Female adnexal tumors of probable Wolffian origin: morphological, immunohistochemical, and molecular analysis of 15 cases.

Female adnexal tumors of probable Wolffian origin are rare and present a diagnostic challenge due to their morphological and immunohistochemical overlap with more common ovarian and broad ligament entities. We evaluated the morphological, immunohistochemical, and molecular features of 15 tumors of probable Wolffian origin. Patients ranged from 32 to 69 (mean 47) years and tumors from 1.8 to 30 (mean 10) cm. All except one arose in para-adnexal soft tissues. Follow-up was available for six patients, five of whom were alive and well, while the sixth, who had extra-adnexal disease at diagnosis, died from unrelated causes. The following patterns were noted: tubular (all tumors), solid 11/15 (73%), sieve-like 7/15 (47%), and reticular 1/15 (7%). A myxoid background was present in 3/15 (20%) of tumors and eosinophilic luminal secretions in 11/15 (73%). Most tumors (12/15, 80%) had low-grade nuclear atypia, while three showed foci with scattered high-grade atypia. Mitotic index ranged from 0 to 17 (mean 4) per ten high-power fields. Tumors were positive for pankeratin and negative for TTF-1. EMA, GATA3, and PAX8 were positive in 2/10 (20%; focal), 3/15 (20%; focal), and 1/15 (7%; focal) of tumors, respectively. CD10, SF-1, calretinin, inhibin, ER, PR, cytokeratin 7, and WT1 were variably expressed. Pathogenic mutations were rare and included STK11 (n = 3), APC (n = 1), and MBD4 (n = 1). Copy number variations were detected in the three tumors with STK11 mutations and a myxoid background. These data demonstrate that female adnexal tumors of probable Wolffian origin are morphologically and immunohistochemically diverse, but infrequently harbor pathogenic mutations. However, their lack of mutations in contrast to their mimickers may be a valuable tool in diagnostically difficult cases.

Comparison of Qualitative (Time Intensity Curve Analysis), Semi-Quantitative, and Quantitative Multi-Phase 3T DCEMRI Parameters as Predictors of Malignancy in Adnexal.

Objective: The present study aimed to compare the qualitative (time intensity curve analysis), the semi-quantitative and the quantitative multiphase 3T dynamic contrast-enhanced (DCE) MRI parameters as predictors of malignancy in adnexal masses. Materials and Methods: In this prospective study, women with an adnexal mass who were scheduled for surgical resection or were followed for more than one year period to confirm the benignity of their lesions, underwent multiphase 3T DCE-MRI. The qualitative (time intensity curve), semi-quantitative (SImax, SIrel, WIR) and quantitative (Ktrans, Kep, Vb) analyses were performed on DCE-MRI sequences and their predictive values were compared. Results: A total of 17 benign and 14 malignant lesions were included. According to the qualitative analysis, none of the lesions with Type I time intensity curves (TIC) were malignant and none of the masses with Type III TICs were benign. The accuracy of the quantitative parameters in detection of malignancy was found to be higher than that of semi-quantitative variables, particularly when calculated for a small ROI within the high signal area of the mass (sROI) rather than the largest ROI including the whole mass (lROI), and when inter-MRI variations were omitted using ratios. The Kep(tumor)/Kep(myometrium) ratio measured from sROI was the best parameter for differentiating a malignant lesion with a sensitivity of 100% and a specificity of 92.3%. Conclusion: We concluded that a Type I TIC confirms a benign lesion, and a type III TIC confirms the malignancy and further evaluation is not recommended for these lesions. So complementary quantitative analysis is only recommended for adnexal masses with type II TICs.

Icon immunoconjugate treatment results in regression of red lesions in a non-human primate (Papio anubis) model of endometriosis.

Endometriosis is a common condition in reproductive-aged women characterized by ectopic endometrial lesions of varied appearance, including red, white, blue, black or powder burn coloration, which contribute to chronic pain and infertility. The immunoconjugate molecule (Icon) targets Tissue Factor, a transmembrane receptor for Factor VII/VIIa that is aberrantly expressed in the endothelium supporting ectopic endometrial tissue. Icon has been shown to cause regression of endometriosis in a murine model of disease but prior to this study had not been tested in non-human primates. This study evaluated Icon as a novel treatment for endometriosis in non-human primates (Papio anubis) using an adenoviral vector (AdIcon) delivery system. Female baboons (n = 15) underwent surgical induction of endometriosis. After laparoscopic confirmation of endometriosis lesions 6-weeks post-surgery, the treatment group (n = 7) received weekly intraperitoneal injections of viral particles carrying the sequence for Icon, resulting in expression of the protein, while the control group (n = 8) received no treatment. Icon preferentially reduced the number and volume of red vascularized lesions. Icon may present a novel treatment for endometriosis by degrading red vascularized lesions, likely by targeting tissue factor aberrantly expressed in the lesion vasculature.

Usefulness of Flow Cytometry in Diagnosis of IgG4-Related Ophthalmic Disease and Extranodal Marginal Zone B-Cell Lymphoma of the Ocular Adnexa.

BACKGROUND/AIM: Although flow cytometry (FCM) is used to evaluate cell surface markers of various leucocyte populations quantitatively, little is known about the usefulness of FCM in lymphoproliferative disorders of the ocular adnexa. The aim of this study was to disclose results of FCM, which were compared among IgG4-related ophthalmic disease (IgG4-ROD), idiopathic orbital inflammation (IOI), and extranodal marginal zone B-cell lymphoma (EMZL). MATERIALS AND METHODS: This is a retrospective observational study. Sixty-nine tumors comprising of 16 IgG4-ROD, 24 IOI, and 29 EMZL were enrolled in the study. All tumors, surgically excised, were diagnosed based on histopathology, immunoglobulin (Ig) heavy chain gene rearrangement, and FCM. In FCM, the percentage of T-cell markers (CD2, CD3, CD4, CD5, CD7, CD8), B-cell markers (CD10, CD19, CD20, CD23), NK cell marker (CD56) and cell surface kappa/lambda was searched based on medical records. Ig light chain restriction was evaluated from results in kappa/lambda deviation by FCM. RESULTS: The percentage of CD2, CD3, CD4, CD7, and CD10 was significantly higher in IgG4-ROD/IOI than EMZL (p<0.05 in every factor). In contrast, CD19 and CD20 percentages were significantly greater in EMZL than IgG4-ROD/IOI (p<0.01). There was no significant difference in any marker between IgG4-ROD and IOI. Kappa-positive cells were significantly greater in EMZL than IgG4-ROD/IOI (p<0.05). In kappa/lambda deviation, false-positive was noted in 3 (7.5%) benign IgG4-ROD/IOI and false-negative was observed in 10 (34.5%) EMZL cases. Sensitivity and specificity of Ig light chain restriction were 65.5 and 92.5%, respectively. CONCLUSION: Analyses of cell surface markers using FCM were useful in differentiating EMZL from IgG4-ROD/IOI. Sensitivity of Ig light chain restriction was relatively low in diagnosis of EMZL using FCM.

MYB, CD117 and SOX-10 expression in cutaneous adnexal tumors.

BACKGROUND: Elevated MYB expression has been documented in adenoid cystic carcinoma (ACC), cylindroma, and spiradenoma, but the specificity of this finding is unknown. CD117 and SOX-10 expression also occurs in some cutaneous adnexal tumors. This study assesses MYB, CD117 and SOX-10 expression in cutaneous adnexal tumors. METHODS: Retrospective analysis of 184 benign adnexal tumors (140 eccrine/apocrine, 40 follicular and 10 sebaceous), and 30 malignant adnexal tumors was performed with MYB, SOX-10 and CD117 immunostaining. RESULTS: In the benign adnexal tumors, 16% (23/140) significantly expressed MYB. MYB expression was limited to cylindromas and to a lesser extent, spiradenomas in the benign cohort. Elevated MYB expression was detected in mucinous carcinoma, endocrine mucin-producing sweat gland carcinoma and 1 and 4 cases of extramammary Paget's disease (EMPD) in the malignant cohort. CD117 and SOX-10 had similar overall positivity rates in benign apocrine and eccrine tumors (45% and 68% respectively), and were generally negative in other benign and malignant adnexal tumors. CONCLUSION: Expression of MYB appears limited to a small number of cutaneous adnexal tumors, including cylindromas, spiradenomas, ACCs, mucinous carcinoma, endocrine mucin-producing sweat gland carcinoma and some cases of EMPD.

Utility of human epididymis protein 4 serum marker for the detection of adnexal malignancy: a multicentric prospective study.

The aim of this study was to assess the usefulness of the human epididymis protein 4 (HE4) serum biomarker in predicting malignant disease in a clinical setting in comparison with other diagnostic tools, such as serum CA125 and ROMA score. A multicentric prospective observational study was carried out between January 2010 and December 2011 in four European centres (Italy, Portugal, Latvia and Spain). Data from 981 healthy controls and patients diagnosed with adnexal pathology were collected. Data on the ROMA index, CA124 and HE4 tumour markers were analysed. The receiver-operator characteristics curve and the area under the curve were analysed to discriminate between malignant and nonmalignant disease. Predictive values were also calculated. In total, 642 (65.4%) patients presented with a pelvic mass, with 324 (33%) of them being diagnosed with malignant disease. Sensitivity for HE4 was 64.1%; specificity was 95.7%; and positive predictive value was 88.1%, with a 4.3% false-positive rate. On comparing malignant disease versus nonmalignant/healthy patients, there was a significant difference (P<0.001) in the area under the curve. The receiver-operator characteristic for CA125 was 0.79 [95% confidence interval (CI): 0.76-0.83], for HE4 was 0.89 (95% CI: 0.87-0.91) and for ROMA was 0.71 (95% CI: 0.68-0.75). The HE4 serum marker showed similar sensitivity, but better specificity, than CA125 and can improve the detection of malignant pathology in women diagnosed with adnexal pathology.

p40 in Conjunction With CK20 and E-Cadherin Distinguishes Primary Adnexal Neoplasms of the Skin.

Although basal cell carcinomas (BCC) show typical histomorphologic features, they sometimes remain difficult in distinction from benign adnexal skin tumors of follicular origin like trichoepithelioma (TE) or trichoblastoma (TB). Consequently, an immunohistochemical marker panel separating described entities would be helpful in clinical routine. Thus, we stained 22 skin lesions (BCC, TE, and TB) against ß-catenin, CK20, E-cadherin, p40, and p63. The staining pattern was described and quantified using an immunohistochemical score. Although p40 and p63 revealed a strong staining intensity of all skin lesions without distinction between BCC and benign lesions (P=1.000), established Merkel cell marker CK20 illustrated a loss of staining in BCC compared with TE and TB (P=0.007). In contrast, BCC exhibited an increased expression of E-cadherin in relation to TE and TB (P=0.009). Single application of CK20 or E-cadherin could predict diagnosis of BCC in 81.8% or 72.7%, respectively. Combining consecutive staining of E-cadherin and CK20 could even enhance specificity toward diagnosis of TE or TB. Hence, findings of our study imply that sequential staining of CK20 and E-cadherin prevents false-positive classification of BCC. Furthermore, our study demonstrated that p40 exhibits the same staining pattern in BCC, TE, and TB. Therefore, p40 might replace p63 equivalently establishing diagnosis of primary adnexal neoplasms of the skin in the form of BCC as well as benign adnexal tumors. As a result, the depicted immunohistochemical marker panel may be applied for adnexal skin neoplasms as a diagnostic adjunct especially in surgically challenging body regions.

Differentiating between benign and malignant adnexal lesions with contrast-enhanced transvaginal ultrasonography.

OBJECTIVE: To analyze the relationship between contrast kinetics in tumorous vessels and lesion histologic type in an attempt to differentiate between malignant and benign disease. METHODS: In a single-center prospective study, patients who had been referred for elective surgery because of a diagnosis of unilateral and/or bilateral adnexal masses were enrolled at Dr Jan Biziel University Hospital, Bydgoszcz, Poland, between January 2012 and September 2013. Participants underwent contrast-enhanced ultrasonography examination (CEUS). Contrast kinetics were obtained and compared with the neovascularization of the tumor. Accuracy, and positive and negative predictive values were calculated. RESULTS: Among 160 enrolled patients, 84 underwent CEUS examination and 51 lesions were studied. Baseline and maximum color Doppler intensities were significantly higher in malignant than in benign tumors (P < 0.001 for both). Similarly, the absolute and relative increases in color Doppler intensity were significantly higher in malignant tumors (P < 0.001). The estimated positive predictive value was 97.1%, the negative predictive value was 100%, and the accuracy was 100%. Peak enhanced intensity of fractional color Doppler Area and area under the time-intensity curve (S-parameter) correlated significantly with the histology of the lesion (P < 0.001). Probability curves demonstrated that higher S-parameter values were correlated with a higher risk of malignancy. CONCLUSION: Transvaginal CEUS is a reliable and reproducible way to differentiate between benign and malignant adnexal lesions.

Improved Diagnostic Accuracy in Characterization of Adnexal Masses by Detection of Choline Peak Using 1H MR Spectroscopy in Comparison to Internal Reference at 3 Tesla.

BACKGROUND: The aim of this study was to assess the role of the presence of a choline peak in 3 Tesla 1H magnetic resonance spectroscopy (MRS) for differentiating benign from malignant adnexal masses. MATERIALS AND METHODS: A total of 46 adnexal masses (23 malignant and 23 benign) underwent 1H MRS study prior to surgery to assess the presence of choline peak. RESULTS: A choline peak was detected in 16 malignant masses (69.5%) and was absent in the other 7 (30.5%). A choline peak was only detected in 6 (26%) of the benign adnexal masses. The presence of an MRS choline peak had a sensitivity of 69.5%, a specificity of 74%, a positive predictive value (PPV) of 72.7%, and a negative predictive value (NPV) of 71% for diagnosing malignant adnexal masses. A significant difference between the frequency of mean choline peaks in benign and malignant adnexal masses was observed (P value<0.01). CONCLUSIONS: A 1H MRS choline peak is seen in malignant adnexal masses more frequently than the benign masses, and may be helpful for diagnosing malignant adnexal masses.

MR Spectroscopy for Differentiating Benign From Malignant Solid Adnexal Tumors.

OBJECTIVE: The purpose of this article is to investigate the proton MR spectroscopy ((1)H-MRS) features of solid adnexal tumors and to evaluate the efficacy of (1)H-MRS for differentiating benign from malignant solid adnexal tumors. MATERIALS AND METHODS: Sixty-nine patients with surgically and histologically proven solid adnexal tumors (27 benign and 42 malignant) underwent conventional MRI and (1)H-MRS. Single-voxel spectroscopy was performed using the point-resolved spectroscopy localization technique with a voxel size of 2 × 2 × 2 cm(3). Resonance peak integrals of choline, N-acetyl aspartate (NAA), creatine, lactate, and lipid were analyzed, and the choline-tocreatine, NAA-to-creatine, lactate-to-creatine, and lipid-to-creatine ratios were recorded and compared between benign and malignant tumors. RESULTS: A choline peak was detected in all 69 cases (100%), NAA peak in 67 cases (97%, 25 benign and 42 malignant), lipid peak in 47 cases (17 benign and 30 malignant), and lactate peak in eight cases (four benign and four malignant). The mean (± SD) choline-tocreatine ratio was 5.13 ± 0.6 in benign tumors versus 8.90 ± 0.5 in malignant solid adnexal tumors, a statistically significant difference (p = 0.000). There were no statistically significant differences between benign and malignant tumors in the NAA-to-creatine and lipid-to-creatine ratios (p = 0.263 and 0.120, respectively). When the choline-to-creatine threshold was 7.46 for differentiating between benign and malignant tumors, the sensitivity, specificity, and accuracy were 94.1%, 97.1%, and 91.2%, respectively. CONCLUSION: Our preliminary study shows that the (1)H-MRS patterns of benign and malignant solid adnexal tumors differ. The choline-to-creatine ratio can help clinicians differentiate benign from malignant tumors.

Adnexal mass secondary to extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma) with associated amyloid deposition.

Systemic amyloid light chain amyloidosis (AL amyloidosis) is usually seen in association with a plasma cell disorder. Amyloid deposition associated with extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma) is a rare phenomenon that is not typically associated with systemic AL amyloidosis. We describe the unusual case of a patient with an adnexal mass secondary to MALT lymphoma with associated amyloid deposition.

Role of a risk of malignancy index in clinical approaches to adnexal masses.

OBJECTIVE: The aim of this study was to evaluate predictive role of risk of malignancy index in discriminating between benign and malignant adnexal masses preoperatively. METHODS: This retrospective study was conducted with a total of 569 patients with adnexal masses/ovarian cysts managed surgically at our clinic between January 2006 and January 2012. Obtained data from patient files were age, gravidity, parity, menopause status, ultrasound findings and CA125 levels. For all patients ultrasound scans were performed. For the assessment of risk of malignancy index (RMI) Jacobs' model was used. Histopathologic results of all patients were recorded postoperatively. Malignancy status of the surgically removed adnexal mass was the gold standard. RESULTS: Of the total masses, 245 (43.1%) were malignant, 316 (55.5%) were benign and 8 (1.4%) were borderline. The mean age of benign cases was lower than malign cases (35.2±10.9 versus 50.8±13.4, p<0.001). Four hundred and five of them (71.2%) were in premenopausal period. Malignant tumors were more frequent in postmenopausal women (81% versus 29%, p<0.001). All ultrasound parameters of RMI were statistically significantly favorable for malignant masses. In our study ROC curve analysis for RMI provided maximum Youden index at level of 163.85. When we based on cutoff level for RMI as 163.85 sensitivity, specificity , PPV, NPV was calculated 74.7%, 96.2%, 94% and 82.6%, respectively. CONCLUSIONS: RMI was found to be a significant marker in preoperative evaluation and management of patients with an adnexal mass, and was useful for referring patients to tertiary care centers. Although utilization of RMI provides increased diagnostic accuracy in preoperative evaluation of patient with an adnexal mass, new diagnostic tools with higher sensitivity and specificity are needed to discriminate ovarian cancer from benign masses.

Histopathological diagnoses of adnexal masses: which parameters are relevant in preoperative assessment?

OBJECTIVE: The aim of the study was to assess which clinical, laboratory and ultrasound characteristics of adnexal masses might predict the histopathological nature of the disease. MATERIALS AND METHODS: The study involved all women treated at the Clinic of Gynecology and Obstetrics Clinical Centre of Serbia for adnexal tumors between July 1, 2010 and December 31, 2011. On admission, detailed anamnestic and laboratory data were obtained, expert ultrasound scan performed and RMI was calculated for all patients. Data were related to histopathological findings and statistically analyzed. RESULTS: The study included 540 women out of which 85 had malignant (seven diagnoses), 435 benign (seven diagnoses) and 20 borderline tumors. All types of malignant and borderline tumors were more frequent in postmenopausal women (p=0.000). Only papillary adenocarcinoma significantly more often produced early metastases (p=0.000). Ascites is a common finding in Krukenberg tumors, granulose cell tumors and papillary adenocarcinomas. There were significant differences between tumor diagnoses regarding the levels of Ca 125 and CEA, erythrocyte sedimentation rate (ESR) and risk of malignancy index (RMI) (p<0.05). No significant differences were found within the group of malignant tumor types regarding the levels of all examined tumor markers, ESR as well as RMI (p>0.05). CONCLUSIONS: In the light of our results, patient age, menopausal status, blood levels of Ca 125, CEA and ESR, as well as calculated RMI, can predict the nature of adnexal masses. Unfortunately none of the examined parameters can accurately determine the exact histopathological diagnosis of the adnexal tumor.

Tumor of the mesosalpinx: case report of a female adnexal tumor of probable Wolffian origin.

We report a rare case of a 45-year-old woman who underwent laparoscopy for a right mesosalpinx mass. Pathologic examination showed a female adnexal tumor of probable Wolffian origin (FATWO). FATWO represents a rare gynecologic tumor and its clinical and pathological features are often ignored. Immunohistochemistry plays the most part in the diagnosis of FATWO. Through this report, we aimed to call attention to this disease in order to better understand the correct treatment and surgical possibilities, and to evaluate and perform the prognosis properly.

Ocular adnexal IgG4-related disease: comparative analysis with mucosa-associated lymphoid tissue lymphoma and other chronic inflammatory conditions.

AIMS: Making a differential diagnosis of IgG4-related disease from mucosa-associated lymphoid tissue (MALT) lymphoma or any other chronic inflammation is often challenging. Moreover, the association with secondary lymphoma of ocular adnexal IgG4-related disease needs to be elucidated. METHODS AND RESULTS: We investigated 14 cases of IgG4-related disease, nine MALT lymphomas and 12 other chronic inflammations involving the lacrimal gland and orbit. Bilateral involvement was frequent in IgG4-related diseases. The number of IgG4-positive cells and the ratio of IgG4/IgG-positive cells were higher in patients with IgG4-related disease than in those with MALT lymphoma (P = 0.016; P < 0.001) and other types of inflammation (P < 0.001; P < 0.001). Monoclonal B cell proliferation was suspected in two cases (14.3%) of IgG4-related disease. One of these patients also displayed monomorphous features suggesting secondary MALT lymphoma. In the other case, κ-chain restriction in IgG4-positive cells was observed, raising the possibility of IgG4-producing MALT lymphoma. Trisomy 3, trisomy 18 or MALT1 translocation was observed in none of the IgG4-related cases. Regulatory T-cell infiltration was higher in cases of IgG4-related disease than in MALT lymphomas (P < 0.001) and other types of inflammation (P = 0.006). CONCLUSIONS: Some genetically and morphologically complicated cases of ocular adnexal IgG4-related disease emphasize the need for in-depth studies to differentiate this disease from MALT lymphoma, and to exclude secondary lymphoma.

Quantitative dynamic contrast-enhanced MR imaging analysis of complex adnexal masses: a preliminary study.

OBJECTIVE: To evaluate the ability of quantitative dynamic contrast-enhanced magnetic resonance imaging (DCE-MRI) to differentiate malignant from benign adnexal tumours. METHODS: Fifty-six women with 38 malignant and 18 benign tumours underwent MR imaging before surgery for complex adnexal masses. Microvascular parameters were extracted from high temporal resolution DCE-MRI series, using a pharmacokinetic model in the solid tissue of adnexal tumours. These parameters were tissue blood flow (F(T)), blood volume fraction (Vb), permeability-surface area product (PS), interstitial volume fraction (Ve), lag time (Dt) and area under the enhancing curve (rAUC). Area under the receiver operating curve (AUROC) was calculated as a descriptive tool to assess the overall discrimination of parameters. RESULTS: Malignant tumours displayed higher F(T), Vb, rAUC and lower Ve than benign tumours (P < 0.0001, P = 0.0006, P = 0.04 and P = 0.0002, respectively). F(T) was the most relevant factor for discriminating malignant from benign tumours (AUROC = 0.86). Primary ovarian invasive tumours displayed higher F(T) and shorter Dt than borderline tumours. Malignant adnexal tumours with associated peritoneal carcinomatosis at surgery displayed a shorter Dt than those without peritoneal carcinomatosis at surgery (P = 0.01). CONCLUSION: Quantitative DCE-MRI is a feasible and accurate technique to differentiate malignant from benign adnexal tumours and could potentially help oncologists with management decisions. KEY POINTS: Quantitative DCE MR imaging allows accurate differentiation between malignant and benign tumours. Quantitative DCE MRI may help predict peritoneal carcinomatosis associated with ovarian tumors. Quantitative DCE MRI helps distinguish between invasive and borderline primary ovarian tumours.

Laparoscopic radical excision of primary round ligament perivascular epithelioid cell tumor mimicking leiomyoma.

Perivascular epithelioid cell tumors (PEComas) are a group of rare mesenchymal tumors including angiomyolipoma, clear cell sugar tumor, lymphangioleiomyomatosis, and other unusual clear cell tumors at various locations. We describe a 45-year-old female patient presenting with a painless mass at the left lower abdomen. Computed tomography showed a circumscribed mass 8 x 7 x 8 cm in the left round ligament of the uterus. The provisional diagnosis was leiomyoma. The patient underwent initial laparoscopic excision. The histological and immunohistochemical diagnosis was malignant PEComa. She subsequently underwent laparoscopic radical excision of the residual left round ligament and surrounding tissue. At 18 months after surgery, she remained well without clinical and radiographic evidence of recurrent disease. According to this report, primary PEComa of the round ligament can mimic leiomyoma. Laparoscopic radical excision might be a feasible and safe alternative treatment of this tumor with a favorable outcome.

Low-grade adnexal carcinoma of the skin with multidirectional (glandular, trichoblastomatous, spiradenocylindromatous) differentiation.

The conjoint occurrence of follicular, sebaceous, or apocrine differentiations in a cutaneous adnexal neoplasm is a known event, more often encountered in benign neoplasms, whereas reports of cutaneous malignant adnexal tumors with bilineage or trilineage differentiation are few. A new case of a cutaneous malignant adnexal neoplasm with multidirectional differentiation is reported here. A 57-year-old woman presented with a long-standing, slowly growing, asymptomatic solitary tumor the size of a large nut in the coccygeal area, which was surgically excised. Ten years after the surgery, there was no evidence of recurrence or metastasis. Microscopically, the neoplasm was located in the dermis with focal extension into the subcutis. It was asymmetric, horizontally oriented, and mostly composed of small nodules that varied in shape from round and oval aggregations to elongated strands and irregular islands; the nodules were either clustered, formed a jigsaw puzzle-like pattern or were dispersed. The nodules were composed of small basaloid cells sometimes intermixed with larger cells with ample cytoplasm forming glandular structures. Rare nodules resembled elements seen in a spiradenoma by containing scattered lymphocytes and globules of hyalinized eosinophilic basal membrane material. The stroma was paucicellular, but focally it resembled that seen in perifollicular mesenchyme. Mitotic figures, including abnormal ones, were infrequent, but mild nuclear pleomorphism, nuclear crowding, and individual cell necrosis were easily appreciable in both small basaloid cells and cells with clear cytoplasm. Perineural invasion was apparent. We classified this tumor as a well-differentiated adnexal carcinoma demonstrating combined follicular and apocrine differentiation. It differs from previously published cases of malignant adnexal tumors with multidirectional differentiation and further exemplifies the spectrum of diversity encountered in malignant proliferations with differentiation toward the folliculosebaceous-apocrine unit.

Female adnexal tumor of probable wolffian origin: clinicopathological, immunohistochemical and cytofluorimetric analyses of a 22-year-old virgin. case report.

The term female adnexal tumor of probable Wolffian origin "FATWO" designs this tumor wich arises by the rare persisting remnants of the mesonephric duct (Wolffian duct). About 40 cases have been reported in literature. Few cases of recurrence have been reported, FATWO usually shows no signs of hormonal activity. We report a case of the youngest patient affected by FATWO in October 2002. At laparotomy the left adnexa were deformed by a well-capsulated mass, totally removed and sent to the pathologist with a specimen of peritoneal fluid and of the omentum. The histological examination showed a prevalent tubular structure with focal retiform area, without intraluminal mucines. Immunohistochemical findings of the case reported are similar to those described by other authors, except for inhibin which has not been detected by us. The cytofluorimetry showed the low presence of aneuploid cells, with a very low prolifing component (< 1%).