help to perform each part of the surgery in a logical sequence, making the procedure ergonomic and easier to adopt and learn. Moreover, the standardization of the surgical techniques could reduce the learning curve.
Topics: Adult; Cytoreduction Surgical Procedures; Dissection; Female; Humans; Laparoscopy; Ovarian Neoplasms; Ovariectomy; Teratoma
PubMed: 31125721
DOI: 10.1016/j.jmig.2019.05.009 -
International Journal of Gynecological... Nov 2022Sebaceous carcinoma is an uncommon primary cutaneous neoplasm which may be associated with mismatch repair (MMR) abnormalities and sometimes with Muir-Torré syndrome.... (Review)
Review
Sebaceous carcinoma is an uncommon primary cutaneous neoplasm which may be associated with mismatch repair (MMR) abnormalities and sometimes with Muir-Torré syndrome. These neoplasms rarely arise in the ovary within a teratoma/ dermoid cyst. We report a sebaceous carcinoma arising in an ovarian teratoma in a 49-yr old (the 14th case reported in the literature) which exhibited loss of expression of MMR proteins MSH2 and MSH6. A germline mutation c.1102C>T was present in exon 7 of the MSH2 gene, the first report of a germline mutation associated with this tumor type. In reporting this case, we review prior reports of primary ovarian sebaceous carcinoma. We recommend that all sebaceous carcinomas of the ovary undergo immunohistochemistry for MMR proteins for investigation of possible Lynch syndrome.
Topics: Female; Humans; MutS Homolog 2 Protein; DNA Mismatch Repair; DNA-Binding Proteins; Muir-Torre Syndrome; Teratoma; Germ-Line Mutation; Carcinoma
PubMed: 35077082
DOI: 10.1097/PGP.0000000000000847 -
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 -
Cytopathology : Official Journal of the... Jul 2022Germ cell tumours infrequently metastasise to body cavities, where early detection on fluid samples is possible and can spearhead early treatment and survival.
BACKGROUND
Germ cell tumours infrequently metastasise to body cavities, where early detection on fluid samples is possible and can spearhead early treatment and survival.
MATERIALS AND METHODS
A total of seven cases of metastatic germ cell tumours were retrieved out of 7500 effusion samples received for cytopathological examination from 2015 to 2021. Detailed cytological features of metastatic germ cell tumours in effusion samples were studied, along with a correlation between clinical, radiological, and histopathological features.
RESULTS
A total of seven cases of metastatic germ cell tumours were analysed in effusion samples which included dysgerminoma (2), immature teratoma (2), yolk sac tumour (1), embryonal carcinoma (1), and mixed germ cell tumour (1). The smears showed predominantly discrete or loose clusters of cells. The cells with round nuclei and prominent nucleoli were helpful in detecting dysgerminoma and yolk sac tumours. Immature teratoma showed tiny groups of small cells and mature squamous cells. Serum tumour markers were raised in the majority of cases.
CONCLUSION
Metastatic germ cell tumours in effusion are uncommon, but detailed clinical history, including serum markers and characteristic cytological features, are helpful in their diagnosis.
Topics: Dysgerminoma; Female; Humans; Male; Neoplasms, Germ Cell and Embryonal; Neoplasms, Second Primary; Ovarian Neoplasms; Teratoma; Testicular Neoplasms
PubMed: 35347771
DOI: 10.1111/cyt.13122 -
Cureus Dec 2023Gliomatosis peritonei (GP) is a rare condition characterised by mature glial nodules that implant in the peritoneum, lymph nodes, or omentum. GP is typically associated...
Gliomatosis peritonei (GP) is a rare condition characterised by mature glial nodules that implant in the peritoneum, lymph nodes, or omentum. GP is typically associated with mature or immature ovarian teratomas and usually affects adolescent females. Although neuroglia may be a standard feature of mature ovarian teratomas, widespread peritoneal glial nodules, ascites, and pleural effusion are rare, particularly in the paediatric population. We report a case of a giant left mature ovarian teratoma associated with GP and omental splenunculus in a 12-year-old female who presented with constipation, an adnexal mass, ascites, pleural effusion, and elevated CA-125 levels. The patient successfully underwent fertility-sparing surgery in the form of a left salpingo-oophorectomy, omentectomy, and resection of peritoneal glial deposits. In light of the current scarcity of data on this clinical entity in the literature, we hope to raise awareness of this rare presentation of mature ovarian teratoma, the challenges associated with preoperative diagnosis, and the impact of fertility-sparing surgery on potential oncological and reproductive outcomes in a paediatric patient.
PubMed: 38179400
DOI: 10.7759/cureus.49945