Cases reported "Teratoma"

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1/21. Establishment and characterization of human immature teratoma cell line (TES-1).

    A new human immature teratoma cell line, TES-1, was established from a surgical specimen from a 12-year old male with third ventricular immature teratoma. TES-1 shows polygonal morphology rich in neurites, and proliferated as adherent monolayer, with an approximate population doubling time of 48 hours. Electron microscopic analysis revealed the presence of swollen rough endoplasmic reticulum, and prominent lipid droplets, lysosomes and microfilaments. The chromosome numbers were between 41 and 160 (mode 78), including abnormal karyotypes 1p-, 5q-, 12p and 17p (G-band analysis). Hetero-transplantation of TES-1 into BALB/c nude mice produced no visible tumors. Multipotential differentiation was not induced in TES-1 monolayer culture, but significant neuron specific enolase activity was expressed in both extracellular (by RIA method) and intracellular fractions (by immunohistochemical method), suggesting the differentiation toward neurocytes. This cell line provides a useful in vitro model for the pathophysiological analysis of immature teratoma.
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2/21. Second trimester prenatal diagnosis of epignathus teratoma in ring x chromosome mosaicism with inactive ring x chromosome.

    We report the second trimester prenatal echographic diagnosis of an epignathus teratoma in a female fetus with ring x chromosome mosaicism. The ring x chromosome mosaicism was present in the amniotic cell culture and in the teratoma and the ring X was inactive (X-inactive specific transcript (XIST) locus expressed). Hypoplastic left heart with valvular aortic stenosis and non-immune hydrops were additional findings, and are well-documented in turner syndrome. The occurrence of epignathus teratoma in turner syndrome has not been documented sofar.
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3/21. Juvenile fulminant adnexal tuberculosis caused by gastrointestinal tuberculosis immediately after ovarian cystectomy.

    Genital fulminant tuberculosis is rarely a primary site of infection. We report a case of juvenile adnexal tuberculosis possibly related to tuberculosis immediately after cystectomy in the ileocecal region in a virginal girl. A 13-year-old virginal girl visited our clinic because of a huge ovarian tumor, which was resected by cystectomy. On postoperative day 39, fever episodes were noted, and she received antibiotics, which were not effective. An abscess developed in the region of the cecum and the right adnexa. laparotomy revealed chronic abscess formation, especially in the right adnexa. Resection of the abscess was done. The pathological diagnosis was tuberculosis with caseation, although bacterial culture and molecular biological diagnosis for mycobacterium tuberculosis were negative in sputum, vaginal discharge, urine, and stool of the patient and her family. She was placed on anti-tuberculosis drugs, including isoniazid, rifampicin, and ethambutol, and this regimen was effective. This patient represents a rare case of fulminant adnexal tuberculosis without pulmonary disease.
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4/21. Physiological activity in mediastinal teratomata.

    The clinical details of two patients with benign mediastinal teratomata are presented. Both patients developed inflammation of the root of the neck, the first after a small dose of radiotherapy and the second after a larger dose of radiotherapy and exploration of the thoracic inlet. In both cases, exploration of the inflamed area was followed by persistent discharge of fluid which was sterile on culture. In the first case, this was found to have a high cholesterol, lipid, and amylase content. In both cases, a benign mixed teratoma, with contents including intestinal epithelium and pancreatic tissue, was removed at thoracotomy. The suggestion is made that leakage of digestive enzymes from pancreatic, intestinal or salivary tissue may be a cause of inflammation in and around teratomata, especially after surgical exploration. Early thoracotomy is advised when the condition is recognized.
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5/21. Mediastinal immature teratoma with yolk sac tumor and myelomonocytic leukemia associated with Klinefelter's syndrome.

    A 14-year-old male presenting with a short history of right subclavicular chest pain was found to have a mediastinal tumor. Hematologic investigations and bone marrow examination showed features of myelomonocytic leukemia. The mediastinal tumor was excised, but the surgery was complicated by massive hemorrhage. The patient's condition deteriorated postoperatively and he died a week later. The histology of the mediastinal tumor showed the typical features of an immature teratoma with a yolk sac tumor. A prominent infiltrate of leukemic blast cells was present within blood vessels and in close proximity to the yolk sac component. The karyotypic analysis of leukemic cells isolated and cultured from the bone marrow showed 50XXY, 8, 21, iso G-group marker chromosome karyotype.
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6/21. Evidence of parthenogenetic origin of ovarian teratoma: case report.

    This case report represents one of the few documented cases of parthenote embryo retrieval from an IVF patient with a history of ovarian teratomas. A 29-year-old woman presented at our centre with a history of primary infertility for 6 years due to male factor. She had undergone left oophorectomy 4 years before due to an ovarian teratoma. An ultrasound scan performed during basal evaluation revealed two complex images in the right ovary suggesting teratomas, measuring 2.5 x 2.4 and 1.7 x 1.3 cm. A significant extent of sonographically normal ovarian parenchyma was present, and the patient underwent the long leuprolide acetate protocol of ovarian stimulation with recombinant FSH for an IVF-ICSI cycle. She had 13 metaphase II (MII), four metaphase I (MI), two germinal vesicle (GV) oocytes and one 4-cell embryo retrieved. Eight out of nine injected oocytes were fertilized normally while one was unfertilized. embryo transfer was carried out 72 h after retrieval. The 4-cell (parthenote) embryo recovered at oocyte retrieval continued to cleave in culture, developing into a 7-cell embryo by the next day. The embryo was morphologically normal, presenting an evident nucleus in each blastomere. Fluorescent in situ hybridization (FISH) returned two signals for the x chromosome in each blastomere that was analysed. Of the eight normally fertilized embryos, three were transferred, resulting in a normal singleton pregnancy and the birth of a healthy baby.
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7/21. Establishment and characterization of a nerve cell line (NC-HIMT) from HIMT cells derived from a human ovarian immature teratoma with special reference to the induction of neuron differentiation by retinoic acid.

    A nerve cell line designated NC-HIMT was established from a HIMT cell line derived from a benign ovarian, three germ layer immature teratoma removed from a 21-year-old Japanese female. The HIMT cells were elongated, ellipsoid or spherical in shape, whose karyotype was on the high side of normal diploidy. Small amounts of retinoic acid enhanced differentiation and maturation of the HIMT cells into nervous tissue, and the NC-HIMT cell line was established by the colony isolating technique when the HIMT cell line was cultured in the presence of retinoic acid-supplemented medium. After establishment, the NC-HIMT cell line was cultured and maintained in retinoic acid-free growth medium. Even though these cells were cultured without retinoic acid, the phenotype of nerve cells remained and the cells were also maintained in a state of high normal diploidy. The nerve cells contacted each other with their long cell projections and formed networks. Immunocytochemical observations using anti-bovine NSE, alpha-internexin, neurofilament 200kD, peripherin and GFAP confirmed that the cells were either nerve cells or glia cells. These results assume that HIMT cells, which were derived from an immature teratoma, have progenitor and/or stem cells which can differentiate into nerve and/or glial cells.
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8/21. Ovarian abscess from salmonella: a case report.

    BACKGROUND: An ovarian site of salmonella bacteremia is rare. The contents of a teratoma may hide the organisms easily. Clinicians should be alert to this possibility when the patient had a teratoma with salmonella infection. CASE: A 19-year-old woman presented with a missed menstrual period. An adnexal mass was found during a routine gynecologic examination. The patient had had gastroenteritis 2 months earlier but did not complain of a gastrointestinal problem at presentation. Exploratory laparotomy was performed for a suspected ovarian tumor. An infective teratoma was considered, but the infection source was unknown until the culture report showed a salmonella infection. CONCLUSION: salmonella infection is a self-limiting, febrile disease and is unlikely to involve organs other than the gut. A nontyphoid ovarian abscess became a rare late complication of acute gastroenteritis. Clinicians should pay special attention to the differential diagnosis of ovarian tumor in patients with a history of salmonella infection, especially those with such ovarian lesions as endometrioma or teratoma and with recent abdominal pain, as noted in this case.
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9/21. Fusion of the tumor-suppressor gene CHEK2 and the gene for the regulatory subunit B of protein phosphatase 2 PPP2R2A in childhood teratoma.

    We characterized the molecular genetic consequences of a balanced chromosome translocation t(8;22)(p21;q12), which occurred as the sole cytogenetic aberration in short-term cultured cells from an intrathoracic mature teratoma in a 15-year-old girl. fluorescence in situ hybridization and reverse transcription-polymerase chain reaction disclosed that t(8;22) resulted in the fusion of the genes PPP2R2A and CHEK2, with an inserted fragment belonging to class I endogenous retrovirus-related sequences at the junction. Sequencing of the two genes did not reveal any additional mutation. None of the three detected PPP2R2A/CHEK2 fusion transcripts resulted in an in-frame PPP2R2A/CHEK2 chimerical open reading frame; however, in all of them, the known open reading frame of CHEK2 was preserved. Thus, promoter swapping leading to deregulated CHEK2 expression would be the most likely oncogenic mechanism. Whereas inactivating mutations of CHEK2 previously have been described in a variety of sporadic tumors and in inherited cancer-predisposing syndromes, PPP2R2A, encoding a regulatory subunit of the multimeric enzyme phosphatase 2, has not been directly implicated in tumorigenesis. Our findings suggest that deregulation of CHEK2 and/or PPP2R2A is of pathogenetic importance in at least a subset of germ cell tumors.
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10/21. Pseudomeningocele as a complication of teratoma resection and aseptic meningitis following craniofacial reconstruction: a case report.

    This is a report of two extremely unusual complications of craniofacial surgery on a single patient. A 14-year-old female underwent resection of a recurrent teratoma four times within the first 9 years of life. This left her with two large cranial cavities in the left temple and posterior to the orbit communicating with the subarachnoid space constituting a pseudomeningocele and pulsatile mass in the left temple--an unusual complication. Following resection of the pseudoepithelial cavity linings, the spaces were filled with a split temporalis muscle. Two weeks following surgery, she developed signs and symptoms of meningitis with negative cultures from the drain sites and lumbar puncture. Six days following an unsuccessful attempt to treat her with wide spectrum antibiotics, she was treated with dexamethasone. As a result, there was a dramatic disappearance of signs and symptoms in less than 36 hours; she has not experienced any recurrence since. We feel that this aseptic meningitis was the result of cerebrospinal fluid exposure to muscle--a rare, yet, previously reported complication. The details of the case history and discussion of complications and the way in which they can be avoided, are subjects of this report.
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