American Journal of Case Reports

Anti-Estrogen Therapy Achieves Complete Remission and Stability in Recurrent Cervical Cancer: A Case Study

BACKGROUND Studies using transgenic mouse models have demonstrated that estrogen is necessary for the development of cervical cancer, particularly in tissues responsive to estrogen. Estrogen also protects cervical cancer cells from apoptosis, suggesting its role in the survival and persistence of cancer cells. CASE REPORT An 84-year-old woman with diabetes mellitus, hypertension, and stage III chronic renal failure was diagnosed with cervical squamous cell carcinoma, FIGO stage IB2. She underwent complete concurrent chemoradiotherapy, but central recurrence was found 9 months later. However, instead of salvage chemotherapy, substitutionary anti-estrogens were given due to her poor medical condition and advanced age. Complete remission was noted after tamoxifen therapy. Since the cervical cancer relapsed again 40 months after tamoxifen use, the anti-estrogen therapy was shifted to letrozole. The SCC-Ag level decreased dramatically after letrozole therapy, and disease stability was achieved until 29 months afterward. After 5 years and 9 months of anti-estrogen use only, the patient died due to noncancer-related pneumonia and heart failure. CONCLUSIONS This report demonstrates the tumor-stabilizing and therapeutic effect of anti-estrogens in the treatment of squamous cervical carcinoma. Further clinical trials are warranted to evaluate the efficacy of anti-estrogen therapy in cervical cancer patients.

Emergency Radiation Therapy for Hemorrhage in Botryoides Sarcoma: A Pediatric Case Report

BACKGROUND Botryoid rhabdomyosarcoma is a subtype of embryonal rhabdomyosarcoma and is a rare and highly vascular malignant childhood tumor, with the most common sites of origin being the vagina and cervix in young girls. This report describes the case of a 15-year-old girl presenting with vaginal hemorrhage due to botryoid rhabdomyosarcoma treated with emergency radiation therapy. Hemorrhage in botryoid rhabdomyosarcoma can present as a life-threatening emergency requiring immediate intervention. This case highlights the role of emergency external radiation therapy as a valuable adjunct in managing hemorrhage in botryoid rhabdomyosarcoma, especially when surgical options are limited. CASE REPORT We report a case of a 15-year-old girl who experienced repeated profuse episodes of vaginal bleeding and protruding mass in the vagina for 2 months. She had no significant medical history or family history of cancer. Magnetic resonance imaging (MRI) confirmed a massive malignant mass in the vaginal introitus that infiltrated the cervix, extending to the uterus. Based on the histopathology result, the diagnosis was botryoid rhabdomyosarcoma with a primary site located on the cervix, and due to the emergency situation and the risks associated with surgical intervention, emergency external radiation therapy was initiated. After 1 session of radiation therapy, the bleeding was significantly reduced. CONCLUSIONS Hemorrhage in botryoid rhabdomyosarcoma is a rare but critical complication. Emergency external radiation therapy helps stabilize the patient with acute bleeding caused by botryoid rhabdomyosarcoma. Further research is needed to find the optimal treatment strategies.

A Rare Case of Ruptured Appendicitis Secondary to Metastatic Ovarian Cancer

BACKGROUND Patients with advanced stage ovarian cancer typically have vague non-specific abdominal symptoms related to pelvic tumor, metastasis, and ascites. When these patients present with more acute abdominal pain, appendicitis is rarely considered. Acute appendicitis due to metastatic ovarian cancer has been sparsely documented in the medical literature; only twice, to our knowledge. CASE REPORT A 61-year-old woman with a 3-week history of abdominal pain, shortness of breath, and bloating was diagnosed with ovarian cancer after computed tomography (CT) demonstrated a large pelvic cystic and solid mass. Five weeks later she underwent an omental biopsy to determine cell type and potential upstaging of the ovarian cancer to stage IV, as other aggressive cancers such as breast cancer can also involve the pelvis/omentum. Seven hours after her biopsy, she presented with increasing abdominal pain. Post-biopsy complications such as hemorrhage or bowel perforation were initially suspected to be the cause of her abdominal pain. However, CT demonstrated ruptured appendicitis. The patient underwent an appendectomy and histopathologic examination of the specimen revealed infiltration by low-grade ovarian serous carcinoma. CONCLUSIONS Given the low incidence of spontaneous acute appendicitis in this patient's age group, and the lack of any other clinical, surgical, or histopathological evidence to suggest another cause, metastatic disease was ruled to be the likely source of her acute appendicitis. Providers should be aware of appendicitis in a broad differential diagnosis and have a low threshold for ordering abdominal pelvis CT when advanced stage ovarian cancer patients present with acute abdominal pain.

Necrotizing Fasciitis In A Patient with Metastatic Clear Cell Ovarian Carcinoma Treated with Bevacizumab

BACKGROUND Necrotizing fasciitis is a life-threatening infection of the deep soft tissues that leads to progressive destruction of the fascia and subcutaneous fat. It typically spreads along the muscle fascia planes because of the relatively poor blood supply. Muscle tissue is usually spared because of its better blood supply. The usual risk factors for necrotizing fasciitis include trauma, malnutrition, obesity, uncontrolled diabetes mellitus, alcoholism, cirrhosis, neutropenia, and recent surgery. CASE REPORT We present a case of a middle-aged female who presented with necrotizing fasciitis of the right gluteal region. Her medical history was significant for well-controlled diabetes mellitus (hemoglobin A1c: 6.6), and clear cell carcinoma of ovaries (stage IV). She was on active chemotherapy with bevacizumab, paclitaxel, and carboplatin. She underwent incision and debridement of right gluteal abscess with drainage of 200 ml of foul-smelling pus and was started on intravenous antibiotics. Her blood cultures were negative, but the cultures taken from the right gluteal abscess showed moderate growth of Escherichia coli. The antibiotics were de-escalated and the patient was discharged with outpatient follow-up. CONCLUSIONS Bevacizumab, a humanized monoclonal IgG antibody, is a novel treatment for metastatic ovarian cancer. It is associated with necrotizing fasciitis due to anti-angiogenic, pro-thrombotic, and poor wound healing properties. It should be stopped in the patients presenting with necrotizing fasciitis.

Concurrent Mature and Immature Teratomas in Contralateral Ovaries in a 30-Year-Old Woman: A Case Report

BACKGROUND Mature ovarian teratomas account for 20% of all adnexal tumors and consist of mature elements from all 3 germ layers. Immature teratomas are much rarer, comprising only 3% of clinical cases. These tumors, which arise from primordial germ cells, can contain both differentiated and undifferentiated tissue, primarily neuroepithelium. It is very rare for a single patient to have both gonads affected, especially by 2 different types of teratomas. This report describes the case of a 30-year-old woman with a right ovarian mature cystic teratoma and a left ovarian immature teratoma. CASE REPORT This report discusses the case of a 30-year-old woman who presented with tumors on both ovaries. Both lesions were surgically removed and histologically analyzed. An immature teratoma was diagnosed on the left ovary and a mature one on the right one. The patient received chemotherapy, which was discontinued due to complications. Follow-up imaging revealed no recurrences. The patient remains under oncological care with regular follow-ups. CONCLUSIONS In cases of diagnostic uncertainty, computed tomography (CT) is recommended, as it provides detailed imaging of the tumor's structure. The most important element of diagnosing both mature and immature teratomas, providing a clear and indisputable diagnosis, is a histopathological examination. Unfortunately, these tumors do not usually cause specific symptoms. This report highlights the importance of thorough histological sampling and evaluation of ovarian tumors, including ovarian mature cystic teratoma, so that malignant elements are not missed and patients receive appropriate management.

Incidental Serous Tubal Intraepithelial Carcinoma that Developed into Primary Peritoneal Serous Carcinoma in a Patient without BRCA Mutation

BACKGROUND Serous tubal intraepithelial carcinoma (STIC) is proposed as the precursor of ovarian, tubal, and peritoneal high-grade serous carcinoma, but the clinical significance remains unclear, especially in the normal population. We report a rare case of STIC in a patient undergoing non-prophylactic surgery who developed PPSC without a strong family history or BRCA mutations. CASE REPORT A 62-year-old woman presented with an abnormal pap smear (ASC-H). She underwent vaginal wall biopsy, endocervical curettage, and HPV testing, which revealed vaginal wall intraepithelial neoplasia 3 and cervical intraepithelial neoplasia 3, HPV 68 positive. Laparoscopic total hysterectomy, including an upper vagina and bilateral salpingo-oophorectomy, was performed. Postoperative histopathologic examination revealed carcinoma in situ of the cervix, and, incidentally, a serous tubal intraepithelial carcinoma (STIC) in situ of both fallopian tubes. During follow-up, the patient was diagnosed with primary peritoneal serous carcinoma (PPSC), 22 months after the initial operation. BRCA mutations were not detected. The findings in our case, coupled with current evidence, suggest the distal fallopian tube as the source of PPSC. CONCLUSIONS After an incidental diagnosis of STIC, we recommend surveillance for BRCA mutations. Standard management remains unclear, but further surgical evaluation and/or chemotherapy should be considered in patients with isolated STIC.

Colorectal Cancer with Ovarian Metastasis After Panitumumab and FOLFOX4: A Case Report

BACKGROUND Colorectal cancer is now one of the most common malignancies worldwide. Systemic treatment options for metastatic colorectal cancer (mCRC) are unsatisfactory, and the disease recurs despite the use of multiple drug combinations. Patients with mCRC and left-sided KRAS/NRAS/BRAF wild-type tumors may benefit from epidermal growth factor receptor (EGFR) inhibitors such as cetuximab or panitumumab in combination with chemotherapy. This report describes the case of a 67-year-old woman with advanced wild-type (WT) KRAS sigmoid colon adenocarcinoma in remission following treatment with panitumumab-FOLFOX4, presenting with ovarian metastases. CASE REPORT We present the case of a 67-year-old woman who was diagnosed with sigmoid colon cancer with metastasis to the periaortic lymph nodes. Due to the wild-type tumor subtype, targeted treatment with panitumumab and FOLFOX4 chemotherapy was implemented. Upon the third follow-up evaluating the effectiveness of therapy, complete remission (CR) was confirmed. During a routine follow-up at the gynecologist, a gynecologic ultrasound performed showed a questionable left adnexal mass, suspicious for primary ovarian cancer. Histopathological examination revealed metastatic adenocarcinoma of the colon. Imaging studies confirmed significant cancer progression. The chemotherapy regimen was changed to FOLFIRI. Imaging studies evaluating the effectiveness of therapy were performed every 3 months, confirming disease stabilization. The patient is still alive and continues to receive systemic therapy. CONCLUSIONS This case report supports previous studies, reports, and clinical trial data showing that treatment with panitumumab-FOLFOX4 can lead to a response in patients with advanced colonic adenocarcinoma and shows the importance of patient selection using molecular diagnostics, including KRAS mutation testing.

Invasive Hydatidiform Mole Mimicking Ectopic Pregnancy: A Case Report and Literature Analysis

BACKGROUND Gestational trophoblastic diseases (GTDs) are a group of benign and malignant tumors that arise from placental tissue. Ectopic pregnancies most commonly occur within the fallopian tubes. The estimated incidence of ectopic gestational trophoblastic diseases (GTDs) is approximated at 1.5 per 1 000 000 pregnancies, which makes it a very rare condition. The diagnosis is challenging since the clinical presentation often mimics conventional ectopic pregnancy, but management approaches are significantly different. CASE REPORT A 41-year-old woman (G7 T4 P0 A2 L4) presented to the Emergency Department with a period of amenorrhea followed by vaginal bleeding and abdominal pain. Right-side abdominal tenderness was noted upon physical examination. Laboratory investigations showed elevated serum ß-hCG level. Ultrasound examination showed a normal-size uterus. No intrauterine gestational sac was identified. An echogenic mass with central lucency was noted in the right adnexa, measuring 2.1×1.8 cm, with minimal free fluid present in the pouch of Douglas. The findings were suggestive of a ruptured right-side ectopic pregnancy. The patient underwent laparoscopic salpingectomy. Histopathological examination of the specimen revealed a complete hydatidiform mole with vascular invasion. The patient was treated conservatively and was followed up by a serial ß-hCG level until she achieved 3 consecutive negative ß-hCG titers. CONCLUSIONS Invasive moles located in the fallopian tube are very rare in the literature, and further research is needed to ascertain the proper management of such cases. Histopathological examination of the salpingectomy specimen has an essential role in confirming the diagnosis of ectopic GTDs and guiding the management.

Rapid Progression of Malignant Peritoneal Mesothelioma Mimicking a Postoperative Complication in a Young Woman: A Case Report

BACKGROUND Malignant peritoneal mesothelioma is a rare disease with a poor prognosis that often presents with vague symptoms and inconclusive laboratory test results. Causes include industrial pollutants, primarily asbestos, and certain genetic mutations, such as BAP1. Due to the nonspecific symptoms, it is often incidentally diagnosed during or after other surgical procedures. CASE REPORT A 35-year-old healthy woman underwent an uncomplicated laparoscopic left salpingo-oophorectomy for a symptomatic large ovarian mature cystic teratoma. She subsequently presented with late-onset postoperative fever, leukocytosis, and multiple intra-abdominal masses. Following an exploratory laparotomy, extensive infectious disease evaluation, and multiple biopsies requiring interdisciplinary collaboration, malignant peritoneal mesothelioma was diagnosed by positive histologic staining of an omental biopsy for D2-40 and CK5/6. This first specimen was positive for BAP1, with the second, a liver biopsy, testing negative for BAP1. The tumor cell testing was also notable for mutations in NF2, MLL2, and ARID1A, and the hereditary cancer genetic testing was overall unremarkable. Her disease progressed rapidly, and she died 6 months after her initial procedure. CONCLUSIONS This case of rapidly developing malignant peritoneal mesothelioma following surgical management of an ovarian mature teratoma highlights the complexity in diagnosing a rare disease that presents with nonspecific symptoms in an otherwise young and healthy woman. The rapid disease course was likely accelerated by expansive intraperitoneal spread and multiple somatic oncogenic mutations in BAP1, NF2, MLL2, and ARID1A. Gynecologists should keep a broad differential for postoperative complications, as occult malignancies can present with symptoms that mimic postoperative complications.

Torsion of Granulosa Cell Tumor of the Ovary in a Preschool Patient: A Rare Cause of Acute Abdomen

BACKGROUND Granulosa cell tumor of the ovary is very rare in childhood; its most common clinical manifestation is isosexual precocious puberty. Clinical presentation as acute abdomen due to pain and ovarian torsion is rare, but a granulosa cell tumor must be suspected in a patient with this acute presentation and signs of early puberty. Adult-type granulosa cell tumor is an even rarer occurrence in children. CASE REPORT We report a case of torsion of adult-type granulosa cell tumor of the ovary in a 5-year-old patient with acute abdominal pain and ovarian torsion and highlight the importance of histological diagnosis of this tumor for the therapeutic plan and progression of these patients. CONCLUSIONS Precocious puberty, pain, abdominal distension, and an ultrasonography with suspicion of ovarian torsion are warning signs that may indicate the presence of a gonadal stromal tumor in pediatric patients seen at an emergency unit. These patients require long-term follow-up by a pediatrician and gynecologist because of the potential for late recurrence.

Complete Laparoscopic Removal and Staging of a Large Ovarian Malignancy: A Case Report

BACKGROUND Advances in minimally invasive surgery are essential for the improvement of patient care, overall health care efficiency, and total cost reductions. Morcellation, the technique used in the present case, is often used to aid minimally invasive laparoscopic procedures, whereby larger tissue specimens are broken down and divided to be removed via smaller incisions. Morcellation has become controversial in the medical community owing to the risk of the procedure spreading an occult malignancy. CASE REPORT A 47-year-old woman with obesity presented with right-sided pelvic pain and suspicion of ovarian torsion. The patient experienced severe acute pain and was taken to the operating room for laparoscopy to remove the left-sided adnexal mass seen on imaging. During surgery, there was no evidence of torsion, and the mass was perfused. During removal of the entire tumor, blunt instruments were utilized for in-bag manual morcellation to avoid spillage of the contents of the bag, which were then sent for frozen section analysis, and a diagnosis of adenocarcinoma of the ovary was made. Once the tumor was removed, a board-certified gynecologic oncologist was consulted to assist with the remainder of the procedure. Following this, the ovarian cancer staging procedure was able to be completed laparoscopically. CONCLUSIONS This technique of in-bag manual morcellation using blunt instruments allows laparoscopic procedures to be performed on large tissue specimens with potential malignancies without spreading cancerous tissue, overcoming the associated risks of dissemination of malignancy. This technique is therefore more beneficial to patients.

Giant Ovarian Leiomyoma Mimicking a Malignant Intra-Abdominal Tumor

BACKGROUND Leiomyoma is a benign gynecological neoplasm most commonly seen in the uterus, while primary ovarian leiomyomas are exceptionally uncommon. These tumors can grow to a large size and undergo degenerative changes, including infarction. Large ovarian leiomyomas with a solid-cystic appearance on imaging can closely resemble malignant ovarian neoplasms, leading to diagnostic uncertainty. We present a case of giant ovarian leiomyoma exhibiting clinical and radiological features closely mimicking those of an ovarian malignancy. CASE REPORT A 68-year-old para 4 woman presented with a four-year history of progressive abdominal distension, which had notably increased over the past two years. A computed tomography (CT) scan of the thorax, abdomen, and pelvis (CT-TAP) revealed a well-defined, lobulated, heterogeneously enhancing, solid-cystic mass, predominantly occupying the left lower abdominal quadrant and originating from the right ovary. Tumor marker analysis showed normal levels of CEA and CA19-9 with a mildly elevated CA-125 level of 51 U/ml. The calculated Risk of Malignancy Index (RMI) was 153, raising suspicion for an ovarian malignancy. In light of these findings, the patient was planned for a total abdominal hysterectomy and bilateral salpingo-oophorectomy (TAHBSO). The intraoperative findings revealed a right ovarian mass measuring 20×20 cm. Histopathological examination confirmed an ovarian leiomyoma with cystic degeneration. CONCLUSIONS The case report demonstrates the preoperative diagnostic challenges associated with giant ovarian leiomyoma, which can result in an unnecessary radical surgical procedure. This underscores the importance of meticulous preoperative and intraoperative assessment to ensure accurate diagnosis and appropriate management.

Giant Uterine Fibroid Complicated by Abnormal Erythrocytosis in a 33-Year-Old Woman: A Case Report

BACKGROUND Uterine fibroids are common benign gynecologic tumors characterized by abnormal uterine bleeding, menorrhagia, and anemia. Myomatous erythrocytosis syndrome (MES), a rare secondary erythrocytosis associated with fibroids, presumably results from ectopic erythropoietin production by the fibroids, which activates the Janus kinase 2 (JAK-2) pathway and increases red blood cell counts. MES clinically presents with abdominal distension, skin discoloration, and menstrual irregularities. Management is complex due to substantial risks of intraoperative bleeding and thromboembolism. This report describes a 33-year-old woman with MES. After myomectomy, her erythrocytosis normalized, but she developed postoperative bleeding. CASE REPORT A 33-year-old woman presented with a 2-year history of progressive abdominal distension and an abdominal mass comparable in size to a 20-week gravid uterus, plethoric facies, and reddish-purple nail beds. Her hemoglobin level was 19.7 g/dL; hematocrit was 60.4%. Ultrasound and computed tomography confirmed a 25×20×12 cm fibromatous uterus. To reduce thromboembolic risk, she received low-molecular-weight heparin and intravenous hydration preoperatively. Abdominal myomectomy was performed, during which substantially increased uterine and fibroid vascularity was observed. However, the patient experienced severe postoperative hemorrhage (1800 mL/24 h), warranting a second operation for hemostasis. She recovered well and was discharged on postoperative day 14; her hemoglobin level normalized. Pathologic examination confirmed uterine leiomyoma, supporting the diagnosis of MES. CONCLUSIONS MES is a rare condition characterized by large uterine fibroids and erythrocytosis; postoperative bleeding and thrombosis are common complications. This case highlights the importance of accurate diagnosis and effective management. Erythropoietin-related therapeutic targets for MES are needed.

Conception After Chemotherapy for Intraplacental Choriocarcinoma: Favorable 5-Year Follow-Up of the Mother and Child

BACKGROUND Intraplacental choriocarcinoma (IC) is a rare subtype of gestational choriocarcinoma that can be associated with placental hemorrhage and metastases to the mother and fetus. Chemotherapy can be effective in cases of placental localization and may not adversely affect fertility. This report describes a 32-year-old woman who conceived 10 months after chemotherapy for IC and achieved successful term delivery of that pregnancy. CASE REPORT A 32-year-old woman delivered her first child by vacuum extraction due to weak labor and fetal distress. The newborn presented with severe anemia, as well as elevated alpha-fetoprotein and fetal hemoglobin levels, suggesting fetomaternal hemorrhage. Placental histopathology revealed IC. Postpartum elevation of human chorionic gonadotropin levels prompted EMA-CO (etoposide, methotrexate, actinomycin D/cyclophosphamide, vincristine) chemotherapy; 7 treatment courses were completed without side effects or metastasis. Ten months after completion of chemotherapy, the patient spontaneously conceived. She subsequently delivered a healthy boy at 38+5 weeks of gestation; placental examination did not reveal malignancy. During the 5-year follow-up period, both children developed normally, and no maternal recurrence or metastasis was observed. CONCLUSIONS This report describes a case of postnatal diagnosis of IC via placental histopathology, followed by successful treatment and subsequent pregnancy within 1 year after chemotherapy completion. The findings highlight the importance of accurate diagnosis and appropriate management of IC; they support previous observations that chemotherapy does not preclude future successful pregnancies. Additionally, the report underscores the clinical challenges of IC, its implications for future pregnancies, and the need for long-term follow-up concerning both mother and child.

A Huge Malignant Ovarian Tumor: A Case Report

BACKGROUND Huge ovarian tumors are rare. In developing countries, many women with huge ovarian tumors only seek consultation when the tumor has become very large. Most cases are benign, and only a few cases were reported to be malignant. This case report presents a case of huge malignant ovarian tumor with a final diagnosis of stage III epithelial ovarian cancer. The tumor was completely removed. The huge malignant ovarian tumor in this case report was completely excisable. CASE REPORT A 43-year-old woman visited the tertiary hospital in Surabaya Indonesia in early September 2020 with a complaint of an enlarged abdomen. The patients had 3 children and normal menstrual periods. Her bowel function was normal. The patient started to notice the abdominal enlargement at 3 months prior to seeking treatment. Ultrasound examination showed a unilocular cystic mass with a diameter of >25 cm, and a solid nodule with normal vascularity was seen. No ascites was noted. Histopathologic examination showed an ovarian mass weighing 9700 g with a size of 30×28×14 cm. The final result showed that the tumor was malignant; specifically, the tumor was a sero-mucinous adenocarcinoma of the left ovary, grade II, which had metastasized to the omentum. CONCLUSIONS Huge malignant ovarian tumors tend to be at an early stage when the diagnosis is made, and they are completely excisable.

Radiotherapy-Related Bladder Fistula: A Hidden Cause of Recurrent Abdominal Symptoms

BACKGROUND Non-traumatic bladder rupture, a rare yet potentially life-threatening condition, can stem from diverse factors such as malignancies, bladder inflammation, or bladder diverticulum rupture. Pelvic radiotherapy, in extremely rare instances, can lead to radiation cystitis and subsequent bladder fistula formation. Patients with such conditions often present with abdominal pain, hematuria, oliguria, and urinary ascites. The diagnosis of radiotherapy-related bladder fistula poses significant challenges, particularly in patients with protracted illnesses and atypical abdominal symptoms, as it can be easily overlooked. CASE REPORT We present the case of a 60-year-old woman who, 14 years following a hysterectomy, bilateral adnexectomy, and radiotherapy for cervical carcinoma, developed recurrent abdominal pain and ascites. Initially, her symptoms were misattributed to gastrointestinal disorders. However, ascites analysis, which revealed markedly elevated creatinine and urea nitrogen levels in the ascitic fluid compared to serum, led to the suspicion of urinary ascites. The definitive diagnosis of an intermittent bladder fistula was confirmed through the instillation of a saline-methylene blue solution via the urinary catheter, which resulted in the drainage of blue ascitic fluid. CONCLUSIONS The diagnosis of vesical fistula is often challenging due to its diverse presentations and the possibility of being overlooked by CT. However, early recognition and appropriate management are crucial to prevent severe complications. This case highlights the importance of considering bladder fistula in the differential diagnosis of unexplained ascites and peritonitis, especially in patients with a history of pelvic radiotherapy or cystoscopic treatment.

AMH and Kisspeptin Receptor Expression in Rare Hydropic Leiomyoma: A Case Study

BACKGROUND Leiomyomas are common benign uterine tumors (BUMTs) with diverse histopathological subtypes and variable clinical presentations. While most are asymptomatic, some cause significant morbidity, including abnormal uterine bleeding, infertility, and pain. Hydropic leiomyomas (HLMs) are rare variants histopathologically characterized by zonal edema and may pose diagnostic challenges, particularly when located in atypical sites such as the retroperitoneal space. This report presents a case of a retroperitoneal HLM with strong expression of anti-Müllerian hormone (AMH) and its receptor (AMHR2), and kisspeptin (KISS1) and its receptor (KISS1R), suggesting potential new therapeutic targets. CASE REPORT A 44-year-old woman presented with acute lower abdominal pain. Magnetic resonance imaging (MRI) revealed a well-circumscribed, pedunculated retroperitoneal mass originating posteriorly from the uterine body-cervix junction. MRI findings suggested a benign mesenchymal tumor but could not exclude malignancy. Surgical excision was performed, and histopathological examination confirmed HLM. Immunohistochemical analysis demonstrated strong nuclear and cytoplasmic expression of AMH, AMHR2, KISS1, and KISS1R in tumor cells, making this the first reported case of such expression in HLM. The patient had an uneventful postoperative course, and no recurrence was observed during a 2-year follow-up. CONCLUSIONS This case underscores the diagnostic complexity of retroperitoneal HLMs and the importance of MRI in differentiating BUMTs from malignancies. Strong AMH, AMHR2, KISS1, and KISS1R expression suggests a potential role of these regulatory proteins in HLM pathophysiology. Further research on targeted modulation of these pathways may provide novel therapeutic approaches for BUMTs, particularly in cases where conventional treatments are limited.

Self-Assembling Hemostatic Peptide Gel Used to Successfully Treat Rectal Ulcer in Radiation-Associated Vascular Ectasia: A Case Report

BACKGROUND Radiation-associated vascular ectasia, formerly known as radiation proctitis, is a known gastrointestinal complication of radiation therapy. Rectal ulcers in the setting of radiation-associated vascular ectasia are particularly challenging to treat. They are often refractory to conventional medical and endoscopic therapies and cause significant morbidity for patients. Self-assembling hemostatic peptide gel is an endoscopic tool used primarily for hemostasis but has also been hypothesized to have mucosal repair properties. CASE REPORT A 41-year-old woman with a history of cervical adenocarcinoma presented with abdominal pain and rectal bleeding several months after brachytherapy and external beam radiation therapy. Conservative therapy with topical pramoxine was ineffective, so she underwent colonoscopy, which revealed a non-bleeding rectal ulcer in the setting of bleeding radiation-associated vascular ectasia. Argon plasma coagulation was used to treat the bleeding vascular ectasia, but the ulcer was treated solely with self-assembling hemostatic peptide gel. A follow-up endoscopy two months later showed complete resolution of the ulcer. CONCLUSIONS The mucosal repair properties of the self-assembling hemostatic peptide gel, an endoscopic product initially designed for hemostasis, are highlighted by this case in which a non-hemorrhagic ulcer was successfully treated. Furthermore, this is a novel use of the self-assembling hemostatic peptide gel in the setting of radiation-associated vascular ectasia and may represent a useful addition to the endoscopist's toolkit for managing a challenging condition.

Giant Cell Carcinoma of the Endometrium: A Case Report of a Rare Entity with Aggressive Behavior

BACKGROUND Giant cell carcinoma of the endometrium is one of the rare variants of endometrial carcinoma, with a very limited number of reported cases and limited follow-up data. The purpose of this case report is to present yet another example of endometrial giant cell carcinoma, discuss its differential diagnosis and management course, and review all previously reported cases. CASE REPORT We report a case of a 71-year-old woman who presented with vaginal bleeding. Her laboratory investigations were within normal limits except for her glycated hemoglobin, which was 10%. Ultrasound and computed tomography scans showed an endometrial mass invading the myometrium. Microscopically, the tumor is comprised almost exclusively of multinucleated giant cells. The patient underwent a total robotic hysterectomy with bilateral salpingo-oophorectomy and lymph nodes dissection, and she is currently undergoing adjuvant radiotherapy. CONCLUSIONS Giant cell carcinoma of the endometrium is a rare diagnosis that can be established by histopathological examination after excluding the other common giant cell-rich lesions that may occur in the endometrium.

Cervical Neuroendocrine Carcinoma Presenting as Isolated Large Ovarian Metastasis: A Case Report

BACKGROUND Neuroendocrine carcinoma (NEC) of the cervix is rare and has high mortality and recurrence rates. The clinical symptoms of cervical NEC, such as abnormal vaginal bleeding and discharge, are similar to those of other cervical cancers. Here, we describe a case involving a 42-year-old woman with cervical NEC accompanied by an isolated large ovarian metastasis. CASE REPORT A 42-year-old woman had experienced abdominal discomfort for the past 4 months, along with a larger abdominal circumference. Physical examination revealed a 15-cm, solid, mobile, abdominal mass and a smooth cervix. Abdominal computed tomography revealed a hypoattenuating solid mass with a calcified component and indistinct borders, measuring 16.6×15.5 cm. Tumor marker levels were as follows: cancer antigen 125, 803.9 U/mL; carcinoembryonic antigen, 241.9 ng/mL. Preoperatively, we suspected a malignant ovarian tumor without any suspicion of cervical cancer. Intraoperatively, a 25×20-cm solid mass was found on the left adnexa with peritoneal wall and rectosigmoid adhesions. We performed a total abdominal hysterectomy with bilateral salpingo-oophorectomy, followed by peritoneal biopsy and omentectomy. Histopathological examination showed a 2.5-cm endocervical mass and a normal ectocervical epithelium. Immunohistochemistry revealed a small-cell cervical NEC with metastasis to the left ovary. The final diagnosis was a stage IB2 cervical NEC with ovarian metastasis. For treatment, we administered an etoposide-cisplatin adjuvant chemotherapy regimen. CONCLUSIONS NEC of the cervix can manifest as a large ovarian tumor, lack the usual indications for cervical cancer, and spread to the ovaries without metastasis to other organs.

A 46-Year-Old Woman with Primary Infertility and a Diagnosis of Microcystic Stromal Tumor of the Ovary Confirmed by Histology and Gene Sequencing: A Case Report and Review of the Literature

BACKGROUND Microcystic stromal tumor (MCST) of the ovary is a rare entity with distinct pathological and molecular features. However, a lack of awareness of ovarian MCST can lead to delayed diagnosis or misdiagnosis. We present a case of ovarian MCST and review all previously reported cases and discuss their clinical and pathological characteristics. CASE REPORT A 46-year-old woman with primary infertility due to polycystic ovary syndrome presented with bleeding and pain. Radiological images showed a complex solid and cystic adnexal mass. Microscopically, the tumor was lobulated with cellular regions separated by fibrous plaques and small anastomosing cysts, consistent with an ovarian MCST. The tumor cells showed positive staining for vimentin, CAM 5.2, CD10, ß-Catenin, CD99, and cyclin D1. Genetic sequencing showed a point mutation in the CTNNB1 gene, with no mutations in the APC, BRCA1, and BRCA2 genes. The patient underwent surgery and was disease-free at 24 months after her initial diagnosis. CONCLUSIONS The diagnosis of ovarian MCST should consider the differential diagnosis of cystic tumors of the ovary. Further research is encouraged to elucidate the various molecular pathways involved in the pathogenesis of this tumor and to determine its optimal treatment and long-term prognosis.

Disseminated Peritoneal Leiomyomatosis of the Peritoneum Following Laparoscopic Supracervical Hysterectomy with Morcellation: A Case Report

BACKGROUND Disseminated peritoneal leiomyomatosis is a rare complication of laparoscopic procedures involving uterine morcellation. These fibroid-like lesions, which derive their blood supply from non-uterine sources, may develop after myomectomy, supracervical hysterectomy, or total laparoscopic hysterectomy - particularly when mechanical morcellation is used. Although the incidence is low, disseminated leiomyomatosis should be considered in the differential diagnosis of abdominal or pelvic pain in patients with a history of such surgeries. CASE REPORT A 40-year-old woman presented with intermittent lower abdominal pain. She had undergone a laparoscopic supracervical hysterectomy with salpingectomy 5 years earlier due to abnormal uterine bleeding, during which uncontained mechanical morcellation was performed. At present, transvaginal ultrasound revealed a solid mass adjacent to the bladder with features suggestive of a leiomyoma. Magnetic resonance imaging (MRI) showed multiple lesions, including a polycyclic mass located above the bladder and additional foci near the sigmoid colon and the bladder dome. Laparoscopy confirmed the presence of leiomyomas on the anterior abdominal wall, sigmoid colon adventitia, and bladder dome. All lesions were excised using laparoscopic techniques, and the postoperative course was uneventful. Histopathological examination confirmed leiomyomas with positive estrogen and progesterone receptor expression. CONCLUSIONS This case underscores the importance of considering disseminated peritoneal leiomyomatosis in the differential diagnosis of abdominal or pelvic pain in patients with a history of laparoscopic hysterectomy with morcellation. It also emphasizes that this condition can mimic malignancy on imaging studies and highlights the essential role of histopathological examination in diagnosing this benign disease.

A Rare Case of Ruptured Malignant Ovarian Brenner Tumor

BACKGROUND Ovarian cancer is the leading cause of death in women with gynecological cancers. Ovarian Brenner tumor (BT) is an extremely rare type of epithelial ovarian cancer that accounts for about 1-3% of all ovarian cancers. Herein, we report a rare case of ruptured malignant ovarian Brenner tumor. CASE REPORT A 39-year-old P0A0 woman came to the Emergency Department (ED) with abdominal pain and tenderness. Perforated appendicitis was initially suspected and an emergency laparotomy was performed by the General Surgery Department. Then, a 25×20×15 cm grayish cystic mass originating from the right adnexa was found. We consulted intraoperatively with the Gynecology Oncology Department and decided to perform complete surgical staging. Histopathological examination confirmed the diagnosis of malignant Brenner tumor (MBT). The patient was then given adjuvant chemotherapy with a paclitaxel carboplatin regimen. In this case report, we present our case along with a review of the current literature regarding the diagnosis and therapy of malignant Brenner tumor. CONCLUSIONS Ovarian MBT is an extremely rare ovarian cancer. Diagnosing MBT can be challenging as there are no clinical, laboratory, or imaging features typical for it. Surgery is the mainstay treatment in MBT cases. The role of adjuvant chemotherapy in MBT is still being debated.

A 12-Year-Old Girl with Juvenile Granulosa Cell Tumor of the Ovary, Presenting with Adolescent Hyperprolactinemia, Galactorrhea, and Amenorrhea

BACKGROUND Juvenile-type granulosa cell tumors (JGCTs) are a rare subtype of sex cord stromal tumor with a characteristic histology that is commonly found in the first 3 decades of life. It most commonly presents with symptoms of hyperestrogenism, which may present as precocious pseudopuberty or as menstruation-related symptoms, allowing for early detection of the tumor. CASE REPORT We present the case of a 12-year-old girl who presented to her primary care provider (PCP) with secondary amenorrhea with intermittent abdominal pain, who underwent an ultrasound for further evaluation, which revealed a large incidental pelvic mass. She was admitted to the Emergency Department (ED) and had findings of galactorrhea and hyperprolactinemia on examination. Imaging studies demonstrated a large ovarian mass measuring 15.0×9.0×18.8 cm that was resected, and subsequent pathology results showed JGCT stage 1A. CONCLUSIONS Prognosis of granulosa cell tumors (GCT) largely depends on its initial size, stage at diagnosis, residual tumors after surgery, and the subtype of GCT. If the patient is of reproductive age, fertility-sparing surgical options must be considered and patients must be regularly monitored for recurrence. JGCTs can present with minimal to no symptoms of precocious puberty in young girls but may present with amenorrhea, which may be considered normal for their developmental age. Although JGCTs are rare, they are important to include in differential diagnoses of younger female patients with abdominal pain, especially if accompanied by hormonal irregularities.

A Case of Vaginal Malignant Melanoma Completely Resected by Radical Surgery

BACKGROUND Primary vaginal malignant melanoma is a rare and aggressive tumor with a high risk of local recurrence and distant metastasis. Although there are several available treatment options, none are considered as standard. Surgical resection is the first treatment choice because of its superior survival benefits. CASE REPORT The patient was a 56-year-old woman with a vaginal mass. At the first visit to our institution, a 20×20 mm black and flat lesion on the lower third of the posterior vaginal wall and a polypoid mass near the vaginal fornix were detected by gynecologic examination. Study of the tumor on the posterior vaginal wall suggested that it did not extend to the uterine cervix. The preoperative diagnosis was vaginal malignant melanoma FIGO stage I (cT1, cN0, cM0). The patient underwent a total vaginectomy, pelvic and inguinal lymphadenectomy, modified radical hysterectomy, and bilateral salpingo-oophorectomy. The tumor cells were arranged in sheets and nests and exhibited nuclear pleomorphism, eosinophilic cytoplasm, brisk mitotic activity, and melanin production. The overlying mucosa was ulcerated. The tumor thickness was 2.5 mm and no residual lesion was found at the surgical margin. No adjuvant therapies were performed. The patient is alive without recurrence 15 months after the initial treatment. CONCLUSIONS This is a case of vaginal malignant melanoma for which complete response was achieved by radical tumor resection, without severe adverse effects and with no observed recurrence 15 months after the surgery.

A 68-Year-Old Woman Presenting with Recurrent Abdominal Pain and a Diagnosis of a Presacral Retroperitoneal Benign Schwannoma that Mimicked an Ovarian Tumor on Pelvic Magnetic Resonance Imagining

BACKGROUND Schwannomas are benign tumors and their appearance in the pelvic region is rare and poses a major diagnostic problem. They can be sporadic or associated with genetical syndromes. They have a slow growth rate and may be asymptomatic for many years. Symptoms are usually nonspecific and due to compression of adjacent structures. Abdominal imaging modalities may not be able to differentiate a benign schwannoma from a malignant retroperitoneal tumor. This report is of a case of a 68-year-old woman presenting with recurrent abdominal pain and a diagnosis of a presacral retroperitoneal benign schwannoma that mimicked an ovarian tumor on pelvic magnetic resonance imaging. CASE REPORT The patient had a history of a femoral hernia repair and recurrent lower abdominal pain. Pelvic imaging raised the suspicion of a primary ovarian tumor. The mass appeared to have clear cleavage planes with the surrounding structures, so the patient was proposed for an exploratory laparotomy. Prior to the surgery, an additional pelvic computed tomography (CT) was performed (10 months after the first one), which did not show progression of the disease. The histological examination result was compatible with a benign retroperitoneal schwannoma and not an ovarian tumor. CONCLUSIONS This report highlights that the diagnosis of retroperitoneal and pelvic masses can be challenging. In women, a primary ovarian tumor should be excluded on imaging and the diagnosis of a benign tumor, such as schwannoma, must be confirmed by histopathology, either preoperatively or following tumor resection.

Fatal Progression of Vulvar Cancer in Pregnancy: A Case Report on Delayed Intervention

BACKGROUND Vulvar cancer (VC) in pregnancy is a rare case, and its incidence is unknown until now. This case report highlights the rapid progression of disease 3 months after delivery, and our limitation in low resource setting. CASE REPORT A pregnant woman in her early 40's presented with a left vulvar lesion. The lesion was 7×5 cm, with clitoris involvement. No infiltration to adjacent organs was present. Her biopsy showed well-differentiated squamous cell vulva carcinoma. After C-section delivery at 34 weeks of gestation, the diagnosis remained FIGO stage IB (T1b N0 M0). MRI and radical vulvectomy were planned immediately, but due to National Health Insurance limitations the procedure was performed 3 months later. After evaluation 3 months later, the clinical diagnosis had progressed to advanced-stage vulva carcinoma. This case report highlights rapid progression of VC in pregnancy and 3 months after delivery. The possibility of rapid progression of VC during the postpartum period should not be overlooked, and timely treatment of VC in pregnancy is essential to ensure better outcomes. CONCLUSIONS Gynecologists should not overlook the possibility of rapid progression of VC after delivery due to several factors, such as immunologic and physiological changes during the puerperium phase. The operation should be carried out as soon as possible, but could be delayed after delivery, considering inguinal lymphadenectomy is an integral part of frozen section surgical staging.

Myxoid Liposarcoma of the Vulva: A Rare Malignancy Mimicking Benign Vulvar Mass

BACKGROUND Liposarcoma of the vulva is an exceptionally rare malignant tumor. Clinically, vulvar liposarcoma often mimics benign lesions, thus misdiagnosis is common. Herein, we present a case of myxoid liposarcoma of the vulva. To the best of our knowledge, this is the first case report of vulvar liposarcoma from Indonesia. CASE REPORT We present a case of a 39-year-old woman with left vulvar mass of 6 years duration that progressively increased in size. The patient reported having pressure and discomfort, especially during movement, but reported no pain. Lipoma was initially suspected. Surgical excision was performed and histopathological examination revealed a well-differentiated myxoid liposarcoma. The base and excised margins of the tumor were free of malignant cells. Post-operative course was uneventful and she was discharged in a satisfactory condition. The patient had been under regular follow-up and is currently symptom- and recurrence-free. We also review other cases of vulvar liposarcoma to further comprehend characteristics of this rare malignant tumor. CONCLUSIONS Liposarcoma of the vulva occurs very rarely, but clinicians and pathologists should always consider it as a differential diagnosis when presented with vulvar mass. Biopsy of a vulvar mass is crucial. Surgical excision remains the mainstay of treatment. Adjuvant radiotherapy may be considered in certain cases. Comprehensive follow-up for recurrences or metastasis is recommended throughout life.

Musculoskeletal Pain and Right Leg Paresthesia Revealed as Large Ovarian Mucinous Cystadenoma: A Case Report

BACKGROUND Epithelial neoplasms are the most common and heterogenous group of ovarian tumors. Approximately 10-15% are primary ovarian mucinous neoplasms. Almost 80% of these consist of benign mucinous neoplasms, while the rest are borderline neoplasms, non-invasive (intraepithelial and intraglandular) carcinomas, and invasive carcinomas. Small ovarian cystadenomas are generally asymptomatic and are mainly found incidentally during an ultrasound examination for another gynecologic disorder. As their size increases, nonspecific symptoms and clinical signs develop as a result of mass effect to adjacent structures or because of tumor torsion. The main clinical symptoms are abdominal and/or pelvic pain, fullness, and discomfort. Large cystadenomas have also been associated with nausea and vomiting, urinary problems, persistent cough, back pain, metrorrhagia, and feminization. CASE REPORT We report a case of a 31-year-old woman with a body mass index of 39 who presented with increasing sacrococcygeal pain and right leg paresthesia over a 2-year period. She was treated for possible musculoskeletal and spine problems. She was finally diagnosed with a large right ovarian mucinous cystadenoma expanding in the sacrococcygeal region. She was successfully treated with complete excision of the tumor and achieved complete remission of all her symptoms. CONCLUSIONS Large ovarian mucinous cystadenomas, which develop in the sacrococcygeal region, can lead to symptoms that mimic musculoskeletal and spine problems. Early diagnosis is of great importance towards the goal of implementing proper therapeutic approaches and achieve complete remission of all clinical symptoms.

Malignant Pericardial Effusion from Cervical Squamous Cell Carcinoma: A Case Study

BACKGROUND Cervical cancer ranks fourth globally among women's cancers. Squamous cell carcinoma constitutes 70% of cervical cancer cases, often metastasizing to lungs and paraaortic nodes. Uncommon sites include the brain, skin, spleen, and muscle, while pericardial fluid metastasis is highly rare. We report a case of squamous cell carcinoma of the uterine cervix that was metastatic to the pericardium and was detected on cytologic evaluation of pericardial fluid. CASE REPORT A 42-year-old woman who was previously treated for stage III squamous cell carcinoma of the cervix presented with symptoms of cough, fever, and shortness of breath for 8 days, and chest pain for 3 days. Clinical workup revealed pericardial effusion, with spread to the lungs and mediastinal and hilar lymph nodes. Cytological analysis of the fluid showed malignant cells, consistent with metastatic squamous cell carcinoma. Immunohistochemistry demonstrated cells positive for p63 and p40, while negative for GATA-3, D2-40, calretinin, and WT1. These findings in conjunction with patient's known history of cervical squamous cell carcinoma was consistent with a cytologic diagnosis of metastatic squamous cell carcinoma to pericardial fluid. CONCLUSIONS History and clinical correlation plays a vital role in determining the primary site causing malignant pericardial effusions. While the occurrence of cervical cancer metastasizing to the pericardium is uncommon, it should be considered, particularly in cases involving high-grade, invasive tumors, recurrences, or distant metastases. This possibility should be included in the list of potential diagnoses when encountering pericardial effusions with squamous cells in female patients.

Hypophysitis Induced by Tislelizumab: A Case Report of Somnolence and Delirium with Elevated Eosinophils

BACKGROUND Tislelizumab is an immune checkpoint inhibitor (ICI) widely used in various solid tumors. Nevertheless, immune-related adverse reactions can arise during the administration of this medication. Hypophysitis is a relatively uncommon adverse reaction, typically manifesting as lethargy and hyponatremia. Although ICI-induced hypophysitis is reported, cases presenting with somnolence and delirium are extremely rare. This report describes the case of a 69-year-old woman with advanced endometrial cancer who presented with somnolence, delirium, and eosinophilia after commencing treatment with tislelizumab and was subsequently diagnosed with hypophysitis. CASE REPORT A 69-year-old woman with advanced endometrial cancer, following multiple lines of prior therapy, presented to our hospital with somnolence as the primary symptom. The patient developed worsening daytime somnolence and nocturnal delirium following tramadol administration for pain associated with a metastatic tumor in the left axilla. After a complex diagnostic and therapeutic process, it was determined that the patient had hypophysitis induced by tislelizumab. After receiving hydrocortisone treatment, the patient's symptoms of somnolence and delirium vanished. Two weeks later, the patient once more developed an adrenal crisis following an interventional procedure. We treated her anew with glucocorticoids, and she recovered. CONCLUSIONS This case underlines the necessity for timely assessment of the hypothalamic-pituitary-adrenal axis function in patients presenting with neurological symptoms, such as somnolence and delirium, particularly in the presence of elevated peripheral blood eosinophil counts. Simultaneously, it is essential for us to understand that patients with hypophysitis induced by ICIs require long-term hormone replacement therapy due to adrenocortical insufficiency.

A Case of Synchronous Primary Tumors of the Left Ovary and Uterus

BACKGROUND Synchronous tumors occur when 2 separate primary tumors are diagnosed within 6 months. They can originate from the same site or different locations. For example, synchronous primary tumors of uterine and ovarian origin are a common type. Diagnosis can be challenging, however is critical to determine whether a patient has multiple primary tumors or a single tumor with metastasis to guide effective treatment. Compared with endometrial cancer that has spread to the ovary, synchronous primary tumors of the uterus and ovaries typically require less aggressive treatment. CASE REPORT A 45-year-old woman with nonspecific symptoms of headache and confusion had imaging studies that revealed a neoplasm in her brain, which was likely causing her symptoms. The masses were metastatic lesions, and the primary cancer was determined to be synchronous endometrial ovarian cancer (SEOC). She underwent bilateral frontal craniotomy for tumor resection and diagnostic tests. She had an exploratory laparotomy, total abdominal hysterectomy, bilateral salpingo-oophorectomy, and omentectomy. She was stable during hospitalization but lost to follow-up after discharge. CONCLUSIONS Regular gynecologic examinations, including bimanual palpation of the ovaries during cervical cancer screenings, are essential for detecting cancer early and improving chances of recovery. This case also highlights the indolent growth and high risk of metastasis associated with SEOC. Although this type of cancer is rare, patients with it can be at increased risk of developing metastatic lesions in other parts of their bodies. To manage synchronous tumors effectively, a multidisciplinary approach and close collaboration between medical professionals are necessary to ensure best patient outcomes.

Sentinel Lymph Node Mapping and Staging Surgery via Gasless Transvaginal Natural Orifice Transluminal Endoscopic Surgery: A Case Report of Patient with Endometrial Cancer Patient and Comorbid Rheumatic Heart Disease

BACKGROUND Conventional laparoscopic surgery and transvaginal natural orifice transluminal endoscopic surgery (vNOTES) both use CO2 pneumoperitoneum to expose the surgical space. However, CO₂ pneumoperitoneum is undoubtedly dangerous for patients with rheumatic heart disease (RHD) and can cause cardiopulmonary impairments. Therefore, we selected the sentinel lymph node (SLN) mapping strategy to guide the staging surgery via gasless vNOTES for an endometrial cancer (EC)-patient with comorbid RHD. Here, we discuss whether our selected surgical method was safe and feasible for this patient. CASE REPORT A 43-year-old woman with a history of RHD, severe mitral regurgitation, and pulmonary hypertension for more than 30 years received diagnostic curettage for irregular vaginal bleeding for more than 1 month. Pathological examinations revealed the occurrence of highly differentiated intrauterine endometrioid adenocarcinoma. She was admitted to the gynecological ward of our hospital for further surgery. We performed EC staging surgery with SLN mapping via gasless vNOTES and adopted a series of effective measures to solve the intraoperative complications of surgical space exposure. Surgery was successful. The patient recovered well and was discharged 5 days after surgery. She has been followed up in the gynecological clinic for nearly 1 year. At the time of this report, she had good recovery, no recurrence and metastasis, and normal tumor markers. CONCLUSIONS For EC patients with comorbid RHD pathology, application of staging surgery with SLN mapping via gasless vNOTES was shown to be safe and feasible. This approach is expected to be highly effective for patients with contraindications to CO2 pneumoperitoneum laparoscopy.

Endometrioid Carcinoma Arising from an Endometriosis-Associated Abdominal Wall Scar

BACKGROUND Carcinoma arising from an endometriosis-associated abdominal wall scar is a rare entity, with only a few case reports published in the literature. The management is very controversial due to on its own rarity, and there are no specific guidelines. Treatment with a multidisciplinary team is important to achieve the best outcome. CASE REPORT We report the case of a 45-year-old woman diagnosed with a growing painless lesion in the right lower quadrant. We decided to perform Tru-Cut biopsy of the abdominal wall lesion, but unfortunately the pathological report was inconclusive at that time. Due to the presence of a highly suspicious lesion, the gynecologic oncologist together with the plastic surgeon and connective tissue surgeon decided to perform a wide resection of the abdominal wall along with hysterectomy and salpingo-oophorectomy. The final pathology report demonstrated endometriosis associated with an endometrioid adenocarcinoma grade II in the abdominal wall tumor. She was restaged with new imaging exams before the definition of the best adjuvant treatment, which showed suspicious bilateral inguinal and right axillary (1.9 cm) lymph nodes, with no other sites of metastatic disease. She was treated with megestrol acetate 160 mg/daily for 8 months, with a partial response. CONCLUSIONS Carcinoma arising from an endometriosis-associated abdominal wall scar is a rare entity, and there are no no specific treatment guidelines. Such patients must be assessed by a multidisciplinary team for decision making. Options for adjuvant and palliative treatment for endometrial cancer are generally used for the treatment of this entity. The main purpose of this article is to report this rare presentation and perform a review of the literature about diagnosis, clinical presentation, treatment, and prognosis.

Peritoneal Post-Radiation Angiosarcoma in a Patient With a History of Radiation for Cervical Cancer

BACKGROUND Post-radiation angiosarcoma (PRA) is a rare, invasive mesenchymal tumor associated with prior cancer radiotherapy. The diagnostic criteria for PRA include a history of radiation exposure, the development of a new malignant tumor with a long latency period within the irradiated field, and a histological type different from that of the primary malignancy if radiation was administered for the original cancer. Although PRA can arise in any part of the body, it most commonly occurs in the skin. Only a few cases of vascular sarcoma secondary to radiotherapy for cervical cancer have been reported, and reports of peritoneal angiosarcoma following pelvic irradiation for cervical cancer are exceedingly rare. CASE REPORT A 69-year-old woman presented with abdominal discomfort 22 years after receiving radiation therapy for cervical cancer. Imaging and laboratory evaluations failed to reveal a definitive cause, leading to suspicion of recurrence or metastasis of cervical cancer. The patient underwent surgical exploration; postoperative histopathology with immunohistochemistry confirmed the diagnosis of angiosarcoma. She subsequently received 4 cycles of chemotherapy with paclitaxel, carboplatin, and bevacizumab. However, 4 months after surgery, computed tomography revealed new lesions in the abdominal cavity and intestinal wall. The patient ultimately died of disease progression. CONCLUSIONS This case illustrates a rare instance of peritoneal angiosarcoma occurring after radiotherapy for cervical cancer. Clinicians should maintain a high index of suspicion for secondary malignancies in long-term cancer survivors presenting with new-onset peritoneal symptoms. Secondary peritoneal angiosarcoma should be considered in patients with prior radiation exposure and peritoneal involvement.

Two Cases of Primary Melanoma of the Uterine Cervix

BACKGROUND This study reports on 2 cases of cervical melanoma with similar presentations but at different stages, and the treatment strategy varied accordingly, and we review the literature on the characteristics, diagnosis, and management of cervical melanoma. CASE REPORT Case 1: A 69-year-old woman with abnormal vaginal bleeding was diagnosed with advanced cervical melanoma, staged as International Federation of Gynecology and Obstetrics (FIGO) Stage IVB, involving multiple metastases. Despite chemoradiotherapy and immunotherapy (nivolumab), the disease progressed rapidly, and the patient died 4 months after diagnosis. Case 2: A 75-year-old woman with cervical melanoma, staged as FIGO Stage IIIc, underwent concurrent chemoradiotherapy, surgery, and adjuvant brachytherapy. After initial stability, recurrence occurred 17 months later, followed by metastases. Palliative care was started after treatment intolerance, and the patient died 7 years after the diagnosis. CONCLUSIONS Cervical melanoma is an exceptionally rare and highly aggressive malignancy with a poor prognosis, often characterized by nonspecific symptoms. Diagnosis is based on pathological findings and immunohistochemical markers such as HMB-45, Melan-A, and S-100. The FIGO staging system is used for staging, and radical hysterectomy remains the primary treatment for achieving clear margins. Adjuvant therapies, including chemotherapy, radiation, and immunotherapy, are essential for managing advanced and metastatic cases. However, challenges persist regarding standardized treatment protocols, and further research into emerging therapies is necessary. Increasing awareness and improving diagnostic methods are vital for improving survival and quality of life for patients with this disease.

Sacral Neuromodulation for Post-Hysterectomy Hydroureteronephrosis in a Patient with Cervical Cancer: A Case Report

BACKGROUND Uterine cervical cancer in its early stage is managed with a standard treatment protocol of radical hysterectomy with bilateral pelvic lymphadenectomy. Common complications after the surgery include hydronephrosis, partially leading to renal failure. Currently, there are few reports on upper urinary tract function impairment caused by delayed lower urinary tract dysfunction after radical hysterectomy. Here, we present a case in which sacral neuromodulation (SNM) was successfully used to treat severe hydroureteronephrosis after radical hysterectomy for uterine cervical cancer. CASE REPORT A 44-year-old woman was diagnosed with bilateral hydroureteronephrosis and renal insufficiency (serum creatinine at 287 µmol/L) more than 1 year after undergoing radical hysterectomy for uterine cervical cancer. After utilizing an indwelling catheter for 3 months, her renal function showed a progressive improvement and stabilized at around 126 µmol/L. Videourodynamic study (VUDS) indicated decreased bladder compliance, bilateral ureteral reflux, with the reflux graded as IV-V. After confirmation that the upper urinary tract damage was caused by the elevated bladder pressure and not by invasion of the bladder by the primary tumor, SNM was performed to restore lower urinary tract function and to maintain low-pressure urine storage and micturition. Currently, 4 years after the surgery, the creatinine level has progressively declined to 75 µmol/L. Ultrasonography revealed no signs of hydronephrosis. Routine urine tests and post-void residual urine volumes are normal. CONCLUSIONS In select patients, sacral neuromodulation can protect the upper urinary tract, probably by improving lower urinary tract function.

Neurological and Cardiac Adverse Events in Cervical Cancer Treatment: A Case of Postoperative Sintilimab-Induced Encephalitis and Myocarditis

BACKGROUND Immune checkpoint inhibitors (ICIs) have shown considerable promise in enhancing patient outcomes and improving survival rates, offering a new frontier in cancer treatment. As a result, their use in clinical practice has become more widespread. However, the adverse effects associated with ICIs can compromise treatment efficacy. Among these, immune-related neurological adverse events are relatively uncommon, with an incidence rate of approximately 17%. Central nervous system (CNS) symptoms, although less frequent (around 6%), are particularly concerning due to their higher risk compared to peripheral nervous system involvement. Additionally, in recent years, the incidence of cardiac toxicity has been increasing, often indicated by elevated cardiac biomarkers, but most cases are asymptomatic. CASE REPORT This report presents a case of a middle-aged woman with cervical cancer who developed both encephalitis and myocarditis during postoperative consolidation therapy with an immune checkpoint inhibitor. A thorough evaluation, including laboratory tests, imaging studies, and an assessment of the patient's medical history and clinical presentation, excluded infection and paraneoplastic encephalitis as potential causes. She was treated with high-dose corticosteroids and intravenous immunoglobulin (IVIG), resulting in gradual resolution of her central nervous system symptoms and normalization of cardiac biomarkers. CONCLUSIONS Although the incidences of immune-related encephalitis and myocarditis are generally low, they can be very severe. With the increasing use of ICIs in clinical practice, the incidence of immune-related neurological symptoms may rise. This highlights the need for increased vigilance in clinical applications, including early preventive measures and prompt diagnosis and treatment to mitigate the adverse effects of these therapies, thereby maximizing patient benefits.

Challenges in Diagnosing Metastatic Uterine PEComa: Insights from Two Case Studies

BACKGROUND Perivascular epithelioid cell tumor (PEComa) is usually a benign perivascular tumor that expresses both melanocytic and myogenic cell markers. We report 2 cases of advanced malignant uterine perivascular epithelioid cell tumor (PEComa) in a 74-year-old woman and a 50-year-old woman undergoing surgery in our center. CASE REPORT Case 1: A 74-year-old woman presented with a painful and massive abdominal mass. The imaging revealed a 19-cm necrotic mass close to the mesentery, a suspicious lesion in the uterus, and a probable liver metastasis. The pathological diagnosis was quite difficult with mixed features of leiomyosarcoma and PEComa with an uncommon immunohistochemistry staining pattern. Therefore, we gave a diagnosis of sarcoma with PEComa-like features. Case 2: A 50-year-old woman with metrorrhagia and abdominal pain. Imaging revealed a 7-cm mass in the uterus and suspicious metastatic lesions in the lung and the liver. The immunohistochemistry pattern was typical, with a strong positivity of Human Melanoma Black-45 (HMB-45) and focal positivity of H-Caldesmon. The patient benefited from targeted adjuvant therapy (MTOR inhibitor-based), with 8-month a follow-up showing no recurrence for this Grade IV PEComa mutated for TP53, ATRX, and TSC1. CONCLUSIONS We have report 2 cases of metastatic PEComa with different clinicopathological features. An overlap remains between characteristics of PEComas and smooth-muscle tumors. At present, there are no known pathognomonic findings or specific diagnostic markers.

Successful Surgical Removal of a Giant Serous Ovarian Cyst in a 52-Year-Old Woman with Presurgical Body Mass Index of 50.5: A Case Report and Review of the Literature

BACKGROUND In contemporary gynecological practice, encountering giant ovarian tumors is a rarity. While most are benign and of the mucinous subtype, the borderline variant only accounts for approximately 10% of these cases. This paper addresses the paucity of information about this specific subtype, emphasizing critical elements of managing borderline tumors that can pose life-threatening complications. Additionally, a review of other documented cases of the borderline variant in the literature is also included to foster a deeper understanding of this uncommon condition. CASE REPORT We present the multidisciplinary management of a 52-year-old symptomatic woman with a giant serous borderline ovarian tumor. Preoperative assessment showed a multiloculated pelvic-abdominal cyst responsible for compression of the bowel and retroperitoneal organs, and dyspnea. All tumor markers were negative. Together with anesthesiologists and interventional cardiologists, we decided to perform a controlled drainage of the cyst of the tumor, to prevent hemodynamic instability. Subsequent total extrafascial hysterectomy, contralateral salpingo-oophorectomy, and abdominal wall reconstruction, followed by admission to the intensive care unit, were also conducted by the multidisciplinary team. During the postoperative period, the patient experienced a cardiopulmonary arrest and acute renal failure, which were managed by dialysis. After discharge, the patient underwent oncologic followup, and after 2 years, she was found to be completely recovered and disease free. CONCLUSIONS Intraoperative controlled drainage of Giant ovarian tumor fluid, planned by a multidisciplinary management team, constitutes a valid and safe alternative to the popular choice of "en bloc" tumor resection. This approach avoids rapid changes in body circulation, which are responsible for intraoperative and postoperative severe complications.

Uncommon Hematogenous Metastasis: Orbital Involvement in Uterine Cervical Cancer

BACKGROUND Although screening for uterine cervical cancer (UCC) and vaccination programs for human papilloma virus (HPV) have been implemented in many countries, women >65 years may not have access to or comply with cervical cancer screening. Women >65 years may present with advanced-stage cervical carcinoma with a poor outcome. Metastatic UCC is often diagnosed, and there are 2 types of metastases related to different treatments and survival rate: hematogenous metastasis and lymphatic metastasis. Hematogenous metastasis is relatively unusual, and it most commonly involves lung and bone locations. Orbital metastasis is an extremely rare hematogenous metastasis in patients with UCC. CASE REPORT A 70-year-old woman receiving dialysis presented to a local hospital due to general fatigue for 5 months. She was diagnosed with locally advanced UCC and underwent radiation therapy (RT). Twenty days after RT, skin masses appeared, and 34 days after RT, right exophthalmos induced by an orbital mass appeared. We diagnosed skin and orbital masses as metastases from UCC, and performed RT to the orbital tumor. The tumor shrank and the visual symptoms disappeared. Regrettably, the patient died of cancer 7 months after the orbital RT; however, no eye symptoms recurred until her death. CONCLUSIONS This report describes a rare presentation of UCC with metastasis to the orbit, and highlights that cervical cancer may still present at an advanced stage, particularly in older women. In this case, RT to the orbital metastasis from UCC was effective and contributed to the patient's quality of life.

Rectal Adenocarcinoma Presenting as a Cervical Mass: A Case Report

BACKGROUND Invasive cervical tumors are often seen in clinical practice. However, there are multiple structures within the pelvis, and invasion of the cervix from another site must be included in the differential diagnosis. In such cases, a multidisciplinary approach is needed to define the organ of tumor origin. Ensuring proper staging and histologic analysis are critical for optimal management. CASE REPORT We present a case of a 68-year-old woman who presented to her gynecologist with painless post-menopausal vaginal bleeding. She was diagnosed with a locally aggressive cervical adenocarcinoma, which was histologically confirmed by an in-office biopsy. She was referred to the gynecologic oncology service at a tertiary care hospital for definitive management, where a thorough clinical workup was performed. Physical exam revealed that the mass had invaded the anterior rectal wall. Through a multidisciplinary approach and a repeat biopsy, she was correctly diagnosed with an invasive rectal adenocarcinoma. She was treated with neoadjuvant chemoradiotherapy and underwent curative surgery. Had she been incorrectly treated as having a primary cervical adenocarcinoma, there would have been no role for surgery. The change in the organ of primary drastically altered the patient's management and outcome. She is currently undergoing surveillance with cross-sectional imaging. CONCLUSIONS Cervical masses originating from non-gynecologic organs can be difficult to differentiate on physical exam and histologic analysis. When a mass involves the rectum, an invasive primary rectal adenocarcinoma must be included in the differential. This will have a significant impact on patient management and ultimately on patient survival.

Arthroscopic Treatment of Septic Arthritis of the Ankle Caused by Group B Streptococcus: A Case Report

BACKGROUND The incidence of septic arthritis of a native joint caused by group B streptococcus (GBS, Streptococcus agalactiae) has been on the rise in non-pregnant women. GBS commonly colonizes the female genital tract. However, only a few reports have discussed serious cases of GBS infection, endocarditis, and joint infection associated with the Papanicolaou (Pap) smear test, which is routinely conducted to detect cervical cancer. Specifically, to the best of our knowledge, there have been few reports about arthroscopic treatment for septic arthritis of the ankle caused by GBS. CASE REPORT A 60-year-old woman, who had previously completed the treatment of total laparoscopic hysterectomy with bilateral adnexectomy and postoperative chemotherapy for ovarian cancer, underwent a routine Pap smear test. Four weeks later, she suddenly presented with high fever and abdominal pain. The pain and swelling in her left ankle gradually worsened. Finally, septic arthritis of the ankle was diagnosed, and thus the patient underwent emergent arthroscopic irrigation and debridement. GBS was isolated from both the ankle fluid and blood culture. After surgical intervention and intravenous antibiotic administration, the patient's symptoms gradually improved. Four months later, the patient had no ankle pain or restriction of ankle motion. CONCLUSIONS Although cervical cytology tests are essential in screening for cervical cancer, transient bacteremia can be induced by the tests. Thus, physicians must watch out for the development of septic arthritis caused by GBS when patients present with fever or swollen joints after a recent Pap smear test. Emergent diagnosis and appropriate surgical intervention is also important.

Avatrombopag Optimizes Response to Niraparib by Managing Thrombocytopenia Associated with Poly-ADP Ribose Polymerase (PARP) Inhibition in Ovarian Cancer and Breast Cancer: A Case Series

BACKGROUND Thrombocytopenia is a potentially treatment-limiting adverse event of particular interest with the PARP inhibitor niraparib. This adverse event may necessitate niraparib dose reduction or treatment discontinuation, resulting in suboptimal treatment outcomes. Here, we report on niraparib dose optimization in 2 patients with breast cancer and 4 patients with ovarian cancer through concurrent administration of the thrombopoietin receptor stimulating agent avatrombopag to mitigate thrombocytopenia, enabling niraparib reescalation and improved clinical response. CASE REPORT Three of 6 patients received niraparib 300 mg daily, the highest recommended dose, for a sustained period. Avatrombopag therapy enabled niraparib dose escalation that led to reductions in biomarkers associated with disease progression. Before initiation of avatrombopag, increases in CA-125 levels, a marker for ovarian cancer, were observed in association with niraparib dose interruption, and in 2 patients with ovarian cancer CA-125 levels fell in response to niraparib dose escalation enabled by concurrent avatrombopag therapy. Further, in 2 patients with metastatic breast cancer, intracranial response was observed in association with avatrombopag-enabled niraparib therapy. In 1 patient with metastatic breast cancer, niraparib induced an intracranial response, while previous use of talazoparib had not, confirming preclinical findings of superior blood-brain-barrier penetrance with niraparib. CONCLUSIONS Avatrombopag is currently approved for use in chronic immune thrombocytopenia and thrombocytopenia associated with chronic liver disease in patients undergoing a surgical procedure. A clinical trial of avatrombopag for chemotherapy-induced thrombocytopenia is ongoing. Preliminary results in these 6 patient cases demonstrate the need for a confirmatory trial of avatrombopag for optimizing the dose of niraparib.

Clinically Relevant Response to Treatment with Olaparib in a Patient with Refractory Multidrug-Resistant Ovarian Cancer and Central Nervous System Involvement: A Case Report

BACKGROUND Despite advances in diagnosis and treatment, epithelial ovarian cancer (EOC) continues to be highly lethal. Undoubtedly, the introduction of poly(adenosine diphosphate-ribose) polymerase inhibitors such as olaparib will alter this clinical picture. Phase III studies have already documented clinically relevant outcomes, particularly among patients with BRCA mutations and homologous recombination deficiency. CASE REPORT Here we present a case report that documents the evolution of refractory multidrug-resistant, BRCA1-mutated EOC in a patient who had advanced clinical deterioration, carcinomatosis, and central nervous system (CNS) involvement that responded favorably to olaparib, resulting in a tripling of her progression-free survival. CONCLUSIONS Olaparib proved to be a safe and effective option for the treatment of a patient with multidrug-resistant, BRCA1-mutated EOC with CNS metastases. This suggests that early initiation of the drug in similar cases can be very useful.

Breast and Axillary Lymph Node Metastasis from Ovarian Cancer: A Case Report

BACKGROUND Breast metastasis (BM) is extremely rare. Ovarian cancer accounts for approximately 0.03% to 0.6% of all BMs. BM diagnosis is challenging and the prognosis very poor. The treatment is multidisciplinary and strictly related to multiple clinical and biological factors. CASE REPORT A 70-year-old non-smoking Caucasian woman was hospitalized for a 4-month history of abdominal pain, anorexia, and weight loss of 10 kg. During the clinical examination, we found multiple axillary lymph nodes and a painless tumor lesion in the superior internal quadrant of the right breast. Whole body CT-scan and ¹⁸F-fluorodeoxyglucose PET scan documented a right ovarian tumor associated with multiple metastases, a hypermetabolic lesion of the right breast, and multiple axillary lymphadenopathies that were confirmed by breast ultrasonography. The percutaneous biopsy of both the right axillary lymph node and breast tumor showed a metastasis from a high-grade serous papillary ovarian adenocarcinoma. Considering the tumor aggressiveness and the lack of BRCA1 and BRCA2 mutations, we started systemic chemotherapy with a 3-week carboplatin/paclitaxel regimen combined with bevacizumab, which quickly improved the patient's symptoms and induced a biological tumor response. CONCLUSIONS This case reports a synchronous breast metastasis from an ovarian cancer and highlights this uncommon entity, which is very difficult to diagnose and treat. A differential diagnosis from a primary breast cancer should be considered as the treatment and prognosis of these 2 tumors are different.

Ovarian Leydig Cell Tumor: Cause of Virilization in a Postmenopausal Woman

BACKGROUND Only 0.5% of all ovarian tumors are Leydig cell tumors and they are generally benign and unilateral. These androgen-secreting tumors lead to virilizing symptoms, most often in postmenopausal women. Because Leydig cell tumors are typically small, diagnosing them accurately can be challenging. CASE REPORT We report the case of a 77-year-old woman who was referred to our Endocrinology Clinic because of a 5-year history of hirsutism (Ferriman-Gallwey score of 11) with no discernible cause. The patient had high levels of serum testosterone and a normal level of dehydroepiandrosterone sulfate. Imaging, including transvaginal ultrasound and pelvic magnetic resonance, revealed a 16-mm uterine nodule, which was suspected to be a submucous leiomyoma, but no adrenal or ovarian lesions. Despite the lack of findings on imaging and because of the high suspicion for an androgen-secreting ovarian tumor, bilateral laparoscopic oophorectomy was performed. Histological examination of the specimen revealed a non-hilar Leydig cell tumor that measured 8 mm in its largest axis. After the surgery, the patient had significant clinical improvement and her laboratory test results normalized. Her sister had the same symptoms and laboratory findings at a similar age, which raised the suspicion of a possible familial genetic syndrome. No genetic testing was performed, however, because the patient's sister declined further diagnostic investigation. CONCLUSIONS Leydig cell tumors are rare, and even when they are small, they can cause symptoms related to androgen excess. As a result, diagnosing them often is challenging.

Vulvar Extramammary Paget Disease Detected by Cytology for Cervical Cancer Screening: A Case Report and Literature Review

BACKGROUND Vulvar extramammary Paget disease (EMPD) with abnormal cervical cytology is extremely rare. We encountered a case of secondary EMPD derived from urothelial carcinoma diagnosed after cytological examination for cervical cancer screening. We diagnosed the case promptly owing to suspicion based on the patient's medical history and vulvar appearance. We report the case and present a review of published cases of EMPD with abnormal cervical cytology. CASE REPORT A 77-year-old Japanese woman visited a hospital because cervical cancer screening raised the suspicion of adenocarcinoma. Findings of the cytological examinations of the cervix, endometrium, and urethral meatus corresponded to those of other malignant neoplasms of the Bethesda system. The patient had undergone total urethral cystectomy for urothelial carcinoma 5 years earlier. In our hospital, we found erythema extending from the urethral meatus to the vulva and performed a vulvar biopsy based on the suspicion of recurrence of the urothelial carcinoma. We diagnosed secondary EMPD derived from the urothelial carcinoma based on the findings of Paget cells in the epithelium and immunohistochemistry. CONCLUSIONS A review of all the reported cases of EMPD with abnormal cervical cytology shows that the frequency of primary lesions is high in primary EMPD and secondary EMPD derived from urothelial carcinoma. These cases demonstrated the difficulty of suspecting EMPD based on cervical cytology alone. It should be considered that the cells derived from vulvar EMPD can be observed in cervical cytology, particularly in patients with a history of primary EMPD or urothelial carcinoma and with vulvar symptoms.

A 33-Year-Old Woman in the Third Trimester of Pregnancy Diagnosed with Advanced-Staged Squamous Cell Cervical Carcinoma by Magnetic Resonance Imaging and Biopsy

BACKGROUND Cervical cancer is the most common gynecologic malignancy diagnosed in pregnancy. When cervical cancer is diagnosed late in pregnancy, pelvic lymphadenectomy is avoided. Advanced imaging adds an alternative way to assess nodal involvement. The aim of this case report is to demonstrate how magnetic resonance imaging (MRI) can contribute to the clinical staging of cervical cancer and inform treatment decisions when diagnosis is made late in pregnancy. We report the case of a woman in the third trimester who was diagnosed with advanced-stage squamous cell cervical carcinoma by MRI and biopsy. CASE REPORT A 33-year-old woman at 33 weeks of gestation was admitted to our hospital for recurrent vaginal bleeding. Examination revealed a large cervical mass. A gynecologic oncologist was consulted, an examination under anesthesia was performed, and cervical biopsy samples were obtained. Pathology results revealed squamous cell carcinoma of the cervix. A clinical stage of IB3 was assumed. MRI revealed a 5.2×5.8-cm cervical mass and an enlarged left pelvic lymph node. These findings upstaged the patient to IIIC1. Instead of undergoing a radical cesarean hysterectomy, the patient had a cesarean delivery and pelvic lymph node dissection. Four weeks later, she began chemotherapy and pelvic radiation. CONCLUSIONS There is significant value in advanced imaging for cervical cancer staging. This is especially relevant in pregnancy, where the under-staging of disease is a concern. This case report demonstrates the value of MRI in cervical cancer staging, particularly in pregnant women, in whom treatment and the timing of treatment should be individualized.

Pregnancy and Delivery After Ovarian Yolk Sac Tumor: A Case Report

BACKGROUND A yolk sac tumor (YST) is a rare, malignant tumor of cells that line the yolk sac of the embryo. It most frequently occurs in the ovary (ovarian yolk sac tumor: OYST) in children and adolescents. Thus, fertility-preservation treatment is a concern. CASE REPORT A 24-year-old nulliparous woman visited us for infertility treatment and then right OYST was detected. A unilateral right salpingo-oophorectomy, infra-colic omentectomy, ipsilateral lymph node dissection, and peritoneal biopsies were performed. Histological examination confirmed the diagnosis of a stage IC OYST. Six cycles of bleomycin-etoposide-cisplatin chemotherapy were performed. She had no recurrence over the next 16 months. She conceived by in-vitro fertilization, and abdominally gave birth to a term infant. Both mother and baby had a smooth recovery. CONCLUSIONS This case adds further evidence to the 5-year survival and progression-free survival following surgery and chemotherapy in OYSTs, while preserving fertility.

Anti-N-methyl-D-aspartate Receptor Encephalitis as a Paraneoplastic Presentation of Mature Ovarian Teratoma

BACKGROUND Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is a potentially fatal form of autoimmune encephalitis that involves autoantibodies directed against the NR1 subunit of the receptor. This leads to dysregulation of neurotransmission and resultant psychotic and neuroanatomical symptoms. Anti-NMDAR encephalitis classically presents in women who have ovarian teratomas, but it also has been associated with a preceding herpes infection, testicular germ cell tumor, small cell lung cancer, and neuroblastoma. CASE REPORT The present case report illustrates the course of severe anti-NMDAR encephalitis in a patient who had poor prognostic factors, including a high anti-NMDAR titer in cerebrospinal fluid and extreme delta brush electroencephalography pattern. In addition, it underscores the importance of a multidisciplinary approach when treating these patients. CONCLUSIONS Despite being the most common form of autoimmune encephalitis, anti-NMDAR encephalitis remains underrecognized in clinical settings because of discrepancies in patient presentations and their resulting hospital courses. These variations make it difficult to devise an appropriate immunotherapy regimen and plan for intensive care management. It has been estimated that 25% of patients with anti-NMDAR encephalitis experience permanent neuropsychiatric debilitation or death even when they receive mainstay treatment. Relapse is estimated to occur in 15% to 24% of patients and is more common in individuals who do not have underlying tumors. Nonetheless, approximately 75% of patients with anti-NMDAR encephalitis recover or have only mild sequelae.

Hypercalcemia and Unilateral Ovarian Mass in a Young Adult: A Case Report of Small Cell Ovarian Carcinoma

BACKGROUND Small cell ovarian carcinoma, hypercalcemic type is an uncommon malignant ovarian tumor entity with an unfavorable prognosis and a short overall survival rate. It mainly affects women of childbearing age. CASE REPORT We report a case in which a 31-year-old woman with small cell ovarian carcinoma, hypercalcemic type presented with unspecific symptoms. We emphasize the importance of treatment planning and address fertility-sparing surgical procedures, which remain a therapeutic dilemma. CONCLUSIONS The occurrence of unspecific abdominal symptoms, unilateral tumor masses, and hypercalcemia may indicate the presence of malignant ovarian neoplasm in young adults. Histopathological examination of the mass should be performed by an experienced gynecological pathologist. A misdiagnosis can lead to inadequate surgical and adjuvant treatment. Adjuvant multi-agent chemotherapy and high-dose chemotherapy with autologous stem cell rescue may prolong the progression-free interval and overall survival.

Mixed Endometrial Stromal and Smooth Muscle Tumor of the Uterus with Unusual Morphologic Features in a 35-Year-Old Nulliparous Woman: A Case Report

BACKGROUND Mixed endometrial stromal and smooth muscle tumors (MESSMT) of the uterus are rare disease entities. The histogenesis is unclear, but its clinical manifestations are similar to those of other mesenchymal tumors. This unique uterine tumor was originally reported as having ultrastructural characteristics of both endometrial stromal and smooth muscle cells. Subsequently, MESSMT was defined as having at least 30% of each component present. Here we present an MESSMT with unusual features in a nulliparous woman and describe its morphological and immunohistochemical characteristics. CASE REPORT A 35-year-old nulliparous woman presented with menorrhagia for 2 months. Transvaginal ultrasonography and magnetic resonance imaging revealed an enlarged uterus with a 6.0×5.5×4.5 cm mass and cystic degeneration. The patient underwent abdominal mass excision. Microscopically, the tumor consisted of 3 distinct components. The outermost area consisted of smooth muscle cells. Well-differentiated endometrial stromal cells that were centrally located showed irregular borders and were merged with smooth muscle cells. In addition, the endometrial stromal component showed focal sex-cord-like differentiation. Morphological and immunohistochemical evaluations were performed, and a MESSMT with focal sex-cord-like differentiation was diagnosed. The patient's postoperative course was uneventful for 29 months. CONCLUSIONS The diagnosis of MESSMT is challenging due to its many overlapping features with other mesenchymal uterine tumors. Although this rare tumor was histologically and clinically consistent with low-grade endometrial stromal sarcoma, it can cause recurrence and metastasis. Therefore, regular follow-up with radiologic examination is essential for the timely detection of local recurrence and distant metastasis.

A Rare Case of Ovarian Serous Borderline Tumor Recurrence with Muscle Metastasis

BACKGROUND Ovarian serous borderline tumors (SBTs) generally have a favorable prognosis, with a very low recurrence rate. However, in rare cases, they can recur as invasive low-grade serous carcinoma (LGSC) after a prolonged follow-up period. Here, we report a case of LGSC originating from SBT that recurred 23 years after the initial surgery, with metastasis to the quadratus lumborum muscle - an exceptionally rare site of metastasis. CASE REPORT A 50-year-old woman, initially diagnosed with stage IIIC SBT and treated with complete tumor resection 23 years prior, presented with an asymptomatic recurrence detected by an elevated serum cancer antigen 125 (CA125) level. Contrast-enhanced computed tomography (CT) revealed multiple nodules suspected of peritoneal dissemination and a tumor infiltrating the quadratus lumborum muscle, suggesting recurrent SBT. A CT-guided needle biopsy confirmed that the tumor within the quadratus lumborum was a recurrence of SBT. Complete cytoreductive surgery was performed with the assistance of an orthopedic surgeon. Histopathological examination revealed progression to LGSC with cytoplasmic expression of the BRAF proto-oncogene (BRAF) V600E, indicating the presence of the BRAF V600E mutation, which is a characteristic feature of both SBT and LGSC. A retrospective review of CT images taken 10 years prior to the recurrence diagnosis showed a peritoneal tumor with calcification attached to the ileocecum, suggesting that the patient had remained asymptomatic for more than a decade after the actual onset of recurrence. CONCLUSIONS This case illustrates a rare instance of recurrent SBT with metastasis to the quadratus lumborum muscle. Given the exceptionally slow progression of recurrent SBT, long-term follow-up with CT imaging and serum CA125 monitoring is crucial for timely intervention and appropriate management upon recurrence.

A 40-Year-Old Woman with Inoperable Uterine Fibroids Treated with Combined Uterine Artery Embolization and Relugolix

BACKGROUND Surgery was once considered the only possible treatment for uterine fibroids. However, a more conservative treatment approach can preserve women's reproductive capacity. In recent years, uterine artery embolization (UAE) and medical treatments have been introduced as a minimally or non-invasive therapeutic option. Relugolix is a non-peptide gonadotrophin-releasing hormone (GnRH) receptor antagonist used to reduce the release of luteinizing hormone (LH) and follicle-stimulating hormone (FSH). This report presents the case of a 40-year-old woman with inoperable uterine fibroids managed with combined uterine artery embolization and Relugolix, a non-peptide gonadotrophin-releasing hormone (GnRH) receptor antagonist. CASE REPORT A woman in her 40s presented with recurrent menometrorrhagia and an 80-mm uterine fibroid causing bleeding and anemia. Due to her medical history and previous surgeries, surgery was deemed risky. Instead, a conservative approach involving UAE followed by Relugolix combination therapy (Relugolix-CT) was pursued before performing minimally invasive surgery. Following bilateral UAE, the ultrasound scan showed there had already been a reduction in the fibroid size. Right after the UAE, the patient was discharged with Relugolix-CT, which reduced the symptoms and helped further reduce the fibroid's size and vascularity. Six months later, a mini-resectoscopic myomectomy was performed under local anaesthesia. CONCLUSIONS This case underscores the effectiveness of UAE and Relugolix-CT as a pre-surgical strategy for large uterine fibroids in patients who are not immediately suitable candidates for major surgery. This new combined approach can lead to improved patient outcomes and reduced surgical risks.

Ruptured Ovarian Mature Teratoma in a Pregnant Woman with Severe Chemical Peritonitis: A Case Report

BACKGROUND Mature cystic teratomas (MCTs) account for about 25% of ovarian lesions. They are usually asymptomatic, but can complicate pregnancies if they lead to ovarian torsion or chemical peritonitis due to spontaneous rupture. CASE REPORT A 31-year-old woman who was gravida 4, para 1, aborta 1 at 26 weeks 0 days gestation presented with nonspecific, severe, acute-onset abdominal pain, which persisted despite conservative measures. Initial imaging showed a pelvic fluid collection and she was taken for a diagnostic laparoscopy, which showed purulent fluid in her pelvis. While the differential diagnosis included acute appendicitis and ruptured tubo-ovarian abscess, the source of the pain was determined to be a ruptured mature cystic teratoma. CONCLUSIONS A ruptured MCT is a reasonable addition to the differential diagnosis for pelvic pain in pregnancy. A pelvic washout during a diagnostic laparoscopy is an ideal way to manage the chemical peritonitis due to a spontaneously ruptured MCT.

Inflammatory Myofibroblastic Tumor of the Uterus: A Case Report and Literature Review

BACKGROUND Inflammatory myofibroblastic tumor (IMT) is a rare disease, and uterine IMT is even rarer. IMT is hard to distinguish from endometrial polyp and submucous myoma. The treatment of IMT is still controversial. Here, we report a case of uterine IMT, discussing both pathological and therapeutic aspects. CASE REPORT A 32-year-old woman was admitted to our hospital for a uterine mass, hypermenorrhea, and anemia. She had been suffering from these symptoms for almost a year. Pelvic ultrasound and MRI revealed a mass about 7 cm in diameter at the bottom of the uterus. Serum tumor markers were negative. She was diagnosed with submucous fibroids of the uterus. Then she underwent hysteroscopic mass resection. Histopathological and immunohistochemistry stain analysis revealed IMT of the uterus. Due to the malignant potential of IMT, she was advised to undergo a total hysterectomy, but she refused because she wanted to retain the uterus and fertility. A watch-and-wait strategy without any therapy was chosen, and the patient is currently disease-free after 18-month follow-up. CONCLUSIONS IMT is a disease with malignant potential and may recur at a late stage; hence, a correct diagnosis is essential for patients with IMT. Surgery is the preferred treatment for IMT. For early-stage, young women who want to preserve fertility, conservative surgery is acceptable, but close follow-up is required to avoid recurrence and metastasis. If a patient cannot undergo surgery or the disease has metastasized extensively, targeted therapy for ALK gene, immunotherapy, and other methods can be considered.

A Case of Primary Insular Ovarian Carcinoid Tumor with Hyperandrogenism and Carcinoid Heart Disease

BACKGROUND Carcinoid heart disease typically occurs in the presence of metastatic carcinoid tumor deposits in the liver, as vasoactive substances access the systemic circulation through the hepatic vein. Primary ovarian carcinoid tumors are rare neuroendocrine tumors, and can be associated with carcinoid syndrome and carcinoid heart disease. CASE REPORT We describe the case of a 40-year-old woman who presented with secondary amenorrhea, acne, hirsutism, and diarrhea. She was found to have a heart murmur on exam in the absence of severe symptoms of heart failure. Her investigations demonstrated elevated urinary 5-hydroxyindoleacetic acid (5-HIAA), chromogranin A, and free testosterone. Abdominal computed tomography enterography showed a large and hypervascular pelvic mass. Octreotide scintigraphy confirmed the diagnosis of primary ovarian carcinoid tumor in the setting of an intensely octreotide-avid mass with no evidence of distant metastases. Transesophageal echocardiography showed severe tricuspid regurgitation with severe dilation of the right heart chambers. She underwent a total abdominal hysterectomy and bilateral salpingo-oophorectomy. The pathology demonstrated a 14-cm carcinoid tumor of 'insular' type confined to the ovary, pT1apNX, grade 1, positive for chromogranin and synaptophysin (neuroendocrine markers) and positive mib-1 (Ki-67). Postoperatively, clinical and biochemical parameters improved significantly but her cardiac function regressed over time, resulting in a tricuspid valve replacement 6 years later. CONCLUSIONS Primary ovarian carcinoid tumors can result in carcinoid heart disease, even in the absence of liver metastases. Early diagnosis and treatment contribute to favorable outcomes.

Pregnancy Complicated by Rapidly Progressing Vulvar Melanoma: A Case Study

BACKGROUND Vulvar melanoma during pregnancy is exceptionally rare. Hormonal and immunological changes in pregnancy have raised concerns about the potential for accelerated melanoma progression and poorer maternal outcomes. This case report describes an unusual presentation of vulvar melanoma in a pregnant patient, which rapidly progressed despite previous treatments, but resulted in a favorable fetal outcome. CASE REPORT A 40-year-old G3P2A0 woman at 28 weeks of gestation, with a history of vulvar malignant melanoma diagnosed 3 years prior, presented with sudden abdominal pain and hematuria. She had previously received 6 courses of chemotherapy. Physical examination revealed a 3-cm mass in the right vulva, while ultrasonography detected a hyperechoic solid mass in the cervix and elevated LDH levels. Given the advanced disease, the medical team proceeded with a cesarean hysterectomy, colpotomy for uterine corpus involvement, and bladder repair due to an iatrogenic laceration. Histopathological findings confirmed metastatic vulvar melanoma in the cervix and uterine corpus. The pregnancy was terminated at 27 weeks due to the progression of grade IV melanoma, but the neonate was delivered in stable condition. Unfortunately, the patient died 1 month after the operation. CONCLUSIONS This case underscores the potential for aggressive melanoma progression during pregnancy, likely exacerbated by physiological changes, yet highlights a successful fetal outcome. While chemotherapy can adversely affect the reproductive system and may lead to infertility, this patient was able to conceive, and the case illustrates the complex interplay of pregnancy and cancer progression.

Successful Management of HPV-Associated Vulvar Cancer During Pregnancy: A Case Report and Treatment Approach

BACKGROUND Human Papilloma Virus (HPV)-associated Vulvar Squamous Cell Carcinomas (VSCC) present more frequently in young women than HPV- independent tumors. Due to its association with HPV infection, the incidence of vulvar cancer is increasing in young women; however, during pregnancy, it is still extremely rare. CASE REPORT We present the case of a 36-year-old pregnant woman at 23 weeks of pregnancy, diagnosed with HPV 16-associated VSCC, Federation of Gynecology and Obstetrics (FIGO) stage IB. Information on the coexistence of VSCC with pregnancy is unique, so it seems extremely important to disseminate it to develop the most effective treatment regimen. Additionally, making any decisions regarding therapeutic methods during pregnancy encounters great ethical problems. The size of the tumor was 0.5 cm with a depth of invasion 0.3 cm. The patient underwent therapy and gave birth by cesarean section at 38 weeks of pregnancy because of orthopedic indications. Surgical removal of the vulvar tumor was performed, including a margin of 1.5 cm of healthy tissue. Due to the patient's lack of consent, the sentinel node biopsy was not performed. No recurrence has been observed for 9 years. CONCLUSIONS The poorer prognosis of HPV-associated VSCC is independent of age and stage, with worse outcomes even in early-stage disease. For this reason, it is essential to sensitize clinicians to the possibility of such a diagnosis and to pay attention to the possibility of taking effective treatment during pregnancy, but safe for the fetus.

Vulvar Leiomyosarcomas: A Case Series with Clinical Comparison to Uterine Leiomyosarcomas and Review of the Literature

BACKGROUND Leiomyosarcomas of the vulva (VLMS) are very rare among gynecological malignancies, with a lack of knowledge on clinical presentation, prognosis, and therapeutic management. CASE REPORT The database of the German Clinical Center of Competence for Genital Sarcomas and Mixed Tumors in Greifswald (DKSM) was reviewed between the years 2010 and 2020. A total of 8 cases of VLMS were retrieved and analyzed retrospectively. One exemplary case of VLMS was outlined in detail: A 45-year-old premenopausal woman presented with increasing vulvar swelling and discomfort. Given the suspicion of a Bartholin's gland abscess, the mass was excised. Final pathology revealed a solid tumor consistent with a moderately differentiated leiomyosarcoma of the vulva. A wide local excision was subsequently performed followed by adjuvant external beam radiation. The clinical features of these 8 cases of VLMS were compared to 26 cases of VLMS found in a review of the literature and to a total of 276 cases of uterine leiomyosarcoma (ULMS) from the same database (DKSM). CONCLUSIONS In addition to rapid growth, observed in both tumor entities, VLMS most commonly presented as Bartholin's gland abscess or cyst and ULMS as leiomyoma. In this cohort, the prognosis of VLMS was much better than that of ULMS, most probably due to the significantly smaller tumor size of VLMS at diagnosis. Further data and larger studies on VLMS are needed to calculate recurrence and survival rates more accurately and define the role of adjuvant radiotherapy.

Adenoid Cystic Carcinoma of Bartholin’s Gland: A Case Report with Emphasis on Surgical Management

BACKGROUND Adenoid cystic carcinomas of Bartholin's gland are rare among gynecological malignancies, accounting for 0.1% to 7% of vulvar carcinomas and 0.001% of all female genital tract malignancies. There are no specific guidelines regarding treatment recommendations; therefore, they are commonly treated like vulvar cancer. CASE REPORT We present the case of a 42-year-old premenopausal woman with an adenoid cystic carcinoma of Bartholin's gland diagnosed upon biopsy of a palpable, predominantly vaginally located mass causing foreign-body sensation, vaginal pain, and extreme dyspareunia. The adenoid cystic carcinoma of Bartholin's gland was treated by radical resection in an extensive interdisciplinary surgical approach including bilateral inguinal lymph node dissection, partial posterior colpectomy, amputation of the rectum, and creation of a descendostomy, as well as reconstruction of the vagina and defect coverage using flap plastic. CONCLUSIONS With the presentation of this case, we propose a possible therapeutic approach to adenoid cystic carcinomas of Bartholin's gland with emphasis on surgical management. Especially in young patients, we recommend primary radical surgery with the objective to obtain negative resection margins. However, additional data on the adenoid cystic carcinoma of Bartholin's gland is needed to better understand its biological behavior and thus optimize and standardize treatment. The role of systematic inguinal-femoral lymphadenectomy and adjuvant and neoadjuvant treatment modalities need further evaluation.

Vulvar Paget’s Disease Presenting with Fever and Left Inguinal and Peritoneal Lymphadenopathies

BACKGROUND Asymptomatic vulvar Paget's disease is rare and commonly presents with vulvar eczema, erosions, or pruritus. The time from onset to diagnosis of vulvar Paget's disease tends to be rather long because of difficulty making a correct diagnosis owing to similar skin findings with eczema or patients' reluctance to undergo physical examination of their pubic area because of embarrassment. CASE REPORT A 55-year-old woman experienced recurrent episodes of fever for 10 months. Her primary care physician indicated inguinal lymphadenopathy 2 months prior to presentation at our hospital. Contrast-enhanced abdominal computed tomography revealed multiple intra-abdominal lymphadenopathies. With the failure of finding the primary lesion after biopsy, and with a diagnosis of metastatic carcinoma, she was referred to our hospital. On admission, she did not report having vulvar symptoms. As imaging studies revealed no primary lesions, we subsequently added immunostaining to the lymph node biopsy specimens, which suggested Paget's disease. We finally performed a vulvar physical examination and identified eczema. We performed a skin biopsy and histopathological examinations, which provided the final diagnosis of vulvar Paget's disease. CONCLUSIONS We experienced a case of vulvar Paget's disease presenting with inguinal and intraperitoneal lymphadenopathies, without a patient report of vulvar symptoms. When identifying lymphadenopathies, it is crucial to obtain a careful history and perform appropriate physical examinations, suspecting diseases of the vulva or perineum. In addition, immunostaining of lymph node biopsy specimens could be useful in making a correct diagnosis.

Late Recurrence of Metastatic Meningioma in the Lung in a Patient with Endometrial Cancer: A Case Report

BACKGROUND Meningiomas are the most frequently diagnosed of all primary brain tumors, including glioblastomas, and of all other central nervous system tumors. While non-malignant meningiomas account for 36.7% of all primary brain tumors, malignant meningioma is much less common, accounting for just 0.6%. The annual incidence of meningiomas in the United States is 5.3 per 100 000 people. The median age of diagnosis is 64, and incidence rises steadily with advancing age. Furthermore, extracranial metastatic meningioma remains extremely rare (0.1%), with the most common location for metastasis being the lung. CASE REPORT We report a case of a patient with biopsy-proven endometrial adenocarcinoma with suspicious lung nodule, Stage IVB. She was managed with chemotherapy followed by surgery and radiation. During her course of management, she was found to have progressive pulmonary nodules. Later, biopsy from the pulmonary nodules showed a metastatic meningioma. CONCLUSIONS Our case highlights the importance of early recognition of metastatic meningioma, especially when treating patients with a history of intracranial meningioma.