Dermoid cyst with secretion of CA 19-9 detected by 18F-FDG PET/CT: A case report.

INTRODUCTION: Carbohydrate antigen 19-9 (CA 19-9) is a tumor glycolipid, frequently elevated in the serum of patients due to malignancies from gastrointestinal organs; in particular, the pancreas. This carbohydrate antigen is also expressed in benign diseases. PATIENT CONCERNS: A case of a 27-year-old female who has an unknown origin CA 19-9 elevation for 2 years. DIAGNOSIS: After the left ovarian cystectomy and microscopic examination, the final diagnosis is a dermoid cyst. The dermoid cyst shows increased F-fluorodeoxyglucose (F-FDG) uptake in the F-FDG positron emission tomography (PET)/computed tomography (CT) study. INTERVENTION AND OUTCOMES: The laparoscopic oophorocystectomy was performed. It was observed that the patient's CA 19-9 level returned to normal after the surgery 6 months later. This showed that the dermoid cyst was responsible for the abnormal CA 19-9 level. CONCLUSION: In this case, we can learn that the F-FDG PET/CT scan has potential use in patients with unknown origin of elevation CA 19-9.

Ovarian damage due to cyst removal: a comparison of endometriomas and dermoid cysts.

INTRODUCTION: Surgical treatment of endometriomas and potential damage to the ovary have been debated. Studies have described the inconsistent risk of unintended removal of ovarian tissue when a cystectomy of an endometrioma is performed. We evaluated the risk of inadvertently removed ovarian tissue during surgery by comparing specimens of endometriomas and dermoid cysts removed laparoscopically. MATERIAL AND METHODS: The material included 326 women in a retrospective cohort study at Rigshospitalet, University hospital in Copenhagen, Denmark from 2011 to 2013. Surgery was performed laparoscopically for 393 benign cysts with a diagnosis of either endometrioma (n = 294) or dermoid cyst (n = 99). The microscopic existence of ovarian tissue in the cystectomy specimens were compared and correlation between CA 125 and size of cysts was examined. RESULTS: In total, 80.3% endometrioma cystectomies disclosed ovarian stroma compared with 17.2% of the resected dermoid cysts (p < 0.001). The difference was found despite skilled laparoscopic surgeons performing the procedure in the endometriosis cohort. A significant positive correlation between the size of endometriomas and the value of CA 125 was found (p < 0.009). CONCLUSION: The risk of removal of ovarian tissue during laparoscopic surgery is significantly higher for endometriomas than for dermoid cysts.

PTCH1 gene polymorphisms in ovarian tumors: potential protective role of c.3944T allele.

In this study we investigated the types and role of different genetic changes of PTCH1 gene in three different types of ovarian tumors: carcinomas, fibromas and dermoids. LOH of the PTCH1 region was detected in 27.3% ovarian carcinoma samples, 18.18% ovarian fibroma samples and 55.56% ovarian dermoid samples. No point mutations were detected in any of the three types of ovarian tumors. SNP c.3944C>T showed significant differences between ovarian carcinoma and control samples with the minor T allele being significantly higher in controls compared to ovarian carcinomas. Interestingly, a new polymorphism c.-1184G>A was found only in tumor samples and further analyses should be performed in order to elucidate its potential role in ovarian tumors.

Laparoscopic approach to ovarian dermoid cysts.

INTRODUCTION: Ovarian dermoid cysts (mature cystic teratomas) are a benign type of germ cell tumours and the most common ovarian neoplasms in women of fertile age. The aim of this study was to analyze the safety of the laparoscopic approach in ovarian dermoid cysts. METHODS: We performed a prospective study between 2006 and ' 2010 including 38 mature cystic teratomas treated either laparoscopically or by open access. All preoperative and postoperative data were included in an MS Access database and statistically analysed with SPSS v. 17 for Windows. RESULTS: The study group was divided into 2 subgroups according to the approach: laparoscopic (25 cases - 2 conversions) and classic (13 cases). The mean age of the patients was 40.34 years (range 19-74): 36.92 years for laparoscopic group and significantly higher 46.21 years for open approach group. Twelve cases were admitted as emergencies either because of complications (torsion or rupture of the teratoma) (11 cases) or associated with acute appendicitis (one case). The latter did not influence the decision for open or laparoscopic approach. Only 29 out of 38 cases had preoperative measurement of CA 125. CA 19-9 was performed in 27 cases and elevated levels were found in 21 cases (78%). Cysts over 10 cm presented higher values of CA 19-9. The mean cysts diameter was 11.29 cm (range 2-27 cm): 13.93 cm mean cyst diameter for open approach vs 9.75 cm for laparoscopic approach. The specimen removal required aspiration of the content for cysts bigger the 10 cm in laparoscopic approach. Mean hospital stay was 4.05 days (range 2-6 days) for the laparoscopic group, significantly lower when compared with the open approach group: 6.96 days (range 5-16 days). CONCLUSIONS: Laparoscopic management of ovarian dermoid tumours is a safe and efficient procedure. It does not increase complications rate in comparison with the open approach, offering a shorter hospital stay, a quick recovery and very important, it allows a conservative treatment, especially in premenopausal women who want to be pregnant.

Quantitative detection of serum survivin and its relationship with prognostic factors in ovarian cancer.

OBJECTIVE: This study was conducted to determine the clinical significance of serum survivin in ovarian cancer. METHODS: A total of 65 patients with ovarian tumors including 21 epithelial ovarian cancer, 22 benign epithelial ovarian tumor, 13 dermoid cyst and 9 endometrioma cases were investigated. All histologic types of cancer were serous carcinoma except for 1 case of undifferentiated carcinoma. ELISA was employed to evaluate the serum survivin levels. For statistical analysis, we used Fisher's exact test, Spearman's correlation coefficient test, the Mann-Whitney U test, the log-rank test and Cox regression analysis. RESULTS: The survivin level was higher in serous ovarian cancer than in benign epithelial tumors with marginal significance. In ovarian cancer, serum survivin was positively correlated with age, advanced stage and poor disease-free survival. Interestingly, high survivin level positivity was associated with positive peritoneal cytology and omental metastasis of ovarian cancer. CONCLUSIONS: Our data collectively suggest that serum survivin reflects the peritoneal metastasis of serous ovarian cancer and may thus be useful as a prognostic biomarker.

[Comparison of laparoscopic vs. laparotomy treatment in ovarian teratomas]. / Comparación del tratamiento laparoscópico vs laparotomía en teratomas ováricos.

BACKGROUND: Benign cystic teratoma is one of the most common benign tumors of the ovary, according to international series represents between 44 and 62% of all ovarian tumors diagnosed in women younger than 40 years. OBJECTIVES: To evaluate and compare the efficacy and safety between laparoscopy and laparotomy in the management of ovarian teratomas, as well as the recurrence between both techniques. MATERIALS AND METHOD: Retrospective, clinical series study involving 169 cases of ovarian teratomas operated at the Instituto Nacional de Perinatología Isidro Espinosa de los Reyes in the period comprehended between 2000-2008. The cases were divided into the ones solved by laparoscopy, corresponding to group 1 (102 cases) and 67 cases solved by laparotomy, corresponding to group 2. RESULTS: Group 1 (laparoscopic approach) consisted of 102 patients (60.3%), and group 2 (laparotomy approach) had 67 patients (39.7%). Surgical bleeding: 55.7 +/- 21.4 mL vs. 91.6 +/- 29.2 mL (p < 0.0001), hospital stay: 1.6 +/- 0.57 vs. 2.7 +/- 0.42 days (p < 0.0001), surgical time: 110.3 +/- 27.2 vs. 83.6 +/- 26.9 minutes (p < 0.0001), teratoma size: 7.2 +/- 2.3 vs. 11.5 +/- 4.2 cm (p < 0.0001) in groups 1 and 2, respectively. Teratoma rupture had 57.8% incidence (59 cases) in group 1, and 16.4% in group 2 (11 cases). Laparoscopy was a risk factor for broken open for ovarian cyst (OR: 6.9; CI 95%: 3.3-14.8). Not a single case was complicated by chemical peritonitis in any study group. CONCLUSIONS: Laparoscopic approach to teratomas is a safe and efficient procedure, it does not increase complications in comparison to the laparotomy approach; it presents less bleeding and short hospital stay, offering a quick recovery.

Virilizing ovarian dermoid cyst with leydig cells.

BACKGROUND: The clinical observation of virilization is a rare finding that has a number of possible explanations. Overall, ovarian tumors causing virilization are exceedingly rare and mostly occur in post-menopausal women. In fact, there are no reported cases of virilization from a testosterone-producing ovarian dermoid in the adolescent female age group. The most frequent germ cell tumor derived from the ovaries is the benign cystic teratoma (dermoid) which accounts for 25% of all ovarian neoplasms. Teratomas consist of tissues that recapitulate the ectoderm, endoderm, and mesoderm. Usually the tumors are asymptomatic, but they occasionally can cause severe pain if there is torsion or if sebaceous material perforates the cyst wall, leading to reactive peritonitis. CASE: A 12-year-old female was found to have a large 3 5 x 19 x 12 cm ovarian mature cystic teratoma arising from her right ovary. The patient also displayed evidence of masculinization demonstrated by a deepening voice and clitoromegaly. The dermoid was producing large amounts of testosterone from a nest of Leydig cells found pathologically in the mass. CONCLUSION: Benign cystic teratomas can produce active hormones, albeit rarely. This is a finding important to consider when ovarian cystectomy is performed for removal of a benign cystic teratoma.

Squamous cell carcinoma arising in a mature cystic teratoma of the ovary in young patient with elevated carbohydrate antigen 19-9.

Squamous cell carcinoma is the most common type of malignant transformation in mature cystic teratomas. It mainly effects postmenopausal women but is rarely seen in young patients. Carbohydrate antigen 19-9 (CA19-9) tumor marker is a high-molecular-weight glycoprotein, frequently elevated in gastrointestinal adenocarcinomas. CA19-9 levels can increase in both dermoid cysts and in malignant transformation of dermoid cysts. Herein we report a case of squamous cell carcinoma originating from a dermoid cyst in a 31-year-old, gravida 0, para 0, single woman with high levels of CA19-9 and normal levels of CEA. Preoperative CA19-9 was 1000 U/ml (normal range below 27 U/ml). The patient underwent unilateral salpingo-oophorectomy, omentectomy, appendectomy, pelvic and paraaortic lymphadenectomy. After the pathologic analysis of the material, the patient was categorized as FIGO Stage IIa due to metastasis to the left tube. She received six cycles of cisplatin and paclitaxel at 21-day intervals. The postoperative first day, second month, and sixth month CA19-9 values were 602 U/ml, 33.5 U/ml and 22.3 U/ml, respectively. She is now doing well without recurrence of disease six months after the surgery. Squamous cell carcinomas originating from dermoid cysts are rare tumors especially seen in elderly patients with high levels of tumor markers (like CEA, SCCA). Every case may not have the same characteristics and management should be individualized.

CA 19-9 can be a useful tumor marker in ovarian dermoid cysts.

PURPOSE: To evaluate the importance of CA-19-9 as an aiding tool in the diagnosis of mature cystic teratomas. METHODS: We found 43 cases of dermoid cysts that were operated on at our clinic, and whose tumor marker CA 19-9 was studied before the operation. RESULTS: Thirty-seven of 43 patients (86%) had elevated CA 19-9 levels. Using Pearson's correlation, the average diameter and weight of the tumor had a strong possitive correlation with the level of CA 19-9 (p < 0.01). Thirty-one out of the 43 (72%) cases had a dermoid cyst in the right ovary, nine (20.9%) in the left ovary, and three (7%) had bilateral dermoid cysts. Right predominancy was very high among the patients (p < 0.05). CONCLUSION: We suggest that besides ultrasonography and computed tomography, serum CA 19-9 level could be a useful tumor marker in the diagnosis of dermoid cysts.

Peritoneal fluids from patients with certain gynecologic tumor contain elevated levels of bioactive lysophospholipase D activity.

Levels of lysophosphatidic acid (LPA), an important phospholipid mediator, in serum and ascitic fluid from ovarian cancer patients were shown to be higher than those from healthy women and from patients with other type of cancer, respectively. Although LPA in human serum seems mainly to be generated by lysophospholipase D (lysoPLD), the source and pathway for LPA in the ascitic fluid remain still obscure. In this study, we examined whether lysoPLD activity producing bioactive LPA in human peritoneal fluid was significantly elevated under pathological statuses. Lysophospholipase D activity in human peritoneal fluids was measured by quantifying choline released from exogenous lysophosphatidylcholine on their incubation at 37 degrees C. We also compared the activity of lysoPLD in sera from patients with different gynecologic diseases. We found relatively high lysoPLD activity in peritoneal fluids from patients with ovarian cancer, dermoid cyst or mucinous cystadenoma, whereas there were no significant differences in the serum lysoPLD activity among clinical groups and healthy subjects. The lysoPLD in the peritoneal fluid was found to have similar substrate specificity and metal ion requirement to those of serum lysoPLD, that has been identified as autotaxin, a tumor cell-motility stimulating protein. Our results suggest that increased lysoPLD activity in peritoneal fluid from patients with certain gynecologic tumors might be relevant to its potential of tumor progression.

Persistent elevation of CA 19-9 levels in a patient with an extended retroperitoneal dermoid.

CA 19-9 has been established as sensitive tumor marker in a variety of malignant diseases, especially in carcinomas of the exocrine pancreas. The specificity of CA 19-9 is limited by many benign diseases causing abnormal values. A case of a patient with a retroperitoneal dermoid showing high serum levels of CA 19-9 will be described. The patient suffered from a large retroperitoneal mass (7 x 6 x 9 cm). A CT-guided fine needle biopsy of the tumor was performed and the histology was compatible with a benign dermoid. The CT-scans of the abdomen showed also the typical features of a dermoid. Tumor markers and lab counts stayed in the normal range except CA 19-9, which was constantly elevated with value between 131 and 329 U/ml. A benign or malignant disorder was excluded. An immunoscintigraphy and a SPECT with a I-131-labeled monoclonal antibody against CA 19-9 revealed the retroperitoneal mass as the source of the CA 19-9 elevation. This is the first case described in English scientific literature of a retroperitoneal dermoid (benign teratoma) as a source of a persistently elevated level of CA 19-9.

Virilizing ovarian dermoid cyst with peripheral steroid cells. A case study with immunohistochemical study of steroidogenesis.

A case of virilizing ovarian dermoid cyst with peripheral steroid cells and virilization is reported in a 62-year-old woman. The level of testosterone dropped to normal after oophorectomy. The cyst wall was bordered by a discontinuous band of steroid cells focally accompanied by smooth muscle cells. Immunohistochemically, the steroid cells were enzymatically active and displayed a profile similar to the internal theca cells of ovarian follicles. These steroid cells were most probably modified stromal cells associated with smooth muscle metaplasia of the ovarian stroma.

Soluble adhesion molecules in serum and cyst fluid from patients with cystic tumours of the ovary.

The potential of the soluble forms of the adhesion molecules ICAM-1 (sICAM-1), CD44std (sCD44std) and E-cadherin (sE-cadherin) was tested for the diagnosis of benign and malignant cystic epithelial tumours of the ovary. Concentrations of sICAM-1, sCD44 std and sE-cadherin were measured by enzyme-linked immunosorbent assay (ELISA) in the serum and cyst fluid obtained from 23 patients with luteal cysts, 29 with cystadenomas, nine with dermoid cysts, five with borderline tumours and 11 with carcinomas. Serum concentrations of sICAM-1, but not of sCD44std and sE-cadherin, were constantly elevated compared with normal controls. Cyst fluid concentrations of sICAM-1, sCD44std and sE-cadherin were elevated in borderline and malignant tumours compared with cystadenomas (P = 0.034, 0.006 and 0.001, respectively). In conclusion, our results suggest that serum concentrations of adhesion molecules have no diagnostic value in ovarian tumours, whereas cyst fluid concentrations may facilitate distinction between benign lesions and borderline or malignant tumours.

Familial hypercholesterolemia and intracavernous venous spilling of cholesterol in a child with large suprasellar dermoid cyst. Case report.

A unique case of suprasellar dermoid cyst and familial hypercholesterolemia in a child is reported. Such an association, which strengthens the congenital hypothesis of dermoid cysts, could be a manifestation of a complex dyslipidemic inherited syndrome, at least in children. Surgical removal of the dermoid cyst which spilled cholesterol into the cavernous and intercavernous sinuses, allowed reduction of cholesterol levels in the postoperative period.

[Treatment using laparoscopic surgery of dermoid cysts. Apropos of a series of 33 cysts]. / Traitement par coeliochirurgie des kystes dermoïdes. A propos d'une série de 33 kystes.

Laparoscopy is a surgical method which is now widely used for the treatment of ovarian cysts. The authors wish to present their experience concerning the management and treatment of dermoid cysts or dysembryomas since these lesions embody all the problems persisting in this field. They report 33 cases exclusively treated by laparoscopic surgery in a series of 407 adnexal cysts treated over a 4-year period (April 1988 to April 1992). As regards diagnosis, the principal difficulty is not to miss the odd malignant lesion (1 to 2%) encountered mainly in older women. Technically, the most delicate stage in the operation is extraction of the cyst, especially when it is large and has an important solid component. Provided all precautions are taken in the diagnosis and the operative technique, this new method seems to be quite acceptable as it enables women who carry these lesions, which require excision, to benefit from the well-known advantages of laparoscopic surgery.

46,XX gonadal dysgenesis with epibulbar dermoid.

Pure gonadal dysgenesis with 46,XX genotype is a rare abnormality with unknown etiology. Although sensorineural deafness has been described with 46,XX gonadal dysgenesis, the majority of reported cases of 46,XX gonadal dysgenesis have no associated physical abnormalities. We report a patient with 46,XX gonadal dysgenesis associated with epibulbar dermoids and preauricular skin tags, the classic ocular and skin manifestations of Goldenhar sequence (oculoauricular vertebral dysplasia). We propose that our patient may represent a new and previously unreported syndrome.