Kimiya Sato

Malignant mixed Mullerian tumor of the uterine corpus with alpha-fetoprotein-producing hepatoid adenocarcinoma component

OBJECTIVES Hepatoid adenocarcinoma is a rare tumor and has the histological coexistence of well-differentiated adenocarcinoma and nests of hepatoid cells with immunoreactivity for alpha-fetoprotein (AFP). A case of hepatoid adenocarcinoma in malignant mixed Mullerian tumor of the uterus is presented with a review of the literature. CASE The patient is a 63-year-old woman who complained of abnormal genital bleeding. Serum AFP concentration was 5060 ng/ml. Histologically, the tumor was composed of endometrioid adenocarcinoma, neoplastic hepatoid cells, and sarcoma component including leiomyosarcoma and rhabdomyosarcoma. After operation followed by six courses of platinum-based chemotherapy, serum levels of AFP dropped into normal range. CONCLUSIONS This is, to our knowledge, the first report of malignant mixed Mullerian tumor of the uterus with an AFP-producing hepatoid adenocarcinoma component.

Nodular hidradenoma of the breast : Report of two cases with literature review

Two cases of nodular hidradenoma of the breast with possibly different origins are reported. Case 1 is of a 58 year‐old female wlth a breast mass in the left, outer lower‐quadrant. A histogenetical origin in the skin adnexal glands was suspected due to its superficial location and immuno‐histochemical findlngs. Case 2 is of a 44‐yearold male with a subareolar nodule and nipple discharge. Histological examlnation demonstrated that the tumor was located deep in the breast tissue, was surrounded by dilated mammary ducts and exhibited intraductal extenslons, which are all features mlmicking those of breast cancer. Immunohlsto‐chemical positivity against gross cystic disease fluid protein‐15 was weakly Identified and negativity for endo‐plasrnic reticulum was observed. This case can be interpreted as arising In the mammary ducts. It is well known that various kinds of skin adnexal tumors arise in the breast tissue; however, nodular hidradenoma of the breast is still a rare benign neoplasm. Cllnically, nodular hidradenoma of the breast tends to occur in the nipple or subareolar region of the female breast. It should be kept in mind that nodular hidradenoma may occur in mammary ducts and it should be included when differential diagnoses are made of subare olar breast tumors.

Peritoneal seeding of hepatocellular carcinoma: clinicopathological characteristics of 17 autopsy cases.

Peritoneal seeding (PS) of hepatocellular carcinoma (HCC) is uncommon, and the clinicopathological features are poorly understood. A total of 181 autopsy cases of HCC, including 171 cases with detailed clinical information, was investigated for PS and evaluated. PS was identified in 17 cases (9.4%), and was locally (70.6%) or entirely (29.4%) distributed in the peritoneal cavity, involving the diaphragm (76.5%), omentum (47.1%), or alimentary tract serosa (47.1%). Compared with primary HCC, PS showed similar or slightly undifferentiated features (88.2%) and exhibited more differentiated features (11.8%). In 15 cases (88.2%) of HCC with PS, primary HCC showed membranous β‐catenin immunoreactivity. However, in five cases (33.3%), respective PS lost this immunoreactivity. PS was significantly associated with rupture of HCC (P= 0.012), direct diaphragmatic invasion (P= 0.001), and lymph node metastasis (P < 0.001), indicating these are high risk factors for PS; there was no significant association with a past history of percutaneous fine‐needle biopsy, percutaneous ethanol injection and/or radiofrequency ablation (P= 0.97), or metastasis to lung (P= 0.13), bone (P= 0.71), or adrenal gland (P= 0.79). PS can infrequently proliferate aggressively with more differentiated features. Loss of membranous β‐catenin expression may be associated with PS of HCC.

Aberrant cytokeratin 7 expression of centrilobular hepatocytes: a clinicopathological study

Matsukuma S, Takeo H, Kono T, Nagata Y & Sato K 
(2012) Histopathology 61, 857–862

Primary pulmonary myxoid sarcoma with EWSR1‐CREB1 fusion, resembling extraskeletal myxoid chondrosarcoma: Case report with a review of Literature

Reported herein is an extremely rare case of primary pulmonary myxoid sarcoma (PPMS). A 31‐year‐old man presented with a 2.7 cm‐sized pulmonary tumor surrounded by capsule‐like fibrosis. The patient has been free of disease for 5.8 years after surgery. This tumor focally showed endobronchial features, and consisted of reticular cords of oval, short spindle, or polygonal cells with swollen vesicular nuclei accompanied by an abundant myxoid stroma, closely resembling extraskeletal myxoid chondrosarcoma. Tumor cells were diffusely positive for vimentin and focally positive for epithelial membrane antigen, but were negative for cytokeratin, TTF‐1, Napsin A, S‐100 protein, CD34, desmin, smooth‐muscle actin, CD10, p63, calponin, h‐caldesmon, c‐kit, HMB‐45, synaptophysin, or glial fibrillary acid protein. Our reverse transcription‐polymerase chain reaction using the formalin‐fixed, paraffin‐embedded tumor tissues detected EWSR1‐CREB1 fusion transcript, but could not demonstrate EWSR1‐ATF1 fusion or EWSR1/TAF15/TFG‐NR4A3 fusion. These findings indicate that the current tumor is an additional case of PPMS with EESR1‐CREB1 fusion, recently reported by Thway et al. Some cases of PPMS can behave in an indolent manner.

Numerous eosinophilic globules (skeinoid fibers) in a duodenal stromal tumor : An exceptional case showing smooth muscle differentiation

A unique case of duodenal stromal tumor In a 51‐year‐old man is reported. The tumor histologically showed spindle cell proliferation and numerous eosinophilic globules. Most globules were composed of tangled 45 nm thick fibrils, which were ultrastructurally Identical to ‘skelnoid fibers’. The presence of glycogen granules in the tumor cells and the Immunoreactivity for α‐smooth muscle actin suggested smooth muscle differentiation. Focal ultrastructural findings also supported the smooth muscle nature of this tumor. There were no immunohistochemical and ultra‐structural features indicating neural differentiation. In previous studies, the presence of such ‘skeinoid fibers’ was suggested to be a histological marker for neural differentiation in gastrointestinal stromal tumor. However, the findings In the present case suggest that numerous ‘skeinoid fibers’ can be Identified in duodenal stromal tumor with smooth muscle differentiation, although this condition may be rare.

Chondrosarcomatous differentiation in metastatic deposit of serous papillary cystadenocarcinoma.

A rare case of serous papillary cystadenocarcinoma of the ovary showing chondrosarcomatous differentiation in a metastatic deposit late in the clinical course is reported. A 49‐year‐old female underwent a total abdominal hysterectomy with bilateral salpingo‐oophorectomy for bilateral ovarian tumors. Histological diagnosis was serous papillary cystadenocarcinoma of both ovaries with lymph node metastasis. After six courses of chemotherapy, she was confirmed to be in complete remission following a second laparotomy. Following additional chemotherapy, a third laparotomy disclosed swollen left inguinal lymph nodes. In one of these nodes, approximately 5.0 cm in greatest diameter, the predominant histological features were: chondrosarcoma of the bone and soft tissue, with small foci of serous papillary adenocarcinoma and squamous epithelium. A histological transition between mesenchymal and epithelial areas was identified. Immunohistochemical positivity for broad‐spectrum cytokeratin (AE1/AE3), vimentin, epithelial membrane antigen, and S‐100 protein was observed in both chondrosarcomatous and epithelial areas. The current evidence may suggest that the chondrosarcomatous differentiation was derived from the metastatic epithelial component.

Carcinosarcoma of the liver with an osteosarcomatous component

Abstract We report a case of hepatocellular carcinoma associated with mesenchymal differentiation, which was identified as a true carcinosarcoma based on thorough pathologic examination. A 48-year-old man presented with a giant mass in the lateral segment of the liver. It was resected by lateral segmentectomy combined with total gastrectomy. Microscopically, the tumor consisted of hepatocellular carcinoma, osteosarcoma, and undifferentiated sarcomatous components. The undifferentiated sarcomatous components were positive for cytokeratin, whereas the osteosarcomatous components were negative for cytokeratin and positive for S100. To our knowledge, this is only the second known case in which mesenchymal differentiation was immunohistochemically demonstrated in liver carcinosarcoma.

Reversible iatrogenic, MTX-associated EBV-driven lymphoproliferation with histopathological features of a lymphomatoid granulomatosis in a patient with rheumatoid arthritis.

Dear Editor, A 71-year-old man with a 6-year history of rheumatoid arthritis (RA) and a 4-year history of Sjögrens syndrome had been treated with 10 mg/week methotrexate (MTX), 200 mg/day bucillamine and non-steroidal anti-inflammatory drugs for 5 years. He showed little RA inflammatory activity when he was referred to our hospital for general malaise in a febrile state. A hard mass, approximately 6 cm in diameter, was palpable in the left axillary lymph node, and systemic lymphadenopathy was evident. Computed tomography (CT) showed systemic lymph node involvement, and nodular lesions were observed in the lungs, liver, spleen and ileum. Hepatosplenomegaly was also observed (Fig. 1a). Fluorodeoxyglucose positron emission tomography (FDG/PET)/CT showed increased uptake of FDG in the bilateral supraclavicular, left axillary, mediastinal, para-aortic, mesenteric, splenic hilar and left inguinal lymph nodes as well as the left and caudate lobes of the liver and ileum (Fig. 1b). Epstein–Barr virus (EBV) nuclear IgG and EBV capsid IgG were positive, whereas EBV capsid IgM was negative, consistent with a prior non-acute EBV infection. MTX was immediately discontinued. A histological examination of the haematoxylin–eosin-stained sections of the biopsy specimen obtained from the left axilla mass revealed irregularly shaped necrotic foci and an angiocentric and angiodestructive polymorphous lymphoid infiltrate (Fig. 2a–c). A small number of large atypical lymphoid cells were CD20, CD79a, CD15, CD30 and CD3 and were accompanied by CD3 T lymphocyte infiltration with CD4 more frequently expressed than CD8. These large atypical cells demonstrated a latency type II pattern of EBV infection, which is characterised by positive S. Kobayashi :Y. Kikuchi :Y. Matsuki :M. Nagumo : S. Oshima : T. Kondo Department of Internal Medicine, Mishuku Hospital, 5-33-12 Kamimeguro, Meguro, Tokyo 153-0051, Japan

Pancreatic neuroma-like lesions after upper abdominal surgery: a clinicopathological postmortem study

To elucidate poorly understood pancreatic neural changes after upper abdominal surgery (UAS), we histologically examined 57 postmortem pancreases after gastrectomy, esophagectomy, and esophageal transection, and also investigated each clinical manifestation. Six pancreases (10.5%) had unique histopathological lesions composed of prominently thickened and/or proliferated neural fascicles. They focally or multifocally involved the pancreatic lobules and/or interlobular septa and mimicked traumatic neuroma. Three of the six lesions were accompanied by moderate to severe chronic pancreatitis/pancreatic fibrosis. All six patients were asymptomatic, although only one exhibited a low-echoic pancreatic mass. There were no statistically significant associations between the pancreatic neural lesions and the patients’ sex, age, the time interval between the surgery and the patients’ death, impaired glucose tolerance, the presence of hyalinized pancreatic islets, or the presence of moderate to severe chronic pancreatitis/pancreatic fibrosis. No similar prominent neural lesions were identified in an additional 57 age- and sex-matched control pancreases. We concluded that these prominent neural lesions were pancreatic neuroma-like lesions following UAS. This study failed to demonstrate the distinct pathogenesis of the neuroma-like lesions and further investigation may be needed. However, this report is the first to delineate that asymptomatic neuroma-like lesions can occur following UAS. Consideration of this possible development would be useful for accurate follow-up of patients who undergo UAS, although its occurrence may be infrequent.