Radiation therapy for primary breast lymphoma in male gynecomastia: a rare case report and review of the literature.

Primary breast lymphoma is a rare type of non-Hodgkin lymphoma and usually affects women, although a few cases have been reported in men. Chemotherapy and radiation therapy, or a combination of both, are frequently administered for treatment of primary breast lymphoma, as local control by surgical resection is poor. No standard therapy has been established, and the optimal radiation dose and irradiation field for male patients are unknown. The present report describes a 75-year-old man with bilateral cirrhosis-induced gynecomastia who was diagnosed with primary breast lymphoma; specifically, diffuse large B-cell lymphoma. Because of his hepatic dysfunction, he was treated with radiation therapy alone. Radiation therapy was followed by eight cycles of rituximab monotherapy. Clinical response was good, with no signs of relapse. Clinicians may benefit from knowledge regarding effective treatment of primary breast lymphoma in male patients, which has been rarely reported owing to the low incidence of this condition. The outcome in the present case may help to establish effective treatment guidelines in similar cases.

Prognostic significance of programmed cell death-1-positive cells in follicular lymphoma patients may alter in the rituximab era.

Programmed cell death-1 (PD-1) is involved in one of the inhibitory pathways of the B7-cluster of differentiation (CD) 28 family; this pathway is known to be involved in the attenuation of T-cell responses and promotion of T-cell tolerance. PD-1 is known to negatively regulate T-cell receptor-mediated proliferation and cytokine production, lead to alternation in the tumor microenvironment. Although several studies have shown that high levels of PD-1-positive cells in follicular lymphoma (FL) patients influence their prognosis, those studies included patients treated without rituximab, and the prognostic impact of PD-1 positivity in the rituximab era (R-era) has not yet been elucidated. We retrospectively studied 82 patients with FL uniformly treated with standard R-CHOP therapy at six institutions between 2001 and 2009 (median follow-up for survivors: 55 months). We also collected and examined biopsy specimens for diagnosis with respect to PD-1 positivity. The PD-1 positivity was significantly higher in male patients and patients with high beta-2 microglobulin (B2M ≥ 3.0) (P = 0.03 and 0.003, respectively). Three-year progression free survival (PFS) and overall survival (OS) were 60% and 86%, respectively. By univariate analysis, elevated LDH (P = 0.07) worsened PFS. Male gender (P = 0.03), high FLIPI score (P = 0.05), and high B2M levels (P = 0.08) worsened OS. Multivariate analysis detected no significant prognostic factors, including PD-1 positivity. However, in male subgroup, high levels of PD-1-positive cells were found to be a prognostic factor for PFS. Addition of rituximab might have altered the prognostic impact of PD-1-positive cells.

Cytologic findings of epithelioid trophoblastic tumor of the uterus: a case report.

BACKGROUND: Epithelioid trophoblastic tumor (ETT) is a rare entity within trophoblastic tumors. It is difficult to recognize ETT because of its epithelioid appearance. CASE: A 35-year-old female, 5 years after pregnancy, experienced genital bleeding 2 months prior to consulting us. Preoperative laboratory data showed a slightly elevated serum level of human chorionic gonadotropin (hCG). A cytologic cervical smear revealed large, polygonal, atypical cells. These cells had mononucleate, ovoid, irregularly enlarged and hyperchromatic nuclei with 1 or 2 conspicuous nucleoli. The cytoplasm was thin and abundant, with a distinct cell membrane, and sometimes showed vacuolation. The patient was diagnosed with uterine cancer, and hysterectomy was performed. The tumor was present in the uterine corpus, measuring 3 x 2.5 x 2.5 cm. Histologically, it was composed of mainly mononuclear tumor cell nests resembling intermediate trophoblastic cells with zones of hyaline material. Immunohistochemically, the tumor was positive for cytokeratin and placental alkaline phosphatase but negative for hCG and human placental lactogen. The tumor was subsequently diagnosed as ETT. CONCLUSION: It was difficult to make a definitive diagnosis of ETT using only a cytologic specimen. The diagnosis of ETT is facilitated by a combination of cytologic, histopathologic and clinical findings.

[Histopathological and cytological findings in a case of gastrointestinal stromal tumor presenting in the pelvic cavity].

Gastrointestinal stromal tumor (GIST) usually occurs in the gastrointestinal tract, mainly in the stomach. Recently, GIST arising in extragastrointestinal organs was been reported. This case was a 49-year-old female who was diagnosed as having uterine leiomyoma and pelvic tumor in the rectovaginal septum. The pelvic tumor showed no connection with the uterus and rectum. Grossly, the pelvic tumor was light brown and solid. Cytologically, the tumor cells of the pelvic tumor showed vague interlacing fascicles with spindle/fibrous cytoplasm and elongated nuclei. No necrosis or mitosis was present. Histological findings of the pelvic tumor revealed an interlacing bundle fashioned with one to two mitotic figures per 50 HPE No necrosis was found in the tumor. The tumor cells were positive for CD117 (c-kit protein), CD34, and vimentin by immunohistochemistry, but negative for actin, desmin, and S-100. The tumor was diagnosed as GIST in the pelvic cavity. GIST rarely presents in extragastrointestinal regions such as this case. It is difficult to make a differential diagnosis between GIST and other mesenchymal spindle cell tumors only from a cytologic specimen. A positive reaction for CD117 is useful for the diagnosis of GIST.

Histopathological and immunohistochemical findings in a case of glycogen-rich clear cell carcinoma of the breast.

Glycogen-rich clear cell carcinoma (GRCC) of the breast is a rare variant of primary breast carcinoma that was first described by Hull et al. in 1981, and is characterized by carcinoma cells containing an optically clear cytoplasm and intracytoplasmic glycogen. The present case involved a 33-year-old female. She had noticed a lump in the inner quadrant of the left breast. The tumor obtained by enucleation biopsy had an irregular shape. The tumor cells exhibited sharply defined borders, polygonal contours, a clear or finely granular cytoplasm, and moderate nuclear atypia. The tumor cells showed a positive reaction with periodic acid Schiff, eliminated by diastase digestion. The tumor was diagnosed as GRCC. There was no nodal metastasis. Immunohistochemically, the tumor cells were positive for cytokeratin, epithelial membrane antigen, HER2, and p53, but negative for estrogen receptor (ER) and progesterone receptor (PR). Although the biological behavior of GRCC is difficult to predict in view of the very limited number of case reports, the prognosis of GRCC may be associated with not only histopathological subtype but also other clinicopathological factors, such as size, status of invasion, status of nodal metastasis, nuclear grade, ER, PR, HER-2, p53 and so on. To clarify the pathogenesis of mammary GRCC, the systematic study of additional well-documented cases with long-term follow up will be necessary.

Pathological and immunohistochemical findings of an autopsy case of adult moyamoya disease.

Moyamoya disease is vaso-occlusive disease involving the arteries of the circle of Willis that is accompanied by a compensatory recruitment of a vascular network. The pathological and immunohistochemical findings of an autopsy case of hemorrhagic moyamoya disease in a 69-year-old woman are described in the present report. The autopsy findings of the brain revealed cerebral and intraventricular hemorrhage with edema. The left anterior cerebral artery, bilateral middle cerebral arteries and left posterior cerebral artery were marked narrowing, and the other arteries revealed mild narrowing. Microscopically, the arteries of the circle of Willis showed narrowed lumen, fibrocellular intimal thickening, marked tortuousness of internal elastic lamina and attenuation of media. The thickened intima was composed of smooth muscle cells. The vessels with dilated or irregular-shaped lumen suggested abnormal vascular networks demonstrated by angiography. In this case, no correlation between the abnormal vascular network and expression of VEGF or VEGF receptor was disclosed. It was hypothesized that abnormal vascular networks might be composed of collateral vessels in relation to various pathological changes of the arteries, such as occlusion and stenosis, and intracranial hemorrhage in patients with moyamoya disease might occur as a result of rupture of arteries including abnormal vascular networks.

[Two cases of parasitic granuloma found incidentally in surgical specimens].

Occasionally, parasitosis demonstrates no clinical symptoms, and is found incidentally. We report 2 cases of parasitic granuloma found incidentally in surgical specimens in rare sites for parasitosis. Case 1 was a 40-year-old female. She was diagnosed with inguinal hernia, and operation was performed. A white nodule, measured 1.5 x 1 x 1 cm in size, was found in the hernia sac. The nodule was elastic hard and solid. Histologically, eosinophilic granuloma was demonstrated, and the parasite was characterized by renette cell and Y-shaped lateral cord, and suggesting extra-gastrointestinal anisakiasis. Case 2 was a 71-year-old female. She was diagnosed with colonic adenocarcinoma, and colectomy was performed. A white nodule, measured 1 x 1 x 0.8 cm in size was found in the omentum, at first identified as peritoneal dissemination of colonic carcinoma. However, histological findings of the nodule showed eosinophilic granuloma, and parasite was observed in the granuloma. The parasite was characterized by thick cuticle and muscular cells, and suggestive of dilofilariasis. In each case, no clinical symptoms of parasitosis were recognized, and each lesion presented in a rare site as anisakiasis or dirofilariasis. When any lesions as in the present cases is found in a surgical specimen, it is important to make differential diagnosis with consideration for parasitic granuloma.

Radiologic features of uterine lipoleiomyoma.

Rare cases of uterine lipoleiomyoma are reported with the presentation of plain film, computed tomography, and magnetic resonance imaging.