FUKUSHIMA JOURNAL OF MEDICAL SCIENCE

Malignant transformation arising from mature cystic teratoma of the ovary presenting as ovarian torsion: a case report and literature review

Ovarian torsion is an acute gynecological condition. Torsion is more likely to occur with benign rather than malignant tumors. Mature cystic teratoma of the ovary (MCTO) is frequent in women of reproductive age; however, the incidence of malignant transformation is approximately 2%. We report a case of malignant transformation of MCTO presenting as ovarian tumor torsion. A 51-year-old premenopausal woman was diagnosed with mature cystic teratoma in the left ovary 7 years ago. The patient visited our hospital because she had been experiencing of pain in left lower abdomen for the past two days. She was diagnosed with ovarian tumor torsion and underwent emergency surgery. The left ovarian tumor was twisted, and left salpingo-oophorectomy was performed. Histopathological examination revealed squamous cell carcinoma arising from the MCTO. We carefully followed the patients without performing staging laparotomy. On postoperative day 112, multiple lymph node metastases in the pelvic and para-aortic areas were found by positron-emission tomography and computed tomography. After referral to a university hospital, total hysterectomy, right salpingo-oophorectomy, partial omentectomy, and pelvic and paraaortic lymphadenectomy were performed. Metastases of squamous cell carcinoma were confirmed in the pelvic and para-aortic lymph nodes. Six courses of adjuvant chemotherapy with paclitaxel and carboplatin were given following radical surgery to prevent the recurrence of malignant transformation of MCTO. No recurrence of the disease has been observed during 2 years of follow-up. When physicians diagnose large ovarian tumor torsion cases, preoperative examinations should be performed, with the possibility of malignancy in mind.

Successful laparoscopic resection of virilizing ovarian steroid cell tumor, not otherwise specified, in a 22-year-old woman: a case report and evaluation of the steroidogenic pathway

Ovarian steroid cell tumor (SCT) is a rare tumor with steroid-producing ability. We report a 22-year-old woman with secondary amenorrhea and hirsutism caused by an ovarian SCT-not otherwise specified (NOS), who underwent successfully laparoscopic resection of the tumor. A 22-year-old null gravida woman presented to a hospital, having amenorrhea for 18 months and increasing facial hair. Physical examination revealed obesity (body mass index, 37.3 kg/m

Differentiation of ovarian serous carcinoma from ovarian clear cell carcinoma using a 10-gene signature selected by comprehensive gene expression analysis

Ovarian serous carcinoma (OSC) and ovarian clear cell carcinoma (OCCC) are two major histological types of epithelial ovarian carcinoma (EOC), each with different biological features and clinical behaviors. Although immunostaining is commonly used for differential diagnosis between OSC and OCCC, correct identification of EOC with mixed-type histology is sometimes a diagnostic challenge. The aim of the present study was to explore candidate genes as potential diagnostic biomarkers that distinguish OSC from OCCC. A total of 57 surgical specimens were obtained from EOC patients who had previously undergone primary debulking surgery. Total RNAs were extracted from fresh-frozen tissues of EOC patients, and were used for comprehensive gene expression analysis using DNA microarray technology. Ten candidate genes, FXYD2, TMEM101, GABARAPL1, ARG2, GLRX, RBPMS, GDF15, PPP1R3B, TOB1, and GSTM3 were up-regulated in OCCC compared to OSC. All EOC patients were divided into two groups according to hierarchical clustering using a 10-gene signature. Our data suggest that the 10 candidate genes would be an excellent marker for distinguishing OSC from OCCC. Furthermore, the molecular signatures of the 10 genes may enlighten us on the differences in carcinogenesis, and provide a theoretical basis for OCCC's resistance to chemotherapy in the future.

Successful management of pelvic recurrence of MSI-High endometrial cancer by total pelvic exenteration followed by administration of pembrolizumab:A case report

Surgery can be curative treatment for pelvic locoregional recurrence of endometrial cancer; however, a cure is contingent on complete resection. Here, we report the case of a patient in whom recurrent endometrial tumor remained in the pelvis after resection; long-term control was achieved with postoperative administration of pembrolizumab.The patient had recurrent endometrial cancer of stage IA and was treated with chemotherapy and radiation, but tumor persisted in the pelvic cavity. We therefore attempted total pelvic exenteration, but the tumor was adherent to the pelvic wall and complete resection could not be achieved. However, postoperative administration of pembrolizumab controlled the residual tumor for more than two years without regrowth. We believe that since the resected tumor was MSI-High, the residual tumor responded well to pembrolizumab. It is not known whether cytoreductive surgery contributes to a long-term response to pembrolizumab, but at least in our patient, pembrolizumab appeared to be a very effective drug therapy for MSI-High endometrial cancer that was refractory to chemotherapy and radiotherapy.

The utility of MRI for the preoperative differential diagnosis of uterine sarcoma and leiomyoma: a single-center study

Although uterine sarcoma is a rare disease, its prognosis is extremely poor;thus, it is important to differentiate it from uterine leiomyoma.　In this retrospective study, we examined the association between preoperative MRI findings and postoperative pathology results in 170 patients with uterine tumors who underwent preoperative MRI examination at Fukushima Red Cross Hospital.　In 4 cases of sarcoma / smooth muscle tumor of unknown malignant potential (STUMP), abnormal findings were found at a high frequency with T1-weighted imaging (T1WI) (75%), T2-weighted imaging (T2WI), diffusion-weighted imaging (DWI), and contrast enhancement (CE) (100%).　In cases of ordinary leiomyoma, on the other hand, abnormal findings were less frequent.　The rates of high DWI signal intensity for degenerated and cellular leiomyoma were 31% and 64%, respectively, and the CE-positive rates were 31% and 57%, respectively.　Apparent Diffusion Coefficient (ADC) values appeared to be useful in differentiating degenerated leiomyoma from sarcoma.　The relatively characteristic findings of uterine sarcoma on MRI images may overlap with those of degenerated leiomyoma and cellular leiomyoma, making it difficult to diagnose sarcoma on imaging alone.　However, findings that distinguish sarcoma from ordinary, degenerated, and cellular leiomyoma cases are worthy of attention, to avoid overlooking sarcoma.

Four cases of pseudomyxoma peritonei with ovarian tumors at our hospital

We describe four cases of pseudomyxoma peritonei (PMP) that were diagnosed and treated at our hospital.Case 1: A 26-year-old woman with a large multicystic ovarian tumor and massive ascites was diagnosed with PMP originating from a borderline mucinous ovarian tumor. She underwent fertility-preserving staging laparotomy and was treated with three courses of intraperitoneal chemotherapy. There has been no recurrence in the 15 years since her first operation. Case 2: A 72-year-old woman with a giant ovarian tumor and massive ascites was diagnosed with PMP originating from low-grade appendiceal mucinous neoplasm (LAMN). After laparotomy, the patient was managed conservatively because she did not want aggressive treatment. She has remained asymptomatic with a small amount of ascites for 3 years. Case 3: A 82-year-old woman with ovarian tumors, massive ascites, and suspected PMP underwent emergency laparotomy due to appendiceal perforation and pan-peritonitis. She was diagnosed with PMP originating from LAMN. She has remained asymptomatic with a small amount of ascites for 2 years. Case 4: A 42-year-old woman with multicystic ovarian tumors and massive ascites underwent laparotomy. She was diagnosed with PMP originating from LAMN. Since multidisciplinary treatment was indicated and desired, the patient was referred to a specialized facility where cytoreductive surgery and hyperthermic intraperitoneal chemotherapy was performed. The patient has done well since the treatment.Although most cases of PMP originate from mucinous tumors of the appendix, female patients with PMP often present with ovarian tumors and are commonly referred to gynecology clinics. It is therefore important for gynecologists to be familiar with PMP and to be able to diagnose it accurately and select the most suitable management including multidisciplinary treatments.

Ovarian strumal carcinoid: a case report

Carcinoid tumors of the ovary are rare tumors, histopathologically classified as monodermal teratomas and somatic-type tumors arising from dermoid cysts. Their malignancy varies from borderline to malignant. Carcinoid tumors can occur in young and elderly women, and are sometimes seen in mature teratoma, struma ovarii, or mucinous cystadenoma as a nodule or tumor. Strumal carcinoid and mucinous carcinoid present as special types of carcinoid tumors of the ovary. This report describes a 56-year-old woman who presented with a large pelvic mass on abdominal ultrasonography during a medical examination. The diameter of the pelvic tumor was approximately 11 cm and was suspected to be ovarian cancer. The values of CA125 and CEA were above their reference intervals on preoperative examination. Abdominal total hysterectomy and bilateral salpingo-oophorectomy were performed. Intraoperative frozen-section histopathology suggested a diagnosis of mucinous adenocarcinoma; therefore, partial omentectomy and pelvic lymphadenectomy were also performed. Permanent-section histopathology led to a final diagnosis of strumal carcinoid of the ovary, stage IA (FIGO 2014). Six years post-operation, the patient had no sign of recurrence.

Struma ovarii with massive ascites mimicking ovarian carcinoma treated with conservative laparoscopic surgery: a case report

Struma ovarii is a rare taratoma that accounts for 0.5-1% of all ovarian tumors. It is sometimes difficult to differentiate struma ovarii from ovarian carcinoma. We encountered a case of struma ovarii that was suspected to be malignant due to the accumulation of massive ascites and an elevated CA125 level. It was successfully treated with laparoscopic surgery.A 37-year-old nulliparous woman consulted a local physician with a chief complaint of abdominal distention. Computed tomography (CT) of the abdomen revealed a pelvic tumor with a large amount of ascites. She was referred to our department. Contrast-enhanced magnetic resonance imaging (MRI) and CT showed bilateral ovarian tumors with multicystic and solid components. CA125 level was markedly elevated. Two cytological examinations of ascites showed no malignant cells. Preoperatively, malignancy was strongly suspected, but considering the possibility of a benign ovarian tumor, laparoscopic surgery was scheduled. During laparoscopic surgery, 4,850 mL of ascites were aspirated, and the left adnexa was removed. Intraoperative rapid pathology suggested struma ovarii with no evidence of malignancy. Postoperative pathology showed mature teratoma and struma ovarii.Although struma ovarii is benign in 90-95% of cases, there have been scattered case reports in which suspected malignancy led to unnecessary or excessive surgery. We propose that appropriate preoperative imaging and accurate intraoperative rapid pathology can prevent excessive surgery, conservative or laparoscopic excisions should be considered.

A case of polypoid endometriosis mimicking advanced ovarian carcinoma with rapid growth, invasion, and dissemination

Polypoid endometriosis is a rare form of endometriosis characterized by polypoid masses that histologically often resemble endometrial polyps. We report a case of rapidly progressing polypoid endometriosis that was preoperatively assumed to be advanced ovarian cancer. A 46-year-old woman, para 0, underwent laparoscopic myomectomy and left adnexectomy for uterine fibroids and a left ovarian endometrial cyst after administration of gonadotropin releasing hormone (GnRH) agonist for 4 months. Eleven months postoperatively, rapid right ovarian enlargement occurred. CT and MRI (both contrast-enhanced) showed masses in the right adnexa, cecum, sigmoid colon, and omentum, and PET-CT demonstrated increased uptake, suggesting ovarian cancer and peritoneal dissemination. The patient later developed intestinal obstruction, and colonoscopy revealed multiple polypoid lesions in the sigmoid colon. The omental tumor and right adnexa were biopsied during exploratory laparotomy, and diagnosed as polypoid endometriosis with no malignancy by permanent pathology. The right adnexal tumor shrunk markedly after 4 months of GnRH antagonist treatment. Second laparotomy was then performed for right adnexal tumor resection and ileocecectomy. Pathological examination revealed polypoid endometriosis extending from the ovary to the cecal mucosa. The patient has been asymptomatic for over 1 year postoperatively. The sigmoid colon tumor shrunk but is still present.Polypoid endometriosis predominantly affects the ovaries, colon, peritoneum, and omentum of patients in their 40s and 50s. It is a benign disease but is often difficult to distinguish from malignancy preoperatively because it rapidly forms numerous solid lesions. Although polypoid endometriosis is rare, with no specific imaging findings, including it in a differential diagnosis may facilitate preoperative identification.

Two cases of low-grade appendiceal mucinous neoplasm (LAMN) mimicking ovarian tumors successfully treated with laparoscopic surgery

We report two cases of low-grade appendiceal mucinous neoplasm (LAMN) mimicking ovarian tumors that were treated with laparoscopic surgery. Case 1: A 57-year-old woman was referred to our hospital because of a pelvic mass noted during physical examination. Imaging revealed an oval, cystic, 81×39-mm mass diagnosed as a right ovarian tumor, and surgery was performed. Intraoperative findings revealed an appendiceal tumor, and laparoscopic appendectomy was conducted. The pathological diagnosis was LAMN.Case 2: A 76-year-old woman was referred to our hospital because of a pelvic mass noted during physical examination. Imaging revealed an oval, cystic, 36×23-mm mass in the pelvic cavity, which was suspected to be a right ovarian tumor. Since it was slightly displaced to the cephalic side, surgery was performed with the possibility of a gastrointestinal tumor in mind. Intraoperative findings revealed an appendiceal tumor, and a laparoscopic appendectomy was conducted. The pathological diagnosis was LAMN.LAMN is a rare lesion with a risk of serious complications if the appendiceal wall ruptures and the tumor extends beyond the appendix. Although its preoperative diagnosis is challenging, it is important that gynecologists differentiate it from adnexal tumors.

Hydropic leiomyoma-like ovarian tumor: a case report

Uterine leiomyomas, benign tumors common in reproductive-aged women, can display rare variants such as hydropic leiomyoma (HL), which exhibit unique histological features like zonal edema and increased vascularity. However, due to its rarity, comprehensive clinical knowledge about HL is limited. We report a case of a 49-year-old Japanese woman who was premenopausal and nulliparous, presenting with a two-year history of abdominal distension. An MRI scan revealed a 20 cm mass in the posterior part of the uterus, exhibiting characteristics suggestive of an ovarian tumor. During laparotomy, a cystic tumor connected with a swollen fibroid was found, and pathology confirmed HL. This case emphasizes that hydropic leiomyomas can mimic malignant tumors on ultrasonography due to their atypical features, necessitating additional evaluations using alternative imaging techniques or histopathological examinations for accurate diagnosis and appropriate management. The patient recovered uneventfully, broadening our understanding of HL's clinical presentation.