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Endometrial adenofibroma is an uncommon mullerian mixed tumor composed of benign epithelial and mesenchymal components. This tumor must be distinguished from other malignant lesions of the uterus, particularly adenosarcoma. The authors report three cases of endometrial adenofibroma and discuss their clinical and histopathologic features. The tumors were diagnosed in patients 31, 55 and 63 years of age. In all three cases polypoid lesions of 13, 2 and 5 cm, respectively, were found in the uterine cavity. A polypectomy was performed in two cases; one patient underwent hysterectomy. Follow-up was available for two patients who are today alive and well.

Most clear cell neoplasms of the ovaries are carcinomas; benign and borderline clear cell tumors are uncommon. To date, only 12 cases of benign clear cell adenofibroma have been reported in the literature. Here we report a case of benign clear cell adenofibroma of the left ovary in a 51-year-old postmenopausal woman. Histological examination revealed widely spaced simple glands embedded in a dense fibrous stroma. The glands were lined by one to two layers of cells with abundant clear cytoplasm. The nuclei were bland and uniform in size and shape. There was minimal cytologic atypia in some areas. In this article we discussed the criteria for the diagnosis of benign and borderline clear cell adenofibromas and reviewed the literature., (Copyright 2007 S. Karger AG, Basel.)

This report describes an ovarian serous cystadenofibroma, in a postmenopausal woman, which exhibited extensive sex cord differentiation, in the form of solid and hollow tubules resembling Sertoli cell elements, within the stroma. The sex cord elements, which were located just beneath the epithelium, were positive with both alpha inhibin and calretinin and negative with epithelial membrane antigen. They were also CD56 positive but negative with other neuroendocrine markers. True sex cord structures or sex cord-like elements have been described in ovarian adenosarcomas and pure stromal tumors, especially fibromas. However, as far as we are aware, these elements have not been reported in a serous cystadenofibroma. The endometrium exhibited simple hyperplasia, perhaps secondary to estrogenic activity of the sex cord elements. We discuss the significance of CD56 positivity of the sex cord elements.

We present a case of a rare small bowel tumor. A 73-year-old female patient presented at our department with vague abdominal pain. Ultrasound examination discovered an inhomogeneous vascularised mass originating from a small bowel loop, mesenteric enlarged lymph nodes and a nodule in the liver. Multislice Computed Tomography of the abdomen confirmed the ultrasonographic findings and found omental implants together with a left-sided ovarian mass. Surgery was performed. Pathology revealed a rare ileal collision tumor consisting of an adenocarcinoma and a small cell neuroendocrine tumor with peritoneal metastasis of neuroendocrine origin and coincidental benign lesions on both ovaries.

Nodular breast lesions of noncarcinomatous origin are often of fibroepithelial origin. They may cause classification problems when they are hypocellular or hypercellular; the latter setting may also raise the differential diagnosis of phyllodes tumors. Thirty equivocal nodular breast lesions were collected and one hematoxylin and eosin slide from each was assessed by six pathologists with special interest in breast pathology. The overall reproducibility of classifying these lesions into categories of fibroadenoma, phyllodes tumor or anything else was moderate (kappa value: 0.48). The lack of a uniform nomenclature was not felt disturbing for hypocellular lesions, but the discordant diagnosis of tumors resembling or representing phyllodes tumors was acknowledged to require intervention, such as more obvious implication of guidelines and quality assurance programs aiming at assessing diagnoses and prognostic parameters.

Adenofibroma is an extremely rare benign biphasic neoplasm that is classified into the mixed epithelial and mesenchymal tumor group. It typically affects the endometrium, but may occur in the cervix or in an extrauterine location. Preoperative diagnosis of this tumor is usually difficult. We describe the case of a 55-year-old woman with papillary cervical adenofibroma, which appeared as a cervical mass containing multiple cystic components on transvaginal ultrasound. This lesion appears to be clinically and histologically benign but must be differentiated from malignant lesions of the uterus, particularly from adenosarcoma, which can be suggestive of adenofibroma. Accurate diagnosis of these benign tumors permits appropriate counseling of patients., (Copyright 2005 ISUOG. Published by John Wiley & Sons, Ltd.)

To establish diagnostic criteria using comparison of cell cluster shapes, between benign and malignant tumors, breast tumors demonstrating weak cellular atypia in low grade invasive ductal carcinoma (IDC) were compared. Fine-needle aspiration (FNA) specimens of breast tumors were obtained from 37 patients. Among these, 16 were histologically diagnosed as IDC low-grade and the other 21 as benign fibroadenoma (FA). For evaluation, we examined 740 clusters from these 37 FNA specimens. Nine image morphometric parameters were studied, including the cluster area, circumference, maximal length, maximal breadth, ratio of length to breadth, cluster roundness, cluster size, and the edge and distribution image fractal dimensions for cluster analysis. We evaluated the irregularity in cell cluster shape using fractal dimension analysis, and determined the correlation to cluster size. The irregularity in the IDC cluster shape was higher than that in the FA cluster shape. However, six cases (28.5%) of 21 FA clusters showed high fractal dimensions similar to those for IDC. The clusters were classified by cluster analysis into three types: IDC clusters, FA with irregular cluster shape, and FA with no irregular clusters. The average cell cluster area of the FA with irregular shape was found to be about three times larger than that of IDC clusters. When the differential diagnosis between IDC and FA is difficult, it is important to focus on irregularities in the shape and on overall size of the cell clusters. For accurate diagnosis, the cell cluster shape is as important as the individual cellular atypia., (Copyright 2005 Wiley-Liss, Inc.)

Ovarian cancer is the leading cause of death among women with gynecologic malignancies. Epithelial tumors typically constitute 80-90% of ovarian malignancies and are classified primarily as serous, mucinous, endometrioid or clear cell. Current data indicate that each of these histologic subtypes is associated with distinct morphologic and molecular genetic alterations. We describe such genetic alterations with specific reference to histologic subtypes.

Metanephric neoplasms represent a spectrum of differentiated lesions that seem most likely to be related to Wilms tumor. These neoplasms include a pure stromal lesion, a pure epithelial lesion (MA), and a mixed epithelial-stromal lesion (MAF). The continuity of these lesions with Wilms tumor has been demonstrated best in the epithelial lesions. The relationship of Wilms tumor, MAF with mitoses or combined MA/Wilms tumor lesions, and usual MAF or usual MA may be viewed as analogous to that of neuroblastoma, differentiating neuroblastoma, and ganglioneuroma, in which progressively more mature or differentiated counterparts of malignant embryonal lesions are associated with a greater probability of benign clinical behavior. Such a spectrum already is recognized for cystic ILNR-derived nephroblastic lesions, ranging from cystic Wilms tumor, cystic partially differentiated nephroblastoma, and cystic nephroma. Although this concept implies that the more active lesions (Wilms tumor) mature with time into inactive ones (usual MAFs or MA), the converse (that an active Wilms tumor can arise within an inactive usual MAF or MA) remains possible.

A 48-year-old woman with a 14 cm left ovarian mass had total abdominal hysterectomy and bilateral salpingo-oophorectomy. On cut section the tumor had a solid and a cystic component. There was a 2.5 cm nodule attached to the cyst wall. Sections taken from the solid component revealed a tumor composed of interlacing bundles of fusiform cells, resembling a leiomyoma. Smooth muscle actin positivity confirmed the diagnosis. Sections taken from the nodule in the cystic part revealed an adenofibroma. There was an SMA positive area in the cyst wall which we think is the possible origin of the leiomyoma. As far as we know, this is the first case of a co-existing leiomyoma and serous cystadenofibroma in the ovary, and we think it will broaden the histological spectrum of primary ovarian leiomyomas.

Background & Objective: Telomerase is a ribonucleoprotein enzyme, which plays an important role in cell immortalization and carcinogenesis. Recent studies on the association of telomerase activity with prognostic factors of breast cancer were controversial due to different methods. This study was to establish a feasible assay of quantitative detection of telomerase activity based on silver staining, and investigate possible association between telomerase activity and clinicopathological prognostic factors in primary breast cancer., Methods: Highly sensitive silver-staining telomeric repeat amplification protocol assay (SS-TRAP) was used to quantify telomerase activity in 52 frozen human breast cancer samples and their adjacent breast tissues, 32 benign lesions, and 14 normal mammary gland lesions. The association between telomerase activity and clinicopathological data was analyzed., Results: Telomerase activity was detected in 47 of the 52 breast cancer samples (90.38%), and in 10 of the 32 benign lesions, whereas no activity was detected in 37 of 52 adjacent nonmalignant breast tissues, and all 14 normal mammary gland tissues. The telomerase activity levels were 36.91+/-15.35, 8.27+/-4.37, 14.10+/-5.28, 0 (unit: TPG) in breast cancer, adjacent tissue of cancer, benign lesion, and normal tissue, respectively. The difference of telomerase activity was significant between breast cancer and the other 3 groups by using ANVOA (all P< 0.01). A strong correlation was found between telomerase activity and pathological category, and differentiation degree by logistic regression analysis, i.e. with ongoing tumor progression, telomerase activity appeared to increase in primary breast cancer (P=0.003, and P=0.004). No correlation was seen between telomerase activity and disease course, age, and menopause status of patients (all P >0.05). Telomerase activity level of invasive non-special cancer was higher than that of invasive special cancer(P< 0.05). Telomerase activity level of moderately/poorly differentiated carcer was higher than that of highly differentiated cancer (P< 0.05), while no obvious difference was found between moderately and poorly differentiated cancer (P >0.05)., Conclusions: The activation of telomerase might occur early in breast cancer,and plays a critical role in carcinogenesis and tumor development. Telomerase may serve as a specific marker of early diagnosis and prognosis in mammary gland neoplasm.

We report the fine-needle aspiration (FNA) cytology findings of endometrioid adenofibroma arising in the ovary of a 60-year-old woman who presented with vaginal bleeding. Imaging studies revealed a large pelvic mass, which was sampled by computed tomography-guided FNA and core biopsy. The FNA yielded cellular smears composed of bland endometrioid cells and fragments of ovarian-type stroma. The core biopsy showed a biphasic process comprising bland endometrioid glands in a spindle-cell stroma. Immunohistochemical studies performed on the core showed the stroma to be CD10-negative and smooth muscle actin-positive. Subsequent resection of the tumor confirmed the diagnosis and revealed an adenocarcinoma arising in the tumor that was not sampled by FNA. To our knowledge, the cytologic features of ovarian endometrioid adenofibroma have not been previously described., (Copyright 2004 Wiley-Liss, Inc.)

The main objective was to determine the clinical and radiological features of metanephric neoplasms. The tumors were diagnosed on histopathological examination. The clinical data and imaging features were retrospectively analyzed. Between 1998 and 2003, 3 patients underwent radical nephrectomy for renal masses turning out as metanephric neoplasms on histopathology. Two of these tumors were metanephric adenoma (MA) and one was metanephric adenofibroma (MAF). Clinical and radiological features were reviewed. All patients were adult females who presented with flank pain. Tumor was detected on screening ultrasound as a hyperechoic lesion. In all cases CT showed a hyper-attenuating exophytic lesion with cystic areas that enhanced with IV contrast. Based on combination of clinical and imaging features it may be possible to prospectively identify metanephric neoplasms and thus avoid unnecessary radical nephrectomy in favor of conservative surgery.

Cystadenofibromas are rare benign or borderline malignant neoplasms containing both epithelial and stromal components. We report a case of endometrioid cystadenofibroma in a patient with endometriosis. In this case, the solid component showed very high signal intensity on T2-weighted images and was enhanced after Gd-DTPA administration, reflecting marked stromal edema of fibrous tissue. Endometrial cysts with malignant transformation are reported to have an enhancing solid component and to show various intensities on T2-weighted imaging. However, a solid component showing very high signal intensity on T2-weighted images should also be considered stromal edema in benign or borderline cystadenofibromas.

We describe a peculiar pulmonary lesion, that we interpreted as a pseudopapillary variant of solitary fibrous tumor. The patient was a 62-year-old asymptomatic male, non smoking, presenting with a peripheral nodule, 0.8 cm across, located in the lower lobe of the right lung. The patient is alive and well 18 months after surgical excision of the nodule. Microscopically, the lesion was well-circumscribed and characterized by a diffuse pseudopapillary pattern. Pseudopapillae were large, and were covered by a rim of cubic epithelium devoid of atypia. The stromal axis was fibrous and contained scattered bland spindle cells. Immunohistochemically, the latter were strongly positive for vimentin and CD34, focally positive for BCL2 and CD99, negative for cytokeratin, EMA, TTF1, calretinin, smooth muscle actin, desmin and S100 protein; the epithelial cells were immunoreactive for cytokeratin, EMA and TTF1. We interpret this lesion as a peculiar pseudopapillary variant of solitary fibrous tumor, corresponding to what has been reported in the literature as pulmonary adenofibroma and fibroadenoma. The most important differential diagnostic considerations are briefly discussed.

We report a case of biliary adenofibroma in a 47-year-old woman, who presented with right upper quadrant pain for several months. Abdominal imaging revealed a 16-cm solid and cystic mass in the left hepatic lobe. Histologically, the tumor showed two distinct components: 1) cystic and tubular structures lined by low columnar to cuboidal biliary-type epithelium, and 2) a dense fibrous stroma composed of spindle-shaped cells with only mild nuclear pleomorphism and inconspicuous nucleoli. Mitoses and stromal invasion were absent. The glandular epithelium stained positively for keratin AE.3/Cam 5.2, cytokeratin 7, cytokeratin 19, carcinoembryonic antigen, and epithelial membrane antigen and had a low Ki-67 proliferative index. In addition, the epithelium was positive for D10 but did not stain for 1F6 or acid mucin with alcian blue stain. This staining pattern, similar to bile duct hamartoma (von Meyenburg complex) with which this tumor shares morphologic similarity, suggests that biliary adenofibroma originates from interlobular or larger bile ducts. Three years after a subtotal resection no metastasis or significant tumor growth was noted. However, given the marked nuclear p53 immunoreactivity and tetraploidy status observed in this tumor, we cannot exclude that biliary adenofibroma may represent a premalignant process that warrants complete resection and thorough histopathologic examination.

A serous carcinoma and endometrial intraepithelial carcinoma were encountered in an endometrial adenofibroma in a 61-year-old woman. The carcinoma involved the myometrium and cervix (stage IIa). To our knowledge, this is the third documented case of an adenocarcinoma and the first serous carcinoma involving a uterine adenofibroma.

Objective: To evaluate the role of three-dimensional transvaginal sonography in assessing complex adnexal masses., Methods: Forty-one women (mean age, 49.5 years; range, 23-75 years) with the diagnosis of complex adnexal masses on the basis of two-dimensional transvaginal sonography were reevaluated by three-dimensional transvaginal sonography. Two different sonologists evaluated the two- and three-dimensional transvaginal sonograms. Criteria indicative of malignancy included the presence of gross papillary projections, solid areas, and solid echogenicity for both techniques. Three women (7%) had bilateral masses, giving a total of 44 masses that were ultimately assessed. A definitive histologic diagnosis was obtained in every case after surgical tumor removal., Results: Twenty-one tumors (47.7%) were proved malignant, and 23 (52.3%) were benign. Sensitivity, specificity, positive predictive value, negative predictive value, and accuracy for two- and three-dimensional transvaginal sonography were 90%, 61%, 68%, 87%, and 75% and 100%, 78%, 81%, 100%, and 89%, respectively. There were no statistical differences between two- and three-dimensional transvaginal sonography (McNemar test, P = .687). The agreement between both examiners was high (kappa index = 0.71)., Conclusions: The use of three-dimensional transvaginal sonography does not significantly improve the two-dimensional transvaginal sonographic morphologic assessment of complex adnexal masses; however, we found it useful for reinforcing initial diagnostic impressions.

Paratesticular adenofibroma is an uncommon benign tumour. Although it has previously been reported in the female reproductive organs, to our knowledge only three cases have previously been reported in the male genital organs. Herein we describe an adenofibroma that developed in the tunica vaginalis and rete testis. We discuss its histogenesis and conservative treatment.

Hepatic biliary cystadenoma is a well-delineated neoplasm in some domestic animals, especially in cats, but it has not been reported in equines. We report on a case of hepatic biliary tumor, incidentally found in a 10-year-old horse, with gross and microscopic features similar to those observed in biliary adenofibroma of humans. The tumor presented as a solid mass measuring 16 cm in diameter and histologically was composed of complex tubulocystic biliary components embedded in an abundant fibrotic stroma. We regarded this tumor as a morphological variant of biliary cystadenoma of domestic animals. Differential diagnoses from other hepatic biliary tumor-like and tumor lesions are provided.

A 32-year-old woman with Turner's syndrome complained of abnormal genital bleeding, which was diagnosed as a delivered myoma. Vaginal myomectomy was performed, and the tumor was histopathologically diagnosed as an adenofibroma of the uterine corpus. As these are extremely rare, we report this case with a review of some of the literature.

Identification of inheritable mutations associated with the development of malignancy has led to prophylactic surgeries to remove tissues at risk. We report seven unrelated patients with family histories of breast and/or ovarian cancer, five of whom underwent prophylactic salpingo-oophorectomy with hysterectomy. Four had proven BRCA-1 or -2 mutations. Malignant cells were found unexpectedly in the peritoneal washings of two patients, leading to the discovery of early-stage fallopian tube carcinoma. After changing the sampling technique at our institution, two more cases of unexpected fallopian tube carcinoma in situ were discovered. Another patient had a significant family history and underwent hysterectomy for uterine fibroids, leading to the discovery of fallopian tube carcinoma. Another patient with BRCA-1 mutation had unexpected widespread primary peritoneal papillary serous adenocarcinoma. The final patient had a borderline malignant clear cell adenofibroma. These cases underscore the importance of peritoneal cytology and thorough sampling in the management of patients undergoing hysterectomy with a family history of breast/ovarian cancer and/or known BRCA-1/BRCA-2 mutations. As prophylactic surgeries are becoming more common secondary to advances in molecular diagnostics, pathologists need to be aware that surgical specimens from these patients may require more rigorous examination to uncover early neoplastic changes.

An unexpected association with breast cancer and thyroid disorders was found during a review of 91 cases of benign and malignant ovarian adenofibromas. Sixty-three tumors were benign, 11 had areas of borderline neoplasms, and 17 had a component of carcinoma. Such tumors were divided into glandular/cystic (61 cases) and papillary (30 cases) according to their gross and microscopic appearance. Among the 61 patients with glandular/cystic adenofibromas, 13 (21%) had breast cancer and 19 (31%) also had thyroid disorders. Among the 30 patients with papillary adenofibromas there were no cases of breast cancer and only 2 patients had thyroid disorders. The average age of the patients with ovarian adenofibroma and breast cancer or thyroid disorders was higher (66 years) than that of patients without breast cancer or thyroid disorders (55 years). More patients with breast cancer and thyroid disorders had bilateral adenofibromas than patients without breast cancer or thyroid disorders. We also reviewed the medical records of 100 patients with ovarian cancer without adenofibroma component, 100 patients with breast cancer, and 100 patients with ovarian and breast cancer. Six percent of patients with ovarian cancer had breast cancer and 16% of each one of these groups had thyroid disorders. This unexpected association found between glandular/cystic adenofibromas, breast cancer, and thyroid disorders might be explained by defects common to these organs. Disorders of some of these organs have been linked by common genetic changes and it is known that these organs are under the influence of similar hormones. Mutations of PTEN have been found in breast and thyroid cancer. The thyroid and ovaries are controlled by glycoprotein hormones of the pituitary gland, which have common alpha subunits.

A case of a 30-year-old patient with a serous papillary borderline tumor of the right ovary who underwent a simple mono-lateral salpingo-oophorectomy is presented. Conservative surgery allowed a subsequent pregnancy with spontaneous delivery 27 months later.

Objective: To investigate the endocrine activity and clinical characteristics of benign ovarian fibro-epithelial tumors., Methods: A retrospective analysis was done on 16 cases of benign fibro-epithelial tumor collected from the department of obstetrics and gynecology, Peking Union Medical College Hospital from 1981 to 2000., Results: The examinations of endocrine activity was measured in 15 cases. Serum estradiol and follicle-stimulating hormone levels in 4 cases, it was (166.5 +/- 83.0) pmol/L and (39.4 +/- 12.6) IU/L respectively; low to moderate level of estrogen influence was observed in 8 cases by vaginal cytology; endometrium proliferation was observed in 10/15 cases by pathological examination., Clinical Manifestations: post-menopausal vaginal bleeding in 10 cases, lower abdominal recessive pain in 3 cases, pelvic mass in all of the 16 cases with both pelvic examination and ultrasonography., Conclusion: Most of the benign fibro-epithelial tumor have endocrine activity and related clinical manifestations.

Objective: To assess the usefulness of cytology in the diagnosis of 147 histologically established adnexal cysts., Study Design: Retrospective, macro-microscopic study based on fluid aspirated from 132 ovarian and 15 extraovarian cysts and projected as a cytohistologic correlation., Results: Typical macroscopic features were identified in 76% of endometriotic cysts, in 53% of mucinous neoplasms and in 67% of dermoid cysts. Cytology helped to identify 67% of nonneoplastic and 56% of neoplastic cysts. The lowest diagnostic sensitivities were observed in functional cysts and benign serous neoplasms (50%), while the highest were shown by endometriotic cysts (76%) and malignant epithelial neoplasms (71%). Inadequate samples were obtained from all types of cysts, even malignant ones (two mucinous cystadenocarcinomas). Diagnostic cytology was useless in extraovarian cysts (33% sensitivity). An adult granulosa cell tumor was erroneously diagnosed as a follicular cyst by cytologic examination., Conclusion: Examination of the cyst fluids obtained by aspiration demonstrated low sensitivity, with 43% of inadequate samples obtained from all types of cysts. Malignant cystic neoplasms may be overlooked in inadequate samples. Our study also revealed that specificity in this type of analysis is high in inadequate samples, provided that the technique is carried out correctly.

We report a rare case and description of a benign pediatric renal mass. To our knowledge, this tumor has never been described in urologic published reports. It is possible that the identification of this renal tumor could spare a child the toxic adjuvant chemotherapy that would be administered if confused with histologically similar tumors such as clear cell sarcoma of the kidney.

Objective: To describe the sonographic characteristics of ovarian cystadenofibromas., Methods: We conducted a retrospective study of 23 ovarian tumors histopathologically confirmed as ovarian cystadenofibromas that were preoperatively evaluated by transvaginal color Doppler sonography., Results: In all cases the mass was predominantly cystic. Septations appeared in 30.4% of the tumors. Papillary projections or solid nodules appeared in 56.5% of the cases. The most frequent appearance was a unilocular complex cystic mass. In 47.8% of the tumors, vascularization was detected, having a typical pattern of peripheral vascularization with scattered vessels of high blood flow impedance., Conclusions: We have described the sonographic spectrum of findings of ovarian cystadenofibromas. The most frequent appearance was a unilocular cystic mass with gross papillary projections or solid nodules. In almost half of the tumors, vascularization could be detected.

Objective: Positive emission tomography (PET) provides a novel means of imaging malignancies. The following study was undertaken to evaluate the predictive value of PET in determining a pathologic complete response in patients with advanced ovarian or peritoneal carcinoma who had a complete clinical response following primary chemotherapy., Methods: Twenty-two patients with advanced-stage ovarian (N = 17) or peritoneal (N = 5) carcinoma who had achieved complete clinical and radiologic remission and normal CA-125 level after six cycles of chemotherapy and who had consented to a second look laparotomy procedure were studied. All patients received platinum based therapy and all but one patient, treated elsewhere, received paclitaxel in combination with platinum. Following IV administration of 20 mCi [(18)F]fluorodeoxyglucose (FDG), the entire abdomen and pelvis were scanned. Various technical modifications including bladder activity dilution, intravenous hydration with diuretic therapy, and mechanical bowel preparations, were used to reduce background activity. Second-look laparotomy findings were classified as negative, macroscopically positive if a biopsy of a suspicious area was histologically positive, or microscopically positive if only a nonsuspicious area was histologically positive. The effect of patient preparation prior to PET imaging was evaluated., Results: Persistent disease was found in 13 of the 22 patients (59%). Only one of nine sites with macroscopic and none of four with microscopic disease were accurately predicted. The sensitivity was only 10% and the specificity 42%. Intravenous hydration, diuretic therapy, and bowel preparation did not improve the results., Conclusions: These results suggest that despite technical modifications the sensitivity of PET before second-look laparotomy for small-volume persistent disease is low., (Copyright 2001 Academic Press.)

We report an example of metastatic metanephric adenoma containing foci of papillary carcinoma in the primitive tumor arising in the left kidney of an 11-year-old girl. Histology revealed a monomorphous population of small cells with bland cytology arranged in pseudoglandular, tubular, papillary, and glomeruloid structures with frequent psammoma bodies. Intermixed there were foci of and small cavities lined by larger cells with eosinophilic cytoplasm and larger nuclei with small nucleoli. A regional lymph node contained metastatic deposits with the former histologic pattern. By immunohistochemistry the small cells were negative for carcinoembryonic antigen (CEA) and keratin 7 while these antibodies reacted positively in the larger cells. A fluorescent in-situ hybridization (FISH) study for chromosome 17 in imprints from the primitive tumor revealed 3 signals in about 10% of the nuclei while the rest was disomic. Disaggregated cells from the metastatic lymph node consistently revealed 2 signals for chromosomes 7, 16, and l7. Histology of the primitive tumor resembled the epithelial component of the so-called metanephric adenofibroma while the metastatic lymph node exhibited histologic and FISH genomic features of metanephic adenoma. Int J Surg Pathol 9(3):241-247, 2001

We report a case of papillary adenofibroma of the uterine corpus in a 31 year-old woman who was initially examined for vaginal bleeding. Pelvic examination showed a large polypoid mass protruding through the cervix canal. A pelvic ultrasound revealed a polypoid cystic mass apparently arising from the uterus. A polypectomy was performed. The tumor was composed histologically by begin epithelial and mesenchymal components. Uterine adenofibroma is a extremely rare tumor which considered to be a mixed tumor of Müllerian origin. This lesion appears to be clinically and histologically benign but must be differentiated from other malignant lesions of the uterus, particularly from the adenosarcoma.

The authors report nine new metanephric adenofibroma (MAFs; previously termed nephrogenic adenofibroma) and 16 related tumors from the files of the National Wilms Tumor Study Group Pathology Center (NWTSGPC). All tumors contained a variable amount of a bland spindle cell stroma, which is essentially identical to the recently described metanephric stromal tumor (MST). Features that distinguish this stroma from congenital mesoblastic nephroma (CMN) include intratumoral angiodysplasia, concentric cuffing of entrapped tubules ("onion skinning"), and heterologous differentiation. The epithelial components of these lesions spanned a wide range of appearances. All tumors contained at least focally an inactive embryonal epithelium identical morphologically to metanephric adenoma (MA), and hence each case could be classified as containing MAF. The epithelium of nine tumors had this appearance throughout, and hence these were considered usual MAFs. The epithelium of four tumors demonstrated increased mitotic activity but was otherwise similar to MA. The epithelial component of seven tumors spanned a morphologic spectrum from inactive MA to malignant epithelial predominant Wilms tumor (WT), with gradual transitions noted in several cases. Five other tumors contained a carcinomatous component distinct from these lesions but identical morphologically to papillary renal cell carcinoma (PRCC). In one of these cases, this component had metastasized to the regional lymph nodes at the time of diagnosis. No tumor recurred during follow-up, although almost all patients received adjuvant therapy for WT regardless of their tumor's histology and NWTSGPC diagnosis. In conclusion, MAF is a biphasic tumor that spans the morphologic spectrum between benign pure stromal (MST) and pure epithelial (MA) lesions, and can merge with the morphology of WT, supporting the concept that these are all related lesions. A relationship to PRCC is also evident.

Adenomyolipoma of the uterus is a rare, benign, polypoid lesion considered to be of hamartomatous origin or represent an unusual type of benign Müllerian mixed tumour with a heterologous element. The authors present a case of uterine adenomyolipoma and discuss its pathogenesis. A 62-year-old woman complained of lower abdominal pain and postmenopausal bleeding. Imaging techniques revealed a solid ovarian mass and a polypoid intrauterine lesion. The frozen section diagnosis of the ovarian mass was a thecoma. A total hysterectomy and bilateral salpingo-oophorectomy were performed. On gross examination a pedunculated, polypoid lesion of 7x4.5x3cm was found in the uterine cavity. Microscopically, the polypoid lesion contained both epithelial and mesenchymal elements. The epithelial elements were endometrial glands of various size, formed by proliferative endometrial cells. The mesenchymal elements were composed of endometrial stroma, smooth muscle and mature adipocytes. Both the epithelial and the mesenchymal elements showed a benign appearance, were intermingled with each other and periglandular stromal condensation was absent. The lesion had an irregular surface. Microscopic diagnosis was an adenomyolipoma. The peculiar shape and microscopic features of this lesion suggested that it was a variant of benign Müllerian mixed tumour.

We present a case of serous cystadenofibroma 2 cm in diameter in the epiploic appendix of the sigmoid as incidental finding in a 72-year-old patient who underwent hysterectomy and oophorectomy for endometrial carcinoma. The tumor showed the same histology as analogous tumors of the ovary and was associated with endosalpingiosis. Further findings were large adhesions between the epiploic appendices of the sigmoid and the parietal peritoneum and atypical cells in the peritoneal washings. Both may be explained by occult peritoneal endosalpingiosis. The histogenesis, histology, and locations of extraovarian müllerian tumors are reviewed.

A case of adenomyofibroma with skeletal muscle differentiation is described. A 55-year-old asymptomatic woman had atypical glandular cells of undetermined significance on a routine Papanicolaou smear. The endometrial biopsy revealed fragments composed of benign endometrial glands and myofibromatous stroma with foci of skeletal muscle differentiation. The stroma exhibited focal mild cytologic atypia and hypercellularity without periglandular cuffing or mitoses. Electron microscopy and immunohistochemical staining for myoglobin confirmed the skeletal muscle differentiation. A diagnosis of low-grade adenosarcoma with heterologous differentiation was made in the biopsy specimen based on the atypical stroma, the skeletal muscle differentiation, and previous observations that adenosarcomas may contain bland areas indistinguishable from an adenofibroma. The patient underwent hysterectomy, bilateral salpingo-oophorectomy, and pelvic lymphadenectomy. The hysterectomy specimen revealed small foci of residual tumor. In light of these findings the diagnosis was revised to adenomyofibroma with skeletal muscle differentiation. Uterine adenomyofibroma with skeletal muscle differentiation should be distinguished from a low-grade adenosarcoma in an endometrial biopsy.

The clinicopathologic features of neoplasms arising in gastrointestinal endometriosis have not been well characterized. In this series, we report 17 cases of gastrointestinal endometriosis complicated by neoplasms (14 cases) or precancerous changes (three cases). Four patients, one of whom also had hypermenorrhea, presented with chronic abdominal pain and five had obstructive symptoms; one of these also had rectal bleeding. One patient presented with an acute abdomen and fecal peritonitis, one had vaginal bleeding, and one had a progressive change in bowel habits. Nine patients had a long history of endometriosis, 11 patients had had hysterectomies, and eight of these had also received unopposed estrogen therapy. The lesions involved the rectum (6), sigmoid (6), colon, unspecified (2), and small intestine (3), and comprised 8 endometrioid adenocarcinomas (EA), 4 mullerian adenosarcomas (MAS), 1 endometrioid stromal sarcoma (ESS), 1 endometrioid adenofibroma of borderline malignancy (EBA) with carcinoma in situ, 2 atypical hyperplasias (AH), and one endometrioid adenocarcinoma in situ (ACIS). The tumors ranged in size from 2 to 15 cm and all involved the serosa and muscularis propria. Two tumors extended into the mucosa, with mucosal ulceration in one. Follow-up was available in 11 cases. One patient with EA was dead of disease at 1 year, one had two recurrences at 1 and 2 years, and three were alive with no evidence of disease (ANED) at 9 months to 13 years (mean, 68 mos). The patient with the EBA was ANED at 3 months. Two patients with MAS were ANED at 2 and 3 years. The patient with ESS had a recurrence at 3 years and was ANED 6 years after her original diagnosis. One woman with AH was ANED at 60 months and the patient with ACIS was ANED at 16 months. One of the carcinomas was originally misdiagnosed as a primary intestinal adenocarcinoma. The pathologist should be aware of the possibility of a tumor of genital tract type when evaluating intestinal neoplasms in females, particularly if they have a history of endometriosis and have received unopposed estrogen therapy.

This clinicopathologic study of primary Mullerian tumors of the bowel arising in foci of endometriosis is based on six new cases and an analysis of 17 previously reported cases. Varieties of Mullerian tumors occur in the bowel; the most common types are endometrioid carcinoma, followed by various mixed Mullerian tumors and stromal sarcomas. Seventy-eight percent develop in the rectosigmoid colon, the remaining in the cecum or ileum. Those in the latter area tend to be sarcomas or mixed Mullerian tumors. Certain architectural growth characteristics, derived from precursor endometriosis, are common to most endometriosis-associated intestinal tumors (EAITs). Seventy percent of EAITs occur in the outer bowel wall. Transmural tumors tend to form luminal polyps and assume an hourglass shape. Metachronous or synchronous Mullerian tumors occur in 39% of cases. Seventy percent of women with EAITs are in their mid 30s to early 50s. Common presenting symptoms are abdominal or pelvic pain, melena, and an abdominal or pelvic mass. Documented in 26% of patients is a history of prolonged unopposed estrogen therapy. Only 28.5% of cases die of their tumors, but follow-up is less than 5 years in all but 2 patients.

Eccrine syringofibroadenoma is an uncommon benign eccrine tumor, which was first described by Mascaro in 1963. It usually develops on the extremities of elderly persons. We report on a 74-year-old man who presented with a 2-year history of a slowly growing lesion on his face. A detailed histologic and immunohistochemical study was performed on the biopsy material. The tumor consisted of epidermal-derived anastomosing thin epithelial cords embedded in a fibrovascular stroma. The epithelial cords contained ductal and cystic structures lined by luminal cells, which were decorated by antibodies against carcinoembryonic antigen, keratin K19, K8, and K18. Antibody to keratin K6 decorated the luminal walls of the acrosyringia. Antibodies to filaggrin decorated the superficial luminal structures. These results suggest dual acrosyringial and dermal duct differentiation in syringofibroadenoma.

We report six cases of carcinomas arising within fibroadenomas. Fibroadenoma is a benign neoplasm occurring in young women. Its association with carcinomas is unfrequent and particularly reported in older women. Few data are available on the histologic features of fibroadenomas harboring malignant lesions. In this study, most cases of fibroadenomas showed cysts, sclerosing adenosis, epithelial calcifications or papillary apocrine changes. These fibroadenomas are classified as complex and are a long-term risk factor for breast cancer. The complex fibroadenoma may be specific of fibroadenoma associated with carcinoma.

The DNA base excision repair pathway is responsible for the repair of alkylation and oxidative DNA damage. A crucial step in the base excision repair pathway involves the cleavage of an apurinic/apyrimidinic (AP) site in DNA by an AP endonuclease (APE). The major AP endonuclease in mammalian cells is APE/ref-1, a multifunctional enzyme that acts not only as an AP endonuclease but as a redox-modifying factor for a variety of transcription factors. The purpose of this study was to determine the expression of APE/redox factor-1 (ref-1) in ovarian tissues, particularly ovarian cancers. Formalin-fixed, paraffin-embedded specimens of ovarian tissues (normal, various benign conditions, and epithelial cancers) were studied using both polyclonal and monoclonal antibodies to APE/ref-1. The relationship between APE/ref-1 protein levels and DNA repair activity was studied in ovarian Hey and Hey-C2 cell lines using Western blot and a specific AP-site oligonucleotide cleavage assay. Hey and Hey-C2 cells were fractionated, and the nuclear and cytoplasmic extracts were quantitated for protein levels and assessed for APE/ref-1 with Western blot. Normal ovarian tissues consistently demonstrated strong nuclear staining of the surface epithelium, epithelial inclusions, corpora lutea and albicantia, and stroma. Cytoplasmic staining was absent. A similar pattern was seen for benign conditions including endometriosis. Low malignant potential ovarian cancers stained in a pattern similar to normal ovarian and nonneoplastic tissues; however, two specimens also had areas of cytoplasmic staining. Epithelial ovarian cancers were remarkably different from all other ovarian tissues studied. Both nuclear and cytoplasmic staining of the malignant epithelium were seen and ranged from strong to weak, often with considerable staining heterogeneity within the same tumor. The AP-site oligonucleotide cleavage assay indicated that APE/ref-1 protein levels correlate well with DNA repair activity. The increased levels of APE/ref-1 in the Hey-C2 cells was mainly attributable to increased cytoplasmic enzyme. APE/ref-1 immunoreactivity is altered in malignant ovarian tumors. Further studies will determine whether the altered expression and subcellular location reflect changes in redox regulatory functions.

We report a case of tubulocystic ovarian clear cell carcinoma (OCCC) with abundant fibrous stroma associated with an endometriotic cyst. Most OCCC show a small amount of fibrous stroma; however, the tumor presented in this case had abundant stroma, that qualifies it as a malignant clear cell adenofibroma. This unusual type of clear cell carcinoma may be misinterpreted as a benign lesion or as metastatic carcinoma on frozen section. In permanent sections, the stromal invasive foci are focal, small and subtle. Therefore, extensive sampling of the specimen to search for evidence of invasion is recommended for a fibrous ovarian tumor that appears benign on gross examination.

Nephrogenic adenofibroma is a benign renal tumor in children and young adults described by Hennigar and Beckwith in 1992. Seven cases have been described, and we report the first case in an 11-month-old child, in good health, revealed by a macroscopic hematuria. Nephrogenic adenofibroma is an unusual tumor, which was difficult to distinguish from nephroblastoma and mesoblastic nephroma. Beckwith makes a distinction between this principal differential diagnosis in child renal tumors based upon morphologic and immunohistochemical patterns. In our observation, the diagnosis remained difficult and needed several reviews of our case. Beckwith proposed the final diagnosis: nephrogenic adenofibroma with stromal predominance. The prognosis is excellent and no treatment is indicated. A FISH analysis of the tumor cells found a trisomy 11. Trisomy 11 has been reported in mesoblastic nephroma as the most frequent chromosomal abnormality. This finding in tumor cells provides an argument for excluding the diagnosis of nephroblastoma but can not clarify the difference between nephrogenic adenofibroma and mesoblastic nephroma.