Ovarian remnant syndrome with paraintestinal ovarian serous cystadenofibroma arose 30 years after bilateral salpingo-oophorectomy: A case report.

RATIONALE: Ovarian cystadenofibroma is a relatively rare benign ovarian tumor. Ovarian remnant syndrome (ORS) is a rare complication of bilateral salpingo-oophorectomy (BSO). We report a rare case of ORS with paraintestinal ovarian serous cystadenofibroma that developed 30 years after total abdominal hysterectomy and BSO in a 73-year-old woman. PATIENT CONCERNS: A 73-year-old woman complained of long-term lower abdominal discomfort. DIAGNOSIS: She was diagnosed with a cystic lesion in the lower abdomen on transabdominal ultrasonography. Further diagnostic imaging and laboratory tests could not exclude a diagnosis of malignancy. INTERVENTIONS: The patient underwent laparoendoscopic single-site surgery. We found one cystic lesion 5 cm in size with multiple septa that was adhered to the small bowel. We consulted a general surgeon for tumor resection. Dissection was performed and the specimen was then removed from the umbilical wound. OUTCOMES: Histopathological examination revealed an ovarian serous cystadenofibroma. The postoperative recovery was uneventful. LESSONS: Patients with lower abdominal pain after a previous hysterectomy and BSO should be examined with transabdominal sonography for ORS.

A case of ovarian serous cystadenofibroma with scattered lesions in pelvic cavity, like malignant disseminations.

Ovarian serous cystadenofibroma is a relatively rare subtype of serous cystadenoma classified as ovarian benign epithelial tumor. We report a rare case of ovarian serous cystadenofibroma with scattered lesions in pelvic cavity, like malignant disseminations. The patient was 22 years old, gravida 0, para 0. In the laparoscopic surgery, numerous hard yellowish-white solid masses of various sizes were present in the bilateral ovaries. Grossly similar masses were scattered in the fimbria of the fallopian tubes, peritoneum, and great omentum. Because the intraoperative rapid histological diagnosis was benign tumor, surgery was completed for only tumor excision. Postoperative histopathological diagnosis is serous cystadenofibroma. Similar pathological findings were noted in the scattered lesions in the peritoneum and great omentum. No malignant or borderline malignant finding was observed. Because of a benign disease, careful treatment taking fertility preservation into consideration is necessary, especially for young patients.

Intrauterine device found in an ovarian tumor: A case report.

RATIONALE: Intrauterine devices (IUDs) are one of the most common and effective methods of contraception worldwide. Migration of an IUD to an extrauterine site is a rare complication. The aim of this study was to report an extremely rare case in which an IUD was found in an ovarian tumor. PATIENT CONCERNS: A 63-year-old Chinese woman presented with vaginal bleeding and lower abdominal pain during hospitalization due to pneumonia. Preoperative imaging showed bilateral cystic masses in the adnexal region, and ring hyperdensity was found in the right ovarian mass. Endometrial thickening and multiple uterine leiomyomas were found on ultrasonography. Hysteroscopy showed partial septate uterus and a small endometrial polyp. DIAGNOSIS: Bilateral ovarian cystadenomas with perforation of the IUD into the right ovarian tumor were considered based on preoperative imaging and the patient's medical history. Furthermore, early endometrial carcinoma was suspected. INTERVENTIONS: The patient underwent hysterectomy, bilateral salpingo-oophorectomy, and omentectomy. A stainless steel ring IUD was confirmed within the right ovarian tumor during the operation. OUTCOMES: The pathology results demonstrated bilateral ovarian serous cystadenofibromas with focal epithelial proliferation and endometrial atypical hyperplasia with malignant transformation. The patient has been followed up for 7 months, and there has been no recurrence at present. LESSONS: The presence of an IUD within an ovarian tumor is extremely rare. This is the second reported case in the English literature describing an extrauterine IUD within an ovarian tumor. The correlation between ovarian cancer tumorigenesis and IUD translocation is unclear and requires further investigation.

Oncologic and fertility impact of surgical approach for borderline ovarian tumours treated with fertility sparing surgery.

BACKGROUND: Even if borderline ovarian tumours (BOTs) in young women treated with fertility-sparing treatment (FST) have an excellent outcome, the type of surgery might affect relapse and fertility. We investigated the effect of surgical approach (open surgery vs. laparoscopy) and type of surgery (salpingo-oophorectomy [SO] vs. cystectomy [Cy]) on oncologic and fertility outcomes in patients with BOT. PATIENTS AND METHODS: Patients with BOT treated at San Gerardo Hospital, Monza, with FST in 1978-2013 period were included. Cox models, stratified by decade of surgery, were used to investigate the association between time to first recurrence or conception and clinical factors. RESULTS: Among 535 patients included, 271 underwent unilateral SO and 264 underwent Cy. Median follow-up was 13.5 years. Ten-year (10-yr) recurrence rate was 23% (95% confidence interval [CI]: 18-29%) for SO and 31% (95% CI: 24-38%) for Cy group (P = 0.10) in patients with unilateral tumour, whereas it was 62% (95% CI: 44-79%) and 72% (95% CI: 59-84%), respectively, (P = 0.35) in patients with bilateral tumour. Multivariable analysis showed no association between recurrence and surgical approach (P = 0.44), type of surgery (P = 0.06) and a negative association with advanced stage (hazard ratio [HR] = 3.18; 95% CI: 2.11-4.78; P < 0.001) and bilateral tumours (HR = 2.48; 95% CI: 1.78-3.47; P < 0.001). Among 252 patients (47.1%) with pregnancy desire, multivariable analysis showed no association between conception success and the type of surgery, surgical approach, histology and tumour laterality. Fertility after surgery was positively associated with prior pregnancy (HR = 1.68; 95% CI: 1.17-2.41; P = 0.005) and negatively associated with the number of surgical procedures (HR = 0.62; 95% CI: 0.53-0.73; P < 0.001). CONCLUSIONS: The type of surgical procedures did not influence recurrence rate or fertility. However, additional surgical procedures decreased the fertility potential. These data can support clinicians in tailoring the best strategy for FST in young patients with BOT.

Massive Ovarian Growth in a Woman With Severe Insulin-Resistant Polycystic Ovary Syndrome Receiving GnRH Analogue.

CONTEXT: Ovarian hyperandrogenism from polycystic ovary syndrome (PCOS) and hyperinsulinemia from insulin resistance are modulators of ovarian follicle development. We report on a woman with PCOS and hyperandrogenism and severe insulin resistance from metabolic syndrome who received long-term GnRH analogue therapy preceding bilateral salpingo-oophorectomy for massive ovarian enlargement. Ovarian histological examination showed proliferating granulosa cells within antral follicles coexistent with serous cystadenofibromas, demonstrating a unique link between hyperinsulinemia and granulosa cell mitogenesis. CASE DESCRIPTION: A 30-year-old woman with PCOS with hyperandrogenism, severe insulin resistance from metabolic syndrome, and nonalcoholic steatohepatitis experienced abdominal pain from bilaterally enlarged ovaries. She had previously experienced a pulmonary embolism while taking oral contraceptives and hepatotoxicity from metformin and spironolactone therapies. Long-term GnRH analogue therapy to induce pituitary desensitization to GnRH successfully decreased gonadotropin-dependent steroidogenesis without improving insulin resistance. Despite GnRH analogue therapy, progressive ovarian enlargement in the presence of hyperinsulinemia from worsening metabolic function eventually required bilateral salpingo-oophorectomy for removal of massively enlarged ovaries. Histological examination showed both ovaries contained proliferating granulosa cells within antral follicles coexistent with serous cystadenofibromas. CONCLUSIONS: In women with PCOS and hyperinsulinemia from severe insulin resistance due to metabolic syndrome, granulosa cell proliferation within antral follicles can occur despite long-term GnRH analogue therapy, implicating hyperinsulinemia as a granulosa cell mitogen in the absence of gonadotropin-dependent ovarian function.

Pitfalls of Frozen Section in Gynecological Pathology: A Case of Ovarian Serous Surface Papillary Adenofibroma Imitating Malignancy.

Serous cystadenofibromas are uncommon benign ovarian lesions, consisting of both fibrous and epithelial components, that are usually cystic but may contain solid or papillary architecture that can be confused with a malignancy on imaging. Papillary architecture seen on frozen section may also falsely steer the pathologist in the direction of a diagnosis of a borderline serous tumor. Overcalling the lesion may lead to more aggressive surgery than necessary, so extensive tissue sampling and consideration of this entity is important in possibly avoiding this mistake.

Unusual case of anti-N-methyl-D-aspartic acid-receptor (NMDA-R) encephalitis and autoimmune polyglandular syndrome (APS).

Anti-N-methyl-D-aspartic acid-receptor (NMDA-R) encephalitis is a novel disease discovered within the past 10 years. It is an autoimmune disease (AD) that has been associated with other ADs, such as Graves' disease. However, association with autoimmune polyglandular syndromes (APS) has not been previously described. A 58-year-old woman presented with altered mental status and an 8-month history of weight loss, apathy and somnolence. Laboratory evaluation confirmed Graves' disease with thyrotoxicosis and type 1 diabetes mellitus. Despite treatment, she continued to have a fluctuating mental status. Further diagnostic evaluation included an abdominal MRI that showed a cystic lobular left adnexal mass. Serum anti-NMDA-R antibodies were positive, raising concern for NMDA-R encephalitis. Bilateral salpingo-oophorectomy was performed, with pathology consistent with cystadenofibroma. She had a favourable recovery with marked clinical improvement. Anti-NMDA-R antibodies were negative 2 months following surgery. The concomitant occurrence of APS and anti-NMDA-R encephalitis suggests a shared mechanism of autoimmune pathophysiology.

Short-term Outcomes and Pregnancy Rate After Laparoscopic Fertility-Sparing Surgery for Borderline Ovarian Tumors: A Single-Institute Experience.

OBJECTIVE: We investigated the short-term outcomes and pregnancy rate after a laparoscopic approach to fertility preservation in patients with borderline ovarian tumors (BOTs). METHODS: Clinic-pathologic variants of patients with BOTs who underwent conservative surgery at the Tianjin Central Hospital of Obstetrics and Gynecology between January 2009 and July 2015 were retrospectively analyzed. RESULTS: Among 211 patients with BOTs, 74 (35.1%) received conservative surgery (44 cases using a laparoscopic approach and 30 cases using a laparotomy approach). The mean age of the laparotomy group was significantly younger than that of the laparoscopic group (P = 0.024). The maximal longitude of the tumor in the laparotomy group was significantly longer than that in the laparoscopic group (P < 0.001). The number of incomplete surgery cases in the laparoscopic group was significantly greater than that in the laparotomy group (P < 0.001). The 2 groups showed no significant differences in gravidity and parity before surgery, abnormality of serum tumor makers, tumor lateralities, ascites, histology, duration of follow-up, pregnancy rate after surgery, or postoperative recurrence. Total recurrent rate was 6.7% (5/74). Two cases in laparotomy group and 3 cases in laparoscopic group relapsed respectively. There was no significant difference of recurrent rate between the 2 groups. The total pregnant rate was 33.8% (25/74). Nine patients (30%) in the laparotomy group and 16 patients (36.4%) in the laparoscopic group became pregnant during follow-up respectively. There were no significant differences in the postoperative durations of pregnancy, pregnancy type, age at pregnancy, tumor lateralities, ascites, or type of pathology between 2 groups. The pregnancy rate of incomplete surgery cases in laparoscopic group was significantly higher than that of laparotomy group (P = 0.011). No recurrence occurred among the pregnant cases. CONCLUSIONS: A comprehensive laparoscopic surgery was not performed in incomplete surgery patients undergoing complete exploration. Good short-term outcomes and pregnancy were observed in patients receiving conservative laparoscopic surgery for BOTs, especially in patients receiving incomplete conservative laparoscopic surgery.

Atypical Proliferative (Borderline) Serous Tumor in the Brain: A Case Report.

A 59-year-old woman with a remote history of invasive ductal carcinoma of the breast was found on a follow-up computed tomography scan of her brain to have a 1-cm lesion in the right frontal lobe in 2008. In the ensuing years, before her current admission, multiple imaging studies of the brain revealed that the lesion was stable and it was, therefore, interpreted as a small area of encephalomalacia related to a thrombosed cortical vein, a cavernoma, or treated metastatic breast cancer. In 2013, the patient underwent a bilateral salpingo-oophorectomy for ovarian tumors that were diagnosed as bilateral serous cystadenofibromas. A partial omentectomy showed no evidence of implants. In June 2016, the brain lesion was completely excised and diagnosed as an atypical proliferative (borderline) serous tumor. Immunohistochemical staining demonstrated that the tumor cells were immunoreactive for Pax8, WT-1, ER, and CK-7 and negative for Gata-3, PR, TTF-1, CDX-2, Napsin A, and CK-20, which was consistent with that diagnosis. We present a brief review of possible mechanisms to account for this unusual presentation and speculate that the most likely one is exfoliation of fallopian tube epithelial cells into the peritoneal cavity, which then gain access to lymphatics resulting in cells implanting in the brain and subsequently progressing to an atypical proliferative (borderline) serous tumor.

Clinical Predictors of Recurrence and Prognostic Value of Lymph Node Involvement in the Serous Borderline Ovarian Tumor.

AIM: This study was aimed to evaluate the risk factors of recurrence and the value of nodal involvement in patients with serous borderline ovarian tumors (SBOT). METHODS: Two hundred twenty-five patients who underwent surgery and were diagnosed with SBOT were retrospectively studied. Univariate and multivariate analyses were used to assess the risk factors for recurrence. Patients' clinical pathologic characteristics were compared between the patients who presented lymph node involvement and those who did not. The significant values of lymph condition influencing 5-year disease-free survival were also evaluated by statistical analysis. RESULTS: Both univariate and multivariate analyses showed that risk factors for recurrence were micropapillary (P = 0.021), fertility-preserving surgery (P = 0.014), and laparoscopic approach (P = 0.009). Of these 112 patients on whom lymphadenectomy was performed, 17 cases showed lymph node positive, whereas the remaining 95 patients did not. Significant differences in terms of lymph node numbers (P < 0.0001), invasive implant (P = 0.022), and International Federation of Gynecology and Obstetrics staging (P < 0.0001) were observed between the 2 groups of lymphatic node involved or not. Kaplan-Meier curves of 5-year disease-free survival revealed that there were no significant differences either between groups of lymphatic node involved or not (P = 0.778) and groups of removed nodes whether more than 10 or not (P = 0.549). CONCLUSIONS: Micropapillary, fertility-preserving, and laparoscopic approach were factors significantly affecting the recurrence of SBOT by both univariate and multivariate analysis. Lymph node metastasis did not seem to be correlated to a worse prognosis of SBOT.

Three-dimensional ultrasound and HDlive in tubal serous cystadenofibroma: a case report and literature review.

Cystadenofibromas of the fallopian tubes are very rare benign tumors and very few cases have been reported in the literature worldwide. Usually, the tumor is asymptomatic, and for almost all cases reported, the tumors were incidentally discovered during surgery for other genital pathology. We report the case of a 30-year-old woman with a serous cystadenofibroma of the fallopian tube, presenting with chronic abdominal pain and secondary infertility. The diagnosis of tubal tumor was formulated before surgery and confirmed during laparoscopic surgery. Both 3D ultrasound and HDlive were useful tools for the diagnosis. The ultrasound diagnosis was helpful in planning appropriate surgical management.

The feasibility of fertility-sparing surgery in treating advanced-stage borderline ovarian tumors: A meta-analysis.

OBJECTIVE: To evaluate the feasibility of fertility-sparing surgery in treating advanced-stage borderline ovarian tumors (BOTs). MATERIALS AND METHODS: The databases of PubMed, Cochrane Library, EMbase, Web of Science, Chinese National Knowledge Infrastructure (CNKI), Weipu (Chinese), and Wanfang (Chinese) were searched using the keywords "advanced-stage borderline ovarian tumors", "fertility-sparing surgery", "conservative surgery", and "borderline ovarian tumor" to collect the clinical controlled trails (CCTs) regarding fertility-sparing surgery for the treatment of advanced-stage BOT. The references of those CCTs were also searched manually. Data extraction and quality assessment were done using Review manager Version 5.1 and R software Version 2.11.1. RESULTS: Four studies involving 74 patients were included. The results of meta-analysis showed that: (1) compared with radical surgery, the recurrence of the fertility-sparing surgery during the follow time is higher with significant difference [odds ratio (OR)=3.87, 95% confidence interval (CI) (1.20,12.44), p=0.02]; (2) the difference of survival rate between the two groups was not significant [5-year survival: OR=0.85, 95%CI (0.03, 23.82), p=0.92; 7-year survival: OR=0.80, 95%CI (0.08, 8.41), p=0.85]; and (3) concerning fertility results in fertility-sparing surgery, 18 patients tried to become pregnant, 15 pregnancies were achieved in the 11 patients, 11 full-term deliveries. CONCLUSION: The rate of ovarian recurrence in patients who underwent conservative treatment was higher than in patients with radical treatment, but it did not affect patient survival; fertility-sparing surgery could be induced to preserve the fertility potential of young patients.

Serous papillary cystadenofibroma of the fallopian tube: A case report and short review of literature.

Serous papillary cystadenofibromas (SPCAFs) of the fallopian tube are very rare benign tumors of the female genital tract. They are usually asymptomatic and are found incidentally. Until now, only 18 cases of this tumor have been reported in the world literature. We report a case of SPCAF of the left fallopian tube in a 30-year-old female who presented with a large abdominal mass and pain. On computed tomography, a diagnosis of ovarian neoplasm was given. However, during surgery the tumor was found to arise from the fallopian tube and was treated with tubal cystectomy with sparing of the ovary. We present this unique case on account of its rarity, unusual presentation, and huge size along with a short review of literature.

Simple descriptors and simple rules of the International Ovarian Tumor Analysis (IOTA) Group: a prospective study of combined use for the description of adnexal masses.

OBJECTIVE: External validation of the IOTA group's three-step diagnostic model (Strategy 1) and comparison with assessment by an expert sonographer (Strategy 2). STUDY DESIGN: Prospective study in patients with persistent adnexal masses, in which an inexperienced sonographer performed transvaginal ultrasound applying simple descriptors (SD) and rules (SR) for classifying as benign or malignant. Any non-classifiable mass was then submitted to an expert examiner for subjective assessment (SA). RESULTS: Eighty-one patients (mean age, 43; 27.2% postmenopausal) were included in this prospective study. Surgery was performed for 30 (8 malignant and 22 benign) masses; 51 masses were considered as benign and managed expectantly (they were assumed to be benign for statistical purposes). Diagnostic performance for Strategy 1 (SD+SR+SA) was sensitivity (SN): 87.5% (7/8, 95% CI, 47.3-99.7%) and specificity (SP): 100% (73/73, 95% CI, 95.1-100%). For Strategy 2 (SA only) it was SN 87.5% (7/8, 95% CI, 47.3-99.7%), SP 98.6% (72/73, 95% CI, 92.6-100%). CONCLUSIONS: The three-step diagnostic strategy designed by the IOTA group for adnexal masses has a diagnostic performance comparable to that of subjective expert assessment and could be used as a triage method by nonexpert sonographers.

Pregnancy after laparoscopic bilateral partial ovarian decortication for stage IC borderline ovarian tumour.

BACKGROUND: Fertility preservation techniques are a growing area of research as more women in the reproductive age group develop gynaecologic cancers. We report here a novel technique of fertility preservation used in the treatment of a patient with borderline ovarian tumour. CASE: A 29-year-old woman with stage I borderline ovarian tumour was referred to our tertiary level hospital. She had a history of infertility and requested fertility preservation be considered in treatment decisions. We performed bilateral laparoscopic partial decortication of the ovaries, and the patient successfully conceived spontaneously following the procedure. CONCLUSION: Fertility-preserving surgery should be an option for young women with borderline ovarian tumours who wish to retain fertility. Removing abnormal ovarian tissue may restore fertility. The laparoscopic approach is safe and feasible for these patients.

Contexte : Les techniques de préservation de la fertilité constituent un champ de recherche en évolution, de plus en plus de femmes en âge de procréer étant atteintes de cancers gynécologiques. Nous nous penchons sur une technique novatrice de préservation de la fertilité utilisée dans le cadre de la prise en charge d'une patiente présentant une tumeur ovarienne à la limite de la malignité. Cas : Une femme de 29 ans présentant une tumeur ovarienne à la limite de la malignité de stade I a été orientée vers notre hôpital de niveau tertiaire. Elle présentait des antécédents d'infertilité et souhaitait que la préservation de la fertilité soit prise en considération dans le cadre du processus de prise de décision quant au traitement. Nous avons pratiqué une décortication laparoscopique bilatérale partielle des ovaires et la patiente a été en mesure de connaître une grossesse spontanée à la suite de l'intervention. Conclusion : La chirurgie visant à préserver la fertilité devrait constituer une solution possible pour les jeunes femmes présentant une tumeur ovarienne à la limite de la malignité qui souhaitent demeurer fertiles. L'excision de tissus ovariens anormaux pourrait permettre de rétablir la fertilité. L'approche laparoscopique est sûre et praticable dans le cas de ces patientes.

External multicentre validation of a nomogram predicting the risk of relapse in patients with borderline ovarian tumours.

BACKGROUND: The Obermair nomogram was recently developed to predict the risk of relapse in patients with borderline ovarian tumours (BOTs) based on five readily available clinical, biological, and pathological characteristics. We set out to externally validate and assess its robustness using a multi-institutional BOT database. METHODS: All consecutive patients treated for BOTs in the two participating centres between January 1980 and December 2008 and who had all the nomogram variables documented were identified for analysis. RESULTS: Three hundred and fourteen eligible patients were identified and used for external validation analysis. The median follow-up and initial relapse time were 46.43 (range: 0.1-360) and 66.64 (range: 8-77) months, respectively. The nomogram concordance index was 0.54 (95% CI, 0.52-0.56). The correspondence between the actual relapse and the nomogram predictions suggests a limited calibration of the nomogram in the validation cohort. CONCLUSION: This external validation study of the Obermair nomogram showed limitations in its generalisability to a new and independent patient population.

Large functional benign endometroid cystadenofibroma of the ovary leading to endometrial cystic glandular hyperplasia and postmenopausal bleeding.

Benign endometroid cystadenofibroma of the ovary is a rare tumour with few reported cases. The association of endometroid cystadenofibroma with endometrial cystic glandular hyperplasia has not been previously reported to authors' knowledge. We are reporting a 75-year-old postmenopausal woman who presented with a large abdominopelvic mass corresponding to 30 weeks size gravid uterus and postmenopausal bleeding. She has a well-oestrogenised vagina. Ultrasound pelvis revealed a large cystic mass extending from pelvis to epigastrium with no solid component and few incomplete septations and no internal echoes. MRI findings showed mainly cystic component with few moderately enhancing, fine, incomplete septa. Endometrial aspiration reported histopathology of cystic glandular hyperplasia without atypia. Serum oestrogen level reported to be high (210 pg/mL). Hysterectomy with bilateral salpingo-oophorectomy was performed. Histopathology of the specimen revealed benign endometroid cystadenofibroma. She remained asymptomatic and disease free during her 6-month follow-up.