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The identification of benign prostatic tissue within ovarian and testicular mature teratomas is an unusual occurrence. While a few documented reports exist in the literature regarding the emergence of benign prostatic tissue within teratomas, the occurrence of prostatic-type adenocarcinoma in a mature ovarian teratoma is an exceptionally rare phenomenon. To date, only two prior reports have documented such instances, and no tumors have been previously reported with prostate-type tissue with morphologically two different malignancies. We outline our experience with two tumors involving prostatic-type carcinoma, both arising in ovarian mature teratomas. Microscopic examination of the first tumor revealed small areas of infiltrative atypical glandular proliferation within the mature teratoma. In the second tumor, prostate-type tissue exhibited a low-grade basal cell carcinoma. Additionally, adjacent minute foci of adenocarcinoma of the prostate (Gleason score 3 + 4 = 7, <5% pattern 4) were identified. Goblet cell adenocarcinoma of appendiceal type was also evident in the latter tumor. In both tumors, immunostains (NKX3.1, PSA) were performed to establish the prostatic origin of these atypical glands and PIN4 was performed to document the absence of basal cell in the atypical glands. On clinical follow-up, both patients have no signs of recurrence at 14 and 11 months after the surgery. Further reports on such neoplasms would contribute to a better understanding of the prognosis and management of such occurrences.
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Background: Intracranial teratomas represent a rare subset of neoplasms characterized by tissues derived from multiple germ layers within the cranial cavity. These tumors, originating from primordial germ cells, exhibit diverse clinical presentations and histopathological features. While predominantly located along the midline axis, including the suprasellar cistern and pineal region, they can also manifest in less common areas such as ventricles and hypothalamic regions. Histopathologically, they are classified as mature, immature, or malignant based on the degree of tissue differentiation. Case Description: Male patient with prenatal care for congenital hydrocephalus born at 38 weeks gestation with a bulging fontanelle. Postnatal imaging revealed an intraventricular lesion, later diagnosed through magnetic resonance imaging as a mature teratoma invading the lateral ventricle and extending to the hypothalamus. Surgical resection achieved total macroscopic removal followed by successful postoperative ventriculoperitoneal shunting due to evolving hydrocephalus. Conclusion: Teratomas are uncommon tumors, and prognosis depends on tumor size and location, especially considering the rarity of mature teratomas. Complete surgical resection is paramount for treatment, leading to a better prognosis and quicker recovery. In cases where complete removal is challenging, adjuvant therapies and cerebrospinal fluid diversion may be required to enhance therapeutic outcomes and ensure successful resection.
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BACKGROUND: The incidence of monodermal teratomas of the reproductive system is low, and most doctors lack adequate understanding, which can easily lead to missed diagnoses and/or misdiagnosis. Therefore, it is important to fully understand the clinical characteristics, diagnosis, differential diagnosis, and treatment of monodermal teratomas of the reproductive system. CASE SUMMARY: Case 1: A 14-year-old boy was admitted to the hospital with a right testicular mass for 1 wk and underwent surgical resection. He was finally diagnosed with right testicular monodermal teratoma with no special postoperative discomfort. Case 2: A 40-year-old woman was admitted to the hospital for uterine abnormalities indicated by ultrasound 20 d prior and underwent laparoscopic surgery. She was finally diagnosed with a left ovarian monodermal teratoma with a satisfactory postoperative quality of life. Case 3: A 49-year-old woman was admitted to the hospital with a pelvic mass that was discovered on B-ultrasound a week prior and underwent laparoscopic resection of the left adnexa. She was finally diagnosed with left ovarian monodermal teratoma, and her postoperative quality of life was satisfactory. CONCLUSION: Monodermal teratoma is a rare tumor whose clinical manifestations are primarily benign. Simple surgical resection of the tumor is effective.
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Retroperitoneal teratomas are uncommon neoplasms that cause diagnostic dilemmas in neonates during the evaluation of abdominal cystic mass lesions. Down syndrome (DS) is a chromosomal abnormality with an extra third copy of chromosome 21. Due to chromosomal instability, DS is inferred to be a cancer predisposition syndrome. The malignancy pattern in DS is unique with higher incidence of hematological malignancies and solid tumors are rarely reported. Down syndrome neonate was incidentally diagnosed with a retroperitoneal cystic lesion along the left kidney during evaluation for poor feeding on ultrasonography, raising suspicion of an adrenal hemorrhagic cyst. CECT abdomen and pelvis revealed complex cystic lesions along the left renal hilum, with the possibility of cystic neuroblastoma, retroperitoneal cystic teratoma, and cystic lymphangioma. Tumor markers were within normal limits. Surgical exploration revealed a left renal hilar solitary cystic mass lesion, which was excised in toto with a probable intraoperative diagnosis of cystic lymphangioma. The postoperative course was uneventful. Histopathological examination confirmed that the lesion was a mature cystic teratoma. The child is doing well postoperatively at the one-year follow-up. Neonatal retroperitoneal teratomas are unusual neoplasms. A favorable outcome can be achieved, with early diagnosis and treatment.
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Fetal intracranial teratoma presents a rare and devastating diagnosis. Typically, this condition is first detected during routine prenatal ultrasounds, appearing as an irregular heterogeneous lesion. Further insights are gained through fetal magnetic resonance imaging (MRI), better characterizing the anomaly. The combination of these modalities provides detail-oriented high resolution MRI images, while follow-up ultrasounds capture dynamic growth changes, serving as a cost-effective and easily accessible adjunct. This fast-growing tumor leads to macrocephaly and ventriculomegaly, causing severe distortion of the brain parenchyma. Early detection is crucial for effective fetal management and preventing maternal complications. Unfortunately, treatment options are limited due to the tumor's aggressive nature, typically resulting in fetal demise shortly after birth. Here, we present the sonographic and MRI findings of a congenital intracranial teratoma, reaching massive proportions and replacing the entire cerebral hemisphere.
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Introduction and relevance: Paraneoplastic hyponatremia is often secondary to syndrome of inappropriate antidiuretic hormone secretion (SIADH) by tumour cells. Immature ovarian teratomas (IOT) are uncommon and may present with SIADH. Case report: A 26-year-old female presented with a 3-month history of abdominal pain and constipation. Imaging identified a mixed solid-cystic right ovarian mass containing fat and peritoneal deposits. Biochemistry showed severe, refractory hyponatremia (117 mmol/l). She underwent diagnostic fertility-preserving right salpingo-oophorectomy and resection of peritoneal nodules with the aim to achieve symptom control and hyponatraemia resolution. Pathology revealed a FIGO Stage 2 Grade 2 IOT with extensive benign peritoneal gliomatosis. Initial management was conservative. After 6 months of active follow-up, a rise in AFP, and recurrent hyponatremia supported the decision to administer three cycles of Bleomycin-Etoposide-Cisplatin chemotherapy. One month later, given radiological disease progression despite satisfactory biomarker response, cytoreductive surgery with complete macroscopic resection was performed. Pathology consisted solely of peritoneal mature glial elements: a growing teratoma syndrome (GTS). The patient remains disease-free after 2 years of surveillance. Clinical discussion: Specimen histological assessment from the patient's initial surgery showed immature neuroectodermal tubules, which are thought to be the source of vasopressin secretion. The authors hypothesise that recurrent hyponatremia and rising AFP levels represented postoperative disease relapse. Biochemical response despite radiological disease progression was pathognomonic of a GTS. Conclusion: Paraneoplastic SIADH secondary to an IOT must be considered in female patients presenting with abdominal symptoms and hyponatremia. Management requires a multidisciplinary approach. Serum electrolytes are useful surveillance biomarkers supplementary to tumour markers.
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PURPOSE: The aim of this case report is to emphasize the significance of the growing teratoma syndrome. Growing teratoma syndrome is frequently misdiagnosed due to its low prevalence, with an estimated incidence of 19% among all immature ovarian teratomas and a lack of experience among healthcare professionals. It is characterized by the growth of benign tumoral tissue during or after chemotherapy for malignant germ cell tumors. CASE REPORT: Our case is about a 46-year-old patient diagnosed with an immature teratoma who was treated unsuccessfully with surgery and chemotherapy. The patient was then referred to our hospital for a second opinion, where this unknown entity was diagnosed and underwent complete surgical debulking, including abdominal wall resection and subsequent repair. CONCLUSION: Physicians need to be aware of rapidly growing masses during or after chemotherapy because early recognition of this syndrome is essential for the adequate treatment of our patients.
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INTRODUCTION AND IMPORTANCE: Oropharyngeal teratoma is a rare congenital tumor that grows slowly and can be suspected prenatally. It entails the obstruction of upper airway and upper digestive tract. Clinical examination is always completed by imaging particularly the MRI. CASE PRESENTATION: We present a case of an eight- month -old infant who presented an oropharyngeal mass which caused intermittent dyspnea and feeding difficulties. DISCUSSION: The treatment is only surgical and histopathological examination confirms the diagnosis. CONCLUSION: Early diagnosis is key to successful management of oropharyngeal teratomas.
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Intracranial growing teratoma syndrome (iGTS) is a rare phenomenon in patients with non-germinomatous germ cell tumor (NGGCT) after chemotherapy or radiotherapy. It manifests as paradoxical growth of teratomatous components, with multiple cystic lesions on cranial imaging despite normalized tumor markers. This paper presents a 22-year-old male with iGTS, diagnosed one month after chemotherapy against NGGCT. Initially diagnosed with presumptive pineal NGGCT causing obstructive hydrocephalus, the patient underwent endoscopic third ventriculostomy and extraventricular drainage with tumor biopsy followed by two chemotherapy cycles. Despite normalization of tumor markers, follow-up MRI showed increased tumor size with honeycomb-like cystic patterns. The patient underwent suboccipital craniotomy for tumor removal via combined telovelar and infratentorial supracerebellar approaches. The final pathology confirmed mature teratoma. However, postoperative bleeding and left thalamic infarction occurred, resulting in severe neurological deficits. Despite challenges, the patient eventually regained the ability to follow simple commands. To understand iGTS pathophysiology, several hypotheses, including the differentiation of immature components and the uninhibited growth of mature components induced by chemotherapy or radiotherapy, were explored. Surgical intervention remains as an ideal treatment, while clinical trials investigate chemotherapy options. Frequent imaging follow-ups are crucial for early detection in iGTS for NGGCT patients.
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Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is the most common form of autoimmune encephalitis, presenting with various psychiatric manifestations, including behavioral and cognitive impairments, movement disorders, decreased consciousness, dysphasia, seizures, and autonomic dysfunction. Autonomic dysfunction may involve hyperthermia, apnea, hypotension, tachycardia, and life-threatening manifestations of sinus node dysfunction (SND), such as bradycardia, sinus pause or arrest, and asystole. The severity and significance of SND are critical, as it is not uncommon for these patients to progress into asystolic cardiac arrest, potentially contributing to morbidity and mortality. Accordingly, we present the case of an 18-year-old female with anti-NMDAR encephalitis who experienced multiple episodes of sinus pause/arrest and asystolic cardiac arrest, achieving a return of spontaneous circulation after successful CPR in all instances, ultimately requiring permanent pacemaker implantation. Additionally, we performed a literature review and analyzed 23 similar anti-NMDAR encephalitis cases with SND manifestations, including sinus pause/arrest or asystolic cardiac arrest, to identify common risk factors and describe management strategies and outcomes. Moreover, we investigated the potential association between teratoma and permanent pacemaker use in SND.
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Malignant transformation (MCT) of ovary is rare complications affecting elderly, squamous cell carcinoma being the most common. The prognosis worsens with extraovarian spread. We present two cases of MCT-derived SCC. Patients exhibited abdominal lump, pain, bowel symptoms, sometimes with weight loss; imaging revealed MCT. Age (51-60), postmenopausal status, large size (>20 cm), bilaterality, and complex ovarian lesions raised suspicion of malignancy. Elevated tumor markers (e.g., cancer antigen-125 and lactate dehydrogenase) were noted in one case. Intraoperative frozen section confirmed malignancy, guiding staging laparotomy. One case was advanced stage on histopathology. Intraoperative frozen section aids optimal staging.
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Testicular tumors are rare in children, representing a small percentage of pediatric solid tumors, with an incidence of 2 cases per 100,000 males. Teratomas, which are the most prevalent tumors in infants, may manifest in mature, immature, or malignant forms. While mature teratomas are typically found in the abdomen, intratesticular prepubertal-type teratomas in infants are infrequent. The present study describes the case of an infant with an intratesticular mature teratoma. A 6-month-old male infant presented with right-sided scrotal swelling, which was noted by his parents. There was no family history of similar conditions, and an investigation of his medical history did not reveal any notable findings. A physical examination revealed a non-reducible, solid mass indistinguishable from the right testicle, with no signs of inflammation or systemic symptoms. A scrotal sonography confirmed a large intratesticular cyst. The levels of α-fetoprotein and ß-human chorionic gonadotropin were normal. Surgical tumor enucleation was performed, and the histopathological examination revealed a benign, prepubertal-type teratoma composed entirely of mature elements. Surgical intervention is commonly used for the management of benign testicular tumors in pediatric patients, including prepubertal teratomas. This approach demonstrates an excellent prognosis as it does not elevate the likelihood of recurrence. Prepubertal-type teratomas have rarely been reported in the infantile testis. They may present as a solid mass indistinguishable from the testicle, with no signs of inflammation.
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Background: Spiradenocylindroma is a benign tumor of skin adnexal origin with overlapping features of two distinct neoplasms: spiradenoma and cylindroma. This cutaneous tumor typically presents on the head and neck and extracutaneous presentations are uncommon. The presentation described below involves a spiradenocylindroma within a mature ovarian teratoma is very rare. Aim: The aim of this article is to portray the diagnostic process of this unusual spiradenocylindroma presentation. Case presentation: A 65 year-old female with a left adnexal mass underwent ultrasonography and magnetic resonance imaging (MRI) which showed a left ovarian multiseptated lesion, with mural calcifications and projections into the mass. Excisional surgery was performed and histopathological examination revealed a spiradenocylindroma. Conclusion: Spiradenocylindroma is rare, hard to identify, and often misdiagnosed. Our study described the process of diagnosis and depicts the rare presentation of this lesion arising within a mature teratoma.
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Background: Mature intrapulmonary cystic teratomas are rare. The clinical manifestations are nonspecific and may mislead to other diseases. The radiographic findings are often perplexing. There are few complete reports on intrapulmonary teratomas, including patient history, radiologic and pathologic findings, patient management, and outcomes. We present a case report of an intrapulmonary mature cystic teratoma diagnosed at our hospital, along with an extensive review of the relevant literature. Case presentation: A 47-year-old non-smoking female patient presented with hemoptysis and intermittent dyspnea for 5 months without fever or weight loss. Chest computed tomography revealed an inhomogeneous, hypodense, lobulated mass with internal fat, calcification, and soft tissue components. The patient underwent a left thoracotomy and left upper lobe lobectomy. Pathological examination revealed a cystic tumor containing various components, including fat, keratin debris, teeth, and hair. A diagnosis of intrapulmonary mature cystic teratoma was made. Three months after the operation, no recurrent tumor was identified, and the patient was scheduled for follow-up next year. Conclusion: Intrapulmonary mature cystic teratomas are unusual. Owing to its general clinical symptoms and radiographic findings, it was misdiagnosed before surgery. After surgery, pathological and radiological findings confirmed the diagnosis. Therefore, intrapulmonary mature cystic teratomas should be considered in the differential diagnosis, and sufficient examinations should be conducted to rule out the condition.
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BACKGROUND: Testicular germ cell tumor (TGCT) is the most common type of tumor in young men. Type II germ cell tumors including postpubertal-type teratomas are derived from the germ cell neoplasia in situ (GCNIS), whereas prepubertal-type teratomas arise independently of the GCNIS. The consomic mouse strain 129.MOLF-Chr19 (M19) is a suitable murine model of such tumors, but its characterization remains incomplete. OBJECTIVE: Here, we interrogated the suitability of testicular tumors in M19 mice as a model of human TGCT by analyzing their histological features and gene expression signature. MATERIAL AND METHODS: Testes collected from M19 mice of different ages were categorized by macroscopic appearance based on size and the degree of suspected tumorigenesis. Histological sections from selected tumors were stained with Hematoxylin and Eosin, and expression of genes associated with tumorigenesis was determined in frozen tissue samples from a large range of tumors of different subclasses using RT-qPCR and Fluidigm Dynamic Arrays. RESULTS: Macroscopically, testicular specimens appeared very heterogeneous concerning size and signs indicating the presence of a tumor. Histological analysis confirmed the development of teratomas with areas of cells corresponding to all three germ cell layers. Gene expression analyses indicated upregulation of markers related to proliferation, vascular invasive potential and pluripotency, and revealed a strong correlation of gene expression with tumor size and a significant intercorrelation of individual genes. DISCUSSION AND CONCLUSION: TGCT in M19 mice is reminiscent of human testicular teratomas presenting with areas of cells derived from all germ layers and showing a typical gene signature. We thus confirm that these mice can serve as a suitable murine model of pure teratomas for preclinical research.
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STUDY OBJECTIVE: The main objective is to evaluate the feasibility of laparoscopic fertility-sparing surgery in women with growing teratoma syndrome. DESIGN: Retrospective cohort study. SETTING: Chinese tertiary university hospital. PATIENTS: Patients with growing teratoma syndrome who underwent fertility-sparing surgery between January 2015 and August 2023. INTERVENTIONS: Baseline characteristics and surgical outcomes were evaluated, including clinical information, surgical procedures, operative time, intraoperative blood loss, complications, length of hospital stay, and follow-up information. MEASUREMENT AND MAIN RESULTS: Twenty-six patients with ovarian growing teratoma syndrome underwent fertility-sparing surgery: 12 had laparoscopic surgery and 14 underwent laparotomic surgery. In the laparoscopic group, the median age of the patients during initial management of immature teratoma or mixed malignant ovarian germ cell tumor was 14.0 years (interquartile range: 13.0-24.5 years). Eleven patients were nulliparous. The primary ovarian tumor was pure immature teratoma in 10 patients and mixed ovarian germ cell tumor in 2 patients. Complete laparoscopic tumor resection was achieved in 11 patients. Patients in laparoscopic group had shorter median operative time (76.5 vs 180.0 minutes, pâ¯=â¯0.001), lower estimated blood loss (20.0 vs 400.0 ml, p < 0.001), decreased postoperative hospital stay (2.0 vs 7.0 days, p < 0.001), compared with laparotomic surgery. There was no conversion to laparotomy and no perioperative complications. Histological examination confirmed mature teratoma in all cases. During a median follow-up of 21.9 months (interquartile range: 7.6-44.9 months), 11 patients were alive without disease and 1 was alive with disease. One pregnancy was achieved postoperatively. CONCLUSION: Laparoscopic fertility-sparing surgery may represent a feasible option in well-selected patients with ovarian growing teratoma syndrome. Surgery should be performed in gynecologic oncology centers by experienced staff trained in endoscopic procedures. More research and long-time follow-up are needed to determine the oncological outcomes and safety of laparoscopic surgery in this population.
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Growing teratoma syndrome (GTS) is a rare condition that arises secondary to malignant germ cell tumors. It is characterized by an enlarging abdominal mass during or after chemotherapy, normal tumor markers, and histopathological indications of mature teratoma components. Awareness of GTS is limited, and it is often mistaken for disease progression or recurrence. This misdiagnosis can lead to delayed treatment and increased risk of complications. Therefore, early identification of GTS is crucial to avoid unnecessary systemic treatments and reduce financial burden. GTS is unresponsive to chemotherapy or radiotherapy and complete surgical resection is the sole therapeutic strategy. In this report, we present a case of GTS in a 20-year-old female following treatment for immature teratoma, alongside a review of the relevant literature aimed at enriching our insight into the clinical manifestations of GTS.
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We present a rare case of a 40-year-old nulliparous lady, with no past medical or surgical history, who was diagnosed with metastatic squamous cell carcinoma of the right ovary that originated from a mature cystic ovarian teratoma. Our patient underwent debulking total abdominal hysterectomy, bilateral salpingo-oophorectomy and partial omentectomy followed by postoperative carboplatin and paclitaxel chemotherapy. Rapid disease progression ensued, complicated by severe parathyroid hormone-related protein-induced hypercalcaemia resistant to medical therapy. The patient was treated in a palliative manner and died five months after her diagnosis.