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Biomedicines May 2023Anti-NMDAR encephalitis has been associated with multiple antigenic triggers (i.e., ovarian teratomas, prodromal viral infections) but whether geographic, climatic, and... (Review)
Review
Anti-NMDAR encephalitis has been associated with multiple antigenic triggers (i.e., ovarian teratomas, prodromal viral infections) but whether geographic, climatic, and environmental factors might influence disease risk has not been explored yet. We performed a systematic review and a meta-analysis of all published papers reporting the incidence of anti-NMDAR encephalitis in a definite country or region. We performed several multivariate spatial autocorrelation analyses to analyze the spatial variations in the incidence of anti-NMDA encephalitis depending on its geographical localization and temperature. Finally, we performed seasonal analyses in two original datasets from France and Greece and assessed the impact of temperature using an exposure-lag-response model in the French dataset. The reported incidence of anti-NMDAR encephalitis varied considerably among studies and countries, being higher in Oceania and South America (0.2 and 0.16 per 100,000 persons-year, respectively) compared to Europe and North America (0.06 per 100,000 persons-year) ( < 0.01). Different regression models confirmed a strong negative correlation with latitude (Pearson's R = -0.88, < 0.00001), with higher incidence in southern hemisphere countries far from the equator. Seasonal analyses showed a peak of cases during warm months. Exposure-lag-response models confirmed a positive correlation between extreme hot temperatures and the incidence of anti-NMDAR encephalitis in France ( = 0.03). Temperature analyses showed a significant association with higher mean temperatures and positive correlation with higher ultraviolet exposure worldwide. This study provides the first evidence that geographic and climatic factors including latitude, mean annual temperature, and ultraviolet exposure, might modify disease risk.
PubMed: 37371620
DOI: 10.3390/biomedicines11061525 -
Frontiers in Pain Research (Lausanne,... 2022The anesthetic management of fetal operative procedures (FOP) is a highly debated topic. Literature on fetal pain perception and response to external stimuli is rapidly... (Review)
Review
OBJECTIVE
The anesthetic management of fetal operative procedures (FOP) is a highly debated topic. Literature on fetal pain perception and response to external stimuli is rapidly expanding. Nonetheless, there is no consensus on the fetal consciousness nor on the instruments to measure pain levels. As a result, no guidelines or clinical recommendations on anesthesia modality during FOP are available. This systematic literature review aimed to collect the available knowledge on the most common fetal interventions, and summarize the reported outcomes for each anesthetic approach. Additional aim was to provide an overall evaluation of the most commonly used anesthetic agents.
METHODS
Two systematic literature searches were performed in Embase, Medline, Web of Science Core Collection and Cochrane Central Register of Controlled Trials up to December 2021. To best cover the available evidence, one literature search was mostly focused on fetal surgical procedures; while anesthesia during FOP was the main target for the second search. The following fetal procedures were included: fetal transfusion, laser ablation of placental anastomosis, twin-reversed arterial perfusion treatment, fetoscopic endoluminal tracheal occlusion, thoraco-amniotic shunt, vesico-amniotic shunt, myelomeningocele repair, resection of sacrococcygeal teratoma, ligation of amniotic bands, balloon valvuloplasty/septoplasty, ex-utero intrapartum treatment, and ovarian cyst resection/aspiration. Yielded articles were screened against the same inclusion criteria. Studies reporting anesthesia details and procedures' outcomes were considered. Descriptive statistical analysis was performed and findings were reported in a narrative manner.
RESULTS
The literature searches yielded 1,679 articles, with 429 being selected for full-text evaluation. A total of 168 articles were included. Overall, no significant differences were found among procedures performed under maternal anesthesia or maternal-fetal anesthesia. Procedures requiring invasive fetal manipulation resulted to be more effective when performed under maternal anesthesia only. Based on the available data, a wide range of anesthetic agents are currently deployed and no consistency has been found neither between centers nor procedures.
CONCLUSIONS
This systematic review shows great variance in the anesthetic management during FOP. Further studies, systematically reporting intraoperative fetal monitoring and fetal hormonal responses to external stimuli, are necessary to identify the best anesthetic approach. Additional investigations on pain pathways and fetal pain perception are advisable.
PubMed: 36246050
DOI: 10.3389/fpain.2022.935427 -
Cancers Mar 2023To determine the role of adjuvant chemotherapy in stage IA G2-3 and stage IB-IC pure ovarian immature teratoma (POIT), we performed a systematic review and meta-analysis... (Review)
Review
To determine the role of adjuvant chemotherapy in stage IA G2-3 and stage IB-IC pure ovarian immature teratoma (POIT), we performed a systematic review and meta-analysis by searching PubMed, Embase, Cochrane library, Web of Science, and ClinicalTrials.gov. Randomized controlled trials or cohort studies on stage IA G2-G3 or stage IB-IC POIT between 1 January 1970 and 15 December 2022 were enrolled. The recurrence rate and mortality rate were the primary outcomes, and subgroup analysis based on the tumor stage and grade was also conducted. In total, 15 studies with 707 patients were included. Compared with surveillance, adjuvant chemotherapy significantly decreased the mortality rate (RR 0.31, 95% CI 0.11-0.88, = 0.03), but not recurrence (RR 0.74, 95% CI 0.39-1.42, = 0.37), in the overall population. Subgroup analysis showed no statistical difference in the recurrence rate and mortality rate between patients who received adjuvant chemotherapy and surveillance in pediatric POIT, stage IA G2-3 POIT, stage IB-IC POIT, and stage IA-IC G3 POIT (all with > 0.05). However, patients who underwent adjuvant chemotherapy appeared to have a lower risk of both recurrence (RR 0.17, 95% CI 0.03-0.83, = 0.03) and death (RR 0.04, 95% CI 0.00-1.00, = 0.05) in adult POIT. Adjuvant chemotherapy significantly decreased the mortality rate in patients with stage I POIT and lowered the risk of recurrence in the adult subgroup. Surveillance administered in stage I POIT over IA G1 should be cautious, especially in adult patients.
PubMed: 36980627
DOI: 10.3390/cancers15061741 -
Frontiers in Endocrinology 2022Ovarian strumal carcinoid is a rare tumor in which thyroid (struma) and carcinoid components coexist. The disease is generally considered to be a borderline malignancy,...
BACKGROUND
Ovarian strumal carcinoid is a rare tumor in which thyroid (struma) and carcinoid components coexist. The disease is generally considered to be a borderline malignancy, however, cases with metastatic disease have been described. No data in the literature are available to guide diagnosis and therapy.
METHODS
We performed a pooled analysis and a systematic review of histopathological-confirmed strumal carcinoid cases published in the literature using the following keywords: "strumal carcinoid of the ovary", "strumal carcinoid case report". A case of strumal carcinoid tumor diagnosed and followed-up at the Medical Oncology Unit of Spedali Civili (Brescia, Italy) was also described and included.
RESULTS
Sixty-six eligible publications were identified, providing data from one hundred and seventeen patients, plus a case diagnosed at our institution. At presentation, among the eighty-eight patients with symptomatic disease, 37% of patients suffered from abdominal distention and 49% from pain due to a growing abdominal tumor mass, 37% from constipation (peptide YY was analyzed in only nine of them, resulting above the physiologic range). Surgery was the primary therapy in 99% of the patients. Three patients had metastatic disease at diagnosis and five patients underwent recurrence after radical surgery. Histology at disease recurrence concerned the thyroid component in two patients, the carcinoid component in two patients, both histologies in one patient. Median disease-free survival and overall survival in this series were not attained.
CONCLUSION
Strumal carcinoid of the ovary generally presents a benign behavior and surgery is curative in most cases. However, a small group of patients with this disease can undergo disease recurrence due to both the thyroid and the neuroendocrine (carcinoid) components. A follow-up in radically operated patients is therefore needed, particularly in those with a voluminous disease at diagnosis.
Topics: Carcinoid Tumor; Female; Humans; Neoplasm Recurrence, Local; Ovarian Neoplasms; Struma Ovarii
PubMed: 35528006
DOI: 10.3389/fendo.2022.871210 -
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 -
Orphanet Journal of Rare Diseases Oct 2014The association of ovarian teratoma and anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is a serious and potentially fatal pathology that occurs in young women...
The association of ovarian teratoma and anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is a serious and potentially fatal pathology that occurs in young women and that is under-recognized. Our objectives were to analyze prevalence and outcome of this association, and increase awareness over this pathology. MEDLINE and SCOPUS for all studies published prior to November 30, 2013 including the search terms: "encephalitis" and "teratoma" were considered. All articles (119) reporting one or more cases of anti-NMDAR encephalitis and confirmed ovarian teratoma (174 cases) were included. No language restrictions were applied. Suspicious cases with no evidence of ovarian teratoma (n = 40) and another type of encephalitis also associated to ovarian teratoma (n = 20) were also considered for comparison and discussion. Data of publication and case report, surgery and outcome were collected. The distribution of published cases is heterogeneous among different countries and continents, probably in relation with level of development and health care. The mean patient age is 24 years and in the majority of cases (74%), a mature teratoma was identified, sometimes microscopically following ovarian removal or at autopsy. The clinical presentation featured psychiatric symptoms and behavioural changes, with a median delay for surgery of 28 days. Twelve women died (7%), most frequently from encephalitis-related complications. In conclusion, the association ovarian teratoma and anti-NMDAR encephalitis is relatively unknown or not reported in many countries and among gynecologists. Heightened recognition of behavioral changes, diagnosis through transvaginal ultrasound and subsequent tumor removal in addition to diagnostic confirmation through the presence of anti-NMDAR antibodies must be emphasized.
Topics: Adolescent; Adult; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Female; Humans; Ovarian Neoplasms; Teratoma; Young Adult
PubMed: 25312434
DOI: 10.1186/s13023-014-0157-x -
Frontiers in Immunology 2022Anti--methyl-d-aspartate receptor encephalitis (NMDARe), a common autoimmune encephalitis, can be accompanied by demyelinating disorders, including multiple sclerosis... (Review)
Review
Anti--methyl-d-aspartate receptor encephalitis (NMDARe), a common autoimmune encephalitis, can be accompanied by demyelinating disorders, including multiple sclerosis (MS), neuromyelitis optica spectrum disorder (NMOSD), and myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD). To compare the clinical characteristics of patients with different overlapping syndromes, we searched the PubMed database and performed a systematic review. Of the 79 patients with overlapping syndromes, 15 had MS, 18 had aquaporin-4-antibody-positive NMOSD (AQP4-Ab-positive NMOSD), and 46 had MOGAD. Compared with classical NMDARe, overlapping syndromes showed atypical symptoms, such as limb weakness, sensory disturbance, and visual impairments in addition to the main symptoms of NMDARe and a lower ratio of ovarian teratoma. Patients with MOGAD overlap were the youngest, while patients with MS and AQP4-Ab-positive NMOSD overlap tended to be older than patients with classical NMDARe. A majority of patients with NMDARe who overlapped with MS or AQP4-Ab-positive NMOSD were female, but this was not the case for patients overlapped with MOGAD. When NMDARe and demyelinating diseases occurred sequentially, the interval was the longest in patients with NMDARe overlapped with MS. A favorable outcome was observed in patients overlapping with MOGAD, but no robust comparison can be drawn with the patients overlapping with AQP4-Ab-positive NMOSD and MS regarding the small number of available data. The long-term prognosis of overlapping syndromes needs further investigation.
Topics: Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Autoantibodies; Female; Humans; Male; Multiple Sclerosis; Myelin-Oligodendrocyte Glycoprotein; Neuromyelitis Optica; Receptors, N-Methyl-D-Aspartate
PubMed: 35837405
DOI: 10.3389/fimmu.2022.857443 -
Cureus Dec 2019The role of preoperative CA 19-9 levels in patients with ovarian mature cystic teratoma (MCT) and the association of elevated levels of the biomarker with patients' and... (Review)
Review
The role of preoperative CA 19-9 levels in patients with ovarian mature cystic teratoma (MCT) and the association of elevated levels of the biomarker with patients' and tumor characteristics were evaluated. Four electronic databases were searched for articles published up to September 2019. Trials that evaluated the significance of elevated CA 19-9 in patients with ovarian MCTs and publications with > 20 patients were considered eligible for inclusion. Seven studies that included 995 patients with an ovarian MCT who were evaluated with elevated (n = 364) or normal (n = 631) CA 19-9 levels were included. Mean tumor size was significantly increased in patients with elevated CA 19-9 levels (p = 0.038). The rate of ovarian torsion was significantly increased in the elevated CA 19-9 group (p = 0.04). The present study highlights the importance of CA 19-9 as a marker in the diagnosis of MCT, and a meta-analysis supports that it could raise a high degree of clinical suspicion of early recognition of torsion and early surgical management due to complications related to increased size. Nonetheless, the diagnostic value of CA 19-9 is still limited and CA 19-9 can still serve only as a supplementary diagnostic tool in patients with MCTs.
PubMed: 31938630
DOI: 10.7759/cureus.6342 -
Translational Psychiatry Mar 2024The global impact of SARS-CoV-2 infection has raised concerns about secondary diseases beyond acute illness. This review explores the significance and potential... (Meta-Analysis)
Meta-Analysis
The global impact of SARS-CoV-2 infection has raised concerns about secondary diseases beyond acute illness. This review explores the significance and potential underlying mechanisms of how SARS-CoV-2 infection might elicit an immune response targeting N-methyl-D-aspartate (NMDA) receptors, and its implications for autoimmune-driven neuropsychiatric manifestations. We identified 19 published case reports of NMDA receptor encephalitis associated with SARS-CoV-2 infection or vaccination by a systematic literature search. The significance of these reports was limited since it is not clear if a coincidental or causal relationship exists between SARS-CoV-2 infection or vaccination and manifestation of NMDA receptor encephalitis. The included studies were hampered by difficulties in establishing if these patients had pre-existing NMDA receptor antibodies which entered the brain by infection- or vaccination-associated transient blood-brain barrier leakage. In addition, four cases had comorbid ovarian teratoma, which is a known trigger for development of NMDA receptor encephalitis. Considering that billions of people have contracted COVID-19 or have been vaccinated against this virus, the publication of only 19 case reports with a possible link to NMDA receptor encephalitis, indicates that it is rare. In conclusion, these findings do not support the case that SARS-CoV-2 infection or vaccination led to an increase of existing or de novo encephalitis mediated by an autoimmune response targeting NMDA receptor function. Nevertheless, this work underscores the importance of ongoing vigilance in monitoring viral outbreaks and their potential impact on the central nervous system through basic, epidemiological and translational research.
Topics: Humans; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Antibodies; COVID-19; Receptors, N-Methyl-D-Aspartate; SARS-CoV-2
PubMed: 38459000
DOI: 10.1038/s41398-024-02831-0 -
Frontiers in Pediatrics 2021The use of robotics-assisted surgery in oncology has been proved effective and safe in adults. Despite these results, the use of robotics has been rarely reported for...
AIM
The use of robotics-assisted surgery in oncology has been proved effective and safe in adults. Despite these results, the use of robotics has been rarely reported for pediatric oncology. Our review aims to evaluate the safety and feasibility of robotics-assisted surgery in this field, analyzing our experience and performing a systematic review of the most recent studies.
METHODS
We reviewed all patients affected by an oncological disease who underwent a robotics-assisted procedure at our institute. We performed a systematic review of the literature from 2012 to 2021 on the subjects.
FINDINGS
A total of 14 patients underwent robotics-assisted tumor resection. Eleven procedures (median age 13.2-years old) were carried out in children with adnexal lesions (seven tumor excision and four ovariectomies). Histological diagnosis was mature teratoma (six), serous papillary cystadenofibromas of the fallopian tube (two), ovarian serous cystadenoma (one), ovarian mucinous cystadenoma (one), and ovarian seromucinous cystadenoma. The median length of stay was 2 days. No recurrences or complications at a median follow-up of 2.1-years were observed. A 5-year-old girl underwent a complete posterior resection of a type 3 sacrococcygeal tumor with a robotics-assisted approach for the dissection of a possible intraabdominal residual component of the lesion. No intra- and postoperative complications were recorded. Complete excision of a recurrent differentiating neuroblastoma of the left para-renal region was performed on a 9-year-old girl. An idiopathic anaphylactic shock occurred 1 day after the procedure. At 9 months' follow-up, no local recurrences of the lesion were observed. Overall, we reported no conversion to open surgery. Lastly, a robotic excision of a growing left superior mediastinal intermixed ganglioneuroblastoma was performed on an 8-year-old girl with no postoperative complications. Follow-up was uneventful (7 months). In the literature, the rate of complications ranges from 0 to 28%, mainly related to difficult dissection and impaired anatomy. Conversion is reported in 5% of all oncological procedures, due to more invading tumors and altered anatomical features. No robotics-related complications were reported.
CONCLUSION
Robotics-assisted surgery in pediatric oncology has proven to be feasible. Nevertheless, its use should be limited to selected cases and performed by highly trained oncological surgeons. Preparation and patient positioning, alongside a correct port placement, are crucial to carrying out these procedures. Further innovations in robotics may allow a wider application of this technology in pediatric oncology.
PubMed: 35118030
DOI: 10.3389/fped.2021.780830