We report the clinical and autopsy findings of a 71-year-old Japanese woman with rheumatoid meningitis. This patient developed subacute meningitis during an inactive stage of rheumatoid arthritis (RA), and despite intensive examinations no causative agents or underlying disease could be identified except for RA. Based on persistent hypocomplementaemia and increased serum levels of immune complexes she was suspected of having vasculitis, and was treated with intravenous methylprednisolone (1000 mg/day for 3 days) followed by oral prednisolone. Soon after beginning treatment with corticosteroid her symptoms improved, in parallel with a decrease in cell counts and interleukin-6 in the cerebrospinal fluid. During tapering of oral prednisolone she died of a subarachnoid haemorrhage which was ascribed to a relapse of the meningitis. Autopsy demonstrated infiltration of mononuclear cells, including plasma cells, in the leptomeninges, mainly around small vessels, leading to a definite diagnosis of rheumatoid meningitis. When RA patients manifest intractable meningitis with a subacute course, this disease is important as a possible diagnosis even if the arthritis is inactive, and intensive treatment, including corticosteroid and immunosuppressants, should be positively considered as a therapeutic option as soon as possible because of the poor prognosis.
Objective. Lupus-prone BXSB mice develop monocytosis characterized by selective accumulation of the Gr-1-monocyte subset. The aim of this study was to explore the possible role of activating IgG Fc receptors (Fc␥R) in the development of monocytosis and to characterize the functional phenotype of the Gr-1-subset that accumulates in lupus-prone mice bearing the NZBtype defective Fcgr2b allele for the inhibitory Fc␥RIIB.Methods. The development of monocytosis was analyzed in BXSB and anti-IgG2a rheumatoid factortransgenic C57BL/6 mice deficient in activating Fc␥R. Moreover, we assessed the expression levels of activating Fc␥R and inhibitory Fc␥RIIB on Gr-1؉ and Gr-1-monocyte subsets in C57BL/6 mice bearing the C57BL/ 6-type or the NZB-type Fcgr2b allele.Results. We observed monocytosis with expansion of the Gr-1-subset in anti-IgG2a-transgenic C57BL/6 mice expressing IgG2a, but not in those lacking IgG2a. Moreover, monocytosis barely developed in BXSB and anti-IgG2a-transgenic C57BL/6 mice deficient in activating Fc␥R. The Gr-1-subset that accumulated in lupus-prone mice displayed a unique hyperactive phenotype. It expressed very low levels of inhibitory Fc␥RIIB, due to the presence of the NZB-type Fcgr2b allele, but high levels of activating Fc␥RIV. This was in contrast to high levels of Fc␥RIIB expression and no Fc␥RIV expression on the Gr-1؉ subset. Conclusion. Our results demonstrated a critical role of activating Fc␥R in the development of monocytosis and in the expansion of a Gr-1-Fc␥RIIB lowFc␥RIV؉ hyperactive monocyte subset in lupus-prone mice. Our findings further highlight the importance of the NZB-type Fcgr2b susceptibility allele in murine lupus, the presence of which induces increased production of hyperactive monocytes as well as dysregulated activation of autoreactive B cells.
Abstract. A case of an incidental endoscopic finding of pneumatosis cystoides intestinalis in the large bowel is presented. Diagnosis was verified by fine needle aspiration cytology. Basic facts about this uncommon entityare reviewed with an emphasis on endoscopic differential diagnosis. CASE REPORT / KAZUISTIKAPneumatosis cystoides intestinalis is a rare gastrointestinal condition, in the majority of cases an incidental finding which represents a differential diagnostic challenge for endoscopist, radiologist, pathologist and physician. Case reportA 51-year-old man underwent colonoscopy elsewhere for diarrhoea and slight haematochezia and he was then referred to our endoscopy unit for supervision because of finding several polyps in the hepatic flexure region. During colonoscopy we found several sessile polyps clustered in streaks, covered with normal mucosa, quite firm on probe palpation (Fig 1 and 2). Biopsy attempts just unroofed normal mucosa away, but polyps disappeared during the puncture and suction with a cytology needle. This typical endoscopic picture led us to suspicion of pneumatosis cystoides intestinalis. Material aspirated from cysts was smeared on cytology glass. Microscopically, two cell populations were visible -epithelial cells of colonic mucosa, arranged in a honeycomb pattern and numerous multinucleated giant cells of foreign body type (Fig 3) and this confirmed our suspicion.The patient was symptom-free at that time, no pathology was found on abdominal plain X-ray. DiscussionPneumatosis cystoides intestinalis is a rare condition characterized by the presence of gas-filled pseudocysts in the wall of gastrointestinal tract. This condition can be combined with the presence of gas in omentum, mesentery, peritoneal cavity, retroperitoneum and portal venous system (2,6,25,28). The first cases of pneumatosis cystoides intestinalis were described by DuVernoi in the 18th century in postmortem studies (cited from ref. 27).Aetiology and pathogenesis of pneumatosis cystoides intestinalis are not clearly known -intraluminal pressure, bacterial flora, intraluminal gas and muco-
We report a case of intrarenal teratoma in a 6-year-old boy. Two years before his operation, multicystic masses had been found in the left side of his abdomen. In the operation, three main cystic masses were located in the upper and lower poles of the left kidney, which were removed in pieces. Histologically, the cyst wall was lined mainly with keratinizing squamous epithelium with hair follicles, shafts and sebaceous glands. The adjacent renal parenchyma showed atrophy, with partially dysplastic and angiomyolipoma-like lesions. Based on these findings, the lesion was diagnosed as mature cystic teratoma of dermoid cyst type. Extragonadal teratoma occurs predominantly along the median line of the body. Intrarenal teratoma is extremely rare; however, it should be distinguished from teratoid Wilms' tumor and other renal cystic lesions.
scite is a Brooklyn-based organization that helps researchers better discover and understand research articles through Smart Citations–citations that display the context of the citation and describe whether the article provides supporting or contrasting evidence. scite is used by students and researchers from around the world and is funded in part by the National Science Foundation and the National Institute on Drug Abuse of the National Institutes of Health.
customersupport@researchsolutions.com
10624 S. Eastern Ave., Ste. A-614
Henderson, NV 89052, USA
This site is protected by reCAPTCHA and the Google Privacy Policy and Terms of Service apply.
Copyright © 2024 scite LLC. All rights reserved.
Made with 💙 for researchers
Part of the Research Solutions Family.