Journal

Journal of Medical Case Reports

Papers (75)

Complete mole coexisting with a normal fetus in a dichorionic diamniotic pregnancy: a case report

A molar pregnancy coexisting with a normal fetus is a very rare occurrence. It can present as a complete mole with a normal fetus or a partial mole with a normal fetus. There is paucity of data on optimal management of such patients who have this presentation, which mostly ends with a poor prognosis. We present a case of 26-year-old woman of African descent, a para 1 + 0 gravida 2, who came with a compete molar pregnancy coexisting with a normal fetus in the second trimester and the complexity of her management. Patient presented in second trimester for an anomaly scan, in which the complete mole coexisting with a normal fetus was diagnosed. The molar pregnancy was also covering the cervical os. The patient was advised to terminate the pregnancy due to the risk of massive hemorrhage, but she was keen on continuing with the pregnancy. She was admitted two times with bleeding and in the last admission she also had chorioamnionitis, which warranted a hysterotomy at 25 weeks 1 day. Patient made a good recovery, unfortunately the neonate had an early neonatal death at a different facility due to respiratory complications. In this case a patient with a normal fetus coexisting with a complete mole and complete previa was closely monitored, but a hysterotomy was done at 25 weeks 1 day gestation due to chorioamnionitis. The diagnosis was made by ultrasound, in which there was a snowstorm appearance of one of the gestational sacs while the other sac had a normal fetus. This is a rare entity that puts the doctor in a dilemma due to potential maternal and fetal complications. The take-home message is that a patient who presents with a complete mole and a coexisting normal fetus can be supported to a viability stage. Chorioamnionitis interfered with the continuation of this pregnancy.

Arthritis as an initial presentation of malignancy: two case reports

Abstract Background Arthritis is rarely reported as a paraneoplastic manifestation of occult malignancy. We report herein two cases of paraneoplastic arthritis due to occult malignancy. Case 1 The patient was a 65-year-old woman of asian descent who was a former smoker with a history of spine surgery performed for L4/L5 degenerative disc disease. She presented with a 1-month history of oligoarthritis affecting both ankle joints and early morning stiffness of about 3 hours. Laboratory tests were positive for antinuclear antibody at a titer of 1:320 (speckled) but negative for rheumatoid factor. She was treated for seronegative spondyloarthritis and started on prednisolone without much improvement. A routine chest radiograph incidentally revealed a right lung mass which was found to be adenocarcinoma of the lung. She was treated with gefitinib and her arthritis resolved. Case 2 The patient was a 64-year-old woman of asian descent, nonsmoker, who presented with a chief complaint of asymmetrical polyarthritis involving her right wrist, second and third metacarpophalangeal joints, and first to fifth proximal interphalangeal joints. She was treated for seronegative rheumatoid arthritis (RA) and started on sulfasalazine, with poor clinical response. Six months later, she developed abdominal pain which was diagnosed as ovarian carcinoma by laparotomy. Her arthritis resolved following treatment of her malignancy with chemotherapy. Conclusion In summary, paraneoplastic arthritis usually presents in an atypical manner and responds poorly to disease-modifying antirheumatic drugs. Accordingly, we recommend screening for occult malignancy in patients presenting with atypical arthritis.

A giant symptomatic primary ovarian leiomyoma with prominent necrosis and degenerative changes in a 75-year-old female: a case report

Adnexal masses represent a prevalent gynecological condition that may affect females of any age, although particularly those of reproductive age. These masses can be classified as either physiological or neoplastic, with the majority of adnexal neoplasms being benign. Primary ovarian leiomyoma is an exceedingly rare type of extrauterine leiomyomas, Its occurrence rate accounts for approximately 0.5-1% of all benign smooth muscle tumors found in the ovaries. These tumors are mostly asymptomatic, unilateral, non-malignant, often measuring less than 3 cm in size, and tend to be reported in women aged 20-65 years, with a considerably higher incidence in premenopausal women compared with postmenopausal women. These tumors lack distinguishing clinical features, thereby requiring comprehensive preoperative and postoperative investigations to avoid confusion with other differential diagnoses. A 75-year-old gravida 5 para 5 Syrian female reported abdominal pain, heaviness, mild vaginal bleeding, and frequent urination. She had a history of well-controlled hypertension. Physical examination identified a large abdominopelvic mass, and subsequent imaging modalities demonstrated a 24 cm × 15.5 cm mass with notable cystic components. Following adequate preoperative assessment, a total hysterectomy and bilateral salpingo-oophorectomy were performed. Microscopic study of the resected mass revealed findings suggestive of primary ovarian leiomyoma with no cell atypia, and subsequent immunohistochemical staining corroborated the diagnosis. Mitotic activity was scant, thereby excluding malignancy, and the diagnosis of primary ovarian leiomyoma was established (please refer to the graphical abstract). This case highlights the importance of considering ovarian tumors in elderly females with a palpable abdominopelvic mass. Primary ovarian leiomyoma should not be excluded on the basis of the age of the patient. The definitive diagnosis remains dependent on postoperative histological evaluation. Leiomyosarcoma should be excluded in cases of large ovarian tumors, particularly in postmenopausal women, regardless of their benign features on microscopic examination.

Small cell carcinoma of the ovary, hypercalcemic type complicated by enterocutaneous fistula: a case report

Small cell carcinoma of the ovary, hypercalcemic type, is a rare, highly aggressive malignancy that predominantly affects young women and is associated with poor prognosis and limited treatment options. We report the first documented case of small cell carcinoma of the ovary, hypercalcemic type, in a 30-year-old Pakistani woman who presented with abdominal pain, vomiting, weight loss, and biochemical evidence of hypercalcemia. Imaging revealed a large left adnexal mass measuring 9.3 cm × 10.7 cm × 9.7 cm. Subsequent histopathological and immunohistochemical analysis demonstrated characteristic features of small cell carcinoma of the ovary, hypercalcemic type with complete loss of SMARCA4 (BRG1) expression, confirming the diagnosis. The patient underwent total abdominal hysterectomy, bilateral salpingo-oophorectomy, and lymph node dissection, followed by multi-agent chemotherapy. Despite her initial response, she developed widespread metastatic disease with tumor infiltration of the abdominal wall and encasement of ileal loops. This ultimately resulted in a contained ileal perforation and the formation of an enterocutaneous fistula-an uncommon and previously unreported complication of small cell carcinoma of the ovary, hypercalcemic type. This case highlights the diagnostic and therapeutic challenges associated with small cell carcinoma of the ovary, hypercalcemic type, and underscores the critical role of SMARCA4 immunohistochemistry in confirming the diagnosis, particularly in resource-limited settings where molecular testing may not be widely accessible.

Mixed sex cord stromal tumor of the testis composed of granulosa, Leydig, and Sertoli cell tumor components: a case report

Mixed sex cord stromal tumor is defined as a tumor consisting of various combinations of sex cord stromal elements, and the tumor is extremely rare. A 76-year-old Japanese male visited our hospital complaining of left scrotal swelling. Magnetic resonance imaging of the mass showed a multilocular cystic pattern with different degrees of intensities in each cyst. The solid component was observed in part showing hypointensity on the T2-weighted image. Although there was no apparent evidence of malignancy in cytology of punctured fluid of the cystic tumor, malignant potential was not ruled out completely. Therefore, transinguinal radical orchiectomy was performed, and the tumor was diagnosed as mixed sex cord stromal tumor consisting of adult-type granulosa cell tumor, Leydig cell tumor and Sertoli cell tumor components. The patient recovered without any postoperative event. In addition, neither apparent recurrence nor metastasis was observed at 7 years after surgery. The tumor showed a multilocular cystic appearance with solid component, which was similar in appearance to previous reports of the same pathological features. Pathological findings of each component were compatible with those of mixed sex cord stromal tumor, and immunohistochemical analysis was useful for accurate diagnosis. The tumor was successfully resected, and no apparent recurrence was observed at 7 years after surgery.

Primary ovarian neuroendocrine neoplasia with concurrent large epithelial borderline ovarian tumor, coexistent with non-malignant pleural effusion and multiple uterine fibroids: a case report and review of the literature

Neuroendocrine neoplasms are a group of neoplasms often originating from the neuroendocrine cells in the gastrointestinal tract, pancreas, and respiratory tract. Neuroendocrine neoplasms rarely occur in female reproductive organs and less than a hundred cases of ovarian high-grade lesions have been reported in the literature so far. Fewer still are cases reported in the literature associated with a borderline epithelial tumor in the same ovary. Owing to the rarity of the condition, there is a lack of specific guidelines for staging, and optimal management of these tumors. We are reporting a case of primary ovarian neuroendocrine neoplasm in association with an epithelial borderline tumor. She is a 50-year-old Filipino woman who presented with nonspecific symptoms. Initial imaging revealed a large mass with suspicion of widespread metastasis. However, further imaging and laparotomy revealed early-stage neuroendocrine neoplasm, a large borderline epithelial tumor, with no evidence of pulmonary metastasis, despite having pleural effusion. She was lost to follow-up, presented again after a year with evidence of residual disease/metastasis, and was treated with chemotherapy. The case posed significant difficulty owing to a lack of typical symptoms at presentation, nonmalignant changes in lungs in imaging, and therapeutic challenges due to the noncompliance of the patient. This report highlights the importance of considering the combination of borderline tumors of the ovary with neuroendocrine carcinoma as a possible differential diagnosis in ovarian tumors, the use of imaging and specific bio-markers for early identification, timely treatment, and follow-ups.

Challenges in the diagnosis and treatment of pure non-gestational uterine choriocarcinoma in a child: a case report

Abstract Background Diagnosing non-gestational uterine choriocarcinoma in children is challenging because of its rarity and nonspecific imaging findings. Herein, we report a case of non-gestational uterine choriocarcinoma in a child, which was unexpectedly found during exploratory laparotomy and confirmed by histopathological findings. However, the tumor did not respond to chemotherapy. Case presentation A 4-year-old Indonesian female patient was brought into the emergency unit with chief complaint of vaginal bleeding. She had suffered from vaginal spotting 4 months before being admitted to the hospital. Physical examination revealed a distended abdomen in the left lumbar region and a palpable fixed mass with a smooth surface. Abdominal computed tomography scans revealed a large mass (10 × 6 × 12 cm) with fluid density and calcification. Thus, we suspected left ovarian teratoma. The patient’s luteinizing hormone, follicle-stimulating hormone, and lactate dehydrogenase levels were 25.2 mIU/ml, 0.1 mIU/ml, and 406 U/l, respectively. According to the clinical and radiological findings, we decided to perform an exploratory laparotomy and found a tumor originating from the uterus, not the ovarium. We did not observe liver nodules and any enlargement of abdominal lymph nodes. Subsequently, we performed hysterectomy. The histopathological findings supported the diagnosis of choriocarcinoma. The patient was discharged uneventfully on postoperative day 5. Thereafter, the patient underwent nine cycles of chemotherapy, including carboplatin (600 mg/m2 IV), etoposide (120 mg/m2 IV), and bleomycin (15 mg/m2 IV). However, on the basis of the clinical findings of a palpable mass and partial intestinal obstruction, the tumor relapsed soon after the ninth cycle of chemotherapy. Currently, the patient is undergoing chemotherapy again. Conclusions Although pure non-gestational uterine choriocarcinoma is rare, it should be considered as one of the differential diagnoses for intraabdominal tumors in a child, so as to better guide and counsel families regarding the surgical plan and prognosis, respectively. In the present case, the patient’s response to chemotherapy was poor, implying that the treatment of non-gestational choriocarcinoma is still challenging, particularly in the pediatric population.

Pseudomyxoma peritonei leading to “jelly belly” abdomen: a case report and review of the literature

Abstract Background Pseudomyxoma peritonei is an infrequent condition with a global annual incidence of only one to two cases per million people. Mucinous neoplasms, widespread intraperitoneal implants, and mucinous ascites characterize it. Currently, most clinicians misdiagnose this condition, which leads to delayed management. Case presentation A 44-year-old North Indian female presented with a 1.5-month history of an abdominal lump. Physical examination revealed a sizeable abdominopelvic mass at 36 weeks. Contrast-enhanced computed tomography showed a massive multiloculated right ovarian cystic mass measuring 28 × 23 × 13 cm with mild ascites and elevated carcinoembryonic antigen levels (113.75 ng/ml). A provisional diagnosis of ovarian mucinous neoplasm was made, for which the patient underwent laparotomy. Intraoperatively, there were gross mucinous ascites, along with a large, circumscribed, ruptured right ovarian tumor filled with gelatinous material. The appendicular lump was also filled with mucinous material along with the omentum, ascending colon, right lateral aspect of the rectum, splenic surface, and small bowel mesentery. Cytoreductive surgery was performed along with an oncosurgeon, including total abdominal hysterectomy with bilateral salpingoophorectomy, omentectomy, right hemicolectomy, lower anterior resection, ileo-transverse stapled anastomosis with proximal ileal loop diversion stoma, excision of multiple peritoneal gelatinous implants, and peritoneal lavage. Histopathology and immunohistochemistry confirmed the presence of intestinal-type mucinous carcinoma. Postoperatively, the patient was given six cycles of chemotherapy. She tolerated it without any specific morbidity and had an uneventful recovery. Postoperative follow-up at 15 months revealed normal tumor marker levels and abdominal computed tomography findings and no signs suggestive of local recurrence or distal metastases. Conclusions Pseudomyxoma peritonei is a rare disease that is frequently misdiagnosed in the preoperative phase. Therefore, radiologists and clinicians should maintain a high index of suspicion for accurate diagnosis and multidisciplinary management.

Mature cystic teratoma with co-existent mucinous cystadenocarcinoma: describing a diagnostic challenge—a case report

Abstract Background Mature cystic teratoma co-existing with a mucinous cystadenocarcinoma is a rare tumor that few cases have been reported until now. In these cases, either a benign teratoma is malignantly transformed into adenocarcinoma or a collision tumor is formed between a mature cystic teratoma and a mucinous tumor, which is either primarily originated from epithelial-stromal surface of the ovary, or secondary to a primary gastrointestinal tract tumor. The significance of individualizing the two tumors has a remarkable effect on further therapeutic management. Case presentation In this case, a mature cystic teratoma is co-existed with a mucinous cystadenocarcinoma in the same ovary in a 33-year-old Iranian female. Computed Tomography (CT) Scan with additional contrast of the left ovarian mass suggested a teratoma, whereas examination of resected ovarian mass reported an adenocarcinoma with a cystic teratoma. A dermoid cyst with another multi-septate cystic lesion including mucoid material was revealed in the gross examination of the surgical specimen. Histopathological examination revealed a mature cystic teratoma in association with a well-differentiated mucinous cystadenocarcinoma. The latter showed a CK7−/CK20 + immune profile. Due to the lack of clinical, radiological, and biochemical discoveries attributed to a primary lower gastrointestinal tract tumor, the immune profile proposed the chance of adenocarcinomatous transformation of a benign teratoma. Conclusions This case shows the significance of large sampling, precise recording of the gross aspects, histopathological examination, immunohistochemical analysis, and the help of radiological and clinical results to correctly diagnose uncommon tumors.

Post molar choriocarcinoma with solitary renal metastasis in the absence of primary uterine tumor: a case report and review of the literature

Abstract Background Choriocarcinoma is a rare and highly malignant form of gestational trophoblastic disease that may develop following pregnancy, abortion, or a hydatiform mole. Renal metastatic involvement by post molar choriocarcinoma is even rarer. In this case report, we describe a unique case of post molar choriocarcinoma with a solitary renal metastasis in the absence of a primary uterine tumor and metastases in other sites, which presented with urological symptoms and spontaneous renal hemorrhage. Case presentation A 41-year-old Persian woman with history of complete hydatiform mole presented with severe flank pain, nausea, vomiting, gross hematuria, and vaginal bleeding. Laboratory tests demonstrated a serum beta human chorionic gonadotropin hormone level of 60,000 mIU/mL. Imaging studies showed a lesion at the lower pole of the left kidney with active bleeding surrounded by hematoma, as well as an empty uterine cavity. Additionally, bilateral pleural effusion was detected without any lesion within the lungs. Subsequently, the patient underwent laparotomy, partial nephrectomy, and left para-ovarian cystectomy. Endometrial curettage was also carried out. The histopathology report revealed choriocarcinoma renal metastasis with high expression of beta human chorionic gonadotropin, cytokeratin 7, and Ki 67. Moreover, there were no malignant cells in the endometrial curettage specimens, and a corpus luteum cyst was found within the para-ovarian cyst. Further investigations revealed that the pleural effusion was free of malignant cells, and there was no evidence of metastatic lesions in the brain. As a result, the patient was referred to the oncology department to receive chemotherapy, and the beta human chorionic gonadotropin levels dropped to 5 mIU/mL after receiving courses of a standard regimen of etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine/oncovin over 3 weeks. Finally, monthly measurements of beta human chorionic gonadotropin levels for 6 months indicated that levels have constantly remained within normal ranges, showing no evidence of recurrence or new metastasis. Conclusions Urological symptoms such as hematuria or spontaneous renal hemorrhage might be the only presentation of post molar choriocarcinoma with renal involvement. Thus, it can be beneficial to measure serum beta human chorionic gonadotropin levels among females of childbearing age who present with unexplained urological symptoms, especially if there is a history of prior hydatiform mole.

High-grade endometrial stromal sarcoma presenting with multiple brain metastases: a case report

High-grade endometrial stromal sarcoma is a rare and aggressive subtype of uterine sarcoma, characterized by a poor prognosis. While typical initial manifestations include gynecological symptoms such as abnormal vaginal bleeding (for example, postmenopausal bleeding, intermenstrual bleeding, or menorrhagia), abdominal pain, and abdominal distension, an initial presentation involving distant metastases, particularly to the brain, is exceptionally uncommon. This case highlights a highly atypical initial presentation of this rare and challenging disease. This report details a unique clinical scenario of high-grade endometrial stromal sarcoma in a 58-year-old Japanese woman. She presented with left hemiparesis and unexplained weight loss, leading to the discovery of multiple brain tumors on cranial computed tomography. Following craniotomy and tumor resection, pathology suggested metastatic sarcoma. Positron emission tomography-computed tomography subsequently identified the uterus as the most probable primary site. Despite initial negative endometrial biopsy results, a targeted transabdominal computed tomography-guided biopsy ultimately confirmed the diagnosis of high-grade endometrial stromal sarcoma. Furthermore, detailed imaging and pathological findings raised the intriguing possibility that the high-grade endometrial stromal sarcoma may have originated from pre-existing adenomyosis. Despite initial chemotherapy and Gamma Knife radiosurgery for brain metastases, the patient's condition rapidly deteriorated, leading to her death approximately 4 months after initial presentation, highlighting the aggressive nature and poor prognosis of this disease. The inherent rarity of high-grade endometrial stromal sarcoma, combined with its highly unusual initial presentation as neurological symptoms, significantly contributed to the diagnostic delay in this case. This report critically underscores the vital importance for clinicians to maintain a high index of suspicion for high-grade endometrial stromal sarcoma in the differential diagnosis of patients presenting with brain metastases, even when typical gynecological complaints are absent.

Partial hydatidiform mole coexists with a living fetus: a case report

Hydatidiform mole is a benign gestational trophoblastic disease (GTD) that is classified into two types: complete and partial. Partial molar pregnancy is usually presented as a missed or incomplete miscarriage in the first trimester of pregnancy. A partial molar pregnancy coexisting with living molar pregnancy fetus is a rare occurrence. A dead fetus and placenta product of normal vaginal delivery arrived at our hospital. This dead fetus belonged to the first pregnancy of a 21-year-old Persian woman, without gross anomaly, male gender, and was compatible with 5-6 lunar months of pregnancy. The placenta was intact with many grape-like vesicular cystic structures. Microscopic evaluation of the placenta showed large hydropic villi with cistern formation and peripheral trophoblastic proliferation mixed with normal and small-sized villi that were compatible with partial molar pregnancy. Partial molar pregnancy has a triploid karyotype. The diagnosis and follow-up of patients with molar pregnancy are important because it has been reported that the molar pregnancy can become an invasive mole or choriocarcinoma. Many pregnancies cannot continue. If the molar pregnancy continues, its management is challenging. The coexistence of a molar pregnancy with a live fetus is a rare condition, and it increases the risk of neonatal morbidity and mortality. To achieve good outcomes, close follow-up of both the mother and fetus is advised; however, the optimal approach remains unknown.

Cecal dermoid masquerading dermoid cyst of ovary: a case report and review of the literature

Abstract Background The ovary is the most common site of occurrence of mature cystic teratomas (dermoid cysts). These are the most common ovarian germ cell tumor in the reproductive age group, accounting for 10–20% of all ovarian neoplasms, with a 1–2% risk of malignancy. A cecal dermoid cyst is a rare entity with only ten cases having been reported so far, eight of which could be retrieved as the rest were reported in different languages. None of these cases were managed laparoscopically. Here we present the first case of cecal dermoid managed laparoscopically. Case presentation A 35-year-old nulliparous Indian Hindu woman presented with complaints of on and off abdominal pain for 10 months. The abdominal examination revealed a well-defined mass of about 10 × 5 cm size, palpable in the right iliac fossa. On sonography, it was suggestive of a right-sided ovarian dermoid cyst. The lesion measured 10 × 7 × 5 cm on a contrast-enhanced computed tomogram (CT) scan. It was well defined and hypodense and located in the right lower abdomen. The ovarian tumor markers were normal. On laparoscopy, the uterus, bilateral tubes, and ovaries were found to be healthy. The cyst was seen arising from the right medial wall of the cecum at the ileocecal junction, which was excised laparoscopically. Histopathological study revealed it to be a mature cystic teratoma. Conclusion Ovarian mature cystic teratoma commonly has an indolent course and can present with palpable abdominal mass, pain, or vomiting due to complications like torsion, hemorrhage, or infection. Alternatively, these cysts can be asymptomatic and incidentally detected. Clinicians should be aware of the variety of presentations of dermoid cysts of the bowel as well as mesentery. The exact location of the teratoma eluded us till the laparoscopy despite adequate imaging including a contrast-enhanced CT scan having been performed preoperatively. We are reporting this as it is a rare entity, and this knowledge will help gynecologists and surgeons make an appropriate surgical decision.

Krukenberg tumor as an incidental finding in a full-term pregnancy: a case report

Abstract Background Krukenberg tumor is a rare metastatic tumor of the ovary with histopathological features known as signet ring cells. It usually develops in women around 45 years of age. However, here we describe an uncommon case in a 38-year-old pregnant woman. We report this case due to our unusual findings, the uncommon presentation in this younger age bracket, its diagnostic challenge, and poor prognosis. Case presentation We describe a unique case of a young Mexican woman with a history of painful vaginal bleeding at 13 weeks of pregnancy and treated for abruptio placentae. In her routine prenatal visit at week 20 of pregnancy, she was found to have a uterine fundus exceeding the expected measure for her gestational age and was referred to the hospital to discard polyhydramnios. Upon admission, a pelvic ultrasound was performed displaying normal findings in a 25-week pregnancy, and also showing bilateral enlarged ovaries with heterogeneous echogenicity. Magnetic resonance imaging revealed a left tumoral lesion with dimensions of 22.1 × 13.6 × 16.3 cm presenting regular lobulated contours with displacement of peripheral structures and mild compression of the bladder, the left ureter, and the inferior vena cava. The lesion was heterogeneous with irregular borders. The patient was scheduled for a cesarean section; during the operation, the abdominal cavity showed bilateral tumors compatible with MRI findings. The ovarian tumors were sent to pathology, and the results showed poorly differentiated mucinous adenocarcinoma (World Health Organization grade III) with extensive signet ring cells, indicative of a Krukenberg tumor. Conclusion This case report describes an uncommon example of a young pregnant woman without identifiable risk factors for gastric cancer who manifested a Krukenberg tumor. This incidental finding suggests that pregnancy obscured the cancer’s clinical appearance. The rapid deterioration in the patient’s condition corresponds to what is described in the literature. The limited information regarding this neoplasm in Mexico and the torpid evolution of the case highlight the importance of conducting additional studies to generate therapeutic interventions aimed at increasing overall survival.

Multifocal Rosai Dorfman disease and simultaneous endometrioid ovarian cancer: a case report

Abstract Background Rosai Dorfman disease is a rare histiocytosis, which is characterized by accumulation of histiocytes in lymph nodes, but also in extranodal locations. Histologically, one of the key features of Rosai Dorfman disease is emperipolesis. Treatment options involve surgical resection, radiation, chemotherapy, corticosteroids, and immunotherapy. Rosai Dorfman disease treatment depends on disease location and symptomatology. Patients with mild nodal or cutaneous involvement usually have a self-limited course and require observation only. The exact pathogenesis of Rosai Dorfman disease is still being evaluated, but mutations of the Kirsten rat sarcoma virus gene are found in up to a third of the patients and are linked to multifocal disease. Recent publications and case reports have shown that treatment with the mitogen-activated protein kinase kinase inhibitor Cobimetinib is a possible option in Rosai Dorfman disease patients with Kirsten rat sarcoma virus gene mutation. Case presentation In this case report, we are presenting and discussing the case of a 47-year-old Caucasian female patient with multifocal Rosai Dorfman disease and simultaneous endometrioid ovarian cancer, who underwent median laparotomy for tumor debulking of the endometrioid ovarian cancer and in which a Kirsten rat sarcoma virus gene mutation was detected and a treatment with Cobimetinib was initiated, leading to sustained remission. Conclusion While there are case reports on patients with Rosai Dorfman disease with a simultaneous cancer diagnosis, and while some authors describe Rosai Dorfman disease as a possible neoplastic process itself, it remains unclear if there is a causal association between Rosai Dorfman disease and cancer.

Invasive lobular carcinoma of the breast with multifocal gastrointestinal, ovarian, and peritoneal metastases: a case report

Invasive lobular carcinoma of the breast exhibits distinct biological behavior and metastatic patterns compared with invasive ductal carcinoma, with a recognized tendency to involve unusual sites such as the gastrointestinal tract, peritoneum, and ovaries. These metastases are often clinically occult and may mimic primary gastrointestinal or gynecologic malignancies, leading to delayed diagnosis. We report the case of a 43-year-old Iranian woman with estrogen receptor-positive invasive lobular carcinoma who developed sequential multifocal metastases to the stomach, ovaries, peritoneum, rectum, and jejunum over a 9-year disease course. Initial biochemical progression, manifested by a rising serum cancer antigen 15-3 level, preceded overt radiologic or endoscopic findings. Gastric metastasis was confirmed by biopsy 6 years after primary treatment. Despite negative pelvic imaging, bilateral salpingo-oophorectomy revealed occult ovarian and peritoneal metastases. Subsequent rectal and small-bowel involvement were diagnosed following new gastrointestinal symptoms. The patient was managed with multiple lines of systemic therapy and remains under active treatment with disease confined to the abdominal cavity. This case highlights the insidious metastatic behavior of invasive lobular carcinoma and the limitations of conventional imaging in detecting gastrointestinal and ovarian involvement. Persistent or unexplained gastrointestinal symptoms and rising tumor markers in patients with a history of invasive lobular carcinoma should prompt consideration of metastatic disease and early histopathological evaluation.

Synchronous atypical lobular endocervical glandular hyperplasia of the cervix and endometrium: a clinicopathologic case report and review of literature

Abstract Background Atypical lobular endocervical glandular hyperplasia is an infrequent precursor to gastric-type cervical adenocarcinoma, often posing diagnostic challenges owing to its occult nature and high misdiagnosis rate. The synchronous presentation in both the cervix and endometrium, a form of synchronous mucinous metaplasia and neoplasia of the female genital tract, is even rarer. Case presentation We report the case of a 53-year-old Chinese woman with increased vaginal discharge and postmenopausal bleeding, initially misdiagnosed as cervical and endometrial polyps. Postoperative pathology following hysteroscopy revealed synchronous atypical lobular endocervical glandular hyperplasia of the cervix and endometrium. Notably, while atypical lobular endocervical glandular hyperplasia is typically an occult finding diagnosed after cervical conization, hysteroscopy in this case enabled direct visualization and targeted biopsy, overcoming a significant diagnostic hurdle. Conclusion This case underscores the insidious clinical presentation of synchronous atypical lobular endocervical glandular hyperplasia and highlights the pivotal role of hysteroscopy as a diagnostic tool. It can facilitate the early and accurate identification of these occult lesions, thereby preventing potential misdiagnosis and guiding appropriate management.

Granulosa tumor: two spontaneous pregnancies after combined medico-surgical treatment: case report and review of the literature

Abstract Background Granulosa tumor is a rare tumor that arises from the mesenchyme and the sexual cord of the ovary. The prognosis is generally excellent, and treatment is mainly based on surgery, followed by chemotherapy depending on the extension of the disease. However, “the obstetrical prognosis” is compromised. Case presentation We report the case of a 32-year-old Caucasian patient who was diagnosed during a primary infertility assessment with an ultrasound image of a 39 mm organic left ovarian cyst confirmed on pelvic magnetic resonance imaging with infiltration of the uterosacral space. Tumor markers, including cancer antigen 125, alpha fetoprotein, and β-human chorionic gonadotropin, were normal. Histological study of biopsies of the ovarian lesion taken during exploratory laparoscopy confirmed the diagnosis of adult granulosa tumor. After a normal extension assessment including a thoracoabdominopelvic computed tomography scan and a positron emission tomography scan, the patient underwent complete conservative surgery and the disease was classified as stage Ic. Three cycles of adjuvant chemotherapy according to the “BEP” protocol combining bleomycin, etoposide, and cisplatin were performed after oocyte cryopreservation. After a 5-year follow-up period, the patient had no sign of tumor progression and had two spontaneous pregnancies, the first occurring 3 months after the end of chemotherapy and the second 14 months later. Conclusion Granulosa cell tumor remains a rare tumor whose management considerably compromises fertility and reduces the chances of having a spontaneous pregnancy. The particularity of our observation is that the diagnosis of the granulosa tumor was made following a primary infertility assessment and that the patient had two spontaneous pregnancies 3 months after the end of a medico-surgical treatment known to be very gonadotoxic.

Cutaneous metastasis from cervical cancer to the scalp and trunk: a case report and review of the literature

Abstract Background An estimated 119,300 new cases of cervical cancer occur annually in China, accounting for 372,00 deaths. Cutaneous metastasis from cervical cancer is a rare event, with an incidence of 0.1–1.3% and typically a preterminal occurrence. Scalp metastasis from cervical cancer is exceptionally anecdotal, with only a dozen examples well documented. Case presentation The patient is a 33-year-old Chinese woman who was diagnosed with International Federation of Gynecology and Obstetrics stage IVB cervical cancer in November 2021. From December 2021 to April 2022, the patient was enrolled in the clinical trial of sintilimab combined with chemotherapy and radiotherapy for treatment of stage IV cervical cancer and underwent six cycles of immunotherapy and chemotherapy (sintilimab plus paclitaxel liposome and cisplatin). Treatment was well tolerated and led to a partial response. The masses adjacent to the spine and iliac bone was largely reduced. Thus, radiotherapy of the metastatic residues was carried out and followed by radiotherapy to the primary tumor at the cervix uteri. However, by the time of the radiotherapy completion in October 2022, the patient noticed painless nodules at the left scapular region and the right hypochondrium. The following month, more nodules occurred on the scalp and trunk, including the left axilla, anterior abdomen, and left back, along with a lesion invading the sternum that caused acute bone pain. The cutaneous masses were white, discrete with a rubbery consistency, and fixed to the skin. Several nodules increased in size and eventually ulcerated. Fine‑needle aspiration cytology of the left back swellings revealed metastatic squamous cell carcinoma, P16 positive. No visceral or brain metastasis was observed at this point. Conclusions Cervical cancer metastases to the scalp are extremely uncommon. When a scalp metastasis is present, it might be the only symptomatic sign of disease progression or widespread metastatic lesions. So far, there is no clear guideline regarding skin metastases treatment. Such skin lesions warrant a thorough radiologic and pathologic workup to form a comprehensive management plan.

Aggressive (deep) angiomyxoma of the vulva: a case report

Abstract Background Aggressive angiomyxoma of the vulva is a benign, slow-growing tumor originating from myxoid cells of connective tissue. The tumor is known for multiple local recurrences with a low tendency to metastasize. Only around 350 cases have been documented in the scientific literature so far. Case presentation We report a case of a 40-year-old North Indian, unmarried woman with a swelling on the left labium majora for 7 years, along with surface ulceration over the mass. Local examination showed a well-circumscribed, 8 × 8 cm pedunculated  mass arising from the left labium majora with an overlying ulcer measuring 6 cm × 4 cm. After taking informed written consent, wide local excision of the mass and surrounding margins was done under anesthesia. Histopathology was suggestive of aggressive angiomyxoma. Immunohistochemistry was done, which was positive for estrogen and progesterone receptors. Her postoperative recovery was uneventful. The patient was given three doses of gonadotropin-releasing hormone agonist (injection, leuprolide 3.75 mg) once a month. No recurrence has been reported so far on follow-up visits for 1 year. Conclusion Aggressive angiomyxoma is one of the differential diagnoses for vulvovaginal growth in a female. As the tumor is well known for local recurrences, correct diagnosis and appropriate management using a multidisciplinary approach are crucial to managing such patients.

A frontier in oncology: managing stage IVB primary ovarian leiomyosarcoma with gallbladder metastasis: a case report and review of literature

Primary ovarian leiomyosarcoma is an exceedingly rare and aggressive tumor, accounting for less than 0.1% of ovarian malignancies. Owing to its nonspecific clinical presentation, primary ovarian leiomyosarcoma is often diagnosed at an advanced stage. To date, metastatic spread to the gallbladder has not been documented. We present the first reported case of gallbladder metastasis arising from stage IVB primary ovarian leiomyosarcoma. A 55-year-old Iranian woman initially presented with a large ovarian mass and bilateral pulmonary metastases. She underwent radical surgical management-including total hysterectomy, bilateral salpingo-oophorectomy, omentectomy, and pelvic lymphadenectomy-followed by adjuvant chemotherapy with ifosfamide and doxorubicin. Owing to persistent pulmonary lesions, her regimen was switched to gemcitabine-docetaxel, which led to a partial response. During follow-up, imaging unexpectedly revealed gallbladder lesions, which were confirmed as metastatic primary ovarian leiomyosarcoma via laparoscopic cholecystectomy and histopathological analysis. Despite further systemic therapies, including sunitinib and a return to gemcitabine-docetaxel, the disease progressed with additional metastases to the liver, spine, and pancreas. The patient died 49 months after the initial surgery. This case highlights an unprecedented metastatic pattern in primary ovarian leiomyosarcoma and suggests that individualized, multidisciplinary care may offer meaningful benefit in advanced disease. Given the rarity of primary ovarian leiomyosarcoma, cautious interpretation is warranted, and standardized treatment guidelines remain urgently needed. Vigilant long-term follow-up is crucial for detecting atypical disease progression.

Rare virilizing tumor: ovarian steroid cell tumor, not otherwise specified: a case report

Abstract Background Ovarian steroid cell tumors, not otherwise specified is a rare sex cord-stromal tumor. Almost 60% of all steroid cell tumors are categorized as not otherwise specified and represent less than 0.1% of all ovarian neoplasm. Some of them are endocrinologically active, producing virilization signs in young women. The recommended treatment is primarily surgical. Case presentation We present the case of a 20-year-old Mexican woman with secondary amenorrhea and virilization signs. She was treated with combined oral contraceptives from 13 years old, due to a misdiagnosis of polycystic ovarian syndrome. However, 4 months after stopping medication, amenorrhea and virilization signs worsened. Biochemically, she had high serum total testosterone and free testosterone levels, and a pelvic and transvaginal ultrasound followed by a pelvic tomography scan demonstrated a right adnexal tumor. She underwent right salpingo-oophorectomy and the histopathological and immunochemistry exams confirmed the diagnosis. The patient was followed for a year after surgery and until then, her menses were regular and she had no recurrence of virilization signs. Conclusion The purpose of this case report is to alert physicians to rule out ovarian steroid cell tumor, not otherwise specified diagnosis in young women with increased testosterone after discarding common causes such as polycystic ovarian syndrome. A multidisciplinary team including a gynecologist, endocrinologist, radiologist, and pathologist should be involved for correct diagnosis at the proper time.

Malignant struma ovarii with thyroid-type papillary and poorly differentiated carcinoma: a case report

Abstract Background Malignant struma ovarii is a very rare type of gynecologic cancer. Although its most common histological subtype is a pure type of papillary thyroid carcinoma containing two components, papillary carcinoma and poorly differentiated carcinoma, malignant struma ovarii is still extremely rare. As a result, the optimal treatment for this type of tumor remains uncertain due to its rarity. Case presentation A 62-year-old Japanese female presented with a pelvic tumor and clinical diagnosis of malignant tumor of the ovary. She underwent complete debulking surgery, total abdominal hysterectomy, bilateral salpingo-oophorectomy, and omentectomy. The histology of the ovarian tumor revealed malignant struma ovarii with thyroid-type papillary projections and poorly differentiated carcinoma. Because of the complete resection and the absence of distant metastasis, the patient did not receive any adjuvant therapy. At 24 months after surgery, she was free of disease. Conclusion This is a rare case report of malignant struma ovarii, without recurrence, in which the component was papillary thyroid carcinoma mixed with poorly differentiated carcinoma. Foregoing adjuvant therapy might be one option for malignant struma ovarii in cases with complete resection and no distant metastasis. In addition, we should consider that long-term follow-up is needed for malignant struma ovarii.

Rhabdomyosarcoma of the uterus in an adult patient with osteopetrosis: a case report

Abstract Background Uterine sarcoma accounts for 3–7% of uterine malignant neoplasms. It is more aggressive than epithelial neoplasms, and patients have a poor prognosis. Rhabdomyosarcoma is classified as a heterologous uterine sarcoma. It is the most common soft tissue malignancy in children while rare in adults. In young patients, the majority of genital tract rhabdomyosarcomas occur in vagina; however, the most common site of gynecologic rhabdomyosarcoma is cervix followed by uterine corpus, in adults. Uterine corpus rhabdomyosarcoma is rare in adults. Diagnosis of pure rhabdomyosarcoma in uterus involves widespread and perfect sampling as well as precise histopathological evaluation to uncover any epithelial component. Case presentation Here we report a case of pure rhabdomyosarcoma of uterine corpus in a 60-year-old Iranian postmenopausal female who had osteopetrosis, presenting with 8-month heavy vaginal bleeding and a protruding cervical mass. She is alive on 18-month follow-up after treatment. Conclusions Rhabdomyosarcoma of uterine corpus is rare in adults. Diagnosis of pure rhabdomyosarcoma in uterus involves widespread and perfect sampling as well as precise histopathological evaluation to uncover any epithelial component. Treatment options in adult gynecological rhabdomyosarcoma are based on studies in younger patients, and more studies may help us choose the best approach for improving outcome.

Mucinous cystadenoma and carcinoid tumor arising from an ovarian mature cystic teratoma in a 60 year-old patient: a case report

Abstract Background Mature cystic teratomas (MCT) of the ovary are benign ovarian germ cell neoplasms. Malignant transformation is possible but rare and ovarian carcinoid tumors in MCT are among the most extremely rare subtypes. Case presentation We report a case of a 60-year-old Iranian woman suffering from postmenopausal bleeding and hypogastric pain for the last 40 days. An adnexal mass was detected during the physical examination. Ultrasound imaging showed a (55 × 58) mm mass in the left ovary. Total abdominal hysterectomy, bilateral salpingooophorectomy and comprehensive staging surgery were performed for the patient. Intraoperative frozen section of the left ovarian mass was indicative of a malignant tumor. She was diagnosed with a carcinoid tumor with benign mucinous cystadenoma arising on MCT of the ovary, confirmed in the histopathology and immunohistochemistry examination. The tumor was classified as low grade and no chemotherapy cycles were considered. The patient was followed up long-term and no recurrence was observed during 14 months of examinations. Conclusion Ovarian carcinoids arising from MCT are rare neuroendocrine neoplasms, and proper diagnosis of these tumors requires careful histopathology evaluation and appropriate examination. Therefore, it is necessary to consider these tumors as a possible differential diagnosis and evaluate them in individuals (especially postmenopausal women) who have abdominal pain or abnormal bleeding and a palpable mass.

Exceptional lymph node recurrence of an unusual ovarian tumor 16 years later: a case report

Abstract Background Sex cord-stromal tumors with annular tubules are a rare tumor accounting for less than 1% of all ovarian malignancies. However, they are characterized by very late recurrence, which can be as late as 30 years after diagnosis and treatment. Case presentation A 16-year-old female Caucasian patient was treated in our department for a stage IA ovarian sex cord-stromal tumors with annular tubules. She underwent a left salpingo-oophorectomy and ipsilateral pelvic node biopsy with no adjuvant treatment. She was seen for amenorrhea after being lost to follow up for 16 years. The diagnosis of recurrence was made by radiology and the elevation of serum inhibin B level. The patient underwent resection of the tumor, left segmental colectomy, and paraaortic lymphadenectomy because the mass was massively adherent to the left mesocolon. Histology confirmed the diagnosis with no metastatic lymph nodes. No adjuvant therapy was indicated. The patient was lost to follow-up again for 4 years and re-presented for amenorrhea. Serum inhibin B level was high. A second recurrence was suggested, and the patient underwent a laparoscopic surgery. We performed left pelvic and paraaortic lymphadenectomy, and 3 months after surgery the patient was pregnant. Conclusion Sex cord-stromal tumors with annular tubules is a slow-growing ovarian tumor with a high potential for recurrence and metastasis. Surgery is the mainstay of treatment. Due to the rarity of these tumors, they are often unsuspected and thus incompletely staged before primary surgery; the diagnosis is made by histological examination. The prognosis of these patients is unknown, and they require long-term follow-up.

Clear cell endometrial carcinoma with high microsatellite instability in a complicated pregnancy: a case report

Abstract Background Endometrial carcinomas are the most common female genital malignancies. They are very rare in pregnancy and worldwide less than 60 cases associated with pregnancy are published. No clear cell carcinoma has been described in a pregnancy with a live birth. Case presentation We present the course of a 43-year-old Uyghur female patient with the diagnosis of endometrial carcinoma with a deficiency in the DNA mismatch repair system in the pregnancy. The malignancy with clear cell histology was confirmed by biopsy following the delivery via caesarean section due to preterm birth of a fetus with sonographically suspected tetralogy of Fallot. Earlier whole exome sequencing after amniocentesis had shown a heterozygous mutation in the MSH2 gene, which was unlikely to be related to the fetal cardiac defect. The uterine mass was initially deemed an isthmocervical fibroid by ultrasound and was confirmed as stage II endometrial carcinoma. The patient was consequently treated with surgery, radiotherapy and chemotherapy. Six months after the adjuvant therapy, re-laparotomy was performed due to ileus symptoms and an ileum metastasis was found. The patient is currently undergoing immune checkpoint inhibitor therapy with pembrolizumab. Conclusion Rare endometrial carcinoma should be included in the differential diagnosis of uterine masses in pregnant women with risk factors.

Mucinous carcinoma originating from uterine adenomyosis: a case report

Abstract Background Uterine adenomyosis is rarely a precursor of malignant tumors, but the most frequent histological subtype is endometrioid carcinoma. We observed a rare case of mucinous carcinoma originating from uterine adenomyosis. Case presentation A 63-year-old Japanese woman presented to our hospital with lower abdominal pain. She had no atypical genital bleeding. Ultrasound demonstrated thickening of the entire uterine wall, but the endometrium was not thick. Magnetic resonance imaging demonstrated an enlarged uterus with thickening of the entire uterine wall, suggesting adenomyosis. On the basis of the specimen of endocervical curettage, adenocarcinoma originating from the endometrium was suspected. Total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed to confirm the diagnosis. Macroscopically, the resected enlarged uterus had no nodules and exudation of mucin was observed from the cut surface of the thickened myometrium. The surface of the endometrium was smooth. On histological examination, mucinous carcinoma invaded almost the entire myometrium. Adenomyotic lesions were distributed focally in the uterine wall, and transition from adenomyotic glandular epithelium to mucinous carcinoma was detected within several foci. Although adenocarcinoma cells proliferated adjacent to the endometrium, the primary endometrial epithelium was atrophic without atypia. Throughout the myometrium, the mucinous carcinoma cells proliferated and floated in dilated lymph vessels with abundant mucin pools. We diagnosed this case as mucinous carcinoma originating from adenomyosis. Although the patient received 11 courses of intravenous adjuvant chemotherapy, she died of disease 18 months after the first operation. Conclusion As only one case of mucinous carcinoma originating from adenomyosis has been reported to date, this is the second case report of mucinous carcinoma. Moreover, an abnormal manner of proliferation with marked lymphatic permeation of the tumor cells throughout the myometrium was observed.

Complete remission of advanced low-grade endometrial stromal sarcoma after aromatase inhibitor therapy: a case report

Abstract Background Low-grade endometrial stromal sarcoma is a rare neoplastic growth in the uterine cavity, representing less than 1% of uterine tumors. Such tumors usually affect premenopausal and perimenopausal women, with a mean age of 46 years. Treatment generally starts with surgical resection of the tumor, followed by chemotherapy, radiotherapy, or hormonal therapy. Case presentation In the current report, we again present a case of low-grade endometrial stromal sarcoma in a 51-year-old Mediterranean woman presenting with abdominopelvic pain. Computed tomography scan revealed a primary uterine tumor measuring 17 × 9 × 9 cm metastasizing to the lungs, bladder, and ureteral orifice, along with lymphovascular involvement. The patient underwent total abdominal hysterectomy, omentectomy, and lymph node dissection. Estrogen deprivation was accomplished by bilateral salpingo-oophorectomy. Lifelong hormonal therapy consisting of letrozole 2.5 mg per day was prescribed, which demonstrated remarkable efficacy, resulting in a partial remission of lung metastasis within 8 months after surgery. Full remission was observed after 18 months of hormonal therapy, with no recurrence. Another scan was performed after 2.5 years, revealing complete remission with no recurrence. Conclusion We again report a case of complete remission of low-grade endometrial stromal sarcoma after surgical removal of the tumor along with first-line hormonal therapy without the use of chemotherapy or radiotherapy, emphasizing the role of hormonal therapy in the treatment of such tumors.

Hereditary breast and ovarian cancer triggered by occult fallopian tube cancer: a case report

Abstract Background At the time of benign gynecological surgery, a prophylactic salpingo-oophorectomy or salpingectomy is increasingly being performed concurrently to reduce the risk of future ovarian and fallopian tube cancer. We herein describe a case of hereditary breast and ovarian cancer syndrome in which a hysterectomy and bilateral adnexectomy were performed with a preoperative diagnosis of benign tumor. A detailed pathological examination revealed occult fallopian tube cancer, and additional staging surgery provided an accurate pathology diagnosis. Case presentation A 72-year-old Japanese woman with a past history of breast cancer underwent a hysterectomy and bilateral oophoro-salpingectomy for the preoperative diagnosis of uterine myoma and a right para-ovarian cyst. In the detailed pathological examination, high-grade serous carcinoma of the right fallopian tube was detected incidentally, and a subsequent staging laparotomy confirmed single para-aortic lymph node metastasis. Furthermore, a mutation in germline BRCA2 was detected postoperatively, and the patient was finally diagnosed with hereditary breast and ovarian cancer syndrome. She was diagnosed with fallopian tube cancer International Federation of Gynecology and Obstetrics Stage IIIA1(i) and started on adjuvant therapy (six courses of paclitaxel and carboplatin followed by maintenance therapy with olaparib), and 18 months after surgery, she was free of disease. Conclusion This is a case of fallopian tube cancer that was diagnosed incidentally and then accurately staged with additional advanced staging surgery. Even in the absence of grossly malignant findings, a detailed pathological search of the fallopian tubes and accurate staging surgery are important to make the necessary treatment decisions for the patient.

Uterine leiomyosarcoma diagnostic challenges in a resource-limited setting: a case report and review of the literature

Uterine leiomyosarcoma, constituting only 1-2% of all uterine malignancies, is a rare and aggressive cancer arising from the smooth muscles of the uterine wall. Owing to its rarity, identifying risk factors and clinical symptoms can be challenging, often leading to misdiagnosis as benign leiomyomas, particularly in settings with limited access to advanced imaging modalities. This case report presents a 40-year-old Ethiopian woman diagnosed with uterine leiomyosarcoma after presenting with nonspecific symptoms, underscoring the diagnostic difficulties associated with this malignancy in resource-limited environments. A 40-year-old Nulliparous woman from North Western Ethiopia presented to the Gynecologic Outpatient Department with complaints of lower abdominal pain, intermittent vaginal bleeding, and abdominal swelling lasting 1 month. Initial imaging suggested a diagnosis of intramural myoma. She underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy, with histopathological examination confirming the diagnosis of uterine leiomyosarcoma. Unfortunately, she experienced rapid recurrence, presenting 77 days postoperation with a vaginal mass, foul-smelling discharge, and fever. Despite the suspicion of local metastasis based on magnetic resonance imaging findings, the patient was lost to follow-up before definitive treatment could be initiated. This case represents the first documented instance of uterine leiomyosarcoma in East Africa, underscoring significant diagnostic and management challenges faced in resource-limited settings. The case underscores critical diagnostic challenges, particularly the difficulty in distinguishing uterine leiomyosarcoma from benign tumors such as leiomyomas. Furthermore, it reveals the impact of limited access to advanced imaging techniques on timely diagnosis and treatment. Despite complete surgical resection, the patient's follow-up revealed rapid disease recurrence, compounded by the loss to follow-up before definitive treatment could be initiated. This case emphasizes the aggressive nature of uterine leiomyosarcoma and the need for heightened clinical suspicion, particularly in low-resource environments. It calls for the development of a preoperative risk assessment tool tailored to leiomyosarcoma, enabling better differentiation from benign conditions and informing surgical management. The rapid progression of this patient's disease highlights the urgent need for effective follow-up strategies and access to comprehensive oncological care in developing regions.

Sarcomatoid transformation of uterine myoma identified through physical examination: a case report

Uterine myomas are the most common benign tumors in the female reproductive system. The incidence of malignant transformation within a uterine myoma is low, but once it occurs, the prognosis is usually poor. The clinical symptoms of this disease are not specific, which can easily lead to misdiagnosis or missed diagnosis. Therefore, early diagnosis and timely treatment are crucial for improving the prognosis of patients. A 58-year-old married Chinese female patient had undergone annual check-ups at our hospital from 2007 to 2024, totaling 18 visits. During her initial examination in 2007, uterine fibroids were detected. She experienced natural menopause in 2010. By 2019, no significant changes were observed in the size or characteristics of the uterine fibroids. In 2020, follow-up examinations revealed an increase in the size of the fibroids, although the patient reported no significant subjective symptoms. A preliminary diagnosis of sarcomatous transformation of uterine myomas was made. Subsequently, the patient underwent surgical intervention at an alternative medical facility. Postoperative pathology confirmed the diagnosis of sarcomatous transformation of uterine fibroids. As of the time of writing, she has been attending regular follow-up appointments annually since then, with the most recent visit occurring in November 2024. Currently, the patient's overall health status remains stable, with no reported discomfort. Gynecological examinations and tumor markers (carbohydrate antigen 125 [CA125], carbohydrate antigen 199 [CA199], alpha-fetoprotein [AFP], and carcinoembryonic antigen [CEA]) are all within normal limits. The malignant transformation of a uterine myoma is a rare but extremely serious complication. Timely and accurate diagnosis, followed by appropriate therapeutic interventions, is crucial for improving patient prognosis. Clinicians should maintain heightened vigilance for postmenopausal uterine fibroids that do not undergo expected regression but instead demonstrate growth.

Pulmonary metastatic gestational choriocarcinoma following an uncomplicated term pregnancy: a case report

Abstract Background Choriocarcinoma is a highly malignant pregnancy-related trophoblastic neoplasm, characterized by early metastasis to the lungs. Therefore, patients may manifest nongynecological symptoms owing to distant metastases. The incidence of choriocarcinoma after a term pregnancy is really rare (1/160,000 pregnancies). Case presentation We report a case of a 20-year-old Iranian woman, gravida 2 para 1 live 1 abortion 1, who was referred to our gynecology department with sudden onset dyspnea and pain in the left hemithorax the day after her labor. The index pregnancy was without any complications. After the initial workup, the elevation of β-human chorionic gonadotropin (HCG) levels (> 1,000,000) along with the identification of clinical (vaginal lesions) and radiological evidence of distant metastases (bilateral pulmonary nodes) directed us toward pulmonary metastatic choriocarcinoma diagnosis. After the oncology consult, the etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine chemotherapy regimen was started for the patient. She responded well to the treatment and is currently continuing her chemotherapy process. Conclusion The prognosis of choriocarcinoma is very good if the treatment is started on time. We suggest that clinicians should consider gestational trophoblastic neoplasia in their differential diagnosis of the post-natal period complications, especially after a term and nonmolar pregnancy.

A 42-year-old woman with abnormal uterine bleeding—leiomyoma (AUB-L) reporting a hemoglobin of 1.6 g/dL: a case report

Abstract Background Abnormal uterine bleeding, formerly known as menometrorrhagia, is estimated to occur in up to one-third of women, commonly at menarche or perimenopause. Among many other causes, abnormal uterine bleeding is known to be caused by leiomyomas, and is itself a leading cause of severe iron deficiency and iron deficiency anemia in women. Rarely, abnormal uterine bleeding can lead to critically low hemoglobin values of less than 2 g/dL. We report here a case of a woman with abnormal uterine bleeding caused by leiomyomas presenting with severely low hemoglobin. Case presentation We report the case of a 42-year-old Asian American woman who presented to the emergency department with chronic abnormal uterine bleeding and symptoms of anemia, including multiple syncopal episodes and abnormally pale skin but otherwise alert and oriented. Laboratory tests found a record-low hemoglobin of 1.6 g/dL and hematocrit of 6%. Transabdominal pelvic ultrasound revealed a lower uterine segment/cervical fibroid measuring 7.5 × 5 × 7.8 cm (length × depth × width). Patient was diagnosed with abnormal uterine bleeding–leiomyoma and received five units of packed red blood cells, one unit of fresh frozen plasma, Venofer infusions, tranexamic acid, and medroxyprogesterone. She was discharged from the hospital after 4 days. Conclusion To date, only a handful of cases have been reported of female patient survival following severely low hemoglobin caused by abnormal uterine bleeding. This case adds to this literature, highlighting the remarkable degree of compensation that can lead to an alert, ambulatory, and oriented patient with abnormal uterine bleeding caused by leiomyoma.

Elevated CA 125 level in a mucinous cystadenoma and a teratoma: a case report

Abstract Background The presence of a suspicious ovarian cyst with elevated cancer antigen 125 level in a woman of reproductive age poses a serious therapeutic dilemma. Mature cystic teratomas and mucinous cystadenomas may also cause an increase in cancer antigen 125. Case presentation A 43-year-old Sinhalese woman with a history of anovulatory subfertility for 5 years presented with heavy menstrual bleeding and secondary dysmenorrhea of 6 months’ duration. Imaging (pelvic ultrasound and computed tomography of the abdomen and pelvis) revealed a hemorrhagic cyst (6 × 4 cm) on the right side and a multilocular cyst with solid areas (10 × 7 cm) on the left side. Her cancer antigen 125 level was 2715 U/ml. Following a multidisciplinary team meeting, a fertility-sparing staging laparotomy was performed, which included right cystectomy, left oophorectomy, infracolic omentectomy, and peritoneal washings. Histology revealed a mucinous cystadenoma of the right ovary and a mature cystic teratoma on the left ovary. No malignant cells were observed in peritoneal washings. The patient’s cancer antigen 125 level dropped to 74.8 U/ml 1 month after surgery. Conclusion Rarely, teratomas and mucinous cystadenomas may also give rise to an extremely high cancer antigen 125 level. The risk of malignancy index and risk of malignancy algorithm may both be misleading in these instances. Therefore, multidisciplinary input, fertility-sparing surgery, and follow-up are paramount to achieve optimal treatment and patient satisfaction.

Carbon ion radiotherapy for mesonephric adenocarcinoma of the uterine cervix: a case report

Abstract Background Mesonephric adenocarcinoma is an extremely rare subtype of uterine cervical cancer that is associated with a poor prognosis and for which a standardized treatment protocol has not been established. Carbon ion radiotherapy (CIRT) is an emerging radiotherapy modality that has been shown to have a favorable anti-tumor effect, even for tumors resistant to conventional photon radiotherapy or chemotherapy. However, there is no report on CIRT outcomes for mesonephric adenocarcinoma of the uterine cervix. Case presentation We treated a 47-year-old Japanese woman with mesonephric adenocarcinoma of the uterine cervix (T2bN0M0 and stage IIB according to the 7th edition of the Union for International Cancer Control and International Federation of Gynecology and Obstetrics, respectively) with CIRT combined with brachytherapy and concurrent chemotherapy. CIRT consisted of whole pelvic irradiation and boost irradiation to the gross tumor; 36.0 Gy (relative biological effectiveness [RBE]) in 12 fractions and 19.2 Gy (RBE) in 4 fractions, respectively, performed once a day, four times per week. Computed tomography-based image-guided adaptive brachytherapy was performed after completion of CIRT, for which the D90 (i.e., the dose prescribed to 90% of the target volume) for the high-risk clinical target volume was 20.4 Gy in a total of 3 sessions in 2 weeks. A weekly cisplatin (40 mg/m2) dose was administered concomitantly with the radiotherapy for a total of five courses. From 4 months post-CIRT, the patient developed metastasis of the lung, with a total of 10 lung metastases over 70 months; these lesions were treated on each occasion by photon stereotactic body radiotherapy and/or systemic therapy. At 8 years from initial treatment (i.e., 2 years after the last treatment), the patient is alive without any evidence of recurrence and maintains a high quality of life. Conclusions This is the first report of CIRT for treatment of mesonephric adenocarcinoma of the uterine cervix. The present case indicates the potential efficacy of CIRT in combination with brachytherapy for treatment of this disease.

An isolated vaginal metastasis from rectal cancer: a case report

Abstract Introduction Vaginal metastasis from colorectal cancer is a rare occurrence, typically associated with other metastatic lesions. Isolated metastasis is exceedingly uncommon, with only a few cases documented in the literature. Vaginal involvement in colorectal cancer primarily results from direct contiguous spread from the primary tumor. Case presentation We present the case of a 70-year-old African woman diagnosed with adenocarcinoma of the middle rectum. She underwent chemotherapy, radiotherapy, and subsequent anterior resection. After 2 months, an isolated metastasis of rectal cancer was identified in the lower third of the left vaginal wall, confirmed by biopsy. Colonoscopy ruled out colorectal recurrence. Thoraco-abdominal computed tomography scan showed no distant metastases. The patient underwent abdominoperineal resection, removing the lateral and posterior vaginal wall with free macroscopic margins and a definitive colostomy. The final histopathological analysis confirmed the diagnosis of moderately differentiated adenocarcinoma of the vagina, measuring 5 × 4.5 cm. The rectal wall was extrinsically invaded by the tumor down to the muscularis propria while respecting the rectal mucosa. Resection margins were negative. The patient was discharged 1 week postoperation with no complications. Adjuvant chemotherapy was indicated, and the patient is currently tolerating the treatment well. Conclusion Vaginal metastases from colorectal cancer are extremely rare. A vigilant gynecological examination is recommended during the follow-up of colorectal cancer patients. Diagnosis can be challenging, especially if the metastatic lesion is small and asymptomatic, even after standard radiological examination. Surgical resection followed by chemotherapy is a valid option for patients with early isolated metastases.

A vocal cord palsy caused by the uterine cancer metastatic tumor in the mediastinum revealed in a patient with a thyroid lesion: a case report and review of the literature

Abstract Background The main cause of vocal cord palsy (VCP) is idiopathic impairment of the recurrent laryngeal nerve (RLN). However, solid tumors along the pathway of the RLN can also impact the nerve’s function. We presented a patient with a thyroid lesion and VCP due to a bulky metastatic mass (uterine cancer) on the aortic arch field in the mediastinum. The report aims to show the significance of comorbid tumors in thyroid pathology and the importance of additional diagnostic methods in avoiding unnecessary surgeries. A patient’s lifetime and the outcome of the disease were also presented. Case presentation A 58-year-old Ukrainian woman with a hoarse voice, intermittent dry cough, and weakness was presented to an endocrine surgeon. Thyroid pathology included signs of hypothyroidism treated with Thyroxine 112.5 µg and a nodule in the left lobe. The lesion is located on the posterior aspect of the lobe, which could probably be a cause of RLN involvement. Fine needle aspiration biopsy (FNAB) was performed twice with Bethesda category 2 result. Fibrolaryngoscopy (FLS) revealed the median position of the left vocal cord. Idiopathic, laryngeal, and thyroid causes of the VCP were excluded. Additionally, the patient displayed her anamnesis of the endometrial clear cell carcinoma following hysterectomy, external beam radiation therapy, and chemotherapy. The mediastinal metastasis was revealed sixteen years later. A chest computed tomography (CT) with intravenous contrast was done. A bulky tumor was found right under the aortic arch. Subsequently, the voice complaints reduced significantly after 4 chemotherapy courses. Cancer progression had led to the appearance of lymph node metastases on the supraclavicular region. Following six months the 60-year-old patient had passed away. Conclusion A history of the disease should always be kept in mind when assessing a patient’s complaints. VCP in case of thyroid pathology and previous secondary malignancy may be caused by metastatic tumor anywhere along the RLN pathway. Such a rare case shows the importance of additional methods of examination which may avoid unnecessary thyroid surgeries.

A Sister Mary Joseph’s nodule in fallopian tube cancer: exploring the metastatic pathway through gene expression profiling—a case report

A Sister Mary Joseph's nodule is an umbilical metastasis from an intra-abdominal or pelvic malignancy, associated with a poor prognosis. Three possible metastatic pathways for Sister Mary Joseph's nodule have been postulated: hematogenous spread, lymphatic dissemination, and direct invasion. However, detailed analyses of these metastatic pathways, particularly those involving gene expression profiling, are lacking in literature. We investigated the metastatic patterns of Sister Mary Joseph's nodule by performing RNA microarray analysis of the primary tumor and each metastatic site in a case of fallopian tube cancer presenting with Sister Mary Joseph's nodule and inguinal lymph node metastases. A 48-year-old Japanese woman presented with swelling in an inguinal lymph node. Positron emission tomography-computed tomography imaging revealed multiple lymph node metastases, right ovarian tumor, umbilical metastasis, and peritoneal dissemination. The patient underwent a laparoscopic right adnexal resection, left inguinal lymph node biopsy, and umbilical resection. Pathological examination confirmed the diagnosis of primary high-grade serous carcinoma of the right fallopian tube. Metastatic high-grade serous carcinoma was identified in the lymph nodes and umbilical tissue. Tumor tissue samples were collected from the primary lesion, umbilical metastasis, and inguinal lymph node metastasis for RNA microarray analysis. The results showed that genes involved in cell adhesion, migration, and stromal remodeling associated with the metastatic processes were more highly expressed in both inguinal lymph node metastasis and Sister Mary Joseph's nodule than in the primary lesion. Interestingly, distinct differences in gene expression profiles were observed between umbilical and lymph node metastases, suggesting different metastatic mechanisms. Our findings suggest differences in the RNA expression patterns between Sister Mary Joseph's nodule and lymph node metastases in fallopian tube cancer, indicating the possibility of distinct metastatic mechanisms. Further examination of similar cases and longitudinal studies are necessary to elucidate the metastatic patterns of Sister Mary Joseph's nodule. This case highlights the potential value of molecular profiling for understanding the complex metastatic processes in gynecological malignancies.

Diffuse large B-cell lymphoma in the uterus with unexpected manifestations: a case report

Abstract Background Lymphoid neoplasm is a common disease, arising from lymphoid cells. It is divided into Hodgkin lymphoma and non-Hodgkin lymphoma. Non-Hodgkin lymphoma can be intranodular or extranodular, which happens in 25% of primary cases. The most common locations of extranodular non-Hodgkin lymphoma are the skin and gastrointestinal tract. The genital tract is a rare location; most lymphomas arise from the cervix and vagina, while the uterine corpus is an extremely rare location. In our case, the patient was diagnosed with primary extranodular non-Hodgkin lymphoma in different locations of her genital tract. Case presentation A 48-year-old nonparous Syrian woman complained of diffuse abdominal pain, fatigue, debility, high fever, vomiting, and urinary retention for a week. The last menstrual period of the patient was 5 years previously. The physical examination showed periodic abdominal pain with severe fatigue and increased abdominal size. The laboratory investigations were within normal limits except for a low level of hemoglobin and a high level of cancer antigen 125. The radiological investigations showed a uterine sizable lobulated mass with irregular borders and high and heterogeneous density, extending to the right and left ovaries, enlargement lymph nodes around the abdominal aortic and right iliac vessels, and severe right pleural effusion with right inferior lobe atelectasis. A total hysterectomy and oophorectomy were done. The histopathological examination showed that the patient had non-Hodgkin lymphoma (primary tumor). Conclusion Primary non-Hodgkin lymphoma in the female genital tract is an extremely rare disease. Fast diagnosis and treatment can improve the outcomes, so this differential diagnosis should be in our minds even in the absence of systematic manifestations of lymphoma. More studies are needed to explain the pathology of this disease and to put guidelines that determine the perfect methods for diagnosis and treatment.

The effect of physical therapy and mechanical stimulation on dysfunction of lower extremities after total pelvic exenteration in cervical carcinoma patient with rectovesicovaginal fistula induced by radiotherapy: a case report

Abstract Background Total pelvic exenteration is the ultimate solution for rectovesicovaginal fistula caused by radiation therapy, yet total pelvic exenteration frequently causes intraoperative complications and postoperative complications. These complications are responsible for the dysfunction of lower extremities, impaired quality of life, and even the high long-term morbidity rate, thus multidisciplinary cooperation and early intervention for prevention of complications are necessary. Physical therapy was found to reduce the postoperative complications and promote rehabilitation, yet the effect on how physiotherapy prevents and treats complications after total pelvic exenteration and pelvic lymphadenectomy remains unclear. Case presentation A 50-year-old Chinese woman gradually developed perianal and pelvic floor pain and discomfort, right lower limb numbness, and involuntary vaginal discharge owing to recurrence and metastasis of cervical cancer more than half a year ago. Diagnosed as rectovesicovaginal fistula caused by radiation, she received total pelvic exenteration and subsequently developed severe lower limb edema, swelling pain, obturator nerve injury, and motor dysfunction. The patient was referred to a physiotherapist who performed rehabilitation evaluation and found edema in both lower extremities, right inguinal region pain (numeric pain rate scale 5/10), decreased temperature sensation and light touch in the medial thigh of the right lower limb, decreased right hip adductor muscle strength (manual muscle test 1/5) and right hip flexor muscle strength (manual muscle test 1/5), inability actively to adduct and flex the right hip with knee extension, low de Morton mobility Index score (0/100), and low Modified Barthel Index score (35/100). Routine physiotherapy was performed in 2 weeks, including therapeutic exercises, mechanical stimulation and electrical stimulation as well as manual therapy. The outcomes showed that physiotherapy significantly reduced lower limb pain and swelling, and improved hip range of motion, motor function, and activities of daily living, but still did not prevent thrombosis. Conclusion Standardized physical therapy demonstrates the effect on postoperative complications after total pelvic exenteration and pelvic lymphadenectomy. This supports the necessity of multidisciplinary cooperation and early physiotherapy intervention. Further research is needed to determine the causes of thrombosis after standardized intervention, and more randomized controlled trials are needed to investigate the efficacy of physical therapy after total pelvic exenteration.

Post-radical hysterectomy ovarian torsion—A gynecological emergency: a case series

Ovarian torsion is a surgical emergency whereby the ovary twists, or torts, on its ligamentous support and potentially compromises vascular supply to both the ovary and Fallopian tube. It accounts for 2-3% of all acute gynecological emergencies, yet is a condition that is rarely reported to occur after hysterectomy. While it is well established that ovarian torsion can occur after laparoscopic hysterectomy, we report on the rare occurrence of ovarian torsion occurring post the supposedly preventative ovarian transposition suspension procedure or ovariopexy. This highlights that further evaluation of laparoscopic surgical techniques are required to prevent this exquisitely painful gynecological condition occurring postoperatively. We report two cases of ovarian torsion post-primary laparoscopic radical hysterectomy performed for cervical malignancy. Both patients were Caucasian, multiparous, premenopausal female patients, aged 37 and 39 years old. Ovarian transposition suspension was performed at the primary procedure to allow ovarian function to be preserved if the patient's required postoperative pelvic irradiation if surgically upstaged. Both patients underwent emergency laparoscopic detorsion, with one torsion due to reoccurrence of her cervical cancer. Adnexal torsion is estimated to occur between 1% and 8% in post-hysterectomy cases. It can be a difficult to diagnose condition, with prudent history-taking, relevant investigations, and a low threshold to consider laparoscopy forming essential clinical acumen. Unfortunately, the suspension of the ovaries post-hysterectomy does not exclude future ovarian torsion. With rising rates of cervical cancer in premenopausal women, reportedly increasing by 37% in the 25-34 age range in the United Kingdom (National Cancer Registration and Analysis Service, Public Health England. 2021), further research is needed to improve techniques to allow for safe ovarian conservation for these patients while undergoing operative treatment of cervical cancer.

Spontaneous uterine rupture as a life-threatening presentation of molar pregnancy: a case report

Gestational trophoblastic disease is the term used to describe the heterogeneous group of interrelated lesions that arise from abnormal proliferation of placental trophoblasts. The clinical presentations of gestational trophoblastic tumor are vaginal bleeding, uterine enlargement greater than expected for gestational dates, hyperemesis, secondary clinical hyperthyroidism, and less common presentations such as pregnancy-induced hypertension in the first or second trimester and theca lutea cyst torsion. It is a rare incident to find uterine rupture due to a molar pregnancy. Most uterine ruptures reported so far were all malignant histologic types of the spectrum. We present a case of ruptured uterus as a complication of complete molar pregnancy presenting with shock and severe anemia. A 37-year-old Ethiopian gravida 6 para 5 with 5-month amenorrhea came with vaginal bleeding, abdominal pain, and symptoms of anemia. Her hemoglobin was 2.8 g/dl, and there was a honeycomb-appearing endometrial mass and free fluid in the general peritoneum. She underwent emergency laparotomy, where a hysterectomy was done for cornual uterine rupture. She was followed with serial serum human chorionic gonadotropin and declared cured after 6 months of surveillance. Even though it is rare, a ruptured uterus in a molar pregnancy can be a catastrophic complication, presenting with massive hemoperitoneum and hemorrhagic shock. Hysterectomy, along with vascular filling with crystalloid and transfusion of blood products, can save a patient's life. Patients can be followed with serial serum human chorionic gonadotropin for any transformation to gestational trophoblastic neoplasia.

Complete pathological response to olaparib and bevacizumab in advanced cervical cancer following chemoradiation in a BRCA1 mutation carrier: a case report

Abstract Background Homologous recombination deficiency is a marker of response to poly(ADP-ribose) polymerase inhibitors in different cancer types including ovary, prostate, and pancreatic cancer. To date, no report about poly(ADP-ribose) polymerase inhibitors has been published on cervical cancer. Case presentation Here we present the case of a patient with cervical cancer treated in this setting. A 49-year-old woman diagnosed with International Federation of Obstetricians and Gynecologists stage 2018 IIIC2 locally advanced undifferentiated cervical cancer received first-line chemoradiotherapy followed by carboplatin, paclitaxel, and bevacizumab with partial response. Because of a family history of cancers, the patient was tested and found positive for a pathogenic BRCA1 germline and somatic mutation, which motivated bevacizumab plus olaparib maintenance treatment. A simple hysterectomy was performed after 2 years stable disease; pathological report showed complete pathological response, and 12 months follow-up showed no recurrence. Conclusion Poly(ADP-ribose) polymerase inhibitors could be an alternative maintenance treatment for patients with persistent advanced cervical cancer previously treated with platinum, especially when familial history of cancers is reported. Clinical trials using poly(ADP-ribose) polymerase inhibitors for advanced cervical cancer are warranted.

Metastatic ovarian cancer spreading into mammary ducts mimicking an in situ component of primary breast cancer: a case report

AbstractBackgroundAccurate diagnosis of metastatic tumors in the breast is crucial because the therapeutic approach is essentially different from primary tumors. A key morphological feature of metastatic tumors is their lack of anin situcarcinoma component. Here, we present a unique case of metastatic ovarian carcinoma spreading into mammary ducts and mimicked anin situcomponent of primary carcinoma. To our knowledge, this is the second case (and the first adult case) confirming thein situ-mimicking growth pattern of a metastatic tumor using immunohistochemistry.Case presentationA 69-year-old Japanese woman was found to have a breast mass with microcalcifications. She had a known history of ovarian mixed serous and endocervical-type mucinous (seromucinous) carcinoma. Needle biopsy specimen of the breast tumor revealed adenocarcinoma displaying anin situ-looking tubular architecture in addition to invasive micropapillary and papillary architectures with psammoma bodies. From these morphological features, metastatic serous carcinoma and invasive micropapillary carcinoma of breast origin were both suspected. In immunohistochemistry, the cancer cells were immunoreactive for WT1, PAX8, and CA125, and negative for GATA3, mammaglobin, and gross cystic disease fluid protein-15. Therefore, the breast tumor was diagnosed to be metastatic ovarian serous carcinoma. Thein situ-looking architecture showed the same immunophenotype, but was surrounded by myoepithelium confirmed by immunohistochemistry (e.g. p63, cytokeratin 14, CD10). Thus, the histogenesis of thein situ-like tubular foci was could be explained by the spread of metastatic ovarian cancer cells into existing mammary ducts.ConclusionMetastatic tumors may spread into mammary duct units and mimic anin situcarcinoma component of primary breast cancer. Thisin situ-mimicking growth pattern can be a potential pitfall in establishing a correct diagnosis of metastasis to the breast. A panel of breast-related and extramammary organ/tumor-specific immunohistochemical markers may be helpful in distinguishing metastatic tumors from primary tumors.

Well-differentiated cerebellum in an ovarian mature cystic teratoma: a case report and review of the literature

Abstract Background Mature teratoma is the most common germ cell tumor as it represents 95% of germ cell tumors. Although common in children and young adults, ovarian teratoma can occur at any age. Mature teratomas are composed of mature tissues representing elements derived from more than one embryonic germ layer (ectoderm, mesoderm, and endoderm), with ectodermal derivatives being the usual predominant component; however, the finding of a well-differentiated cerebellum is extremely rare. Case presentation A 20-year-old Saudi female presented to the emergency department with severe abdominal pain of 1-day duration. Pelvic ultrasound showed a large ovoid- to bilobed-shaped cystic pelvic structure extending to the lower abdomen. The patient underwent left ovarian cystectomy. Microscopic examination showed a cyst wall with skin tissue, including adnexal structures (sebaceous glands), a well-differentiated cerebellum, and mature glial tissue. After extensive sampling, no immature component was identified. Thus, the final diagnosis of a mature cystic teratoma with well-differentiated cerebellum was established. The patient was well postoperatively and was discharged in a stable condition. Conclusion We report this case of well-differentiated cerebellum within ovarian teratoma to expand the pool of cases reported in literature of this extremely rare entity, as only 22 cases with such findings have been reported in literature to the best of our knowledge. This finding poses a diagnostic challenge to the pathologist due to its rarity and its similarity to immature teratoma. We thus emphasize that thorough sampling of ovarian teratoma is of paramount importance and to keep the aforementioned diagnosis in mind and not confuse it with immature elements, especially in intraoperative consultation and frozen sections.

Giant hamartomatous polyp of the uterine cervix with heterologous mesenchymal tissue in a child: a case report

Abstract Background Polyps of the uterine cervix are one of the most common benign hyperplastic lesions occurring in the female genital tract that usually arise from the endocervical canal and are believed to be the result of reactive changes due to long-standing chronic inflammation, multiparty, and foreign bodies. Cervical polyps are usually small in size (less than 4 cm) that are commonly found in adult women; however, a few cases of giant polyps and the rare occurrence of these lesions in children have also been reported. Heterotopias and malignant transformation in cervical polyps are considered to be very rare. Case presentation We present the case of a 10-year-old Afghan girl with a giant pedunculated mass protruding out of the uterine cervix that was accompanied by abdominal pain and mass sensation. The mass was completely excised by surgical intervention and the specimen was submitted for histopathological evaluation. Upon gross and microscopic examination, the characteristic findings of a hamartomatous polyp with heterologous mesenchymal tissue in the form of mature cartilage were seen. To the best of our knowledge, this is the first case of a giant (17.5 cm) hamartomatous polyp of the uterine cervix in this age group. Conclusion Giant hamartomatous cervical polyps rarely occur in patients below 10 years of age. The majority of these lesions are benign; however, a few cases with malignant transformation are also reported, which demands elaborate investigations into the etiopathogenesis and nature of the lesions.

Vulvar leiomyoma in an adolescent girl: a case report and review of the literature

Abstract Background Vulvar leiomyoma is a rare soft tissue tumor, with only around 300 cases described in the literature. Owing to its low incidence of just 0.03% of all gynecological tumors, it often poses a great diagnostic challenge, especially in teenagers. We report this rare occurrence of vulvar leiomyoma in a teenage girl who was primarily left untreated due to cultural taboos and fear of loss of virginity. The main aim in presenting such rare case studies is to raise awareness and expand the diagnostic horizon of the surgeon for appropriate management. Case presentation We describe a case of a 15-year-old North Indian, sexually inactive unmarried girl, who presented with a history of painless swelling in the left labia majora for the last 1 year, which gradually increased in size. There was no associated pain or any other difficulty. Local examination revealed a 14 × 10 cm solid, unilateral nonpedunculated mass on the left labia majora with superficial vascularity. Differential diagnoses of sarcoma, lipoma, Bartholin cyst, and fibroid were kept in mind. Ultrasonography revealed a solid mass with superficial vascularity with normal internal genitalia. The mass was enucleated with an intact capsule under anesthesia. Histopathology confirmed it to be benign vulvar leiomyoma. The patient was discharged after 3 days in a satisfactory condition. Conclusion Leiomyoma of the vulva is an exceptionally rare tumor and is seldom seen in teenagers. It is often misinterpreted as a Bartholin cyst and should be kept as one of the differential diagnosis in teenage girls presenting with unilateral vulvar swelling. Vulvar leiomyoma can be completely cured by surgical removal if diagnosed timely without compromising virginity, so should never be missed in adolescents.

A case of intestinal fistula following surgery for a large ovarian tumor complicated by abdominal tuberculosis: a case report and review of the literature

Abstract Background Abdominal tuberculosis is a rare and diagnostically challenging form of extrapulmonary tuberculosis that can closely mimic advanced ovarian cancer both clinically and radiologically. This case is reported to highlight this diagnostic dilemma and the serious postoperative complications that can arise, to enhance clinical awareness and reduce misdiagnosis. Case presentation A 68-year-old female of Asian ethnicity presented with a 1-year history of heartburn and significant weight loss. Preoperative evaluation revealed a large pelvic mass, elevated cancer antigen-125 (309.2 U/mL), and ascites, highly indicative of ovarian malignancy. She underwent laparoscopic exploration converted to laparotomy for left adnexectomy owing to extensive adhesions. Postoperative pathology confirmed a benign ovarian mucinous cystic adenoma and, unexpectedly, necrotic granulomatous tissue was observed in the abdominal wall tissue and mesosalpinx, consistent with tuberculosis. A total of 2 weeks post surgery, the patient developed an enterocutaneous fistula, which was managed conservatively with targeted antituberculosis therapy, prolonged drainage, and nutritional support. After over a year of treatment, the fistula healed completely and the patient regained weight, achieving a full recovery. Conclusion This case underscores the importance of considering abdominal tuberculosis in the differential diagnosis of ovarian cancer, especially in endemic areas. A high index of suspicion, utilizing a combination of diagnostic tools, and careful intraoperative assessment are crucial. Once diagnosed, conservative management with sustained antituberculosis therapy and nutritional support can lead to excellent outcomes even for complex complications such as intestinal fistula, avoiding the need for high-risk reoperation.

Publisher

Springer Science and Business Media LLC

ISSN

1752-1947