Histological and immunohistochemical characteristics and p16 status studied by FISH in six incidentally detected cases of well-differentiated papillary mesothelioma of the peritoneum.

Well-differentiated papillary mesothelioma (WDPM) is an uncommon mesothelial neoplasm, which is generally regarded as benign or indolent in terms of its clinical behavior. However, details about WDPM have remained relatively unknown. Therefore, in this study, we examined six incidentally detected cases of WDPM of the peritoneum. All six cases were surgically excised, without any additional therapeutic measures. None of the cases showed recurrence. All six cases presented single lesions and the tumor sizes ranged from 2 to 10 mm. Histologically, all six cases exhibited papillary proliferation of cytologically bland mesothelial cells with a fibroconnective tissue core. One of the cases (Case 6) presented small invasive foci in the stalk. The tumor cells were immunohistochemically positive for mesothelial markers and negative for GLUT-1, p53, and CD146. The Ki-67 labeling index of the tumor cells was lower than 5% at the hot spots. All samples were BAP1-positive. None of the samples presented p16 homozygous deletion, as assessed by fluorescence in situ hybridization (FISH). None of the patients deceased due to WDPM. However, in Case 3, death occurred due to pancreatic cancer. The results of this study indicate the importance of analyzing immunohistochemical markers and p16 status to diagnose WDPM accurately.

Nevus lipomatosus cutaneous superficialis on the shoulder: A case report with MRI findings.

Nevus lipomatosus cutaneous superficialis is a rare disease, and its magnetic resonance imaging features have not been reported. A 1-year-old male infant was admitted to our hospital for examination of a mass on his right shoulder. Magnetic resonance imaging revealed hypertrophic skin and a lipomatous subcutaneous mass, consistent with a hamartoma-like lesion or mesenchymal tumor; after surgery, the tumor was pathologically diagnosed as nevus lipomatosus cutaneous superficialis. To the best of our knowledge, this is the first case report focusing on the magnetic resonance imaging features of this disease. Hypertrophy of all skin structures involved (epidermis, dermis, and subcutaneous fat) may be specific to nevus lipomatosus cutaneous superficialis, enabling its differentiation from other tumors.

Gastric cancer metastatic to neck lipoma: a case report with imaging consideration.

A 71-year-old man visited our hospital for examination of a soft neck mass. Computed tomography and magnetic resonance imaging scans showed a well-circumscribed large lipomatous tumor with multiple nodules inside. Atypical lipomatous tumor or lipoma involving the lymph nodes was considered. Pathological examination of the surgical specimen suggested typical lipoma including multiple metastatic foci from gastric cancer. Subsequent endoscopy revealed a gastric tumor, which was histologically proven to be signet-ring cell carcinoma. Considering these findings, this case was diagnosed as tumor-to-tumor metastasis to lipoma. We present the first such case with imaging and clinical characteristics.

[A case of non-invasive IPMC with fibrotic lesion-like "tubular complex"].

A 73-year-old man was incidentally diagnosed with a cystic lesion in the pancreatic body. Ultrasonography, abdominal computed tomography, magnetic resonance imaging, endoscopic retrograde cholangiopancreatography, and endoscopic ultrasound were performed, and a nodule was detected in the cystic lesion along with an irregularity of the main pancreatic duct. An initial diagnosis of a mixed-type intraductal papillary mucinous neoplasm was made, and a central pancreatectomy was performed. However, the final diagnosis was altered to non-invasive intraductal papillary mucinous carcinoma (IPMC). The histopathological examination revealed a fibrotic lesion that was similar to "tubular complex" findings observed in mouse models of pancreatic cancer. The fibrotic lesion was placed between the main pancreatic duct lesion and branch-duct cystic lesion. The changes reflected in branch-level stenosis may be caused by IPMC growth.

Cytokeratin 7-positive/cytokeratin 20-negative cecal adenocarcinoma metastatic to the uterine cervix: a case report.

BACKGROUND: The vast majority of uterine cervical malignancies are primary carcinomas, and secondary neoplasms that metastasize to the uterine cervix from a distant organ are uncommon. Although relatively rare, metastases to the uterine cervix from a primary colon cancer have been reported. We report a rare case of metastatic carcinoma originating from a cecal adenocarcinoma with an unusual cytokeratin 7/cytokeratin 20 immunophenotype. CASE PRESENTATION: A 74-year-old postmenopausal Japanese woman was referred to our hospital for the evaluation of a uterine tumor. She had a past medical history of cecal cancer and had undergone laparoscopically assisted right hemicolectomy at the age of 69 years. During follow-up, she was found to have elevated levels of the tumor markers carbohydrate antigen 19-9 (179.7 IU/mL) and carcinoembryonic antigen (26.9 µg/L). Positron emission tomography/computed tomography showed a focus of high 18F-fluorodeoxyglucose uptake in her uterus. Examination of a cervical biopsy found a poorly differentiated adenocarcinoma that was immunopositive for cytokeratin (CK)7 and caudal-related homeobox 2 (CDX2) expression and immunonegative for cytokeratin 20 expression. The patient underwent radical hysterectomy and bilateral salpingo-oophorectomy. Histopathological examination found invasive growth of irregular and atypical ductal hyperplasia. Immunohistochemical staining of the tumor specimen revealed the same immunophenotype as the biopsy specimen. The cecal cancer specimen from her previous surgery was also examined and found to have the same immunophenotype. The histopathological diagnosis was cecal adenocarcinoma metastatic to the uterine cervix. The patient is currently receiving adjuvant chemotherapy and to date is without evidence of recurrent disease. CONCLUSIONS: Our report illustrates the importance of immunohistochemistry for the correct diagnosis of the origin of a uterine cervical adenocarcinoma in a patient with a medical history of colorectal cancer. Re-examination of a previous oncological specimen is critical for cases with a uterine lesion that is difficult to identify as primary or metastatic cancer.

Solitary uterine metastasis of invasive lobular carcinoma after adjuvant endocrine therapy: a case report.

INTRODUCTION: Solitary uterine metastases from extragenital cancers are very rare. Breast cancer is the most frequent primary site of metastasis to the uterine corpus, with invasive lobular carcinoma more likely to spread to gynecologic organs than invasive ductal carcinoma. CASE PRESENTATION: A 62-year-old postmenopausal Japanese woman was diagnosed with uterine leiomyomata more than 20 years ago and had been managed conservatively until menopause. Seven years prior to her presentation, she was diagnosed with breast cancer and underwent a partial resection of her right breast for stage IIA invasive lobular carcinoma. She underwent adjuvant chemotherapy, radiotherapy, and five years of anastrozole hormonal therapy. She presented with a growing uterine mass. Her tumor marker levels were markedly increased over the course of her follow-up, but a systemic examination revealed only a solitary uterine tumor. She underwent a total abdominal hysterectomy with bilateral salpingo-oophorectomy. A histopathological examination, including detailed immunohistochemistry, confirmed metastatic invasive lobular carcinoma, infiltrating both her uterine myometrium and fibroid tissue. CONCLUSION: We report a very rare metastatic pattern of invasive lobular carcinoma and demonstrate that gross cystic disease fluid protein-15 and mammaglobin are useful in the diagnosis of metastatic breast cancer.

Immunoglobulin G4-related disease with several inflammatory foci.

We herein report the case of a 62-year-old Japanese man who presented with jaundice, dry eyes and abdominal discomfort. Imaging studies revealed swelling of the periorbital tissue, parotid and submandibular glands, pulmonary hilar lymph nodes, pancreas, bile ducts, gall bladder walls, bilateral kidneys, arterial walls and prostate. A significant increase in the serum level IgG4 was seen, and the patient was diagnosed with IgG4-related disease after undergoing a biopsy of the pancreas and prostate. We herein report a case of IgG4-related disease with multiple ten organ involvement at the onset of the disease that was successfully treated with prednisolone (PSL) therapy.

Efficient palliative involved-field radiotherapy on highly progressive diffuse large B-cell primary gastric lymphoma with liver cirrhosis.

We report the case of a 73-year-old woman having diffuse large B-cell primary gastric lymphoma with a cirrhotic liver caused by hepatitis C virus infection. She visited our hospital with symptoms of nausea and vomiting, which appeared to be caused by stenosis due to the tumor. Metastatic tumors were seen in the gastric and jugular lymph nodes. The clinical stage was IVB with a high risk of poor prognosis according to the international index. Because of poor hepatic reserve function, standard chemotherapy could not be administered. To maintain her quality of life, palliative involved-field radiotherapy was performed. The symptoms and tumor markers significantly improved, and computed tomography and endoscopy indicated the disappearance of the primary gastric tumor. Two months after radiotherapy and her return home, she died of pneumonia. Autopsy showed neither lymphoma cells nor stenosis of gastric lesion. The significant anti-tumor effect on primary tumor in our case suggests that the involved-field radiotherapy, although palliative, can be a therapeutic option for primary gastric lymphoma patients with various complications.

A patient with severe hypercalcemia in multiple organ dysfunction syndrome: role of elevated circulating 1alpha,25(OH)2 vitamin D levels.

A 54-year-old man was transferred to our ICU because of systemic inflammatory response syndrome (SIRS) and multiple organ dysfunction syndrome (MODS). He died after 38 days of intensive care. During treatment, his serum calcium (Ca) levels continued to increase and reached 3.95 mmol/L, while the ionized Ca levels reached 2.30 mmol/L before his death. He presented with severe kidney injury, pancreatitis, and hemorrhagic gastric erosion that worsened his prognosis; these were possibly associated with the hypercalcemia. His circulating 1alpha,25-dihydroxyvitamin D [1,25(OH)(2)D] level was elevated (75.7 to 204 pg/mL), whereas the levels of 25-hydroxyvitamin D, parathyroid hormone, and parathyroid hormone-related peptide were not. Liver histology revealed immunoreactivity for 25-hydroxyvitamin D 1alpha-hydroxylase (CYP27B1) in some of the hepatocytes, in which the localization pattern was similar to that of lysozyme-positive hepatocytes. Our ICU has previously encountered 22 similar MODS patients who presented with hypercalcemia over the last 8 years. SIRS with severe kidney and liver injuries are common clinical findings in hypercalcemic patients with MODS. Of the 23 hypercalcemic MODS patients, including the present patient, 17 had circulating 1,25(OH)(2)D levels exceeding 70 pg/mL despite severe kidney injury. Extrarenal activation of CYP27B1 seems to play a role in the development of hypercalcemia in this disease condition. Clinicians need to be aware that severe hypercalcemia may occur in MODS patients.

Extranodal Rosai-Dorfman disease involving bilateral ovaries in a patient with a ventriculoperitoneal shunt.

Rosai-Dorfman disease (RDD) is a rare condition of unknown etiology, and female genital tract involvement in RDD is uncommon. We describe the first case of RDD with bilateral ovarian involvement in a patient implanted with a ventriculoperitoneal (VP) shunt. The patient was a 17-year-old Japanese woman who had undergone radiotherapy, surgery for extranodal RDD involving the brain, and VP shunt insertion at age 12. Bilateral pelvic masses were incidentally detected on a computed tomography scan. She underwent laparotomy for lesion extirpation. On abdominal washing cytology, histiocytes showing emperipolesis were identified. Bilateral salpingo-oophorectomy was performed instead of extirpation, as it was difficult to identify the lesion margins. At 24 months after surgery, the patient is well and has not developed local recurrence. Thus, RDD can recur because of implantation of lesion cells into the abdominal cavity through a VP shunt, as is observed in the case of cerebral neoplasms.

Macrophage colony-stimulating factor is indispensable for repopulation and differentiation of Kupffer cells but not for splenic red pulp macrophages in osteopetrotic (op/op) mice after macrophage depletion.

We previously reported that macrophage colony-stimulating factor (M-CSF, CSF-1) played important roles in the process of the repopulation of Kupffer cells after their elimination by administration of liposome-entrapped dichloromethylene diphosphonate (lipo-MDP). In this study, we examined the repopulation of Kupffer cells and splenic red pulp macrophages in osteopetrotic (op/op) mice defective in the production of functional M-CSF and their littermate mice by using the lipo-MDP model. In untreated op/op mice, numbers of F4/80-positive Kupffer cells in the liver and F4/80-positive splenic red pulp macrophages were reduced. Repopulation of Kupffer cells and splenic macrophages was observed in littermate (op/+) mice liver by 14 days after depletion. However, in op/op mice, repopulation of Kupffer cells was not observed in Kupffer-cell-depleted op/op mice until 56 days after depletion, whereas splenic red pulp macrophages repopulated and recovered to the level of control op/op mice by 10 days after depletion. Single injection of M-CSF was effective for the induction of the repopulation of Kupffer cells, and daily administration of M-CSF induced remarkable repopulation and maturation of Kupffer cells and proliferation of macrophage precursor cells in the liver of Kupffer-cell-depleted op/op mice. These results suggest that Kupffer cells are completely M-CSF-dependent tissue macrophages, whereas splenic red pulp macrophages are composed of M-CSF-dependent macrophages and M-CSF-independent macrophages. This mouse model provides a useful tool for the study of effects of growth factor on Kupffer cell differentiation in vivo.