Disordered hypothalamus-pituitary-ovary axis in heterotopic extraovarian sex cord-stromal proliferation: a case report of fallopian tube serous adenofibroma.

BACKGROUND: Fallopian tube serous adenofibromas are uncommon tumors of the female genital tract, only dozens of cases have ever been reported. Earlier study indicated that they might be derived from embryonic remnants of the Müllerian duct. Clinical presentation of these tumors is usually asymptomatic. Small cysts of 0.5-3 cm in diameter are mostly incidentally found at the fimbriae end, with coarse papillary excrescences lined by epithelial cells and connective tissue stroma without nuclear pleomorphism or mitosis. CASE PRESENTATION: A 23-year-old woman with normal secondary sexual characters and 46, XX karyotype, presented to the gynecology clinic complaining of irregular menstrual cycles. Laboratory studies reported unique discrepancy of hormone levels; anti-Müllerian hormone (AMH): 6.05 ng/mL (The normal range of AMH is 1.70-5.63 ng/mL in women aged under 35 years old), follicle stimulating hormone (FSH): 31.9 mIU/mL (reference range: 3.85-8.78, follicular phase; 4.54-22.51, ovulatory phase; 1.79-5.12, luteal phase; 16.74-113.59, menopause), and luteinizing hormone (LH): 52.0 mIU/mL (reference range: 2.12-10.89, follicular phase; 19.18-103.03, ovulatory phase; 1.20-12.86, luteal phase; 10.87-58.64, menopause), mimicking gonadotropin-resistant ovary syndrome. The ultrasound reported a right adnexal cyst of 10.4 × 7.87 × 6.7 cm. Laparoscopic evaluation was performed; pathology revealed serous adenofibroma of the fallopian tube with ovarian stroma contents. Heterotopic extraovarian sex cord-stromal proliferations was most probable. The patient's hormone levels returned to the reproductive status two weeks after surgery; FSH: 7.9 mIU/mL, LH: 3.59 mIU/mL,and AMH: 4.32 ng/mL. The patient's menstrual cycles have resumed to normal for over two years after removal of the fallopian tube cyst. CONCLUSIONS: This case of fallopian tube serous adenofibromas presented a discrepancy of serum AMH and FSH mimicking gonadotropin-resistant ovary syndrome. The clinical picture derived from heterotopic extraovarian sex cord-stromal proliferation indicated a disordered hypothalamus-pituitary-ovary axis.

Endometrioid adenofibroma of ovary: A case report and review of literature.

RATIONALE: Endometrioid adenofibroma is a benign epithelial neoplasm of the ovary, most of which are often unilateral. The symptoms of endometrioid adenofibroma are often nonspecific and misleading. Therefore, a full understanding of the characteristics, diagnosis, and treatment methods of this disease is of great importance. In this study, we report a 34-year-old woman who was found with an unidentified mass on the right ovary during the physical examination 3 years ago with nosymptoms or signs. PATIENT CONCERNS: A 34-year-old Chinese female was found with an unidentified 6 cm mass on the right ovary for 3 years that presented with no symptoms or signs. DIAGNOSIS: Pelvic ultrasound revealed a 6 cm cystic solid mixed mass on the right ovary. Through histological and immunohistochemical examinations, the tumor mass was finally diagnosed as endometrioid adenofibroma of ovary. INTERVENTIONS: To confirm the diagnosis, the ovarian tumor was laparoscopically resected. OUTCOMES: The patient returned to hospital after 3 months with no recurrence or postoperative complications. LESSONS: Endometrioid adenofibroma is a benign epithelial neoplasm of the ovary. Complete surgical resection is required and rare cases can recur. Postsurgical pathologic and immunohistochemical testing can confirm a diagnosis of endometrioid adenofibroma. It is important to understand of the key points of differential diagnosis of the disease due to the different prognosis and clinical treatment.

Synchronous ovarian and uterine mesonephric-like carcinoma that potentially arose from endometrioid adenofibroma: A case report.

Mesonephric-like carcinoma histologically resembles mesonephric adenocarcinoma (MA) of the cervix. MA arises from mesonephric duct remnants. However, the origin of mesonephric-like carcinoma is not extensively studied because of its rarity. Here, we present a case of synchronous ovarian and uterine mesonephric-like carcinoma that potentially arose from endometrioid adenofibroma. A 69-year-old woman presented with an abdominal mass with no genital bleeding. She underwent simple total abdominal hysterectomy and bilateral adnexal resection. Histological and immunohistochemical analyses were consistent with mesonephric-like carcinoma involving both ovaries and the uterus. Endometrioid adenofibroma was present in both ovaries, while adenomyosis was observed in the uterus. The glandular duct of the endometrioid adenofibroma in the right ovary had areas suggestive of precursor lesions of mesonephric-like carcinoma. All tumors exhibited the KRAS G12D mutation. These findings suggest that the origin of the mesonephric-like carcinoma was the Müllerian duct, and that the ovarian and uterine tumors were monoclonal.

Tumor fibroso solitario intrapulmonar de patrón adenofibromatoso: un reporte de caso / Solitary intrapulmonary fibrous tumor with an adenofibromatous pattern: A case report

El tumor fibroso solitario es una neoplasia de origen mesenquimal que suele darse en la pleura y que normalmente tiene un curso benigno. La localización intrapulmonar es rara y aún más raro es el patrón adenofibromatoso. Presentamos un caso de un tumor fibroso solitario intrapulmonar con características adenofibromatosas en un paciente con antecedente de adenocarcinoma de próstata. El tumor mostró características inmunohistoquímicas de positividad para STAT6, CD34 y Bcl-2.(AU)

Solitary fibrous tumor is a neoplasm of mesenchymal origin that generally occurs in the pleura and is usually benign. An intrapulmonary location is rare and the adenofibromatous pattern is even more infrequent. We present a case of a solitary intrapulmonary fibrous tumor with adenofibromatous characteristics in a patient with a history of adenocarcinoma of the prostate. Immunohistochemically, the tumor was positive for STAT6, CD34 and Bcl-2.(AU)

Cervical adenofibroma without clinical symptoms: report of a rare case.

Adenofibroma is an extremely rare benign biphasic tumour composed of glandular and fibrous tissues. It occurs more often in the endometrium but it can also occur in the cervix and extrauterine sites. This case report describes a 39-year-old asymptomatic woman with cervical adenofibroma. The patient was treated successfully with surgical removal of the tumour. As adenofibromas are very rare, the report is presented with a brief review of the literature.

Imaging features of biliary adenofibroma of the liver with malignant transformation: a case report with literature review.

BACKGROUND: Biliary adenofibroma (BAF) is a rare primary hepatic tumor with the potential risk of malignant transformation. Given the extreme rarity of the disease, the imaging features of BAF are unclear. We presented a case of malignant BAF and conducted a systematic literature review. We highlighted the key imaging features in the diagnosis and aggressiveness assessment of BAF, as well as the role of various imaging modalities in evaluating BAF. CASE PRESENTATION: We reported a 64-year-old woman with a 5-months history of pain in the right upper quadrant abdomen. US of the liver showed a hypoechoic subcapsular nodule. CT scan revealed a subcapsular solid-cystic mass in segment V of the liver. The mass showed a marked enhancement in the arterial phase followed by wash-out in the venous phase. The patient underwent partial resection of liver's right lobe. The mass was diagnosed as BAF with malignant transformation by postoperative pathology. CONCLUSIONS: CT and MRI are helpful in recognizing and characterizing BAF. The imaging features of BAF include a solitary, large solid-cystic mass with a well-defined margin, lobulated shape, and internal septa; subcapsular location; no intrahepatic bile duct communication; the presence of von Meyenberg complexes in background liver. The enhancement patterns may have the potential to assess the aggressiveness of BAF, and that marked enhancement in the arterial phase followed by wash-out in the venous phase is suggestive of malignant BAF.

Ovarian Clear Cell Tumors Associated With Seromucinous Borderline Tumor: A Case Series.

Ovarian seromucinous borderline tumors (SMBT) and clear cell tumors are both closely associated with endometriosis and share, in a proportion of cases, a molecular pathway involving ARID1A mutations, but they have been rarely described in association. We report a case series of 4 clear cell tumors (3 carcinomas, 1 borderline adenofibroma) coexisting in the same ovary with SMBT. In all cases, the SMBT was the predominant component and we highlight that adequate sampling of these tumors is important to detect small clear cell carcinomas, thus potentially altering the treatment and prognosis.

Robotic enucleation of a biliary adenofibroma.

A 69-year-old man was referred to the hepatobiliary surgeons for mild enlargement of an asymptomatic cystic liver lesion found on routine screening in 2017 that measured 3.7×3.6×4.3 cm. Work-up with MRI revealed a complex multilocular cyst that had enlarged to 6.6×5.5×4.6 cm. Other work-up was unremarkable. He had a Eastern Cooperative Oncology Group score of 1; therefore, a surgical excision was planned due to the possibility for malignancy. A robotic approach with enucleation of the lesion was undertaken, with plans for return for a wider resection if pathological examination revealed malignancy. The lesion was noted to be a biliary adenofibroma, an exceptionally rare lesion that is thought to be benign, but requires excision due to potential malignant degeneration. The patient was discharged home the following day and has had minimal pain in his postoperative course.

Two cases of adenodermatofibroma.

Dermatofibromas (DF) are common skin lesions composed of a dermal proliferation of fibroblasts and histiocytes. Among the variants of DFs, adenodermatofibroma are characterized by a dense proliferation of fibroblasts and histiocytes admixed with entrapped dilated glandular structures. We report two additional cases of adenodermatofibromas, review the literature, theorize on the histopathogenesis of this variant, and suggest that there are different patterns among adenodermatofibromas, from primarily cystic to primarily glandular.

Ovarian seromucinous cystadenomas and adenofibromas: first report of a case series.

AIMS: To report a series of benign ovarian seromucinous neoplasms, an uncommon and hitherto poorly described category of tumours included in the current 2014 World Health Organisation classification of tumours of the female reproductive organs. METHODS AND RESULTS: We report the clinicopathological features of a series of 22 benign ovarian seromucinous neoplasms (cystadenomas and adenofibromas or admixtures). The neoplasms occurred in patients aged 32-83 years (mean = 62, median = 65.5) and involved the left ovary (n = 14), the right ovary (n = 6) or both ovaries (n = 2). There was a common association with endometrioid elements (endometrioid differentiation within the cystadenoma/adenofibroma and/or endometriosis) and other endometriosis-associated neoplasms. CONCLUSIONS: We speculate that some of these represent benign ovarian endometrioid neoplasms with foci of mucinous and/or serous differentiation, while others represent true mixed neoplasms.

A Case of Pulmonary Adenofibroma Treated by Thoracoscopic Resection.

Pulmonary adenofibroma is a rare biphasic tumor that contains epithelial and stromal components. We report a case of pulmonary adenofibroma in which the tumor was resected by thoracoscopic surgery and the diagnosis was established by histopathology. A 59-year-old woman with a past medical history of pyelonephritis visited our hospital for evaluation of an abnormal opacity on a plain chest x-ray during a comprehensive medical examination. A follow-up chest x-ray showed enlargement of the lesion, and the patient was referred to our department for further management. Chest computed tomography revealed a well-circumscribed nodule measuring 1.4 cm in diameter in the upper lobe of the left lung. The chest imaging findings suggested a benign tumor, but because of evidence of lesion enlargement and elevated serum carcinoembryonic antigen levels, we performed wide wedge resection of the left upper lobe by video-assisted thoracoscopic surgery, for diagnosis and treatment. The resected specimen was submitted for rapid pathological diagnosis during the operation, and a benign tumor, possibly sclerosing pneumocytoma, was suspected. Therefore, we completed the operation with wide wedge resection. The final histopathological diagnosis was pulmonary adenofibroma. The patient had an uneventful postoperative course, and at this writing, 6 months postoperatively, there has been no evidence of tumor recurrence. We have reported this case of pulmonary adenofibroma because the tumor is rare, has not yet been well-characterized, and has an unclear prognosis. Collection of data from a larger number of patients is necessary.

Imaging Features of Carcinosarcoma Arising from Adenofibroma of the Uterus: A Case Report.

BACKGROUND: Adenofibroma is a benign tumor composed histologically of epithelial elements and mesenchymal stroma. Carcinosarcoma is a malignant neoplasm that contains elements of carcinoma and sarcoma. Carcinosarcoma arising from adenofibroma of the uterus has never been reported in the literature in English language. Case Presentaion: We report a case of a 56-year-old woman who complained vaginal spotting persisting for several months. We described here for the first time a case of malignant transformation of uterine endometrial adenofibroma into carcinosarcoma that was depicted as a multilocular cystic lesion with enhancing solid portions and stalk-like structure between the myometrium and endometrial mass.

Giant serous adenofibroma of the fallopian tube.

Serous adenofibroma of the fallopian tube is a rare, benign tumour of the female genital tract. They are usually small, asymptomatic and incidentally diagnosed during a surgery for another gynaecological condition. This report presents an atypical case of a 17-year-old girl with a tubal serous adenofibroma that presented with a palpable mass occupying the entire abdomen accompanied by urinary symptoms. She underwent a laparoscopic surgery with drainage of 1800 mL of yellow, citrine liquid from the cyst and left salpingectomy with no complications.

Computer-assisted Diagnosis of Breast Cancer by Cell Network Matrix Extraction and Multilayer Perceptron Analysis.

OBJECTIVE: Diagnosis of breast cancer is based on identification of various morphologic features by histopathologic examination of the specimen. Ancillary immunohistochemical and molecular analyses provide additional information that is prognostic and guides therapy. Because of subjectivity in this approach, we sought to develop a computer model which could assist in differentiating normal or benign tissue from malignant breast carcinoma. METHODS: Cases of benign sclerosing adenosis (20) and high-grade invasive ductal carcinomas (20) of breast were retrieved and re-examined. Five images of the diagnostic areas from each case were captured (400x). Each image was divided into quadrants and saved as 1-megapixel each. These 800 images were then binarized and segmented using the watershed method. The cell graphs were extracted to identify the matrix of adjacent cells and the network properties were determined for each image. The local network features were fed into a PAM model and global network features were fed into a multilayer perceptrons (MLP) to distinguish between benign and malignant samples. These characteristics were evaluated by training the models on 40% (320) of the randomly assigned images followed by real-time testing of the remaining 60% (480) images. In addition, normal breast tissue from five cases was retrieved and forty (40) images were captured to further test the model. RESULTS: Both local and global network feature models had high area under the curve (AUC) (0.63 and 0.99 respectively), with their adjusted Rand indices (ARI) being 0.61 and 0.87, respectively. Pooling the pseudoprobabilities of the two neural networks greatly increased the accuracy of the model with predictions of the combined model at image level being 100% accurate with AUC of 1. CONCLUSION: This study shows that using a combination of cell-graph extraction and a deep learning algorithm computers can accurately distinguish between benign and malignant breast lesions.

Prominent entrapment of respiratory epithelium in primary and metastatic intrapulmonary non-epithelial neoplasms: a frequent morphological pattern closely mimicking adenofibroma and other biphasic pulmonary lesions.

As one of the most common target organs for hematogenous spread from diverse cancers, biopsy interpretation of lung tumors is complicated by the challenging question of primary versus metastatic and by frequent entrapment of native respiratory glands. Nevertheless, the literature dealing with this issue is surprisingly sparse and no single study has been devoted to this topic. We reviewed 47 surgical lung specimens of non-epithelial neoplasms (38 metastases, mainly from sarcomas and 9 primary lesions) for frequency and pattern of intralesional epithelial entrapment. Respiratory epithelium entrapment was noted in 23/47 (49%) cases (diffuse in 15 and peripheral in 8). Entrapped glands frequently showed prominent regenerative and reactive changes mimicking neoplastic glands. Based on cellularity of the mesenchymal component and the extent, distribution and shape of entrapped respiratory glands, four morphological patterns were recognized: paucicellular sclerosing low-grade neoplasms containing leaflet-like glands indistinguishable from adenofibroma and fibroepithelial hamartomas (n = 11), and biphasic cellular lesions mimicking adenomyoepithelioma (n = 1), biphasic synovial sarcoma (n = 2), and pleuropulmonary blastoma (n = 1). Only a single genuine pulmonary adenofibroma was identified. This study highlights frequent respiratory epithelium entrapment in diverse non-epithelial lung tumors, both primary and metastatic. Recognition of this finding and use of adjunct IHC combined with clinical history should help to avoid misinterpretation as primary pulmonary biphasic neoplasm or as harmless adenofibroma. The vast majority of morphologically defined lung adenofibromas represent adenofibroma-like variants of histogenetically diverse entities so that a diagnosis of adenofibroma should be rendered only very restrictively and then as a diagnosis by exclusion.

Pulmonary Adenofibromas: A Clinicopathologic Correlation of 13 Cases.

Thirteen cases of primary pulmonary adenofibromas are presented. The patients are 8 women and 5 men between the ages of 41 and 73 years (average: 57 y). The patients presented with nonspecific symptomatology or their tumor was identified during routine chest films. A wedge resection was performed in all cases with lymph node sampling. Grossly, the tumors varied in size from 1 to 2.5 cm in greatest dimension. The entire tumor was histologically evaluated in all cases. All the tumors shared similar histologic features namely leaf-like/phyllodes-like growth patterns with varying areas of sclerosis, focal inflammation, and entrapped epithelium. A wide panel of immunohistochemical studies was performed including epithelial, neural, muscle, and vascular markers, all of which showed negative staining. The tumors were positive only for vimentin in the stroma and keratin in the entrapped epithelium. Further evaluation in 6 cases using in situ hybridization for the solitary fibrous tumor was performed and was negative. Clinical follow-up in all the patients showed no evidence of recurrence or metastatic disease, during a period of 12 to 36 months. The current cases highlight the unusual occurrence of pulmonary adenofibromas and the importance of separating these tumors from other tumors that may have the potential to recur or metastasize. The use of proper immunohistochemical stains/molecular analysis aids in the proper classification of these tumors.

Magnetic Resonance Imaging Findings of Biliary Adenofibroma.

Biliary adenofibroma is a rare tumor with a bile duct origin characterized by a complex tubulocystic non-mucin secreting biliary epithelium with abundant fibrous stroma. The MRI features of biliary adenofibroma are not well established. The authors encountered two patients with biliary adenofibroma and reviewed the literature focusing on the MRI findings. A well-circumscribed multicystic tumor with septal enhancement and no intrahepatic bile duct communication may be the characteristic MRI findings of biliary adenofibroma.

Mucinous Neoplasms of the Ovary: Radiologic-Pathologic Correlation.

Mucinous neoplasms of the ovary account for 10%-15% of ovarian neoplasms. They may be benign, borderline, or malignant. The large majority are benign or borderline, accounting for 80% and 16%-17%, respectively. Mucinous neoplasms of the ovary most commonly affect women in their 20s to 40s. The clinical manifestation is nonspecific, but most mucinous ovarian neoplasms manifest as large unilateral pelvic masses. At gross pathologic analysis, mucinous ovarian neoplasms appear as large multiloculated cystic masses. The contents of the cyst loculi vary on the basis of differences in internal mucin content. At histologic analysis, mucinous ovarian neoplasms are composed of multiple cysts lined by mucinous epithelium, often resembling gastrointestinal-type epithelium. Imaging evaluation most commonly includes US and/or MRI. The imaging findings parallel the gross pathologic features and include a large, unilateral, multiloculated cystic mass. The cyst loculi vary in echogenicity, attenuation, and signal intensity depending on the mucin content. Mucinous neoplasms of the ovary are staged surgically using the FIGO (International Federation of Gynecology and Obstetrics) staging system. Primary treatment is surgical, with adjuvant chemotherapy considered in the uncommon case of mucinous carcinoma with extraovarian disease. Since most mucinous ovarian neoplasms are benign or borderline, the overall prognosis is excellent.