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Annals of Medicine 2023The prognostic factors for patients with pure ovarian immature teratoma (POIT) and the role of adjuvant chemotherapy in stage IA G2-G3 and IB-IC POIT remains...
BACKGROUND
The prognostic factors for patients with pure ovarian immature teratoma (POIT) and the role of adjuvant chemotherapy in stage IA G2-G3 and IB-IC POIT remains controversial.
METHODS
We conducted a retrospective study of 155 POIT patients treated in our hospital between 2000 and 2022. The recurrence-free survival (RFS), disease-specific survival (DSS), and potential prognostic factors of POIT patients were evaluated. Subgroup analysis was conducted in stage I other than stage IA G1 POIT.
RESULTS
The median age at diagnosis was 23.0 years (range: 4.0 - 39.0), and 126 (81.3%), 2 (1.3%), 26 (16.8%), and 1 (0.6%) patients had FIGO stage I, stage II, stage III, and stage IV disease, respectively. Twenty-three patients relapsed and five died of the diseases after a median follow-up of 7.6 years, with a 5-year RFS and DSS rate of 86.0% and 97.0%, respectively. Multivariate analysis showed that positive postoperative tumour markers (TM) were the risk factor for recurrence in the overall cohort (hazard ratio [HR] 4.058, 95% CI 1.175 - 14.019, = 0.027) and subgroup (HR 10.237, 95% CI 2.175 - 48.179, = 0.003), and FIGO stage II-IV was the only factor for DSS in overall cohort (HR 7.751, 95% CI 1.281 - 46.895, = 0.026). In 110 patients subjected to subgroup analysis, 29 patients received surveillance without chemotherapy and 81 patients were administered adjuvant chemotherapy. Multivariate analysis revealed active surveillance significantly increased the recurrence rate (5-year RFS of 75.7% vs. 93.6%, HR 7.562, 95% CI 2.441 - 23.424, < 0.001) but not the death related to POIT ( = 0.338).
CONCLUSION
Positive postoperative TM and FIGO stage II-IV were the prognostic factors for POIT. Active surveillance in stage I POIT of any grade may be practical for those with negative postoperative TM.
Topics: Female; Humans; Child, Preschool; Child; Adolescent; Young Adult; Adult; Prognosis; Retrospective Studies; Neoplasm Staging; Ovarian Neoplasms; Chemotherapy, Adjuvant; Teratoma
PubMed: 37909181
DOI: 10.1080/07853890.2023.2273984 -
International Journal of Environmental... Jun 2023Nonepithelial ovarian cancers (NEOC) are a group of rare malignancies, including germ cell tumours (GCT) and sex cord-stromal tumours (SCST), along with small-cell... (Review)
Review
Nonepithelial ovarian cancers (NEOC) are a group of rare malignancies, including germ cell tumours (GCT) and sex cord-stromal tumours (SCST), along with small-cell carcinomas and sarcomas. GCTs represent 2-5% of ovarian cancers, with a yearly incidence of 4:100,000, and they usually affect young women and adolescents. Precursory germ cells of the ovary form the basis of GCT. They are histologically classified into primitive GCT, teratomas, and monodermal and somatic-type tumours associated with dermoid cysts. A primitive GCT can be either a yolk sac tumour (YST), dysgerminoma, or mixed germ cell neoplasm. Teratomas are either mature (benign) or immature (malignant). Given that malignant GCTs occur rarely compared to epithelial ovarian tumours (EOC), greater focus is required in their diagnosis and treatment. In this article, we review the epidemiology, clinical manifestations, diagnosis, and molecular biology, along with the management and therapeutic challenges.
Topics: Adolescent; Humans; Female; Neoplasms, Germ Cell and Embryonal; Ovarian Neoplasms; Teratoma
PubMed: 37372675
DOI: 10.3390/ijerph20126089 -
Children (Basel, Switzerland) Oct 2022: Ovarian teratomas are the most common type of ovarian mass during childhood and adolescence. There is no clear guidance for the management of the ovarian teratomas in... (Review)
Review
: Ovarian teratomas are the most common type of ovarian mass during childhood and adolescence. There is no clear guidance for the management of the ovarian teratomas in children and adolescents. It is necessary, however, to understand the feature of these tumours and the indications for operating on them, and to put subjective criteria for the management in elective or emergency presentations. : Medical records of patients between the age of 12 and 20 years old that underwent surgery for ovarian teratomas at the Department of Obstetrics and Gynaecology in University Hospital of Ioannina, between January 2000 and August 2022, were reviewed. A medical database was searched between January 2000 and August 2022 with the following keywords: ovarian teratoma, paediatrics and adolescents. : We present the results of forty patients, with a mean age of 11.8 years of age. All patients had surgery, where three out of four underwent oophorectomy and only one out of four underwent ovary-sparing operation; similar results were found in the literature. : Ovary-sparing treatment is more common recently compared with the past, such as MIS compared with laparotomy. Better-organised and -planned prospective multi-centre studies are necessary to gain a deeper knowledge of the physiology and prognostic factors of teratomas in children.
PubMed: 36291507
DOI: 10.3390/children9101571 -
BMJ Neurology Open 2023Opsoclonus-myoclonus-ataxia syndrome (OMAS) is characterised by the combination of opsoclonus and arrhythmic action myoclonus with axial ataxia and dysarthria. In...
BACKGROUND
Opsoclonus-myoclonus-ataxia syndrome (OMAS) is characterised by the combination of opsoclonus and arrhythmic action myoclonus with axial ataxia and dysarthria. In adults, a majority are paraneoplastic secondary to solid organ tumours and could harbour antibodies against intracellular epitopes; however, certain proportions have detectable antibodies to various neuronal cell surface antigens. Anti-N-methyl-D-aspartate (NMDAR) antibodies and ovarian teratomas have been implicated in OMAS.
METHODS
Report of two cases and review of literature.
RESULTS
Two middle-aged women presented with subacute-onset, rapidly progressive OMAS and behavioural changes consistent with psychosis. The first patient had detectable antibodies to NMDAR in the cerebrospinal fluid (CSF) alone. Evaluation for ovarian teratoma was negative. The second patient had no detectable antibodies in serum or CSF; however, she had an underlying ovarian teratoma. Patient A was treated with pulse steroids, therapeutic plasma exchange (TPE) followed by bortezomib (BOR) and dexamethasone, while patient B was treated with steroids, TPE followed by surgical resection of ovarian teratoma. Both patients had favourable outcomes and were asymptomatic at the 6 monthly follow-up.
CONCLUSIONS
With coexistent neuropsychiatric manifestations, OMAS can be considered a distinct entity of autoimmune encephalitis, pathogenesis being immune activation against known/unknown neuronal cell surface antigens. The observation of absence of anti-NMDAR antibody in patients with teratoma-associated OMAS and vice versa is intriguing. Further research on the potential role of ovarian teratoma in evoking neuronal autoimmunity and its targets is required. The management challenge in both cases including the potential use of BOR has been highlighted.
PubMed: 37396795
DOI: 10.1136/bmjno-2023-000414 -
Obstetrics and Gynecology International 2016Although ovarian mature cystic teratomas are the commonest adnexal masses occurring in premenopausal women, there are many challenges faced by gynecologists on deciding... (Review)
Review
Although ovarian mature cystic teratomas are the commonest adnexal masses occurring in premenopausal women, there are many challenges faced by gynecologists on deciding upon the best surgical management. There is uncertainty, lack of consensus, and variation in surgical practices. This paper critically analyzes various surgical approaches and techniques used to treat these cysts in an attempt to outline a unified guidance. MEDLINE and EMBASE databases were searched in January 2015 with no date limit using the key words "ovarian teratoma" and "ovarian dermoid." The search was limited to articles in English language, humans, and female. The two authors conducted the search independently. The laparoscopic approach is generally considered to be the gold standard for the management. Oophorectomy should be the standard operation except in younger women with a single small cyst. The risk of chemical peritonitis after contents spillage is extremely rare and can certainly be overcome with thorough peritoneal lavage using warmed fluid. There is a place for surveillance in some selected cases.
PubMed: 27110246
DOI: 10.1155/2016/2390178 -
BMC Women's Health Sep 2023Struma ovarii (SO) is a rare tumor and may transform into ovarian strumal carcinoid (OSC) and/or malignant struma ovarii (MSO), but the incidence, clinical...
BACKGROUND
Struma ovarii (SO) is a rare tumor and may transform into ovarian strumal carcinoid (OSC) and/or malignant struma ovarii (MSO), but the incidence, clinical characteristics, and survival outcomes have not been well defined.
METHODS
We conducted a retrospective study of patients with ovarian strumal diseases treated in the our hospital between 1980 and 2022. Subgroup analyses of SO, OSC, and MSO were subsequently performed.
RESULTS
A total of 275 cases (2.14%) were identified in a cohort of 12,864 patients with ovarian teratomas, where SO, OSC, and MSO accounted for 83.3%, 12.0%, and 4.7% of cases, respectively. There were no significant differences in age, tumor sizes, elevated tumor markers, and ascites among the three subgroups. At initial treatment, all patients with SO or OSC had FIGO stage I disease except one SO patient presenting metastatic disease, ten patients had MSO confined to the ovary, whereas other three patients had metastatic diseases. Two patients with SO respectively relapsed at peritoneum and anterior mesorectum, while none of the OSC patients presented tumor recurrence or death despite different surgical procedures employed. The 5-year recurrence-free survival rate was 88.9%, and only one death occurred at 9.5 years after diagnosis in patients with MSO. Radioiodine therapy showed satisfactory therapeutic efficacy, but these patients showed poor responses to the chemotherapy.
CONCLUSION
2.14% of ovarian teratoma could be classified as SO, of which 12.0% and 4.7% of SO may transform into OSC and MSO, repsectively. The survival outcomes were excellent even after SO transformed into OSC or MSO.
SYNOPSIS
SO occupied 2.14% of ovarian teratoma, where 12.0% and 4.7% of SO may transform into OSC and MSO, respectively, and had excellent survival outcomes.
Topics: Female; Humans; Ovarian Neoplasms; Struma Ovarii; Retrospective Studies; Carcinoid Tumor; Incidence; Iodine Radioisotopes; Neoplasm Recurrence, Local; Teratoma
PubMed: 37726744
DOI: 10.1186/s12905-023-02624-5 -
Annals of Clinical and Translational... Jan 2024Ovarian teratoma is a common occurrence in patients with anti-NMDA receptor encephalitis (NMDARe), and its removal is crucial for a favorable prognosis. However, the...
INTRODUCTION
Ovarian teratoma is a common occurrence in patients with anti-NMDA receptor encephalitis (NMDARe), and its removal is crucial for a favorable prognosis. However, the initial pathogenesis of autoimmunity in the encephalitic teratoma remains unclear. In this study, we aimed to investigate the genomic landscape and microscopic findings by comparing NMDARe-associated teratomas and non-encephalitic control teratomas.
MATERIALS AND METHODS
A prospective consecutive cohort of 84 patients with NMDARe was recruited from January 2014 to April 2020, and among them, patients who received teratoma removal surgery at Seoul National University Hospital were enrolled. We conducted a comparison of whole-exome sequencing data and pathologic findings between NMDARe-associated teratomas and control teratomas.
RESULTS
We found 18 NMDARe-associated teratomas from 15 patients and compared them with 17 non-encephalitic control teratomas. Interestingly, the genomic analysis revealed no significant differences in mutations between encephalitic and non-encephalitic teratomas. Pathologic analysis showed no discrepancies in terms of the presence of neuronal tissue and lymphocytic infiltration between the encephalitic teratomas (n = 14) and non-encephalitic teratomas (n = 18). However, rituximab-naïve encephalitic teratomas exhibited a higher frequency of germinal center formation compared to non-encephalitic teratomas (80% vs. 16.7%, P = 0.017). Additionally, rituximab-treated encephalitic teratomas demonstrated a reduced number of CD20 cells and germinal centers in comparison to rituximab-naïve encephalitic teratomas (P = 0.048 and 0.023, respectively).
DISCUSSION
These results suggest that the initiation of immunopathogenesis in NMDARe-associated teratoma is not primarily attributed to intrinsic tumor mutations, but rather to immune factors present in the encephalitic patient group, ultimately leading to germinal center formation within the teratoma.
Topics: Female; Humans; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Rituximab; Prospective Studies; Ovarian Neoplasms; Teratoma; Receptors, N-Methyl-D-Aspartate; Genomics
PubMed: 37986706
DOI: 10.1002/acn3.51948 -
Journal of Indian Association of... Oct 2014PEDIATRIC GERM CELL TUMORS (GCT) ARE RARE TUMORS: 80% are benign, 20% malignant (2-3% of all malignant pediatric tumors). The gonadal sites (ovary and testis) account... (Review)
Review
UNLABELLED
PEDIATRIC GERM CELL TUMORS (GCT) ARE RARE TUMORS: 80% are benign, 20% malignant (2-3% of all malignant pediatric tumors). The gonadal sites (ovary and testis) account for 40% of cases.
OVARIAN GCTS
Represent 30% of GCTs and 70% of neoplastic ovarian masses, being the most common ovarian neoplasms in children and teenagers. Benign and immature forms (teratomas) constitute about 80% of all ovarian GCTs, malignant forms represent 20% increasing during adolescence. The most common malignant entity in children is the yolk sac tumors (YST); dysgerminoma is frequent during adolescence and being bilateral in 10% of cases. Presentation is similar in malignant and benign lesions; abdominal pain (70-80%) and lower abdominal mass are common symptoms. Evaluation of alpha-fetoprotein (αFP) or beta subunit of human chorionic gonadotropin (βHCG) is essential to address the nature of the tumors: Their elevation means presence of malignancy. Surgery includes intraoperative staging procedures and requires ovariectomy or ovarosalpingectomy for malignant lesions, but may be conservative in selected benign tumors. Since malignant GCTs are very chemosensitive, primary chemotherapy is recommended in metastatic or locally advanced tumors.
TESTICULAR GCT
Represent 10% of pediatric GCT, and about 30% of malignant GCT with two age peaks: Children <3 years may experience mature teratoma and malignant GCTs, represented almost exclusively by YST, while adolescents may also show seminomas or other mixed tumors. The main clinical feature is a painless scrotal mass. Surgery represents the cornerstone of the management of testicular GCTs, with an inguinal approach and a primary high orchidectomy for malignant tumors, while a testis-sparing surgery can be considered for benign lesions. A retroperitoneal lymph node (LN) biopsy may be necessary to define the staging when the involvement of retroperitoneal LN is uncertain at imaging investigations.
CONCLUSION
Patients with gonadal malignant GCTs fare better than those with extragonadal mediastinal germ cell tumors (MGCTs) and survival rate exceeds 90% in localized forms. Chemotherapy has significantly improved the outcome of malignant forms since the introduction of platinum based regimens. The surgical procedure has to be performed in agreement with the ongoing protocols.
PubMed: 25336799
DOI: 10.4103/0971-9261.141995 -
Journal of the Chinese Medical... Jun 2018
Topics: Adolescent; Biomarkers, Tumor; Female; Humans; Ovarian Neoplasms; Teratoma
PubMed: 28882729
DOI: 10.1016/j.jcma.2017.08.002 -
Journal of Indian Association of... 2019Ovarian cystic mature teratomas (OCMTs) are the most frequent ovarian tumors in childhood. This review aimed to determine the feasibility and safety of laparoscopic... (Review)
Review
Ovarian cystic mature teratomas (OCMTs) are the most frequent ovarian tumors in childhood. This review aimed to determine the feasibility and safety of laparoscopic management of OCMT. Literature was searched for terms "mature," "ovarian," "teratomas," and "laparoscopy." Primary endpoints were age at surgery, laparoscopic and surgical technique, intraoperative complications, postoperative morbidity, and associated pathology. Literature search revealed 11 articles published between 1998 and 2014 that met the inclusion criteria. There were 105 ( = 95 unilateral; = 10 bilateral) patients for this analysis, with mean age at surgery being 13 years. Four laparoscopic approaches were opted: gasless transumbilical laparoendoscopic single-site (LESS) surgery ( = 19), gasless multiport surgery ( = 24), single-incision laparoscopic surgery (SILS) ( = 3), and pneumoperitoneum multiport laparoscopy ( = 59). The 10 patients with bilateral OCMT underwent ovary-sparing surgery: LESS-assisted extracorporeal bilateral cystectomy in which tumors were punctured by a balloon catheter ( = 2), intracorporeal cystectomy for gasless multiport laparoscopy ( = 5) with use of endobags to prevent spillage, and transperitoneal multiport laparoscopy ( = 3). OCMT was associated with ipsilateral and unilateral ovarian torsion in five and bilateral ovarian torsion in one patient with bilateral OCMT. In four patients with unilateral OCMT, salpingo-oophorectomy was performed. Intraoperative complications were laceration of utero-ovarian ligament and bladder injury during a suprapubic port placement. The mean follow-up was 31.9 months. Patients with unilateral or bilateral OCMT can be offered ovarian-sparing surgery laparoscopically with one of the following techniques: LESS, SILS or multiport laparoscopy with pneumoperitoneal or gasless. Long-term follow-up of these techniques has shown no recurrence with low postoperative morbidity and low intraoperative complications.
PubMed: 31105392
DOI: 10.4103/jiaps.JIAPS_246_17