Biliary adenofibroma: a precursor lesion of intrahepatic cholangiocarcinoma

ABSTRACT Biliary adenofibroma (BAF) is an uncommon liver tumor with a high propensity for malignant transformation. The histomorphology of BAF with malignant transformation can show a spectrum of changes ranging from benign, dysplastic to frank malignancy. Thus, the diagnosis of BAF imposes the pursuit of dysplasia/ malignancy focus. We presented a case of intrahepatic cholangiocarcinoma arising from BAF in a 49-year-old woman with detailed histomorphology. We also performed a PubMed database search and tabulated all previously reported cases of BAF with dysplasia/ malignant transformation. A statistic comparison of age, sex ratio, size of the tumor, and survival following complete resection between BAFs with and without dysplasia/ malignancy from the retrieved data is presented. Our analysis did not highlight any statistically significant difference between BAFs with and without dysplasia/ malignancy in age, sex ratio, tumor size, and survival following complete surgical resection. Our study highlights the histopathology and immunohistochemistry of a case of BAF with malignant transformation and highlights the importance of this diagnosis in management. Further longitudinal studies on a larger cohort of patients are required to validate our findings.

Clinical and MRI features of ovary adenofibroma: Analysis of 21 cases / 中国医学影像技术

Objective: To observe clinical and MRI features of ovary adenofibroma (OAF). Methods: Clinical and MRI data of 21 patients with OAF confirmed by surgery and pathology were retrospectively analyzed. Results: Among 21 cases, 15 were asymptomatic, while 4 cases complained of abdominal pain and 2 cases of abdominal distension. CA125 increased in 3 cases, while remained normal in 18 cases. OAF displayed as solid mass, solid cysts or polycystic masses on MRI. Most solid masses showed low signals or bottom signals on T2WI with light to medium enhancements. The solid parts of solid cysts showed very low signals of the polycystic and cystic separation on T2WI without or only slight enhancements. T2WI of the multi-sac cystic masses showed low signal of the wall and the separation with slight enhancement. Conclusion: OAF patients often had no characteristic symptoms, and tumor markers were mostly negative. MRI features were helpful to diagnosis of OAF.

Magnetic Resonance Imaging Findings of Biliary Adenofibroma / 대한소화기학회지

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.

A rare pulmonary benign bi-phasic tumor: a case report of pulmonary adenofibroma and literature review / 北京大学学报(医学版)

Pulmonary adenofibroma is an extremely rare benign primary tumor of the lung,with characteristic bi-phasic differential pattern.They are usually sub-pleural solid pulmonary nodules with clear margins.The tumor is composed of glands and peri-gland stroma.The glands are often quite simple,forming long and narrow tubules,with uniformly monolayer lining cells.Combined papillary or phyllodes structures were reported in some cases.The stromal cells are spindle-shaped and look mild,mixed with the collagen bands.Up till now,only a few cases of pulmonary adenofibroma have been reported all over the world.And because of the limited recognition,this tumor is easily misinterpreted as malignancy in frozen section or biopsy specimens.We reported a new case of pulmonary adenofibroma.The mass peripherally located in the left lobe of the lung,found by chance in a 74-year-old woman.The patient underwent a wedge resection of the left lung by the thoracoscope,because of the slowly gradual enlargement of the mass annually.An oval grayish-white nodule was sub-pleural located in the specimen,with solid and rubbery texture,but without a distinct capsule.Two distinct components of simple glands and mild spindle cell stroma were found to mix together uniformly under the microscope.Collagen bands of various widths evenly surrounded each stromal cell.A few small coarse papillae or phyllodes structures were randomly distributed in some area.The immunohistochemical staining pattern of the glandular cells was accordant with type Ⅱ alveolar epithelium.Stromal cells were positive with CD34,B-cell lymphoma-2 (Bcl-2),CD99and estrogen receptor (ER),while S-100,smooth muscle actin (SMA) and all the mesothelium markers were negative.The patient was disease free after the surgery,although the follow-up time was only one year.Besides the new case above,we also reviewed all the reported cases,and tentatively discussed the probable histological origin of pulmonary adenofibroma.

The analysis of ovarian adenofibroma on clinical ,CT and MRI features / 实用医学杂志

Objective To explore the clinical,CT and MRI features of ovarian adenofibroma(OAF)and improve the diagnostic level. Methods Ninety-two cases of OAFs were analyzed retrospectively and summarized the features including clinical manifestations ,CT and MRI images. Results The probability of unilateral ,serous and benign OAFs were 91.3%,89.1%and 97.8%. OAFs associated with uterine or/and ovarian lesions was 40.2%. The clinical manifestations of OAF was not typical. These OAFs were divided into 2 groups according to whether or not accompanied with uterus or/ and ovarian lesions. The clinical manifestations and laboratory indexes were only found abnormal menstruation rate(P<0.05),but there were no significant difference between other indexes. The performance of CT plain scan and CT/MRI enhanced scan had not characteristic.The percentage of the"black sponge"on T2WI image and hyperintensity on DWI image of OAFs were 40.0% and 65.0%. Conclusion The"black sponge"on T2WI image and hyperintensity on DWI image may be helpful to diagnose OAF.

The analysis of ovarian adenofibroma on clinical ,CT and MRI features / 实用医学杂志

Objective To explore the clinical,CT and MRI features of ovarian adenofibroma(OAF)and improve the diagnostic level. Methods Ninety-two cases of OAFs were analyzed retrospectively and summarized the features including clinical manifestations ,CT and MRI images. Results The probability of unilateral ,serous and benign OAFs were 91.3%,89.1%and 97.8%. OAFs associated with uterine or/and ovarian lesions was 40.2%. The clinical manifestations of OAF was not typical. These OAFs were divided into 2 groups according to whether or not accompanied with uterus or/ and ovarian lesions. The clinical manifestations and laboratory indexes were only found abnormal menstruation rate(P<0.05),but there were no significant difference between other indexes. The performance of CT plain scan and CT/MRI enhanced scan had not characteristic.The percentage of the"black sponge"on T2WI image and hyperintensity on DWI image of OAFs were 40.0% and 65.0%. Conclusion The"black sponge"on T2WI image and hyperintensity on DWI image may be helpful to diagnose OAF.

Clear Cell Adenocarcinoma Arising from Adenofibroma in a Patient with Endometriosis of the Ovary

Ovarian clear cell adenocarcinomas (CCACs) are frequently associated with endometriosis and, less often with clear cell adenofibromas (CCAFs). We encountered a case of ovarian CCAC arising from benign and borderline adenofibromas of the clear cell and endometrioid types with endometriosis in a 53-year-old woman. Regions of the adenofibromas showed transformation to CCAC and regions of the endometriosis showed atypical endometriotic cysts. This case demonstrates that CCAC can arise from CCAF or endometriosis.

Clinicopathological analysis of recurrent uterine Mullerian adenofibroma / 重庆医学

Objective To analyze and summarize the clinicopathological features of recurrent uterine Mullein adenofibroma . Methods 8 cases of recurrent uterine Mullein adenofibroma in our hospital from February 2003 to February 2014 were selected and analyzed on the clinical data for summarizing its clinicopathological features .Results In 8 cases of recurrent uterine M ullerian ade‐nofibroma ,the lobulated neoplasm could be found on outside of the cervix or inside of the uterus ,which was formed by the benign epithelial and mesenchymal tissue .Immunohistochemistry showed that the estrogen receptor (ER) was expressed in 8 cases ,some cases expressed progesterone receptor(PR) ,smooth muscle actin(SMA) ,CD10 and p53 ,and the mean average value of Ki‐67 (+ ) was 8% .Conclusion Recurrent uterine Mullerian adenofibroma has no specific clinicopathological changes ,its differential diagnosis with sarcoma is difficult .Only CD10 and Ki‐67 may be conducive to diagnosis .

The Ultrasound and MRI Findings of Uterine Adenofibroma: A Case Report

Adenofibroma is an extremely rare benign mullerian mixed tumor composed of epithelium and mesenchymal cells. Most uterine adenofibromas occur in the endometrium, but they rarely protrude into the vagina. To date, only a few such cases with the imaging findings have been reported. Therefore, we report here on the sonographic and magnetic resonance (MR) imaging findings of a case of endometrial adenofibroma protruding into the vaginal cavity in a 28-year-old woman. The uterine adenofibroma appeared as a large intracavitary echogenic mass containing multiple small internal cysts, and it was distending the vaginal cavity on transrectal sonography. T2- weighted MR images showed a large intracavitary mass with heterogeneous high signal intensity protruding into the vaginal cavity. On gadolinium-enhanced T1-weighted MR images, heterogeneous septa-like enhancement was noted in the mass. Although uterine adenofibroma is extremely rare, adenofibroma can be suggested as a possible diagnosis when an intracavitary uterine mass, with multiple internal small cystic components and enhancing septa-like structures, is protruding into the vaginal cavity on imaging.

Endometrial mullerian adenosarcoma after toremifene treatment in breast cancer patients: a case report / 부인종양

Toremifene is an anti-estrogen which has been shown to be effective in the treatment of breast cancer, and is thought to be a less uterotrophic agent than tamoxifen. The risk assessment concerning endometrial cancer has been inconclusive because of its rare use up to the mid-1990s. We report a case of an adenosarcoma, which is a very rare type of uterine malignancy, after toremifene treatment for 5 years in a breast cancer patient. After 1 year of toremifene use, the patient had a benign Mullerian adenofibroma. After an additional 4 years of toremifene treatment, the endometrial polypoid lesion was transformed into a Mullerian adenosarcoma. Although toremifene is a promising anti-estrogenic agent in the treatment of breast cancer patients, clinicians should not neglect the possibility of a uterine malignancy.

A case of adenofibroma of uterine cervix / 대한산부인과학회지

Adenofibromas of the uterine cervix, which are classified as benign tumors of mixed epithelium and mesenchymal cells, are extremely rare. Most common symptom is usually abnormal genital bleeding. It is very difficult to diagnose preoperatively. Recommended method of treatment is total hysterectomy, because it usually recurs. We experienced a case of adenofibroma of the uterine cervix with increased CA125, so report it with a review of the literature.

A Case of Uterine Adenofibroma / 대한산부인과학회지

Uterine adenofibroma is an extremely rare benign biphasic neoplasm that is classified into mixed m?llerian tumor. This mass lesion appears to be clinical and histologically benign but it should be differentiated from other malignant neoplasms of uterus. We have experienced a case of uterine adenofibroma in a 53-year-old woman and report this case with a brief review of literature.

A Case of Ovarian Leiomyoma accompanied with Ovarian Serous Adenofibroma / 대한산부인과학회잡지

Ovarian leiomyoma is a rare mesenchymal tumor, accounting for only 1% of all benign ovarian neoplasm. Most patients are asymptomatic and the tumors are usually found incidentally during operation or at autopsies. Ovarian adenofibroma is a rare benign tumor which originates from the germinal lining and the stroma of the ovary. This tumor may be solid, semisolid or cystic, depending on the relative amount of the epithelial and stromal component. We have experienced a case of ovarian leiomyoma accompanied with ovarian serous adenofibroma in a 42-year-old woman and report this case with a brief review of literature.

Malignant Endometrioid Adenofibroma of the Ovary: A case report

Ovarian endometrioid adenofibroma is rare and characterized by prominent stroma. Its histologic classification is controversial but the malignant counterpart is distinguished from the borderline by the presence of confluent growth pattern of epithelium with invasion of the stroma by the endometrioid cells. A fifty-year-old woman was admitted with one month history of abdominal enlargement. Total abdominal hysterectomy with bilateral salpingo-oophorectomy was performed under the clinical diagnosis of ovarian malignancy. Grossly, the righy ovary had round, encapsulated, solid and whitish gray mass which measured 9 cm in the greatest dimension and showed peripheral small cysts. Microscopic examination revealed that the tumor consisted of endometria type glands set in fibrous stroma. The glands varied from tubules to cysts and the lining cells showed complicated architectural pattern with occasional papillary infoldings, atypical mitosis and malignant nuclear characteristics. Some cysts of glands showed intraluminal mucin products. Stromal invasions by individual epithelial cells showing malignant characteristics were often found. A brief summary of the histopathologic aspect of this tumor is presented together with review of literatures.

Malignant Endometrioid Adenofibroma of the Ovary: A case report

Ovarian endometrioid adenofibroma is rare and characterized by prominent stroma. Its histologic classification is controversial but the malignant counterpart is distinguished from the borderline by the presence of confluent growth pattern of epithelium with invasion of the stroma by the endometrioid cells. A fifty-year-old woman was admitted with one month history of abdominal enlargement. Total abdominal hysterectomy with bilateral salpingo-oophorectomy was performed under the clinical diagnosis of ovarian malignancy. Grossly, the righy ovary had round, encapsulated, solid and whitish gray mass which measured 9 cm in the greatest dimension and showed peripheral small cysts. Microscopic examination revealed that the tumor consisted of endometria type glands set in fibrous stroma. The glands varied from tubules to cysts and the lining cells showed complicated architectural pattern with occasional papillary infoldings, atypical mitosis and malignant nuclear characteristics. Some cysts of glands showed intraluminal mucin products. Stromal invasions by individual epithelial cells showing malignant characteristics were often found. A brief summary of the histopathologic aspect of this tumor is presented together with review of literatures.

Juvenile Cellular Adenofibroma of Breast: A case report

Juvenile cellular adenofibroma of the breast is a unique neoplasm of the breast that should be differentiated from other important benign and malignant lesions of the juvenile breasts. We report a case with it's characteristic clinical, gross and histological features. The tumor was in the right breast with the size of 20 cm in maximum extent. This patient was also associated with hemihypertrophy of the right side. Microscopically the masses were characterized by prominent stromal cellularity associated with pericanalicular duct proliferation.

The clinicorpathological analysis of ovary adenofibroma and ovary cystadenofibroma / 解放军医学杂志

Objective To investigate the morbidity, clinical characteristics, diagnosis, the pathological classification, character and special behaviour of transformation and the tendency of development of ovarian adenofibroma and cystadenofibroma, so as to provide information for clinical work. Methods 7 patients of ovarian adenofibroma and cystadenofibroma were retrospectively analysed on their onset age, clinical manifestations, ultrasonographic findings, pelvic examination and postsurgical pathology. Results All the seven tumors were serous. The ultrasonography revealed a cystic mass in the overy or near the overy, with clear boundary, and regular in shape. Three tumors were papillary (with rich signal of circulation in the papillary wall in two cases). In one case there was rich circulatory signal in the wall of the cyst. Three tumors had neither papilla on the wall of the cyst, nor blood signal. Pathological examination showed that 4 tumors were benign, and three were borderline. Conclusion The tumor is more prevalent in women of childbearing age. Unilateral tumor is more common than bilateral. The tumor is mainly serous, most of them are benign, and malignant tumors are rare. Ultrasonograply may be helpful to defining the nature and differentiation of the tumor

A Case of Adenofibroma of Left Renal Pelvis / 대한비뇨기과학회지

A case of a 17 years old Korean adolescent who suffered from secondary hydronephrosis and chronic pyelonephritis of the left kidney due to a obstructive adenofibroma, a rare tumor, arising at the ureteropelvic junction of the left renal pelvis was presented.