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A 32-year-old woman with Turner's syndrome complained of abnormal genital bleeding, which was diagnosed as a delivered myoma. Vaginal myomectomy was performed, and the tumor was histopathologically diagnosed as an adenofibroma of the uterine corpus. As these are extremely rare, we report this case with a review of some of the literature.
A case of papillary adenofibroma of the endometrium is reported in a 78-year-old parous woman. Diagnosis was made by curettage. The patient was treated by total abdominal hysterectomy with bilateral salpingo-oophorectomy. This is the eighth case known in the literature. Clinical and pathological aspects of this relatively new pathological entity are reviewed.
The expression of beta-endorphin, Met-enkephalin and Leu-enkephalin was studied in 63 malignant and benign human breast tumors using immunohistochemical methods. Among invasive ductal carcinomas, 93% were positive for beta-endorphin, 87% for Leu-enkephalin and 90% for Met-enkephalin, in both the tumor stroma and the cell bodies. Enkephalin was predominant in cells, whereas endorphin was predominant in stroma. Nearly the same distribution was found in adenofibromas. In pericancerous normal tissue, neuropeptides were predominantly expressed in the stroma. Although the neuropeptide expression is not cancer-specific, it could be cancer-related, since the results suggest that the neuropeptide expression could reflect the host response to cancer cells and not only the cancer cell activity. The possibility of a direct action of the nervous system on stroma reaction and then on cancer cells is discussed.
We report on two similar cases of young women with irregular menstrual bleeding. Curettage and biopsy revealed an adenofibroma of the uterus in case 1 (31 yrs) and an uterine adenosarcoma in case 2 (30 yrs). Hysterectomy was subsequently performed in both patients: histological examination of the hysterectomy specimen showed no remnants of the tumour in case 1. There was adenosarcoma tissue in the uterine cavity of case 2 but no myometrial infiltration. The difficulties of differential diagnosis of uterine soft tissue tumours in young women are discussed and the literature is reviewed.
A unique case of bilateral papillary adenofibroma of the fallopian tube is reported. The seven-year disease-free follow-up after simple excision without salpingectomy further confirms the benign nature of this entity.
We report two cases of a primary lung tumour characterized by complex gland-like spaces lined by simple cuboidal to columnar epithelium surrounded by a hyalinized spindle-cell fibroblastic proliferation reminiscent of adenofibromas of the female genital tract. The lesions occurred in a 54-year-old woman and a 56-year-old man. The tumours presented clinically as 1-2 cm, solitary 'coin' lesions and were discovered incidentally on routine chest X-rays. Both lesions were treated by lobectomy. One patient is alive and well with no evidence of disease after 8 years; the other died of myocardial infarction 5 years following resection of his tumour without evidence of recurrence. We interpret these lesions as benign hamartomatous growths; their main importance lies in distinguishing them histologically from other types of pulmonary hamartomas, pulmonary blastomas, intrapulmonary solitary fibrous tumours, and metastases from soft tissue and visceral sarcomas.
This is a case report of malignant endometrioid adenofibroma of the ovary with serous cystadenoma in a 42-year-old female. It was classified as FIGO serous IA (2) ovarian tumors. Serous epithelium transformation into endometrioid glandular epithelium was disclosed. The patient is now in good condition and followed-up periodically.
The light and electron microscopic characteristics of an adenofibroma of the rete testis in a 51-year-old man are described. The tumor was 5.5 cm in greatest diameter and situated in the anterior superior portion of the right testis. It was composed mainly of mesenchymal and secondary epithelial proliferation. Long slit-like spaces and short tubules lined by a layer of epithelial cells were seen in the mesenchymal tissue. The epithelium was histochemically and ultrastructurally similar to that of the rete testis, and the tumor was considered to be of rete testis origin.
A well-differentiated endometrioid adenocarcinoma coexistent with benign and borderline-malignant endometrioid adenofibroma was found in the ovary of a 64-year-old woman. She had vaginal bleeding caused by simple hyperplasia of the endometrium due to high levels of sex steroid hormones. A FIGO stage Ia solid ovarian tumor was identified. It was composed of irregularly shaped endometriotic glands with benign and borderline malignant cytologic features embedded in abundant fibromatous stroma. Well-differentiated malignant epithelium was adjacent to these areas, but fibromatous stroma was not predominant. She was treated by surgery and three cycles of chemotherapy. This paper describes this unusual tumor and reviews the literature.
Hepatic biliary cystadenoma is a well-delineated neoplasm in some domestic animals, especially in cats, but it has not been reported in equines. We report on a case of hepatic biliary tumor, incidentally found in a 10-year-old horse, with gross and microscopic features similar to those observed in biliary adenofibroma of humans. The tumor presented as a solid mass measuring 16 cm in diameter and histologically was composed of complex tubulocystic biliary components embedded in an abundant fibrotic stroma. We regarded this tumor as a morphological variant of biliary cystadenoma of domestic animals. Differential diagnoses from other hepatic biliary tumor-like and tumor lesions are provided.
We report a case of tubulocystic ovarian clear cell carcinoma (OCCC) with abundant fibrous stroma associated with an endometriotic cyst. Most OCCC show a small amount of fibrous stroma; however, the tumor presented in this case had abundant stroma, that qualifies it as a malignant clear cell adenofibroma. This unusual type of clear cell carcinoma may be misinterpreted as a benign lesion or as metastatic carcinoma on frozen section. In permanent sections, the stromal invasive foci are focal, small and subtle. Therefore, extensive sampling of the specimen to search for evidence of invasion is recommended for a fibrous ovarian tumor that appears benign on gross examination.
We describe a peculiar pulmonary lesion, that we interpreted as a pseudopapillary variant of solitary fibrous tumor. The patient was a 62-year-old asymptomatic male, non smoking, presenting with a peripheral nodule, 0.8 cm across, located in the lower lobe of the right lung. The patient is alive and well 18 months after surgical excision of the nodule. Microscopically, the lesion was well-circumscribed and characterized by a diffuse pseudopapillary pattern. Pseudopapillae were large, and were covered by a rim of cubic epithelium devoid of atypia. The stromal axis was fibrous and contained scattered bland spindle cells. Immunohistochemically, the latter were strongly positive for vimentin and CD34, focally positive for BCL2 and CD99, negative for cytokeratin, EMA, TTF1, calretinin, smooth muscle actin, desmin and S100 protein; the epithelial cells were immunoreactive for cytokeratin, EMA and TTF1. We interpret this lesion as a peculiar pseudopapillary variant of solitary fibrous tumor, corresponding to what has been reported in the literature as pulmonary adenofibroma and fibroadenoma. The most important differential diagnostic considerations are briefly discussed.
Nephrogenic adenofibroma is a novel kidney tumor of young people (mean age of presentation, 13 years), who present with polycythemia, hypertension, or hematuria, which resolve following nephrectomy. The typical nephrectomy specimen contains a solitary, nonencapsulated, vaguely circumscribed, irregularly shaped or spherical, firm mass with either tan, gray-white, or pale yellow coloration. Cysts are sometimes present within the tumor. The histologic appearance is distinctive and characterized by a marked proliferation of spindled mesenchymal cells resembling the classical type of congenital mesoblastic nephroma, encasing discrete nodules of embryonal epithelium similar to the hyperplastic nephrogenic rests (nephroblastomatosis) usually associated with Wilms' tumor. The mesenchymal component consists of a fascicular proliferation of tightly interlaced, uniform, benign-appearing spindled cells that immunostatin for vimentin and fibronectin, but not desmin or actin. The epithelial component consists of discrete islands of blastemal cells that are partially or fully differentiated toward tubular, tubulopapillary, or papillary structures. Psammoma bodies are plentiful. Embryonal epithelium immunostains for cytokeratin but not epithelial membrane antigen. The overall histologic appearance of the mesenchymal and epithelial components is benign, and preliminary clinical data suggest that the tumor has a benevolent course. Two cases, however, contained small, well-circumscribed papillary lesions near the renal pelvis that resembled low-grade collecting duct carcinoma. The clinical implications of the latter finding are unclear.
Juvenile adenofibromas are large, benign breast tumors common to adolescents and young women that often present as a disturbing breast asymmetry, as in the case described here.
A case of ovarian clear cell adenofibroma of borderline malignancy in a 59-year-old woman is presented with histochemical and electron microscopic observation. The tumor was histologically composed of tubular component lined in part with flat or cuboidal clear as well as hobnail cells with mild cytologic atypia and abundant stromal component containing foci of calcification. The epithelial cells had diastase-digestive PAS-positive material in the cytoplasm, and occasionally the luminal surface and intraluminal substance were stained with mucicarmine. Some of the epithelial cells showed positive lipid staining with Sudan III stain. On ultrastructural study, the tumor featured a moderate number of small mitochondria, poorly developed rough endoplasmic reticulum (rER), short microvilli, intercellular tight junction and desmosome and small amount of glycogen granules. The patient is alive and well with no evidence of recurrence six and half years after the operation.
Significant increases in the serum levels of cancer antigen 125 (CA125) and carbohydrate antigen 19-9 (CA19-9) were observed over one month prior to the removal of an ovarian adenofibroma. The serum levels of CA125 and CA19-9 decreased rapidly after surgery. The surface of the tumor at surgery showed marked inflammation, probably induced by the necrosis produced by torsion. Pathologically, most of the tumor was necrotic, and histoimmunochemical staining of the viable cells was weak for CA125 but intense for CA19-9. Clinicopathological observations of the case suggested that CA125 and CA19-9 might be stimulated in the cells by inflammation or that originally existing CA125 and CA19-9 were released from the tumor cells following the cell necrosis.
Endometrioid adenofibromas are rare tumours of the ovary characterised by proliferation of endometrial type glands without any particular atypical cell structure, within a dense and thick stroma. We report a case of such a tumour which showed an outline of stromal luteinisation.
Benign biliary tumors of the liver are uncommon. In this report, we describe a distinctive biliary tumor of 7-cm diameter occurring in the right lobe of the liver of a 74-year-old Chinese woman. The lesion, characterized by a complex tubulocystic nonmucin secreting biliary epithelial and an abundant fibroblastic stromal components, is distinct from other well-recognized biliary lesions. A number of unusual features are focally present, namely, intraluminal bile concretions, apocrine-like epithelial change, acute inflammation, and granuloma. The tumor shows a striking resemblance to Meyenburg's complex (MC), but the large size of the lesion and the absence of any typical MC in the background liver are exceptional for the latter. Its expansile growth, possession of mitoses, and foci of epithelial tufting and cellular atypia favor a neoplastic process. Previous reported cases of adenomatous neoplastic transformation of MC are dissimilar. We therefore conclude that this is a hitherto unrecognized biliary tumor that may be yet another neoplastic form of MC and propose the designation biliary adenofibroma. The course appears benign, but malignant epithelial transformation may supervene if the lesion is left untreated.