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A type of cholangiocarcinoma (CCA) characterized by peculiar histologic patterns and underlying adenofibromatous lesions has been reported in the literature mostly as individual case reports. This study aims to further clarify the defining characteristics of this spectrum of lesions. Clinicopathologic analysis of 8 biliary tumors with tubulocystic architecture arising in the background of adenofibroma-type lesions was performed. Three of these were also investigated with next-generation sequencing with a 174 genes panel. The patients were 5 males and 3 females, with a mean age of 64.6. All tumors were intrahepatic except for one perihilar that protruded into soft tissues. The mean size was 4.4 cm. At histology, all cases showed a peculiar and cytologically bland tubulocystic pattern that closely resembled tubulocystic-type kidney cancers, including back-to-back microcystic units that formed relatively demarcated nodules, and occurring in the background of adenofibromatous lesions. One case showed perineural invasion by otherwise deceptively benign-appearing microcystic structures, one had areas transitioning to intraductal tubulopapillary neoplasm, and 3 cases harbored more conventional small-duct CCA foci. In those 3 cases, both the tubulocystic and conventional CCA components were investigated by next-generation sequencing separately, and they shared the molecular alterations, including recurrent mutations in chromatin remodeling genes, such as ARID1A , BAP1 , and PBRM1 , and the actionable FGFR2-MCU fusion gene. In the limited follow-up, all but one were alive and free of disease after surgical resection. In conclusion, we described a distinct entity of CCA with specific histo-molecular features, for which we propose the designation of tubulocystic carcinoma of bile ducts., Competing Interests: Conflicts of Interest and Source of Funding: A.S.: consulting and speaker bureau from: MSD, Incyte, Medica s.r.l., NTP. C.L.: consulting and speaker bureau from: MSD, Medica s.r.l., NTP. For the remaining authors, none were declared., (Copyright © 2024 Wolters Kluwer Health, Inc. All rights reserved.)

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., (© The Author(s) 2024. Published by Oxford University Press on behalf of the British Institute of Radiology. All rights reserved. For permissions, please email: journals.permissions@oup.com.)

Competing Interests: The authors declare no conflict of interest.

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., Competing Interests: The authors have no conflicts of interest to disclose., (Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.)

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., (© 2022. The Author(s).)

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., Competing Interests: The authors declare no conflict of interest., (Copyright © 2021 by the International Society of Gynecological Pathologists.)

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., (© 2020 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

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., (© 2020 John Wiley & Sons Ltd.)

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.

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.

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.

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., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.)

Semaphorins are a large family of evolutionarily conserved morphogenetic molecules that are associated with repelling axonal guidance. Intriguingly, recent researches indicate that semaphorins are involved in cancer progression. Semaphorin 4C (SEMA4C) has long been considered a neuronal migration gene, but we detected that it is also highly expressed in many malignant human cancers. During an investigation of subcutaneous tumor models, we found that SEMA4C expression promoted tumor growth and progression. We discovered that SEMA4C was involved in maintaining tumor cell self-renewal, likely by regulating the p53 pathway. Inhibiting the expression of endogenous SEMA4C in tumor cells impaired growth and induced senescence and cell-cycle arrest in the G 2 -phase. In addition, we found that SEMA4C induced the production of angiogenin and colony-stimulating factor-1 (CSF-1) in tumor cells by activating the NF-κB pathway in a plexinB2-dependent manner. In conclusion, SEMA4C expression in breast cancer cells promotes cancer cell proliferation, macrophage recruitment, and angiogenesis. Thus, inhibition of SEMA4C activity may be a novel therapeutic strategy for human breast cancer. IMPLICATIONS: In breast cancer, therapeutic targeting of the SEMA4C pathway may prevent tumor growth, angiogenesis, metastasis, and progression., (©2019 American Association for Cancer Research.)

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.

Pulmonary adenofibroma (PAF) is a rare neoplasm that may be related to solitary fibrous tumor (SFT). A subset of PAFs harbor the NAB2-STAT6 fusion that is typical of SFT, but a significant proportion do not. Their distinction is clinically important as SFTs can potentially have an aggressive clinical course, while there has been no report of a PAF behaving in a malignant fashion. We report a case of a 60-year-old male who developed a SFT and PAF in the same lung. The SFT harbored a NAB2-STAT6 fusion, while the PAF did not have any identifiable fusion. This case represents the first instance of a single patient with both of these tumors occurring simultaneously in the same lung.

A solitary firm nodule was found in the lung of a sika deer (Cervus nippon yesoensis). Histologically, it was a biphasic lesion composed of epithelial and stromal cell elements and exhibited a leaf-like growth pattern. The epithelial cells were immunohistochemically positive for pancytokeratin, cytokeratin 7, napsin A, and thyroid transcription factor-1, and the stromal cells were positive for vimentin and partially positive for desmin and α-smooth muscle actin. These observations were consistent with pulmonary adenofibroma, which is an extremely rare lesion in humans. To the best of our knowledge, this is the first reported case of pulmonary adenofibroma in an animal.

Cholangiocarcinoma is the second most common hepatobiliary cancer following hepatocellular carcinoma, and 20% to 25% are intrahepatic. We describe 2 cases of intrahepatic cholangiocarcinoma arising within unusual and rare hepatic lesions, fibropolycystic liver disease form of ductal plate malformation and biliary adenofibroma, whose association with malignancy is rarely reported in the literature.

Ovarian clear cell adenofibroma is uncommon, and borderline clear cell adenofibroma (low malignant potential) is extremely rare. Borderline clear cell adenofibromas may represent the precursor lesion of clear cell adenocarcinoma of the ovary, but this has not been established. We present a case of a woman in her mid-50s with a clear cell adenofibroma ranging from benign to borderline to frankly invasive. While some clear cell adenocarcinomas are thought to arise from endometriosis, this range of findings supports the theory that some ovarian clear cell adenocarcinomas originate from borderline tumors.

Objective: MicroRNA-331 (miR-331) has shown regulatory activity against several genes whose expression has been claimed to be deregulated in breast tumors, including that of epidermal growth factor receptor 2 (HER2). Herein, the clinical value of miR-331 expression was investigated by analyzing its levels in breast benign and malignant tumors., Methods: The expression levels of miR-331 were quantified via real-time PCR in 130 malignant and 66 benign breast tissue specimens collected after surgical resection of primary tumors. The generated data were analyzed by applying several statistical tests in order to examine the relationship of miR-331 expression with various established clinicopathological features and survival data of patients., Results: Our data showed that miR-331 was overexpressed in malignant breast tumors compared to their benign counterparts both overall (P = 0.026) and individually when the subgroups of fibroadenoma and invasive ductal carcinoma were analyzed with each other (P = 0.001). ROC curve analysis confirmed the diagnostic value of these variations, providing an AUC value equal to 0.597 (P = 0.026) and 0.663 (P = 0.001), respectively. Furthermore, miR-331 levels were elevated (P = 0.026) in ductal cancerous specimens compared to the lobular ones but failed to correlate with other clinicopathological features or survival data of the breast cancer patients., Conclusions: Our results provide evidence that miR-331 levels might provide valuable information regarding the differential diagnosis of benign and malignant breast tumors but present no prognostic value for breast cancer., (Copyright © 2018. Published by Elsevier Inc.)

The Bi-RADS (Breast Imaging Reporting and Data System) classification developed by the ACR (American College of Radiology) is the classification system for radiological images recommended for breast cancer screening. The ACR 4 microcalcification is an indeterminate or suspected abnormality with 2-95% probability of malignancy, according to studies. This disparity pushed us to conduct this retrospective study of 181 patients in the Department of Obstetrics and Gynecology I at the Hassan II University Hospital, Fez, over a period of 5 years. This study aimed to report the histological results of breast lesions radiologically classified as ACR4 in order to assess their radio-histological correlation and to improve therapeutic approach. All patients underwent breast imaging examinations and then anatomopathologic examination was performed using different techniques. There was a clear predominance of benign lesions with a rate of 62% versus 29% of malignant lesions and only 9% were intermediate lesions. Adenofibroma was the most common histological finding (30% of cases), invasive ductal carcinoma was the most frequent malignant lesion (17% of cases). We performed a literature review which showed that our results were in line with findings of other studies, with a positive predictive value of 29%. Nevertheless, ACR classification divided into subcategories 4a, b and c should be used due to the significant number of unnecessary surgical interventions.

We present an autopsy case of a "so-called adenosarcoma with ossification of the liver" in a 63-year-old man. Macroscopically, the well-circumscribed tumor with portal vein invasion was observed in the right lobe of the liver. The cut surface of the tumor had a solid and microcystic appearance. Microscopically, the tumor was characterized by a benign epithelial component and a malignant mesenchymal component. We believe the presence of biliary adenofibroma-like areas and the von Meyenburg complexes suggests that the tumor is possibly associated with a biliary adenofibroma. In addition, the present tumor was unique in that it showed scattered heterotopic ossification. Immunohistochemical study showed that the mesenchymal atypical spindle cells had characteristics of undifferentiated mesenchymal cells. This is the first report of a primary hepatic so-called adenosarcoma., (Copyright © 2017 Elsevier Inc. All rights reserved.)

Microscopic, heterotopic extraovarian sex cord-stromal proliferations have only recently been reported in the literature. We describe the largest series to date, of 30 cases of microscopic, incidentally detected, heterotopic extraovarian sex cord-stromal proliferation, in women aged 25-79 yr who had undergone surgery for a range of benign and malignant gynecologic conditions. In 14 patients the foci of proliferation comprised ovarian cortical stroma, in some cases with an ovarian fibroma-like appearance. Ten cases of adenofibroma and cystadenofibroma were also identified, including 1 Brenner adenofibroma; 2 cases comprised both ovarian cortical stroma and serous cystadenofibroma; 4 cases showed sex cord proliferation resembling microscopic adult granulosa cell tumors. Immunohistochemistry, where possible, confirmed the sex cord nature of the heterotopic proliferations. The foci of proliferation were <1-7 mm, and most were at the fimbrial end of the fallopian tube. These proliferations are likely to be encountered with increasing frequency as we sample the adnexa more extensively. Previous reports postulated that the proliferations probably represent embryonic rests caused by anomalous migration but we suggest that incorporation of exposed ovarian parenchymal tissue into the fimbrial stroma at the time of ovulation may be another possible cause.

Multiple adenofibromas or adenofibromatosis is characterized by the presence of at least 3 mono- or bilateral adenofibromas increasing significantly in size, causing trophic disorders. We report the case of a 46-year old female patient who had been followed up since she was 30 years old for bilateral adenofibromatosis that required 4 surgical procedures. Patient's medical data were collected in the Department of Respiratory Diseases at the Ibn Rochd University Hospital Center, Casablanca. Basing on preoperative assessment before bilateral mastectomy, the patient underwent chest X-ray that showed opaque right hemithorax exerting compression on the mediastinum. Clinical examination showed effusion syndrome in the right hemithorax and left supraclavicular cervical adenopathy. Pleural puncture biopsy confirmed the presence of poorly differentiated invasive carcinoma in the pleura, supporting breast origin. Bronchoscopy after pleural puncture objectified infiltration of the whole bronchial tree; biopsies confirmed the anatomopathological results. The recommended treatment strategy was based on multidrug chemotherapy. Patient's evolution was marked by the occurrence of hepatic metastases. This study shows that adenofibromas require regular monitoring given the risk of trasformation to breast cancer, which is a frequent cause of pleuropulmonary metastases.

Ovarian borderline tumors can show histologic features, such as different degrees of cellular proliferation, eosinophilic cells, autoimplants, and adenofibromatous architecture, the importance of which is not known. The aim of the study was to describe these features and correlate them with clinical characteristics. Eighty-three ovarian borderline tumors were studied for the aforementioned features. These were correlated with clinicopathologic features. Epithelial proliferation was associated with the T stage in serous tumors (P=0.0009), but not in mucinous tumors (P=0.97). It was positively associated with bilateral tumors (P=0.01) and the presence of autoimplants (P<0.0001). It was associated with the presence of eosinophilic cells, as tumors with extensive eosinophilic cells had a mean proliferation of 80.7%, for those with no such cells it was 23.8% (P<0.0001), and for those with a limited presence of eosinophilic cells it was 48.7% (P=0.03). Cellular proliferation was not associated with the size of the tumor. An adenofibromatous architecture was associated with unilateral tumors (P=0.02) and showed a trend (P=0.08) with regard to T stage in serous tumors. It was not associated with the size of the tumor. The presence of autoimplants was marginally associated (P=0.07) with bilateral tumors and it was not associated with the size of the tumor or the T stage. The presence of eosinophilic cells was not associated with the T stage, the size of the tumor, or bilateral tumors. The degree of epithelial proliferation, autoimplants, adenofibromatous architecture, and the presence of eosinophilic cells are important features in ovarian borderline tumors.

Sclerosing polycystic adenosis (SPA) is a rare benign neoplasm of the salivary gland which resembles the fibrocystic disease of the breast clinically as well as morphologically. This entity has varied morphological presentation on fine needle aspiration. Only a few case reports and occasional case series are found in the literature describing its cytology. Here we are presenting a case of SPA in the parotid gland in a 13-year-old male patient who presented with a slow growing infra-auricular mass since one year without any other symptoms. Radiological investigation suggested a possibility of matted level II cervical lymph nodes. Fine needle aspiration of the same showed only crystalloids in a dirty background. Hence, a possibility of retention cyst was given and excision was advised. On histopathological examination, the mass was diagnosed as Sclerosing polycystic adenosis of the parotid gland. Since SPA is a rare entity with diverse morphological features, it is more likely to be misdiagnosed on cytology. Recognition of this benign entity is important since the differential diagnosis includes other more common benign and malignant salivary gland neoplasm particularly mucoepidermoid carcinoma and other tumors with cystic and oncocytic features. Repeat aspiration from multiple sites should be considered in cystic lesions of the salivary gland so that its varied cytological components can be yielded to help with the diagnosis. Diagn. Cytopathol. 2017;45:640-644. © 2017 Wiley Periodicals, Inc., (© 2017 Wiley Periodicals, Inc.)

Aims: Pulmonary adenofibromas are rare benign fibroepithelial tumours of the lung with unknown histogenesis and an indolent clinical behaviour. Their stroma resembles that of solitary fibrous tumours, whereas the glands are composed of respiratory epithelium organized in a phyllodes-like architecture. Differentiation of pulmonary adenofibromas from other more aggressive intrathoracic tumours is clinically relevant. However, their biology is unknown. Here, we sought to characterize pulmonary adenofibromas at a clinicopathological level and to define whether they could be underpinned by a highly recurrent somatic genetic alteration akin to tumours with similar morphology., Methods and Results: Seven pulmonary adenofibromas were subjected to immunohistochemical analysis for thyroid transcription factor 1 (TTF1), napsin A, cytokeratin 7, E-cadherin, CD99, CD34, CD31, STAT6, oestrogen receptor (ER), progesterone receptor, androgen receptor, bcl-2, and vimentin, as well as electron microscopy and capillary sequencing on microdissected samples to evaluate the presence of NAB2-STAT6 fusion genes and MED12 exon 2 mutations in their discrete components. A control group comprising pulmonary solitary fibrous tumours, pulmonary hamartomas and breast fibroadenomas was also analysed. We confirmed that the stromal elements of pulmonary adenofibromas pertain to the fibroblastic lineage, and show ER overexpression in 71% of cases, whereas the epithelium consists of TTF1-positive, E-cadherin positive bronchiolar elements. A highly recurrent NAB2-STAT6 fusion variant (exon 4-exon 2) was detected in the stroma but not in the epithelium. No MED12 mutations were identified., Conclusions: Here, we demonstrate that pulmonary adenofibromas are neoplastic lesions harbouring the molecular hallmark of solitary fibrous tumours., (© 2017 John Wiley & Sons Ltd.)

Biliary adenofibroma is a rare primary hepatic neoplasm, recognized in the World Health Organization classification, although only 14 cases have been reported to date. This series includes extended follow-up from 2 of the early case reports and 4 novel cases. Clinical history and histology were reviewed in all 6 cases. Tumor DNA was analyzed for point mutations by multiplex polymerase chain reaction and copy number alterations by array comparative genomic hybridization. The patients included 4 females and 2 males presenting between 46 and 83 years of age, with tumors ranging from 7 to 16 cm in diameter. The tumors had similar morphology, with tubules and cysts lined mainly by bland to mildly atypical cuboidal epithelium embedded in fibrous stroma. Multiplex polymerase chain reaction did not identify mutations in 4 tumors tested. Three tumors tested by array comparative genomic hybridization showed chromosomal copy number alterations, including 1 with amplifications of CCND1 and ERBB2. Three patients underwent resection with no recurrence at 21, 20, and 3 years of follow-up. One patient is alive after 14 months with no resection. Two patients with margin-positive resections had local recurrence at 1 and 6 years after surgery. No patient had distant metastasis. The distinct morphology and multiple clonal cytogenetic alterations in biliary adenofibromas indicate that the lesions are neoplastic. Amplifications of CCND1 and ERBB2 are not typical of benign neoplasms, and suggest that these tumors may have the ability to behave aggressively. However, the clinical outcomes in these patients suggest the neoplasms are only slowly progressive.

We are reporting one case of multiple pulmonary adenofibromas in a 57-year-old non-smoking female. Ten well-circumscribed masses were identified in both lungs. The masses are characterized by gland-like structures lined by a single layer of simple cuboidal or columnar epithelium. The stroma is abundant and demonstrates compact spindle-cells. The epithelial component is generally positive for CK7, TTF-1, Napsin A. The stromal component displays expression of vimentin, desmin, SMA, h-CALD, ER, PR, Bcl-2, and is negative for CD34, CD117, CD99. We are postulating that the possible histogenesis of these lesions is via proliferation of mesenchymal component of the peribronchial wall, which entraps the epithelium as it expands. Hitherto, this is the first case with multiple lesions reported. Currently, the patient is 11 months post-surgery and doing well., (© 2016 by the Association of Clinical Scientists, Inc.)

Purpose: To retrospectively compare the MR findings of histological subtypes of clear cell carcinomas (CCCs) of the ovary., Materials and Methods: This single-center retrospective study was approved by the institutional review board, and the requirement for informed consent was waived. Between April 2005 and August 2015, we found 51 consecutive patients with histopathologically proven CCCs. Among them, 44 CCCs in 37 patients who underwent preoperative MR imaging were included. CCCs were pathologically divided into three subgroups: (1) four clear cell adenofibroma-associated CCCs, (2) 21 endometriosis-associated CCCs, and (3) 19 indeterminate CCCs. The statistical tests were used to compare the frequency of qualitative assessments and value of quantitative measurements among the histological subtypes., Results: On T2-weighted images, hypointense areas within solid components were more frequently observed in clear cell adenofibroma-associated CCCs [3/4 (75%)] than in the remaining CCCs [1/40 (3%)] (p < 0.01), and the signal intensity ratios (SIRs) of solid components were significantly lower in clear cell adenofibroma-associated CCCs (0.27 ± 0.13) than in the remaining CCCs (0.61 ± 0.18) (p < 0.01). On T1-weighted images, hyperintensities of cystic components were more frequently observed in the endometriosis-associated CCCs [17/21 (81%)] than in the remaining CCCs [5/20 (25%)] (p < 0.01), and the SIRs of cystic components were significantly higher in endometriosis-associated CCCs (2.99 ± 1.51) than in the remaining CCCs (1.82 ± 1.10) (p < 0.01)., Conclusion: MR features may help differentiate between adenofibroma-associated and endometriosis-associated CCC.

Identifying precursor lesions of cholangiocarcinoma (CC) is relevant for early diagnosis and management. While for distal, perihilar and large duct cholangiocarcinoma, premalignant lesions are being recognized and studied actively, precursors of peripheral CC are still poorly described. Biliary adenofibroma is a rare benign tumor that has been proposed as a possible precursor lesion of peripheral CC. In the present report, we describe a unique case of a peripheral ICC that originated from a benign biliary adenofibroma with areas of atypical papillary proliferation, which is an expression of a multistep process of carcinogenesis., (Copyright © 2016 Elsevier GmbH. All rights reserved.)

Aims: The carcinogenesis of ovarian clear cell carcinoma (CCC) has been hypothesized to comprise two different pathways: an adenofibroma-carcinoma sequence and an endometriosis-carcinoma sequence. However, the difference in the genetic basis of these two pathways remains unclear. Recent studies have suggested that an ARID1A mutation and the loss of the corresponding protein, BAF250a, are frequent events in CCC. Herein, we investigated the difference in the loss of BAF250a expression in adenofibroma-related CCC and endometriosis-related CCC., Methods and Results: In total, 93 cases of surgically treated CCC were evaluated. The presence of adenofibroma and endometriosis associated with carcinoma was determined by reviewing haematoxylin and eosin-stained slides for each case. BAF250a expression in carcinoma was examined immunohistochemically. The loss of BAF250a expression was detected in carcinomas in 50 of 93 (54%) cases, including five of 18 (28%) with adenofibroma alone, 30 of 45 (67%) with endometriosis alone, eight of 18 (44%) with both conditions and seven of 12 (58%) with neither condition. The loss of BAF250a expression was significantly less frequent in CCC cases with adenofibroma than in cases with endometriosis (P = 0.01, Fisher's exact test)., Conclusions: The action of ARID1A in carcinogenesis differs between adenofibroma-related CCC and endometriosis-related CCC., (© 2015 John Wiley & Sons Ltd.)

Tamoxifen is a widely used in anti-oestrogen treatment of breast cancer. Previous reports showed that tamoxifen is associated with proliferative endometrial lesions. We herein reported an unusual case of concurrent hyperplastic lesions in the uterine cavity and right ovary in a 45-year-old woman with tamoxifen therapy. Regular vaginal ultrasonography showed the progressive endometrial thickening and right ovary enlargement during the period of drug use. Both lesions in the uterine cavity and right ovary showed characteristics resembling that of Müllerian adenofibroma. There were also foci of endometriosis in her bilateral ovarian surfaces. We suggest that women taking tamoxifen with a known history of endometriosis should be followed with transvaginal ultrasonography periodically.

We reported a case of metanephric adenofibroma in a 10-year-old boy to describe the clinical, radiologic, and pathologic features and discuss its treatment and differential diagnosis. Nephrectomy was performed for the patient; final histopathologic evaluation was that of a metanephric adenofibroma. Epithelial and stromal elements were both positive for WT-1, Vimentin, PAX2, and the epithelial tumor cells were also positive for S100, AE1/AE3, PAX8, CK8/18, EMA and a few cells were positive for CK7. Larger vessel wall components were positive for SMA, Des, caldesmon while capillary components were positive for CD10, CD31, and CD34. CA-9, α-inhibin and CD-56 were negative in the neoplasm. The Ki-67 labeling index was <1%. Metanephric adenofibroma is a rare benign renal tumor; the diagnosis of it relies on pathology and immunohistochemistry. As its rarity, there is no standard treatment for this disease. The majority of patients underwent nephrectomy and had good prognosis, as it is a benign neoplasm.

Biliary adenofibroma (BAF) is a rare, benign liver tumor. Herein, we report a case of BAF with histological features of imminent malignant changes. Ultrasound and CT revealed a solid 2.5-cm mass in the right liver lobe. The patient was asymptomatic and had no past medical history including liver disease. A general examination that included the abdomen and the laboratory data were normal. Because of the increase of its size, this tumor was surgically resected. Grossly, a 3.5-cm nodular mass abutted on the hepatic capsule was found, and its cut surface showed a well-circumscribed, whitish, and firm lesion that showed microcystic changes in the periphery and solid changes in the central parts. Histologically, the tumor showed a proliferation of tubulocystic structures embedded in a fibrous stroma. Microcysts were prevalent in the periphery, while tubular components with abundant fibrous stroma were in the central parts. The tubules were variably dilated and branched. This case closely resembled the previously reported cases of BAF, except that there were complicated papillary projections with fine fibrovascular cores in some of the microcysts and that the epithelial component in papillary projections showed dysplastic changes and increased cellular proliferative activities, implicating ominous features of imminent malignant changes. These dysplastic and papillary changes may be an intermediate lesion leading to malignancy, which have occasionally been reported in BAF.

Pulmonary adenofibroma is a rare benign biphasic tumor of the lung composed of epithelial and stromal components. We report 3 cases of this unusual lesion of lung in a male (25 years old) and 2 female (40 and 55 years old) patients. Breathlessness on exertion and mild left-sided chest pain of 1 month's duration were the main concerns in 2 patients, whereas the third had cough and hemoptysis for 3 months. Chest radiograph and computed tomography scan revealed a well-circumscribed, subpleural homogenous mass in left lower chest fields in 2 cases and solid-cystic lesion in left upper lobe in the third patient. All 3 patients underwent lobectomy, following biopsy in 2 cases. Histology revealed a well-circumscribed lesion composed of complex glandlike spaces lined by cuboidal to columnar epithelium surrounded by a hyalinized spindle-cell fibroblastic proliferation reminiscent of adenofibroma of the female genital tract or fibroadenoma of the breast. Immunohistochemical examination supported the diagnosis of a benign pulmonary adenofibroma. All 3 patients were are alive and doing well with no evidence of recurrent or metastatic disease. Diagnosis on biopsy can be challenging and may be misinterpreted as well-differentiated adenocarcinoma with extensive fibrosis or low-grade sarcoma. Frozen-section consultation will be a valuable adjunct in planning for limited lung resection of this benign lung lesion. Although we described 3 cases of pulmonary adenofibroma, still this is the largest published series of this rare entity till date. The possible histogenesis and various differential diagnoses are discussed along with literature review., (Copyright © 2014 Elsevier Inc. All rights reserved.)

We retrospectively evaluated pelvic magnetic resonance imaging including diffusion-weighted imaging (DWI) of 16 ovarian lesions (5 adenocarcinofibromas, 2 borderline adenofibromas, and 9 benign adenofibromas). All adenocarcinofibromas were detected as large solid areas of strong high signal on DWI, and seven of nine benign adenofibromas and both borderline adenofibromas demonstrated no areas of high signal or small areas of weak high signal. Solid components that appear as areas of strong high signal on DWI might represent a characteristic finding of adenocarcinofibromas., (Copyright © 2014 Elsevier Inc. All rights reserved.)

Objective: To study the clinicopathologic features and differential diagnosis of recurrent Müllerian adenofibroma (MAF) of the uterus., Methods: Clinicopathologic information of 7 cases of recurrent MAF of uterus was retrieved from January 1992 to April 2006 and compared with 12 cases of MAF without recurrence and 14 cases of low-grade Müllerian adenosarcoma (MAS). EnVision immunohistochemistry of estrogen receptor (ER), progesterone receptor (PR), smooth muscle actin (SMA), CD10, Ki-67 and p53 were performed in all cases., Results: All cases of recurrent MAF of the uterus were polypoid, lobulated, and broad based mass arising from the corpus or cervix. Microscopically, the tumor consisted of benign epithelial and mesenchymal components with low mitotic activity ( ≤ 1/10 HPF). The clinical and pathologic features of 3 recurrent tumors were similar to their primary tumors, while 4 cases of recurrent tumor presented with focally higher cellularity and mitotic activity, meeting the diagnostic criteria of adenosarcoma. The stromal expression patterns of ER, PR, SMA and p53 in recurrent MAF were similar to those of clinically benign MAF and low-grade MAS. Negative or focally positive stromal cell expression of CD10 was seen infrequently in recurrent MAF (1/7) and clinically benign MAF (1/12). In contrast, a moderate to strong CD10 staining was frequently seen in MAS (9/14, P < 0.05). The difference of Ki-67-labeling index between MAF and MAS did not reach a statistical significance (P > 0.05). Ki-67-labeling index increased in areas of periglandular stromal cuffing as compared with interglandular areas in all MAS cases, but it was not observed in either recurrent MAF or clinically benign MAF cases., Conclusions: Recurrent MAF may be associated with aggressive behavior. It is difficult to distinguish MAF from low-grade MAS. CD10 and Ki-67 staining pattern in stromal cells may be helpful for the differential diagnosis.

Background: Cytology for breast lesions is a safe, rapid and cost-effective with a high specificity and sensitivity., Objective: To determine the cytomorphologic patterns of breast lesions identified among a group of Sudanese patients., Materials and Methods: This study included 759 patients undergoing either a fine needle aspiration FNA, nipple discharge (ND) smears or breast skin scraping (SS) at a cytology clinic in Khartoum. Clinical and demographic data were reviewed. Stained smears were categorized into: inadequate sample, normal breast, benign lesion, suspicious, or malignant neoplasm., Results: of the 759 cases, 734 (96.71%) were FNA, 18 (2.37%) ND and 7 cases (0.92%) SS. For 28 cases, FNA was done under ultrasound guidance. Females were 720 (94.86%). Benign lesions were 423(55.75%) and 248 (32.67%) were malignant and 77 (10.14%) of smears were normal without any detected abnormality. Ten (1.31%) cases were suspicious for malignancy, and only one case (0.13%) was reported as inadequate. Most lesions were observed among the age group 30 years and above., Conclusion: Most patients investigated have benign lesions, one third of cytological smears were malignant. FNAC is a useful tool for investigating breast lesions in limited-resource settings.

We report a case of pulmonary adenofibroma in a 29-year-old female found by CT scan during work-up for midline chest pain. To our knowledge, the cytological features of this entity have not been previously reported. Cytology demonstrated bland epithelial and stromal cells of varying size without karyorrhexis, pyknosis, or necrosis and with very rare mitoses. Stromal cells were present as either naked bipolar nuclei, as spindle cells with fragile eosinophilic cytoplasm, or as rare larger carrot shaped nuclei. Epithelial cells were present as small loosely cohesive groups with smooth round nuclei and moderate amounts of cytoplasm. Histologically, this lesion consisted of a leaf-like fibroepithelial pattern in which the clefts were lined by a single layer of cuboidal epithelium reminiscent of adenofibroma occurring in the female genital tract. Immunohistochemical analysis demonstrated epithelium that stained positively for pan-cytokeratin and TTF-1. The stroma stained positively for vimentin and desmin, and was weakly positive for SMA-1. The lesion was confirmed to be pulmonary adenofibroma with a smooth muscle component. The differential diagnosis for this lesion includes, but is not limited to, pulmonary hamartoma, pulmonary blastoma, adenomyofibroma, synovial sarcoma, and visceral metastases. It is important for cytopathologists to be aware of this benign entity because it can be encountered on lung FNA specimens. Considering this benign lesion in the differential diagnosis may help plan for minimal lung resection. Confirmatory intraoperative frozen section is a reasonable option., (Copyright © 2012 Wiley Periodicals, Inc.)

We report a case of pulmonary adenofibroma, a rare benign soft-tissue tumor characterized by a combination of glandular and spindle stromal elements, in a 55-year-old man. This case is interesting because of the unusual X-ray and computed tomography (CT) imaging findings of a well-defined, subpleural soft-tissue nodule in the inferior lobe of the left lung, which showed no enhancement after contrast scan. The tumor was resected completely via video-assisted thoracoscopy. Microscopic and immunohistochemical examinations supported the diagnosis of a benign pulmonary adenofibroma. The patient remains well with no evidence of recurrence 16 months after surgery. Although the imaging findings were nonspecific, adenofibroma may be one of diagnostic inclusions of soft-tissue nodules of the lung in middle-aged patients.