Elevated aspartate aminotransferase and alanine aminotransferase in the torsion of ovarian mature cystic teratoma: normalised after operation for torsion.

The aim of the study was to report the elevated liver function test levels in torsion of ovarian mature cystic teratoma (MCT). A retrospective review was performed of 116 patients with MCT who underwent surgery in our hospital between 2010 and 2017. Eleven of 116 patients were with torsion of MCT. Of the 11 torsion of MCT cases, 6 of those showed abnormal elevated levels of aspartate transaminase (AST)/alanine aminotransferase (ALT). After operation, AST/ALT levels recovered to normal ranges. Elevated liver function levels require attention to complications associated with anaesthesia and emergency operation. However, the current report shows that emergency surgery should not be delayed to assess other causes of elevated liver function tests.Impact statementWhat is already known on this subject? Abnormal liver function tests require attention to complications associated with emergency surgery.What do the results of this study add: Our study shows that some patients with torsion of MCT returned to normal levels of AST/ALT after surgery without any medications to improve liver function.What are the implications of these findings for clinical practice and/or further research? This study would offer that in some cases with torsion of MCT, emergency surgery should not be delayed and the assessment of other possible causes of these elevations postponed to postoperative period. Additional studies are required to assess the correlation between elevated AST/ALT levels and torsion of MCT.

N-methyl-D-aspartate receptor antibody production from germinal center reactions: Therapeutic implications.

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.

Primary malignant melanoma arising from ruptured ovarian mature cystic teratoma with elevated serum CA 19-9: a case report and review of literature.

BACKGROUND: Ovarian mature cystic teratomas comprise tissues derived from all three germ layers. In rare incidences, malignant tumors may arise from ovarian mature cystic teratoma, which occurs in 0.2-1.8% of cases. A variety of tumors can arise within mature cystic teratoma, among which malignant melanoma is exceedingly rare. CASE PRESENTATION: A 42-year-old woman presented with abdominal pain. Transvaginal ultrasonography showed mixed echogenic cystic masses in both ovaries. Her serum cancer antigen (CA19-9) level was elevated at 29,770 U/ml. Surgical excision was performed. Histologic examination showed infiltrating nests of pleomorphic cells with prominent nucleoli and black pigments in the background of a mature cystic teratoma. These pleomorphic cells showed strong immunoreactivity for Melan-A and HMB-45. The patient was re-evaluated and the possibility of a melanoma at any other site was ruled out. Based on these findings, we concluded that the malignant melanoma originated from the ovarian mature cystic teratoma. CONCLUSION: We report a rare case of primary malignant melanoma derived from an ovarian mature cystic teratoma.

Antenatal Prediction of Neonatal Survival in Sacrococcygeal Teratoma.

OBJECTIVES: In sacrococcygeal teratoma, the presence of high-output cardiac failure resulting from arteriovenous shunting through the large tumor has been associated with an adverse outcome. The objective of this study was to determine whether the tumor size and cardiac biomarkers in cord blood can predict neonatal survival in sacrococcygeal teratoma. METHODS: The study population consisted of 25 neonates with sacrococcygeal teratoma. Tumor size was calculated by the ellipsoid formula using dimensions measured by antenatal ultrasound ([length × width × depth in cm] × 0.52= volume in cm3 ). To adjust the gestational age, the tumor volume index (tumor volume/biparietal diameter) was adopted in the analysis. Cardiac biomarkers for heart failure (N-terminal pro-B-type natriuretic peptide [NT-pro-BNP] and cardiac troponin T [cTnT]) were measured in cord blood taken at the time of delivery. RESULTS: The rate of neonatal death was 24% (6 of 25). The cases that resulted in neonatal death had a higher tumor volume index and higher concentrations of NT-pro-BNP and cTnT than those with survival. A tumor volume index of greater than 60 cm3 /cm, elevated NT-pro-BNP (>2000 pg/mL), and elevated cTnT (>0.08 ng/mL) had sensitivity of 100% for prediction of neonatal death. CONCLUSIONS: The tumor volume index and cord blood biomarkers for heart failure can be promising prognostic markers for neonatal survival in sacrococcygeal teratoma.

Elevated preoperative neutrophil : lymphocyte ratio as a preoperative indicator of mature cystic teratoma with malignant transformation.

AIM: To examine the usefulness of the neutrophil : lymphocyte (N/L) ratio as a cost-effective and simple diagnostic marker of mature cystic teratoma (MCT) with malignant transformation (MT). METHODS: A retrospective chart review was performed between 1998 and 2013 of 12 MCT patients with MT and between 2009 and 2013 of 130 patients with benign MCT. Data were collected on age, tumor size, white blood cell count with differential counts, tumor marker levels, and presenting features. RESULTS: Older age, greater tumor size, higher CA19-9 or CA125, higher neutrophil count, and higher N/L ratio were associated with MT on univariate analysis. White blood cell count; lymphocyte count; and the tumor marker squamous cell carcinoma antigen were not associated with MT. Older age (≥median), larger tumor size (≥10 cm), and high N/L ratio (≥5.0) were predictors of MT (hazard ratio, 11.51, 5.87, and 11.11, respectively). Six of 12 patients were diagnosed with MT on preoperative magnetic resonance imaging and five of 12 had an N/L ratio ≥5.0. CONCLUSIONS: Neutrophil : lymphocyte ratio is a potential preoperative diagnostic marker of MT. The optimal cut-off should be determined in future large-scale studies.

Serum pentraxin 3 as a possible marker for mature cystic teratomas.

Pentraxin 3 (PTX3) is an inflammatory mediator that is released by a wide range of tissues and cells. Elevated PTX3 levels may represent a useful diagnostic and/or prognostic marker for a number of diseases. The purpose of this study was to investigate serum PTX3 levels in benign gynecological conditions including mature cystic teratomas (MCTs), endometriomas, and uterine leiomyomas. Serum PTX3 levels of the MCT group were found to be significantly higher compared to those of the other groups, including healthy controls (p = 0.001), although carbohydrate antigen 19-9 (CA19-9) did not exhibit a significant difference. Serum PTX3 levels of the MCT, but not the endometrioma group, were also found to have significantly decreased post-operatively (mean ± standard deviation, 4.98 ± 2.10 to 3.61 ± 1.53 ng/mL). Immunohistochemical analyses demonstrated positive staining for PTX3 protein in the sebaceous glands, epidermal tissues, and hair roots of MCT specimens. PTX3 is expressed by MCTs and is associated with increased serum concentrations compared to healthy controls and patients with either endometriomas or uterine leiomyomas. We conclude that serum PTX3 levels could be used as a potential diagnostic marker for MCTs, especially helpful in differentiating them from endometriomas with elevated expression of CA19-9.

Immature ovarian teratoma with hyponatremia and low serum vasopressin level.

Hyponatremia is often caused by the syndrome of inappropriate secretion of antidiuretic hormone (SIADH). Hypersecretion of vasopressin from malignant tumors can be considered a cause of SIADH. Most of these ectopic productions of vasopressin are complications of small cell lung cancer. Cases concomitant with ovarian tumors are very rare, and a specific causative substance from the ovary is often unknown. A 16-year-old woman was diagnosed with an ovarian tumor. She developed hyponatremia that was resistant to medical treatment, but immediately improved after surgical resection of the tumor. Her diagnosis was SIADH caused by an ovarian tumor; however, her serum vasopressin level was normal. It is possible that a vasopressin-like substance causing SIADH was secreted by either nervous system tissue within an immature teratoma or small cell lung cancer. We should be cautious when SIADH is a complication of an ovarian tumor.

The clinical significance of CA19-9 in ovarian mature cystic teratoma.

OBJECTIVE: To evaluate the clinical significance of CA19-9 in patients with ovarian mature cystic teratoma (MCT). MATERIALS AND METHODS: A retrospective study was performed on 65 patients with pathologically-confirmed MCT and 80 patients with benign epithelial ovarian tumors. Serum tumor markers for all patients and tissue CA19-9 for MCTs were measured. The relationships between clinical characteris- tics of MCTs and CA19-9, as well as the correlation between serum and tissue level of CA19-9 in MCTs, were evaluated. RESULTS: The mean serum level of CA19-9 in MCTs was significantly higher than that in benign ovarian epithelial tumors (49.9 ± 73.4 IU/ml vs. 17.08 ± 24.8 IU/ml). CA19-9 was the only tumor marker with a mean serum level above the cut-off value and the elevation rate was 30.76% in MCTs. The positive tissue expression rate of CA19-9 in MCT patients were 50.9% and were higher than that of preoperative serum levels (50.9% vs. 32.7%). CONCLUSION: Serum CA19-9 has the highest positivity rate among other tumor markers in MCT. Elevated serum CA19-9 is not an uncommon finding MCT and could be used as a marker in the differential diagnosis of MCT in patients with pelvic mass.

Expression and clinical significance of tumor markers in ovarian mature cystic teratoma.

BACKGROUND: CA19-9, CA125, AFP, CEA, and CA153 are common cancer biomarkers. This study aimed to detect the expression and assess clinical significance of these biomarkers in ovarian mature cystic teratomas (OMCT). MATERIALS AND METHODS: The clinical data of 312 resected patients with ovarian mature cystic teratoma referred to the third Hospital of Suzhou University from January 2008 to February 2014 were analyzed retrospectively. Levels of serum CA19-9, CA125, CA153, AFP, and CEA were detected. RESULTS: Levels of serum CA19-9, CA125, AFP, and CEA were detected in all patients, but the level of serum CA153 was detected in 36 patients. The positive expression of these biomarkers above is 45.83%, 6.09%, 0.00%, 0.64%, and 2.78%, respectively. Ovarian tumor diameter in CA19-9 and CA 125 elevation is larger than that in negative group. The levels of serum CA19-9 and CA125 in multilocular ovarian tumor patients is higher than that in unilocular ovarian tumor patients. The level of serum CA19-9 in bilateral ovarian tumor patients is higher than that in unilateral patients. The level of CA19-9 in patients three months after operation decreased significantly compared with that before operation. CONCLUSIONS: The present study suggests that elevated CA 19-9 is probably the most reliable marker among many tumor markers in the OMCT. It is valuable for diagnosis and recurrence of OMCT.

[Testicular teratoma in children: Analysis of 64 cases].

OBJECTIVE: To improve the diagnosis and treatment of testicular teratoma in children by analysis of clinical data. METHODS: We retrospectively analyzed the clinical data about 64 cases of testicular teratoma treated in the Children's Hospital of Chongqing Medical University from 1995 to 2014. RESULTS: Sixty-one of the cases presented painless scrotal mass with a sense of bearing down and the other 3 cases were confirmed because of empty scrotum diagnosed as cryptorchidism. The level of serum alpha fetal protein ( AFP) was obviously increased in 46 cases but normal in the other 18 preoperatively. Ultrasonography manifested abnormal inhomogeneous echo zones with calcification or necrosis. X-ray examination presented patchy or curvilinear high-density shadows in 28 cases. Forty-one of the patients underwent testis-sparing surgery (TSS) , 20 received high inguinal orchiectomy, and 3 refused surgical treatment. Pathological examination revealed 3 mature germinal layers in the 49 cases of mature teratoma and immature germinal tissue, including the original neural tube, and 11 cases of immature teratoma. The mature cases were exempted from chemotherapy, while the immature cases received the combination of cisplatin, etoposide, and bleomycin (PEB). The patients were followed up for 2 years postoperatively, which revealed no recurrence or metastasis. CONCLUSION: Most children with testicular teratoma presented painless scrotal mass with a sense of bearing down and with abnormal serum AFP in most cases. Ultrasonography and plain radiography of the scrotum contribute to the diagnosis of the tumor. TSS is the main treatment option and intraoperative frozen-section can help the surgeons decide on the surgical mode. Postoperative chemotherapy is necessitated for immature teratoma but not for mature cases.

Mature cystic teratomas: relationship between histopathological contents and clinical features.

AIMS: The aim of this study was to evaluate the relationship between pathological findings and clinical features in patients with ovarian mature cystic teratomas (MCTs). MATERIALS AND METHODS: We reviewed and compared the demographic and clinical features of 32 patients confirmed pathologically as having MCT at a university hospital from 2009 to 2014, with MCT contents such as skin, hair, sebum, and cartilage. RESULTS: The mean age of the patients was 33.7 ± 13.4 years. The mean tumor diameter was 7.1 ± 3.3 cm (range: 2-15 cm). The mean serum CA-19.9 level was 37.5 ± 79.5 IU/ml and the mean serum CA-125 level was 29.1 ± 33.0 IU/ml. The postmenopausal and pregnancy status rates of participants were 18.8, and 15.6%, respectively. The mean age, postmenopausal and pregnancy status, tumor size, symptoms related to MCT and laterality of the tumor did not differ among the patients according to the MCT contents. CONCLUSIONS: Our findings suggest no relationship between the clinical features and histopathological contents of MCTs.

Cytotoxic reaction mediators: granzymes A and B in women with ovarian cancer.

The purpose of this work was the assessment of cytotoxic reaction mediators - granzymes A and B in the serum of women with ovarian tumors. The study included 120 women with proven ovarian tumors. The control group consisted of 60 healthy women in whom no pathological changes within the reproductive system were detected. Concentrations of granzymes A and B were measured by enzyme-linked immunosorbent (ELISA) assay. The highest concentrations of the studied parameters were observed in serum of women with ovarian cancer. Moreover, the concentrations of granzymes A and B in patients with ovarian cancer were substantially increased in comparison to concentrations in patients with ovarian cystadenomas (P < 0.0001) or ovarian teratomas (P < 0.0001).

Successful treatment of metastatic ßHCG-secreting germ cell tumor occurring 3 years after total resection of a pineal mature teratoma.

UNLABELLED: Patients diagnosed with intracranial teratoma are at risk for developing a recurrent malignant germ cell tumor. We describe a 14-year-old boy initially diagnosed with a mature teratoma in the pineal region that recurred as a metastatic beta-human chorionic gonadotropin (ßHCG)-secreting germ cell tumor 3 years after gross total resection. A surveillance brain MRI scan during follow-up demonstrated multiple lesions within the ventricular and subependymal area infiltrating the brain parenchyma along with concomitant elevated levels of ßHCG in both the serum and cerebrospinal fluid. The patient underwent chemotherapy with PEI (cis-platinum, etoposide, ifosfamide) followed by radiation therapy according to the SIOP CNS GCT protocol. The patient is currently alive without evidence of disease 35 months after starting therapy. CONCLUSIONS: A careful and long-term follow-up including scheduled tumor markers as well as surveillance MRI scans is required for patients with intracranial teratoma in an effort to detect and diagnose recurrent malignant disease, especially since multimodal therapy provides the potential for long-term cure.

Significant simultaneous changes in serum CA19-9 and CA125 due to prolonged torsion of mature cystic teratoma of the ovary.

Mature cystic teratoma is a common benign neoplasm of the ovary. Complications occur in approximately 20% of cases. Clinical manifestations, laboratory findings, and imaging studies can assist in making a diagnosis of ovarian torsion of mature cystic teratoma. Furthermore, serum tumor markers may be helpful for diagnosing mature cystic teratoma and its torsion and, thus, can lead to early surgical intervention. A 56-year-old woman presented with a huge pelvic mass and pelvic pain. Serum CA19-9, CA125, and carcinoembryonic antigen levels were abnormally elevated at >700 U/ml, 282.5 U/ml, and 3.94 U/ml, respectively. The tumor was surrounded by extensive adhesions and showed inflammatory changes. The serum levels of these markers returned to normal levels after surgery.

Comparison of pelvic masses score (PMS) and Risk of Malignancy Index (RMI 3) in the evaluation of pelvic masses.

PURPOSE: Ovarian cancer is the fourth cause of death from cancer in women worldwide and the majority of its diagnoses is made in an advanced stage of the disease. Several sonographic scoring systems have been created for a better preoperative discrimination between benign and malignant pelvic masses. The aim of this study was to evaluate the performances of the Risk of the Malignancy Index 3 (RMI 3) and the Pelvic Masses Score (PMS). MATERIALS AND METHODS: This retrospective study was performed in 55 women admitted to the department of Obstetrics and Gynecology of University of Udine for surgical exploration of pelvic masses between 2009 and 2012. Sensitivity, specificity, positive predictive value (PPV) and negative predictive value (NPV) were calculated for both the scores. RESULTS: PMS showed a sensitivity of 100%, a specificity of 93.8%, a PPV of 70%, and a NPV of 100%, while RMI 3 yielded a sensitivity of 85%, a specificity of 91%, a PPV of 60%, and a NPV of 97.8%. CONCLUSION: The authors found that, in discriminating between benign and malignant pelvic disease, the PMS method was more reliable than RMI3. PMS is a simple scoring system which can be used in clinical practice.

Primary ovarian malignant melanoma arising in teratomatous component of mixed- germ cell tumor in a child: case report.

Primary ovarian malignant melanoma arising in teratomatous component of germ cell tumors is seen extremely rare with most reports being only of single cases and small series in reproductive aged woman and mostly from cystic teratoma, whereas information on pediatric presentation is sparse. This case is reported for being extremely rare tumor.

The Diagnostic Accuracy of Serum Alpha-Fetoprotein Levels in Follow-up for Recurrence of Sacrococcygeal Teratoma; a Nationwide Review of SCT Cases in the Netherlands.

BACKGROUND: Serum alpha-fetoprotein (AFP) is often used as tumour marker for recurrent sacrococcygeal teratoma (SCT). We aimed to assess the normal dynamics of serum AFP levels after initial resection and diagnostic accuracy of serum AFP levels the follow-up for recurrence in SCT. METHODS: This retrospective study included 57 patients treated for SCT in the six pediatric surgical centers in the Netherlands from 1980 to 2018. MAIN RESULTS: 57 patients were included in the study of whom 19 children developed 20 recurrences at a median of 14.0 months after initial resection. No significant difference was found in serum AFP level dynamics between the recurrence and non-recurrence group after initial resection (p = 0.950). Serum AFP levels did not significantly increase before recurrence (p = 0.106) compared to serum AFP levels of children without recurrence at the same time. However, serum AFP levels did significantly increase in malignant recurrences (n = 7) (p = 0.03) compared to patients without recurrence. A cut-off value of 55 µg/L was found to be predictive for recurrent SCT with an Area Under the Curve (AUC) of 0.636 with sensitivity of 50% and specificity of 100%. CONCLUSION: Dynamics of serum AFP levels are not different between patients with and without recurrence after initial resection of SCT. Serum AFP levels are not predictive for mature or immature recurrent SCT and normal AFP levels do not rule out recurrent SCT. However, serum AFP levels exceeding 55 µg/L can indicate recurrent SCT, especially malignant recurrences.

Malignant transformation of ovarian mature cystic teratoma with rupture, elevated serum CA199, CA12, CEA: A case report.

RATIONALE: Reports of mature cystic teratomas (MCTs) with associated complications and changes in serum cancer antigen levels are rare. Herein, we report a rare case of MCT with associated complications (rupture and malignant transformation), high levels of serum cancer antigens (CA19-9, CA12, and CEA), and surgical therapy. PATIENT CONCERNS: An 81-year-old woman was referred to our emergency department because of diffuse abdominal pain and distension for 20 days. DIAGNOSES: Imaging findings, including transabdominal ultrasonography, computed tomography, and magnetic resonance imaging, revealed a complex solid cystic mass in the lower abdomen. Preoperative laboratory test results showed high levels of serum cancer antigens (CA19-9, CA12, and CEA) in MCT. Histopathological examination of the specimen revealed a MCT with rupture and malignant transformation. INTERVENTIONS: The patient underwent a total abdominal hysterectomy, bilateral oophorectomy, and partial omentectomy. The patient did not undergo chemotherapy after surgery. OUTCOMES: The follow-up period was 12 months. The patient recovered well without focal local recurrence or distant metastasis after the surgery. LESSONS: The study aims to report a new case of MCT with associated complications (rupture and malignant transformation) and changes in serum cancer antigen levels. Although this tumor presents as a complex solid cystic mass, detection of the intratumoral fat component is a key diagnostic imaging feature. A high level of serum cancer antigen may indicate the malignant transformation of MCT. In this case, surgery was an effective treatment for the MCT.

Enhancement of ovarian tumor classification by improved reproducibility in matrix-assisted laser desorption/ionization time-of-flight mass spectrometry of serum glycans.

The serum N-glycome is a promising source of biomarker discovery. Matrix-assisted laser desorption/ionization time-of-flight (MALDI-TOF) mass spectrometry (MS) profiling of serum N-glycans was attempted for differentiating borderline ovarian tumor from benign cases, for which a low data spread is essential. An experimental protocol using matrix-prespotted MALDI plates and fast vacuum drying of the loaded N-glycan samples was developed, thereby minimizing the intensity variations in the replicates to an average relative standard deviation (RSD) of 3.96% for the highest N-glycan peak (m/z 1485.53) of the Sigma-Aldrich serum standard. When applied to sera of ovarian tumors, this procedure exhibited an average RSD of 5.74% for m/z 1485.53 and of 7.28% for all MS peaks. This improved reproducibility combined with the OVA-Beyond(®) screening software resulted in 75.1% and 79.4% correct classification for benign and borderline tumor samples, respectively, while the classification rates by the conventional ovarian tumor marker CA-125 were 54.4% and 53.1%, respectively. Both true positive rate and true negative rate fluctuated with small numbers of markers and converged as the number of markers increased. Cross-validations were performed in comparison with CA-125. These results suggest that our optimized process for MALDI-TOF MS of the serum glycome has a great potential for the screening of early stage ovarian cancer.

CA19-9 elevation in ovarian mature cystic teratoma: discrimination from ovarian cancer - CA19-9 level in teratoma.

BACKGROUND: We aimed to identify clinical characteristics of ovarian mature cystic teratoma (MCT) in association with CA19-9 elevation, and to determine if CA19-9 is a useful marker in discrimination of MCT from ovarian cancer (OC). MATERIAL AND METHODS: Medical records of 322 women with pathologically-confirmed MCT or OC (stage 1 or 2) were reviewed retrospectively. The relationships between the characteristics of MCT (mean diameter, bilaterality, and pathologic components) and elevated CA19-9 were evaluated. Tumor markers in MCT were compared to those in OC. RESULTS: MCTs with CA19-9 elevation were correlated with a larger diameter (8.53±3.84 cm vs. 6.95±3.97 cm, p=0.002) and presence of fat component (67.1% vs. 32.9%, p<0.001), compared to those with normal CA 19-9. Although the incidence of CA19-9 elevation was not different between patients with MCT and OC (p=0.700), the mean value of CA19-9 was higher in those with OC (114.66±20.66 U/mL vs. 508.58±261.63 U/mL, p=0.013). In addition, simultaneous elevation of CA125 and CA19-9 was associated with a higher probability of malignant neoplasm (p<0.001; odds ratio: 23.7; 95% confidence interval: 8.863-63.576) than single elevation of CA 19-9. CONCLUSIONS: CA19-9 could be an important tool in the diagnosis of ovarian mature cystic teratoma. CA19-9, in combination with CA125, might be a useful marker in discrimination of MCT from cancer.