Imaging Features of Biliary Adenofibroma With Malignant Transformation: A Case Report and Literature Review.

Biliary adenofibroma (BAF) is a rare benign tumor, but it has the potential for malignant transformation. The differentiation between benign and malignant forms of BAF before surgery is of great importance for clinical decision-making. We report a case of BAF with invasive carcinoma. The patient did not present any clinical symptoms but had a history of hepatitis B virus infection for more than twenty years. Magnetic resonance imaging (MRI) revealed a solid and cystic 4 cm mass in segment II of the liver exhibiting hypointense signals on T1-weighted images and intermediate-to-high intensity signals on T2-weighted images. Enhancement scanning revealed markedly rim-like enhancement on the arterial phase, with the left inter-hepatic artery as the tumor-feeding artery, and wash-out on the venous and delayed phases. To the best of our knowledge, BAF with invasive carcinoma is uncommon. Preoperative qualitative diagnosis based on imaging features can achieve the maximum benefit for patients.

MRI combined with clinical features to differentiate ovarian thecoma-fibroma with cystic degeneration from ovary adenofibroma.

OBJECTIVE: To explore the value of magnetic resonance imaging (MRI) and clinical features in identifying ovarian thecoma-fibroma (OTF) with cystic degeneration and ovary adenofibroma (OAF). METHODS: A total of 40 patients with OTF (OTF group) and 28 patients with OAF (OAF group) were included in this retrospective study. Univariable and multivariable analyses were performed on clinical features and MRI between the two groups, and the receiver operating characteristic (ROC) curve was plotted to estimate the optimal threshold and predictive performance. RESULTS: The OTF group had smaller cyst degeneration degree (P < .001), fewer black sponge sign (20% vs. 53.6%, P = .004), lower minimum apparent diffusion coefficient value (ADCmin) (0.986 (0.152) vs. 1.255 (0.370), P < .001), higher age (57.4 ± 14.2 vs. 44.1 ± 15.9, P = .001) and more postmenopausal women (72.5% vs. 28.6%, P < .001) than OAF. The area under the curve of MRI, clinical features and MRI combined with clinical features was 0.870, 0.841, and 0.954, respectively, and MRI combined with clinical features was significantly higher than the other two (P < .05). CONCLUSION: The cyst degeneration degree, black sponge sign, ADCmin, age and menopause were independent factors in identifying OTF with cystic degeneration and OAF. The combination of MRI and clinical features has a good effect on the identification of the two. ADVANCES IN KNOWLEDGE: This is the first time to distinguish OTF with cystic degeneration from OAF by combining MRI and clinical features. It shows the diagnostic performance of MRI, clinical features, and combination of the two. This will facilitate the discriminability and awareness of these two diseases among radiologists and gynaecologists.

Endometrial Adenofibroma in a Patient Receiving Toremifene: A Case Report

INTRODUCTION: Adenofibroma is a rare benign Müllerian mixed tumor composed of epithelial and mesenchymal cells. This tumor may occasionally be associated with toremifene therapy which is used as an adjuvant drug for breast cancer. CASE PRESENTATION: We describe a case of a 55-year-old woman with adenofibroma of the endometrium. This patient was receiving toremifene after surgery and neoadjuvant chemotherapy for breast cancer. She underwent a total abdominal hysterectomy and bilateral salpingectomy. There was no evidence of tumor residual or recurrence at 32 months of MRI follow-up. CONCLUSION: In conclusion, we report a rare case of endometrial adenofibroma in a patient receiving toremifene. It must be borne in mind that long-term toremifene therapy may increase the frequency of endometrial neoplasms.

Biliary adenofibroma: a precursor lesion of intrahepatic cholangiocarcinoma.

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.

Benign Biphasic Tumors of the Mullerian Tract - a Mimic of Phyllodes Tumor. A Clinical-Pathologic Description of 21 Cases.

OBJECTIVE: A review of the clinical-pathologic characteristics and outcomes of biphasic polyps occurring in the female genital tract, not meeting the diagnostic criteria of Mullerian Adenosarcoma (MA). METHODS: An archival database search was run, after IRB approval, between 2001 and 2019, using terminology such as "Mullerian adenofibroma," "atypical Mullerian adenofibroma," "polypoid adenofibroma," and "atypical polyp with increased stromal cellularity." Two pathologists (JW and MRQ) reviewed all the retrieved cases and documented the morphologic features with particular emphasis on the presence of any features of Mullerian adenosarcoma. Follow-up data were also abstracted. RESULTS: Twenty-one cases, 12 cervical and 9 endometrial lesions, constituted the study cohort. Patients ranged from 26 to 64 years (median 49 years). On review, 20 of 21 of those cases showed Phyllodes-like architectural patterns. However, only one case showed all four features of MA, all of which were focal and inconspicuous. Follow-up (median duration of 5 years) did not document any recurrences in any of the 21 cases after excision. CONCLUSION: This series adds to the growing body of literature affirming the existence of benign biphasic Mullerian polyps encountered in the endometrium and cervix that fall short of the Mullerian adenosarcoma diagnosis.

Mesonephric-like adenocarcinoma arising from endometrioid adenofibroma in a patient with in-utero exposure to diethylstilbestrol: A case report.

Mesonephric-like adenocarcinomas are newly classified rare neoplasms of the upper genital tract. They share identical features with mesonephric adenocarcinomas, with the exception of location. There is ongoing discussion on whether mesonephric-like adenocarcinomas arise from mesonephric remnants or are of Müllerian origin. A 65-year-old woman (G2P1) presented with pelvic pain. Transvaginal ultrasound revealed multiple fibroids, and a robotic total laparoscopic hysterectomy with bilateral salpingo-oophorectomy was planned. Intraoperatively, a complex mass with cystic and solid components was found on the left ovary, which also adhered to the rectosigmoid colon. Pathologic reports documented mesonephric-like adenocarcinoma that appeared to be arising in association with an endometrioid adenofibroma. This case is notable due to the patient's unique background of in-utero exposure to diethylstilbestrol (DES) and multiple gynecological malignancies within her mother, as well as the associated endometrioid adenofibroma. This case contributes evidence to two seemingly opposing theories of mesonephric-like adenocarcinoma histogenesis: the mesonephric remnant theory (supported by the patient's DES exposure), and the Müllerian theory (supported by the associated endometrioid adenofibroma).

Recurrent cervical adenofibroma progressing to adenosarcoma: a rare case report.

OBJECTIVES: Cervical adenofibroma is a rare form of mixed mesodermal tumor that can present as cervical polyps with a tendency for local recurrence and progression. Few cases progressing to adenosarcoma have previously been reported. We report a case of cervical adenofibroma progressing to adenosarcoma, and we seek to remind clinicians of the method and importance of the differential diagnosis of this disease. A fertile woman was admitted in our department for the eighth recurrence of a cervical polypoidal mass which for the past 10 years. Recurrence of cervical adenofibroma was confirmed by ultrasound and MRI. A wide local excision under hysteroscopy was performed due to her strong desire to preserve the uterus. Surgical pathology and immunohistochemical interpretation revealed cervical adenosarcoma. A hysterectomy with conservation of the ovaries was recommended, with regular follow-ups for evidence of disease recurrence. CONCLUSIONS: Differential diagnoses of cervical adenofibroma are hard to prove. Adenosarcoma should be ruled out, especially in women presenting with recurrent cervical polypoidal masses. A combined histological/immunohistochemical investigation is mandatory.

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.

Multimodal imaging findings of intrahepatic cholangiocarcinoma arising from a biliary adenofibroma: a case report with radiological-pathological correlation.

PURPOSE: Biliary adenofibroma is a solid microcystic epithelial neoplasm in the liver, comprising microcystic and tubuloacinar glandular tissues lined by a non-mucin secreting biliary epithelium and supported by a fibrous stroma. It is an extremely rare benign tumor with potential for malignant transformation. Herein, we report the case of a 64-year-old woman diagnosed with intrahepatic cholangiocarcinoma arising from biliary adenofibroma. METHODS: Imaging studies revealed a tumor of 50 mm diameter, consisting of two components in S1 of the liver. The ventral portion of the tumor showed an ill-defined mass with early peripheral and gradual centripetal enhancement invading to the middle hepatic vein on computed tomography (CT), diffusion restriction on magnetic resonance images, and high fluorine-18-2-deoxy-D-glucose (FDG) uptake on positron emission tomography, like conventional intrahepatic cholangiocarcinoma. The dorsal portion showed a well-defined and low-attenuated mass with heterogeneous early enhancement and partial wash-out on CT, marked hyperintensity on heavily T2-weighted images, and low FDG uptake. The patient subsequently underwent extended left hepatectomy. RESULTS: Pathologically, the former was diagnosed as cholangiocarcinoma and the latter as biliary adenofibroma. We discuss the radiological-pathological correlation of the tumor with a literature review. CONCLUSION: Preoperative diagnosis of biliary adenofibroma is extremely challenging; however, clinically, it is crucial not to miss the presence of malignant findings.

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.

A Case of Multicystic Biliary Hamartoma with a Marked Peribiliary Gland Component Successfully Treated by Purely Laparoscopic Anatomical Liver Resection.

BACKGROUND: Multicystic biliary hamartoma (MCBH) is an extremely rare benign liver lesion characterized by a gross well-circumscribed multicystic honeycomb appearance. This report presents a MCBH case with a marked peribiliary gland component which showed unusual histology. CASE PRESENTATION: A 63-year-old Japanese male was admitted to our hospital for a detailed examination of a hepatic cystic lesion, which was originally detected 14 years ago and had slowly enlarged. A preoperative imaging study revealed a well-demarcated multicystic lesion without communication to the biliary tracts. The possible clinical diagnoses were mucinous cystic neoplasm (MCN) or MCBH. The lesion was successfully resected by purely laparoscopic right anterior sectionectomy. The cut surfaces of resected specimens grossly exhibited a well-circumscribed multicystic lesion with a thick septum. Histologically, the cyst wall was covered by cuboidal epithelial cells resembling epithelium of the bile duct while abundant small ducts, which morphologically resembled peribiliary glands, were observed among the fibrous stroma of the thick septum. Although possible pathological diagnosis varied, including intrahepatic cholangiocarcinoma, intraductal papillary neoplasm of the bile duct, biliary adenofibroma, MCN and MCBH, the lesion was finally diagnosed as MCBH with a marked peribiliary gland component. CONCLUSIONS: MCBH can contain abundant peribiliary glands in the fibrous stroma. A pathologist should be careful not to diagnose such peribiliary glands in MCBH as neoplastic glands.

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.

A case report of adenofibroma arising from the seminal vesicle: A rare giant pelvic mass.

Primary seminal vesicle tumors are extremely rare. Several rare pathological types of primary seminal vesicle tumors have been reported, such as adenocarcinoma, but there is no report on adenofibroma. We report the first case of adenofibroma arising from the seminal vesicle. A 50-year-old man, with no history or clinical evidence of any other tumors, accidentally found a pelvic mass during an ultrasound examination. As the mass grew, the patient developed mild constipation, without genitourinary or other symptoms. All laboratory examinations were normal. MRI of the pelvis revealed a mixed density, measuring 11.7×9.9×8.2cm, well circumscribed mass. The rectum, bladder, prostate and lymph nodes were normal. We successfully performed the open surgery and removed the mass. Histopathological results confirmed that the mass was a primary seminal vesicle adenofibroma. In literature, we found that for biphasic differentiated tumors, it is easy to reduce the accuracy of pathological diagnosis because of insufficient puncture. Therefore, preoperative puncture biopsy for seminal vesicle tumors should be investigated further.

Atypical Biliary Adenofibroma of the Liver: Related Treatment and Performance.

Biliary adenofibroma is an extremely rare benign liver tumor, but it may be a precancerous lesion of cholangiocarcinoma. So far, only 29 cases have been reported in the literature. A 30-year-old woman was admitted to our department for upper abdomen mass. The computed tomography scan showed a huge cystic and partly substantial mass between the left lobe of the liver and the descending duodenum, which was considered to be an exophytic tumor derived from the left lobe of the liver. Laparoscopic liver segment IVb resection and cholecystectomy were performed. Microscopic examination showed that the tumor was composed of glandular cavities of varying sizes and fibrous interstitium. The glandular cavity was covered with cubic or columnar epithelium without atypia. Some of the mesenchymal cells are myofibroblast-like and spindle-shaped with red-stained cytoplasm. The mesenchymal cells in some areas proliferate densely with moderate atypia. It was considered to be an atypical biliary adenofibroma with focal necrosis and active cell proliferation which may have malignant transformation potential. There was no recurrence and metastasis at a 6-month follow-up. Biliary adenofibroma is a rare benign tumor derived from the bile duct, but it may progress to malignancy and develop distant metastasis. It is difficult to distinguish it from other liver tumors through imaging examination and the gold standard of diagnosis is histopathological examination. Close clinical follow-up is recommended.

Malignant transformation of biliary adenofibroma combined with benign lymphadenopathy mimicking advanced liver carcinoma: A case report.

BACKGROUND: Biliary adenofibromas (BAFs) are rare primary hepatic neoplasms, some of which can potentially undergo malignant transformation. Here, we describe a rare case of malignant transformation of BAF. CASE SUMMARY: A 51-year-old female was referred to our hospital with epigastric pain. Computed tomography showed a solitary liver mass combined with the enlargement of multiple mediastinal and cervical lymph nodes, clinically mimicking a liver carcinoma with extensive lymph node metastasis. However, core needle biopsy suggested BAF with malignant transformation. Finally, the patient underwent curative resection of the neoplasm and was recurrence-free for 12 mo. CONCLUSION: Our case serves as an example of a rare manifestation of BAF. Our report and the previously published experience, reinforce that curative resection should be considered the primary treatment for BAFs with malignant transformation, leading to a favorable prognosis.

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.

Pulmonary adenofibroma with central liquefaction necrosis: A case report.

Pulmonary adenofibroma (PAF) is a rare benign tumor. Computed tomography (CT) imaging of PAF show well-defined, homogeneous and solitary nodules. To the best of our knowledge, there is no report of PAF presenting with central liquefaction necrosis on CT images. The present study reports the case of a 70-year-old man who was hospitalized due to an inguinal hernia without respiratory symptoms. Chest CT scan revealed a tumor (~6.5x5.5x4.4 cm) in the lower lobe of the left lung, characterized by uneven density and unclear boundary with the pleura. Contrast-enhanced scan revealed that the lesion was slightly enhanced and liquefaction necrosis appeared in its center. Wedge resection was performed using video-assisted thoracic surgery. Histopathological and immunohistochemical examination confirmed the diagnosis of PAF.